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A 72-year-old Caucasian female patient was referred to the respiratory clinic with a 6-month history of exertional breathlessness. She gave a history of yearly chest infections, particularly in winter months. She, however, had no history of childhood respiratory problems or any significant respiratory symptoms during her adult life. She had a 30-pack-year smoking history but she quit 25 years prior to presentation. She worked as a nursing auxillary at a local hospital and had no prior asbestosis. She was on a 1 mg maintenance dose of prednisolone for polymyalgia rheumatica. Otherwise, she had no significant medical condition and was not on any regular medications. Her exercise tolerance was unlimited and she enjoyed a good health.\nHer O2 saturation was 95% on room air and her spirometry showed obstructive pattern with FEV1 1.14L (55%), FVC 2.09L (83%) and FEV1/FVC ratio 54%.\nAn initial chest radiograph 2 years prior to presentation suggested an increased radiolucency of the left lung but this was not investigated further (). Serial CXRs 2 years later showed a relatively unchanged appearance of the left hemithorax with progressively increased opacification in the periphery of the middle and lower zones, and accompanying reduction in lung volume, on the right side ().\nThe patient underwent a contrast-enhanced CT of the chest in view of the repeated chest infections and lack of complete resolution to rule out the possibility of an adenocarcinoma in situ or endobronchial lesion, respectively. Cross-sectional imaging was also carried out to assess the possibility of an underlying bronchiectasis in the presence of the recurrent chest infections and to further assess the interstitial changes seen on the CXR on the right side.\nHer CT showed hypoplastic left pulmonary artery (; arrow), attenuated peripheral pulmonary artery branches on the left (: arrows), hyperinflated left lung with air trapping and cystic bronchiectasis of the lingua and left lower lobe (). Features were in keeping with the diagnosis of Swyer-James-MacLeod syndrome (SJMS). Interestingly, the right lung was small in size with evidence of subpleural reticulation, traction bronchiectasis and ground-glass opacification suggesting an underlying pulmonary fibrosis (). The oligaemia of the left lung is shown as reduced FDG uptake () on PET CT, which was done to investigate a different pathology.\nSputum culture grew pseudomonas species and bronchoalveolar lavage from the right lung showed mixed cellularity with 22% eosinophils and a 36% neutrophils. |
A 22-year-old female was referred to the King Saud Medical City Outpatient Urology Clinic for the assessment of large right abdominal swelling. She gave a history of right-sided abdominal swelling for the past 3 years which slowly increase in size and was associated with vague abdominal pain. Otherwise, she had no other urinary or gastrointestinal symptoms. She was diagnosed recently to have hypertension treated with angiotensin-converting enzyme inhibitor (ACEI).\nOn examination, the abdomen was soft with large abdominal swelling, smooth surface, solid in consistency, and not associated with tenderness, with superficial dilated veins in the anterior abdomen, lower back, and right lower limb [].\nAll laboratory investigations were within the normal values. Computed tomography (CT) abdomen and pelvis with contrast showed multilocular multicystic fluid containing mass arising from the right kidney, measuring 29 cm × 23 cm × 21 cm with no calcifications or soft-tissue enhancement, nor fat content []. There was no enlarged lymph node in the abdomen and pelvis. The chest X-ray was normal. Initial diagnosis was multilocular cystic nephroma, but renal hydatid cyst was suspected. Therefore, we started prophylactic albendazole 4 weeks before the surgery. An informed consent was obtained from the patient for the intervention and its possible complications. Actually, we have decided to perform the right radical nephrectomy based on preoperative images showing that the huge mass originates from the right kidney, as we believe that other treatment options such as partial nephrectomy (PN) and/or angioembolization would not be feasible treatment for such a huge mass. The right hemi-chevron approach with midline extension incision [] was performed with all precautions for hydatid cyst lesion.\nGrossly, the mass was well circumscribed, measured 30 cm × 22 cm × 20 cm and weighted 9000 g with an attached 3 cm segment of dilated ureter []. Cross-section cuts revealed tan-white-yellowish, homogenous gelatinous mass arising from the kidney parenchyma with no areas of hemorrhage or necrosis. Multiple dilated cystic spaces filled with clear yellowish fluid with smooth thin walls were seen []. There was a thin normal cortex in the lower pole. Microscopic examination [Figure and ] revealed a mass composed predominantly of smooth muscles within an edematous stroma, island of mature fat, and thick blood vessels are also noted throughout the tumor, with no evidence of malignancy or epithelioid elements. Tissue immunohistochemical staining showed a strong positivity of smooth muscle actin, caldesmon, and estrogen receptor. No definite staining is seen with human melanoma black (45) and CD117. CAE1/AE3 highlighted entrapped dilated tubules. These features are consistent with AML. Tumor is entirely excised with no evidence of malignancy seen.\nPostoperatively, she was doing fine and had a normal renal function during follow-up, as well as, she no longer use ACEI for the treatment of hypertension. Radiologically, there was no evidence of tumor recurrence at a 15-month follow-up. |
A 61-year-old female of Chinese descent first reported in the eye clinic of our hospital almost 10 years back. She had complained of progressively increasing ptosis and limitation of ocular movements in both eyes. During those visits, her retinal examination had shown a few depigmented spots near the superior arcade in the right eye and a single spot in the inferior periphery in the left eye. Over the years the patient had come for regular follow up and the spots were consistently noted by different examiners.\nHowever, on a recent review in our clinic, she was found to have the following findings: her best corrected visual acuities were 20/40(OD) and 20/70(OS). She had severe bilateral ptosis and extraocular movements were severely restricted in all directions of gaze []. Both direct and consensual pupillary reactions were briskly present. Examination of anterior segments showed nuclear sclerosis in both eyes. However, the fundus examination was remarkable in its findings. The right eye showed a hypopigmented patch with central hyperpigmentation below the inferior arcade []. The left eye had no significant retinal findings []. The hypopigmented spots, which had been noted previously, were not seen at this time.\nThe patient underwent a cardiac assessment, which revealed a first-degree atrioventricular block. Her blood sugar was elevated and an endocrine referral was made. She was subsequently started on oral hypoglycemic agents by the endocrinologist. The patient had difficulty in hearing and an otorhinology consultation has been done. A neurologic consultation was also done to rule out any other neurologic conditions like myasthenia gravis. Apart from the raised blood sugar the hematological workup did not reveal any other abnormality. Antiacetylcholine antibodies were also negative. A muscle biopsy of the orbicularis muscle was performed. Modified Gomori trichrome staining revealed the presence of ragged red fibers in the muscle specimen. These findings confirmed the diagnosis of KSS in this patient. |
A 60-year-old female presented to us on August 13, 2013 at Lata Mangeshkar Hospital and NKP Salve Institute of Research Sciences with chief complaints of lump in lower abdomen since 12 months and pain in abdomen since last 3 weeks. Lump was incidentally noticed and gradually increasing in size. The pain was constant and dull in nature with no radiation. She had no history of fever, vomiting. She had no bowel or bladder complaints. Patient had her menarche attained around 10 years back. She had regular periods.\nOn per abdominal examination a large lump involving whole of the abdomen palpable. The overlying skin was shiny and stretched. The lump was nonmobile, nonpulsatile, and firm in consistency [].\nUltrasonography abdomen study showed a large well-defined heterogeneous lesion appearing to arise from pelvis suggestive of the possibility of a pelvic mass likely ovarian in origin.\nContrast-enhanced computed tomography abdomen revealed a rim enhancing, well defined lobulated large intraperitoneal hypo dense cystic lesion of size 21 cm × 19 cm × 17 cm in dimension with volume of approximately 4 L, arising in the pelvis, with few thick walled septa within the lesion, with multiple calcific foci suggestive of benign etiology. Both ovaries seen separately []. A possibility of primary intraperitoneal cystic lesion like hydatid cyst was considered. Her laboratory investigations were normal.\nThe patient was operated on August 29, 2013 for exploratory laparotomy with excision of the cyst. A midline incision was given. Abdomen opened. A huge cystic swelling, completely extraperitoneal from the anterior wall of the dome of the urinary bladder and extending superiorly up to xiphisternum []. The cyst was extending behind the pubic symphysis and was adherent to the pubic bone posteriorly. Aspiration tried with wide bore needle but failed. Cyst opened. Thick mucinous fluid could not be sucked due to repeated blockage []. Some amount of fluid was drained [] and the cyst was closed. Cyst separated from anterior bladder wall by blunt dissection. During a separation from the bladder wall, bladder opened at the dome. Closure of bladder wall was done in two layers. Cyst excision was done. Uterus, ovaries, and ureter were normal. Drain placed in retroperitoneal space. Closure was done.\nHistopathology was suggestive of the benign cystic lesion with calcification []. Patient recovered well and discharged after 2 weeks []. |
The patient was a 35-year-old man—who operated a jackhammer. He appeared at the Emergency and Accidents Department with constant and intense pain in the lower left limb, moderate ipsilateral paresis in the ankle, foot, and toes, and coldness. He had suffered these symptoms for around 48 hours. In addition, he had suffered from an irregular fever (not measured using a thermometer) for two weeks with oropharyngeal discomfort and spontaneous pain in the groin and left lower quadrant which increased with time. He did not mention any previous trauma but when pressed recalled hyperpulsatility beneath the inguinal ligament at least a year before. He did not consult a physician as the problem did not appear to bother him.\nHis medical history included a cerebral venous thrombosis (left transverse and sigmoid sinus thrombosis) which was attributed to mastoiditis in 2006, resulting in occasional attacks of epilepsy being treated successfully with oxcarbazepine. He was a smoker with no known allergies.\nA physical examination revealed feverishness, a generally good physical condition, normal thorax and abdomen features, and an expansive throbbing mass below the inguinal ligament and left minor pelvis. The absence of femoral and infrainguinal pulses was obvious as well as severe and acute ischemic symptoms. The other limbs were found normal.\nA blood test discovered leukocytosis (16 × 103/microliter) and neutrophilia (N = 80.4%) with a hemoglobin value of 13 g/dL. In a CT scan we observed a saccular aneurysm with a transversal diameter of 45 mm located in the left external iliac artery with no sign of rupture. This was associated with a short occlusion in the bifurcation between the femoral and profunda arteries (). A deep venous thrombosis in the iliofemoral area was found. There was no evidence of aneurysms or malformations in other abdominal organs.\nThe patient underwent urgent surgery under general anesthesia. During the surgery, we decided first to perform a left retroperitoneal approach in order to prepare field for the clamping of the iliac artery, above the aneurysmatic lesion. Then, we used a longitudinal approach in the left groin to dissect the common femoral, femoral, and profunda arteries, preparing for the clamping.\nThe dissection was made with difficulty due to advanced fibrosis. With full heparinization we clamped the external iliac, femoral, and profunda arteries. We opened the anterior wall of the pseudoaneurysmatic mass. There was not any mural thrombosis and it had the appearance of a common traumatic pseudoaneurysm. We performed a thrombectomy with a Fogarty catheter and removed the short thrombus formed in the common femoral bifurcation. Neither pus nor any friable tissue was found macroscopically to support an infectious etiology. We easily observed the neck of the pseudoaneurysm, with an endothelized appearance () suggesting a traumatic etiology. However, the patient could not recall any direct trauma previously. We took specimens from the common femoral artery around the neck and from the wall of the pseudoaneurysm, for further culture and pathological studies.\nA revascularization of the femoral and profunda arteries was performed using a sequential inverted bypass originated in the healthy part of the external iliac artery. We used the saphenous vein of the contralateral thigh, prepared for this purpose (). The pseudoaneurysmatic capsule was not removed due to the high level of morbidity associated with this action, its adherence to adjacent tissues, and the absence of any local signs of sepsis.\nIn the immediate postoperative period, the patient recovered his distal pulses and the pain and paresis of the limb disappeared. However,the patient did experience significant edema of the limb, difficulties in venous return, and reperfusion. A Doppler scan found a deep venous thrombosis from the iliac to the popliteal veins.\nDuring the early stages of postoperative distress,the patient had a fever of up to 39°C with no evident site of infection and was unsuccessfully treated with piperacillin/tazobactam. The microbiology results were negative for blood, urine, bronchial aspirate, mural thrombus, and tonsillar thrush exudate. Serology was negative for hepatotropic viruses, HIV, and syphilis. The postoperative blood analysis showed some changes, which are presented in for the purpose of clarity. The pathological examination of the artery only showed unspecific fibrous thickening and loss of elastic and muscle fibers. The examination of the capsule confirmed the nature of the pseudoaneurysm, without the integrity of the vessels wall, showing mainly fibrous tissue. The microbial culture of the specimen was negative.\nThe persistence of an irregular fever of up to 38.5°C with no clear focus, oral aphthous lesions, massive venous thrombosis, previous cerebral venous thrombosis, and pseudoaneurysm supported a diagnosis of BD. The pathergy test was negative, but although this test is very specific it is known to have low sensitivity and excessive geographical variability in our environment []. In addition, the patient recalled having an ulcerated scrotal lesion which was by then a jagged scar of about 1 cm.\nFollowing the diagnosis of BD, an extensive examination was carried out. On examining the eyes, we ruled out a retinal vasculitis. When screening for other vascular lesions, CT angiography of the skull, thorax, abdomen, and pelvis revealed no abnormalities. Thrombophilia and antibody screening proved negative, there were no common tumor markers, and the transthoracic echocardiography showed no pathological signs.\nThe patient was treated with Prednisone at doses of 1 mg/kg/day with a very good response. The fever disappeared within 24 hours, as did the oral thrush and acute phase reactants. This dose was reduced progressively as much as we could and this treatment was continued during 22 months. The patient was also treated with anticoagulant therapy (Acenocumarol, INR: 2,5) during 15 months (until we could check the complete recanalization with ultrasound) and he wore an elastic high-compression stocking. He was treated with immunosuppressive drugs (Azathioprine 150 mgr/24 h) and Colchicine (1 mgr/24 h) too, and both drugs are his treatment actually, with lower dose of Azathioprine (100 mgrc/24 h). The outcome at two years shows a normal duplex scan in the bypass, a complete recanalization with venous insufficiency in femoropopliteal venous system, and a complete clinical remission of his vasculitis. |
A 63-year-old woman with metastatic breast carcinoma presented to the ophthalmology clinic with diplopia in right gaze and head turn to the right. Medical history revealed that she was diagnosed with estrogen receptor (ER)-positive and progesterone receptor (PR)-positive invasive ductal carcinoma 1 year earlier with mediastinal lymph node and bone metastasis at the time of diagnosis. She was treated with zoledronic acid 4 mg monthly and paclitaxel 80 mg/m2 weekly for 12 weeks, followed by endocrine therapy with letrozole.\nOn ophthalmological examination, best corrected visual acuity was 20/25 in both eyes. Slit-lamp examination of the anterior segment and fundus was unremarkable other than bilateral posterior chamber intraocular lenses. On motility exam, abduction was totally limited in the right eye with globe retraction and narrowing of the palpebral fissure on attempted abduction (). Abnormal head position towards the right side was noted. Magnetic resonance imaging (MRI) revealed isolated enlargement of the right medial rectus muscle (). Clinical evaluation and laboratory studies were carried out for differential diagnosis. There were no clinical findings suggestive of thyroid eye disease and thyroid function tests were normal. Rheumatologic assessment for inflammatory and vasculitic diseases was not contributory. Biopsy of the right medial rectus muscle was performed to establish a definite diagnosis and initiate appropriate treatment.\nHematoxylin and eosin staining of the biopsy specimen revealed large, round to polygonal epithelioid tumor cells arranged in loosely cohesive clusters and sheets infiltrating fibrocollagenous tissue and muscle fibers (). Immunohistochemical analyses using streptavidin-biotin peroxidase complex method revealed panCytokeratin and cytokeratin 7 positivity (). ER, PR and human epidermal growth factor receptor 2 (HER2/neu) were negative (triple-negative). Based on the patient’s clinical history and the morphological and immunohistochemical features of the tumor, she was diagnosed with breast carcinoma metastasis to the right medial rectus muscle. Pathological examination demonstrating a triple-negative breast carcinoma indicated discordance with the primary tumor, which was ER- and PR-positive at the time of diagnosis.\nThe patient was referred to the radiation oncology department for external beam radiation therapy. The orbital mass was irradiated with 45 Gy in 15 fractions. Following radiotherapy, chemotherapy with docetaxel 100 mg/m2 once every 21 days was initiated. After 15 months of follow-up, abduction of the right eye has partially recovered; the patient is stable and continuing to receive palliative chemotherapy. |
A 27-year-old Mongolian man from the central hilly region of Nepal presented with a 10-day history of pain in his left upper abdomen. Pain was acute in onset, crampy, nonradiating, and increasing in severity for the previous 3 days. He had had three episodes of vomiting during the last 2 days with no history of altered bowel habit or stool color. A systemic review was normal. He did not have significant past medical, surgical, or family history. Apparently, he had been managed at a local hospital in his home district with a diagnosis of acute peptic ulcer disease (APD) that did not relieve his symptoms, and he went to a teaching hospital in the capital. Again reassured it was APD and advised to follow up on an outpatient basis, he made frequent visits with similar complaints. Our patient’s symptoms increased in severity and he presented in the emergency department of the same hospital where investigations were carried out with normal hematological, biochemical, ultrasonography (USG), and upper gastrointestinal (UGI) endoscopy findings. With diagnostic uncertainty, an abdominal and pelvic CECT scan was done which revealed: complete occlusion of the superior mesenteric vein (Fig. , white arrow) and its tributaries due to acute thrombus extending into the main portal vein (Fig. , white arrow) narrowing its lumen; complete occlusion of the left portal vein and anterior division of the right portal vein without significant collaterals around its tributaries; a thickened edematous wall of 20 cm mid-jejunum (Fig. , white arrowhead), and ischemic changes to liver segments VII and VI. With the diagnosis of acute mesenteric ischemia secondary to portomesenteric vein thrombosis, our patient was referred to our hospital and we admitted him into the surgical intensive care unit for further management.\nAt admission, our patient was agitated but his vital signs were stable. His abdomen was soft with mild tenderness on the left hypochondrium and with normal bowel sounds. Other systemic examinations were normal. Routine investigations showed a white blood cell (WBC) count of 11,290/mm3 (N70), and a hemoglobin (Hb) level of 14.7 gm/dL, His platelet count was 366,000/mm3, and prothrombin time/international normalized ratio (PT/INR) was 16 sec/1.33. His liver function test, renal function test, and amylase level results were normal. Arterial blood gas analysis showed a lactate acid level of 0.5 mmol/L (ref, 0.7–2.5 mmol/L). Abdominal/pelvic X-rays showed no dilated bowel loops and no features of obstruction or pneumoperitoneum.\nOwing to the young age of our patient, the unusual location, and absence of acquired risk factors of venous thrombosis, investigations to find possible prothrombotic disease were started. Clot-based coagulometer results showed plasma protein S activity of 17 s (ref. 77–143) and protein C activity of 114 s (ref. 67–195), whereas a chromogenic assay showed plasma antithrombin III activity of 75 s (ref. 70–122). A final diagnosis of acute mesenteric ischemia following acute superior mesenteric and portal vein thrombosis due to protein S deficiency was made.\nOn admission, we immediately started anticoagulation therapy with intravenous infusion of unfractioned heparin (UFH) monitored 6 hourly with activated partial thromboplastin time (aPTT). The therapeutic target range of 60–80 s of aPTT was achieved by the second day. We kept our patient on nil by mouth, prophylactic intravenous antibiotics, regular monitoring of his vital signs and routine investigations. Plasma lactate level and clinical features were used as markers for progression to extensive bowel ischemia and bowel infarction, which were maintained within normal limits. During the course of treatment, our patient developed episodes of malena for 4 days. Our patient’s symptoms gradually improved, he started tolerating an oral diet and developed no other significant issues. Warfarin bridging with heparin for 2 days with a target INR of 2–3 was done. We discharged our patient after 10 days with warfarin 5 mg taken orally twice daily, advised him to avoid exertion, any physical trauma, and strenuous work, and to follow up with a PT/INR report. After 2 weeks and subsequent 5-monthly follow-ups, our patient was comfortable, had normal bowel function, and maintained an INR of 2–3. We advised him on lifelong anticoagulation therapy with warfarin and regular PT/INR monitoring. |
A 50-year-old man was admitted with progressive left-sided visual disturbance, severe headache, and was in a state of lethargy for one year. The initial modified Rankin Scale (mRS) of the patient was 2 at admission. Painful ophthalmoplegia began in April 2012. A left orbital mass biopsy was performed in another hospital, revealing highly suspected IPT, so he underwent steroid pulse therapy, radiation therapy (RT; 30 Gy/15 fx), and chemotherapy (Cyclophosphamide), though the patient did not respond well to such medical procedures. In April 2013, when he visited our hospital, he has lost his vision and light reflex in the left eye, suggesting that the 3rd, 4th, and 6th nerves were affected. The computerized tomography (CT) scan showed that a spindle shaped, slightly high density mass was noted in the left anterior cavernous sinus and in the orbital apex with pressure erosion of the adjacent bones (). A magnetic resonance image (MRI) of the brain showed enhanced lesion in the cavernous sinus bilaterally with sphenoid sinus fullness, left pituitary fossa, left orbital apex, a mass in the left temporal lobe measuring about 4.9×4.3×4.3 cm, left distal internal carotid artery, left proximal anterior cerebral artery A1, and left middle cerebral artery M1 segment involvement (). The positron emission tomography-CT scan showed no high metabolism lesion except in the brain (). Lab findings showed an infective state with elevated white blood cell, erythrocyte sedimentation rate, C-reactive protein in the blood and in the cerebrospinal fluid, but did not detect bacteria or fungi.\nWe thought it might be an IPT or metastatic cancer, and applied dexamethasone and amphotericin B. Later, craniotomy was done on the left side of the brain to remove a tumor. The pathologic findings presented infiltrating small lymphocytes and uninucleated histiocytes in the fibrous stroma. The lymphoid follicle has surrounding lymphocytes in the hematoxylin and eosin stain. In the keratin finding stain, there were no definite carcinoma with multiple infiltration (). More than 7 days after surgery, the patient slightly recovered left side visual acuity. However, left eye vision and right eye visual acuity decreased at the postoperative 10th day. A follow-up brain enhancement CT showed decreased mass size and enhancement with hydrocephalus (). An additional procedure, ventriculoperitoneal shunt, was performed. The patient was then administered TB medication 1 month after open craniectomy. About 2 weeks later after TB medication and shunt procedure, blindness in the left eye continued while visual acuity of the right eye improved daily. The follow-up brain CT showed decreased high density lesion at the left anterior cavernous sinus and orbital apex (), and the MRI revealed markedly decreased hyper intensity with strong enhancement lesion (). The mRS of the patient was 2 upon discharge. |
A 14-year-old female was referred by her paediatrician in outpatient clinic on 30 January 2018 for an elective hospital admission on 1 February, 2018. She was referred because she had recently lost weight; was complaining of a 2-week history of intermittent epigastric abdominal pain; and, upon investigation, the paediatrician found that she had an iron deficiency anaemia and an epigastric/left upper quadrant mass was found on clinical examination. She had lost 1 kg of weight in the past week, but had a normal appetite. The abdominal pain was not associated with nausea, vomiting, diarrhoea or other constitutional symptoms. She had noticed some dark stools, (consistent with her being on Ferrograd C) and a slight reduction in her exercise tolerance, but felt well within herself.\nShe had been seen in the paediatric outpatient clinic due to her history of congenital adrenal hyperplasia. Other relevant past history includes delayed puberty and poor growth, with poor weight and height gain in the past 3 years. Further history revealed that she had been suffering from the abdominal mass and abdominal pain for approximately a year, having had roughly 3–4 episodes during this time. An abdominal ultrasound was ordered in June 2017 to investigate, which showed no abnormalities, apart from a small left kidney. The girl’s medications included Hydrocortisone, Fluticasone and Ferrograd C. Her mother provided consent for medical imaging and procedures.\nOn admission, physical examination revealed a firm and non-tender mass in the epigastric and left upper quadrant region. Her abdomen was mildly distended, but soft and non-tender. Examinations of her heart and lungs revealed no abnormalities. Additionally, there was no evidence of mouth ulcers, skin changes, obvious alopecia or eye irritation. Her weight was 34.6 kg and height 146 cm (first percentile). Her vital signs showed no significant abnormalities. The differential diagnosis at this stage was thought to be of spleen pathology. The plan on admission was for laboratory investigations, stool tests, commence an iron infusion, an abdominal ultrasound and chest radiograph. On admission (1 February 2018) her abnormal blood tests showed: Hb 76 g l−1 (L), MCV 67fL (L), Albumin 32 g l−1 (L), Ferritin <5 ug l−1 (L) and ESR 25 mm hr–1 (H). Extensive urine, stool and blood tests (including autoimmune, endocrine, infective and vitamin testing) were all negative.\nAbdominal ultrasound on the 1 February 2018 showed grossly enlarged lymph nodes at the left upper quadrant, free intraperitoneal fluid at the right iliac fossa and an atrophic left kidney with cystic changes in the lower pole. The paediatric team requested a second read, which found a prominent bowel with the cause of the findings unclear. The report conclusion stated that these findings were atypical for mesenteric adenitis and CT or MRI would be appropriate. However, on ultrasound, both radiologists missed the finding of a large thick and curved echogenic band with posterior acoustic shadowing (), which is typical of a trichobezoar. This diagnosis needs to be differentiated from other differentials including food remains, barium concretions (if used) and bile ileus. Given the initial abdominal ultrasound findings, a chest radiograph was requested to check for mediastinal lymphadenopathy. The chest radiograph was reported by the radiologist as having no evidence of mediastinal lymphadenopathy or any other pathology. However, the radiologist missed the subtle evidence of a trichobezoar on plain chest radiography. It is seen as a curvilinear shadow below (), and projecting into the gastric air bubble. This finding could easily have been confused for normal food contents. Later that evening, a CT scan of the chest, abdomen and pelvis with i.v. contrast was performed (). This showed a grossly distended stomach filled by a suspected solid content, surrounded by a rim of gas, occupying as much as 13 × 6 × 6 cm in size. Additionally, mild to moderate free fluid and adjacent perigastric nodes anteriorly were found with no obvious duodenal lesion found. The suspected diagnosis was a gastric bezoar. The following day the patient admitted to pulling/playing with her hair and eating a substantial amount of hair as a child, but denied having consumed any for years. The gastroenterology team were consulted and an upper gastrointestinal endoscopy was organised later that day. This showed a large gastric trichobezoar with an associated gastric ulcer on the lesser curve of the stomach. The duodenum appeared unremarkable to the D2 segment. The general surgical team were subsequently consulted and she underwent a laparotomy, gastrotomy and retrieval of the trichobezoar on the 3 February 2018.\nShe recovered uneventfully post-operatively, being discharged from hospital after 4 days. Her last bloods before discharge were on 5 February 2018, which showed a Hb of 86 (L), MCV 73 (L), Alb 26 (L). She was discharged with nil issues from the general surgical outpatient clinic after her first visit 3 weeks post-discharge from hospital. On her most recent paediatric outpatient appointment on the 17 April 2018, she had been very well, with increased energy, exercise tolerance and her blood results had normalised. |
The patient is a 49-year old African American female G2P2002. The patient’s medical history is significant for hypertension and notably on an angiotensin receptor blocker, long-term cigarette smoker, pancreatitis, and mature cystic teratoma of the left ovary status post left oophorectomy a month prior to referral. The patient presented with persistent abdominal and back pain and with an 85-pound weight loss within 1 year. Imaging showed masses in the body/tail of the pancreas and uterus/right adnexa. Serum markers were within normal limits, except a minimally elevated human chorionic gonadotropin of 20 pg/mL. Given the possibility of a uterine or right adnexal primary, a total abdominal hysterectomy with right salpingo-oophorectomy and exploration of the pancreatic mass were performed. The surgery revealed a sclerotic mass in the body and tail of the pancreas that grossly involved the superior mesenteric artery (unresectable); therefore, the tumor was left in situ and pancreatic biopsy was taken for histopathology.\nThe hysterectomy specimen revealed a fibroid uterus. The mass adjacent to the right ovary was a benign subserosal uterine polypoid leiomyoma. The right cystic ovary was entirely submitted for histologic evaluation and was a mature teratoma. Histology of the pancreatic biopsy showed a high-grade spindle cell sarcoma and adjacent moderately differentiated adenocarcinoma ( and ). Around the sarcoma were a concentration of myofibroblasts marking with desmin and smooth muscle actin (SMA), and overall the background tissues were prominently fibrotic. Additionally, the hepatocholedochal lymph node submitted showed a focus of metastatic adenocarcinoma of 0.85 mm. Immunohistochemical stains were performed on the current and prior biopsy specimens (). The adenocarcinoma component was, on immunohistochemistry, compatible with a pancreatic primary (). The sarcoma stained focally for SMA, focally for desmin, and showed strong diffuse staining with CD10 (). The diagnosis of pancreatic carcinosarcoma was made.\nAfter discussion in the multidisciplinary tumor board, the patient received chemotherapy with gemcitabine and docetaxel with no response, followed by ifosfamide and Adriamycin with progression. After failing these 2 lines of chemotherapy, palliative radiation therapy was planned; however, the disease rapidly progressed and the patient died approximately 10 months postdiagnosis. |
A 67-year-old man was admitted to another medical center with right flank pain lasting for more than one month. An ultrasound scan revealed a mass in the right kidney. His subsequent computed tomography (CT) scan and magnetic resonance imaging (MRI) revealed a mass on the upper pole of right kidney, 4.5 cm diameter, exophytic and solid character suggestive for malignancy. MRI scan also showed a 2 cm diameter, hypodense mass on the upper pole of right kidney which was thought to be a benign cystic lesion (). However this lesion was not revealed at CT. He was referred to our department. Physical examination was normal. All hematological and biochemical tests were unremarkable except total PSA (7.07 ng/dL). Patient had no previous medical history. He was a smoker for 40 years.\nThe tumor was clinically diagnosed as a right renal tumor and classified as cT1bN0M0, according to tumor-node-metastasis system. Patient underwent right RN and adrenalectomy. He was discharged at postoperative third day, without any complication. At the sixth month and first year follow-up, the patient was free from the disease.\nPathology. On macroscopic examination, there were 2 different tumors on the upper pole of right kidney. The cut surface of the bigger one was at 6.5 cm diameter, composed of two nodules, one of them was of orange color and the other one was of yellow-brown color and had small cystic cavities. The smaller tumor was in 1 cm diameter, of beige color, and had irregular margins. The distance between two tumors was 0.5 cm. Microscopy revealed two different patterns. The bigger tumor was diagnosed as clear cell type RCC with a diameter of 6.5 cm and smaller one was diagnosed as type 1 papillary RCC with a diameter of 1 cm on microscopic examination. Both of the tumors were limited in the kidney and there was no perirenal and perihilar invasion. There was no invasion of blood and lymph vessels. Pathologic examination revealed a normal adrenal gland.\nAreas of clear cell RCC were characterized by solid nests and sheets of carcinoma cells interspersed by prominent network of delicate blood vessels. Tumor cells have water clear cell cytoplasm surrounded by a distinct cell membrane and had Fuhrman grade 2 morphology with finely granular chromatin but small nucleoli that are not discernible of 10x magnification. On the other hand, areas of papillary RCC were characterized by tightly packed tubulopapillary structures and nuclear morphology was consistent with Fuhrman grade 2 (). |
A 64-year-old man presented in the urology outpatient department hospital with painless swelling on the glans penis for the past 2 months along with bleeding from the mass. There was no history of urethral discharge or any urinary complaints. The patient was a chronic smoker and had undergone angiography for coronary artery disease 1 month back. However, there was no history of any local trauma or other comorbidities.\nLocal examination of the penile lesion revealed a lesion arising out of the meatus, at the tip of the glans, measuring 3 cm × 2 cm []. The meatus was visualized and partially obliterated and bled on touch. There was no induration beyond the lesion. The penile shaft was normal. Multiple bilateral inguinal lymph nodes were palpable each measuring <1 cm in size, firm in consistency, and mobile. The provisional diagnosis of carcinoma penis was made.\nComplete blood count and biochemical investigations were within the normal limits, except for eosinophilia (13%). The patient's serum was nonreactive for HIV and hepatitis B surface antigen. The routine urine examination showed field full of red blood cells and 2–4 pus cells. On contrast-enhanced computed tomography abdomen, a simple cortical cyst in the right kidney was noted; otherwise, no renal or bladder mass was seen.\nWedge biopsy of the penile lesion was taken under aseptic conditions. Histopathology showed partly ulcerated keratinized stratified squamous epithelium covered by dense fibrinous exudates and subepithelium showed a proliferation of endothelial cells with mild-to-moderate nuclear enlargement and having a moderate amount of eosinophilic cytoplasm [Figure and ]. Occasional mitotic figure (<1/10 high power field) was noted; however, no atypical mitosis was found. There was mild-to-moderate inflammatory infiltrate composed of eosinophils, lymphocytes, and neutrophils []. Few areas also showed interconnecting vascular channels; however, those were lined by endothelial cells with the same morphology as described above. The immunohistochemical examination showed immunoreactivity for CD31 in epithelioid endothelial cells and smooth muscle antigen (SMA) (in myopericytes) [Figure and ]. The histopathological features in combination with immunohistochemical markers were those of EH of the penis. |
A 33-year-old man presented to the hospital with a complaint of sudden gross hematuria. He had no associated pain or additional complaints. His laboratory workup revealed normal levels of hemoglobin and tumor markers (CEA, CA19-9, NSE, SCC, sIL-2R, and IgG4). Atypical urothelial cells were not detected in the urinary cytological test. In a cystoscopic examination, gross hematuria from the left ureteral orifice was found, although no apparent abnormalities were observed in bladder mucosa. In contrast, enhanced CT revealed a 35-mm mass with slight enhancement, which significantly compressed the left renal vein (Fig. a,b). Collateral vessels were not apparent between renal parenchyma and inferior vena cava. Abdominal enhanced magnetic resonance imaging identified a low signal intensity of tumor at T1 as well as T2-weighted images with a slight enhancement and an almost normal intensity in diffusion weighted image suggesting a benign tumor. A CT-guided needle biopsy was not performed because the tumor was encroaching the renal vein, thus having a possible risk of hemorrhage. After obtaining the informed consent concerning surgery and subsequent publication, the patient underwent open tumor resection through retroperitoneal approach for easy extension of the resecting area in case the frozen section identified malignancy. During surgery, a yellowish, non-necrotic tumor compressing the renal vein was identified corresponding to the CT findings. The result of the intraoperative rapid pathological analysis suggested a benign tumor. Therefore, the left kidney was spared and the surgery was completed.\nPathological examination identified the mass was directly surrounded by adipose tissue, lacking a distinct capsule, and was composed of adrenocortical-like cells. Medullary cells were not observed. The final pathological diagnosis was adrenocortical adenoma (Fig. ).\nAccording to the Weiss criteria, the estimated malignant potential of the tumor was low, with only one of the nine criteria met, which was clear cells comprising ≤25% of the tumor. Accordingly, the tumor was diagnosed as benign. The hematuria improved immediately after surgery, and no evidence of tumor recurrence was found during the 2-year follow-up, supporting the benign nature of the tumor.\nTo further investigate the etiology and hormonal function of the mass, we performed immunohistochemical analysis of key steroidogenic enzymes, as previously reported:, 3βHSD, CYP11B2, CYP17, and CYP11B1 (Fig. ). The PCA per TA of each stained section was measured by the Color Deconvolution software and the ImageJ software. The PCA/TA ratio of 3βHSD and CYP11B1 was 39.4% and 93.4%, respectively. The latter indicated that the tissue was of an adrenocortical origin. The PCA/TA of CYP17 was 10.0%, suggesting that some cells might have produced cortisol. The CYP11B2 staining result was positive only in a few cells (0.3%), indicating that the mass unlikely produced aldosterone.\nAfter removal of the tumor, the narrowing of left renal vein disappeared in the CT image (Fig. c). At present, more than 2 years after the operation, there is neither recurrence nor even microscopic hematuria. |
The patient was a 58-year-old man, on treatment for hypertension, who was admitted to a vascular surgery service because of an incidental finding of two aneurysms of the SMA, identified during preoperative imaging exams preparatory to repair of an incisional hernia. The patient had no abdominal symptoms and on physical examination his abdomen was flaccid and painless and with a pulsating mobile mass in the epigastrium. Abdominal ultrasonography indicated a partially thrombosed saccular aneurysm in the retroperitoneal space, with no communication with the aorta. Multislice angiotomography revealed two aneurysms of the SMA, a proximal one measuring 5.9 × 5.2 × 5.0 cm and a distal one measuring 5.3 × 3.5 × 3.2 cm ( \n ). Since multiple collateral branches emerged from both aneurysm bodies, which meant the endovascular treatment would have involved a risk of damaging the intestinal blood supply, the decision was taken to perform open surgical repair. During the operation, by explorative laparotomy, access to the retroperitoneal space was achieved after performing the Cattell-Braasch maneuver, with medial displacement of the ascending colon and part of the transverse colon, exposing the infrarenal aorta and its branches. This revealed two true aneurysms of the SMA, the larger of which was around 3 cm from the arterial ostium and the smaller approximately 2 cm from the end of the first ( ). It was also possible to observe collateral branches (right colic, ileocolic, jejunal artery, and ileal arteries) projecting from the bodies of these aneurysms. A mesenteric-mesenteric, end-to-end bypass was therefore constructed, using a dacron prosthetic graft, excluding both aneurysms but preserving branches distal of the proximal aneurysm. It was decided to ligate and resect the aneurysms – sending specimens for cultures – and their lumens were opened, revealing large quantities of intraluminal thrombi ( ). Inspection of the abdominal cavity found the intestines to be viable and free from any sign of injury. During the postoperative period, the patient suffered gastrointestinal atony, but responded satisfactorily to prolonged conservative measures. Additionally, on the fifth day after the operation, a control computed tomography revealed signs of hematoma in the hepatorenal recess, managed conservatively to resolution. The patient was discharged from hospital to outpatients follow-up in good clinical condition, 18 days after the operation ( ). There was no bacterial growth in the culture of the aneurysm segment. |
A 7-year-old Japanese boy presented to a nearby clinic owing to an increasing mass and pain in his left ankle for 6 months. He experienced a sprain of his left ankle several months before the onset of his symptoms. Gradually increasing pain hindered him from walking long distances and exercising. He was diagnosed as having synovial osteochondroma by a local physician and referred to our hospital. Physical examination revealed a palpable hard mass on the anterior and posteromedial sides of his left ankle. He had a full ankle range of motion of 15° in dorsiflexion and 40° in plantar flexion with his knee flexed. Instability and apprehension were evoked by an anterior drawer test on his left ankle. Left ankle plain radiography revealed multiple oval free bodies 10–15 mm in size at the anterior and posterior ankle joint spaces (Figures and ). Computed tomography images showed intra-articular multiple ossified oval mass lesions and some protrusions arising from the talus (Figures –). Magnetic resonance imaging showed an intra-articular oval mass with low intensity on T1-weighted images and partially high intensity on T2-weighted and short T1 inversion recovery (STIR) images. Diffuse STIR high-intensity lesions were confirmed on the protuberance from the talus and talar body underneath it, suggesting a bone marrow lesion of the protuberance and talus (). Owing to the intractable pain and inability to walk, surgical treatment was performed. As the mass lesions were quite large to be removed under the arthroscopic procedure, open surgery was selected. With anterior midline and posteromedial incisions, intra-articular multiple mass lesions were removed (Figures and ). Excisions of the protuberances from the talus rendered a circular articular cartilage defect 5 × 3 mm in size. The microfracture technique was performed for the cartilage defect (). No obvious mass lesion was found in the synovial tissue. As ankle instability was not the chief complaint of the patient, we did not address ankle lateral ligament repair. The wound was irrigated and closed, and a sterile dressing was applied.\nOn macroscopic examination, some mass lesions were colored brown, including the protuberance from the talus, while others were white (). Microscopic examination revealed no hyaline cartilage component in the synovium (). The brown mass lesion was trabecular bone tissue covered almost exclusively by fibrous tissue, and only a small amount of cartilage was found (). The white mass lesions, all of which were loose bodies, were thick hyaline cartilage (cartilage cap) with central ossification, which resembled the epiphysis (). Chondrocytic clusters were detected, and the central osseous tissue was mostly necrotic. No sarcoma component was found. As the protuberance had arisen unilaterally from the talar epiphysis of the juvenile and the synovium did not contain a cartilage component, we diagnosed it as DEH. A below-knee cast was applied for 2 weeks with non-weight-bearing for 4 weeks. Half weight-bearing was started 4 weeks after the operation, and full weight-bearing was achieved in 6 weeks. The preoperative ankle pain disappeared after the primary surgery.\nHowever, 3 months after the primary surgery, the patient experienced recurrent left ankle sprain, and 10 months postoperatively, he first experienced a temporary locking of his left ankle when he plantarflexed the ankle. He had a sharp pain at the posterior side of the ankle joint during the locking position. Lateral plain radiography revealed the growth of a new oval ossified free body in the posterior joint space of his left ankle during postoperative follow-up (Figures –). Radiographic evaluation using the anterior drawer stress test revealed a significant anterior translation of the talus compared with the contralateral side. A secondary surgery was performed to improve the patient's ankle pain and instability. As a second surgery, arthroscopic removal of the recurrent free body and anterior talofibular ligament repair were performed. Anteromedial and anterolateral portals were created for visualization of the intra-articular free body. The free body existed in the posterior portion of the ankle joint space and was removed using a grasper. The cartilage defect, resulting from the resection of the protuberance from the talus, treated with microfracture in the primary operation was covered with healthy cartilage (). The lateral ligaments were then repaired according to the modified Brostrom-Gould method using suture anchors through a longitudinal incision made over the distal fibula, which extended inferiorly toward the sinus tarsi.\nPostoperatively, a below-the-knee splint was applied for 3 weeks with non-weight-bearing for 1 week. Full weight-bearing was initiated 1 week after the operation. Six months after the operation, the patient experienced neither ankle sprain nor severe ankle pain and could live a daily life without any inconvenience. A recurrence of free body or protuberance was not confirmed by the plain radiographs during the postoperative follow-up of 6 months (). We compared the outcomes of the surgery using an objective standard rating system, the Japanese Society for Surgery of the Foot (JSSF) scale [, ]. The preoperative JSSF scale score of 78 points (maximum score, 100 points) significantly improved to 85 points 6 months after the second surgery. |
A 64-year-old right handed male with a university degree in arts presented to the neurology service with the chief medical complaint of behavioral changes slowly progressing over the course of the past seven years. Initially the patient experienced an insidious onset of decline in work productivity and gradually stopped working within the space of a year. Meanwhile, his family also noticed a short-term memory deficit primarily evidenced by difficulty remembering messages he was supposed to give to his wife, as well as not being able to name some specific objects. In the ensuing years the symptoms kept worsening while new behavioral and motor changes such as apathy, irritability, aggressiveness, delusional thoughts, psychomotor and gait slowing were contributing to progressive cognitive impairment. There were also reports of paroxysmal events characterized by self-limited episodes of experiencing unpleasant smells that lasted for seconds to a few minutes.\nThe patient had consulted several physicians during the course of the disease. He was diagnosed with bipolar disorder by a psychiatrist with the report of accelerated thinking at the time, and was treated with olanzapine and valproate without any significant improvement. A neurosurgeon diagnosed normal pressure hydrocephalus and performed a ventriculoperitoneal shunt with no apparent symptomatic relief.\nAt a later stage, he presented to the Neurology Department of the University of São Paulo with the main complaint of worsening delusional thoughts. The patient stated that his wife had been substituted by a perfect copy of herself, that the house he was living in only resembled his original property, and the city's most famous avenue had been duplicated in two identical copies.\nPast medical history was relevant for Pulmonary Abscess, Hypertension, Depression, Diabetes and former tobacco and alcohol abuse. He had an unremarkable family history. Prescribed medications were Carbamazepine, Olanzapine, Sertraline and Clonazepam.\nNeurological examination was remarkable for a cognitive impairment characterized by dysexecutive syndrome (inappropriate digit span, low fluency verbal test, impairment of abstract thinking and problem solving, inadequate clock drawing test), psychomotor slowing and inappropriate behavior. The Mini-Mental State Examination test score was 26 out of 30 (dropped 1 point in spatial orientation and 3 in recall). Glabellar and palmomental primitive reflexes were markedly present as well as inhibitory paratonia, demonstrating frontal release signs.\nBased on initial impressions, the clinical presentation was attributed to LE (short-term memory impairment, temporal lobe seizures and behavioral abnormalities) of protracted course associated with a delusional misidentification syndrome (Capgras Syndrome concomitant to Reduplicative Paramnesia).\nThe patient was then admitted to the neurology ward for extensive investigation. General laboratory tests were unremarkable, serology for common infectious diseases were negative (HIV, Syphilis, Lyme, Cytomegalovirus, Herpes Simplex), Antinuclear Antibodies (ANA) were positive in high titers (1:2560) but the remaining rheumatologic panel was unrevealing (AntiSm, Anti-Ro, Anti-La, Anti-dsDNA). Brain Magnetic Resonance Imaging (MRI) revealed high signal intensity on T2/ FLAIR in the left temporal pole and left mesial temporal region. Cerebral spinal fluid (CSF) was relevant for moderate lymphocytic pleocytosis (85 cells/mm3), elevated protein levels (116mg/dl), normal glucose levels (56 mg/dl), negative flow cytometry for malignant cells, and negative infectious markers (cultures for bacteria and fungi, Syphilis, Tuberculosis, Herpes Simplex, Varicella Zoster, Cytomegalovirus, Epstein Barr Virus). Positron Emission Tomography (PET) with fluorodeoxyglucose (FDG) disclosed hypermetabolism in the left mesial temporal region and peri-insular cortex, as well as severe and diffuse hypometabolism in frontal, parietal and lateral temporal cortices. Electroencephalography showed diffuse slow waves (theta and delta) without epileptiform activity.\nWhile the aforementioned initial work-up was being performed, the patient was started empirically on acyclovir, ampicillin and fluconazole for 14 days. Following the negative results for infectious agents associated with the clinical picture, temporal lobe abnormalities on advanced neuroimaging studies and poor response to antimicrobials, the hypothetical diagnosis of Autoimmune LE was strengthened. The patient received methylprednisolone 1 gram daily for five days without significant improvement and investigation for a paraneoplastic syndrome was continued. Tests for autoantibodies associated with neurological paraneoplastic syndromes in serum and CSF were negative (Anti-Hu, Anti-Ri, Anti-Yo, Anti-Ma2, Anti-NMDAR, Anti-VGKC, Anti-GAD, Anti-TPO, Anti-TG). Computed Tomography (CT) of the neck, thorax, abdomen and pelvis disclosed no significant findings. Ultrasound of the testicles was also normal. However, whole-body FDG-PET revealed an anomalous increase in glycolytic metabolism in cervical external inguinal and iliac portacaval lymph nodes. A cervical lymph node biopsy was performed but anatomopathological study showed nonspecific neutrophilic inflammatory reaction and absence of signs suggestive of malignancy. The patient was then discharged for clinical follow-up scheduled over the next few months.\nTwo months following previous hospital admission, the patient exhibited progressive worsening of cognitive and behavioral symptoms and therefore another work-up for occult malignancy was undertaken. The repeat whole-body CT scan disclosed enlarged mediastinal, hilar, cardiophrenic and axillary lymph nodes. The hypothesis of lymphoproliferative disease was proposed and the patient was admitted to the hematology ward for fine-needle aspiration of the axillary lymph node. Papanicolaou and Giemsa-stained cytology slides, and the cell block preparation from the sample material, showed morphologic features of a large cell lymphoma: intermediate-to-large sized cells, nuclei of different sizes and shapes, clumping artifact of naked nuclei, frequent mitosis and apoptotic bodies. Immunophenotyping showed a B-cell phenotype with a high Ki-67 labelling index (). The tumor cells were negative for CD 3, CD 30 and pan-cytokeratin AE1/AE3. In light of these immunohistochemical results and morphologic features, the diagnosis of diffuse large B-cell lymphoma was reached. After the procedure, the patient acquired a nosocomial respiratory infection followed by septic shock and evolved to death. |
A 63-year-old, right-handed Caucasian woman with an unremarkable medical history was referred to the hand clinic at our institution with a large firm mass in her left hand that had been growing steadily over the previous 3 years (Figures\n and\n). The mass had grown to a point that it interfered with many of her daily life activities. She complained of pain and occasional paresthesia in the first three rays of the hand. Ultrasound examination was inconclusive. Magnetic resonance imaging (MRI) was performed, which revealed a large mass spanning almost the entire palmar aspect of the hand, superficially to the flexor tendons and with no evidence of bone involvement (Figures\n and\n). A pre-operative angiogram showed a tumor with a rich blood supply (Figure\n). The tumor vessels were given off by the deep palmar arch, the external branch of the superficial palmar arch and the palmar digital arteries of the first, second and third fingers. With the patient under local anesthesia, an incisional biopsy was performed, which identified a synovial sarcoma.\nSubsequently, with the patient under general anesthesia, the mass was excised via a palmar approach, preserving the flexor apparatus of the fingers as well as all major vessels and nerves (Figures\n and\n). Macroscopically, the tumor was grayish and had a maximum length of approximately 10cm (Figure\n). The tumor was multinodular and rubbery and seemed to be circumscribed by a fibrous pseudocapsule. The post-operative period was uneventful. Recovery was fast, and the patient had no significant functional or aesthetic impairment (Figure\n). She declined any physiotherapy treatments. Her only complaint was pain over the surgical scar that subsided approximately 6 months post-operatively. Pathological examination revealed the typical appearance of synovial sarcoma of the monophasic type (Figure\n). The specimen margins were found free of tumor cells. There was no evidence of systemic disease on the computed tomography (CT) scan of the thorax, abdomen and pelvis that had been done pre-operatively. The patient underwent post-operative radiotherapy and chemotherapy with good tolerance. Adjuvant chemotherapy consisted of one cycle of ifosfamide and doxorubicin following the recommendations described by Kampe et al.\n[]. Ifosfamide was given for 6 days at a dose of 14g/m2. An equimolar dose of mesna was combined with the first dose of ifosfamide of 2g/m2. These two drugs were given as a 4-hour intravenous bolus. The subsequent ifosfamide treatment was given at the rate of 2g/m2/24 hours by continuous intravenous infusion. The patient received an additional dose of mesna at the end of the sixth day. Doxorubicin was administered as a 48-hour continuous intravenous infusion, corresponding to a total dose of 60mg/m2. Adjuvant radiotherapy was delivered at the end of chemotherapy, including not only the tumor excision site but also a 3cm margin. A total dose of 60 Gy was applied, fractioned in 2.0 Gy daily, 5 days per week, during 6 weeks.\nThe patient was followed at the hand clinic at regular intervals for 9 years. The CT scans of the thorax, abdomen and pelvis that were done regularly during this period failed to show any evidence of systemic disease. She then decided to stop coming to the outpatient clinic, arguing that she saw no point in going as she felt perfectly well. Eleven and one-half years after surgery, the patient returned to the outpatient clinic with a recurrence of a sarcomatous mass in her left hand. MRI of the hand showed a large hypervascular mass with foci of necrosis that occupied most of the anterior compartments of the hand and encroached into the metacarpal and carpal bones (Figure\n). No evidence of metastasis was found.\nA multidisciplinary team decided to propose left-hand amputation, which the patient declined. Twelve years after the initial diagnosis, she decided to stop coming to the outpatient clinic again and rebuffed any further contact. Six months later, she was found dead at her home, having had a massive hemorrhage from the recurrence in her hand. |
An 80-year-old female with a past medical history of hypertension and diabetes mellitus, on oral medication, presented to a community hospital with postprandial right upper quadrant pain and dark stools. She was found to have mild thrombocytopenia. Computed tomography (CT) scan of the abdomen revealed cholelithiasis and an eccentric thickening of the medial wall of the gallbladder concerning for malignancy. There were a few esophageal varices seen; evidence of liver cirrhosis was present. There were no intraabdominal ascites. The patient had no history of alcohol abuse. She was referred to us for further care. Her blood work demonstrated a hemoglobin of 13.9, platelet count of 117,000, total bilirubin 1.4, international normalized ratio (INR) 1.3, and creatinine of 1.1. Her complete hepatitis serology was negative. Upon repeat imaging, her contrast-enhanced CT scan of the abdomen and pelvis revealed a 2 cm liver nodule in the left lateral sector with intense early arterial phase-contrast enhancement, with no early washout or fill-in typical of hepatoma or hemangioma, respectively (Figure ). Magnetic resonance imaging (MRI) of the abdomen ruled out hemangioma of the left liver lobe, but could not rule out hepatocellular carcinoma or other malignant liver tumors as a possibility.\nThere was an enhancing polypoid mass within the gallbladder lumen suggestive of soft tissue on the medial wall of the inferior gallbladder (Figure ).\nThe liver was nodular in contour suggestive of cirrhosis, and paraesophageal varices were present. The gallbladder contained a 2.7 cm x 1.8 cm enhancing mass concerning for malignancy. An upper endoscopy revealed grade two esophageal varices.\nThe patient was determined to have compensated Child-Pugh class A liver cirrhosis with some portal hypertension (small esophageal varices, recanalized umbilical vein, and mild thrombocytopenia) and a model for end-stage liver disease (MELD) score of twelve. Operative intervention was decided on with the patient. Laparoscopic examination showed no carcinomatosis; however, it showed a nodular liver, and intraabdominal venous varicosities. Laparoscopic liver ultrasonography showed no additional liver masses aside from the known left liver lobe mass. Ultrasonography of the gallbladder showed a hyperechoic intraluminal mass with a hypoechoic rim of interface between it and the medial gallbladder wall. No evidence of invasion into the liver was seen grossly and the gallbladder wall thickness was normal. Therefore, a laparoscopic non-anatomic wedge resection of a portion of left lateral lobe containing the mass and a laparoscopic cholecystectomy were performed. During cholecystectomy, dissection of the gallbladder off the cystic plate was unremarkable and no evidence of transmural extension of tumor into cystic plate was encountered. The left lateral liver resection specimen and gallbladder were submitted for frozen section analysis (Figure ). This revealed a papillary adenocarcinoma of the gallbladder and a benign vascular tumor of the liver.\nAs a result of the malignant gallbladder diagnosis with no exact T-staging available on frozen section, a decision was made to perform non-anatomic resection of segments IVb and V en bloc with the cystic plate and hepatoduodenal lymphadenectomy. Therefore, a limited right subcostal incision was made and a non-anatomic resection of portions of segment IVb and V, en bloc with the cystic plate, was performed as well as a hepatoduodenal lymphadenectomy. Cystic duct margin, on frozen section, was negative for malignancy. Final pathology revealed a 2.7 cm T1b well-differentiated grade one papillary carcinoma of the gallbladder; no lymph nodes were found in the specimens (Figures -).\nThe liver mass was determined to be an anastomosing hemangioma with sharp demarcation from the surrounding liver parenchyma, associated with a branching vascular pattern with mild nuclear atypia and absence of mitoses (Figures -).\nPostoperative recovery was uneventful and the patient was discharged on postoperative day five. She was doing well 18 months postoperatively with no evidence of recurrence. |
A 74-year-old male presented at our clinic with a 10-day history of mild pain and loss of vision in his right eye. In 2003, he had undergone an exenteratio bulbi with the insertion of a prosthesis in the left eye following the corneal perforation of a Mooren's ulcer. Medical examination revealed acute conjunctival hyperaemia and a large perforated Mooren's ulcer in the right eye. The anterior chamber was absent and a seclusio pupillae with cataract were also present (Figure ). Visual acuity was hand movements. Under general anaesthesia, we performed a 14 mm corneoscleral transplantation in association with the extracapsular extraction of the cataract, intraocular lens implantation and superior basal iridectomy.\nThe surgical technique included a total limbal peritomy in attempt to save limbal stem cells. A 14 mm trephine was used to mark and partially cut the scleral surface. After careful haemostasis, the anterior chamber was entered obliquely with a diamond knife and the sclera was cut with Castroviejo's corneal scissors. The entire cornea and scleral ring was removed, viscoelastic material was placed on the recipient bed and a large anterior capsulorhexis was performed. After the expression of the lens nucleus and aspiration of residual masses, an intraocular lens was implanted into the capsular bag and a peripheral iridectomy was performed. The donor tissue was trephined from a whole eye used within 24 hours of the donor's death and stored in a moist chamber using a trephine of the same diameter as that used on the recipient. The donor corneoscleral graft was then sutured into place onto the scleral ledge using 8.0 interrupted silk sutures. The anterior chamber was filled with viscoelastic material and the conjunctiva was closed with Vicryl 8.0 interrupted sutures.\nThe day after surgery we found a mild keratitis with a low flare in the anterior chamber. The intraocular pressure measured with a pneumotonomer was 14 mm/Hg. Postoperative steroid therapy was prednisolone acetate 1% every two hours for four weeks (decreased to four times a day for five months), oral cyclosporine A (5 mg/kg/day) for six months and prednisone 25 mg a day for four months. After six months, the best corrected visual acuity was 6/60 (Figure ). |
A 38-year-old woman with a 3-month history of an anterior cervical mass located in the suprasternal region of her neck was referred to our hospital. Physical examination revealed that the 3-cm movable neck mass was firm and slightly tender on palpation and had a distinct margin from surrounding tissues. An ultrasound examination showed that the well-defined oval mass was 31 × 23 × 17 mm in size and exhibited heterogeneity. Furthermore, a non-enhanced computed tomography (CT) scan of the neck revealed that the distinct neck mass in the subcutaneous tissue had a mixture of soft tissue and fatty components (Fig. ). Based on these findings, the cervical tumor was clinically diagnosed to be an unusual lipoma with degeneration; however, we could not exclude the possibility of it being a malignant tumor such as liposarcoma. Thus, fine-needle aspiration cytology of the tumor was performed, but no diagnosis of malignant cells was obtained. Further examinations were conducted to address concerns related to her menstrual pain, and abdominal magnetic resonance imaging revealed bilateral ovarian cysts in the lower abdomen. Under general anesthesia, the patient underwent neck mass extirpation and bilateral ovarian cystectomy; pathological examination of the cysts resulted in a diagnosis of ovarian endometriotic cysts. During the surgery, the cervical mass was well demarcated and did not adhere to the surrounding tissues. The postoperative course was uneventful. The gross pathology report showed that the neck mass measured 3.0 × 2.5 × 2.0 cm. The cut surface of the specimen was heterogeneous, solid, whitish, and yellowish (Fig. ). Microscopically, the tumor was composed of spindle cells, epithelial nests, and mature adipose tissue (Fig. a, b). Immunohistochemical examination revealed that both spindle cells and epithelial nests were positive for cytokeratin (CK) AE1/AE3 (Fig. ). As these histopathological findings were consistent with the features of EHT, a diagnosis of EHT was confirmed. Over a follow-up period of 30 months, this patient exhibited no evidence of recurrence. |
A 29-year-old, 79.5 kg female patient was known to have migraine and epilepsy for the last 12 years. She was treated for tuberculosis meningitis 12 years back and was on antiepileptic drugs (AED) but stopped AED after consultation with the neurologist for the last 1 year as she was seizures free. She underwent cesarean section under spinal anesthesia due to previous uterine scar and remained stable throughout the procedure. On the 1st postoperative day, the patient had an event of generalized tonic–clonic seizure (GTCS) lasted for 30–60 s. Neurology team was taken on board and the patient was loaded with 1 g IV levetiracetam and 500 mg BID. Magnetic resonance imaging (MRI) brain and electroencephalogram (EEG) advised. MRI was tried but failed due to patient's restlessness. However, EEG showed diffuse theta slowing along with intermittent delta bursts. On the 3rd postoperative day, the patient had another event of GTCS lasted for about 40 s with a drop in Glasgow Coma Scale (7/15) along with loss of protective airway reflexes. The patient was loaded with valproic acid 1.5 g and 500 mg BID. The patient was intubated for airway protection and shifted to ICU for further management. On arrival in the ICU, the patient had only a right forearm 22-gauge IV cannula. An arterial cannula in the right radial artery was placed just after patient's arrival in ICU by the on-call anesthesia resident for the purpose of blood sampling and monitoring blood pressures. Just after the establishment of arterial cannula, patient had another event of GTCS with severe jerky movements and the only IV cannula got dislodged. Due to difficult IV cannulation and intense generalized jerky movements, the on-call team failed to maintain an IV line despite multiple attempts. It was now 10 min and patient was continuously seizing. Therefore, after analyzing risk versus benefit ratio, it was decided by the on-call ICU team to inject a diluted injection of midazolam through the established arterial cannula. Midazolam 5 mg (1 mg/ml) was diluted in 20 cc syringe (5 ml midazolam + 15 ml normal saline) and was injected slowly 1 mg (4 ml)/20 s. Seizures activity was successfully aborted with 4 mg (16 ml) of IA diluted midazolam. Central venous catheterization was immediately done afterward. She was strictly monitored for adverse reactions especially for signs of ischemia and no immediate or delayed adverse reactions were observed. She was successfully extubated after 3 days, and subjectively, there was no complaint of pain or discomfort at the injection site. |
A 50-year-old gravida 2 para 2 woman presented at our hospital for a regular checkup for breast cancer. She had a family history of breast cancer on her maternal side and had undergone surgery for right breast cancer at the age of 43. For the past 6 years, she had taken tamoxifen and was followed up for cervical cytology. She also had childhood asthma and an appendectomy at the age of 12. She had complained of excessive menstruation, and a hot spot in the uterine lumen was detected using positron emission tomography–computed tomography (PET-CT) at the breast surgery department. She was referred to the obstetrics and gynecology department for further examination and treatment.\nPhysical and abdominal examination findings were normal. The results of the pelvic examination finding were also normal. Transvaginal ultrasonography revealed a 21.8-mm uterine mass and an enlarged right ovary. Cervical cytology was negative for malignant intraepithelial lesions. Endometrial cytology was negative. Her blood test showed a hemoglobin level of 8.7 g/dL and hematocrit of 29.2%. Laboratory findings such as platelet count and solidification systems were normal, and tumor markers including CA-125 and CA-19-9 were negative.\nPelvic magnetic resonance imaging (MRI) was performed to examine the uterine mass, which revealed an endometrial polyp and submucosal leiomyoma in the uterine lumen and a 5-cm right ovarian tumor []. The MRI signal pattern suggested that the right ovarian tumor was a fibroma or thecoma. Thus, the cause of anemia was thought to be due to the endometrial polyp and submucosal leiomyoma. Therefore, she underwent laparoscopic surgery under the clinical diagnosis of endometrial polyp, submucosal leiomyoma, and right ovarian tumor for polyp, leiomyoma, and tumor excision, respectively.\nLaparoscopy showed that the right ovarian tumor was approximately 5 cm in diameter, a white solid tumor, suggestive of a fibroma or thecoma. The uterus was slightly enlarged. Laparoscopic total hysterectomy, right salpingo-oophorectomy, and left salpingectomy were performed. Tissues were excised out vaginally. Uterus and ovarian tumor weights were 276 g and 57 g, respectively. Surgery time was 163 min, and the amount of bleeding was 100 ml.\nHistopathological examination revealed polyp-like ridges containing interlacing endometrial glands with mild alignment disorder and endometrial stroma in the uterine endometrium. There were ectopic endometrial tissues in the uterine muscle layer and serosal surface and some uterine intramural leiomyomas (maximum 6-mm diameter). The ovarian tumor measured 5.5 cm × 3 cm × 3 cm, and the cut section of the tumor showed a hard gray-white solid area which was well encapsulated []. Furthermore, the ovarian tumor contained interlacing bundles of fusiform cells with wide fibrosis and hyalinization. The cell density of the ovarian tumor was low. Nuclear atypia, pleomorphism, and necrosis were absent. The mitotic count was <1/10 high-power fields []. Immunohistochemical staining showed strong and diffuse positive staining for α-smooth muscle actin (α-SMA) [].\nThe diagnosis was endometrial polyps, uterine adenomyosis, and intramural leiomyomas and an ovarian leiomyoma. The ovarian leiomyoma was larger than any other uterine leiomyomas, and there were no findings of metastatic leiomyomas on PET-CT. Therefore, the final diagnosis was a primary ovarian leiomyoma. The postoperative period was uneventful. Her blood test showed a hemoglobin level of 6.0 g/dL 1 day after surgery and 8.3 g/dL with iron administration 1 week after surgery. At the 6-month follow-up, the patient had no complaints and no signs of tumor recurrence. |
A 27-year-old male medical technician presented with a 12-month history of right-sided inguinal and testicular pain. The patient’s symptoms were insidious in onset with no history of trauma, injuries related to sports or work activities, medications used to account for the symptoms or familial history of genitourinary conditions. At the time of initial occurrence, the patient was seen in the primary care clinic with a complaint of painful urination with bilateral suprapubic pain, bilateral flank pain, and back pain. Pain severity ranged from 6/10 to 8/10 on a visual analogue pain scale. No hematuria or testicular symptoms were present. A microscopic urinalysis and abdominal and pelvis computed tomography (CT) scan were ordered to rule out nephrolithiasis and potential inguinal hernia. The urinalysis was normal. While no nephrolithiasis or obstructive uropathy was identified on the CT scan, a left-sided prostatic calcification was noted. The initial treatment consisted of rest and indomethacin.\nSymptoms did not improve over one week and the patient returned to his physician. A urinalysis was obtained and showed increased nitrites and protein suggesting a urinary tract infection, which was empirically treated with sulfamethoxazole and trimethoprim for three days. While there was a mild improvement initially, the symptoms reoccurred. Based on a repeat CT finding and an International prostate symptom score of 25, the differential diagnosis seemed to favor prostatitis. Over the next six weeks, the working diagnosis evolved to include pelvic pain syndrome. Further, urology and gastroenterology workup were performed and found to be non-contributory for a definitive diagnosis. The patient underwent a right cord nerve block targeting the genital branch of the genitofemoral nerve and the ilioinguinal nerve. By two days following the injection, the patient reported an increase in testicular pain at the injection site and worsening pain when the testicle was not supported. He reported waking one to two times per night due to pain.\nThe patient underwent a five-week course of pelvic floor physical therapy. Treatment consisted of pelvic floor and abdominal muscle retraining utilizing biofeedback. Biofeedback-based pelvic floor muscle re-education has been shown to be effective in treating men with chronic prostatitis and chronic pelvic pain syndrome []. Upon discharge, the patient reported improvements in his hip flexibility and urination frequency. However, there were no long-term improvements in pain and the patient continued to have pain that limited his physical activities, marital relationship, and sleep. The patient was not satisfied that all treatment options had been exhausted. He was subsequently referred to the Interdisciplinary Pain Management Clinic (IPMC) for evaluation and management. Upon presentation, the patient’s goals were diagnostic clarity, avoiding interventional injections, and resolution of symptoms.\nPhysical examination/evaluation\nPhysical evaluation by the IPMC physical therapist revealed that the patient’s pain was localized to the right inguinal area and testicle. Digital pressure applied distal to the inguinal canal at the external inguinal ring resulted in both inguinal and the testicular pain, consistent with his index pain. This finding is seen in approximately 94% of patients with a sports hernia and tenderness with palpation is part of the diagnostic criteria for the inguinal-related groin pain classification per the Doha agreement [,]. The patient completed a Defense and Veterans Pain Rating Scale (DVPRS) (Figure ) with his pain level rated at 1/10 []. The subsets of the pain supplemental questions for activity, sleep, mood, and stress were rated at the initial encounter as 2/10, 2/10, 3/10, and 3/10, respectively, and were assessed at each follow-up visit (Table ). The patient did not report any pain along the lumbar spine. The patient demonstrated a full lumbar range of motion, but he described a pulling-type sensation along the groin with end range extension. The right hip extension was decreased compared to the left side. Posterior anterior pressure applied to the spinous processes of L1 through L5 into the end range of joint motion did not change his pain.\nSacroiliac joint (SIJ) motion tests\nIn standing, the physical evaluation revealed asymmetry in the patient’s pelvic landmarks with his posterior superior iliac spine (PSIS) and iliac crest elevated on the right side as compared to the left. The SIJ motion tests utilized were the forward flexion test and the thigh thrust test. In the forward flexion test, the PSIS’s are assessed for motion as the patient bends forward from a standing position. The response is positive if the PSIS moves first and/or is higher on the painful side. For the thigh thrust, the patient is supine with hip and knee flexed. The examiner cups the sacrum with one hand and applies force axially through the knee providing a shear force to the SIJ. The test is positive if the pain is reproduced in the SIJ region. In this case, both tests were positive demonstrating right-sided SIJ hypomobility.\nStrength and flexibility assessment\nDuring the initial evaluation, the patient’s flexibility was also assessed. The patient presented with the tightness of the right iliopsoas. Manual resistance was then applied to the iliopsoas reproducing the patient’s pain mildly. This evaluative technique is recommended by Knockaert, D’Heygere, and Bobbaers as a maneuver to diagnose ilioinguinal nerve entrapment [].\nDiagnosis/prognosis\nDifferential diagnosis specific to this patient’s symptoms upon initial evaluation to the IPMC was extensive and included chronic testicular pain, sports hernia, neuropathic pain, genitofemoral nerve entrapment, ilioinguinal nerve entrapment, and abdominal cutaneous nerve entrapment syndrome (ACNES). As symptoms upon onset were painful urination and bilateral suprapubic pain, ACNES was not considered. The spermatic cord block increased the patient’s pain; therefore, nerve entrapment within the scrotum and varicocele were discarded. Similarly, the specific diagnosis of sports hernia was low on the differential due to a lack of trauma or activity-related injury; however, ilioinguinal nerve and the genital branch of the genitofemoral nerve entrapment are pathoanatomic components of a sports hernia []. Instead, tissue-based nerve entrapment proximal to the scrotum was the likely mechanism since the patient’s testicular pain was reproduced with deep pressure applied to the superficial inguinal rings, a mild reproduction of symptoms with the loading of the iliopsoas, and no benefit from the scrotal block.\nThe prognosis for full resolution of symptoms was poor given the chronicity of symptoms, the poor response to conservative measures, and the patient’s request to avoid interventional procedures. While the pain levels reported would appear comparatively low, the patient’s behavioral health and marital relationship challenges were an overriding factor in proceeding with care.\nIntervention\nThe plan of care included manual therapy, stretching, strengthening, instrument-assisted tissue mobilization (IASTM), and activity modification (Table ). The impairment-based rehabilitation program was designed to leverage osteopathic manipulation techniques (OMT) and IASTM in the clinic with a self-management program.\nUpon initial treatment, the patient underwent a common OMT directed at the right ilium to address the SIJ dysfunction (Figure ). This maneuver did correct the SIJ mechanics, but there was no change in pain symptoms with digital palpation at the external inguinal ring. Manual stretching was then applied to the right iliopsoas for 30 seconds per repetition over a 10-minute period. There was a mild decrease in pain with pressure applied to the external inguinal ring. The patient was started on a home exercise program consisting of iliopsoas stretching (Figure ). This stretch was selected to reduce the anterior translation of the ilium, which could have counteracted potential benefit from the OMT maneuver's posterior translation of the ilium. The patient was asked to follow-up seven days later.\nAt the first follow-up, the patient reported that the stretching program only provided short-term benefit. His pain level was a 3/10, which represented a two-point increase in his baseline pain level. The SIJ exam was negative. Testicular pain was again reproduced with digital pressure applied at the area of the external inguinal ring. The patient was then treated with directional cupping (Figures , ) along the length of the inguinal canal from the anterior superior iliac spine (ASIS) to the external inguinal ring so that all potential entrapment areas were addressed [,,]. To determine if this treatment was effective, the patient was asked to strength train at the gym prior to his next visit.\nBy the third visit three days later, the patient reported a small improvement in pain; however, there continued to be a 2/10 pain level after working out. The directional cupping treatment was repeated followed by manual iliopsoas stretching. The patient was placed on the left side lying position and pulled the left knee towards the chest. The provider held the right leg with knee flexed to 90 degrees, and moved the leg posteriorly until a stretch was felt. The stretch was held for 30 seconds and repeated five times. The patient was asked to follow-up in one week.\nAt the fourth visit, the patient reported no pain since the previous treatment session. The patient returned to pain-free jogging and engaged in intercourse without pain. The patient reported that he consistently stretched the iliopsoas. Upon examination, no tenderness or radiating pain was reproduced with digital pressure over the external inguinal ring. The patient performed exercises that would have previously exacerbated his pain to include squatting, lunging, and eccentric sit-ups. No pain was reproduced. The patient was asked to continue his stretching program to equalized iliopsoas length as compared to the opposite side and to follow-up as needed if any return of pain. He followed-up two days later wanting to undergo another treatment to stay ahead of re-injury. The patient reported no pain across all DVPRS subsets (Table ). The final intervention provided was an active release technique to inhibit the iliopsoas and compliment manual stretching. He was encouraged to return to the clinic for any recurrence in inguinal or testicular pain.\nThe patient was seen in the IPMC for an unrelated musculoskeletal complaint one-year following completion of therapy for this inguinal pain. He reported one episode of inguinal pain during this period that resolved after stretching of the iliopsoas and performing deep tissue massage along the inguinal canal utilizing a Thera Cane (Thera Cane Company, Denver, CO). |
A 19-year-old African-American female presented to the emergency department for evaluation of subacute onset of left-sided hemianesthesia that started in the left hand and gradually progressed proximally to the entire left upper and lower extremities, and then the right side of the body over a course of two weeks. The patient reported associated weakness and bifrontal throbbing headache. She also reported decreased bowel and bladder sensation, but no episodes of bowel or bladder incontinence.\nOn physical examination, the patient had 4/5 strength in the left upper extremity and 5/5 strength in the right upper extremity. She had decreased sensation to light touch on the left side of her body, and a mild decrease in rectal tone.\nMRI of brain and spine showed sharply delineated ovoid mass within the spinal cord at the level of C3 that measured approximately 1.7 cm (SI) x 6.4 mm (AP). This lesion had high intensity on T2 and was isointense to the spinal cord on T1, with partial central enhancement. No syringomyelia or surrounding cord edema was identified, but the cord was expanded at the level of the mass (Figure ).\nBased on the radiological findings, low-grade astrocytoma, ependymoma, and inflammatory or demyelinating lesion were in the differential diagnosis.\nThe patient was then started on steroids and cerebrospinal fluid (CSF) was obtained. CSF analysis was positive for oligoclonal bands. This raised the suspicion for a demyelinating lesion, and the patient was treated with combination of high-dose steroid and five plasma exchange sessions.\nAfter completing the treatment inpatient, the patient started to have significant improvement in her left side weakness and numbness. She was cleared by physical and occupational therapy to be discharged home. At two-month follow-up, the patient continued to do well, and the repeat MRI showed mild reduction in the size of the lesion and no contrast enhancement, indicating an inactive inflammatory lesion (Figure ). |
A 70-year-old female presented to a rural hospital with painless jaundice. She had undergone a routine open cholecystectomy 30 years earlier at the same hospital. Biochemical markers showed an obstructive pattern: Bilirubin 155 μmol/L (6–24), ALP 262 U/L (30–110), ALT 272 U/L (0–55), AST 161 U/L (0–45). The white cell count was within the normal range at 6.65 × 109/L (4–11), and the CRP was mildly elevated at 11 mg/L (<8.0). In the 30-year interval, she had experienced no other episodes of jaundice, abdominal pain, or unexplained fever and had undergone no other abdominal surgery. Unfortunately the hospital records (on microfilm) were incomplete so no operation note from the cholecystectomy 30 years previously was available. The anaesthetic chart and drug chart were retrieved and revealed that no antibiotics were given and no specific intraoperative difficulty or postoperative complications were recorded in the clinical records. The patient also confirmed an uneventful recovery.\nShe was referred to the gastroenterologists at a tertiary centre for endoscopic retrograde cholangiopancreatography (ERCP). The images at ERCP demonstrated a round filling defect approximately 2 cm in diameter associated with a smooth stricture consistent with extrinsic compression in the proximal bile duct (). The common bile duct was cannulated without difficulty and measured 8 mm in diameter. A guidewire was easily placed past the area of compression and insertion of a 10 French; 12 cm Cotton Leung biliary stent was performed. The jaundice resolved within 72 hours of the procedure. No antibiotics were given at the time of the ERCP. No malignant cells were identified from the aspirated bile sent for histology. The patient was discharged the day following the ERCP and referral to the hepatopancreaticobiliary surgical team was made for further investigation into the cause of the jaundice.\nCT imaging demonstrated mild intrahepatic dilatation. No lesions other than a rounded soft tissue mass immediately lateral to the stent and below the porta hepatis were identified (). This was consistent with the ERCP findings. The mass measured slightly smaller on the CT images at approximately 1.5 cm in diameter. The mass had varying density but seemed complex in nature and as such the possibility of a choledochal cyst was raised. Diagnostic laparoscopy was performed one month following the ERCP. The view was obscured by adhesions from the previous open cholecystectomy completed via a right paramedian incision. No antibiotics were given at the time of the laparoscopy. An esophagogastroduodenoscopy and colonoscopy excluded primary malignant lesions in those areas. Following extensive discussion at the hepatopancreaticobiliary multidisciplinary meeting and with the patient, it was decided that due to the diagnostic uncertainty of the obstructing tumour in the hepatic duct, laparotomy and attempted excision of the lesion should be performed.\nAt operation, the common bile duct was identified and the lesion located within the proximal bile duct at the usual position of the insertion of the cystic duct, though the cystic duct stump itself could not be identified. The dimensions of the lesion were similar to those measured on the CT images and the common bile duct again measured 8 mm in diameter. During dissection pus started exuding from the duct wall. A sample was sent for microbiological analysis. No bile was seen in the purulent discharge and probing of the underlying cavity did not reveal a communication with the common bile duct. Because of the possibility of malignancy, the common bile duct was opened and choledochoscopy showed normal primary and secondary bifurcations of the common bile duct, a normal duct distally and no apparent communication with the abscess cavity. The lateral wall of the abscess cavity was resected and sent for histology. This confirmed mild, chronic nonspecific inflammation and fibrosis only. The stent placed at ERCP three months previously was removed and the tip sent for culture. Because the medial wall of the common bile duct was found to be thinned and delicate due to the abscess formation and consequent resection, a T-tube was inserted and sutured into position.\nIntravenous cephazolin and metronidazole were given intraoperatively as part of the hospital microbiology guidelines for antibiotic prophylaxis of biliary tract surgery. These antibiotics were given for 4 days postoperatively with the addition of gentamycin, whilst culture results were awaited. Klebsiella ornithinolytica and Enterococcus casseliflavus were subsequently identified and were sensitive to the prescribed antibiotic regime. The postoperative recovery was uneventful with no episodes of sepsis. A T-tube cholangiogram thirteen days postoperatively showed no abnormalities (). The tube was clamped following the cholangiogram and removed without incident two weeks later. |
A 63-year-old female with no significant past medical history presented to our facility with headache and neck stiffness. The headache progressed to being the most severe pain she had ever experienced. She was also mildly lethargic but did not exhibit any focal deficits. She denied history of trauma or use of aspirin or blood thinners. Initial brain computerized tomography (CT) demonstrated evidence of scattered subarachnoid hemorrhage and diffuse cerebral edema which was most prevalent within the posterior fossa. Subsequent brain magnetic resonance imaging (MRI) with and without contrast demonstrated persistent right superior cerebellar hemisphere subarachnoid hemorrhage with associated stable parenchymal hematoma and evidence of a distal flow-related right superior cerebellar artery aneurysm secondary to an underlying posterior fossa AVM. Cerebral angiography showed two ruptured flow-related aneurysms on an enlarged branch of the right superior cerebellar artery associated with a small tectal AVM (Figure ).\nHowever, initial attempts at endovascular treatment were unsuccessful due to profound tortuosity of the left vertebral artery, which was the only potential transfemoral or transradial access to the flow-related aneurysm (Figure ).\nGiven the marked tortuosity of the vessels, the patient underwent treatment of the flow-related aneurysms beginning with percutaneous access of the vertebral artery at the C1 level, obtained using roadmap guidance from selective catheterization of the ostia of the left vertebral artery (Figure and Figure ).\nUsing a standard microcatheter system over a microcatheter guidewire, a super-selective catheterization of the two flow-related aneurysms was performed (Figure ).\nMultiple detachable platinum coils were then deposited into the aneurysmal branch resulting in occlusion of the vessel. Final control angiography revealed marked stasis in the two flow-related aneurysms well into the venous phase suggesting that they will rapidly thrombose (Figure ).\nThe patient subsequently underwent surgical resection of the AVM and made complete neurological recovery. Repeat angiographic evaluation at six months showed no evidence of vascular abnormality (Figure ). |
A 59-year-old male patient was admitted to coronary angiography for recurrent chest pain for 1 day. Based on ECG and elevated troponin T, acute coronary syndrome without ST elevation was diagnosed. He had no significant past medical history. Using the right radial approach and a Judkins diagnostic catheter, the very short LMCA was visualized with almost independent take-off of the left anterior descending coronary artery (LAD) and left circumflex coronary artery (LCX). First selective contrast injection into the LCX did not show any significant lesions. In order to selectively engage the LAD, excessively deep catheter intubation was performed, which caused iatrogenic LMCA dissection, propagating to the LAD and proximal LCX. Acute LAD closure and contrast extravasation was visualized (). Immediate, percutaneous attempts to open the LAD were undertaken. However, despite support with inflated 1.25 mm and 2.0 mm balloons, it was impossible to insert any of the different guidewires into the true lumen of the LAD or diagonal branch. Thus no contrast flow in the mid/distal LAD segments was restored. The patient developed CS, requiring continuous infusion of inotropes, intubation and mechanical ventilation. Thus, he was immediately transferred to a tertiary hospital with the department of cardiac surgery, where rescue off-pump coronary artery bypass grafting (CABG) was performed just a few hours after dissection. Entering the operating room the patient had a sinus rhythm with a heart rate of 120 beats/min and blood pressure of 85/55 mm Hg. The LAD dissection extended to the mid segment, distal to which the vein graft was sewn. The second vein graft was sewn to the obtuse marginal artery. The postoperative course was characterized by progressive impairment of the left ventricle (LV) systolic function. The LV ejection fraction (LVEF) decreased from 25% preoperatively to only 10–15% in the early postoperative period. No repeat early post-CABG coronary angiography has been performed to document graft patency. Maximal troponin T was 20650 ng/l (UNL < 14) and the peak lactate was 84 mg/dl (UNL < 20) on the second day. An intra-aortic balloon pump (IAPB) was inserted percutaneously through the left femoral artery. Due to progressive haemodynamic instability despite maximal medical therapy and IABP support, an ECMO was inserted on the second postoperative day through the full sternotomy, utilizing an 8 mm graft sewn to the ascending aorta and a VAD venous cannulae introduced into the right atrium to salvage the patient. The ECMO circuit was based on a centrifugal pump and a hollow-fiber membrane oxygenator. All circuit components were heparin surface coated. After surgical bleeding had been controlled, continuous infusion of heparin was initiated with the activated partial thromboplastin time target of 40–60 s. The pump flow was maintained at 2.5 l/min/m2, obtaining a mean systemic pressure of 60–70 mm Hg. Packed red blood cells and fresh frozen plasma were transfused as required. Haematocrit was maintained above 30% to optimize oxygen delivery. No exploration for bleeding was necessary. The intensive care unit course was characterized by modest but continuous clinical improvement and LVEF increase up to 35–40%. The decision for ECMO weaning was undertaken on stable haemodynamic and inotropic support, with echocardiographic absence of tamponade and of left heart distension. Weaning was done in the operating room. The ECMO flow was gradually reduced under continuous transesophageal echocardiography monitoring of LV function and finally stopped 14 days after its initiation. Simultaneously systemic blood pressure, filling pressures and cardiac output were monitored during the process. The patient was decannulated after cardiac function stabilization and adequate gas exchange was confirmed. He was extubated on the first postoperative day, and discharged after 35 days.\nDuring 4 months of follow-up the patient required three hospitalizations for exacerbation of heart failure. The NT-pro-BNP varied from 4883 pg/ml to 15 000 pg/ml. Peak oxygen uptake was only 8.3 ml/min/kg (34% of predicted normal value) on the cardiopulmonary exercise testing. Thus, the patient is considered a candidate for orthotopic heart transplantation (OHT). |
A 62-year-old woman was referred to the authors' hospital for evaluation of a corneal ulcer in her left eye. With the patient's consent, a review of the clinic record was conducted. Ten years previously she received an allogeneic hematopoetic stem cell transplant for treatment of myelodysplasia syndrome. Her course was complicated by the development of cGVHD affecting the liver, skin, esophagus, mouth, and eyes. Severe keratoconjunctivitis sicca had been treated with a PROSE lens for more than 2 years prior to developing this corneal ulcer. Her medical status was compromised by systemic steroid dependence and by steroid induced diabetes mellitus.\nAfter presenting with a corneal ulcer in her left eye, the patient was empirically treated with topical 0.5% moxifloxacin (Vigamox, Alcon) that was applied 6 times a day (once before the PROSE lens was inserted in the morning, 4 times during the day inside of the PROSE lens reservoir, and once again at night after the PROSE lens was removed). After failure to improve on 4 days of this treatment, she was referred to Massachusetts Eye and Ear Infirmary for additional corneal ulcer evaluation, culture and modification of treatment. Upon presentation, the central cornea was opaque and neovascularized with tissue loss of approximately 60% of the corneal thickness. There was an epithelial defect that measured 2.5 mm by 1 mm, with an underlying 2 mm by 1 mm infiltrate. Microbiological smears and cultures were performed with the specimen from corneal scraping. No bacteria or fungi were evident with Gram stain or Calcofluor white stain. At the time, frequency of application of topical moxifloxacin was increased to every 2 hours while awake (approximately 8 times per day), delivered as one drop added to the PROSE reservoir after removal and cleaning reinsertion of the device and replenishment of the reservoir with preservative free saline (B).\nFour days after culture and modification of antibiotic delivery regimen, the corneal ulcer resolved, with re-epithelialization of the cornea surface and resolution of the infiltrate. A strain of E. coli cultured from the lesion in 5% sheep blood agar media exhibited resistance to fluoroquinolones, trimethoprim/sulfamethoxazole and ampicillin and ampicillin-sulbactam according to breakpoints set by the Clinical Laboratory Standard Institute-CLSI (). The strain carried multiple single point mutations in the quinolone resistance-determining region (QRDR) of gyrA (Ser83Leu, Asp87Asn), parC (Ser57Thr, Ser80Ile) and parE (Leu416Phe) genes. These mutations were correlated with varying levels of resistance to the fluoroquinolones: ciprofloxacin (MIC 256 μg/mL), levofloxacin (8 μg/mL) and moxifloxacin (16 μg/mL), as determined by reference broth microdilution. |
A 46-year-old male with no prior medical history presented to UC Irvine with a several month history of episodes of right and left hemiparesis, progressive bulbar weakness, paresthesia, dysarthria, and headache. He also reported a 30-pound unintentional weight loss. He was admitted to the neurology service and underwent an extensive evaluation for autoimmune, demyelinating, vascular, and neoplastic processes. MRI brain demonstrated several small discrete foci of restricted diffusion in the white matter, enhancement in large portions of the bilateral corticospinal tracts, and significant pontine involvement (). Lumbar puncture demonstrated CSF pleocytosis and elevated protein. Histology of the right frontal brain lesion was consistent with diffuse large B-cell lymphoma (DLBCL), and a bone marrow biopsy demonstrated normocellular marrow. The patient was diagnosed with primary diffuse CNS lymphoma (PCNSL) and started on high dose steroid therapy.\nEarly in the hospital course the patient was found to be in acute respiratory distress presenting with tachycardia, tachypnea, and increased work of breathing. He was emergently intubated and transferred to the neuro ICU. CT angiogram of the chest was negative for pulmonary embolism but demonstrated interval left lower lobe consolidation. The patient was placed on multiple conventional ventilator modes with a set tidal volume of 400 ml and pressure support of 5 cm H2O. Despite these standard settings, large tidal volumes were observed ranging from 700 to 1,400 ml. Initial arterial blood gas (ABG) was significant for a pH of 7.61 and PaCO2 of 13.1 mmHg. Due to the significant hypocarbia, a number of ventilator tubing segments were added to increase the mechanical dead space and increase the rebreathing of exhaled CO2. In addition, whole brain radiation and treatment with rituximab were initiated. Before these interventions, the patient's neurological exam was poor. He did not follow commands or open eyes to noxious stimuli. However, the patient had intact brainstem reflexes and trace extremity movement to noxious stimuli.\nCO2 was monitored with ABG and immediate response to the increase in dead space was measured with VBG. Before intervention, VBG showed pH 7.57 and PvCO2 18.4 mmHg which immediately showed signs of improvement after 1 day. Two days after the increase in dead space a follow-up ABG showed pH 7.45 and PaCO2 30.5 mmHg. Over the coming days the patient required ventilator and dead space adjustments as he was weaned off of fentanyl and midazolam to dexmedetomidine and oxycodone.\nThe patient required a tracheostomy due to prolonged ventilator time, but 2 weeks after the initiation of dead space therapy the patient was weaned from the ventilator with T-piece uncapped. He was off sedation and had been weaned off of opioids. The patient was transferred to the step-down unit with ABG showing pH 7.51 and PaCO2 28.5 mmHg without tachypnea. In those 2 weeks the patient's neurological exam had improved significantly, and he was awake, alert, and attending to the examiner. He followed motor commands in all extremities (distal strength better than proximal strength) and also followed commands to close eyes and stick out tongue. On discharge 1 week later (3 weeks after the increase in dead space) he was smiling and interactive with family. |
We report a case of 30-year-old Caucasian woman in her first pregnancy with a BMI of 25. The couple were nonconsanguineous. The partner had been tested in the past and was found to be a carrier of a balanced translocation that involves chromosomes 7 and 12. Partner's brother had an unbalanced form of this familial translocation between chromosomes 7 and 12 with severe learning disability. Also partner's mother was found to be a carrier of balanced translocation that involved the same chromosomes.\nWoman's dating ultrasound at 11 weeks and 3 days showed an intrauterine pregnancy with a CRL (crown rump length) of 47.5 mm and a nuchal translucency of 2.0 mm. The risk of trisomy 21 was reported to be 1 : 3,300.\nA routine anomaly scan was performed by the fetal medicine specialist at 20 weeks and 3 days in view of the family history of balanced and unbalanced translocations. Anomaly scan revealed several abnormalities (). There was marked microcephaly and alobar holoprosencephaly. Multiple facial abnormalities were detected including an absent nasal bone, microphthalmia, hypotelorism, flat profile, and single nostril. The bowel appeared hyperechogenic. All the other systems appeared normal.\nThe patient and her partner were counselled regarding diagnosis and poor prognosis. Due to the brain abnormalities, the prognosis was deemed to be poor with a high likelihood of miscarriage, neonatal death, handicap, and learning disability. In view of the multiple abnormalities, the possibility of an underlying chromosomal or genetic abnormality was also discussed with the couple.\nThe patient opted for an amniocentesis and it was performed on the same day with no complications. The patient and her partner opted for termination of pregnancy in view of associated fetal brain abnormalities. A medical termination regime was commenced and completed four days later with the delivery of a male infant weighing 320 grams. The couple opted for a full postmortem examination. |
A three-day old female patient in the nursery ward was consulted for assessment of multiple, red skin lesions on the chest and left shoulder. The baby was born full-term and by a spontaneous vaginal delivery. Soon after delivery the patient noticed to have low platelet count for which a pediatric hematologist was consulted, and the patient diagnosed with autoimmune thrombocytopenia, treated with platelet transfusion and intravenous immunoglobulin (IVIG). Two of the patient's siblings were also born with similar skin lesions and diagnosed with autoimmune thrombocytopenia (Figure ). Her grandmother and two of the patient’s aunts had a history of similar lesions, which were stable since birth. On receiving the mother's history, all her previous pregnancies were complicated with gestational thrombocytopenia (Figure ). The patient's parents showed positive consanguinity. All affected family members are completely normal with normal development/growth and no systemic symptoms. On examination, the patient was vitally stable with normal growth parameters, no syndromic features, macro/microcephaly, or limb deformities. The physical examination showed well-defined red to rose-colored patch over the left shoulder and arm measuring 4 cm x 3 cm with irregular borders (Figure ); also, there were multiple smaller round to oval erythematous macules and patches on the right side of the chest and over the right knee (Figure ). There was no tenderness or thrill. Nails, mucous membrane, and hair examination were unremarkable; the patient showed a normal systemic examination including eye and neurologic examinations. Histopathologic findings showed dilated capillary venular channels in the upper dermis and no arteriovenous malformations. Ultrasound was done for the patient, and the report revealed dilated dermal postcapillary venule with an increase in Doppler flow compared to the adjacent dermis. There were no fistula and no arteriovenous or lymphatic malformations. Genetic studies were not done due to the patient's family refusal. The patient was finally diagnosed with a distinct type of capillary malformation-arteriovenous malformation syndrome newly named as rhodoid nevus. The patient's parents were reassured and a pulse dye laser treatment was offered to be done in a later stage of the patient's life. |
This is a case report of a 46-year-old female with a past medical history notable for depression, asthma, and uterine leiomyomas who presented to an urgent care with 5 days of progressive abdominal pain, bloating, nausea, and subjective fevers. The patient endorsed a several month history of gaining weight, though she attributed it to her lifestyle, accompanied with strong, intermittent, crampy right lower quadrant pain. The pain was random in onset and would dissipate very quickly. However, 5 days prior to presentation, her pain dramatically increased and was persistent in nature.\nHer surgical history included a myomectomy performed 5 years prior followed by a laparoscopic hysterectomy 2 years later. She was a regular drinker, consuming four to five alcoholic drinks per night, but stopped when her symptoms worsened and had no history of withdrawal. Family history was significant for a maternal grandmother with breast cancer, mother with skin cancer, and an uncle with colon cancer.\nUpon arrival to the emergency room, the patient was tachycardic, mildly hypotensive, and febrile to 103 °F. Her abdomen was soft, distended, and diffusely tender without peritonitis. Labs were notable for a leukocytosis of 15 K. CT of the abdomen and pelvis demonstrated moderate volume, complex fluid within the abdomen and pelvis with extensive amount of gas and peripheral rim enhancement, a thickened appendix filled with fluid, and a soft tissue/cystic lesion in the anterior abdominal wall (Fig. ).\nDifferential included pseudomyxoma peritonei with a ruptured appendiceal mucocele versus PMP secondary to an adnexal ovarian neoplastic pathology with an infectious component. Per the radiology report, the origin was unclear based upon imaging and stated a ruptured adnexal cyst should be considered given the markedly enlarged septated cystic lesions in the pelvis. The patient was resuscitated, and her blood pressure and heart rate normalized with 2 L of fluid. Given the patient’s hemodynamic stability, the decision was made to attempt conservative management with antibiotics and interventional radiology (IR) biopsy and drainage.\nOn the floor, the patient remained hemodynamically stable with intermittent fevers. Her abdominal exam also remained unchanged, with persistent pain that was under control with pain medication. It was unclear if the abdominal wall mass was neoplastic, and given our initial nonoperative approach, a biopsy was performed for tissue diagnosis to properly guide further treatment. A core needle biopsy of the abdominal wall mass demonstrated a uterine leiomyoma implant, and the cytology aspirate of the peritoneal fluid showed pools of acellular mucoid material. On hospital day 7, the patient’s leukocytosis rose to 23 K, and repeat imaging demonstrated a more organized collection with intraperitoneal air in addition to multiple thick, wall-enhancing, complex cystic multiseptated lesions. Given these findings on imaging as well as her clinical presentation, the patient was taken to the operating room (OR) jointly with surgical oncology and gynecology oncology for a peritoneal washout and bilateral salpingoopherectomy. Intraoperative findings included a large amount of mucin in the abdomen and pelvis with a pocket of purulent fluid in the mid-abdomen, a very large (12 cm) cystic left ovary, dilated right fallopian tube with enlarged right ovary, a very dilated and thickened appendix, and dense, diffuse small bowel intraabdominal adhesions (Fig. ). Her peritoneal cancer index (PCI) score was unable to be determined due to the degree of inflammatory adhesions. She underwent bilateral salpingoopherectomy given the amount of inflammation in the cecum and base of the appendix, the decision was made not to perform an appendectomy to avoid leakage at the staple line and further infectious complication. The patient’s abdomen was hostile, and no larger operation was deemed necessary at this time, as the patient was nonobstructed and the origin of PMP was not yet verified. The primary goal of the operation was to clear the infection, with the intent to return at a later date when her intraabdominal contents could be properly mobilized for a complete PCI score and an appropriate oncologic resection could be performed.\nHer hospital course was complicated by recurrent pleural effusions requiring multiple thoracocenteses, negative for cytology, and persistent need for supplemental oxygen. She also developed a secondary intraabdominal abscess requiring IR-guided drainage. After the patient clinically improved, she returned to the OR 2 months after her initial presentation for completion cytoreductive surgery (CRS) and hyperthermic intraperitoneal chemotherapy (HIPEC) with 40 mg of mitomycin-C at a target temperature of 42–43 °C over 90 min, as per our institutional protocol []. Her PCI at the second surgery was calculated to be 13. She had minimal peritoneal adhesions, and her intraabdominal infectious process had completely resolved. The patient underwent an appendectomy, omentectomy, and tumor debulking. The appendicular base was healthy and easily stapled across. The patient was discharged home with lovenox on postoperative day 5, saturating well on room air, tolerating a diet with oral pain medication, and with return of bowel function. Six months after the CRS/HIPEC, the patient had surveillance imaging with no evidence of recurrence (Fig. ). The abdominal wall mass was unchanged from prior imaging and, given the biopsy of leimyoma, presumed to be an implant at the port site from the patient’s prior hysterectomy.\nAfter the discharge from the second surgery, the patient’s postoperative recovery was complicated by new-onset shortness of breath after the completion of her 30 day course of lovenox. She was diagnosed with bilateral pulmonary emboli, with no evidence of deep vein thrombus on lower extremity duplex. Therapeutic lovenox was initiated, and her symptoms gradually improved. An interval angio CT of the chest demonstrated resolution of the clot. Anticoagulation has since been discontinued, and the patient will continue with 6-month interval surveillance imaging for PMP recurrence.\nThe pathology of the left ovary and tube following the first operation resulted as an ovarian cyst containing mucin pools and low-grade intestinal type mucinous glands, consistent with metastasis from appendiceal mucinous neoplasm. The right ovary and tube showed mucin, acute inflammation, and adhesions. Immunostains were positive for CK7, CK20, and CDX2 and negative for PAX8 (Fig. ). Following the completion cytoreduction, the appendiceal pathology demonstrated a low-grade appendiceal mucinous neoplasm (LAMN) with acellular mucin outside the appendix and associated fibroinflammatory response. The proximal margin, the appendicieal base, was negative for tumor. Additional specimens sent during the debulking included the falciform ligament, omentum, pelvic tumor nodules, and tumor deposits overlying the sigmoid, small bowel, left colon, and liver. All additional specimens consisted of acellular mucin. Pre-operative carcinoembryonic antigen (CEA) and CA-125 were elevated at 26.8 and 101 respectively. CA 19-9 was within the normal limits. Using AJCC (8th ed.) staging, the final staging was determined to be T4a NX M1a (stage IVA). Since the patient had a complete cytoreduction and the pathology was determined to be LAMN, no further adjuvant therapy was given. |
A 53-year-old female patient was diagnosed with stage IIIC (pTxpN3M0) melanoma involving left inguinal lymph nodes after biopsy in March of 2012. Work-up did not reveal a primary site. She was treated with lymph node dissection with a plan for adjuvant immunotherapy, but she rapidly developed metastatic adenopathy in multiple sites, including her left iliac and right axillary chains (biopsy confirmed in latter site). She participated in a phase II clinical trial with high dose IL-2 followed by intermittent low dose temozolomide. The patient was noted to have BRAF V600E mutation and an M1a pattern of spread; with marked LDH elevation. She received one course of therapy but had marked progression with new sites of disease in multiple lymph nodes, subcutaneous and mesenteric nodules by October 2012. She did develop vitiligo post IL-2 treatment. The patient was then started on ipilimumab, but had marked progressive disease after 2 cycles, as shown by PET/CT in December 2012 ( left). She developed bone, adrenal gland and splenic metastases, as well as worsening of existing lymph node metastases (M1c pattern). We continued the last 2 cycles of ipilimumab and began concurrent vemurafenib at 960 mg twice daily, given the massive symptomatic disease progression, along with radiation therapy (RT) to the larger masses in the cervical and inguinal regions (30 Gy in 10 fractions). The RT was done both for palliation and for synergism with ipilimumab, as it could theoretically render the tumor more immunogenic. She rapidly achieved a clinical response to vemurafenib (along with a pruritic rash requiring a dose reduction and a brief course of steroids). The patient developed fevers, chills and marked synovitis in a symmetric fashion, particularly in her wrists, hands and feet. She completed a course of ipilimumab, with 4 doses at 3mg/kg every 3 weeks. The patient did not develop autoimmune hepatitis but her vitiligo became more prominent () and she also developed autoimmune hypophysitis, which required hydrocortisone replacement. She had a transient positive antinuclear antibody test. PET/CT scan in February 2013, 3 months after the vemurafenib therapy, showed CR (, right). We gradually weaned the vemurafenib for 3 reasons: 1) the arthritis was nearly incapacitating despite dose reduction of the vemurafenib and initiation of celecoxib, 2) the patient had signs of marked autoimmunity, suggesting that there could be a beneficial synergy with the prior immune therapy and 3) in theory, to forestall development of resistance to the vemurafenib. At 3 months, we began treatment alternating one week on then one week off of the agent. Subsequent PET scans revealed persistent CR. She received a total of 12 months of intermittent vemurafenib treatment. Her arthritis did not improve on the weeks she was off therapy and has persisted to date but is gradually reducing in severity over time. She remains in CR at 15 months from the completion of the vemurafenib course. |
A 90-year-old male presented to the emergency department with complaints of midepigastric and right upper abdominal pain. According to the patient the pain started the same day; it was a rapidly developing discomfort which he described as an “aching sensation.” There was associated nausea with 2-3 bouts of vomiting. He denied fever or chills. He did move his bowel twice since the pain started. Past surgical history was significant for history of right hemicolectomy for carcinoma colon, sigmoid colon resection for diverticulitis, laparoscopic cholecystectomy, appendectomy, cataract, and history of small bowel obstruction leading to small bowel resection 2 years back. Physical examination revealed an elderly white male patient. Vitals include temperature of 96.3 F, pulse rate of 87, respiratory rate of 27, and blood pressure on presentation of 194/83.\nThe abdomen was grossly doughy and mildly distended. However on palpation there was diffuse tenderness across the abdomen, especially in the right upper quadrant with guarding but no definite rebound.\nSerum biochemistry was within normal limits.\nPreoperative imaging consisted of X-ray abdomen followed by the CT scan.\nA plain film abdomen () showed diffuse gaseous distention of bowel with loops of dilated small bowel measuring up to 6.5 cm in diameter. A large hiatal hernia distended with gas containing the stomach along with possible bowel protruding into the hernia was also seen.\nThe CT scan (Figures and ) showed large type IV hiatus hernia, which was distended by fluid and gas. Based on the new finding of air-fluid level containing fluid crescent communicating with the general peritoneal cavity, there was suspicion of bowel perforation relating to either the hiatus hernia or the small bowel obstruction.\nBased on physical examination and imaging, decision was made to take up the patient for surgery. Generous midline laparotomy incision was made; due to prior surgeries and significant adhesions, extensive lysis of adhesions was done. All herniated small bowel and stomach protruding through the hiatus were reduced back into the peritoneum. The perforation was identified in proximal diverticula of the jejunum. Due to limited length available and advanced age of the patient, wedge resection of the diverticulum was performed and it oversewn in two layers. Hiatus was repaired with interrupted figure of "eight" stitches using #0 Prolene suture. No mesh was placed because of perforated viscus. The patient tolerated the procedure well. He was started on clears on postoperative day 4 which he tolerated and gradually advanced to regular diet. The patient was discharged on postoperative day 7. |
A 15-year-old boy was diagnosed to have mitral regurgitation with a floppy anterior mitral leaflet. He was also found to have retinitis pigmentosa and Marfan's syndrome. His exercise tolerance was adequate, and he did not have any symptoms suggestive of pulmonary hypertension or heart failure. No symptoms suggesting unstable atlanto-occipital joint were present. He was scheduled for mitral valve replacement. The decision to replace the valve as opposed to a repair was taken because extensive annular calcification was noted in the preoperative transthoracic echocardiography.\nAfter induction of anesthesia, right IJV cannulation was attempted in the standard position with the insertion point at the apex of the triangle formed by the sternal and clavicular heads of sternocleidomastoid using triple lumen catheter set (Arrow, 7F) using Seldinger's technique.\nIn the first attempt, blood was freely aspirated at around 4 cm depth with the finder needle and the introducer needle. During insertion of the guide-wire mild resistance was felt at about the depth the guide-wire was expected to enter the vein. However, the senior resident (who had nearly 3 years of experience after postgraduation) doing the cannulation, managed to thread the guide-wire in, with minimal manipulation. Following dilation, the catheter was inserted over the guide-wire without any difficulty. Since a total of five operating rooms share a single ultrasound machine, it was not available for immediate use at that point of time. It was decided to fix the catheter at 10 cm, but blood could be aspirated from only one side port of the triple lumen. Withdrawing the catheter did not help, and the guide-wire was inserted and the catheter once again advanced. Free flow of blood was not obtained even with this, and the catheter was removed and pressure applied.\nThe landmark was re-identified, and the finder needle inserted. The initial attempt was unsuccessful, and the angle of the needle was reduced. Blood was aspirated at the same depth as in the previous attempt, and free flow was observed. After passing the guide-wire freely, dilation of the vein was done with the dilator. The catheter was passed again to 10 cm and blood could be aspirated from only one lumen, this time from the distal one. A small amount of saline was pushed through the other lumens, and there was no resistance. Again misplacement of the central venous catheter was suspected, and the subsequently catheter was removed and the pressure was applied at the insertion site. A more medial point of entry was chosen (medial to the sternal head of sternocleidomastoid).[] The cannulation was done this time without difficulty. All the three lumens were patented this time as evident by free aspiration of blood. No swelling was found above the clavicle. The patient was hemodynamically stable, and the surgery proceeded. Before going on bypass after heparinization, the only thing noted was a slightly lower blood pressure (BP) of 90/50 mm Hg and a relatively low central venous pressure (CVP) of 3–4 cm of H2O.\nRelative hypotension was ensured at the time of aortic cannulation to prevent dissection of the aorta which is possible in Marfan's syndrome. Operating surgeon was able to trace the tip of the catheter inside superior vena cava (SVC) during CPB. On weaning from bypass, after removing the SVC cannula, it was noticed that the medial portion of the right side pleura was bulging into the operating field. Initially, seepage of blood into the pleura from the operating field was suspected, and pleura was opened. About 1.5 L of clots and blood was removed. At this point, the BP fell suddenly from 90 to 50 systolic. A single, 2 mm tear in the right subclavian vein at the portion where it crosses the apex of the lung was noticed.\nThe tear was sutured in about 10 min and the BP improved. Adequate replacement of packed red cells was done, and the patient was separated from bypass uneventfully. The postoperative course was uneventful. |
A 48-year-old African American woman with a past medical history of autoimmune hepatitis and possible overlap syndrome with primary sclerosing cholangitis status post multiple stent placements and endoscopic retrograde cholangiopancreatography (ERCP) presented with tearing, redness, and irritation of the left eye. She was diagnosed with autoimmune hepatitis for over ten years before the visual symptoms began and was on a low-dose steroid maintenance treatment for the condition. She had strictures that extended to her intrahepatics with accompanying cholangitis. Histological examination confirmed hepatitis, and laboratory values confirmed a cholestatic liver enzyme profile. She also was positive for anti-nuclear and anti-smooth muscle antibodies and negative for hepatitis B and C. Visual acuity was 20/20 in the right eye and 20/40 in the left eye, and the intraocular pressures (IOPs) were 21 and 19 mmHg, respectively. Slit lamp exam showed a crescent-shaped left peripheral corneal ulcer measuring 4.5 × 0.5 mm extended from 5 to 6:30 o'clock with about 50% thinning and no anterior chamber reaction. The adjacent sclera was also inflamed with injected and edematous conjunctiva. The remainder of the eye exam was unremarkable. The patient had previously been on 10 mg of oral prednisone for prior medical history but was started on topical prednisolone acetate 1% eye drop four times daily. Prophylactic topical antibiotic eye drops, moxifloxacin, and oral doxycycline 100 mg twice daily along with preservative artificial tears were also started.\nOver the next 6 weeks, there was continued worsening of the ulcer. One week after initial presentation, the patient returned with no improvement. Three weeks later, the ulcer base became widened and deepened with significant thinning of about 70%. At this time, the patient was started on azathioprine 150 mg and prednisone 60 mg daily by her rheumatologist. One month later, there was continued worsening of the ulcer extending to the 3 o'clock position with about 70–80% thinning (). Moreover, her right eye also showed a small area of ulceration along the limbus at the 2 o'clock position. Because of lack of progress and concern of poor medication compliance, the patient was admitted to the hospital for intravenous methylprednisolone and rituximab treatment by her rheumatologist.\nWhile the right eye ulcer started epithelialization, the left eye showed no improvement. The patient underwent conjunctival resection in the area of ulcer in the left eye with application of fibrin glue and a bandage contact lens. Meanwhile, the patient finished a course of 1000 mg rituximab infusion along with intravenous methylprednisolone sodium succinate, 1 gram daily for 3 days, and discharged on mycophenolate mofetil and a tapering dose of oral prednisone. A month after the procedure, the ulceration in the left eye had epithelialized to about 2 or 3 clock hours, and within a subsequent month, the lesion had healed entirely with residual thinning. Visual acuities were 20/20 OD and 20/30 OS.\nThe patient was monitored on a monthly basis afterwards. After five months, the patient presented with 20/100 vision in the left eye. Examination showed a small linear corneal perforation near the limbus with iris prolapse at the site of previous ulcer in the left eye. The perforation was repaired with direct closure. The patient was discharged on mycophenolate mofetil. A month after the surgery, the corneal perforation had healed with superficial neovascularization, and there was a small area of iridocorneal adhesion. In a three-month follow-up visit, uncorrected visual acuity in the left eye was 20/100 that was corrected to 20/40. The intraocular pressure was measured to be 16 mmHg. There were no signs of recurrent inflammation or ulceration. |
A 72-year-old Caucasian female patient was referred to the respiratory clinic with a 6-month history of exertional breathlessness. She gave a history of yearly chest infections, particularly in winter months. She, however, had no history of childhood respiratory problems or any significant respiratory symptoms during her adult life. She had a 30-pack-year smoking history but she quit 25 years prior to presentation. She worked as a nursing auxillary at a local hospital and had no prior asbestosis. She was on a 1 mg maintenance dose of prednisolone for polymyalgia rheumatica. Otherwise, she had no significant medical condition and was not on any regular medications. Her exercise tolerance was unlimited and she enjoyed a good health.\nHer O2 saturation was 95% on room air and her spirometry showed obstructive pattern with FEV1 1.14L (55%), FVC 2.09L (83%) and FEV1/FVC ratio 54%.\nAn initial chest radiograph 2 years prior to presentation suggested an increased radiolucency of the left lung but this was not investigated further (). Serial CXRs 2 years later showed a relatively unchanged appearance of the left hemithorax with progressively increased opacification in the periphery of the middle and lower zones, and accompanying reduction in lung volume, on the right side ().\nThe patient underwent a contrast-enhanced CT of the chest in view of the repeated chest infections and lack of complete resolution to rule out the possibility of an adenocarcinoma in situ or endobronchial lesion, respectively. Cross-sectional imaging was also carried out to assess the possibility of an underlying bronchiectasis in the presence of the recurrent chest infections and to further assess the interstitial changes seen on the CXR on the right side.\nHer CT showed hypoplastic left pulmonary artery (; arrow), attenuated peripheral pulmonary artery branches on the left (: arrows), hyperinflated left lung with air trapping and cystic bronchiectasis of the lingua and left lower lobe (). Features were in keeping with the diagnosis of Swyer-James-MacLeod syndrome (SJMS). Interestingly, the right lung was small in size with evidence of subpleural reticulation, traction bronchiectasis and ground-glass opacification suggesting an underlying pulmonary fibrosis (). The oligaemia of the left lung is shown as reduced FDG uptake () on PET CT, which was done to investigate a different pathology.\nSputum culture grew pseudomonas species and bronchoalveolar lavage from the right lung showed mixed cellularity with 22% eosinophils and a 36% neutrophils. |
On December 13, 2016, a 30-year-old Han Chinese woman in 34+6 wk of pregnancy was admitted to the Department of Obstetrics, First Hospital of Jilin University, due to premature rupture of the fetal membrane and regular contractions for 7 h.\nThe patient reported no menstrual abnormalities. Her last menstrual period was on April 13, 2016, and the expected delivery date was January 20, 2017. She did not undergo regular prenatal examinations during her pregnancy.\nThe patient reported a development delay in learning to walk at the age of 6 years and had poor exercise endurance. She usually experienced troubled breathing, dizziness, and heavy perspiration after fast walking or running. However, she had not undergone systematic diagnostic procedure and had not been diagnosed officially.\nThe patient was unemployed, and had no history of smoking or drinking. No pertinent family history was presented.\nA physical examination upon admission revealed that her general condition was poor (temperature: 36.5 °C, pulse: 126 beats per min, respiratory rate: 28 breaths per min, blood pressure: 153/78 mmHg, oxygen saturation: 83%), and she exhibited cyanosis and shortness of breath. An abdominal examination revealed that she was in the third trimester, and the fetal heart rate was 145 beats per min. Pelvic examination revealed that her cervix was dilated to approximately 10 cm.\nThe patient’s serum troponin level was normal, as were her liver function, renal function, and routine blood test results.\nNo imaging examination was performed in the Department of Obstetrics because of the emergency situation.\nShe was transferred to the labor room immediately. Considering her low oxygen saturation, a heart disease could not be ruled out. Therefore, perineal incision and forceps were used to assist the vaginal delivery and shorten the second stage of labor. A pre-term female neonate was delivered vaginally. Fentanyl (0.002 mg/kg) was slowly intravenously injected to relieve her pain and reduce the cardiac load. However, she continued to experience shortness of breath. Her cyanosis worsened, her heart rate was 120 beats per min, and her blood oxygen saturation ranged from 80%-85%. She was transferred to the intensive care unit immediately.\nThe patient was cyanotic and lacked spontaneous breathing when admitted to the intensive care unit. Immediately, endotracheal intubation was performed and the patient was put on ventilator-assisted respiration. Although, physical examination revealed a body mass index of 15.6, all other findings were normal. Recovery of spontaneous breathing was observed soon after intubation. Her oxygen saturation reached 100% with low respiratory support parameters. Tracheal intubation was removed on the second day postpartum.\nHer cardiac color Doppler ultrasound and electrocardiography were normal. A careful physical examination revealed that she had certain characteristic facial features (Figure ), most notably a thin, elongated face, and a high arched palate. Moreover, muscle volume was less in the limbs, trunk, face, and neck (Figure ), and she exhibited scoliosis with a waddling gait. Computed tomography of the chest (Figure ) revealed scoliosis. Considering the clinical manifestation and physical examination of the patient, motor neuropathy and respiratory muscular disease could not be excluded. Electromyography suggested a myopathic process (Figure ). Biopsy of the left bicep brachia revealed a predominance of type 1 muscle fiber atrophy in the absence of other structural abnormalities, such as that of the rods or cores (Figure ). Our patient's parents and brothers exhibited no signs of CM. They declined to undergo genetic testing. |
A 74-year-old postmenopausal woman was referred to the gynecology clinic of the Jeonju Presbyterian Medical Center with complaints of a palpable abdominal mass that she first recognized approximately 2 months prior. The patient had a history of gravida 2 and para 2. She had no other medical history but hypertension. First, during routine physical examination, an approximately 16–18 gestational week-sized mass was palpated. It was movable, painless, and was suspected to be a large ovarian mass based upon transvaginal ultrasonography. The echogenicity of the mass was heterogeneous with irregular contours. Pelvic computed tomography (CT) indicated that the mass was 10×8.5×13 cm in size with cystic and solid-enhancing features. Enhancement of the internal component of the mass was complex, but there was no definite pathologic enlargement of lymph nodes and ascites (). Additionally, there was a unilocular right ovarian simple cyst and a uterine myoma. Two suspicious retroperitoneal lipomas were observed in the para-aortic region and infrarenal portion. According to CT findings, the sigmoid colon bordered the huge left ovarian tumor. A colonoscopy was performed and confirmed the absence of tumor invasion into the bowel mucosa. Laboratory findings showed only mild anemia and other laboratory analyses revealed normal values. Tumor markers, including cancer antigen (CA) 125, CA19-9, carcinoembryonic antigen, α-fetoprotein, and β-human chorionic gonadotrophin were also in normal ranges. A laparotomy was performed to assess the pathological findings of the left ovarian mass. During surgery, the large ovarian mass seemed to adhere tightly to the ovarian wall, and its smooth capsule seemed to be connected to the ovarian germinal epithelium. There was no sign of tumor capsule rupture. The tumor looked benign in nature without any ascites or peritoneal carcinomatosis. There was slight adhesion between the tumor base and the peritoneal surface (). Since the patient was elderly with a uterine myoma and a right ovarian cyst, total hysterectomy, and bilateral salpingo-oophorectomy were performed. The 2 suspicious lipomas in the para-aortic and infrarenal area were not dissected because of their benign features in the CT. Final pathology of the left ovary indicated dedifferentiated liposarcoma mixed with well-differentiated, myxoid, spindle cells in the liposarcomatous area. The mitotic rate was 20/10 HPF (French Federation Nationale des Centres, cancer grade 3). No progesterone and estrogen receptors were located in the liposarcoma tissue, as confirmed by immunohistochemical testing. There were also no adipose cells in the ovarian stroma. The liposarcoma was considered to have originated not from ovarian stroma but from fat tissues surrounding the left adnexa. Adjacent retroperitoneal fat tissue was especially strongly suspected. After surgery, positron emission tomography (PET)-CT was performed to assess distant organ metastasis and to determine if remnant tissues were present. The fat-containing masses in the left para-aortic and left infrarenal space did not take up fluorodeoxyglucose (FDG) abnormally. There were also no metastatic lesions on other organs. Furthermore, there was no abnormal FDG uptake in the pelvic cavity. Before deciding to start RT, pathologic findings and radiologic findings were further reviewed. Furthermore, pelvic magnetic resonance imaging (MRI) was performed one month after surgery to assess the patient's status. In doing so, a pelvic fatty mass approximately 8 mm in size in the lower pelvic wall was found. At this point, there were three options for planning the patient's further treatment. First, after histologic differential diagnosis via surgical biopsy of the small mass found on MRI, further treatment could be determined. Second, prophylactic radiotherapy might be recommended for local control if surgery was difficult. Third, the fatty mass could just be monitored. Although the pathologic grade indicated a high risk, if the pathologic resection margin status was negative, postoperative RT would confer no survival benefit. Therefore CT scan monitoring could be another choice. Finally, the patient chose the third option because of her age and the economic burden.\nThe radiation oncology department chose to perform a follow-up MRI after 6 months to assess changes in the fatty lesion. If the gross lesion showed growth, RT or secondary en bloc resection would be planned. Until now, the patient had no other complications after surgery and no signs of aggravation of the disease. It was decided to follow up on the progress of the patient's disease every 3 to 6 months for 5 years. |
In 2004, an 80-year-old Caucasian male underwent bilateral cordectomy for Tis carcinoma in the left cord and microinvasive T1 carcinoma at the right one. In the same year, he was diagnosed with prostatic carcinoma (Gleason score 3 + 3) which was treated with radiation therapy for a final total dose of 64 Gy. Since then, he has been controlled regularly on a 6-month basis. His family history was negative for cancer, but he suffered from mild hypertension that has been treated with a sartan drug for 17 years, and he also suffered from mild dyslipidemia without any medication besides dietary control.\nIn January 2014, he was admitted at the dermatologic unit of our institution for a pigmented cutaneous lesion at the occipital region of the scalp and an ulcerated lesion in the left temporal region, which were both radically excised. The pathologist reported a BRAF, c-KIT, and N-RAS wild-type lentigo maligna melanoma in the occipital scalp, while the ulcerated lesion at the temporal region was classified as an actinic keratosis. Full skin and ocular examinations, chest X-rays, and abdominal sonogram excluded the presence of clinical detectable neoplastic deposits. Due to his medical history, the patient was admitted to a three-month follow-up program. In January 2015, for the appearance of a swelling in the left parotid gland, he received an MRI and a subsequent biopsy, which led to a diagnosis of pleomorphic adenoma.\nIn May 2014, he was diagnosed with hypochromic, iron-deficiency anemia in the absence of overt clinical signs of bleeding and normal urine analysis. Fecal occult blood test was strongly positive. The patient was put on oral iron therapy without positive results for three months and therefore was referred to the endoscopy unit for upper and distal gastrointestinal tracts examination. Esophagogastroduodenoscopy showed the presence of several nonbleeding, bluish to black, gastric lesions of a few millimeters in diameter (Figures and ). Such lesions were present in the whole stomach with an increased frequency in the corpus and the antrum but were absent in the esophageal tract and the duodenal level until Treitz's ligament. Biopsy of the three lesions was performed without significant bleeding. Morphologically, the lesions appeared suggestive for metastatic melanoma. Pan-colonoscopy was performed two days later and showed no widespread injuries except for two 5 mm wide, brownish, nonbleeding areas 2 cm away from each other at the rectal mucosa about 15 cm from the anal verge. Biopsy was suddenly performed although the morphological features of these rectal lesions were consistent with mucosal angiodysplastic lesions similar to those often found after radiation therapy for prostate tumors. Pathology examination of both gastric and rectal specimens reported the presence of HMB45, MelanA-S100+, Cd68 negative, infiltrating malignant melanoma. PET/TC scan showed a metastatic lesion in the lumbar spine and the left clavicle, with the latter being biopsied. Pathology reported metastatic melanoma to the bone. PSA and serum chemistry tests were within the normal limits. Careful skin mapping as well ocular examination failed to show presence of cutaneous melanoma. In April 2015, rectal lesions were endoscopically treated with argon plasma coagulation, while gastric lesions were left untreated. The patient was subsequently referred to the Medical Oncology Unit and, after we obtained written informed consent, we started immunotherapy with ipilimumab 3 mg/kg every three weeks. After 3 months, he showed rapidly progressing disease with the appearance of melena and worsening of anemia. Therefore, ipilimumab was stopped and in July 2015 pembrolizumab was started at the dose of 2 mg/kg every three weeks. Reevaluation of the disease after three months showed a PET/TC-documented stabilization of the disease and a significant improvement of clinical conditions and hemoglobin levels with a dramatic reduction of transfusion needs. Pembrolizumab was very well tolerated with mild gastrointestinal and cutaneous toxicity, easily managed with low dose steroids. Pembrolizumab was given for eight months till March 2016 when progressive disease to the liver and the brain was recorded. Unfortunately, the patient died 4 months later due to massive liver progression. |
Our patient was a 31-year-old man who presented to a regional hospital with the chief complaint of substernal chest pain. His past medical history was significant for Crohn’s disease treated with infliximab every six weeks. He was in his normal state of health until he suddenly developed substernal chest pain associated with nausea and vomiting after he finished his routine workout in the morning. He reported similar episodes of chest pain along with shortness of breath on exertion over the past two years. He was referred to a cardiologist who opted to perform an exercise treadmill stress test with unremarkable results. He denied any family history of cardiovascular diseases. He regularly followed a gastroenterologist for his Crohn’s disease that was complicated by anal fistula. Unfortunately, he had been experiencing daily chronic diarrhea due to Crohn’s disease. He was also scheduled for an abdominal ultrasound for his chronically elevated liver enzymes in the upcoming months. Otherwise, he denied alcohol abuse, smoking history, or illicit medication use. He worked as a security guard and was also a heavy weight lifter. He used chronic anabolic steroids in three-to-four months cycles for approximately the past ten years.\nElectrocardiogram (EKG) revealed anterior wall ST-segment elevations along with an elevated cardiac troponin level (Figure ). The patient was transferred to our hospital status post administering of aspirin 324 milligrams (mg) via helicopter transportation service in order to undergo emergent coronary angiography that revealed a normal left main coronary artery, but a thrombotic occlusion was found in the left anterior descending artery (LAD) (Figure ). There was thrombolysis in myocardial infarction (TIMI) grade flow of 1-2 from collateral arteries of the distal right coronary artery to the septal cascade. The patient underwent balloon angioplasty associated with intracoronary thrombolysis of tissue plasminogen activator (tPA) 10 mg over 10 minutes. Insertion of a 4.5 x 3.8 millimeters of the bare-metal stent was deployed at high atmospheric pressure. The subsequent angiography demonstrated TIMI II/III flow to the anterior lateral wall with respective correlation found on the left ventriculogram that showed severe anterior lateral, anterior apical, inferior apical hypokinesis with an approximate ejection fraction (EF) of 30% (Figure ). He remained to be hemodynamically stable throughout the procedure without significant arrhythmias except for a transient accelerated idioventricular rhythm (AIVR) episode. The patient was subsequently transferred to the cardiac care unit (CCU) status post the intervention for further management. He remained to be stable overnight.\nOn day two of hospitalization, he was hemodynamically stable with normal sinus rhythm while on telemetry monitoring. Laboratory results were unremarkable except for elevated troponin levels that peaked at 440 NG/ML, reactive leukocytosis of 18.0 K/UL without a clinical impression of infection along with elevated aspartate aminotransferase (AST) of 521 U/L and alanine aminotransferase (ALT) of 186 U/L with a normal level of alkaline phosphatase. He had a normal hemoglobin A1c of 5.4% and elevated low-density lipoprotein cholesterol (LDL) of 158 MG/DL despite a normal cholesterol level of 198 MG/DL. Echocardiogram showed a moderate to severely reduced left ventricular systolic function with an estimated EF of 25%-30%. He had no signs of acute congestive heart failure or arrhythmias. He remained to be stable overnight.\nOn day three of hospitalization, he continued to remain hemodynamically stable without signs of arrhythmia or acute congestive heart failure. He was asymptomatic with negative reviews of the system. He was fitted for a wearable cardiac defibrillator to potentially reduce the risk of sudden cardiac arrest. He was subsequently downgraded to the telemetry floor from the CCU. He was discharged from the hospital and recommended against taking further anabolic steroids as it was believed to be the cause of his MI without obvious cardiovascular risk factors. He was advised to follow up with his primary cardiologist in the outpatient setting for the continuation of care. He eventually underwent an automatic implantable cardioverter-defibrillator (AICD) implantation three months later (Figure ). |
A 77-year-old dextral man was referred to the emergency department of our hospital by the police because he was found on the landing outside his flat, unkempt and dishevelled in personal appearance, after reportedly having lived there for 10 days. When questioned about this behaviour, he claimed that he was waiting for his next-door neighbour, who held a spare set of his flat keys, since he had accidentally locked his set in his garage. However, while he believed her to be on holiday, she had actually been living in a retirement home for 2 years. His medical history was significant only for a craniotomy, performed 16 years before, for the excision of a left parietal meningioma, with no residual neurological deficits, and hypertension, for which he did not take any drugs. He admittedly consumed more than a litre of wine per day but denied smoking. His psychiatric history was unremarkable, but in recent years he had begun packing his flat with ‘art pieces’. An electrocardiogram performed in the emergency department revealed the presence of atrial fibrillation, and a chest radiography showed a right lower lobe consolidation, which were treated with oral medication because he refused any infusion therapy. He was initially admitted to the geriatric department and then moved to the psychiatric unit because of repeated instances of wandering and a quarrel with another homeless in-patient. When contacted, the patient's building manager reported behavioural changes over the past few years, and a recent history of pathological hoarding was confirmed by his general practitioner. A mental state examination revealed confabulation, along with a more structured delusion about being married to a counterterrorism spy, daughter of a 19th-century marshal, with whom he would communicate only non-verbally in order for her not to be discovered. Neuropsychological evaluation outlined an impairment in selective attention and letter verbal fluency retrieval, in the context of an overall preserved mental functioning, with a score of 26/30 on the Mini-Mental State Examination (MMSE); a subsequent more thorough testing for language excluded semantic impairment (). Brain magnetic resonance imaging (MRI) showed a mild temporo-insular atrophy on both sides and calcifications in the left lenticular nucleus; fluorodeoxyglucose positron emission tomography (FDG-PET) revealed mild hypometabolism in the left superior temporal gyrus; amyloid PET did not reveal areas of increased radiotracer binding, ruling out an underlying amyloid brain deposition (). The search for expansion in the Chromosome 9 open reading frame 72 (C9orf72) gene gave negative results, and no mutations were detected through next generation sequencing (NGS) analysis. According to Rascovsky criteria, a diagnosis of bvFTD was made. In view of agitation and delusions, the patient was initially prescribed clotiapine, then shifted to zuclopenthixol and eventually to haloperidol, with only partial response. After the hospital admission, the patient was admitted to a care home. |
A 61-year-old African-American male came to the emergency department with a history of back and hip pain, night sweats, and decreased appetite coupled with an unintentional weight loss of 40 lbs. He also reported feeling fatigued and had generalized malaise. His history was also significant for hemoptysis and pleuritic chest pain.\nFive months ago, before the current admission, he had presented to our hospital for similar symptoms. During that time, he underwent a Computed Tomography scan (CT) of the chest that reportedly showed right lower lobe pleural thickening with hilar lymphadenopathy. The patient was then prescribed oral antibiotics for possible community acquired pneumonia and was subsequently discharged. Unfortunately, the patient did not feel better and came to the hospital again for the current admission.\nPast medical history was significant for a period of incarceration during which time he had a PPD placed, which was negative. He also endorsed an eight pack-year history of smoking tobacco and a history of remote IV drug use for two years.\nOn admission, the patient’s vitals were remarkable for tachycardia at 106 beats per minute and an oxygen saturation of 98% on 2 liters. On physical exam, a grade IV/VI holosystolic murmur was heard in all cardiac foci. Lungs were clear to auscultation bilaterally. The abdomen was tender to palpation diffusely with normoactive bowel sounds. There was significant tenderness to palpation in the lumbar spine with a positive straight leg test. Digital clubbing of the fingernails was noted bilaterally. Pertinent labs showed an elevation of white blood cell counts at 14.5 thousand cells per milliliter and sodium of 128. Radiological imaging included a CT scan of the chest that showed multiple opacities bilaterally, more prominent on the right (). Patient was then admitted with a diagnosis of right upper lung community acquired pneumonia, with a high suspicion for tuberculosis. Eventually, tuberculosis was ruled out with three negative acid-fast bacillus sputum samples, negative quantiferon screen, and negative nucleic acid amplification test. Blood cultures came back positive for streptococcus sanguinus. Following positive blood cultures, the patient underwent transthoracic echocardiography (TTE) which confirmed the presence of a large and mobile pulmonic valve vegetation, as well as moderate to severe tricuspid regurgitation (). Infectious disease consult was obtained and he was started on ceftriaxone 2 grams intravenously per day. The treatment was continued for a total of four weeks. Due to the large size of the vegetation and severe pulmonary valve insufficiency, the patient underwent valve replacement (). |
A 77-year-old man with a 10-day history of chronic constipation and abdominal distension was seen at the emergency room. The patient's medical history included right hemiparesis due to left basal ganglia infarction 8 years prior and systemic hypertension. On physical examination, he had abdominal distension with mild tenderness; however, he did not have rebound tenderness and his bowel sounds were increased. On digital rectal examination, there was an edematous soft mass-like lesion at a one-finger depth from the anal verge which was not tinged with blood. Laboratory studies revealed that the white blood cell count, hemoglobin, electrolytes, and liver function tests were within the normal range. The level of carcinoembryonal antigen was 17.0 ng/mL (normal range, 0 to 6 ng/mL), and that of carbohydrate antigen 19-9 was 99.3 U/mL (normal range, 0 to 37 U/mL). Plain abdominal radiography revealed a mechanical ileus with severe bowel distension (). A contrast enhanced computed tomography scan was suggestive of rectal cancer with a diffuse wall thickening that was 7 cm in length and was located 5 cm above the anal verge. The small bowel and colon were dilated with fecal retention due to a mechanical obstruction proximal to the mass lesion (). Colonoscopic findings revealed that the extruding mass was covered with normal mucosa (). Endoscopic biopsy showed only an inflammatory change; however, we clinically suspected rectal cancer, so additional pelvic magnetic resonance imaging (MRI) was performed. The pelvic MRI revealed a submucosal infiltrating enhanced lesion involving the rectum and containing round and tubular cystic lesions with perirectal fat infiltration (). A positron emission tomography scan showed rectal cancer because of increased fluorine-18 2-fluoro-2-deoxy-D-glucose uptake (max standardized uptake value, 7.7).\nBecause of negative biopsy results and radiologic findings that showed a submucosal infiltrating enhanced lesion, we suspected that the tumor was most likely a primary rectal linitis plastica and recommended surgical treatment for the patient. However, the patient declined surgery without pathologic confirmation and chose close observation and symptomatic treatment. One month later, he returned to the emergency room with recurrent abdominal distension and severe constipation. An emergent APR and permanent colostomy was performed due to signs of impending bowel perforation. During surgery which lasted for 5 hours, there were no special events, but pelvic dissection was difficult due to the infiltrative tumor mass and neighboring fibrosis. After surgery, the patient recovered without complications. Pathologic examination of the resected tissue revealed a mucinous adenocarcinoma in the rectal diverticulum involving the rectum (4.2 × 2 × 1.5 cm3) with extension to perirectal soft tissue; however, no lymphovascular perineural invasion or distant metastasis was detected ().\nThe cancer was located within a diverticulum, but the entrance of the diverticulum was not seen during endoscopy (). The negative endoscopic biopsy result was due to the fact that the mucous membrane was unaffected by the adenocarcinoma within the rectal diverticulum. After surgery, the patient received adjuvant radiation therapy, and no recurrence has been detected for 12 months. |
An 8-year-old female patient visited the university hospital due to delayed eruption of her right permanent maxillary central incisor. Her medical history revealed no systemic diseases, and the dental history proved no facial trauma or other tooth abnormalities have occurred. She avowed that she was operated on, at the age of seven years, for a supernumerary tooth delaying the eruption of the left permanent maxillary central incisor. A previous panoramic radiograph (one year ago) revealed the presence of a mesiodens associated to two supernumerary teeth [].\nClinical examination revealed a mixed dentition and the absence of the right maxillary permanent central and lateral incisors []. The left ones have already erupted.\nThe patient was referred to CBCT examination of the maxilla to assist the localization and orientation of the mesiodens. CBCT images were requested for diagnosing accurately the morphology and exact location of the mesiodens and the radicular formation of the permanent maxillary central incisors. The images were created and viewed interactively using a dental computed tomography (CT) software program.\nThe 3D CBCT panorex showed the impaction of the right permanent maxillary incisor and the presence of a supernumerary tooth; moreover, the axial slice image of CBCT revealed the palatal position of the impacted and inverted mesiodens and the sagittal slices showed that the supernumerary tooth was in palatal position over the impacted permanent maxillary central incisor [].\nA comprehensive treatment plan was formulated, which included surgical extraction of the mesiodens and the supernumerary tooth []. The patient was administered local anesthesia. Using a mucoperiosteal elevator, a full thickness palatal flap was raised. The supernumerary tooth was exposed, luxated, and removed. Hemostasis was achieved and the flap was replaced back and sutured. For the mesiodens, a simple palatal anteroposterior incision was done and the extraction followed. Postchirurgical instructions were explained to the patient along a prescription of antibiotic and analgesic treatment. The recall visits were scheduled for the following week to remove suture and evaluate the healing process. The patient was kept under the observation until the successful eruption of permanent central and lateral incisors. |
A 64-year-old male with a past medical history of coronary artery disease, cardiac stent, COPD, HTN, and dyslipidemia presented as a referral for left testicular teratoma and primary bladder leiomyoma. Prior to presenting to our facility, he underwent left radical orchiectomy, and hernia repair due to enlarging left testicle, had a transurethral resection of bladder tumor (TURBT) due to hematuria, and underwent PSA screening. Pathology determined the testicular mass to be a pT1 teratoma without lymphovascular invasion, PSA was found to be elevated (was 4.2 in 2014, now 4.39), and the 40 g of bladder tumor resected during the TURBT was found to be a leiomyoma of the bladder showing no sarcomatoid differentiation. A CT scan was performed which was notable for bilateral pelvic adenopathy with lymph nodes greater than 2 cm and an interaortocaval lymph node of approximately 1.2 cm. Additionally, outside CT scan with IV contrast of abdomen pelvis showed large 6 cm heterogeneous soft tissue mass within the posterior left aspect of the bladder (). The mass was causing complete obstruction of the left ureterovesical junction (UVJ), which resulted in severe left sided hydronephrosis and hydroureter. Although the CT findings were highly concerning for metastatic process, the TURBT specimen did not support the diagnosis of metastatic disease from a bladder primary as it had returned leiomyoma of bladder without sarcomatoid elements. The disparity between imaging findings and pathology results prompted confirmatory testing on both the initial testicular mass and the TURBT specimens. After discussion at multidisciplinary GU tumor board, the consensus recommendation was for CT-guided lymph node biopsy, repeat transurethral resection of bladder tumor, and decompression of the left upper tract via ureteral stent, or nephrostomy. The patient underwent rigid cystourethroscopy, monopolar transurethral resection of bladder tumor (large >5 cm) and monopolar transurethral resection of prostate. During this surgery, a large tumor located on the left wall extending down into the bladder neck and prostatic urethra was resected with an extensive amount of tissue removed (). The right ureteral orifice was uninvolved, but the left ureteral orifice was completely obscured by tumor. The bladder neck and prostate were resected down to the level of the verumontanum which was spared. A 3-way foley catheter was placed at the conclusion of the case and the patient was admitted post operatively. He underwent left percutaneous nephrostomy (PCN) placement and IR guided biopsy of the suspicious pelvic lymph nodes prior to discharge.\nFollowing independent pathology review of the outside facility testicular specimen and review of in-house specimens, the previous diagnosis of testicular teratoma was recharacterized as a Leydig cell tumor of the testis and the patient's lymph node biopsy revealed follicular lymphoma. Discussion at multidisciplinary tumor board initially recommended bone marrow transplant, neoadjuvant chemotherapy, and extirpative surgery at a future date depending on the patient's response to chemotherapy. After bone marrow biopsy, his follicular lymphoma was staged as stage 4 with minimal bone marrow involvement. In advanced stage follicular lymphoma there is no survival benefit to early initiation of treatment, therefore serial PET CT scans 6 months apart were recommended by hematology/oncology to follow his lymphoma.\nThe patient was managed conservatively with nephrostomy tube exchanges every 10–12 weeks and underwent cystoscopy with urodynamic studies after presenting with urinary retention and traumatic foley at approximately 1 year post resection. Cystoscopy revealed a large mass in the left lateral bladder wall extending down into the bladder neck and involving the prostate. He was started on Flomax and discharged without a foley catheter as he was able to void with a normal PVR at the time of the procedure. Due to significant regrowth of leimyoma on cystoscopy, he was encouraged to have repeat debulking of the tumor via transurethral approach as he declined cystoprostatectomy. The patient underwent repeat TURBT with bipolar resectoscope for recurrent left lateral bladder wall, bladder neck, and prostatic mass. The pathology again returned leiomyoma without sarcomatoid differentiation. A month after discharge the patient presented to an outside Emergency Room after developing gross hematuria. He was transferred for continuous bladder irrigation and refractory hematuria which required urgent TURBT to fulgurate bleeding vessels at the left lateral bladder neck and resect additional tumor regrowth.\nThe patient followed up in clinic 1 month later, and after an in-depth discussion of his clinical course, he decided to undergo surgical extirpation of the bladder and prostate with creation of an ileal conduit urinary diversion. When reviewing the preoperative MRI there was loss of fat plane between the prostate and rectum, therefore colorectal surgery was consulted to assist in rectal dissection. He also underwent preoperative colonoscopy in order to rule out invasion into the rectum and screen the remainder of the colon. The patient underwent open radical cystoprostatectomy with ileal conduit urinary diversion and intraoperative angiogram of the involved bowel segment and omental flap interposition. He had intraoperative proctoscopy by colorectal surgery to ensure no rectal injury was present. He was discharged on postoperative day 4 after progressing without incident. The pathology report from his extirpative surgery revealed a 1.8 cm leiomyoma involving the submucosal bladder wall surrounding the left ureteral orifice. Incidental finding of High Grade Prostatic Intraepithelial Neoplasia was noted on the prostate specimen. The findings did not extend through the bladder wall into the perivesical or periprostatic adipose. Margins were negative. The patient was evaluated 8 weeks postoperatively and has had an uncomplicated postoperative course to date. He has been satisfied by his improved functional status and is to be followed with serial renal ultrasound in 6 months to ensure no obstruction at the uretero-ileal anastomoses. |
A 66-year-old male patient was admitted to our institution after he underwent follow-up in another institution. CA 19-9 levels were found to be elevated. He had no known diseases other than hypertension. There were no alarming symptoms in his systemic examination, such as weight loss, abdominal pain, hematemesis, and night sweats. He had no history of alcohol consumption and abstained from smoking for last 20 years, after smoking for 20 years. The patient was found to have chronic reflux symptoms, which did not regress with proton pump inhibitors. The patient had undergone a gastrectomy and a billroth 1 anastomosis long time ago. No pathological findings were observed in the initial laboratory tests; both biochemical parameters and total blood counts were normal, including hemoglobin, which was found to be 16.6 gm/dL. The tumor marker tests were renewed and CA 19-9 was 193 U/mL, carcinoembryonic antigen was 2 ng/mL, alpha-fetoprotein was 4 ng/mL, and CA 72-4 was 1.1 U/mL, which showed that the patient had an isolated CA 19-9 elevation. To rule out a possible malignity concerning the gastrointestinal system, a gastrointestinal system endoscopy and colonoscopy were performed. Endoscopic and colonoscopic examinations had no findings pointing to a malignity. A former gastric operation scar was observed at the upper gastrointestinal endoscopy and the lower esophageal sphincter was found to be relaxed, explaining the reflux symptoms. To rule out a possible pancreatobiliary system malignity, abdominal computerized tomography and magnetic resonance cholangiopancreatography were performed, but no findings related to a malignity that can be at the pancreas or the biliary system were observed in the radiological imaging. As the patient had persistent CA 19-9 elevation with no pathological findings to explain this situation, a follow-up was initiated. As the patient had severe reflux symptoms, during the next 5 years, three gastrointestinal endoscopies were performed. In the endoscopic examination, similar findings were observed with the prior endoscopy. Again, during the follow-up, as the CA 19-9 levels were not stable, another colonoscopic examination was performed, but no findings pointing to a malignity were observed. During this period, the patient was examined with an abdominal ultrasound once in every year but no malignity-related findings were observed. During these years, although CA 19-9 was always 5 to 15 times the normal value, no other tumor markers were found to be elevated (). Due to the fact that the patient had dyspepsia and persistent reflux symptoms, an endoscopy was performed, which showed a mass lesion above the larynx near the vocal cords with a malignant-like appearance. The patient was consulted to surgical departments. Before the operation, positron emission tomography was performed, and at the mass, at the left aryepiglottic area, and at the nodule in the third level of the left side of the neck, pathological 18F-fluorodeoxyglucose uptakes were observed. The patient underwent a total laryngectomy and lymph node dissection. Histopathological diagnosis was consistent with a moderately differentiated squamous cell carcinoma. Immunohistochemically, focal strong CA 19-9 positivity was detected in the apical areas of the cytoplasm of the tumor cells (). After the surgery, CA 19-9 levels were back to normal. |
A 12-year-old girl medically free presented with fever, loin pain and hematuria on February 26, 2020. Her laboratory work revealed anemia (hemoglobin level 8.4 g/dL, reference range: 11.5-15.5 g/dL) with normal platelets and white blood cells count.\nBased on clinical presentation and laboratory data, diagnosed as urinary tract infection and iron deficiency anemia, she was discharged on cefprozil and oral iron supplements. Her other investigations showed normal chest x-ray (CXR) and negative urine culture.\nAfter two weeks from initial presentation, she had developed dry cough and her family sought medical advice in many medical centers as her cough getting worse without improvement. On the 17th of March, the patient admitted to our institution complaining of worsening dry cough with no fever. Cough was associated with left sided chest pain, which increased with movement and deep breathing. She denied any history of contact with patient with confirmed COVID- 19 or any respiratory diseases. No history of travel or contact with any person with travel history. She had no gastrointestinal symptoms, joints pain, or skin rash. No family history of thrombosis or similar illness. On physical examination, the child was clinically stable with normal vital signs including body temperature as well as blood pressure. She was not in respiratory distress and saturated well in room air. Chest examination revealed normal contour, good air entry on the right side, and diminished air entry on the left side with no bronchial breathing or wheeze.\nCXR showed opacity involving the left lung base laterally with obliteration of the left costophrenic angle ().\nThe laboratory investigations at admission are summarized in , showed mild lymphopenia and normal WBC, but later on she developed leukopenia and severe lymphopenia. Kidney and liver function were normal. On the next day of admission both D-dimer level and anti- Phosphatidylserine (IgG and IgM) were elevated whereas other thrombophilia panels were negative (D-Dimer: 4.590, reference range 0.000-0.500 ug/mL, IgG Anti- Phosphatidylserine: 39, reference range 0-1 U/mL, IgM Anti-Phosphatidylserine: 39, reference range 0-24 U/mL). Fibrinogen was normal: 3.8 (reference range 2.0-5.0 g/L). Procalcitoninwas elevated as well: 6.29 (reference range 0.10-0.49 ng/mL).\nBecause of CXR findings, computed tomography pulmonary angiography (CTPA) had been performed on the next day of admission and revealed extensive bilateral pulmonary embolisms (PE) with multiple peripheral subpleural infarctions and peripheral subpleural ground-glass opacity of the right upper lobe (). The patient started initially from ceftriaxone and received a course of azithromycin as a treatment of atypical community acquired pneumonia with unusual PE for investigation.\nImmediately, hematology and cardiology services evaluated the patient condition and documented the stability of the cardiorespiratory condition with no hematuria or potential risk of bleeding. To look for the source of PE, doppler ultrasonography of the lower limbs and echocardiography was reported as normal with no detected thrombi. Electrocardiogram testing was performed as well and revealed normal sinus rhythm with neither right ventricular hypertrophy nor ST segment abnormalities. Multidisciplinary team decided to start the child from subcutaneous low molecular weight heparin (enoxaparin 1 mg/kg with a total dose of 30 mg subcutaneous every 12 hours). Based on the follow up of therapeutic anti-X level, enoxaparin adjusted to 40 mg s/c every 12 hours to keep the therapeutic level at the higher side 0.75-1.0 IU/mL.\nOn 25th of March, follow up CTPA showed the same features of PE and infarction as previous imaging. The child remains afebrile and clinically stable with no signs of respiratory distress until 28th of March (11 days after admission) when she started to have frequent spikes of fever up to 39.5°C with no respiratory distress and her laboratory testing revealed leucopenia and severe lymphopenia (0.6×109/L). Although she had been on broad-spectrum intravenous antibiotics, which were escalated to a combination of tazocin and vancomycin, her body temperature continued to spike daily. Antimicrobials therapy was stopped after the microbiological laboratories had reported negative blood cultures on multiple occasions.\nDue to the clinical manifestations of persistent fever and dry cough, radiological features of ground glass appearance and PE, in addition to laboratory testing of progressive leucopenia and lymphopenia, COVID- 19 was suspected. On March 31st, COVID- 19 testing was performed by (rRT-PCR) of nasopharyngeal swab. While waiting the COVID-19 rRT-PCR result, the patient was started on daily oral hydroxylchloroquine 200 mg as a suspected with antiphospholipid syndrome because of PE and associated elevation of both antinuclear and antiphospholipid antibodies with elevated ESR and CRP\nThree days later, COVID-19 rRT-PCR results reported as positive which led to the addition of azithromycin to hydroxychloroquine. The child remained clinically stable, afebrile, and on room air without any cardiorespiratory assistance. After two COVID-19 negative serial RT-PCR for SARS-CoV-2 RNA from the nasopharyngeal swab, she was discharged home from anticoagulant therapy in a good condition with a repeat of CTPA in two weeks from discharge for evaluation of her clinical condition. |
A 59-year-old woman was admitted to the emergency department with sudden onset of chest pain for five hours. She had hypertension (treated with β-blockers) as a cardiovascular risk factor. She was diagnosed with advanced non-small-cell lung cancer with liver metastases one month previously. The patient was not considered for surgical resection, she was scheduled for chemotherapy, and, consequently, no antineoplastic treatment had been initiated yet. Prophylactic anticoagulation was not indicated On clinical examination, her blood pressure was 150/90 mmHg with a pulse of 100 beats per minute, oxygen saturation of 95%, and normal heart sounds. The 12-lead electrocardiogram showed sinus rhythm with a heart rate of 102 beats for minutes and ST-segment elevation in all leads except aVR where ST-segment depression was noted (Figure ). Her laboratory test results revealed a baseline troponin T level of 1212 ng/ml (normal range < 26 ng/ml). Also, the echocardiography findings revealed akinetic walls from the mid to apical septum and anterior and inferior walls. The ejection fraction was estimated to be 39%. Therapy was started with clopidogrel, aspirin, and low-molecular-weight heparin. The patient underwent a coronary angiogram from the right radial approach, which revealed the presence of an extended thrombus in the left anterior descending artery (LAD), and another thrombus was found in the second segment of the right coronary artery (RCA) with thrombolysis in myocardial ischemia (TIMI) III flow and without any atherosclerotic lesions in the coronary artery tree (Figure and Figure ), therefore, balloon angioplasty and/or stent placement was not considered. The patient was taken to the coronary care unit (CCU) and tirofiban infusion was administered for a period of 48 hours. In view of persistent breathlessness, pulmonary embolism was suspected. There were no symptoms or clinical signs suggestive of deep venous thrombosis. A computed tomographic pulmonary angiography was performed and showed a distal pulmonary embolism (Figure ). The patient was discharged on acenocoumarol to be followed as an outpatient. At her 45 days follow-up, she did not report any episode of chest pain, bleeding, as well as any thrombotic events. |
A 47-year-old male with a past medical history notable for hypertension on metoprolol succinate, morbid obesity, and pre-diabetes presented to the emergency department (ED) with a chief complaint of generalized weakness. The patient tested positive for COVID-19 and exhibited mild unspecified respiratory symptoms. He was subsequently discharged home to recover in isolation per the CDCs COVID-19 response protocols. One week later, the patient returned to the ED for ongoing symptoms and was admitted requiring supplemental oxygen for hypoxia. Upon admission to the hospital, the patient was noted to have difficulty standing and ambulating. Two days into his inpatient stay, the patient developed urinary retention issues requiring intermittent catheterization. Three days later, he developed facial weakness and numbness. The clinical diagnosis of GBS was suspected and subsequent EDX studies reported AIDP. The patient was treated with a five-day course of intravenous immune globulin (IVIG). Three days after the completion of his IVIG treatment, the patient noted improvement with right upper extremity anti-gravity strength.\nThe patient was transferred to inpatient rehabilitation with significant proximal lower extremity weakness. The bilateral upper extremities demonstrated slight weakness. The bilateral lower extremities demonstrated a significant loss of strength, 1/5 dorsiflexion, and 3/5 plantar flexion bilaterally. The patient also reported diminished sensation to light touch in bilateral upper extremities in all dermatomes. His blood pressure upon rehabilitation admission was 110/73 mmHg.\nPrior to hospitalization, the patient was independent with mobility and all activities of daily living. Upon evaluation in the inpatient rehabilitation, the patient was at a significant functional decline from baseline, requiring dependent assistance with toileting hygiene, showering, upper body dressing, lower body dressing, footwear management, rolling left and right, and all transfers. The patient was unable to ambulate due to his level of impairment. In inpatient rehabilitation, the patient completed three hours total of physical, occupational, and speech therapy per day five days a week with exercises aimed to improve balance, mobility, activities of daily living, fine motor skills, cognition, and breath support.\nAfter completing four weeks of inpatient therapy, the patient was independent with supine to sit, upper and lower body dressings, and rolling right and left in bed. He required minimal assistance to lift the left lower extremity into the bed and moderate assistance with wheelchair-to-bed transfers and bed mobility. He was able to stand with the assistance of his spouse and by using significant reliance on bilateral upper extremity support on a walker for stability and offloading lower extremities. He required a power wheelchair for mobility and was unable to transfer in and out of the car thereby requiring a wheelchair van for transportation. Final muscle strength grading was not documented. His blood pressure remained stable throughout his stay on metoprolol succinate and was 121/65 mmHg on discharge. |
A 70-year-old man with antecedent of follicular lymphoma in complete remission presented at the Timone University Hospital (Marseille, France) in 2016 for a squamous cell carcinoma of the hypopharyngeal region. The patient categorically refused any treatment, including preservative surgery, radiotherapy, chemotherapy or supportive care.\nOne year later, he was addressed to our palliative care unit by the hand-surgery department after attempting suicide. The patient explained his action by the fear of suffering. No depressive state was diagnosed by our psychiatrists. Despite persistence fear of suffering, the patient rejected the idea of suicide because of his family, but still wanted to die and asks for assistance. Information on Claeys-Leonetti law was given, especially on assisted-suicide banishment and on the possibility to relieve suffering with adapted treatments.\nOne week after discharge, the patient was readmitted to our department for dyspnea and anxiety. Symptoms were managed by appropriate treatments (oxygen and low dose of midazolam in an anxiolytic purpose). Despite stabilisation, the patient was afraid of dying suffocated and asked for deep and continuous palliative sedation until death. Apart from the fear he expresses, the patient has no symptoms of anxiety, depression or pain after the introduction of appropriate treatments. On the other hand, he clearly states that he refuses to live again knowing that his death is approaching and that he is apprehensive of suffering. He says he wants to rush his death. For us, this is a request for assisted-suicide (active help from a third party for the administration of a lethal product) or euthanasia (act of a third party which intentionally provokes the death of another to put an end to his sufferings), rather than a real demand for deep and continuous sedation. It seems important to note that patient’s requests for deep and continuous sedation until death are not registered officially. The law does not even impose a written request. Thus, the request is most often made orally in the presence of several doctors and clinicians.\nIn order to try to objectify this request and therefore our answer, the patient’s request was examined and denied by palliative multidisciplinary board, in accordance with by the French Oncology Coordination Centre guidelines. This situation did not fulfil the criteria requested by Claeys-Leonetti law. Indeed, prognosis appeared not short term committed (no visible clinical progression of the disease, which commits for sure the short-term vital prognosis), symptoms were managed with appropriate treatments and no life-sustaining treatment arrest could lead to potential unbearable sufferings. Regarding the short-term criterion of life-threatening prognosis, the patient was offered to have a Computed Tomography (CT) scan to measure the progression of the disease. Indeed, no imaging had been performed for one year (time of diagnosis of recurrence). The patient refuses this proposal. The request for deep and continuous sedation was reiterated several times by the patient, who was still refusing any investigations to define the progression of his cancer and wanted parenteral hydration to be maintained. Daily, he questioned each caregiver about the rationale for the refusal of his request. How can the medical staff be sure that his prognosis is not short-term compromise? Why his psychological distress could not be considered as refractory? One week after refusing further investigation, the patient finally agrees to undergo a CT scan. Three days after the exam he dies peacefully, according to our team (no specific questionnaires or objective elements to judge the quality of death exists), of a not predictable respiratory distress certainly linked to the evolution of his cancer of the hypopharyngeal region without introduction of deep and continuous sedation, but with introduction of midazolam for anxiety. Opiates were not introduced because the patient was saying not being painful. The CT scan results, unknown at the time of death, reveal nothing conclusive (pulmonary metastases, but no lymph node involvement) and would have required additional analyzes. |
A 32-year-old Indian woman presented to our gynaecology clinic with secondary subfertility. Routine screening found her to be positive for protein C deficiency. In 1999, the woman had previously delivered a son by elective caesarean section following a diagnosis of transverse lie at 39 weeks of gestation. The baby was subsequently diagnosed with SCID and died at 8 months due to this condition. A specialist genetic centre indicated that this was an X-linked condition and the woman is a carrier of the mutation c.283G>A (W90X) in exon 2 of her IL2RγC gene.\nShe was advised of a 50%-risk of any subsequent male offspring being affected by SCID and was thus offered preimplantation genetic diagnosis. However she subsequently conceived the index pregnancy spontaneously. She was commenced on aspirin, low molecular weight heparin (LMWH) and progesterone pessaries as soon as the pregnancy was confirmed by ultrasound at 6 weeks. Resutls of her nuchal translucency screening and first trimester anomaly scan were normal.\nThe woman presented with leakage of liquor at 14+5 weeks gestation. Spontaneous rupture of the membranes was confirmed clinically and through an ultrasonography. She was commenced on antibiotic prophylaxis with oral erythromycin and her medication of progesterone pessaries was discontinued. Serial specialist ultrasonography at 16, 18, 20 and 22 weeks confirmed a normally grown male fetus with no obvious structural defects. The placenta was posterior and low, and the severe oligohydramnios was persistent. In view of the oligohydramnios due to extreme preterm SROM, an invasive genetic testing was not felt to be appropriate. The risk of this male fetus being affected with SCID therefore remained at 50%. Because of the extremely poor prognosis for the baby, the couple was offered a termination of pregnancy but they declined. The pregnancy continued, septic markers remained negative, and she remained on continuous oral erythromycin, aspirin and LMWH.\nAt 24+4 weeks gestation, the woman self-referred with a history of unprovoked vaginal bleeding. She was given dexamethasone and was transferred to a centre with Level 1 neonatal facilities. She stayed at the centre until her transfer back to her local hospital at 28+5. Erythromycin was discontinued at 25+6 weeks gestation, maternal clinical and laboratory signs of infection remained absent, and ultrasound scan at 28 weeks showed normal growth of the fetus and an amniotic fluid index of 8.4 cm. Upon readmission, she was recommenced on erythromycin and discharged from the hospital. She was advised instead to visit the antenatal day unit twice a week for regular assessments.\nThe woman remained stable until she presented again at 31+4 weeks with lower abdominal pain and recurrent slight vaginal bleeding. It was found that her cervix was dilating and that the baby's presentation was breech. An emergency caesarean section was thus performed under spinal anaesthetic. She made a good postoperative recovery and was discharged after 5 days.\nThe baby boy was born with Apgar scores of 61 and 105 minutes and weighed 1830 g (50th percentile). He required some initial resuscitation but was transferred to our special care baby unit with spontaneous respiratory effort in facial oxygen. His white cell count and C-reactive protein level were within the normal range (6.3 and <5, respectively) while his blood cultures were negative at birth. The baby was treated with oral nystatin, intravenous benzyl penicillin and gentamicin. Isolation and barrier nursing was also advised.\nX-linked SCID (consistent with the mother's carrier type) was confirmed by genetics testing during the neonatal period. He received intravenous therapy of 1 g immunoglobulin on Day 3 and was transferred to a specialist paediatric centre on Day 6. The baby underwent an unconditioned CD34-selected mismatched family donor bone marrow transplant (from his father) on Day 46. He continued to receive monthly 2 g intravenous immunoglobulin and regular outpatient specialist paediatric immunology reviews. As of this writing, the baby is thriving and still breastfeeding at 1 year of life. He is also showing a good response to his treatment. |
A 50-year-old woman positive for hepatitis B virus underwent a right hemihepatectomy extended to a medial part of Couinaud segment IV for HCC in May 2000. The initial tumor was mainly located in Couinaud segment V and was 10 cm in diameter with capsule invasion. Histologically, it was moderately differentiated HCC with thick trabecular pattern with microscopic vascular invasion. In January 2001, partial resection of the medial segment was performed due to recurrence in the form of intrahepatic metastasis. During a period of 8 years after second surgery, no recurrence sign was detected by 5-year regular imaging follow-up at our hospital. In December 2008, the patient consulted a home doctor for chest discomfort and then was recommended to our hospital again for the purpose of general examination. The serum value of alpha-phetoprotein (AFP) increased to 248369 ng/mL (normal rage; <20 ng/mL). Computed tomography (CT) revealed a heterogenous mass, 9 × 7 cm in diameter, in the remnant liver (). The mass had expanded to the adjacent organs, inferior vena cava (IVC) () and the pericardium (), indicating a diagnosis of intrahepatic recurrence from HCC. Angiography revealed that the remnant liver was supplied by left hepatic artery with no tumor stain (). Because selective angiograms showed a hypervascular tumor supplied from the descending branch of the inferior phrenic artery (IPA) () and the phrenic branch of the internal mammary artery (IMA) (), transcatheter arterial embolization (TAE) was selected first. However, TAE could not relieve her chief complaint, and after evaluation that there were no other distant metastases, complete surgical resection was planned on January 2009. Because it was difficult to identify the whole aspect of the tumor from just the abdominal upper midline incision, an additional median sternotomy was carried out (Figures and ). Pericardial fluid was found to have no malignancy after cytological examination. At the superior border of the tumor, the confluence of the pericardium and diaphragm was displaced, but the tumor itself showed general expanding growth that was not invasive. Since the adhesion on the central tendon was too complete to separate, the crus was ligated (). The diaphragm was divided toward the left edge of the sternocostal trigone, and when rotated, the heart was observed. In the opened pericardial cavity, the pericardium was transected to expose the posterior side of the tumor from the IVC (), and following transection of the posterior parietal peritoneum, the specimen was removed (). The diaphragm was sutured and the defect in the pericardium was repaired using a composix mesh (Figures –). Macroscopically, the resected tumor, at 9.5 × 7.4 cm (), was covered with fibrous tissue (diaphragm) without evidence of infiltration and expansive necrosis was detected (). The tumor comprised moderately differentiated HCC showing a thick trabecular pattern and focal pseudogland formation () and also the displaced muscular layer of the diaphragm was observed (, arrow). These histological findings revealed a solitary peritoneal implantation to the diaphragm. The surgical resection could relieve her chest discomfort immediately. The patient is in good health without any postoperative events and no sign of recurrence. |
A 30-year-old married Indian female, diagnosed as SLE 10 years ago presented to the Medical Emergency Department with a 1-month history of mutism, nonresponse to commands, continuous staring, refusal to eat, rigidity of limbs, decreased self-care, urinating in bed and insomnia. She was on oral prednisolone 30 mg/day and hydrochloroquine 200 mg BD for last 10 years in view of her SLE. Some years ago, she also had received one cycle of 200 mg of cyclophosphamide intravenously for 1-month. There was a past history 2 months ago of psychiatric illness in which she had an episode of agitated behavior, tendency to run away from home, muttering to self, which was precipitated by a single episode of seizure. She was worked up for organic precipitating cause, and haloperidol 5 mg intravenously was given immediately at the time of admission, in view of considering delirium as a provisional diagnosis.\nA psychiatric consultation was sought in view of her continuing symptoms. She was conscious, but withdrawn and vacant, with decreased touch from reality. She was mute and did not obey commands showing features of ambitendency. She made no spontaneous movements and could not stand or walk without support. There was rigidity of all limbs more so in upper limbs. The remainder of neurological examination was normal. She was afebrile, and no abnormalities were found in cardiovascular, respiratory and abdominal systems.\nAll baseline investigations including complete blood count, liver function test, kidney function test, blood sugar, serum electrolytes and electrocardiogram were normal. Imaging studies including ultrasonography, magnetic resonance imaging brain and electroencephalogram showed no abnormality. Anti-nuclear antibodies and anti-ds DNA antibody was negative. Examination of the cerebrospinal fluid, including virological studies, was normal. Renal biopsy showed features of membranous nephropathy consistent with lupus nephritis Class Vb with mild tubulointerstitial nephritis.\nHaloperidol was continued for 5 days (5 mg twice daily). On day 6, in view of her catatonic symptoms, she was given lorazepam orally 2 mg/day in three divided doses (6 mg/day). On day 10, the dose of lorazepam was increased to 10 mg/day for the next 10 days. The patient however, showed no improved and the drug was tapered off. She was then given amisulpride 600 mg/day for the next 3 days, but with no response. She was advised ECT by a consultant psychiatrist after the patient showed no response to drugs []. On day 24, ECT was administered bilaterally with 30% energy stimulation with thiopentone (15 mg) and suxamethonium (25 mg) respectively. Her Bush Francis Catatonia Rating scale (BFCRS) score decreased from 27 before ECT treatment to 5 after the fourth ECT. She was given a total of six sessions of modified ECT twice weekly on outpatient department (OPD) basis for a total duration of 3 weeks by the end of sixth ECT session she was completely symptom free (BFCRS of 0) []. On day 45, she was discharged to outpatient follow-up. In the outpatient follow-up, her mental status was stable. Her husband also reported marked improvement in her behavior at home in the form of taking care of herself and her children, doing household work with interest, remaining cheerful and taking part in family discussions. Other drugs were slowly tapered, and she is presently taking only 50 mg cyclophosphamide. She has presently no neuropsychiatric manifestations at 1-year of follow-up at psychiatric OPD []. |
A 29-year-old man, a former semiprofessional handball player, had a traumatic rupture of the proximal side of the patellar tendon of the left knee in 2016 during a match. No associated disease was reported. Primary surgery was performed in another health facility through a median approach using 2 anchors for tendon repair protected by an additional ipsilateral semitendinosus graft (patellar and tibial tunnels). The patient came to our health facility following severe functional deficits after an iterative rupture without having experienced any new trauma 13 months after the initial surgery. The iterative rupture of the knee extensor mechanism was also an iatrogenic fracture of the transverse patellar tunnel (). Clinically, walking was not possible, there was a lack of active extension and hemarthrosis with pain. There were no scar problems, no signs of deep or superficial infection, and no cutaneous wound. A huge gap was clinically observed between the patella and the patellar tendon. Considering the patient's age, his preinjury sports level, and lack of active extension, the decision was made to perform a revision procedure. An artificial ligament (LARS®) and two adjustable loops, free ends of the PULLUP® BTB (SBM SAS, France), were used to enhance the patellar tendon repair.\nA preoperative lateral standard X-ray was taken of the contralateral knee at 30° of flexion to measure the Caton-Deschamps index and patellar height (). The patient was placed in a supine position under general anesthesia with a tourniquet at the proximal part of the thigh. The previous median approach was used. The patellar fracture and the site of the previous rupture were cleaned to remove fibrous tissue and hematoma. Previous anchors were left in place.\nThe first step was to place the ligament advanced reinforcement system (LARS® polyethylene terephthalate fibers 6 mm ref. L030307 ACFAR 32 CK). A new transverse tunnel was drilled in the tibia, distally to the tibial tunnel of the initial surgery, with a 5.5 mm drill, and the LARS® was inserted in the tibial tunnel. The artificial ligament was then passed through the lateral retinaculum and above the patella at the junction with the quadriceps tendon in a Pulvertaft manner and through the medial retinaculum to return to its origin (). Two longitudinal tunnels were drilled in the patella using a 2.4 mm drill. The loops of a PULLUP® BTB (the plate was removed from the device) were first passed into the patellar tendon and then into the patella through the two longitudinal tunnels using a shuttle relay. Next, the free ends were pulled down in the opposite patellar tunnels. At the proximal side of the reconstruction, the two free ends of the PULLUP® BTB were inserted into each braid to close the system (Figures and ). The distal and medial ends of the LARS® were tightened with a clamp in order to restore normal patellar height and secured with 2 staples. Then, the 2 PULLUP® BTB loops were tightened (). The previous tendon rupture was closed and reinforced with separate X-knots using absorbable sutures. The skin was closed. The knee was placed in an articulated brace with compressive ice therapy for 24 hours, and a postoperative X-ray was taken ().\nFor postoperative care, weight bearing was not allowed for 6 weeks and mobilization of the knee was immediately started between 0 and 45° for 3 weeks then from 0 to 90° from 3 to 6 weeks. No complication was reported during the postoperative period. At 3 months, the patient was pain free and could walk without the aid of crutches. He was able to resume handball practice at 6 months after a control MRI. At one-year follow-up, he was able to play handball with complete knee extension strength (compared with the contralateral side) and was able to return to a semiprofessional level. The range of motion of the knee was 0-0-130°. The MRI at 1 year showed complete healing of the patellar tendon and the bone (). |
A 50-year-old Caucasian woman presented to a chest clinic with a 1-year history of a cough. A chest X-ray performed at the time of onset of symptoms had been normal. She had a history of allergic rhinitis and had developed a wheeze, and a provisional diagnosis of allergic asthma was made by her family doctor. There was an initial improvement in her symptoms with courses of prednisolone, but by the third course she was failing to respond. She had also been given a month's trial of a proton pump inhibitor for suspected associated acid reflux. There were no complaints of loss of weight or appetite. She had smoked 20 cigarettes daily from her youth until 10 years prior to presentation.\nOn examination, her vital signs were normal and chest examination was clear. Spirometry revealed a forced expiratory volume in 1 second (FEV1) of 2.14 litre (100% predicted) and a forced vital capacity (FVC) of 2.65 litre (93% predicted). A chest X-ray was performed which showed ill-defined shadowing in the right upper zone. The patient subsequently developed pain over the right shoulder. A CT scan revealed a 6 × 3 cm mass in the right lung apex extending alongside the right side of the mediastinum to a level above the right hilum. A provisional diagnosis of lung cancer was made. Her case was reviewed at the lung cancer multidisciplinary team meeting and further investigations were arranged: a half body FDG-PET/CT scan, a right anterior mediastinotomy and bronchoscopy. Bronchoscopy findings were normal, and cytological analysis of a transcarinal aspiration showed a small number of lymphocytes and macrophages. Similarly, the biopsies from the mediastinotomy showed reactive lymph node tissue only.\nThe FDG-PET/CT scan showed multifocal, ill-defined semi confluent areas of marked increased uptake (maximum standard uptake values (SUVmax) of up to 9.6) within areas of dense consolidation in the right lung upper lobe (Figure ). Focal intense uptake was present in right hilar precarinal and right and left paratracheal nodes (Figure ). There was no abnormal uptake outside the thorax. These findings were consistent with a T4, N3 and M0 bronchial carcinoma.\nBlood tests revealed eosinophilia with very high immunoglobulin E (IgE) of 1304 kAU/L (normal <87). A positive radioallergosorbent test (RAST) revealed Aspergillus fumigates, and Aspergillus precipitins were moderately positive, with a titre of 1 in 8. The eosinophil count was elevated at 0.48 × 109/litre, which was 4.9% of the white cell count. The erythrocyte sedimentation rate was elevated at 64 mm/hr.\nFollowing a multidisciplinary meeting to discuss the patient, the decision was made to proceed with surgery. Repeat bronchoscopy showed inflammation of the right main bronchus and segmental bronchi. A right posterolateral thoracotomy was performed. Adhesions were noted between the apex of the right upper lobe and the parietal pleura as well as the mediastinal pleura. Two poorly delimited tumours were evident in the right upper lobe, one towards the apex and the other in the region of the transverse fissure between the upper and middle lobes. Enlargement of the paratracheal, para-oesophageal and hilar nodes was noted. Biopsies were taken from the right upper lobe, paratracheal nodes, para-oesophageal nodes and hilar nodes. All showed reactive changes only on the frozen section. Following discussion with our oncologist in the operating theatre, and given the possibility that there was an occult neoplasm at the centre of one or both of the upper lobe masses, the upper and middle lobes were resected.\nA histological examination revealed a poorly defined, tan-coloured mass, 65 mm in diameter, in the upper lobe. A microscopic examination of this mass showed organising pneumonia, with numerous macrophages and giant cells. There was no evidence of necrosis or the formation of granulomata. There was an overlying pleurisy, in which giant cells were seen. All the lymph nodes examined showed reactive changes only. Further examination and staining for Aspergillus did not reveal any fungi in the samples.\nTwo months after surgery, the patient was clinically well. In view of the positive Aspergillus precipitins and elevated IgE and the cryptogenic organising pneumonia on the histology, she was put on both prednisolone and itraconazole. She has had no recurrence of her respiratory symptoms since her surgery. She is currently not under therapy, with normal inflammatory markers and no evidence of consolidation on her chest X-ray 18 months after surgery. |
A 42-year-old woman presented to the neurology emergency department with headaches, lack of mobility in right hand and foot and lost speech ability. On physical examination, there was complete Broca's aphasia and right hemiplegia. Brain computed tomography (CT) scan showed an evidence of embolic infarction in the left middle cerebral artery (MCA) zone and the treatment for cerebrovascular accident was started. Then, the heart and carotids were evaluated.\nCarotid Doppler ultrasound and electrocardiogram (ECG) were normal. The heart rhythm in ECG was normal showing a sinus rhythm. Transthoracic echocardiogram (TTE) was normal in chambers and heart valves, A suspicious mobile mass was seen in the aortic arch in assessment of suprasternal notch. To investigate further, the CT angiography of the thoracic aorta was performed and a filling defect was observed in the aortic arch. TEE was performed emergently. Hypermobile mass was seen in the aortic arch with the dimensions of 4.0 × 0.5 × 0.5. On the histopathology, the resected intra-aortic mass showed a blood clot. Regarding the nature of mass, hypermobility and lack of intracerebral hemorrhage in brain CT scan, the decision was made to urgently resect the tumor. The patient was immediately transferred to the operating room.\nMidsternotomy was performed right after receiving heparin and arterial cannulation of the right atrium was performed with a grade 7 cannula. Anonymous artery was cannulated with a cannula number 14 and the patient was cooled to 18 degrees and then was given health care assistant (HCA). The base of the anonymous artery was clamped and arterial flow was maintained through the anonymous artery cannula to 800 cc/kg. First arc opening and cutting was undertaken during septum.\nA red mobile mass of 4.0 × 0.5 × 0.5 cm was observed at the junction of the ascending aorta and arch, with a soft consistency which was attached with a pike of 0.5 × 0.5 cm in aortic tissues. The free end of the mass reached the left carotid artery at the time of mobility which was consistent with the cerebrovascular accident (CVA) signs in the patient. Mass was removed with a margin of 2 mm from the wall of the aorta and aortic wall was repaired and the patient was warmed. After taking the appropriate course of action, the patient was referred to the heart department on the second day. The patient’s clinical condition improved on the second day and she was discharged with aspirin and warfarin after 8 days. A follow-up echocardiogram one month later showed no change in the aortic arch and she had an uneventful surgical resection of the mass. |
A 48-year-old male presented with gradually progressive painless swelling of the left upper neck and preauricular region, dysphagia, change in the quality of voice and frequent disturbed sleep during night,\nA total of 4 years after undergoing transoral tumor excision for pleomorphic adenoma. On intraoral examination, there was a smooth firm bulge of the soft palate and left lateral pharyngeal wall, occluded the oropharynx. Posterior nasal examination showed the extension of the swelling into the nasopharynx.\nMagnetic resonance imaging showed large lobulated well-defined homogenously hypointense lesion on T1 WI and hyper intense lesion on T2 and STIR WI in the left para-pharyngeal space extending from the skull base to the hyoid bone. Medially, it was bulging in the nasopharynx and the oropharynx, significantly compromising the airways ().\nThe deep lobe of the left side parotid gland was not separately identified from the lesion, and it was abutting the neurovascular bundle of the parotid gland from the inner aspect.\nAfter obtaining the patient’s informed consent, surgery was planned to approach the deeper lobe of parotid gland and para-pharyngeal mass by transparotid, transmandibular swing approach in order to achieve radical excision of the mass. Superficial parotidectomy was done to preserve the facial nerve. After exposure of the parasymphysis, body and ramus of the mandible, mandibular swing access osteotomy was planned. The osteotomy cuts were planned by preserving the inferior alveolar neurovascular bundle, and the media surface of the mandible was exposed to visualize parapharyngeal structures. The facial nerve branches around the tumor were dissected out past the mass, so that the tumor could be peeled away from the facial nerve (). It was completely excised in toto. The measurements of the tumor were 16 × 10 × 4.5 cm in size (). Histopathological examination revealed the features suggestive of pleomorphic adenoma. The patient was discharged after 9 days with no facial nerve deficit. |
Our patient is a 40-year-old Asian man with complaints of bleeding and discomfort in his anus of 2 months’ duration. He was an employee with average income who did not smoke tobacco or drink alcohol. He had no weight loss or urinary symptoms, and no substantial family history. He denied any significant medical or surgical history. His abdomen was soft, non-tender, and non-distended, with normoactive bowel sounds. In examination, a mass could be touched by finger tips. The mass was large and bleeding. In subsequent examinations, blood was detected in a stool sample. His vital signs were: blood pressure, 130.77 mm Hg; respiratory rate, 18 breaths/minute; heart rate, 83 beats/minute; and temperature within normal limits. Oxygen saturation was 98% on room air on admission. In colonoscopy, a large lobular tumor was diagnosed at 4 cm above the dentate line, which was suspicious for malignancy. Various samples were taken from the tumor. The rest of his large intestine did not show a clear pathologic lesion in the colonoscopy.\nHigh-grade adenocarcinoma was reported in pathological examinations. In subsequent diagnostic procedures, his carcinoembryonic antigen (CEA) level was normal. Computed tomography (CT) scans revealed that metastatic lesions were not detected in his liver, abdominal viscera, and chest. In CT scans with or without contrast, and magnetic resonance imaging (MRI) scanning, an ectopic kidney was detected incidentally on his right pelvis without any prior urinary symptoms. The left kidney was in its original location, and both kidneys were functional. Renal function tests provided normal results.\nIn subsequent investigations done by MRI scanning for staging the tumor, a pelvic rectum tumor was reported to be interfering with the T3 N1 mesorectal lymph nodes (Figs. , ). The case was discussed in a multidisciplinary cancer team; afterward, our patient was regarded as a candidate for neoadjuvant radiotherapy. He underwent 45 GY radiation in 25 fractions to the pelvis along with capecitabine. He underwent total mesorectal excision (TME) surgery to maintain the ectopic kidney 6 weeks later. After abdominal exploration, his abdominal viscera were examined. There was no metastatic lesion in his liver and abdomen (Fig. ).\nAfter mobilization of the left colon and the splenic flexure, and the closure of the inferior mesenteric artery (IMA), in the avascular plane, the mesorectum was separated from the fascia propria, and the mesorectal lymph nodes and hemorrhoidal vessels in the anterior and pelvic nerves were fully mobilized, and the distal rectum was removed by an appropriate margin (Fig. ).\nHis right kidney was completely inside the pelvis, and while the kidney was carefully protected by the retractor, an attempt was made to minimize the damage to the ectopic kidney because there was a possibility of damage to the pelvic nerve and nephrectomy.\nThe blood of the right kidney appeared to be supplied by the right superior iliac artery. During the surgery, hematuria occurred to our patient, which was resolved by hydrating him. Then, coloanal anastomosis and temporary ileostomy were performed on our patient. He was transferred to our intensive care unit (ICU). He underwent laparotomy again due to anastomosis leakage a week following the surgery. As a result, a colostomy was performed. Postoperatively, after the reappearance of symptoms, stabilization, and healing of the wounds, he was referred to medical oncology and started adjuvant chemotherapy with 5-fluorouracil, folinic acid, and oxaliplatin (FOLFOX). Follow-up testing (for a year) included routine medical history and physical examination (every 3–6 months), blood tests such as serum CEA, colonoscopy, and radiologic imaging. He was dissatisfied with the permanent colostomy after the end of the treatment. However, the satisfying result was that his kidney was preserved (Additional file ). Before the surgery, the potential risks and damage to his ectopic kidney and the possibility of its removal were explained to our patient and his consent was obtained. His general condition is appropriate after 1 year and his quality of life has been reported to be satisfying despite the permanent colostomy. |
Patient 1 was a 38 years-old male. In May 2010, this patient was diagnosed with glioma soon after an episode of seizures. MRI showed intra-axial expansive and infiltrative lesions that were cortical and subcortical, and which affected the anterior half of the right temporal lobe and extending from the pole to the Sylvian fissure superiorly and to the right parahippocampal gyrus, posteriorly, and medially. Partial surgical resection was performed in August 2010 and the first pathologic diagnosis was astrocytoma grade II. He underwent chemotherapy with TMZ at a dose of 2,000 mg with cycles every 28 days for 5 days in the years 2011–2013, with no tumor regrowth until the beginning of 2015. At this time, he underwent MRI, which was used to compare the discrete extension of the signal alteration areas, especially the subinsular regions. In March 2015, he resumed chemotherapy with TMZ at a dose of 100 mg/day and the patient then lost 12 kg of body weight, which was associated with anorexia, insomnia, and depression. In May 2015, he suffered a seizure requiring hospitalization. In June 2015, the patient resumed the old chemotherapy regimen with TMZ (2,000 mg every 28 days for 5 days), and a follow-up with MRI; however, the tumor size continued to increase. In January 2016, the neuro-oncology team decided to discontinue treatment with TMZ considering the risk/benefit and planned a surgical re-approach. This was followed by chemoradiation and lasting 6 cycles of PCV associated with CBD. The CBD dosage was ranging from 300 to 450 mg/day.\nDuring chemoradiation, the patient had an excellent clinical performance, practiced sports and had few symptoms of fatigue and/or nausea.\nAt 1 month after the end of chemoradiation, control MRI (Figure ) was characterized by exacerbation and the ultra-precocious phenomenon of PSD with increased edema and inflammatory disease characterized by extensive areas of contrast enhancement associated with tissue hypoperfusion (not shown). MRI controls demonstrated the progressive reduction of these findings.\nThe result of a pathological study after the first surgery was astrocytoma grade II with Ki67 staining of 5%. After the second surgery, he progressed to GBM grade IV (Figure ), related to increased cellularity, frequent mitosis, presence of micronecrosis, microvascular proliferation/endothelial, Ki67 staining of 30%, and loss of ATRX expression. Biomolecular marker analysis indicated IDH-1 mutated and MGMT methylated. |
A 19 year old boy presented with bilateral slow growing and painful swellings of his achilles tendons of 4 year duration. On clinical examination each swelling was of size 5×2 cm, firm, nodular, tender and localized to the distal portion of the tendoachilles just above its insertion point to the calcaneal tuberosity (). He was more symptomatic on the right side. He was initially advised rest, analgesics and foot wear modification. However, the symptoms did not subside and the situation worsened resulting in significant disability with limitation of his walking distance.\nInitial radiographs of both ankles showed homogenous soft tissue shadow in the lower halves of tendoachilles (). Magnetic resonance imaging showed localized homogenous hyper intense signals with fusiform swellings of tendoachilles (). Surgical excision was offered and the right sided one was selected first as it was more symptomatic. The swollen tendinous portion measuring 6×3 cm was excised () followed by reconstruction using the ipsilateral peroneus brevis tendon. Immediate and early post-operative period was uneventful. Biopsy of the excised specimen revealed it to be a xanthoma characterized by the accumulation of mononuclear cells with foamy cytoplasm and multinucleated giant cells with high concentration of cholestanol. Thus a diagnosis of cerebro-tendinous xanthomatosis was made.\nOn detailed retrospective inquiry, he had surgery for bilateral juvenile cataracts at the age of 8, along with history of chronic intractable diarrhea. He was coherent and co-operative but slow cerebrated with low intelligence. There were no central nervous system symptoms like convulsions, abnormal gait or incoordination of movements. Family history revealed that he was a child of a consanguineous marriage. Other sibling, a girl was normal. He was short statured measuring 144 cm in height (), thin built with a peculiar yellow conjunctiva. Hematological and biochemical investigations including liver function tests, lipid profile, thyroid function tests and ultrasound examination of abdomen were normal.\nA special test - serum cholestanol level was elevated to 4.37 mg/dL (normal value: 0.02-0.12 mg/dL). Based on the clinical picture, pathological and serological analysis, the diagnosis of cerebro-tendinous xanthomatosis was confirmed and was kept on medical therapy with chenodeoxycholic acid (CDCA), a synthetic bile acid.\nRESULT: He was kept under regular follow up with neurologic and neuropsychological evaluation, musculoskeletal examination and serum cholestanol estimation. He started to improve after 3 months of continued medication. According to the latest follow up of 23 months, he achieved complete improvement in his mental status and normal values of serum cholestanol. The pain on the left (non-operated) tendoachilles disappeared completely although swelling persisted. His walking distance improved. However, had reappearance of the swelling in the reconstructed tendon on the right side, although was not painful () |
A 35-year-old male presented with inferior wall ST-elevation myocardial infarction (STEMI) []. The patient was a known diabetic, hypertensive, and with dyslipidemia. He had a history of prior inferior wall infarction a year ago, for which he was treated by primary percutaneous intervention (PCI) to the right coronary artery (RCA). During prior ACS episode, the treating physician noted an additional vessel originating abnormally from the right coronary sinus (RCS) which was assumed to be left circumflex artery (LCX). Left injection was not be taken at that time as patient was hemodynamically unstable. He was discharged on secondary prevention drugs including dual antiplatelet therapy and was advised coronary computed tomography to delineate the course of anomalous artery which patient did not undergo. Noncompliant on treatment, he presented to us again with inferior STEMI. This time he was hemodynamically stable. Transthoracic echocardiogram revealed segmental wall-motion abnormalities in inferior, posterior, basal septal, and lateral walls of left ventricle. His cardiac biomarker (cardiac troponin I) levels were elevated (1.5 ng/ml, N < 0.4 ng/ml), but the rest of blood investigations including hemogram and renal function test were within normal limits. The patient was taken up for primary PCI through right radial access. Despite multiple attempts, left main coronary artery could not be engaged. Hence, a RCA injection was taken first. A short common vessel was noticed in the right sinus, from which two coronary arteries were emanating []. One of the vessels continued as RCA with a normal course. The second vessel originating from the common ostium was left coronary artery which was dividing into LAD and LCX []. Two discrete lesions were noted in RCA, and a stent was visible in proximal RCA which was patent. Proximal to stent, there was 70% stenosis and distal RCA also had 70% stenosis. LAD was also diseased in its proximal segment with 70%–80% stenosis. Because of multiple lesions in RCA and LAD and diabetic status of patient, he was given option between CABG and multivessel PCI. Fortunately, he opted for multivessel PCI. RCA was taken up for PCI, and lesion was wired with a floppy wire (Runthrough NS, Terumo Inc, Somerset, NJ, USA). The lesions were predilated using 2.5 mm × 12 mm semi-compliant balloon catheter []. After that two nonoverlapping everolimus drug-eluting stents deployed in sequence, proximal one first followed by the distal one. The proximal stent was 3.5 mm × 24 mm in size, while the distal stent was 3.0 mm × 18 mm in size, respectively. Both stents were postdilated with upsized noncompliant balloons as IVUS run could not be performed. A staged PCI was planned for the noninfarct-related artery lesion in the proximal LAD.\nThe patient remained stable throughout the hospital course and was discharged on dual antiplatelet therapy (aspirin 150 mg and ticagrelor 90 mg bid) and other guideline-directed medical therapy for secondary prevention. |
A 28-year-old female patient with a chief complaint of swollen gums was referred to the Department of Periodontology, Navodaya Dental College and Hospital, Raichur (India) completing her orthodontic treatment at a private setup. On clinical examination, patient presented with abundance of calculus, with oral hygiene index of 3.3 referring to poor oral hygiene status. Patient presented with generalized erythematous gingiva with diffuse gingival enlargement. There were generalized deep true periodontal pockets with average probing pocket depth of 7-8mm with Grade II to Grade III mobile teeth in relation to11, 21 []. Patient was systemically healthy.\nA provisional diagnosis of generalized chronic periodontitis with inflammatory gingival enlargement was made. Radiographic investigations such as orthopantomograph (OPG) [] and full mouth intraoral periapical radiographs (IOPA) were advised []. OPG revealed moderate-to-severe bone loss. Angular defect was best appreciated in IOPA of tooth region 11-21. The inference from the clinical and radiographic investigations leads to the definitive diagnosis of generalized chronic periodontitis. No radiographs were available for comparing the condition before the orthodontic treatment.\nA comprehensive treatment plan was proposed which included an initial nonsurgical therapy, full mouth flap surgery with platelet rich fibrin (PRF), hydroxyapatite (HA) bone graft and subsequent esthetic rehabilitation along with reevaluation in between the treatment phase. The patient was explained about the treatment plan and informed consent was obtained for the same. Routine blood examinations were carried out and nothing of relevance was noted.\nTreatment started with Phase-I therapy that included patient motivation, thorough scaling and root planning with proper oral hygiene instructions Patient was on maintenance phase and recall visits were scheduled once in a month to assess plaque control measures and evaluate her response to Phase-I therapy. Re-evaluation for clinical parameters such as gingival inflammation, probing pocket depth, gingival enlargement and mobility was done after 3-month period Extracoronal wire and composite splint was fabricated for tooth region 13-23 before regenerative surgical procedure to manage the mobility associated with the teeth 11, 21 On achieving an improvement in patient's gingival health, decrease in clinical signs, and symptoms of gingival enlargement and inflammation. Surgical intervention was performed, that included internal bevel gingivectomy and modified Widman flap surgery to manage the gingival enlargement as well as the persistent pockets Regenerative procedures were performed wherever angular defects were present. Tooth region 11-21 showed a two-walled defect []. Whereas 16 and 26 both showed three walled defect with Grade II furcation involvement seen in 26 []. After complete debridement, root bio-modification was done with tetracycline (prepared by dissolving 500 mg capsule in 10 ml of saline). PRF was prepared by drawing 10 ml of patient's whole venous blood from antecubital fossa into sterile vacutainer tubes without anticoagulant. These tubes were then placed in the centrifugal machine at 3000 rpm for 10 min. A fibrin clot is then obtained in the middle of the tube just between the red corpuscles at the bottom and the acellular plasma at the top. The PRF thus obtained is mixed with the hydroxyapitite bone graft (sybograf ™ - Eucare pharmaceuticals) with particle size ranging between 600 and 700 μand placed in the 11–21 and 16 regions. The PRF was compressed on a glass slab to form a membrane and placed in the furcation defect of 26. Flap sutured and periodontal dressing placed Postoperative instructions were given to the patient. The patient was asked to refrain from mechanical plaque control measures in the surgically treated quadrant for 2 weeks and no intrasulcular brushing for 8 weeks. Advised soft diet >2 weeks. The patient was advised to use chlorhexidine 0.2% rinse until the completion of the treatment. Patient was prescribed with the following antibiotic and analgesic regimen, amoxicillin 500 mg thrice daily for 5 days, ibuprofen twice a day for 3 days. Sutures were removed 7 days postsurgery Patient was scheduled for maintenance therapy, recalled every 3 months in the first year and then recalled every 6 months. Patient was followed up for 2 years. Re-evaluation at the 2-year period showed an improvement in the clinical parameters, with complete resolution of gingival enlargement and establishment of knife edge margin with pink gingiva. There was a decrease in the mobility of the teeth 11, 21 from Grade III to Grade I []. Splint in the 13-23 region was subsequently removed. Postoperative radiographs at 9 months showed significant bone fill in the areas where regenerative techniques were performed [Figures and ].\nDespite extreme care and caution, patient had loss of interdental papilla which presented as black triangles in the anterior esthetic region as a result of the extensive periodontal surgery. Patient was explained about the periodontal plastic surgical procedures. However, due to her unwillingness for another traumatic and extensive surgery and further orthodontic treatment, an alternate treatment modality of fabricating a gingival prosthesis was preferred. Prosthesis was fabricated from flexible valplast material. Custom tray was fabricated from the diagnostic impression of the maxillary arch. After border molding, a secondary impression was made with addition silicone material. Using injection molding technique valplast prosthesis was fabricated. The patient was instructed with the use of prosthesis. The patient was very much satisfied with the esthetics achieved with the prosthesis []. |
A 29-year-old primigravida patient was admitted to the labor and delivery suite at 34 weeks of gestation with a diagnosis of preeclampsia. Her systolic blood pressures ranged between 140 and 156 mmHg and her diastolic blood pressures ranged between 91 and 108 mmHg. Her workup revealed hyponatremia and hypoalbuminemia with no other abnormalities. She was observed for signs of severe preeclampsia. At 35 weeks of gestation, the patient started having vaginal bleeding and mild abdominal pain. Placental abruption was suspected. The patient was remote from delivery. She was counseled and consented to undergo a primary cesarean section.\nAt the time of cesarean section and upon entering the peritoneal cavity, a copious amount (around 800cc) of milky peritoneal fluid was evacuated (). The exact nature and etiology of the fluid were unknown to us at that time. Surgery was completed. A live male newborn was delivered with a birth weight of 2020 g. Apgar scores were 8 at 1 minute and 9 at 5 minutes. Amniotic fluid was clear and an estimated 20% placental abruption was clinically evident. A general surgeon was called for intraoperative consultation. The abdomen was explored by the general surgeon; no abnormalities were noted in the cecum, small bowels, omentum, uterus, ovaries, or fallopian tubes. A Jackson-Pratt (JP) abdominal drain was placed at the end of the procedure.\nThe patient was started on intravenous antibiotics for the possibility of intraperitoneal infection. Culture of the peritoneal fluid was negative. Triglyceride level of the peritoneal fluid was 863 mg/dL (diagnostic of chylous ascites). The patient had no clinical evidence of pancreatitis.\nThe intravenous antibiotics were discontinued after peritoneal culture results were available. The drained peritoneal fluid became progressively serous. The JP drain was removed on the third postoperative day after drainage became clinically insignificant.\nThe patient received magnesium sulfate therapy for seizure prophylaxis for 24 hours postpartum. She remained hemodynamically stable and regained her bowel function. She was discharged from the hospital on the fourth postoperative day.\nA CT scan of the abdomen and pelvis was performed at 6 weeks postpartum and showed no evidence of ascites and no abdominal masses. |
A 62-year-old woman was referred to the authors' hospital for evaluation of a corneal ulcer in her left eye. With the patient's consent, a review of the clinic record was conducted. Ten years previously she received an allogeneic hematopoetic stem cell transplant for treatment of myelodysplasia syndrome. Her course was complicated by the development of cGVHD affecting the liver, skin, esophagus, mouth, and eyes. Severe keratoconjunctivitis sicca had been treated with a PROSE lens for more than 2 years prior to developing this corneal ulcer. Her medical status was compromised by systemic steroid dependence and by steroid induced diabetes mellitus.\nAfter presenting with a corneal ulcer in her left eye, the patient was empirically treated with topical 0.5% moxifloxacin (Vigamox, Alcon) that was applied 6 times a day (once before the PROSE lens was inserted in the morning, 4 times during the day inside of the PROSE lens reservoir, and once again at night after the PROSE lens was removed). After failure to improve on 4 days of this treatment, she was referred to Massachusetts Eye and Ear Infirmary for additional corneal ulcer evaluation, culture and modification of treatment. Upon presentation, the central cornea was opaque and neovascularized with tissue loss of approximately 60% of the corneal thickness. There was an epithelial defect that measured 2.5 mm by 1 mm, with an underlying 2 mm by 1 mm infiltrate. Microbiological smears and cultures were performed with the specimen from corneal scraping. No bacteria or fungi were evident with Gram stain or Calcofluor white stain. At the time, frequency of application of topical moxifloxacin was increased to every 2 hours while awake (approximately 8 times per day), delivered as one drop added to the PROSE reservoir after removal and cleaning reinsertion of the device and replenishment of the reservoir with preservative free saline (B).\nFour days after culture and modification of antibiotic delivery regimen, the corneal ulcer resolved, with re-epithelialization of the cornea surface and resolution of the infiltrate. A strain of E. coli cultured from the lesion in 5% sheep blood agar media exhibited resistance to fluoroquinolones, trimethoprim/sulfamethoxazole and ampicillin and ampicillin-sulbactam according to breakpoints set by the Clinical Laboratory Standard Institute-CLSI (). The strain carried multiple single point mutations in the quinolone resistance-determining region (QRDR) of gyrA (Ser83Leu, Asp87Asn), parC (Ser57Thr, Ser80Ile) and parE (Leu416Phe) genes. These mutations were correlated with varying levels of resistance to the fluoroquinolones: ciprofloxacin (MIC 256 μg/mL), levofloxacin (8 μg/mL) and moxifloxacin (16 μg/mL), as determined by reference broth microdilution. |
A 67-year-old South Asian female, who was diagnosed with diabetes mellitus with peripheral neuropathy and hypertension, was admitted with right lower limb necrotizing fasciitis to the surgical unit of our institution. A thorough wound debridement was performed under subarachnoid block. Wound swab cultures yielded methicillin-resistant Staphylococcus aureus (MRSA) sensitive to teicoplanin. Concomitant blood and urine cultures and rest of the MRSA screening were negative. As she had developed severe reactions to clindamycin, teicoplanin, and penicillin group antibiotics, desensitization for meropenem was carried out in a tertiary-care center under the guidance of a consultant immunologist. Simultaneously, several wound debridements were carried out under spinal anaesthesia. The patient was transferred back to our institution for further wound care. She was on a urinary catheter and utilizing a wheel chair for mobility. A repeat wound debridement was planned under spinal anaesthesia. She had mild back pain with no spinal tenderness or new onset or worsening neurological deficits. History did not reveal any trauma to the back. Throughout her hospital stay, the clotting profile was normal with normal platelet counts, and spinal anaesthesia was conducted under strict aseptic conditions at all instances. While performing the most recent subarachnoid block, the anaesthetist noticed the absence of cerebrospinal flow, and when the spinal needle was withdrawn, pus was noted at the hub (Figures –).\nThe procedure was abandoned. With the suspicion of SEA, she was transferred to the nearest neurosurgical center. An urgent MRI of the spine revealed a large epidural abscess with cauda equina displacement and compression with edema involving cauda equina fibres ().\nVertebral osteomyelitis and discitis were excluded. There was no evidence of psoas abscess or intra-abdominal collections. Urgent bilateral L4 laminectomy with epidural abscess evacuation was carried out under general anaesthesia. The abscess aspirate grew methicillin-sensitive Staphylococcus aureus which was treated with intravenous meropenem for 28 days. Even though the sensitivity for cloxacillin and ceftriaxone was noted in the aspirate, the recent history of severe reactions to penicillin group antibiotics, the cross reactivity between third-generation cephalosporin (ceftriaxone in our patient) and penicillin groups, and her desensitized state for meropenem prompted preference of the latter. She made a complete recovery without general or neurological complications and was discharged 6 weeks later. The routine follow-up in the surgical clinic excluded any recurrence of the SEA. |
A 50-year-old female patient presented to us with tingling sensation over both lower limbs which affected the right side more than the left side for the last 6 weeks. She then developed weakness of both lower limbs which initially affected the right side manifesting as heaviness in legs followed by stiffness and difficulty in walking. Weakness was insidious in onset but rapidly progressed to involve both lower limbs that patient was unable to stand without support in the next 4 weeks. When she came to us, both her lower limbs were severely spastic. Power in both her lower limbs was MRC 1/5. Tone and power in both upper limbs were unaffected. Reflexes in both lower limbs were exaggerated. Bilateral plantars were extensor. She had no sensory deficits. The patient underwent magnetic resonance imaging (MRI) of the cervicodorsal spine [] which revealed an heterogeneously enhancing intraspinal tumor at D1–D2 level on the right side causing cord compression and pushing it to the opposite side. The tumor extended into the right D1–D2 foramen with extension into paravertebral area. Noncontrast computed tomography cervical spine showed mild degenerative changes with anterior osteophyte formation and right facet joint arthropathy from C4 to C7 level. The right side D1–D2 neural foramina was wider and the D1 pedicle thinned out as compared to the left side []. With a possible diagnosis of peripheral nerve sheath tumor with cord compression, the patient underwent D1-D2 laminectomy using posterior midline incision in the prone position. A large reddish pink color extra dural tumor extending from C7 to D2 level was seen on the right anterolateral side of the spinal canal pushing the cord to the left side. The tumor began to bulge out of the spinal canal even as the laminectomy was being performed. D1 and D2 right-sided facet joint was also excised to create a corridor to approach the foraminal and extraforaminal/paraspinal part of tumor. There was a clear plane of cleavage between the tumor and adjacent tissues. Right D1 pedicle was thinned out due to compression by the tumor. Gross total excision of the intraspinal part of the tumor was done. The tumor was densely adherent to the right side D1 nerve root which was sacrificed. The wound was then closed in layers. Power and spasticity in both lower limbs started to improve after surgery. At the time of discharge, power in the right lower limb was 2/5 and in the left lower limb was 3/5. Histopathological examination [] revealed sheets of monomorphic small round cells with scanty cytoplasm and high nuclear/cytoplasmic ratio and hyperchromatic round nuclei consistent with diagnosis of PNET. It was positive for neuron-specific enolase and CD99. It showed focal expression of synaptophysin and cytokeratin. It was negative for leukocyte common antigen, CD3, and CD20. The Ki-67 proliferation index was 20%. MRI brain and whole body positron emission tomography (PET) scan showed no other tumor sites or distant metastases, and thus a diagnosis of psPNET was established. The patient was then referred to an oncologist for adjuvant therapy. |
A 72-year-old male with a history of essential hypertension and without any history of abdominal surgeries presented to the emergency department with several hours of acute onset severe abdominal pain. The pain was colicky, non-radiating, and localized to the periumbilical area. He tried bismuth subsalicylate at home with no relief of symptoms. He had not passed any flatus and had no bowel movements since the onset of symptoms. He denied any fever, chills, diarrhea, nausea, or vomiting. He denied any past occurrences of similar episodes. On further questioning, he admitted to eating a large amount of fried, unshelled sunflower seeds the day before the symptoms started. He had a colonoscopy ten years ago which revealed a tortuous colon but no polyps or diverticulosis. There was no history of colon cancer or inflammatory bowel disease in the family. On presentation, vital signs were within normal limits. The abdominal exam revealed decreased bowel sounds, tenderness in the periumbilical area and right lower quadrant, no rebound tenderness was noted, and McBurney's sign was negative. All laboratory investigations, including a complete blood count, comprehensive metabolic panel, and lipase, were within normal limits. A CT of the abdomen was pertinent for a transition zone in the terminal ileum with wall thickening, fat stranding, and dilation of the bowel proximal to the transition zone (Figure ). These findings were indicative of small bowel obstruction secondary to possible distal ileitis. The patient was managed conservatively with intravenous fluids and was kept nil per os.\nThe next morning, the patient had two large volume bowel movements, after which his abdominal pain significantly improved. Colonoscopy was performed and showed nonspecific ulceration proximal to the ileocecal valve, suggestive of trauma, possibly from ingestion of a large number of sunflower seeds (Figure ).\nAt this point, early Crohn's disease was still in the differential. The pathology report showed focal surface epithelial erosion and florid lymphoplasmacytic and neutrophilic infiltrates. No granulomas were seen in the tissue (Figure ). These findings favored traumatic ulceration, and Crohn's disease was ruled out. The patient continued to improve clinically with complete resolution of his abdominal pain. |
A 50-year-old male patient presented with a 2-year history of left-sided typical HFS. Painless irregular clonic contraction of the facial muscles began initially in the orbicularis oculi muscle of the lower lid. It gradually spread to other muscles innervated by the facial nerve on the left side of the face, including platysma. The paroxysm was induced or aggravated by emotional tension, stress, and voluntary and reflexive movements of the face. He had significant difficulty in his work and social life despite 2 times of botulinum toxin injection. Medical treatment with carbamazepine (up to 600 mg) and baclofen (30 mg) was not effective. He was referred for surgical treatment. His medical history was unremarkable. His physical and neurologic examinations were normal, including hearing. No tinnitus or discernible noise heard in his left ear was found. Only typical nature of clonic hemifacial spasm was evident. Abnormal synkinesis between the orbicularis oculi and orbicularis oris muscles was found by the electromyographic examination of the blink reflex. Despite typical HFS, there was no discernible vascular structure in the REZ of left facial nerve (). However, a meatal loop of AICA abutting to the cisternal portion of the facial nerve was found.\nUnder the impression of HFS caused by neurovascular compression of distal facial nerve, standard microsurgical procedure was performed as described previously [, , ]. In addition to intraoperative monitoring of BAEPs, LSR, which is an abnormal muscle response demonstrated by EMG recordings from mimic muscles that are innervated by a different branch of the facial nerve [], was also monitored throughout the operation. The entire course of the facial nerve and offending arteries were exposed under microscopic vision. Upon exposure of the REZ of the facial nerve, there was no offending vessel in the REZ as expected (). The distal, cisternal segment of the facial nerve was found to be bent by a meatal loop of the AICA (). A small piece of Teflon felt was interposed between the facial nerve and the meatal loop of the AICA with extreme care not to stretch the internal auditory artery and the distal facial nerve (). After interposition of Teflon felt, LSR immediately disappeared and BEAP was stable also (). The closure of the dura and wound was performed in routine manner. The HFS resolved completely following the surgery. The postoperative course was uneventful with no signs of facial weakness or hearing impairment by pure-tone audiometry. No recurrence of HFS or neurologic sequelae was evident at a 12-month follow-up. |
A 37-year-old male was referred with a diagnosis of episcleritis with peripheral keratitis to our center for further management. He had a history of increasing ocular discomfort in the right eye of one week duration. He was not on any medicines at the time of presentation. On examination, his best-corrected visual acuity was 20/20 in both eyes. Slit-lamp examination in the right eye revealed marginal keratitis between 12'0 clock to 2'0 clock positions along with a pannus []. Eversion of the eyelid revealed an insect wing on the superior tarsus with a localized conjunctival granuloma at the area of the foreign body []. The lid margins were healthy with no evidence of blepharitis. The marginal keratitis corresponded to the area of the foreign body and the conjunctival granuloma. Rest of the ocular examination was normal. Left eye examination was normal. The foreign body was removed under topical anesthesia with a forceps and sent to a microbiologist for confirmation. Confocal microscopy (Rostock corneal Module II, Heidelberg retinal tomogram, Heidelberg, Germany) at the area of the keratitis and corneal scraping (KOH, Grams stain and culture) were negative for an infectious etiology. Topical treatment initially with antibiotics (ofloxacin, four times a day for a week) and lubricants (1% carboxy methyl cellulose, six times a day) followed by a course of steroids started three days later (dexamethasone, four times a day for five days) and tapered over three weeks alleviated his symptoms and caused resolution of the conjunctival granuloma over four weeks.\nOur case highlights the need for a complete ocular examination in every case of keratitis or scleritis. Insect wing foreign bodies on the cornea and the peripheral limbus causing vascularization, infiltration and secondary bacterial infections have been reported.[] There is only an isolated report of an insect wing tarsal foreign body causing a localized conjunctival granuloma.[] However, there are no reports of marginal keratitis associated with an insect wing tarsal foreign body (Medline search). When a foreign body gets lodged in the conjunctiva, initially, there is an acute inflammatory response in the form of exudation of plasma and fibrin. This normally dislodges the foreign body. However, when the foreign body has a large surface area, as in this case, this mechanism may be insufficient and the foreign body becomes embedded. This is followed by a chronic inflammatory response resulting in the formation of a granuloma containing epitheloid and foreign body giant cells.[] The presence of the foreign body and the exotoxins released by the bacterial flora around the foreign body may have resulted in sterile infiltrates (marginal keratitis) in the cornea. It is also possible that the corneal findings were secondary to the abrasive effect of the foreign body. Any sustained paralimbal inflammation evokes a fibrovascular proliferation in the form of a pannus as seen in our case. |
A seven-month-old female child presented with history of mass lesion in the left lower lid since birth which was progressively increasing in size. Examination revealed a swelling in the lateral two-third of left lower lid, bluish in color and measuring 3 × 3 cm. The swelling was soft, fluctuant, with ill-defined borders and the surface had engorged blood vessels. It was nontender and did not increase in size on crying. The lower tarsal conjunctiva was everted and mechanical ectropion was visible []. The right eye was normal. Systemic examination revealed a ventricular septal defect and patent ductus arteriosus. Computed tomography (CT) scan revealed an enlarged orbit with no bone erosion []. The cystic lesion occupied most of the orbit displacing the microphthalmic globe and extraocular muscles superiorly. Neuroimaging excluded any communication with the globe or central nervous system. Anterior inferior orbitotomy with total cyst excision was done. the superior part of the cyst wall was found merged with the inferotemporal aspect of the sclera. Using gentle blunt and sharp dissection under the microscope, the cyst could be separated from the globe without breaching its integrity. The eye ball was found to be microphthalmic []. Corneal diameter measured 5 × 6 mm.\nThe specimen consisted of a flattened cyst like structure measuring 2.5 cm in diameter. It had congested outer surface and smooth inner surface. There were large areas containing multiple cysts. Microscopic examination revealed tissue derived from all three germ layers. The wall of the cyst was lined focally by tall columnar epithelium []. There was neuroglial tissue which was confirmed by the glial fibrillary acidic protein (GFAP) immunostain. Surrounding areas showed presence of fat, mature smooth muscle and nerve bundles. The tissues were mature, without mitotic activity [–].\nPostoperative evaluation revealed a microphthalmic eye with no light fixation. The ocular movements were present with some limitation in abduction only. Microcornea was opaque with no view of anterior segment details. Patient's parents were explained about the poor visual prognosis and the need for ocular prosthesis. One year postoperatively there was no recurrence. |
A 6-year-old boy presented with a history of lower back pain for the past 10 days with worsening of symptoms at night time, disturbing his sleep, and restricting his normal activities and play. He gave a history of a trivial fall on the back at the school, a few days before presentation. He had no other constitutional symptoms. The child was initially managed with local analgesic creams and massage therapy. He was later taken to family physician who had advised symptomatic therapy attributing it to mechanical and postural causes. However, in view of worsening of pain, the child was evaluated for tuberculosis spine. On examination, child had no lymphadenopathy or hepatosplenomegaly. On local examination, there was tenderness over the lumbar spine. Antalgic gait and scoliosis were present. The rest of the musculoskeletal system and neurological examination was normal.\nBlood investigations revealed normal erythrocyte sedimentation rate and C-Reactive protein. Chest X-ray showed normal lung and Mantoux and sputum for Acid-Fast Bacilli was negative. The spine X-ray showed collapse of L4 vertebral body. With a provisional diagnosis of infective spondylitis, magnetic resonance imaging (MRI) spine done showed a lesion involving the right side of the L4 vertebral body and pedicle, with extension into the right L4–L5 neural foramen [ and ]. Pediatric oncologist opinion was sought and advised biopsy of the lesion. Computed tomography-guided biopsy of L4 vertebra showed aggregates of eosinophils and histiocytes having nuclear grooving and there was no evidence of granulomas. By immunohistochemistry the lesional cells were diffusely positive for S100, vimentin and focally positive for CD68 and CD1a, confirming the diagnosis of LCH. The work up for diabetes insipidus was normal. Bone scan showed abnormal increased tracer uptake only in the region of body of L4 vertebra. Bone marrow aspiration and biopsy to rule out marrow involvement was normal. He was started on LCH III protocol and brace application for spine stabilization.\nThe pain disappeared within 2 weeks of chemotherapy. MRI lumbosacral spine after 6 weeks of chemotherapy showed compression fracture of L4 vertebra with the lesion predominantly confined on the right side involving the pedicle and the lamina and no soft tissue involvement [ and ]. The child had completed maintenance chemotherapy, is completely symptom-free now and has normal daily activities. |
A 64-year-old woman was admitted to our hospital with a pulsatile mass swelling over the left cervical region and right hemiparesis after cough for 1 day. She had a history of fibromatosis, but no previous history of trauma, operation or inflammation in this region. On physical examination, a 5-cm large pulsatile swelling with blowing bruit was found over the left cervical region and multiple fibromas were found in regions of face, neck, and trunk. Neurological examination revealed muscular strength of grade IV according to the ‘manual muscle test (MMT)' grading system and hypoesthesia of right limbs, including touch and pain sensation. Computed tomography angiography (CTA) and DSA revealed a left high-flow internal carotid-jugular fistula at the first cervical level and twisted left internal carotid artery (ICA) (Figures ). Blood flow from the vertebrobasilar artery and right ICA via the circle of Willis supplied the left anterior cerebral artery and middle cerebral artery and drained backwards into the petrosal segment of left ICA (Figures ).\nWe chose embolization of both the fistula and parental artery, because no appropriate covered stent could be used to pack the fistula in the condition of reservation of left ICA. The purpose of first endovascular treatment was to isolate the fistula, by blocking both the backward blood flow from right ICA and the forward blood flow from left ICA. A Headway-21 stent catheter was selectively inserted into the distal part of fistula in the segment of carotid cavernous sinus, meanwhile an Echelon-10 microcatheter was placed in the distal part of stent catheter. One LVIS 5.5*30 mm stent, with its characteristic relatively compact mesh, was used to cover the distal part of fistula to avoid coils being pushed into internal jugular vein and heart (Figure ). However, the 3.5 cm fistula could not be covered by the LVIS stent. Therefore, 10 coils were additionally used one by one, to block the fistula from the distal to proximal part through an Echelon-10 microcatheter (Figure ). When considering the fast-backward blood flow from the right ICA after embolization, we could not guarantee the safety of blocking by Onyx. We chose one detachable balloon to block the ICA near the proximal part of fistula to stop the forward blood flow, and the backward blood flow would be stopped in the second treatment (Figures ).\nThe blowing bruit was reduced significantly after the first treatment, but became worse after 2 months later. The patient was admitted to our hospital again, and DSA revealed residual blood flow in the fistula from the left ICA and premature balloon deflation (Figures ). It was fortunate that LVIS stent blocked the balloon into the internal jugular vein. An additional 13 coils were used to pack the fistula with double-microcatheter techniques, under the multi-angle DSA projection, to avoid the coil protruding into the internal jugular vein (Figures ). Afterwards, two detachable balloons were used to block the proximal part of left ICA again (Figure ). DSA revealed the forward blood flow disappeared, the low-flow backward blood from right ICA still supplied the fistula, and a normal ipsilateral jugular vein (Figure ). Because the fistula was mostly blocked, and the low-flow backward blood may promote the formation of thrombosis in the distal part of fistula, we stopped the second endovascular treatment and planed a DSA examination to evaluate the effect of embolization and to determine further treatment. After this treatment, the patient did not feel the blowing bruit or any other discomfort.\nHowever, the patient complained of swelling and pain in the left occipitocervical region 2 days later. Physical examination revealed a large subcutaneous mass, with volatility and severe tenderness. Emergent CT revealed a subcutaneous hematoma in left occipitocervical region, without abnormalities in brain (Figures ). DSA in the hybrid operation room showed the coils in the fistula were stable without no forward blood flow in the proximal part of fistula. However, there was still a little backward blood flow through the right ICA and vertebrobasilar artery into right internal jugular vein, via the distal part of fistula. The lower part of fistula was not shown, because the blood drained into the right internal jugular vein through the sigmoid and transverse sinus reversely (Figures ). It was suggested that the occipitocervical subcutaneous hemorrhage was caused by poor ipsilateral jugular drainage, which was the result of the formation of thrombosis in fistula extending to the left internal jugular vein, thus blocking forward and backward reflow. We inserted an Echelon microcatheter through the left vertebrobasilar artery-posterior communicating artery, then selectively to petrosal segment of the left ICA, where 3 coils were put in, and then a 1.2 ml Onyx-18 was injected slowly. DSA examination after this procedure revealed that the backward blood flow distal to the fistula disappeared (Figures ). An open operation to remove the hematoma was preformed after the interventional operation. Multiple capillary hemorrhages were found from behind the sternocleidomastoid muscle after removing clots. The operation to stop bleeding was successful, and the postoperative course was uneventful. A 6-month follow-up DSA demonstrated a totally occluded fistula allowing the patient to engage in light manual labor with normal neurological functioning after the operation (Figures ). |
A 44-year-old male employee presented to the hospital in Fars Province, Iran, with decreased level of consciousness. The patient had no personal or family history of medical problems, and no history of head trauma.\nThe patient began occasional smoking of opium 14 years prior to admission, which increased to daily smoking for the decade leading up to admission. He had a distant history of occasional use of alcohol; however, he stopped using it eight years prior to admission.\nMoreover, the patient had smoked heroine for six years prior to admission; however, there was no history of cannabis or cocaine use in the past.\nEight months prior to admission, the patient commenced smoking methamphetamine, twice weekly, which then increased to daily use in the two months leading up to admission. From the time he commenced daily smoking of methamphetamine, the patient developed persecutory delusions, believing he was being followed by an agent from. He also experienced persecutory delusions regarding his neighbors and had a visual hallucination of a man on his roof. He had a poor appetite and severe insomnia.\nIn short, when he was brought to hospital by his wife, he had been smoking heroine for six years and methamphetamine daily for two months. Physical and neurological examinations were normal. Serology for HIV and hepatitis were negative. Drug screening was positive for methamphetamine and morphine (consistent with opium and heroin use) and negative for cannabis, ecstasy, methadone, buprenorphine, benzodiazepine and alcohol.\nThe patient had taken methamphetamine on the morning of admission. By that evening his level of consciousness was reduced and he was drowsy. He was given buprenorphine 2 mg sublingually twice daily, to reduce opioid withdrawal, and closely monitored. On the second day of admission his drowsiness had increased and it was difficult to arouse him to answer questions. He became disoriented in time and place. On the third day, his condition was further deteriorated and could not speak or take food.\nOn the fourth day of admission, the patient was considered to be in mortal danger and he was given emergency ECT using a Thymatron TM (System IV, Company IND, USA). The electrodes were applied bilaterally, he was administered two seizures during the same anesthetic multiple monitored ECT. Fourteen hours later he was alert and orientated, although still with some psychotic thinking and hallucinations. There was no change on the fifth day.\nOn the sixth day, multiple monitored ECT, as described was repeated. Five hours later the patient was alert and orientated in time, place and person, and free of psychotic features.\nSingle ECT was administered on 9th, 11th and 13th days and he was discharged on the 14th day. He received further single ECT on the 16th and 18th days (as an out-patient) and remained well.\nWithin a week or so, however, he again began to smoke amphetamine and was soon psychotic as before, with hallucinations and delusions. Multiple monitored ECT was again administered, and some hours later, he showed partial improvement. He was administered double ECT on two further occasions over the next 5 days and was discharged free of psychiatric symptoms.\nThe patient was followed-up and consumed buprenorphine for 6 months. He denied any further methamphetamine use and his mental state remained unremarkable. |
We present a case of a 42-year-old man already operated 10 years ago for CE of the liver and 3 years ago for mediastinal and primary spinal disease involvement.\nHe comes to our attention in 2010 after access to our emergency department for progressive paraparesis since 1 week with inability to walk, level of anesthesia below D2 associated with worsening and marked sphincter disorders, and pain associated with cervical-dorsal refractory to analgesic therapy.\nComputed tomography (CT) () demonstrated cystic tissue localized to the mediastinum and the paravertebral muscles with the erosion of the second rib and the vertebral bodies of D1 and D2.\nCervicodorsal spine MRI with intravenous injection of gadolinium () showed multiple and heterogeneous lesions located in the intraspinal extradural space at D1-D2 level, with severe spinal cord compression and involvement of the transverse processes, spinous processes, the left laminae, the second rib, the mediastinum on the right side, and cervical paraspinal muscles from C4 to D2. The patient was subsequently treated with an anthelmintic drug (Albendazole) and steroid therapy for the neurologic deficit. The patient underwent a first surgical approach by right sided paramedian longitudinal incision from C4 to D2 with the removal of the hydatid cyst (2 × 5 cm) located within the paraspinal muscle (); the second surgical approach was performed in the same surgical procedure to remove numerous smaller hydatid cysts () with a median posterior approach, performing decompressive laminectomy and spinal stabilization from C6 to D4 with Universal Clamp Spinal Fixation System-Zimmer ().\nMRI performed after surgery showed the persistence of a small cyst located in the body of D2. It was decided with the thoracic surgeon to remove the small residual cyst during surgery for the removal of mediastinal cysts, with an anterior approach; however, the patient refused the thoracic surgery.\nThree years later (in 2013) after he voluntary stopped medical treatment, the patient develops a rapid worsening of spinal deficits with motor deficit predominantly on the right side with progressive paraparesis, neuralgiform distal pain, marked sphincter disorders, and lack of peripherical sensitivity.\nCT () and MRI with intravenous injection of gadolinium () showed a recurrence of multiple intraspinal extradural cystic lesions, with the spinal cord compression involving the body of D2 and minimally the body of D1. The patient will undergo a second surgical procedure of decompression of the spinal cord, removing the recurred cysts and maintaining the spinal stabilization system. The patient recovered successfully and was able to walk autonomously on the fifth day after surgery. Later the patient began treatment with anthelmintic drug (Albendazole), with stability of the pathology until today. |
A 20-year-old male patient presented with a 6-year history of dental pain and progressive loosening of the posterior teeth on the left mandible. A panoramic radiograph revealed wide-ranging dissolution of the left mandible, while no periosteal reaction or reactive new bone formation were observed around the residual bone tissues (). A CT scan of the craniofacial region corroborated X-ray results, and further revealed that the osteolytic bone ranged from the left ramus to the second premolar, with a continuation of the lower margin of the left mandible only. The teeth appeared to float in the osteolytic tissues (). Three-dimensional reconstruction CT images indicated that the left temporomandibular joint remained uninvolved ().\nAn initial diagnosis of pericoronitis of the wisdom tooth was issued by the patient’s local primary hospital. Following anti-inflammatory therapy, the dental pain was temporarily relieved but the progressive tooth loosening was also aggravated. In 2009, the chewing function in the left mandibular area was completely lost. Dental radiology at that time indicated massive osteolysis of the left mandible, however, the patient refused further examinations and therapy.\nIn May 2012, the patient was referred to our hospital due to an enlarging mass in the left mandible. CT and MRI images showed a 38×43×75-mm, irregular, thick-walled cystic mass in the left ramus of the mandible (). Standard laboratory investigations, including bone metabolism tests, revealed no abnormality. A skeletal survey revealed no other osseous involvement. The loosening posterior teeth (–) were extracted during exploratory surgery and a large quantity of pus was spilled from the incision area. Following repeated rinsing with sterile saline, the wound was sutured. Histopathological examination of the bone biopsy showed proliferation of the fibrous connective tissue, intermixed with irregular bony trabeculae (). Tissues obtained from the cystic wall were also sent for histopathological examination, but only inflammation and granulation tissue formation were revealed.\nOn the basis of the clinical, radiographic and histopathological results, a diagnosis of Gorham-Stout syndrome was made. Radiotherapy and etidronate therapy were proposed to the patient, but were not accepted. At present, the patient remains under observation. The study was approved by the ethics committee of Wuhan General Hospital of Guangzhou Command. Written informed patient consent was obtained from the patient. |
A 45-year-old German Caucasian woman was transferred to our department of general surgery with a suspected esophageal perforation after endoscopic recovery of a pearl onion which was impacted in the middle third of the esophagus. After recovery of the pearl onion, upper gastrointestinal endoscopy revealed slightly bleeding mucosa at the site of impaction as well as an impassable stenosis. Her medical history, included relapsing periods of dysphagia since her childhood that were never examined by endoscopy or gastrografin swallow. At admission, she presented in a stable condition with normal laboratory findings. Clinical investigation revealed no abnormalities other than retrosternal pain as well as emphysema of the skin. Chest and abdominal radiography showed free intra-abdominal air as well as a pneumomediastinum. A gastrografin swallow was performed revealing leakage at the distal esophagus and pneumoperitoneum (Figure ). Explorative laparotomy and direct closure with hemifundoplication were performed and the mediastinum was drained.\nBecause of pathological drainage continuing on the tenth postoperative day as well as increasing white cell counts, we performed a gastrografin swallow that revealed mucosal irregularities and a double-barreled esophagus (Figure ). Upper gastrointestinal endoscopy identified an esophageal intramural dissection with complete obstruction of the true lumen at 35 cm, as well as mucosal bridges (Figure ).\nThese findings left us without any feasible conservative management options. The presence of mediastinitis due to a suspected transmural perforation prompted us to perform a transhiatal esophagectomy with cervical esophagostoma and blind closure of the stomach.\nThe pathology report showed the true esophageal lumen and a transmural perforation as well as a second lumen that was focally covered by a flat squamous epithelium with multiple ulcerations within the submucosal layer (Figure ). The postoperative course was uneventful and 3 months after esophagectomy, a reconstruction with a cervical esophagogastric anastomosis was performed and the patient was discharged on the 12th postoperative day after an uneventful course. |
A 23-year-old Caucasian Greek woman presented with a history of progressively worsening hoarseness during the previous 9 months and recent onset of dysphagia. She reported no weight loss and she denied the use of tobacco or excessive alcohol consumption. She had a history of skin atopy, reporting sensitivity to wool, and a history of an episode of anaphylactic reaction of unknown cause 2 years previously.\nPhysical examination of the neck was normal. Indirect laryngoscopy and endoscopy with a flexible endoscope revealed a smooth, red-yellow, cystic formation localized in the left hemilarynx between the false vocal cord and the aryepiglottic fold. Chest X-ray was normal. A tracheal computed tomography (CT) scan in 5 mm sections confirmed the existence of the mass (Figure ). The thyroid cartilage appeared intact and no nodal involvement was detected.\nWe proceeded to perform microlaryngoscopy under general anaesthesia and removal of the mass (measuring 4 × 2.5 × 2 cm) using a CO2 laser. Pathological examination revealed a mass with a smooth surface. On macroscopic inspection the cut sections were yellow-grey and solid with a soft and elastic consistency. On microscopy, it was not clear whether its structure was bundled or micronodular, consisting of bundles of woven eosinophilic tissue, with sparse fibroblastic cells, several small vessels and rare atrophic glandular regions. There were also a few collections of lymphocytes and granulocytes. Staining with Congo red stain, under polarized light, revealed blue-green birefringence throughout the mass due to the presence of amyloid. No signs of malignancy were seen.\nFurther examinations were done to rule out systemic amyloidosis. The patient's complete blood count, erythrocyte sedimentation rate, basic metabolic and biochemical panel and liver function tests were within normal limits. Serum calcium was also normal. Serum and urine electrophoresis were normal. Rectal biopsy was negative. Based on these findings systemic amyloidosis and multiple myeloma were excluded from the differential diagnosis. The amyloid light chain was lambda (λ) type. The amyloid mass was removed by microlaryngoscopy. |
In August 2012, a previously healthy 28-year-old female patient was hospitalized with a 1-month history of progressing symptoms of raised intracranial pressure and ataxia. The symptoms have been progressing for nearly a month until unassisted walking has become impossible. Neuroradiological examinations were performed upon admission, which revealed a cerebellar tumor positioned deeply and mainly to the left, leading to obstructive hydrocephalus []. Following diagnosis the tumor was resected totally. Postoperative computed tomography displayed resolved obstructive hydrocephalus, which suggested against the implantation of a ventriculoperitoneal shunt. Histopathological analysis revealed the tumor to be desmoplastic medulloblastoma. The patient underwent a postoperative course of total craniospinal axis radiotherapy.\nPosttreatment, the patient was in good condition, without symptoms suggestive of persisting disease or recurrence. The 4th-month follow-up magnetic resonance imaging (MRI) of the head revealed no signs of recurrence.\nHowever, 8 months after the operation the patient began to experience low-back pains with irradiation toward the frontomedial surface of the left lower extremity. MRI of the lumbosacral region did not reveal pathological abnormalities. The symptoms were manageable through conservative treatment and resolved completely. Yet, the stable clinical condition was only temporary and due to reemergence and progression of the symptoms the patient was rehospitalized in June 2013. Head MRI was performed that showed no signs of tumor recurrence. However, the MRI of the lumbosacral and retroperitoneal areas revealed an epidural tumor along L2 to L4 levels, protruding through the two respective left neuroforamens and sheathing the neural radices []; hypo- and hyperintense lesions were found in the lumbar vertebrae, sacrum, and two iliac bones. Additional lesions were found in the region including multiple enlarged retroperitoneal and inguinal lymph nodes predominantly on the left side and nondistinct hypodense zones in the left iliac and medial gluteal muscle []. The patient underwent intervention with the partial extirpation of the tumoral mass in the vertebral canal, the paravertebral, and the gluteal musculature. The histological examination of the formation revealed it as a metastasis from desmoplastic medulloblastoma. Despite certain alleviation of the pain syndrome early after the operation, the intervention was with only a temporary effect and adjuvant radiotherapy of the tumor locus was performed to achieve control of the pain. The patient developed progressive myelodysplastic syndrome, which hindered adequate chemotherapeutic treatment of the metastatic disease. The severity of the patient's condition, unfortunately, lead to fatal end on the 12th month after the initial diagnosis. |
Patient 10 (Tables and ), a 76-year-old female, was referred to the department of neurology in July 2012 because of progressing cognitive decline over the last 12 months, loss of weight, nausea, gait disturbance and tremor. She was seen on May 2011 for the first time by a neurologist with a 3-month history of dull holocephalic headache who ordered a cranial magnetic resonance imaging (MRI) and diagnosed a tension-type headache and a depressive disorder. Treatment with an antidepressant (duloxetine) was started. The patient experienced no improvement and a second examination by another neurologist was undertaken 2 months later. Again no focal neurological signs could be detected. Due to the weight loss, an occult neoplasm was suspected but not detected during an extensive inpatient investigation at a medical department during February 2012; however, the MRI showed bilateral white matter lesions (WML) and an old lacunar lesion located at the left striatum, the latter was not seen in the previous MRI from May 2011. Since the patient also suffered from mild hypertension, vascular encephalopathy was thought to be the cause of the progressive cognitive decline. Extensive neurocognitive testing was carried out in a rehabilitation centre in May 2012 and disclosed a severe decline of attention, memory and executive functions corresponding to subcortical dementia (Fig. ). When the patient was seen for a further diagnostic work-up at the SMZ-Ost-Donauspital in July 2012, the weight was 47 kg and a weight loss of 20 kg was reported over the past year. The gait was insecure with postural instability and with a tendency to fall when turning around. Frontal signs were positive, the voice was quiet, the tonus was mildly elevated and showed a slight hesitancy (“Gegenhalten”), tendon reflexes were brisk, paresis and pyramidal signs missing. There were no signs of ataxia, but a mild bradykinesia. Action tremor was more distinct than a mild resting tremor. Again, neurocognitive testing and gait disturbances were consistent with subcortical dementia (Figs. and ). Regarding the mild signs of parkinsonism, dementia with Lewy bodies (DLB) was also suspected but excluded by a dopamine transporter (DAT) scan. Fluorodeoxyglucose positron emission tomography (FDG-PET) demonstrated hypometabolism in the left striatum and in the left frontotemporal cortex (Fig. ). Cerebrospinal fluid (CSF) showed signs of a chronic lymphocytic inflammation. The CSF markers for dementia, total tau protein and phosphor-tau were within the normal range, while beta-amyloid 1-42 and the Innotest-amyloid-tau index (IATI) were found to be below the reference values (beta-amyloid 1-42: 290 pg/ml, reference value > 500 pg/ml; IATI 0.6, reference values > 1). Finally, LNB was diagnosed when further CSF examinations disclosed a highly elevated Bb-specific-AI indicating local intrathecal Bb-specific antibody synthesis (Table ). The patient was treated with 2 g ceftriaxone daily for 3 weeks.\nNeurological symptoms and impaired cognitive functions, although persistent for a year, recovered rapidly within a few weeks (Figs. , and ) and so did the pathological CSF findings (Table ). A follow-up FDG-PET examination showed the left frontotemporal hypometabolism in remission, while this was not the case for the cystic lacunar lesion in the left striatum. A new and clinically silent small right thalamic lesion was detected that was not present in the pretreatment MRI (Fig. ). The Consortium to Establish a Registry for Alzheimer’s Disease (CERAD) test battery at the last follow-up in April 2014 scored within the age-dependent normal range with the exception of verbal learning and semantic verbal fluency (Fig. ). In a telephone call in February 2018 at the age of 82 years, the patient reported no gait problems or cognitive impairment and had just returned from a trip to Cuba. |
A 29-year-old man, a former semiprofessional handball player, had a traumatic rupture of the proximal side of the patellar tendon of the left knee in 2016 during a match. No associated disease was reported. Primary surgery was performed in another health facility through a median approach using 2 anchors for tendon repair protected by an additional ipsilateral semitendinosus graft (patellar and tibial tunnels). The patient came to our health facility following severe functional deficits after an iterative rupture without having experienced any new trauma 13 months after the initial surgery. The iterative rupture of the knee extensor mechanism was also an iatrogenic fracture of the transverse patellar tunnel (). Clinically, walking was not possible, there was a lack of active extension and hemarthrosis with pain. There were no scar problems, no signs of deep or superficial infection, and no cutaneous wound. A huge gap was clinically observed between the patella and the patellar tendon. Considering the patient's age, his preinjury sports level, and lack of active extension, the decision was made to perform a revision procedure. An artificial ligament (LARS®) and two adjustable loops, free ends of the PULLUP® BTB (SBM SAS, France), were used to enhance the patellar tendon repair.\nA preoperative lateral standard X-ray was taken of the contralateral knee at 30° of flexion to measure the Caton-Deschamps index and patellar height (). The patient was placed in a supine position under general anesthesia with a tourniquet at the proximal part of the thigh. The previous median approach was used. The patellar fracture and the site of the previous rupture were cleaned to remove fibrous tissue and hematoma. Previous anchors were left in place.\nThe first step was to place the ligament advanced reinforcement system (LARS® polyethylene terephthalate fibers 6 mm ref. L030307 ACFAR 32 CK). A new transverse tunnel was drilled in the tibia, distally to the tibial tunnel of the initial surgery, with a 5.5 mm drill, and the LARS® was inserted in the tibial tunnel. The artificial ligament was then passed through the lateral retinaculum and above the patella at the junction with the quadriceps tendon in a Pulvertaft manner and through the medial retinaculum to return to its origin (). Two longitudinal tunnels were drilled in the patella using a 2.4 mm drill. The loops of a PULLUP® BTB (the plate was removed from the device) were first passed into the patellar tendon and then into the patella through the two longitudinal tunnels using a shuttle relay. Next, the free ends were pulled down in the opposite patellar tunnels. At the proximal side of the reconstruction, the two free ends of the PULLUP® BTB were inserted into each braid to close the system (Figures and ). The distal and medial ends of the LARS® were tightened with a clamp in order to restore normal patellar height and secured with 2 staples. Then, the 2 PULLUP® BTB loops were tightened (). The previous tendon rupture was closed and reinforced with separate X-knots using absorbable sutures. The skin was closed. The knee was placed in an articulated brace with compressive ice therapy for 24 hours, and a postoperative X-ray was taken ().\nFor postoperative care, weight bearing was not allowed for 6 weeks and mobilization of the knee was immediately started between 0 and 45° for 3 weeks then from 0 to 90° from 3 to 6 weeks. No complication was reported during the postoperative period. At 3 months, the patient was pain free and could walk without the aid of crutches. He was able to resume handball practice at 6 months after a control MRI. At one-year follow-up, he was able to play handball with complete knee extension strength (compared with the contralateral side) and was able to return to a semiprofessional level. The range of motion of the knee was 0-0-130°. The MRI at 1 year showed complete healing of the patellar tendon and the bone (). |
A 41-year-old male was HBP in the left abdomen during his first at-bat during a baseball game. He was standing close to the plate, and the pitcher who attempted to throw a pitch inside. The ball struck the patient and dropped directly to the ground. Although he felt pain at site of impact, the patient continued to play. However, when swinging at his second at-bat (he hit a ground ball), he experienced abdominal discomfort and subsequently removed himself from the game. After the game while traveling home on a bicycle, he lost consciousness and fell. He was transported to a local hospital where abdominal computed tomography (CT) showed a splenic injury. After transfer to our emergency and critical care center, he was pale but alert and receiving intravenous fluids. Vital signs were stable but systolic blood pressure was relatively low (80 mmHg). The injured area was bruised without underlying rib fracture (Fig. ). CT with contrast showed traumatic splenic injury and intra-abdominal hemorrhage (Fig. ). Emergency laparotomy was then performed. Hemorrhagic ascites was observed during the operation and approximately 2300 mL of blood was aspirated. Abdominal exploration revealed a 4-cm tear in the splenic capsule of the inferior extremity, (medial spleen) and active hemorrhage near the splenic hilum. Further exploration showed vascular injury of the hilum and a deeply extending parenchymal injury. We elected to perform splenectomy because of the severity of injury and hemorrhage. The vessels at the splenic hilum were ligated and hemostasis was achieved, prior to spleen was removal. During the operation, 1120 mL of red blood cells and 1200 mL of fresh-frozen plasma were transfused. Histopathologic examination of the spleen showed hemorrhage in the parenchyma and partial rupture of the capsule. Splenic rupture was diagnosed based on the intraoperative and histologic findings. The patient’s postoperative course was uneventful, and he was discharged home 6 days after surgery. |
A 15-year-old undernourished boy presented to our outpatient department with a 1-month history of progressive breathlessness on exertion. He had a history of intermittent fever of mild to moderate grade and dry cough since 4 months. His breathlessness had progressed relatively rapidly over 1 month and now limited his daily activities. He also had associated anorexia and weight loss. He had been treated symptomatically by his local physician and later underwent investigations for the persistence of symptoms. A chest X-ray [] showed a complete white-out of the left lung with ipsilateral mediastinal shift suggesting complete lung collapse. A contrast-enhanced computed tomography of the thorax showed an endobronchial lesion in the left main bronchus with complete collapse-consolidation of the left lung; it also showed an endobronchial lesion in the right lower lobe []. He underwent a fibreoptic bronchoscopy which confirmed a left main bronchial growth and a right lower lobar bronchial growth and a guided biopsy of these endobronchial lesions [] was done. The histopathology report of the left sided lesion was necrotic tissue and the right sided lesion was reported as a neuroendocrine tumor (carcinoid tumor).\nSubsequent to these investigations, he was transferred to our hospital for increasing the severity of his symptoms. He presented to our hospital in respiratory distress and required endotracheal intubation with mechanical ventilation. We performed a bronchoscopy which confirmed the outside report and repeated endobronchial biopsies. Review of these and the external biopsies showed a high-grade malignant tumor composed of sheets ofhighly pleomorphic cells containing a moderate amount of eosinophilic cytoplasm and eccentrically placed hyperchromatic nuclei and prominent nucleoli. Few cells showed wreath like/horseshoe-shaped nuclei []. On immunohistochemistry (IHC) the tumor cells were strongly and diffusely positive for leukocyte common antigen, while negative for cytokeratin [], desmin and CD3 and CD20. On additional IHC the tumor revealed strong positivity for CD30[] and anaplastic lymphoma kinase-1 (ALK-1)[]. Mib-1 (Ki-67)labelling index was approx. 40-50%. Systemic examination and other investigation did not revealed any evidence of the disease elsewhere. Hence the diagnosis of primary endobronchial Alk-1positive anaplastic large cell lymphoma null cell type was rendered. His cerebrospinal fluid (CSF) cytology was also positive for NHL. We started him on CHVbP protocol chemotherapy, and he showed dramatic improvement in his ventilatory parameters and was extubated in 3 days after initiating chemotherapy. He also received G-CSF prophylactically to prevent neutropenia which is not uncommon and sometimes fatal in these settings. His chest X-ray which had shown complete collapse before treatment also showed dramatic improvement []. |
A 64-year-old man without known significant medical history was referred to our hospital for the treatment of an incidentally found left JFS. He was found to be completely intact on examination and no family history of neurofibromatosis type 2. MRI revealed a dumbbell-shaped solid mass at the left cerebellopontine angle with marked enlargement of the affected jugular foramen and no sign of dural tail, which was 25 × 27 × 18 mm and 22 × 25 × 19 mm for the intradural and extradural portions, respectively, and 8.4 ml in total volume. The mass mildly compressed brainstem and had no evident sign of hemorrhage [ and \n]. There was no other intracranial tumor. MR venography demonstrated that the left jugular bulb was obliterated because of tumor compression []. The tumor showed no signs of calcification or hypervascularity, and positron emission tomography with fluorodeoxyglucose did not exhibit increased uptake consistent with malignancy [] or any evidence of other tumors throughout the body. T1-weighted MRI at the level of the internal auditory canal indicated minimal brainstem compression before treatment []. Based on observations from the aforementioned examinations, two independent radiologists made a diagnosis of sporadic JFS. After thorough discussion, SRS using Gamma Knife (Elekta AB, Stockholm, Sweden) was performed with a marginal and maximal dose of 13 and 26 Gy, respectively.\nThree days after SRS, the patient developed dysphagia, hoarseness, and leftward deviation of the tongue that were confirmed by otorhinolaryngologic examinations, suggesting injuries of the left vagal and hypoglossal nerves. MRI on post-SRS day 5 showed tumor expansion without peritumoral edema []. Administration of dexamethasone gradually relieved the symptoms except for vocal cord palsy that later required additional laryngological interventions.\nAlthough his symptoms improved, follow-up MRI at 5 months from SRS showed further tumor expansion with central necrosis and peritumoral edema, albeit asymptomatic []. This was considered as a slightly more aggressive change than transient tumor expansion typically seen in vestibular schwannomas (VSs), and thus prompted further close follow-ups.\nAt 7 months, he suddenly developed headache, dizziness, mild hearing deterioration (20 dB decrease in pure tone audiometry), nausea, and vomiting. Imaging studies revealed intratumoral hemorrhage with exacerbation of peritumoral edema []. At that time, he was not on any blood thinners, and coagulation tests demonstrated normal function. Since he showed no further progression after restarting the administration of dexamethasone and osmotic diuretics, we continued the conservative management without surgical intervention. He finally recovered almost completely within 2 months after the hemorrhage, and follow-up images at 4 years from SRS demonstrated significant tumor shrinkage []. |
A 30-year-old female with a medical history of hypertension and smoking sustained a left mandibular angle fracture after a fall from a ladder. The patient underwent open reduction and internal fixation of her left mandibular angle through an intraoral approach without complications. The patient developed an infection at the site of the hardware and nonunion of the fracture site as a result of noncompliance—lack of smoking cessation and poor oral hygiene—with postoperative instructions. For treatment of the nonunion and infection, the patient returned to the operating room (OR) approximately 1 month after the initial surgery for debridement and removal and replacement of the hardware. Two months from the time of injury, the patient sustained a fist-to-face assault and developed an abscess at the site of the hardware. Incision and drainage of the abscess were performed at an outside hospital. However, the patient developed an extraoral fistula at the site of the infected hardware. She was scheduled for an elective fistulectomy, debridement of the previous fracture site, and removal and replacement of hardware 4 months from the time of her initial injury.\nIn the OR, all standard American Society of Anesthesiologists monitors were placed. The patient underwent preoxygenation for 8 minutes prior to intubation, and induction of general anesthesia was facilitated with 80 mg lidocaine, 160 mg propofol, 150 mcg fentanyl, 120 mg succinylcholine, and 10 mg rocuronium. Nasoendotracheal intubation was performed with a 7.5 mm nasal right angle endotracheal (RAE) tube, and a MacIntosh 3.5 laryngoscope was placed with direct laryngoscopy; however, 2 minutes after intubation during confirmation of tube placement, the patient was found to have a leak related to cuff rupture. An Eschmann-guided exchange was performed with a 7.0 mm nasal RAE tube and a McGrath video laryngoscope because a Cook airway exchange catheter was not available. Placement was confirmed with bilateral breath sounds, symmetric chest rise, and capnography; however, the patient had diminished breath sounds throughout auscultation with expiratory wheezes bilaterally. Approximately 10 minutes following confirmation of endotracheal tube placement, the patient's peak inspiratory pressures increased from 23 cm H20 to 64 cm H20, resulting in difficult ventilation. Numerous measures were taken, including suctioning the nasal RAE tube and administering 0.5 mg terbutaline subcutaneously and 0.5 mL of 2.25% nebulized racemic epinephrine; however, no improvement in ventilation and peak pressures was noted. Six minutes after the elevation in peak pressures, the 7.0 mm nasal RAE tube was removed, and bag-mask ventilation was initiated. Peak pressures elevation and ongoing difficulty with ventilation continued. The patient's oxygen saturation declined from 98% to 54%, and her heart rate decreased from 104 to 64 bpm, but no significant decline in blood pressure was noted. The patient was reintubated using a McGrath video laryngoscope and a 7.0 mm oral endotracheal tube, and ventilation was initiated via Ambu bag and supplemental oxygen. The patient's oxygen saturation increased to 96% and her heart rate to 92 bpm.\nShe began to develop significant left-sided facial and bilateral neck and chest edema with crepitus, indicating subcutaneous air. Decreased right breath sounds were noted with auscultation. Intraoperative chest x-ray revealed significant bilateral subcutaneous air, pneumomediastinum, and a large right-sided tension pneumothorax (). An attempt at needle decompression failed because of large chest wall thickness. General surgery emergently placed a 32-French chest tube on the right side without complication, resulting in an almost immediate improvement in ventilation and tidal volumes and resolution of the tension pneumothorax. The fistulectomy, debridement, and removal of hardware procedures were aborted.\nThe patient was transferred to the trauma intensive care unit (TICU) where she remained intubated and sedated. Bronchoscopy performed intraoperatively in the TICU following chest tube placement revealed no obvious injuries. Postoperative computed tomography (CT) maxillofacial scan without contrast revealed extensive air within the right parotid, masticator, retropharyngeal, and bilateral carotid spaces, along with right preseptal periorbital, bilateral neck, and anterior chest wall soft tissues. CT of the neck without contrast demonstrated a large amount of soft tissue emphysema within the anterior and posterior neck, superior mediastinum, and chest wall compartments, and CT of the chest without contrast demonstrated persistent right-sided pneumothorax ( and ).\nThe following day, flexible fiberoptic laryngoscopy performed by the TICU service revealed a tissue flap in the posterior hypopharynx with overlying blood clot. After removal of the clot, a tissue defect consistent with a right pyriform sinus tract laceration injury was identified (). Otolaryngology confirmed the pyriform sinus injury. Enteral feeding through an orogastric tube was initiated, and intravenous (IV) ampicillin-sulbactam was administered for aerodigestive organism coverage.\nThe patient remained intubated and sedated until day 5 when she was extubated over an Eschmann catheter by the anesthesia service. The following day, the patient's chest tube was removed, and she was transferred to the floor for monitoring of her hypopharyngeal injury. She remained an inpatient for 2 days and then requested discharge from the hospital. The patient was advised to remain hospitalized until the otolaryngology service could perform a barium esophagram with laryngoscopy to reevaluate the pyriform sinus laceration, but the patient left the hospital against medical advice on postoperative day 8 after the risks had been discussed with her. The patient completed 8 of the recommended 10 days of IV ampicillin-sulbactam and 8 days of enteral feeding.\nThe patient was successfully contacted 1 week after leaving the hospital and agreed to return for follow-up evaluation and another attempt to perform the procedure. One month later, the patient underwent fistulectomy, debridement of the previous fracture site, and removal and replacement of hardware after nasoendotracheal intubation without complication using a C-MAC video laryngoscope. Preoperative evaluation with awake nasolaryngoscopy performed by the otolaryngology service revealed a well-healed pyriform sinus. Surgery was completed without incident, and the patient was extubated postoperatively without incident.\nThe patient was seen postoperatively 3 times in 1 month for outpatient follow-up. She continued to smoke cigarettes daily and did not comply with the recommended no-chew diet. At her final visit 1 month postoperatively, she had developed a superficial intraoral surgical site dehiscence. She did not return for additional follow-up visits. |
An 85-year-old woman presented to the emergency department with complaints of acute-onset abdominal pain. She reported a 1-day history of epigastric pain that radiated to the right upper quadrant and right flank. It was associated with anorexia, nausea, and emesis. She denied fever, chills, change in bowels, or any other symptoms. She was afebrile with mild tachycardia at initial presentation. Physical examination was notable for a soft, nondistended abdomen with tenderness to palpation in the epigastrium and right upper quadrant. She was found to have a positive Murphy sign. Her past medical history was significant for chronic lymphocytic leukemia for which she was currently treated with ibrutinib. Laboratory assay revealed a white blood cell count of 168.7, which was within her baseline levels. Her liver function tests were normal, with a total bilirubin of 0.5, alkaline phosphatase of 98, and normal transaminases. A computed tomography (CT) scan of the abdomen revealed a dilated gallbladder with a thickened wall and pericholecystic edema concerning for acalculous cholecystitis []. No obvious gallstones were noted. An ultrasound of the right upper quadrant was obtained in order to attempt to better visualize gallstones. This ultrasound did not visualize gallstones or sludge, but did confirm the presence of a distended gallbladder with a thickened wall. She was admitted to the general surgery service, and intravenous ciprofloxacin and metronidazole were administered []. Due to the persistence of symptoms, she was taken to the operating room for laparoscopic cholecystectomy with a presumed diagnosis of acute cholecystitis. After the induction of general anesthesia, she had a palpable mass in the right upper quadrant. Upon insertion of the laparoscope into the abdomen, it was noted that the gallbladder was completely gangrenous. There was only one attachment from the underside of the liver to the gallbladder, which measured approximately 1 cm. Distal to this attachment, the gallbladder was completely torsed. After detorsion, the infundibulum was also noted to be gangrenous, but the proximal cystic duct was not, and laparoscopic cholecystectomy was performed. Given the very short attachment of the gallbladder to the liver fossa, great care was taken to completely define the anatomy prior to resection. The total operative time was 135 min with minimal blood loss. Histopathologic findings of the gallbladder specimen revealed acute cholecystitis with involvement by chronic lymphocytic leukemia. Postoperatively, her course was complicated by abdominal distention, right shoulder pain, and hyperbilirubinemia. On postoperative day #3, a repeat ultrasound of the right upper quadrant was performed, which revealed a small amount of perihepatic fluid concerning for bile leak. There was no choledocholithiasis or intrahepatic biliary dilatation. A hepatobiliary iminodiacetic acid (HIDA) scan confirmed leakage of the bile posteriorly and at the dome of the liver. The patient then underwent endoscopic retrograde cholangiopancreatography that showed normal biliary anatomy with peripheral bile leakage therefore a common bile duct stent was placed. She had a CT scan of the abdomen on postoperative day #4 for persistent symptoms, revealing persistent biloma measuring 11 cm at the greatest diameter. She returned to the operating room for laparoscopic washout and drain placement. The patient was discharged to home on postoperative day #9/#5. One week after the discharge, the patient had her drain removed in the outpatient clinic and after further recovery, her biliary stent was removed endoscopically. |
This paper presents the case of a 50-year-old male German academic who developed neurocognitive deficits, affective symptoms, and predominant delusions for over one and a half years. The patient, as well as his family, have given their signed written informed consent for this case report, including the presented images, to be published. The patient reported a pronounced feeling of thirst, although he was drinking 10–20 liters of water a day, and he was absolutely convinced that he would die of thirst. He did not hear an imperative voice encouraging him to drink more. He said that he needed to drink less water and wondered how his body could retain all the liquid. He was delusionally convinced that he had lost the ability to urinate. In contrast, sonography repeatedly detected no residual urine. Moreover, he suffered from severe insomnia (the patient had the feeling of being completely unable to sleep), reduced cognitive performance—for example, being unable to remember dates—and reduced energy levels, as well as complete loss of interests. In the external assessment, disinhibition, flattened affect, accelerated speech and slow thinking were striking. He had suffered from recurrent depressive episodes since he was 34 years old. For the past eight years, depression had been more severe. In the period between 45 years and 48 years, he had taken high doses of Z-drugs in order to treat insomnia. At the same time, alcohol abuse had developed until two years ago (up to the age of 48 years). At the age of 48, he had suffered a single epileptic status, which was interpreted as the result of alcohol and Z-drug withdrawal in an external hospital. At that time, epileptic activity was described in EEG. Over the last two years he had not consumed any substances (). In addition, hyponatremia (i.e., 130 mmol/L; reference range: 136–145 mmol/L) had been revealed half a year prior to diagnosis. On admission to our hospital, a normal sodium concentration was measured. Additional repeated laboratory testing showed a urine osmolality of 73 and 45 mosm/kg, respectively, and a suppressed urine sodium concentration of less than 20 mmol/L. During a thirst trial, urine osmolality increased to 651 mosm/kg, which indicates psychogenic polydipsia and excludes a syndrome of inappropriate antidiuretic hormone (SIADH).\nDiagnostic findings: The diagnostic examinations were conducted approximately one and a half year after symptom exacerbation after admission to our special ward. In serum, anti-LGI1 antibodies were repeatedly positive in different laboratories (see ), whereas CSF antibody testing was negative. A serum titer of 1:80 (reference <1:20) was measured using cell-based assays. CSF analyses showed normal to slightly elevated white blood cell (WBC) counts (maximum 5/µL; reference <5/µL) and evidence of a blood–brain barrier dysfunction (protein concentration up to 557 mg/L; reference: <450 mg/L; albumin quotients up to 9.5, reference: <8). Oligoclonal bands were always negative. Fluid-attenuated inversion recovery (FLAIR) MRI sequences depicted hyperintensities right-mesio-temporally and on the right side of the amygdala. In the left thalamus, a small, possibly microangiopathic, lesion was detected. In addition, there was a slight grey–white matter blurring (cf. [] ). The routine EEG was normal in the visual assessment. Independent components analyses or rather automatic detection of intermittent EEG slowing remained inconspicuous (cf. []). A cerebral FDG-PET examination showed moderate hypometabolism of the bilateral mesial to medial frontal cortices, which was interpreted as being possibly due to an early manifestation of frontotemporal dementia [], whereas a sequela of substance and/or alcohol abuse was rated to be less likely. It should be noted that there was no mesial temporal or striatal hypermetabolism (), which can be present in active limbic or anti-LGI1 encephalitis. A whole-body FDG-PET/computer tomography scan detected no metabolic or structural pathologies suggestive of malignancy or inflammation. Neuropsychological testing using the Consortium to Establish a Registry for Alzheimer’s Disease (CERAD) test battery showed deficits in Mini Mental State Examination (25 of maximum 30 points), word-list savings, and trail-making tests B and A/B. The test for attentional performance by TAP showed deficits in working memory (missings, false alarms), set shifting (overall index) and alertness (reaction times with and without sound). All diagnostic findings are summarized in .\nIllness, somatic, and family histories: The patient’s past medical history was inconspicuous in terms of in-utero and birth complications. He had not suffered from febrile convulsions, inflammatory brain diseases, relevant systemic infections, or craniocerebral traumata during his childhood or adolescence. There was no evidence of neurodevelopmental or personality disorder. Seven years earlier, at the age of 43, he had developed a deep vein thrombosis by unclear coagulopathy. For several years, he had suffered from leg ulcer on the left inner ankle. He had also suffered from arterial hypertension, treated with ramipril 10 mg/day. His history of autoimmune diseases, infections, or cancer was negative. In the family history of neuropsychiatric diseases, only his father had suffered from depression.\nTreatment and outcome: Classic psychiatric treatment with sertraline, venlafaxine, mirtazapine, reboxetine, several tricyclic drugs, lithium, zopiclone, olanzapine, risperidone, haloperidol, quetiapine, and clozapine previously administered had no relevant positive effect on symptom relief. Under clozapine, he had developed myocarditis. After immunological findings gave evidence for chronified limbic autoimmune encephalitis, glucocorticoid pulse therapy with 500 mg of intravenous methylprednisolone daily for five days and subsequent oral tapering over two months (starting with 50 mg) led to slight improvement of mood and neurocognitive symptoms including temporal orientation, executive functions, word list savings and alertness times (with no difference between the conditions with and without sound). However, new deficits were found in fluency and word list learning with more word intrusions, while deficits in working memory and set shifting were persisting. A second steroid pulse 3 months after the first pulse with oral tapering did not lead to relevant improvement. However, the signal hyperintensities in the MRI showed a tendency to reduction. Further treatment (e.g., with plasmapheresis and/or rituximab) was refused by the patient and his legally authorized parents. Furthermore, l-thyroxine (due to hypothyroidism), vitamin B1 (prophylactic after earlier alcohol abuse), and vitamin D (due to serologically proven deficiency) were supplemented. |
A 67-year-old man presented after being struck by a vehicle while riding his motorcycle with complaints of pain in his neck, pelvis, and left upper extremity. There was no loss of consciousness. Urinary function could not be assessed at initial presentation due to the presence of a Foley catheter, but there was no blood at the urethral meatus. Medical history was significant only for hypertension. On examination, he was conscious, oriented, and hypotensive (103/55 mm Hg). There was hypogastric tenderness and exposed bone through an open wound near his left elbow. Neurovascular status was preserved in both the upper and lower extremities. There was no damage to the spinal cord elements. Anteroposterior pelvic radiograph revealed pubic diastasis of 4.7 cm and a midline fracture through the sacral body with 1.0 cm of displacement (). A pelvic binder was placed to decrease intrapelvic hemorrhage and repeat plain films demonstrated acceptable closure of his pelvic diastasis (). After binder application and the placement of two large-bore intravenous cannulas administering crystalloid isotonic solution, hemodynamic stability was achieved and the patient was deemed stable a contrast-enhanced computed tomography CT scan revealed hemoperitoneum, 1.3 cm of pubic diastasis, and a complete, midline sacral fracture through the spinal canal and posterior sacral elements extending from S1 to the coccyx (). To rule out urologic injury, a CT cystogram was obtained, which revealed no evidence of bladder or urethral rupture. An urgent irrigation and debridement with surgical fixation of his left open ulnar shaft fracture were performed. Great care was taken to monitor the patient’s skin condition beneath the binder to assure he did not experience any areas of skin necrosis or compromise. 2 days after hospital admission, he underwent open reduction internal fixation of the pubic diastasis using a pelvic reduction clamp and a 6-hole non-locking symphyseal plate by Pfannenstiel approach (). During reduction and fixation of the anterior pelvis, intraoperative fluoroscopic assessment revealed increased widening of the midline longitudinal sacral fracture. Due to the fracture’s involvement of the posterior sacral elements and subsequent axial plane instability, fixation of the sacral fracture was deemed necessary to prevent further displacement and potential symptomatic deformity. Under fluoroscopic guidance, transsacral-transiliac guide pins were then used to insert 7.3mm fully threaded screws into the S1 and S2 body, measuring 150 mm and 140 mm, respectively. The screws were inserted posterior to the iliaccortical density so as not to injure the L5 nerve root. Using lag-screw fixation technique principles, these screws closed down the midline sacral fracture diastasis and anatomic reduction was achieved. Postoperatively, the patient was mobilized to weight bear for transfers. At 10weeks, he began weight-bearing as tolerated. The patient developed post-operative urinary retention and a Foleycatheter remained in place on hospital discharge. The urology team followed him as an outpatient and the catheter was removed at 1month, at which time he was able to pass urine normally. The patient received routine follow-up evaluations. Sacral fracture union was evident at 5 months with only mild loss of reduction at the pubic symphysis (1.3 cm of diastasis) (). He had persistent impotence with otherwise normal urologic function and no neurologic deficits. |
A 60-year-old male presented to the emergency room with a complaint of weakness and numbness of all extremities from last three days. One week prior to the presentation, the patient visited his primary care physician with a complaint of neck pain and was sent home on acetaminophen with a possible diagnosis of musculoskeletal pain. His symptoms worsened and rapidly progressed to new onset weakness of all extremities and numbness all over below the level of neck, associated with bowel and bladder incontinence. The patient has a past medical history of diabetes mellitus, thalamic stroke three years ago, and a prosthetic left testis because of undescended left testis. Previously, the patient had excision of the skin and a sinus tract of the left scrotal wall; however, there is no evidence of active infection.\nAt the time of presentation, vital signs were noted to be BP of 140/85 mm of Hg, pulse rate of 85 beats per minute, respiratory rate of 18, temperature of 99.7°F, and pulse oximetry of 97% on room air. On examination, appearance was nontoxic, endorsing a low-grade fever. Cardiopulmonary examination was benign. Cranial nerve examination was unremarkable, and neck movement was unrestricted. Upper extremities had a strength of 3/5 in the right and 2/5 in the left, and lower extremities had a strength of 2/5 in the left and 3/5 in the right. Sensation to fine touch and pin prick was grossly reduced below neck; however, pain, pressure, and vibration sense were intact. Reflexes were intact; Babinski's sign was equivocal. Other significant finding in physical examination was pressure ulcer over left scrotal skin from prostheses but no pus collection or drainage.\nInvestigations done at the time of admission revealed high inflammatory activity with a CRP level as high as 133.3 mg/L and leucocytosis with a cell count of 17,000 cells/mL. Blood cultures were drawn, and empiric broad-spectrum antibiotics were initiated. For a new onset quadriparesis with neck pain, an MRI of whole spine with and without contrast () was performed emergently which revealed a collection that was located along the posterior aspect of vertebral bodies C4–C6, suggestive of epidural abscess with severe cord compression and focal T2 hyperintensity at the C3-C4 level, concerning cord edema. There were no evident cervical infiltrations.\nMultidisciplinary care was initiated for this patient. Neurosurgery was contacted for emergent spinal decompression. The pus was drained, and C3-C4 fusion with plating was done. Our patient was started empirically on cefepime and vancomycin which were tailored as per culture and sensitivity findings; the antibiotics were continued for a total duration of 42 days. Both blood and pus culture from the spine grew Streptococcus intermedius, susceptible to ceftriaxone. Testicular prosthesis gross examination was normal. The culture obtained from the pressure ulcer site over scrotum grew mixed flora including Streptococcus intermedius. Testicular prosthesis was removed with excision of affected scrotal wall. Our patient had a remarkable recovery, and one-year follow-up on the patient shows no residual neurologic symptom. |
The proband, a 12 and a half years old female, is the only daughter of healthy and non-consanguineous Caucasian parents (the mother is 36 years old, the father 40 at conception). She was delivered by cesarean section for maternal reasons at 39 weeks of a spontaneous and unremarkable pregnancy. Amniocentesis was performed due to advanced maternal age resulting in a normal female karyotype (46,XX). The birth weight was 3000 g (25th percentile), length 50 cm (50th percentile), head circumference 35 cm (50th percentile), Apgar score was 8 and 10 at 1 and 5 min respectively. After birth she was referred as normal: she was breastfed for a few weeks, then artificially, weaned at 6 months. She walked independently at one year, spoke the first words at 18 months. At the admission to the kindergarten, at the age of 3, she was referred to the Child Neuropsychiatry Unit on account of language impairment. Intensive speech therapy was supplied for 2 years, then continued with less frequent sessions during the primary school with slowly but steadily positive results. During the second year of the primary school, at the age of 7, learning disability and behavioral disorders became evident. Cognitive assessment performed by Leiter R scale (specific for nonverbal intelligence) and by WPPSI-III, gave an IQ of 91 and 77, respectively (with VIQ = 68, PIQ =89 at the latter scale). A diagnosis of a neurodevelopmental disorder compatible with Gilles de la Tourette syndrome was made at age of 11 for the presence of motor and vocal tics, hyperactivity, motor and behavioral mannerisms, together with mild intellectual disability. During instrumental work up audiological examination, EEG and brain MRI were performed with normal results. At the last follow-up, when she was 12 years old, physical examination showed a height of 153 cm (35th percentile), weight of 61 kg (91th percentile). She had her first menstruation at the age of 11 years without dysmenorrhea or other related problems. Motor embarrassment and bilateral flat foot, corrected with orthotics, were persistent. The last cognitive assessment performed in 2015 by Leiter R gave an IQ score of 69, with WISC IV scale the results indicated total IQ of 57 (VCI = 60, PRI = 67, WMI = 61, PSI = 85) while the GAI was 59. The proband now attends the second class of the secondary school with a learning support teacher. Pharmacological treatment with Aripiprazole 8 mg/day is ongoing. |
A 25 year-old male patient presented with a chief complaint of generalized hair loss in the scalp parietaloccipital region for a duration of 1 year, saying that hair loss started gradually and had worsened over time. In addition, this patient indicated that the alopecia area was occasionally accompanied by mild itching. He had scalp skin folds resembling the ridge and furrow of the cerebral cortex in the parietaloccipital region since birth. These were a small protuberant mass at first and had gradually increased in size and volume in proportion to his growth. He was of normal intelligence and denies neuropsychiatric diseases, eye diseases, scalp injuries, endocrine diseases, tumors, chronic dermatitis and similar family history. Physical examination at our department revealed hypertrophy and formation of folds in the parietal-occipital area, forming 5 to 6 furrows and ridges (Fig. ). The size of the cerebriform mass was about 12.0 × 8.5 cm, without other skin lesions. Diffuse non-scarring hair loss was distributed on the posterior-parietal scalp, mid-parietal scalp and superior-occipital scalp. Further examination did not reveal any changes in neurology or ophthalmology, swelling of the maxillofacial or limb joints, skin thickening and tissue growth. A plain head computed tomography (CT) scan revealed a thickened dermis and excessive growth of the scalp, forming the characteristic scalp folds, with no associated involvement of the bone or intracranial tissue (Fig. ). Due to the lack of neurological or psychiatric findings, this patient was not referred for brain magnetic resonance imaging (MRI). All laboratory tests (routine blood count, glycemia, lipid, basic metabolic profile, liver functions, kidney functions, immune function, insulin-like growth factor 1, growth hormone, parathyroid hormone, cortisol and acromegaly screening) were within normal range. The patient did not accept our suggestion about performing a scalp skin biopsy firstly. In contrast, due to the strong desire to improve the appearance, he and his families chose surgical excision diretly. After the hospitalization procedures, completing the relevant examination and eliminating the contraindications of the operation, the diseased tissue of the patient's parietaloccipital area was excised under general anesthesia. During the operation, a π-shaped incision was designed on the surface of the diseased tissue (Fig. a). The skin, subcutaneous tissue, subcutaneous superficial fascia layer and cap aponeurosis layer were cut off successively along the designed incision line with a blade. The diseased tissue was peeled off with an electric knife in the cap aponeurosis layer. Then the subcutaneous tissue on both sides of the incision was separated to form two fascia tissue flaps. After using an electric knife to fully stop the bleeding and indwelling a negative pressure drainage tube, we pulled the bilateral fascia tissue flaps to the middle to suture the incision interruptly with 4-0 absorbable thread and performed an interrupted suture of the skin with 5-0 silk thread. After the operation, the incision healed well, leaving only a π-shaped scar with a total length of about 18 cm (Fig. b, c). Pathological examination of the tissue excised during the operation showed that there were dense intradermal melanocytic nevus (Fig. ). In light of these findings, the patient was diagnosed with secondary CVG caused by CIN. At the 2 year follow-up, there were no obvious changes in the lesions. |
The patient was a 45-year-old female who was admitted to the Department of Gastroenterology on March 2, 2019 for “recurrent hematochezia for half a year after radiotherapy”. In 2017, due to cervical cancer, the patient underwent 28 radiotherapy treatments and 8 rounds of chemotherapy at a local hospital. During radiotherapy, diarrhea consisting of a black pasty stool occurred 7 to 8 times per day. After radiotherapy, the diarrhea resolved. Half a year ago (September 2018), the patient began to repeatedly experience mucus-containing bloody stool alternately with black pasty stool. The blood in the bloody stool was a bright red color accompanied by dark red clots, and mucus was present. The volume was approximately 30 to 50 ml occurring 5 to 6 times per day and approximately 2 to 3 days per week. The black pasty stool occurred 1 to 2 times per day with a volume of approximately 100 to 250 g each time and occurred approximately 2 to 3 days per week. The diarrhea was occasionally accompanied by abdominal pain and tenesmus, and there was no fever. Dizziness, fatigue, and polypnea gradually appeared after daily activities. The patient was admitted to a local hospital 3 times for severe anemia. The routine blood examination showed a minimum hemoglobin concentration of 30 g/L, and after transfusion, dizziness, polypnea, and fatigue improved. However, the above symptoms continued to repeatedly occur, and the patient sought further treatment at our hospital.\nThe patients temperature was 36.4°C, heart rate was 100 bpm, respiratory rate was 20 breaths per minute, blood pressure was 114/76 mm Hg and body mass index was 20.8 kg/m2. The patient presented an anemic face and a soft abdomen, and no mass was palpated. There was tenderness in the lower abdomen and no rebound pain. Bowel sounds occurred 5 times/minute.\nA routine stool and faecal occult blood test showed a positive faecal occult blood test and a positive faecal fat test. The faecal pathogenic bacterial cultures showed fungal growth on March 2. However, there was no fungal growth on March 12, and we hypothesized that contamination was detected on March 2. The Epstein-Barr virus (EBV) antibody and DNA quantity, cytomegalovirus (CMV) antibody, Clostridium. difficile antigen, tuberculosis antibody, T cell spot test for tuberculosis infection, and coagulation function detection showed no abnormalities. The routine blood examinations showed a hemoglobin concentration of 53 g/L, indicating microcytic hypochromic anemia.\nThe magnetic resonance imaging examination of the pelvic cavity with a plain scan and enhancement showed changes after radiotherapy and chemotherapy for cervical cancer, and no obvious tumor recurrence or metastasis was found in the pelvic cavity. The walls of the sigmoid colon and rectum were slightly thickened and swollen, which was considered to have occurred after radiotherapy (Fig. ). Colonoscopy showed obvious congestion of the sigmoid colon and rectal mucosa and a smooth surface, and bleeding was easily induced by touch. The sigmoid colon was narrow, rendering the passage of the colonoscope difficult (Fig. a-2b). The colonoscopy diagnosis was radiation proctitis. The pathological findings revealed chronic mucosal inflammation with mild atypical hyperplasia of the glands (Fig. ).\nBased on the above examination results and after the exclusion of diseases, such as tumor recurrence, invasion of the rectum, fungal infection, Clostridium difficile infection, EBV or CMV infection, and ulcerative colitis, a diagnosis of “chronic hemorrhagic radiation proctitis” was rendered. Considering that many studies have reported that intestinal bacteria are involved in the incidence of radiation enteritis and that both animal model experiments and case reports have shown that FMT can improve the symptoms of radiation enteritis and mucosal inflammation,[ the patient and her family agreed to FMT treatment after being fully informed.\nFaecal bacteria were obtained from 8 healthy people aged 21 to 24 years without digestive system diseases, tumors, infectious diseases, metabolic diseases, genetic diseases, or other related diseases after auxiliary examination. Additionally, the subjects did not take antibiotics, drugs affecting gastrointestinal dynamics or other drugs causing intestinal microecological disorders in the prior 3 months. Fresh faecal liquid was separated by an automatic purification system (GenFMTer; FMT Medical, Nanjing, China). Two FMT treatment approaches[ were utilized as follows: the lower-gut approach was adopted in the first course of treatment, and the mid-gut approach was utilized in the second to fourth courses of treatment because it was difficult to pass the endoscope through the narrow intestinal cavity. The faecal liquid was moved from the donor to the patient within 1 hour to keep it as fresh as possible to achieve maximum clinical efficacy. The regimen of 1 course of treatment was 200 ml/treatment/donor once a day for 3 days.\nTreatment courses: The patient received a total of 4 individual courses of FMT, which were completed in March, April, May, and July.\nThis study was approved and monitored by the Ethics Committee of the First Affiliated Hospital of Guangdong Pharmaceutical University (Approval No. Yilun Shen [2019] No. 188).\nAt the six-month follow-up after all 4 courses of FMT were completed, hematochezia, abdominal pain and diarrhea were resolved, and the patient did not experience any side effects from the FMT treatment. Compared to her symptoms before the treatment, her stool frequency decreased from a maximum of 6 times to 2 times per day, and the mucus-containing bloody stool and pasty stool became soft yellow stool. Additionally, the European Organization for Research and Treatment of Cancer Therapy Oncology Group (RTOG/EORTC) score[ improved from level 2 to level 1. The patient was elated because she did not need a blood transfusion, proving that no blood was lost, and the hemoglobin concentration remained above 60 g/L (Table ). The sigmoid colon and rectal mucosal inflammation showed improvement via colonoscopy (Fig. c-2f).\nThe faecal samples collected from the patient before the treatment (1), 28 days after the first course of the FMT treatment (2), 30 days after the second course of the FMT treatment (3), and 70 days after the third course of treatment (4) and the donors (mix of the first course faecal liquid from 3 donors, GZ) were subjected to 16S rRNA sequencing. On average, 340.06 operational taxonomic units (OTUs) were identified with a minimum of 231 OTUs and a maximum of 500 OTUs. According to the analysis of the alpha diversity index, the species richness, evenness and inter-species differences significantly increased concurrently with the extension of time and increases in the transplantation time (Table ). At the phylum level (Fig. a), the predominant bacteria were firmicutes before the transplantation (1) and bacteroidetes after the transplantation. However, at the genus level (Fig. b), the primary bacteria were bacteroidetes before the transplantation (1) and Prevotella after the transplantation. As the FMT course progressed, the composition of the microbiota of the patient tended to become similar to that of the donors. |
A 32-year-old Caucasian gravida 1 para 0 (G1P0) woman without any significant past medical or surgical history was followed by our department throughout the duration of her pregnancy, which was facilitated by in vitro fertilization. Prior to pregnancy, the patient had regular menstrual periods and cycles lasting 30-32 days on average with a variability of +/- 5 days. Papanicolaou (Pap) smear conducted 5 years prior to pregnancy revealed a low-grade squamous intraepithelial lesion that was negative for human papillomavirus (HPV). A subsequent Pap smear conducted 2 years prior to pregnancy was negative for abnormality. She denied a history of pelvic inflammatory disease (PID), sexually transmitted infections (STIs), tubo-ovarian abscess, colitis, appendicitis, and endometriosis. Despite not having any chronic medical conditions, being a non-smoker, and having a healthy, active lifestyle, the patient had a history of unexplained infertility for which she underwent an extensive medical workup and multiple treatment modalities.\nThe patient previously used an intrauterine device for contraception, which was removed approximately 4 years prior to this pregnancy. Approximately 2 years prior to the start of pregnancy at her routine gynecologic exam (during which she had her second Pap smear, as noted previously), the patient stated that she was trying to become pregnant. Two weeks prior to this exam, her male partner had an overall normal semen analysis. The patient at this time was provided with prenatal guidance regarding timed intercourse and anticipatory guidance. She was also instructed to take daily prenatal vitamins and docosahexaenoic acid (DHA). Roughly 4 months after this appointment (at this point 8-9 months of actively trying to become pregnant) the patient sought out additional procreative guidance after unsuccessfully conceiving. The patient was advised to use an ovulation indicator, continue timed intercourse, and maintain healthy lifestyle habits. Laboratory testing was also conducted, and the following values were reported at this time: dehydroepiandrosterone (DHEA) 7.35 ng/mL, estradiol 48 pg/mL, progesterone 0.26 ng/mL, testosterone 21 ng/dL, sex hormone-binding globulin 73 nmol/L, free testosterone 2.1 pg/mL, follicle-stimulating hormone (FSH) 6.1 mIU/mL, prolactin 15.04 ng/mL, luteinizing hormone (LH) 4.35 mIU/mL, LH:FSH 0.7. These values were all considered to be within normal limits for being in the follicular phase of the menstrual cycle. These results were followed by a hysterosalpingogram to assess potential structural abnormalities, which was conducted 1 month later and shown in Figure . The impression noted a widely patent right fallopian tube and a patent left fallopian tube. Because of the widely patent right side, the contrast flowed very quickly, and it was difficult to appreciate the detail of the endometrial cavity. Overall, no focal abnormalities were noted. The patient subsequently underwent three cycles of clomiphene at a 50 mg dose for 5 days (instructed to be used during days 5-9 of the menstrual cycle) and one cycle of clomiphene at 200 mg with the same regimen. She was unsuccessful at conceiving after a combined four cycles and experienced gastrointestinal-related side effects with the 200 mg dose. She was then unsuccessfully trialed on letrozole 2.5 mg for 5 days using the same regimen and was referred to an infertility clinic.\nThe patient was next managed by our network 8 weeks into her pregnancy in the emergency department with a chief complaint of vaginal bleeding with abdominal cramping. She stated to the provider that she had undergone in vitro fertilization with the implantation of two 5-day-old embryos. A transvaginal ultrasound confirmed a singleton live intrauterine pregnancy with an estimated age of 8 weeks 0 days. She was subsequently discharged home.\nThe patient was reintroduced to our department during her first prenatal visit at 10 weeks 4 days gestational age (GA). At this appointment, she was screened and found negative for the following conditions: asymptomatic urinary tract infection, chlamydia, gonorrhea, hepatitis c, hepatitis b, human immunodeficiency virus (HIV), and syphilis. She was also found to be rubella immune with a negative urine drug screen. Her blood type was confirmed as B positive with a negative antibody screen. The patient elected to have genetic testing at this point in time which revealed an expected representation of chromosomes 21, 18, and 13 (normal result). Y chromosomal material was also detected, consistent with a male fetus. The patient continued with an uneventful pregnancy with a normal fetal anatomy scan at 20 weeks. Her 1-hour glucose tolerance test was borderline at 143 mg/dL and followed up with a 3-hour glucose tolerance test that revealed fasting, 1-hour, 2-hour, and 3-hour results of 84, 146, 111, and 102 mg/dL, respectively. The patient was swabbed for Group B Streptococcus (GBS) in her 36th week of pregnancy and was found to be colonized (positive).\nThe patient presented at 39 weeks and 0 days GA at the labor and delivery unit in active labor claiming rupture of membranes with clear fluid within the last 3-4 hours. Shortly after admission, she was adequately treated for the GBS colonization and provided epidural analgesia. She progressed through labor without augmentation and had a category 1 fetal heart tracing. After complete dilation of the cervix and more than 3 hours of active pushing, the course of labor was augmented by uncomplicated vacuum assistance. This resulted in the delivery of a healthy male infant and a first-degree laceration. After 30 minutes, the placenta had not detached from the uterus with the application of gentle traction. The patient had consented at this point in time for manual removal of the placenta, which was found to be morbidly adherent to the uterus. The attempt of placental removal resulted in atony of the uterus and brisk vaginal bleeding. Bimanual uterine massage and uterotonic drugs (oxytocin, methylergometrine, and carboprost) were sequentially administered and were unsuccessful at controlling the bleeding. The patient was urgently brought into the operating room for further management. First, a Bakri balloon tamponade catheter was placed without improvement, and the decision was made to move on to exploratory laparotomy. After transitioning to an abdominal procedure, the physician placed bilateral O’Leary sutures followed by multiple longitudinal and box-type compression sutures in the myometrium of the uterus. The bleeding remained uncontrolled and the B-Lynch technique was subsequently attempted. This sequence was used in an effort to preserve the patient's fertility.\nUltimately, a peripartum hysterectomy was needed despite following the uterotonic algorithm. A massive transfusion protocol was also declared, requiring the administration of six units of packed red blood cells (RBCs), two units of fresh frozen plasma, and two units of platelets in order to achieve hemodynamic stability. The patient was discharged after a 3-day hospital stay. In follow-up, her pathology report revealed a diagnosis of a 0.1 cm placenta increta. Placenta images are shown inFigure 2 and Figure . |
A 31-year-old woman came to our department with an abrupt and severe headache for half a day. At the time of admission, her Fisher scale was on grade 2 and the Hunt and Hess scale was on grade 1. She had no motor or sensory deficit but had signs of meningeal irritation. There was no history of trauma, hypertension, diabetes, or other disease. Computed tomography (CT) of the brain was conducted immediately and showed a subarachnoid hemorrhage (SAH) (). Thus, the patient was diagnosed with SAH and accepted regular treatment with intravenous mannitol and nimodipine, and the headache was greatly relieved. Seven days after headache symptom onset, the patient underwent digital subtraction angiography (DSA) examination under general anesthesia, and the result showed the presence of an aneurysm of the left PCA at the P1-P2 segments, measuring 4 × 5 mm with a neck of 1.5 mm (). Then, the patient underwent stent-assisted coil embolization of the aneurysm (). Stent-assisted coil embolization did not cause any neurological complication, and the patient's headache disappeared gradually within days postembolization. One week after coil embolization was conducted, brain CT examination was reperformed, and the result showed no abnormality in brain parenchyma. The patient was discharged and required to continue treatment with oral antiplatelet agents aspirin (100 mg/day) and clopidogrel (75 mg/day), which started the day after endovascular coiling treatment.\nOne month after endovascular therapy, aspirin, and clopidogrel were abandoned by the patient due to gingival bleeding and skin ecchymosis. On the day the antiplatelet agents were halted, the patient presented with transient visual disturbances characterized by spotted flashes around the bilateral visual field and a water ripple-like image stemming from the center of the bilateral visual field and spreading around. This visual disturbance lasted for 7–8 min and was then succeeded by severe right-sided throbbing headache lasting for about 2–3 h. The headache was accompanied with nausea, vomiting, and photophobia and aggravated by head movement. Head CT was performed immediately showing no localized hemorrhage or ischemic lesions (). The patient was underwent DSA examination again, showing that the coil was stable and there was no in situ thrombus (). From then on, she experienced episodic visual disturbance and headache attacks similar to the episode on the day the antiplatelet agents were halted every month with a frequency of two times a month. Each episode of headache would last 2–3 h. The headache was moderate to severe in severity and throbbing and pressure-like in character. Photophobia and nausea accompanied when the headache was severe. Before this event, she had neither a personal nor familial history of recurrent headaches. This recurrent event was considered to be either migraine with aura or epilepsy, and an electroencephalography (EEG) examination was conducted. The EEG showed epileptic discharges in the left occipital and temporal areas. A treatment with sodium valproate 500 mg twice a day was chosen for the patient as sodium valproate can usually effectively and prophylatically treat either migraine or epilepsy. The episodic visual aura and succedent headache were still recurring twice or even more times a month during the 3 months of sodium valproate treatment, but the headache severity seemed reduced. Sixteen months after endovascular coiling treatment, the patient accepted EEG examination again in our department, and spike-and-slow wave complexes could be detected in the left occipital and temporal areas (). The patient reaccepted antiplatelet treatment with clopidogrel (75 mg/day), omitting valproate and aspirin, and her migraine-like headache with visual aura relieved during the 4 months of follow up. One month after the readministration with clopidogrel, EEG was reconducted, and neither epileptiform discharges nor asymmetric background activity was observed (). |
We present a case of 68-years-old Indian, Hindu male patient who landed in emergency room with severe pain over lower abdomen associated with distention of abdomen and bloody diarrhea of one day duration. Patient gave history of intermittent lower abdominal pain and diarrhea for last eight months. Before coming to our institute he was diagnosed as a case of inflammatory bowel disease and was being treated with acetyl-salisylic acid (Mesacol 800 mg TDS) and intermittent steroid therapy for eight months. He was a chronic alcoholic and smoker. On examination, patient looked toxic with pulse rate of 126/minute. Tenderness and guarding was present all over abdomen. Blood samples were immediately sent for routine investigations, which were significant for the presence of leucocytosis (18,300/cmm), Na+ 129 mEq/l and K+ 2.9 mEq/l. ELISA test for HIV detection was negative. Urine sample was also sent for routine microscopy which showed few pus cells and presence of E. coli. Abdominal radiograph in erect position showed dilated bowel loops but no free air under the diaphragm. Ultrasonography of abdomen was asked for, which revealed distended large bowel loops with no fluid collection in peritoneal cavity. On per rectal examination rectum was empty and examining finger was tinged with blood and mucous.\nIntravenous antibiotics in the form of a combination of ceftriaxone, amikacine and metrogyl were started and patient was taken for exploratory laparotomy. Intra-operatively, multiple large areas of necrosis were found involving almost entire colon. Almost whole length of ascending and descending colon, and proximal sigmoid colon was perforated into the retro-peritoneum at multiple sites. Pelvic cavity was free from any fluid collection but minimal fluid was found in retroperitoneal space. Total colectomy with proximal ileostomy and exteriorization of distal end of sigmoid colon as mucous fistula was done. Luminal surface of resected colon had multiple ulcers measuring 3 mm to 5 cm, covered at place with necrotic purulent slough (Figure 1, 2, 3). Diagnosis of amoebic colitis was confirmed postoperatively on histo-pathological examination which showed necrotizing trans-mural ulceration with inflammatory exudates, and trophozoites of Entamoeba histolytica as isolated and in colonies at the ulcer bases (Figure 4A & B). Sections from the appendics showed features of nonspecific appendicitis. Terminal 6 cm portion of ileum was essentially normal. Postoperative course was complicated by wound infection and septicaemia. Unfortunately we lost our patient on 12th postop day due to multiple organ failure. |
A 25-year-old female patient known case of hypertrophic cardiomyopathy presented with complaints of chest pain, shortness of breath, and palpitations. The patient had complained of palpitations for many years but it was exacerbated for three months. She also complained of shortness of breath for the last three months (New York Heart Association (NHYA) class III) and chest pain which is central in location and exacerbated on exertion. She denied a previous history of syncope, or any chronic medical disorder, or any drug history. She also denied any sudden death in her family. On examination, a young age female patient well oriented lying on the bed comfortably. Her blood pressure was 100/70 mmHg, pulse was 70 beats/min and regular, she was afebrile and her respiratory rate was 18/min. On her precordial examination, the apex beat was located at the fifth intercostal space lateral to the midclavicular line, and a double apical impulse was appreciated. On auscultation, S1 and S2 heart sounds were audible of equal intensity and there was ejection systolic murmur (Grade 4+) at the aortic area radiating to the carotid area. The basic laboratory workup is mentioned in Table . On further investigation, her electrocardiograph (Figure ) showed sinus rhythm and left ventricular hypertrophy with deep T wave inversions and ST depression depicting LVH with strain pattern. The echocardiography revealed severe asymmetrically hypertrophied left ventricle with normal function and the systolic anterior motion of the mitral valve and dynamic left ventricular outflow tract obstruction. The aortic valve was thickened with no calcification and there was also a subvalvular aortic membrane seen. The specific findings of echocardiography are discussed in Table . The cardiac computed tomography showed a thickened trileaflet tricommissural aortic valve with no calcification on leaflets and aorta with no significant valvular aortic stenosis. There was severe asymmetrical hypertrophied LV, and diverticulum at mid muscular septum bulging of the membranous part of the interventricular septum (IVS) towards the right ventricle (RV) and a subaortic membrane (concentric only sparing anteriorly) resulting in LVOT obstruction, the Video given below shows the details of the scan.\nThe case of this patient was discussed with the heart team and it was decided to carry out left heart catheterization first and later surgical myomectomy and resection of the subaortic membrane. But the patient requested discharge and she was kept on medical therapy with beta-blockers and advised for close follow-up. |
A 2-year-old Asian Indian female presented to us with mild fever and swelling of the right upper lid of 10 days duration. There was no history of diarrhea prior to the onset of the symptoms. There was no history of preceding viral illness or significant medical history necessitating treatment with antibiotics. She was previously treated with oral antibiotics and drainage of the vesicle fluid. Subsequently, edema and blackish discoloration of the right upper eye lid developed. Cutaneous anthrax was unlikely as there was no history of unexplained cattle death in her environment. On examination, the child had low-grade fever and there were no other skin lesions. Ophthalmological examination revealed right upper lid edema with a large black necrotic area of the lid which was adherent to the underlying tissues. There was surrounding erythema and edema with no discharge []. The anterior segment examination was within normal limits. Left eye examination was unremarkable. The child was examined by a pediatrician to rule out any other focus of infection. Dermatological consultation yielded a diagnosis of EG clinically. Microscopic examination of the skin biopsy revealed staphylococci and hence cutaneous anthrax was ruled out. Blood cultures were negative. The child was started on intravenous cefotaxime for a week with resolution of fever and the necrotic area turned to a well-defined eschar with no edema and induration. After 2 weeks, the child underwent escharotomy with wound debridement and full thickness skin graft from the groin []. Under general anesthesia, the groin area was cleaned and draped. The skin was harvested from the groin under strict sterile aseptic precautions. The eschar on the lid was found to be partial thickness, was excised in toto, and the wound margins were debrided. The harvested skin was placed over the lid defect and sutured with 6-0 prolene. The graft took well and suture removal was done after 1 week []. |
This is a case of a 74-year-old male presented to our hospital with a one-week history of pain underneath the right jaw before and during meals. A week earlier, he had developed a sudden onset of swelling and pain underneath the right jaw associated with eating and the anticipation of eating. He did not have any constitutional symptoms such as fatigue, anorexia, weight loss, and fever. He was seen by the primary care physician. During the physical exam, it was noted that he had developed right submandibular salivary gland enlargement and was referred to an otorhinolaryngologist, who observed a stone at the opening of the salivary gland duct and cervical lymphadenopathy, as well as submandibular gland enlargement, on examination. A clinical diagnosis of sialolithiasis complicated by sialadenitis was made initially. It was presumed that cervical lymphadenopathy was a result of sialadenitis. At that time, imaging was not performed, as there were no symptoms concerning salivary gland tumor or abscess. He was prescribed cephalexin 500 mg twice daily for five days, which he took with some relief of pain. However, three days later, the patient presented to the emergency department with generalized weakness and fatigue. On examination, he was found to have a firm, palpable right submandibular salivary gland and bilateral cervical lymphadenopathy, right greater than left. Labs were ordered, which revealed significant leukocytosis of 47 x 109 cells/L (range 3.5 x 109 -10 x 109 cells/L). Peripheral blood smear (PBS) was negative for leukemic blasts. A computed tomography (CT) scan with contrast showed multiple enlarged cervical lymph nodes, the largest measuring 18.86 mm (Figure ) and moderate splenomegaly of 168.7 mm. (Figure )\nFlow cytometry of peripheral blood was performed. The lymphocytes were mostly T cells accounting for approximately 30% of total cellularity with the expression of cluster of differentiation 3 (CD3). These T cells showed a spectrum of expression of CD3 and CD5, where most of the cells were CD3 and 5+, with a small subpopulation showing lesser expression/loss of CD5 and CD3. All T cells expressed CD7 and CD2 and a majority of these cells showed aberrant expression of CD25. Only rare CD4 positive cells were seen (0.1%). CD8+ cells accounted for approximately 16%. The T cells appeared to partially express TCR alpha-beta (small subpopulation) and were negative for TCR gamma delta. They were negative for CD4, CD16, CD56, CD57, cTdT, CD34, and CD10. Thus, there was an abnormal population of CD8 positive T cells. The differential diagnosis of the peripheral blood smear flow cytometry included a T-cell lymphoproliferative disorder and a reactive process characterized by an expansion of a normal T-cell subset. The patient underwent a right submandibular excisional lymph node biopsy. Two days after admission, the patient was found to be encephalopathic. At this time, labs revealed an elevated uric acid level at 9.6 mg/dL (range 3.7-8 mg/dL), decreased ionized calcium level at 1.04 mmol/ L (range 1.17-1.33 mmol/L), elevated phosphorus level at 5 mg/dL (range: 2.6-4.7 mg/dL), elevated potassium at 5.5 mmol/L (range: 3.5-5.3 mmol/L), elevated blood urea nitrogen (BUN) at 110 mg/dL (range: 6-23 mg/dL), creatinine at 3.1 mg/dL (range: 0.67-1.23 mg/dL), which indicated spontaneous tumor lysis syndrome, complicated by acute kidney injury. The patient required daily hemodialysis. His hospital course was also complicated by new-onset atrial fibrillation and mild pericardial effusion. White blood count continued to increase up to 165 x 109 cells/L (range 3.5 x 109 - 10 x 109 cells/L) of which 33.9% were lymphocytes, and, therefore, was started on high-dose steroids (intravenous methylprednisolone 60 mg every 12 hours) and hydroxyurea 1000 mg every 12 hours for cytoreduction. Hemodialysis was continued daily. The patient continued to deteriorate despite supportive measures and was found to be in acute liver failure, leading to severe conjugated hyperbilirubinemia with a total bilirubin of 7.3 mg/dL (range: 0.0-1.2 mg/dL) and direct bilirubin of 7.2 mg/dL (range: 0.0-0.3 mg/dL). The submandibular lymph node biopsy revealed ALK-positive anaplastic large cell lymphoma (ALK-positive ALCL) with the expression of CD30. The specimen was sent to Yale University, and it was forwarded to Mayo Clinic for detailed expert consultation. The lymph node architecture was partly effaced by an abnormal lymphocyte population growing in the interfollicular zones. They were large with rounded and lobulated nuclear contours, dispersed chromatin, distinct nucleoli, and pale cytoplasm. The abnormal large cells uniformly expressed CD30 and ALK1. t(2;5) was not identified. The treatment options were discussed with the family, which included combination chemotherapy with the addition of the new agent brentuximab vedotin and allogeneic stem cell transplant for consolidation. Unfortunately, the patient remained critically ill from multiorgan failure secondary to spontaneous tumor lysis and thus was a poor candidate for chemotherapeutic intervention. On the twelfth day after admission to the hospital and within three weeks from the onset of symptoms, the patient expired from multiorgan failure secondary to the rapid progression of aggressive disease and spontaneous tumor lysis. |
A 36 year-old female patient was under the care of our mental health team with a diagnosis of mixed anxiety and depressive disorder, with emotionally unstable personality disorder traits (emotional dysregulation and sensitivity to abandonment). Her past medical history was unremarkable and she was taking no medications for physical maladies.\nTwo months prior to referral to our services her general practitioner started her on pregabalin 150 mg once daily for anxiety and in this time she had reported no appreciable side effects. She started attending anxiety management groups and there was some improvement regarding her anxiety symptoms. However, the patient was still complaining of generalized anxiety with significant panic attacks on occasion. Her pregabalin dose was increased to 150 mg twice daily and subsequently, after approximately 2 weeks, to 150 mg three times a day. She was also receiving venlafaxine MR 225 mg in the morning, as well as lorazepam 1–2 mg, as required (max 4 mg/24 h). Lorazepam was seldom utilized and only in severe anxiety attacks.\nThe increase in pregabalin dose appeared effective in managing her anxiety symptoms; improving her everyday functioning and reducing the number of panic attacks. After 3–4 days of receiving pregabalin 150 mg three times a day, she reported visual hallucinations and became increasingly agitated. She described having a ‘foggy state of mind’ and experienced visual hallucinations described as ‘black shadows’ in her visual field mimicking people, animals or objects. She further reported that on occasion the visual hallucinations were well defined and colorful; however, these were infrequent occurrences. There were no signs that the patient was experiencing depersonalization-derealization disorder and she remained alert, fully oriented in time, place and person, had organized speech and her memory was intact. She also maintained insight throughout; clearly distinguishing these hallucinations from reality. She did not report any delusions and there were no hallucinations in other sensory modalities. These visual hallucinations were not associated with any diurnal variation and no identifiable pattern emerged in their occurrence.\nPhysical examination including full neurological examination was unremarkable. Her visual acuity was within normal limits and ophthalmological examination was unremarkable. Blood test including FBC, Urea & Electrolytes, LFT, TFT, and CRP were normal. Blood exams excluded infection or any metabolic abnormalities. MRI head and EEG conducted 2 days after symptom onset showed no pathological findings. There was no previous history of psychotic symptoms or family history for psychosis. The patient reported lifetime abstinence from any elicit substances, she was a non-smoker and there was no history of alcohol abuse. We had no reasons to believe that the patient might had been tampering with pregabalin dose. Her sleep pattern remained normal and her food intake was the same as before the onset of visual hallucinations.\nNo other side effects, signs or symptoms were reported apart from visual hallucinations and agitation. The patient herself attributed these symptoms to the increase in pregabalin dose and she requested immediate cessation of pregabalin. Gradual discontinuation of pregabalin was agreed with her and stopped completely after 2 weeks. Visual hallucinations and agitation ceased completely after 5–6 days of pregabalin dose reduction and did not recur. Venlafaxine MR 225 mg in the morning was continued unchanged throughout and as required lorazepam was used to manage agitation symptoms effectively during pregabalin dose reduction. The patient was followed up for almost 1 year after pregabalin cessation and she did not report any visual hallucinations. |
A 73-year-old male patient presented with a complaint of pain and nasal discharge for the past 2 months. On examination, there was a diffuse mild swelling over the right maxillary area which was tender on palpation []. Maxillary right molars showed mobility. The computed tomography scan revealed a large mass filling the right maxillary sinus. Histopathological examination of incisional biopsy revealed a solid tumor mass showing dysplastic epithelial cells involving the underlying tissues. The dysplastic features seen were increased nuclear and cytoplasmic ratio, cellular pleomorphism, nuclear hyperchromatism and increased mitotic figures. In correlation with clinical and histopathological features, squamous cell carcinoma of the maxillary sinus was achieved. The patient was referred to the Department of Prosthodontics, V. S. Dental College and Hospital, before the surgery from ENT Department of KIMS Hospital, Bengaluru.\nOn examination, the maxillary arch had few missing teeth with Kennedy's Class III condition. A primary impression was made and maxillary cast was poured. The case was discussed with an ENT surgeon and the cast was marked with the extent of the tumor and the teeth to be involved in resection []. In contemplated area of resection, teeth were removed from the presurgical stone cast to the level of the gingival line and the cast was smoothed.\nAdams clasp was fabricated on the cast and surgical obturator made with clear acrylic []. The patient underwent surgery and the right premaxilla was resected []. Immediately after completion of the surgery, the surgical obturator was inserted in the patient's mouth and clasps were adjusted to stabilize the obturator []. After a week, the patient was reviewed and the prosthesis was cleaned. Around 15–20 days after surgery, the surgical site was completely healed up and ready for interim obturator [].\nA primary impression was made with alginate, cast was poured and custom tray was fabricated with self-cure acrylic.\nThe lateral defect in the right maxilla was recorded using impression compound, and care was taken not to extend the material medially. Borders were recorded and final impression was made [].\nMaster cast made using dental stone shows clearly the extent and depth of the defect. Adams clasp were done on premolar and molars on the left side and interim obturator was fabricated using heat-cure resin []. Finally, the interim obturator was adjusted in the patient's mouth with all teeth arranged in occlusion []. |
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