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A 29-year-old male, with a past medical history of extraosseous Ewing sarcoma of the right triceps, presented for a follow-up of his disease and was found to have an enlarged gastrohepatic lymph node.\nHe was initially diagnosed with ES of the right triceps (T2b N0 M0) at 12 years of age. He was treated with standard chemotherapy with VAC (vincristine, adriamycin, and cyclophosphamide) alternating with IE (ifosfamide and etoposide). He was then treated with radiation therapy consisted of 4500 cGy (180 cGy/fraction) with a boost to residual gross disease of 200 cGy/fraction for a total dose of 5500 cGy. The residual mass was followed thereafter, and he remained in remission until he turned 26 years.\nAt the age of 26 years, the patient had local recurrence and was found to have an increase in size of the mass within the right proximal triceps and abutting the humerus. FNA and core biopsy was positive for a small blue cell tumor and positive for CD99. These findings were consistent with Ewing sarcoma. He was then treated with temozolomide/irinotecan × 2; cyclophosphamide and topotecan × 6 followed by limb-sparing resection of his right upper arm mass with negative margins (closest 2 mm), with no further evidence of local recurrence.\nNearly 2.5 years after the aforementioned episode of local recurrence, the patient developed metastatic recurrence in his lungs and right orbit. Radiographic studies showed 3 nodules in the right lung and a mass in the right greater sphenoid that extended into his right orbit. An enlarged hepatic lymph node was also noted (). CT-guided biopsy of the lung nodule was positive for malignant cells consistent with metastatic Ewing sarcoma. The patient subsequently received radiation therapy (30 Gy/10 fx) to his right orbit for his sphenoid lesion and 8 courses of cyclophosphamide and topotecan.\nFollow-up imaging after completion of the therapy showed good response of lung metastasis with complete resolution of 2 nodules previously presented and near complete response of the 3rd nodule. Imaging also demonstrated orbital disease responded well to therapy. However, the patient's abdominal mass had minimal response. Positron emission tomography (PET) scan remained positive for the aforementioned mass, thought to be a gastrohepatic lymph node. An MRI of the abdomen subsequently showed a 2.1 cm mass in the pancreatic neck (). An endoscopic ultrasound (EUS) with fine-needle aspiration was performed for further evaluation. EUS showed an 18 × 16 mm hypoechoic mass in the proximal body of the pancreas (). Cytology of the fine-needle aspirate of the mass showed malignant cells consistent with Ewing sarcoma. Furthermore, tissue sections demonstrated a small blue cell tumor. The tumor cells were positive for CD99 but negative for both CD45 and S100, which was consistent with Ewing sarcoma.\nAfter discussion at the Sarcoma Tumor Board Conference at our institution and based on his previous successful response to radiation therapy to his right orbit, he was treated with radiation (30 Gy/10 fx) to the pancreatic metastasis. There was significant regression of the pancreatic mass after radiation therapy based on the follow-up CT imaging (). He remained symptom-free and disease-free at follow-up after 12 months and is currently undergoing periodic surveillance. The follow-up protocol typically followed includes MRI/CT with contrast of primary site, and chest imaging (CT or X-ray) every 3 months for 2 years, every 6-12 from 2 to 5 years, and annually thereafter. |
A 28-year-old female reported in March 2005 for evaluation of renal dysfunction noted during her first pregnancy. She developed bilateral pedal edema, hypertension and proteinuria during the third trimester of the first pregnancy in 1998. Thyroid evaluation during second and third trimester of pregnancy was reported to be normal. She underwent Caesarean section at 8th month of gestation, indication being accelerated hypertension. On evaluation 2 months after delivery, she was found to have persistent hypertension, proteinuria, and elevated serum creatinine (1.72 mg/dl). Tests for HIV, hepatitis B and C were negative. Screening tests for systemic lupus erythematosus were found negative. Kidney biopsy showed focal glomerular sclerosis with an increase in the mesangial matrix and tubular atrophy, suggestive of focal segmental glomerulosclerosis. She was treated with oral prednisolone, but developed pain in both hip joints after 5 months. Regional magnetic resonance imaging showed avascular necrosis of left femur head and so prednisolone was tapered and discontinued. She underwent hemiarthroplasty of left hip. Despite supportive treatment, kidney function deteriorated steadily and in 2005, she was initiated hemodialysis. Thyroid hormone levels were normal [].\nIn 2009, she experienced a sudden loss of weight (4 kg over 4 months). Clinical examination revealed body mass index 18.59 kg/m2, with Grade I diffuse enlargement of thyroid gland and Grade II inactive orbitopathy. Laboratory studies indicated hyperthyroidism []. Technetium 99 thyroid scintigraphy showed diffuse uptake of 6.8% (normal 1-4%) indicating Graves' disease (GD) [].\nShe was placed on carbimazole (20 mg daily as a single dose) and propranolol (20 mg twice daily). Propranolol was discontinued since she developed dizziness and postural hypotension. She became biochemically euthyroid after 4 months of antithyroid drugs. She developed retrobulbar pain and redness of eyes while on carbimazole. Examination revealed Grade II active orbitopathy, which was controlled by symptomatic treatment. The laboratory investigation results are noted in .\nShe developed relapse of hyperthyroidism upon weaning carbimazole after 14 months, and total thyroidectomy was offered. I-131 ablation was not planned considering the deteriorating orbital lesion. She preferred to remain on carbimazole (10 mg daily). She developed generalized body pain, asthenia and muscle weakness suggestive of myopathy and became wheel chair bound by 2012. Bone mineral densitometry studies of right wrist and lumbar spines showed T score of −3 and −2.7, respectively. She was diagnosed to have secondary hyperparathyroidism [].\nAfter control of thyrotoxicosis, total thyroidectomy and subtotal parathyroidectomy retaining portion of right superior parathyroid gland were done. Thyroid gland was diffusely enlarged with nodules in the right lobe and weighed 49 g. Histology showed hyperplasic follicles lined by occasional tall follicular cells. There was scanty pale colloid and occasional florid papillary hyperplasia. There was dense infiltration of lymphocytes in the stroma. The final histology showed features suggestive of GD of the thyroid gland and hyperplasia of parathyroid glands. The patient had uneventful postoperative recovery and symptomatic relief. |
A 69-year-old hypertensive man was found by his wife on the bathroom floor ~4 hours before referral to the neurosurgery unit at the institution. Before that, he had been complaining of right frontal headache and experienced three episodes of vomiting over the course of the preceding 24 hours. He had been suffering intermittent headache for ~6 months.\nInitial assessment in the emergency room revealed a Glasgow Coma Scale (GCS) score of 11/15 (E1 V4 M6), equal and reactive pupils and a dense left hemiparesis—Medical Research Council Grade 1/5 power in both upper and lower limbs. His presenting blood pressure was 162/109 mmHg. He had been taking aspirin with prophylactic indication. Initial non-contrast computed tomography (CT) scan of the brain revealed a massive right frontal intracerebral hematoma with significant vasogenic edema and mass effect as shown in .\nThe patient was intubated, then osmotherapy (20% mannitol 1 g/kg) and platelet transfusion commenced en route to the hospital. Cerebral CT angiogram on arrival did not reveal a vascular abnormality. He underwent emergency right frontal craniotomy, clot evacuation and debulking of a fleshy, pigmented tissue encountered. There were areas of clearly defined plane between normal and abnormal brain parenchyma; however, this distinction was less evident at other regions. The lesion extended to the anterior cranial fossa floor and was adherent to the anterior third of the superior sagittal sinus (SSS) and falx cerebri. There was neither a vascular abnormality nor was there excessive hemorrhage during surgery. Gross total resection was not targeted in the emergency setting given the intimate relationship of the lesion with the SSS. The evacuated clot and abnormal tissue were sent for histology in 10% formaldehyde. The craniotomy was closed after a pedicled pericranial flap duroplasty was fashioned.\nInitial intracranial pressure at closure was 4 mmHg and the patient was monitored in the intensive treatment unit postoperatively. He was successfully extubated and transferred to the open ward within 48 hours after surgery with normal GCS and no focal deficits.\nMRI brain was suggestive of meningioma and confirmed residual tumor (). Histopathological findings of perivascular epithelioid cells arranged loosely in sheets with a papillary architecture, immunohistochemical positive staining for vimentin, epithelial membrane antigen (EMA) and progesterone as well as increased Ki67 (5%) fraction, led to a final report of PM World Health Organization Grade III. No other abnormal mass was found on clinical examination.\nA reoperation was performed with neuronavigation and microsurgical subpial dissection was able to remove the remaining tumor whilst the anterior third of the SSS was coagulated. Eventually, a Simpson Grade II resection was achieved [].\nThe subsequent cerebral MRI found no residual tumor and normal neurological function was preserved. The patient was discharged home in good general condition 12 days after emergency admission. |
A pregnant (19 weeks) patient (27 years old) presented with per-vaginal bleeding and was diagnosed to have abruptio placentae. She did not have any prior medical illness or coagulopathy. The placenta was removed manually by the traction of the cord and digital separation under general anaesthesia, but she continued to have profuse bleeding in spite of maximal oxitotic treatment as per our hospital protocol (oxytocin intravenous infusion 40 IU, methyl-ergometrin 0.5 mg intramuscular, misoprostole 1000 μg per rectal). She became haemodynamically unstable in spite of resuscitation. She was immediately taken for emergency laparotomy under general anaesthesia for surgical control of bleeding after discussing all risks and benefits with the patient and relatives and taking a written consent. Generalized oozing was found mostly at the lower part of the uterus. Bleeding could be temporarily controlled with suture and packing in an attempt to preserve fertility. She lost about 3 L of blood which was replaced with eight units of packed red blood cells and fresh frozen plasma, six units of platelets and Cryoprecipitate. There was no hypothermia and the Acid base status of the patient was also corrected, and empirically calcium (1 g) was also given. But despite all our efforts, the patient started to bleed again. She was then shifted to the radiology room for uterine artery embolisation, but no active bleeding was found. Even after bilateral uterine artery embolisation, she continued to bleed. While obstetricians were considering hysterectomy, we decided to use the rFVIIa concentrate after discussing the risks and benefits with relatives. Bleeding reduced within few minutes after receiving a single dose of rFVIIa (90 μg/kg)[––] and she became vitally stable and her laboratory parameters normalised []. Finally, she was shifted to ICU where she was extubated on the next day. She was put on pneumatic stocking for thrombo-prophylaxis. She had an uneventful recovery without any thromboembolism (TE) or allergic complication. She was discharged after 7 days. |
A 30-year-old female reported with a gradually increasing, painless swelling in the upper left posterior palatal region since childhood. There was history of frequent bleeding from the lesion, and difficulty in swallowing and speaking.\nExtra-oral examination including the lymph nodes was insignificant. Intra-oral examination revealed a 2×2.5 cm growth which was red in color with bluish hue and with slightly pale periphery, arising from the buccal attached gingiva of maxillary left first and second molar, which extended palatally []. The lesion appeared sessile imperceptibly with the papilla and attached gingiva. Although two surface ulcerations were noticed, probably due to trauma during mastication, the remaining entire surface was smooth with several visible small vessels. The growth was gently soft to palpate and showed blanching on application of pressure, and was pulsatile but not reducible []. Intra-oral periapical radiograph of the region exhibited a horizontal pattern of bone loss due to chronic periodontitis []. Carotid angiogram could not be possible because of under-privileged socio-economic status.\nA provisional diagnosis hemangioma was laid down due to clinical appearance and characteristics of the growth. Preoperative hematological examination revealed all findings within normal parameters. Widman's periodontal flap of maxillary left quadrant was carefully raised under local anesthesia (posterior superior alveolar block) and the growth was excised []. Bleeding spots were cauterized and intermittent sutures were given and periodontal dressing was applied. The excised growth [] was sent for histo-pathological examination (HPE) and patient was given necessary hygiene instructions for home care of wound. Satisfactory uneventful healing occurred and follow-up was done for two years to monitor any recurrence.\nThe excised soft tissue mass was subjected to routine processing and several sections were obtained. HPE of the sections obtained from the lesion showed numerous capillaries filled with RBC in a delicate fibrillar connective tissue which was predominantly full of proliferating endothelial cells. Few inflammatory cells were also evident in the stroma. The connective tissue component was covered by parakeratinized stratified squamous epithelium which showed ulceration at few places. Areas of ulceration were covered with fibrin meshwork and were infiltrated by RBCs and mixed inflammatory cells. Correlating the microscopic features with the clinical picture, HPE confirmed the diagnosis as “Capillary hemangioma” [Figures –]. |
A 69-year-old female presented with the complaints of headache and pain in the left eye of one month duration. She was a known patient of unilateral pseudoexfoliation glaucoma, who underwent manual small incision cataract surgery with Mitomycin C (0.2 mg/ml for 2 min) augmented trabeculectomy in her left eye. It was a single site combined surgery in the superior quadrant of the eye, with a fornix based conjunctival flap and a single releasable 10-0 nylon suture anchoring the scleral flap using a modified (first bite in peripheral cornea) Kolker's technique. Surgery was performed 3 years back and she lost to follow-up after 1 month of surgery. At present, she had no history of diminution of vision or trauma to the left eye and was not a diabetic. The patient was pseudophakic in the right eye.\nBest corrected visual acuity was 20/30 in both eyes. Anterior segment examination of the right eye showed pseudoexfoliation material on the pupillary ruff with posterior chamber intraocular lens in the bag and the left eye showed clear cornea with a releasable suture, a low noncystic bleb with an elevated area and congestion overlying it, normal anterior chamber with pseudoexfoliative material on the pupillary ruff and a posterior chamber intraocular lens in the bag []. The intraocular pressure was 12 mmHg in both eyes and fundus examination showed a cup disc ratio of 0.4 with healthy neuroretinal rim in the right eye and 0.9 with bipolar notch in the left eye and age related macular degeneration changes in both eyes. The releasable suture in the left eye had mucus debris and the bleb showed collection of pus [].\nOn gonioscopy, ostium was free and posterior segment examination showed no signs of vitritis. Nasolacrimal duct of both sides were free of infection and patient's random blood sugar was within the normal limits. The releasable suture was removed and inoculated in blood agar and the collection of pus was expressed out along the same suture tract []. The patient was started empirically on oral Ciprofloxacin 500 mg twice daily for 5 days and topical 5% Cefotaxime and 2% Amikacin eye drops hourly.\nThe blood agar culture report revealed growth of Pseudomonas aeruginosa sensitive to the above antibiotics and hence the patient was advised to continue the same medications. After a week, the patient was symptomatically better, but on examination, we noticed a small recollection of pus in the bleb area []. The collection was again expressed and inoculated in blood agar and the patient was continued on the same topical antibiotics. The repeat culture report also revealed growth of P. aeruginosa. After 3 days and her last review visit, her left eye best corrected visual acuity was 20/30, the eye was quiet with a low bleb devoid of collection and ostium free of infection. Intraocular pressure was 10 mmHg and posterior segment continued to be free of infection. |
A 29-year-old woman visited Pusan National University Hospital with mild back pain when coughing and suprapubic discomfort after voiding. According to her medical history, she had undergone a hysterectomy 2 weeks earlier for uterine atony during a caesarean section and right-sided ureteroneocystostomy with bladder rupture repair after a right lower ureteral transaction injury. A ureteral D-J stent (6 Fr, 26 cm) was placed intraoperatively in the right urinary tract to prevent urinary leakage. No intra- or postoperative radiological evaluation was performed. Intravenous pyelography (IVP) on day 2 after admission showed that the D-J stent extruded from the urinary tract (). We could not find the distal end of the ureteral stent during a cystoscopic evaluation and considered removing the stent laparoscopically. Preoperative laboratory findings, including urine analysis, were normal except for a slight increase in C-reactive protein levels (0.75 mg/dL). Abdominopelvic computed tomography (CT) on day 3 after admission revealed that the ureteral stent had migrated further upward into the right cardiac chamber (). We changed our treatment plan and decided on endovascular management to reduce the possibility of any morbidity or mortality related to open surgery.\nBefore the radiological intervention, a scout x-ray examination showed that the distal end of the stent had migrated to the L1 vertebral level. The percutaneous approach was transfemoral on both sides and a 9-Fr sheath and 5-Fr sheath were indwelled in the right and left common femoral veins, respectively. A pigtail catheter extending from the left femoral sheath separated the distal end of the ureteral D-J stent from the right ovarian vein, and a snare loop from the right femoral sheath then drew the stent from the vascular circulation (). Inferior venography after the procedure showed no fistula tract or bleeding focus. The patient was discharged on day 5 after admission, and her postoperative course was uneventful. The follow-up CT and IVP showed normal urinary and vascular structures 5 months later. |
A 47-year old female patient visited our clinic with the main complaint of neck pain that had become severe over the past year. The medical history showed that the patient was undergoing psychiatric treatment for depression and had been diagnosed with ossification of the posterior longitudinal ligament (OPLL) for neck pain that had continued for 15 years. One year before visiting our clinic, she had undergone an operation in a neurosurgery clinic. The symptom did not significantly improve after the surgery and the pain continued. Thus, she underwent continuous rehabilitation therapy and physical therapy under the diagnosis of failed back surgery syndrome.\nWhen the patient came to the clinic, she complained of tearing pain concentrated in the neck and both nuchal regions. This pain became more severe when the patient was doing more than the usual household chores. The visual analogue scale (VAS) score of the pain was as high as 8 and was accompanied by sleep disorders. The physical examination showed pain during neck flexion and extension as well as direct tenderness at both trapezius muscles and the levator scapular muscle.\nA simple C-spine X-ray test and a laboratory test found no explanation for the pain. TPI was then performed, under the assumption that the myofasical pain syndrome originated in the trapezius and the surrounding muscles. Medication for failed back surgery syndrome was suggested, but the patient rejected it since the previously used analgesic anti-inflammatory agent had had no effect on her and she was taking an antidepressant and other drugs. Hence, as an outpatient,a small dose (12.5 µg/hr) of fentanyl patch (Durogesic D-trans®, JanssenPharmaceutica N.V, Tumhoutseweg, Belgium), which is a narcotic analgesic, was given, and TPI was performed on the trapezius muscle.\nWhen the patient returned as an outpatient after the treatment, the patient stated that the VAS score had decreased from 8 to 4-5 for 2-3 days following the TPI but later returned to previous levels. The patient did not show any side effects from the fentanyl patch. TPI was repeated three times, but the pattern of improvement for 2-3 days and exacerbation, thereafter, continued. Treatment methods such as botulinum toxin injection were considered but pulsed RF treatment to the trapezius muscle was performed first due to its lower cost and the fact that it could be used in outpatient treatment.\nFollowing betadine disinfection in the prone position, the accurate trigger point was located by means of an additional physical examination. Then, skin wheal infiltration was performed with local anesthetics. To verify the accurate needle position, 10 cm-RF needles (22 gauge, straight, 5 mm active-tip) were inserted into both trapezius muscles under ultrasound (S-nerve, Sonosite®, Bothell, WA, USA) guidance. Following sensory stimulation performed at 50 Hz with a potential less than 1V, pulsed RF (PMG230, Baylis medical company Inc, Montreal, Quebec, Canada) treatment was performed two times for each muscle (120 s, 20 ms, 2 Hz, 42℃, impedance below 400 ohms) ().\nWhen the patient returned as an outpatient one week after the pulsed RF treatment, the pain at both nuchal regions was much relieved as reflected in the VAS score of 1-2. This pain relief was continuously maintained. Thus, we decided to use only the fentanyl patch. Since the VAS score has been in the range of 2-3 for about two months after the pulsed RF treatment, TPI is no longer performed. |
A 36-year-old female patient [] was reported to the Department of Oral and Maxillofacial Surgery, Yenepoya Dental College, Mangalore, with a chief complaint of swelling in the upper right front and lower right back region of the mouth. She was confronted with a slow progression of the lesion for the past 1 year. On clinical examination of the extraoral features, there was a mild swelling on the right side of her face, with the skin appearing to be normal. While palpation, the swelling was hard, and mild tenderness was felt with the expansion of buccal cortical plate and egg shell crackling with respect to the right mandibular region. Intraorally, a well-defined swelling was present on the buccal vestibule of the right mandible and maxilla measuring about 4 cm × 3 cm extending in the region of root stump of 46 and 13, respectively, with vestibular obliteration.\nRadiographically [], a well-defined homogeneous radiolucency of size 5 cm × 4 cm with sclerotic border was observed in the right side of the maxilla extending from the distal aspect of the middle one-third of the root of the canine to the mesial aspect of the middle one-third of the root of the second premolar tooth. The upper margin of lesion was smooth and pushing the anterior wall of the maxillary sinus. Tooth 13 was tender on percussion. In the mandible, a well-defined radiolucency of size 3 cm × 2 cm with sclerotic border was noted extending from distal aspect of apical one-third of root of 45 to medial aspect of one-third of root of 48. The inferior alveolar canal was not involved, and the inferior margin of lesion appeared smooth. Tooth 46 was tender on percussion with well-defined radiolucency, and sclerotic border of size <1 cm was noted in relation to 23, 24, 25, and 26.\nOn the basis of clinical and radiographical findings, the provisional diagnosis of radicular cysts was remarkably made of both maxillary and mandibular lesions.\nUnder local anesthesia, the mandibular lesion was exposed by raising the mucoperiosteal flap as planned enucleation of the cyst in the mandibular body with extraction of offending root stump, i.e., 46 along with 42, 43, 44, 45, and 48, was performed during the first sitting. Flap closure was done with 3-0 silk suture and recalled after 7 days for suture removal. After 3 weeks, the cyst in relation to the right maxillary sinus was exposed and enucleated along with extraction of 12, 13, and 14. The cyst was enucleated in toto []. Thorough curettage was done. Flap closure was done with 3-0 silk suture and recalled after 7 days for suture removal. Postoperative healing was uneventful.\nThe histopathological report of both the surgical specimens was proved to be a radicular cyst. The report of mandibular lesion came as radicular cysts with fibrocellular cystic capsule with mild-to-moderate chronic inflammatory cells chiefly lymphocytes, plasma cells, and foamy macrophages. In case of maxillary lesion, the reports said inflammatory infiltrate, predominantly lymphocytes and plasma cells. Odontogenic rests were also evident in this section. |
The patient was a 3-year 7-month-old female referred by her speech-language pathologist, presenting with oromyofascial dysfunction characterized by speech sound production errors, difficulty swallowing, open mouth breathing, and noisy breathing during sleep. With respect to sleep, there were reports of difficulties going to sleep, waking up two to three times per night to drink water, getting up to go to the bathroom, open mouth breathing while asleep (), snoring during sleep, and sweating more than usual during sleep. She experienced wheezing that was associated with asthma, which was treated with an Albuterol sulfate inhaler. There were reports of difficulty with effective chewing. In addition, the patient would eat around 50% of her meals, before refusing the rest. She experienced chronic cough and recurrent upper respiratory tract infections.\nPhysical examination () of the patient showed her to be well developed, well nourished, and to appear the stated age. The patient was alert, oriented, able to communicate, and respond appropriately to questions. During the nasal examination, the nose had no external deformity. The nasal septum was straight, and the inferior turbinates were grade 2 bilaterally. There were class 3 dental occlusion and a class 3 facial-skeletal relationship characteristic of anterior-posterior maxillary deficiency. Oropharyngeal examination showed grade 3 modified Mallampati tongue position and grade 2 tonsils. The patient was found to have a restrictive class 2 upper labial frenulum with tethering of the upper lip () and restrictive grade 4 lingual frenulum [] ().\nBased on the patient history and the physical examination, the assessment revealed that the patient had functional and structural mouth breathing due to open mouth posture and low tongue posture in the setting of restrictive labial and lingual frenulum. The risks and benefits of lingual and labial frenuloplasty to facilitate lip closure and proper tongue resting posture were discussed with the parents and included, but were not limited to pain, inflammation, bleeding, scarring, need for revision surgery, and failure for significant improvement. An informed consent document was signed by the parents.\nThe maxillary labial frenuloplasty was performed under general anesthesia. Local anesthesia was achieved by applying 1 cc of 0.25% Marcaine with 1 : 200000 epinephrine to the maxillary labial frenulum via a 27-gauge needle. Pressure was applied lateral to the frenulum to locate the point of maximum tension. The maxillary labial frenulum was incised at the base of attachment with the use of sharp scissors. A V-to-Y lip closure was performed with a 4-0 chromic suture applied in a simple interrupted fashion ().\nThe lingual frenuloplasty procedure was then performed. A 2-0 silk suture was applied to the tip of the tongue as a retraction stitch. Local anesthesia was achieved by applying 1 cc of 0.25% Marcaine with 1 : 200000 epinephrine to the lingual frenulum via a 27-gauge needle. The tongue was retracted in the anteroposterior direction extending to the roof of the mouth and maxillary central incisors. Tension was applied to the floor of the mouth to protect the floor of mouth salivary glands, as well as Wharton's duct. A hemostat was used to clamp the restrictive lingual frenulum 5 mm above the attachments of the sublingual gland duct. The fibrous band was gently excised with the use of iris scissors. The underlying myofascial fibers of the genioglossus muscle were dissected further, with blunt cotton tips and sharp iris scissors being used to release the muscle from the overlying mucosa. The dissection was continued until there was adequate improvement to the tongue range of motion such that the tongue could extend up towards the maxillary central incisors in maximal mouth opening position. Simple interrupted sutures were used to close the diamond-shaped defect into a vertical line, as a means to lengthen the ventral tongue, with 4-0 chromic suture applied in a simple interrupted fashion. In total, the tongue was released from a grade 4 restricted range of motion to a grade 1 range of motion (). All wounds were hemostatic at the completion of the procedure. The patient was gently awoken from anesthesia and taken to recovery in stable condition.\nThe patient returned to the clinic four days after the procedure. The wound sites were healing appropriately, and there were no postoperative complications observed. The patient's mother reported that within the first day of returning home, the patient's issues with chewing had improved significantly, and she was more interested in eating different foods. In addition, her appetite appeared to have increased, and the patient would complete her entire meal before asking for more food (as compared to having only eaten around 50% before treatment). By the fourth day after surgery, the patient exhibited closed-mouth nasal breathing while asleep (). There were no longer any observed events of snoring and/or noisy breathing. The mother reported that the patient had remained compliant with myofunctional and speech therapy.\nThe patient returned for a 2-month postoperative visit. During the examination at this visit, no scar tissue was observed, and the wound sites had closed. Grade 1 tongue range of motion was observed ().\nThe patient's family wrote in a letter to the clinic approximately six months after the procedure, mentioning that the patient was doing very well with no complications. In addition, the patient had been reported to have completely stopped mouth breathing and snoring while asleep. However, the patient was reported to still have occasional episodes of cold and cough, as well as one episode of asthma exacerbation. Finally, the patient was reported by her family to have made progress with myofunctional and speech therapy, but the goals of eliminating the tongue thrust, achieving proper resting posture, and improving speech sound production errors were not met due to early discontinuation of treatment. |
A 75-year-old female never smoker presented to the ear, nose and throat specialist with hoarseness, dry cough, difficulty swallowing and a 3 kg weight loss since 1 month.\n5 years earlier she was treated with radioactive iodine for a toxic multinodular goiter. She also had a past medical history of a stage Ic well-differentiated serous papillary ovarian carcinoma treated with surgery and adjuvant chemotherapy.\nOn clinical examination, a multinodular and enlarged thyroid gland was palpated. A computed tomography (CT) scan of the neck and chest revealed a large mass (8 × 9 cm) in the anterior mediastinum involving the right thyroid lobe and causing deviation of both trachea and oesophagus. There were multiple pathologically enlarged cervical, supraclavicular and mediastinal lymph nodes as well as multiple bilateral lung metastases (). Further staging revealed a bone metastasis in the right iliac crest. During bronchoscopy paralysis of the right vocal cord was noticed as well as invasion of the trachea with reduction of tracheal diameter estimated at 25%. No intracranial metastases were present on baseline brain CT imaging. A biopsy of a cervical lymph node was performed showing diffuse infiltration by an abnormal epithelial cell proliferation. The abnormal cells were large with large and hyperchromatic nuclei. Multiple mitotic figures were present as well as necrosis and several infiltrating lymphocytes. Immunohistochemical studies on the neoplastic cells were negative for thyroid transcription factor-1 (TTF-1), PAX-8, thyroglobulin, cytokeratin AE1/AE3, cytokeratin 7, CD 45, CD 34 and S-100. The pathological findings were compatible with an anaplastic large cell malignancy.\nThe case was discussed at the multidisciplinary tumor board and based on clinical presentation, imaging and pathology a diagnosis of primary anaplastic thyroid cancer was made. Next generation sequencing (NGS, TRUSEQ, MISEQ, ILLUMINA) was negative for molecular drivers. PDL1-staining was positive with a high expression (60% of tumor cells). At that point the clinical condition of the patient was rapidly deteriorating due to dysphagia with impaired food intake. The possibility of palliative stenting of the oesophagus was discussed with the gastroenterologist but was considered technically impossible. The patient was unfit for a clinical trial. Based on occasional case reports describing exceptional response to immunotherapy in patients with anaplastic thyroid cancer, the patient was offered the possibility of treatment with pembrolizumab. After only 2 cycles, the patient noticed a marked improvement of dysphagia and after 3 cycles, a near complete response was seen on CT (). Thyroid function was thoroughly monitored during treatment and remained normal at all times.\nAfter 8 cycles however, the patient developed a severe grade 4 colitis (bloody stools, severe dehydration with hypotension) requiring aggressive fluid resuscitation and treatment with systemic steroids. Due to further clinical deterioration infliximab was administered, after which gradual improvement occurred. 3 months after the last administration of pembrolizumab, there were no signs of tumor progression. As most guidelines recommend permanent discontinuation of immunotherapy after grade 4 gastro-intestinal toxicity, a watchful waiting approach was proposed with follow-up CT of the chest and neck every 3 months.\n6 months later (16 months after diagnosis), the patient presented to the emergency department with gait difficulties, falling tendency to the left, left-sided facial palsy and hypoesthesia of the left upper arm. Magnetic resonance imaging (MRI) of the brain revealed a 19 × 18 × 19 mm lesion in the right thalamus. Stereotactic biopsy of this lesion was compatible with metastasis of known anaplastic large cell carcinoma. There were no signs of extra-cranial tumor progression (). Stereotactic radiotherapy to the brain lesion was performed. However her neurological condition did not improve. Because of poor performance status, previous grade 4 gastro-intestinal toxicity and mainly intracranial treatment failure, a re-challenge with pembrolizumab was not considered nor was second line chemotherapy. The patient was referred for hospice care. She died 18 months after initial diagnosis. |
A 78-year-old immunocompetent Caucasian male with past medical history of numerous BCCs and malignant melanoma of the right posterior shoulder (0.26 mm Breslow depth treated with wide local excision in 2010) presented with an asymptomatic, crusted, pink plaque of unknown duration on the left parietal scalp measuring 1.5 × 1.5 cm2. Shave biopsy revealed a nodular and focally infiltrative BCC. He was referred to the Mohs Surgery clinic for treatment.\nThe patient had a past medical history of multiple aggressive scalp BCCs. Five years prior he underwent Mohs surgery for two BCCs on the left and right parietal scalp. The left parietal scalp BCC recurred 18 months later and he was treated at an outside plastic surgery clinic with a wide local excision. He developed a BCC on the vertex scalp 9 months after that which was treated with Mohs surgery. Due to extension to bone, the patient also underwent adjuvant radiation therapy.\nMohs surgery was initiated for the most recent recurrent BCC on the vertex of the scalp (). Frozen section analysis revealed diffuse infiltrative BCC in the dermis and subcutaneous fat. In the fifth stage, intravascular tumor cells were noted (), and the patient was still positive in five out of six specimens with a defect measuring 6.5 × 7.0 cm2 (). Intravascular invasion, with CD31(+) and CD34(+) vascular channels, was confirmed with permanent sections by a board-certified dermatopathologist. No evidence of perineural involvement was seen in the sections examined.\nMohs surgery was stopped given the concern for widespread disease of the scalp and inability to attain a clear margin. A positron emission tomography/computed tomography (PET/CT) scan was ordered to evaluate for metastatic disease. The PET/CT scan demonstrated diffuse heterogeneous moderate tracer distribution in the left scalp wound favoring malignant involvement with an overlap of activity into the left parietal bone with erosion of the cortex, favoring osseous involvement. Prominent activity was last noted in the left cervical level IIa nodes, right sacrum, right scapula, and the third left rib, concerning for metastatic disease. A right sacral ala biopsy showed infiltrating nests of basaloid tumor cells in a sclerotic stroma, consistent with metastatic BCC (). After discussion at a multidisciplinary tumor board involving head and neck surgery, radiation oncology, and medical oncology, he started vismodegib 150 mg daily. Repeat PET/CT scan 6 months later showed decrease in the activity of right sacrum metastatic lesions, consistent with response to treatment, and stable cervical IIa nodes. However, there was an interval new subcutaneous focus over the right cheek suggestive of new BCC development, which is being managed with vismodegib at this time. |
A 34-year-old lady presented with history of shortness of breath of four-month duration and shortness of breath used to be worse on lying in the left lateral position (trepopnea). She also complained of dull aching central chest pain and dry cough for the same duration. In addition, she also complained of recurrent seasonal episodes of sneezing and nasal obstruction associated with headache. There was no history of fever, wheeze, and hemoptysis or weight loss. General physical examination revealed expiratory stridor. Respiratory rate was 24/min and oxygen saturation while breathing at room air was 95%. Examination of the respiratory system and rest of the systemic examination was normal.\nChest radiograph was normal. In a young female in view of trepopnea and expiratory stridor, intrathoracic (tracheal/major bronchial) obstruction was suspected and contrast enhanced computed tomography (CT) scan of the thorax was performed. In view of symptoms suggestive rhinosinusitis CT of paranasal sinuses was also performed. CT scan of the thorax showed presence of endoluminal polypoidal growth arising from the posterior wall of the tracheal bifurcation measuring 13 mm in anteroposterior diameter and 13 mm in the transverse diameter (). The growth was not enhancing postcontrast administration. There was no mediastinal lymph node enlargement and both lungs appeared normal.\nFlexible fiberoptic bronchoscopy (FOB) was performed for further assessment. It revealed a large fleshy polypoidal growth with well-defined narrow stalk arising from the posterior wall of the lower trachea (). The growth was flopping in and out of the right main bronchus and trachea leading to dynamic airway obstruction of either of the two main bronchi. The major bronchi and the distal bronchial segments were normal.\nIn view of the respiratory distress (central airway obstruction), a decision to attempt flexible bronchoscopic removal of the polyp was taken. Facility for rigid bronchoscopy was standby, in case there was any difficulty during flexible bronchoscopic removal. Olympus BF-1T-180 video bronchoscope (with a 3 mm working channel) via oral route was used. An electrosurgical snare loop (SD 7C-1, Olympus) was used for electrosurgical excision. The snare loop was used to encircle and firmly tighten around the neck of the polypoidal growth and electrosurgical excision was performed. The mass got avulsed from its base completely and stuck on to the snare loop and the same was removed in toto along with the flexible bronchoscope (). The procedure was done under conscious sedation using lidocaine nebulization and intravenous injection of midazolam 2 mg, fentanyl 50 microgram, and Phenergan (promethazine) 25 mg. There was no procedural complication/bleeding. Patient had immediate relief of symptoms of breathlessness and trepopnea in the same day.\nThe histopathology of the tracheal growth was suggestive of carcinoid (). A DOTANOC Positron Emission Tomographic CT scan was performed later which demonstrated no areas of abnormal tracer uptake. Patient is under regular follow-up and follow-up bronchoscopic examination has been normal. |
A 39-year-old woman presented with epigastric pain, intermittent nausea, frequent vomiting, and loss of appetite over the last seven days. She denied weight loss. She could pass gas but had not passed stool over the last four days. She was able to tolerate a full meal the night before presenting to the hospital, but nausea and vomiting had resumed within several hours.\nHer medical history was significant only for laparoscopic oophorectomy for a left ovarian cyst almost 20 years prior. She was taking no medications, herbal remedies, or nutritional supplements. She admitted to chronic alcohol abuse with frequent visits to the emergency department due to alcohol intoxication, but had never smoked cigarettes. She denied any recent sexual contact, overseas travel, raw food ingestion, or sick contacts.\nUpon presentation, the patient appeared to be in moderate distress. Her respiratory rate was 24 breaths per minute, but vital signs were otherwise stable. She complained of discomfort on palpation of the epigastric region. The upper abdomen was soft but mildly distended. No mass was palpated. Small scars from previous laparoscopy were noted.\nLaboratory results were only remarkable for a mildly elevated C-reactive protein of 0.44 mg/dL. Esophagogastroduodenoscopy (EGD) performed 20 hours after the patient's last meal revealed a mildly distended stomach with significant food residue in the esophagus and stomach (). The patient vomited copious food residue during the procedure, precluding a thorough examination for fear of aspiration. No gross abnormalities were found up to the third part of the duodenum. Food residue and intraluminal air were suctioned to the extent possible at the end of the examination.\nDespite the patient's ability to pass gas, an emergency computed tomography (CT) scan was conducted to rule out small bowel obstruction. CT without contract was largely unremarkable, with no visible signs of tumor or bowel obstruction (). However, CT with contrast taken several minutes later revealed the “target sign,” a bowel-in-bowel configuration measuring 7 cm in the proximal jejunum with invaginated mesentery (). No lead point was identified. No proximal distension was observed, most likely as a result of vomiting and suction during EGD. The patient was diagnosed with jejunojejunal intussusception, most likely of a transient nature.\nWhile the possibility of spontaneous reduction was explained, the patient wished to undergo exploratory laparoscopy due to the severity of her symptoms. The surgeons also agreed to exploratory laparoscopy in light of the severe obstructive symptoms, surgical history with possible adhesions, and possible recurrence if left untreated. Consent for intraoperative endoscopy was also obtained to evaluate the jejunum for a possible lead point, as the patient was not in a condition to undergo preoperative enteroscopy.\nExploratory laparoscopy was performed the next day. Severe jejunal hyperactivity was observed intermittently throughout the laparoscopy. However, no intussusception was observed, suggesting spontaneous reduction. Tumors and segmental edema in the proximal jejunum were also notably absent. Adhesions from previous laparoscopic oophorectomy were observed near a port placed in the right lower quadrant (). While adhesiotomy was performed, the adhesions were distant from the proximal jejunum and appeared as an unlikely cause of intussusception.\nIntraoperative peroral jejunoscopy was performed with a long colonoscope (PCF-H290L, Olympus Corp., Tokyo, Japan) and carbon dioxide insufflation. A thorough laparoscopic exploration was completed prior to commencing intraoperative endoscopy, as endoscopic insufflation would hinder the laparoscopic view. The surgeons used laparoscopic grasping forceps to apply gentle pressure to the stomach to facilitate scope insertion (). When the endoscope reached the jejunum, the laparoscopic camera was pointed to a location in the jejunum believed to be distal to the reduced intussusception. Forceps were also gently placed at this location to facilitate insufflation (). The endoscope was inserted until light from the laparoscopic camera was visualized, confirming the absence of tumors or other lesions which may serve as a lead point for intussusception (). Short bowel resection was therefore not performed. The patient was diagnosed with transient jejunojejunal intussusception, more likely associated with chronic alcoholism rather than adhesions from previous surgery.\nThe patient experienced complete resolution of her symptoms after the surgery. The postsurgical course was uneventful and the patient was discharged two days later, with instructions to stop drinking alcohol. No recurrence was observed during 24 months of follow-up. |
The patient was a 16-year-old boy of Han ethnicity who was a local high school student; he had experienced upper abdominal pain and fatigue for 3 months. At the time of admission to our hospital, he had lost 4 kg in weight. He had no other symptoms and no previous history of other diseases. Gastroscopy revealed a mucosal protrusion with a smooth surface in the fundus of the stomach (Fig. a). A miniature ultrasonic probe and EUS revealed a hypoechoic lesion (10.8 mm X 22.4 mm) that was suspected of having originated from the muscularis propria of the gastric wall; the internal echo was uniform, and the boundary was clear (Fig. b and c). Based on our experience and these findings, we considered the lesion to be an interstitial tumor. After communicating with the patient and his family, they decided to choose minimally invasive surgery, namely, endoscopic submucosal dissection (ESD), under general anaesthesia. After completing the preoperative examination (chest CT: obsolete pulmonary tuberculosis and abdominal ultrasonography: no obvious abnormalities), we performed ESD on the patient. During the operation, we did not find any lesions originating from the muscularis propria. However, after opening the muscularis propria, we were surprised to find a lesion with a relatively complete package and multiple nodules on the surface. This lesion severely compressed the muscularis propria and pushing it into the gastric lumen, and it was continuous with a lump similar to the enlarged lymph node outside the gastric wall (Fig. d and e). After the lesion was successfully removed, a histopathological examination (HE staining) was routinely performed. Surprisingly, the pathological results revealed chronic granulomatous inflammation with caseous necrosis, which is consistent with tuberculosis (Fig. f 200X and Fig. g 400X). We next performed ESR (35 mm/h) and PPD (+) analyses. The patient was eventually diagnosed with gastric tuberculosis and given anti-tuberculosis treatment for half a year, at which point the lesion in the fundus of the stomach had disappeared, leaving a slight white mucosal depression with scar formation (Fig. h and i). |
The patient was a 55-year-old African-American man with a history of prostate cancer (diagnosed via core needle biopsy 2 years prior). At that time, the patient had 6 specimens obtained from different prostatic tissue locations submitted for histopathology. Four out of the 6 specimens showed benign prostate tissue in the left apex, right apex, left mid, and left base. The remaining 2 specimens showed prostatic adenocarcinoma in the right base and right mid zones, respectively: a Gleason score of 9 (5+4) was noted in 4 out of 4 submitted core segments from the right base, and a Gleason score of 8 (4+4) in 3 out of 3 core segments from the right mid zone. No therapy (chemotherapy, hormonal, or radiation) was implemented prior to presentation to our facility. He reported previously treated Lyme disease and chronic kidney disease stage 3, and complained of right neck pain that had started after a mechanical fall. He denied any trauma to the head, loss of consciousness, syncopal symptoms, any weakness in his lower extremities, unsteady gait or numbness, or tingling in his extremities. The patient also reported recent onset of coughing spells associated with drinking of fluids.\nMRI of the thoracic spine disclosed significant compression of the T4 vertebral body with mild associated cord compression, and diffuse patchy enhancement throughout the thoracic spine. Initial CT scans revealed no acute bony trauma, but showed multiple destructive lytic lesions and partial sclerosis in the lower calvarium (Fig ), both clavicles, and the lower cervical and thoracic vertebra (Fig ). A paravertebral mass in the right upper mediastinum was also visualized (Fig ). Subsequent MRI images of the brain with and without contrast displayed a right-sided, 1.3 × 4.3 × 2.5 cm, intermediate-signal mass at the base of the skull in both the T1- and T2-weighted sequences. Chest X-rays revealed a small right lung apical opacity with probable destruction of adjacent first and second ribs. Further workup showed small sclerotic densities in the left sacrum and right iliac bone, assessed as metastasis. Multiple myeloma workup, including serum and urine protein electrophoresis, immunofixation, beta-2 microglobulin, and serum free light chain quantification, was negative. The serum PSA level was elevated at 994.47 ng/ml.\nTo further assess the extent of disease, a bone scan was obtained after administering 26 mCi of technetium-99m methylene diphosphonate intravenously. This revealed multiple foci of abnormally increased tracer accumulation involving multiple ribs bilaterally as well as the cervical, thoracic, and lumbar spine (Fig ). Small foci of increased tracer accumulation were also noted in the humeral shafts, left proximal femur, and left mid femoral shaft. MRI of the cervical and thoracic spine with and without contrast revealed diffuse, patchy, bony metastases and a paraspinal mass on the right, extending from T1 to T4 and measuring approximately 7 cm in craniocaudal dimension, with significant compression of the T4 vertebral body with mild associated cord compression (Fig ). MRI of the brain also revealed a contrast-enhancing mass at the base of the skull on both the T1 and FLAIR sequences (Fig ).\nThe patient's symptoms (choking spells and neck pain) were attributed to cord compression as evident on MRI imaging, and he was started on dexamethasone for symptom relief. He was also started on 6 cycles of chemotherapy with Taxotere in addition to intramuscular leuprolide for treatment of his prostate cancer, given the extensive burden of his disease. Radiation oncology was consulted for consideration of radiotherapy to the lesion at the base of his skull and the paravertebral mass. They formulated a treatment plan of 2.5 Gy per fraction doses of external beam radiation to the skull base in 14 fractions for a total dose of 35 Gy, and 2.5 Gy per fraction doses of radiation to the paravertebral mass in 14 fractions for a total dose of 35 Gy. The patient's coughing paroxysms associated with swallowing resolved after just 2 sessions of radiotherapy.\nThe patient has had 4 cycles of chemotherapy thus far and is responding well to treatment. Restaging/surveillance imaging will be obtained once he has completed 6 cycles of chemotherapy. |
An 11-year-old male Rwandan, a known patient of multiple hereditary exostoses (MHE), presented to our hospital with a 6-month history of unrelenting bone pain despite treatment with NSAIDs; he later developed (two weeks prior to admission) a dull persistent aching pain and swelling of his left lower thigh that was worse at night and unresponsive to morphine. His parents noticed that he had also lost weight and appetite. There was no history of trauma or a fall. He was diagnosed with MHE at age 3 and had been living a relatively normal life and attending school. None of his siblings or any other member of his family had MHE. His past surgical history was unremarkable. On general examination, he was well nourished, had mild pallor of the mucus membranes, no jaundice, lymphadenopathy, or skin rash. All the other parameters were normal. Examination of the musculoskeletal system revealed normal stature except for the curving deformity of the left leg. There were multiple palpable bony swellings bilaterally on the upper humeri and lower femurs. The lesion on the left distal femur was markedly enlarged and tender, with induration, reddening, and limited range of motion of his knee joint (). Repeat X-rays confirmed the presence of bony outgrowths (exostoses) on the medial and lateral aspects of the distal femurs bilaterally and left metaphyseal widening common in this condition as had been previously identified when he was diagnosed at age 3, and further investigations of the left distal femur swelling () with magnetic resonance imaging (MRI) revealed a distinct enhancing lesion in the distal aspect of the left thigh at the site of intense swelling and pain. MRI showed a lesion with hypointense signals on T1WI sequences and has heterogeneous signal intensities with moderate and heterogeneous enhancement on T1WI postcontrast study and on T2WI sequences; the lesion had heterogeneous signal intensities (Figures –). At surgery, there was a necrotic lesion, and excision biopsy at the site of the left distal femur exostosis was taken (Figures and ). Hematological evaluation was normal except for a mild leukocytosis. The results of the serum biochemical tests were also normal. We did not do genotyping for EXT-1 and EXT-2 due to lack of facilities.\nHistopathological examination revealed a characteristic fibrous cartilaginous cap with a broad base (1.293 mm thick), covering a layer of normal appearing marrow and bone below which was a tumor-forming osteoid, an osteoblastic tumor as evidenced by presence of numerous bone spicules of varying maturity. There was also marked cellular atypia, grossly pleomorphic osteoblasts in the marrow with frequent mitoses. These features were those of a high-grade osteoblastic osteosarcoma. In conclusion, histopathological revealed an osteochondroma with an underlying high-grade osteoblastic osteosarcoma involving the marrow cavity (Figures –).\nFor staging purposes, CT scans of the chest, abdomen, and pelvis were done to investigate any presence of metastatic lesions (Figures and ). There were no metastatic deposits in the lungs, abdomen, and pelvis, and this was confirmed with PET scan in India where the patient was referred for specialized bone tumor treatment including limb salvage therapy. He was reevaluated, and the diagnosis of MHE and osteoblastic osteosarcoma confirmed. Whole body PET scan showed metabolically active disease in the distal left femur 8.6 × 8.1 × 16 cms in dimension with features consistent with osteosarcoma, multiple hereditary exostoses with evidence of skip lesions, loco-regional lymph node involvement, and no distant metastases. Histopathological review confirmed the earlier diagnosis of osteochondroma with osteoblastic osteosarcoma. The conclusion was that the patient had clinically localized disease. The following treatment plan was proposed and instituted: initiation with neoadjuvant chemotherapy followed by limp salvaging surgery and finally adjuvant chemotherapy. He received doxorubicin 35.5 mgs/m2 per day (day 1 and day 2), cisplatin 60 mgs/m2 for 7 days (days 1 and 2), methotrexate 12 gms/m2 per day, etoposide 100 mgs/m2/day for 5 days, and ifosfamide 2.8 gms/m2/day for 5 days followed by Ifosfamide 3 gms/m2/day for 3 days and subsequently pegylated Interferon-α2b 0.5 mcg/kg - 1mcg/kg. He completed a total of six cycles of chemoimmunotherapy.\nOur patient is still alive and back at school with no evidence of disease after 11 months of treatment and follow-up and continues to be followed up by the oncologist. |
We present the case of a 29-year-old nulliparous, 39-week pregnant woman who was hospitalized for irregular contractions in early labor, no ruptured membranes, no bleeding and very good fetal movements. The pregnancy follow-up was uneventful, with both first and second-trimester anomaly scans within normal limits. There was no other associated pathological medical history. Clinical examination and vital signs were normal. At the ultrasound examination, the placenta was high posterior, there was normal amniotic volume, cephalic presentation and active fetal movements, and estimated fetal growth of 3500 g with normal umbilical, cerebral and uterine arteries doppler.\nDuring routine cardiotocography, a pathological trace was recorded—tachycardia and late deceleration (). As per guidelines [], immediate action to correct reversible causes and additional methods to evaluate fetal oxygenation were required. Because the patient requested a vaginal delivery and chronic hypoxia needed to be excluded, the artificial rupture of membranes was performed to rule out the presence of meconium and to assess other possible causes of fetal hypoxia. The amniotic fluid had a normal aspect, with no signs of meconium, blood or foul smell. Having excluded infection and chronic hypoxia, and because the trace was reassuring with stable baseline and normal variability, the plan was to reassess progress in 2 h under continuous CTG monitoring, IV hydration and maternal repositioning. Two hours later, another event raised the concern of suspected fetal compromise—a prolonged 4 min late deceleration immediately after a high amplitude fetal movement ()\nA decision was made for a category 2 cesarean section. A healthy 3500 g male was delivered in good condition (Apgar score 8). The macroscopic placental examination was normal. During the first routine clinical examination of the neonate, a Wharton’s jelly anomaly was identified at the abdominal umbilical cord insertion. For approximately 1 cm of length, umbilical cord vessels were completely uncovered by Wharton’s jelly. The condition required surgical thread elective ligation of exposed umbilical cord vessels (). |
A 19 month old Brazilian female was admitted at the emergency unit of a teaching hospital with fever, cough, irritability, and dyspnea. Her mother said that the child was on one week’s use of amoxicillin and bronchodilator drug.\nShe was born by C-section at 29 weeks and six days in the Obstetric Emergency Unit of the same institution and was immediately transferred to the PICU and intubated because of her critical clinical state. Her birth weight was 1020 g. By day 13, she presented a bloodstream infection due to Candida albicans, which was successfully treated with amphotericin B (AMB). The heart auscultation showed a presence of a continuous murmur and the echocardiography identified the patent ductus arteriosus (PDA) and tricuspid regurgitation, which led to a surgical correction (A–E). An iatrogenic chylothorax was diagnosed and during the procedure to repair it, the child presented cardiopulmonary arrest and the surgery was stopped. Then, a thoracentesis was performed with a placement of a chest tube, which remained for three months. A few days after the thoracentesis, a positive urine culture for C. albicans was evidenced and she received amphotericin B for 10 days. By day 30, she presented a positive catheter tip culture for Pseudomonas aeruginosa with a negative blood culture, successfully treated with the catheter removal. By day 43, Candida albicans and coagulase-negative staphylococci bacteria were recovered from blood and treated with AMB and penicillin, respectively. Due to the presence of cervical edema, an echocardiogram was performed and a superior vena cava thrombosis was evidenced. She was heparinized for three and a half months. After seven months, she was discharged from the PICU. However, the child was admitted in the same hospital in two different occasions due to fungal and bacterial infections.\nAt admission she was afebrile and weighed 7.0 kg. Pulmonary auscultation: presence of bibasilar crackles and rhonchus. Heart: mild systolic murmur. Abdomen: flaccid and distended. Liver: 3 cm below costal margin. Chest X-ray evidenced cardiomegaly and a right middle lobe consolidation. The next day, she presented cyanosis, anasarca, chest in-drawing, and worsening of her general clinical state. A moderate pericardial effusion with hemodynamic repercussion was evidenced at echocardiogram (F–H). She was admitted to the PICU, intubated, and treated with vasoactive drugs, diuretics, fluconazole, ceftriaxone, and oxacillin due to sepsis hypothesis. One week after, the antibiotics scheme was replaced by vancomycin plus cefepime. A pericardiocentesis was performed and the pericardial fluid analysis showed pH 8.0, density 1.030, glucose 60.8 mg/dL, lactate dehydrogenase 1362.0 U/L, amylase 5.0 U/L, and proteins 5.19 g/dL. Gram, culture for bacteria and fungus, and Ziehl-Neelsen staining were negative. Over the next three weeks, her clinical condition remained unchanged. Despite 12 days on fluconazole (FLZ) prophylaxis (25.0 mg/kg/day), a positive culture for Candida sp. was identified. A second blood culture six days later identified the same microorganism. After 14 days on fluconazole (25.0 mg/kg/day) use, a maintenance dose (6.0–12.0 mg/kg/day) was prescribed and amphotericin B (attack doses: 0.5, 1.5, 3.5, 4.5, 5.5, and 11.5 mg/kg/day) implemented. Two days after the first positive culture, there were a gradual reduction on the hemoglobin level and white blood cells (WBC) count, with severe leukopenia and neutropenia for which she received granulokine and blood transfusion. In a period of 10 days, the WBC count reduced from 10,990 cells/mm3 (bands: 1429 cells/mm3, neutrophils: 8571 cells/mm3, eosinophils: 220 cells/mm3, lymphocytes: 440 cells/mm3, and monocytes: 330 cells/mm3) to 180 cells/mm3 (neutrophils: 14 cells/mm3 and lymphocytes: 166 cells/mm3). Her clinical state worsened presenting anasarca, epistaxis and hemorrhagic suffusions in the mouth and sclera, oliguria, and bradycardia. She died five days after.\nThe blood samples were collected using the BACTEC system (DifcoTM, Sparks, Baltimore, MD, USA) and processed through the BACTEC 9240 (Becton, Dickinson, and Company, Sparks, MD, USA) automated culture system. Presence of yeast cells was confirmed by Gram staining of the positive vials. Blood was subcultured in CHROMAgar Candida medium (Becton, Dickinson, and Company, Sparks, Baltimore, MD, USA) for 24–48 h at 37 °C yielding small pink colonies. The germ tube test was negative. As the first line of identification, colonies were subjected to VITEK® 2 (Biomeriux, Marcy l’Etoile, Lyon, France) and this identified both isolates (UFTM 08.01 and UFTM 08.02) as C. pelliculosa. Antifungal susceptibility tests to AMB, ketoconazole (KET), itraconazole (ITZ), voriconazole (VOR), and FLZ were performed according to CLSI M27A3 []. Both isolates were susceptible to all antifungal tested. The Minimal Inhibitory Concentration, MIC (µg/mL), range were as follows: 0.12–0.25 for AMB, 0.12 for KET and ITZ, 0.12–0.25 for VOR, and 1.0–2.0 for FLZ.\nGenomic DNA, internal transcribed spacer (ITS) region amplification and sequencing were performed as previously described [,]. The Basic Local Alignment Search Tool (BLAST) search revealed that the isolates showed 99% similarity with CBS 260 type strain of W. anomalus. The sequences of the herein described isolates were compared with the ITS sequences of different species of Wickerhamomyces retrieved from CBS-KNAW culture collection, NRRL Culture Collection, and NBRC Culture Collection. The sequences were aligned using the Muscle algorithm in Mega X [,]. The best substitution model, K80, was calculated in jModelTest v. 2.1.10 []. The Maximum Likelihood tree was inferred and visualized using the Interactive Tree of Life. This analysis confirmed the identification of both isolates as belonging to W. anomalus clade, composed of the type strain CBS 5758 and CBS 260, the last one also recovered from blood (). |
A 49-year-old Caucasian lady with history of polysubstance use disorder and related complications including, recurrent cutaneous abscesses, spinal diskitis and septic thrombophlebitis presented to the emergency room with complaints of intermittent fevers and right hip pain. A month prior to the presentation, she had left another hospital against medical advice after being diagnosed with Methicillin-resistant Staphylococcus aureus bacteremia and right hip septic arthritis. Post discharge, she was off antibiotics, but continued heroin and methamphetamine and was taking multiple dose of ibuprofen in addition for pain control. On admission, her physical exam was notable for severe tenderness in her right hip, marked bilateral lower extremity edema and multiple deep, circular, punched-out looking atrophic scars involving all extremities at prior skin popping (subcutaneous injection of illicit drugs) sites. Pertinent laboratory data included chronic anemia with a hemoglobin of 9.8 g/dL, WBC count of 10.23 k/uL and a platelet count of 395 k/uL. She had negative HIV, Hepatitis B antibodies and elevated Hepatitis C antibody with undetectable Hepatitis C viral load. Her basic metabolic profile noted a sodium of 140 mmol/L, potassium of 3.5 mmol/L and a creatinine of 2.9 mg/dL (estimated glomerular filtration rate of 17 ml/min) which was a significant rise from the normal creatinine levels a month earlier. Her urine analysis noted >500mg/dL proteinuria with a protein/creatinine ratio of 28.25. She had no monoclonal proteins on serum or urine electrophoresis. CT scan of her right hip noted marked degenerative changes in the right hip, with right acetabular protrusion and cortical disruption of the medial acetabular wall. She was diagnosed with right hip osteomyelitis and was in acute renal failure with evidence of nephrotic range proteinuria. She was placed on antibiotics (daptomycin) and underwent hip arthroscopy with irrigation and debridement along with lavage shortly after admission. Differential diagnoses considered for her renal disease included acute tubular necrosis due to sepsis, post infectious glomerulonephritis, focal segmental glomerulosclerosis associated with heroin use, acute interstitial nephritis from NSAIDs and membranoproliferative glomerulonephritis associated with Hepatitis C. She underwent a renal biopsy which revealed acute tubular necrosis and secondary (AA) amyloidosis with the classic apple green birefringence when stained with Congo red () and positive immunohistochemical stain for serum amyloid A protein (). Two weeks after admission she underwent Girdlestone arthroplasty. During the hospital stay, she developed intermittent hypotension, had evidence of primary adrenal insufficiency attributed to amyloidosis and was initiated on steroids. She was discharged home after completion of prolonged antibiotic therapy with daptomycin and was maintained on oral doxycycline. She was referred to outpatient opioid treatment program. Eight months after her admission, she remained committed to be off all illicit drugs and underwent right total hip replacement. Her creatine levels normalized (estimated GFR of 82 ml/min) but she continued to have proteinuria from renal amyloidosis (protein/creatinine ratio of 28.25) and required diuretic therapy for symptomatic management of her edema. |
A fifty-eight-year old man was evaluated for painless macroscopic hematuria. The patient had a family history of von Recklinghausen's disease with the typical multiple cutaneous neurofibromas. Initial abdominal ultrasound revealed a tumor on the bladder dome which was confirmed by a subsequent cystoscopy. A CT scan of the abdomen showed no perivesical tissue extension, no lymph node enlargement and no hydronephrosis presence (). Due to its uncommon endoscopically features and extension of the lesion (large base, smooth surface, and approximate size >3 cm) the patient underwent an incomplete transurethral resection of the tumor for staging purposes and to define its origin. Histopathology following tumor resection depicted a neoplasm of mesenchymal origin with immunohistochemical findings indicating the diagnosis of inflammatory myofibroblastic tumor (IMT). Vimentin was diffusely expressed in neoplastic cells and desmin, α-SMA, s-100, CD117, CD34 and keratins of low molecular weight were focally positive. (Figures and ). On the other hand CD30, NSE and keratins of high molecular weight were not expressed at all.\nInterestingly, our patient had a recent 5-month history of suprapubic prostatectomy due to lower urinary tract symptoms, during which no bladder wall pathology was recorded. In addition, preprostatectomy imaging was reviewed and quoted as normal. Although the diagnosis of inflammatory pseudotumor could be made, the patient was consented for the uncertain biological behavior and the malignant potential of the tumor. Upon discussing definitive treatment the patient decided to undergo radical cystectomy. As a consequence, radical cystectomy followed by an ileal conduit was performed. Additionally due to lack of genetic test to confirm his inherited disease, the excised cutaneous lesions from the site of the performed stoma were sent for histopathologic evaluation (). Macroscopic view of the cystectomy specimen revealed two polypoid lesions with smooth, elastic surface, 1 and 1,5 cm in diameter located at the bladder dome (). The ulcerated surface of the polypoid lesion was recognized microscopically (). The microscopic and immunochemistry findings confirmed the initial diagnosis after the transurethral resection of the lesion. |
A 23-year-old man was referred to our clinic because of chest pain, cough and hemoptysis. His physical examination and chest X-ray revealed left pleural effusion. Thoracentesis was performed and exudative aspiration fluid cytology did not show atypical or malignant cells. Routine laboratory tests were normal except a mild increase in lactate dehydrogenase. Echocardiography (echo) revealed a left atrial mass measuring 3.8 × 3.8 cm in diameter, filling the left atrial cavity and extending into the mitral valve during diastole, causing severe mitral stenosis symptomatology (fig. ). The presumptive diagnosis was a cardiac myxoma; because of the symptoms it had caused, tumor excision with mitral valve replacement was performed. Pathological examination revealed a high-grade conventional osteosarcoma arising from the cardiac wall. The tumor cells were mostly osteoblastic in nature. The lace-like pattern of the osteoid matrix was seen between these atypical osteoblasts. S-100 immunostain was negative and vimentin was positive. S-100 negativity excluded the possibility of a malignant nerve sheath tumor with extensive tumoral bone production (fig. , fig. ).\nDue to the possibility of a further primary tumor or distant metastasis, a bone scan and computed tomography (CT) were performed; the bone scan was normal, and the thoracic-abdominal CT scan did not show any signs of distant metastasis or another primary tumor. Adjuvant combination chemotherapy with cisplatin and ifosfamide (plus mesna) was done for six cycles every 3 weeks, owing to the histology and grade of the tumor. At the end of the therapy, physical examination, thoracic-abdominal CT and echo were completely normal, without a clue for recurrent disease.\n9 months after the last cycle of chemotherapy, the patient presented to our clinic with left hemiparesis and abnormal speech. A CT scan of the cranium showed a central pontine mass (3.3 × 3 × 2.5 cm) extending to the right (fig. ). The radiologists diagnosed the mass as a metastatic lesion. Neurosurgery consultation did not recommend surgery because of the close proximity to vital nervous structures. The patient was given radiotherapy with a dose of 37.8 Gy. However, during the course of hospitalization, his clinical status worsened, loss of consciousness occurred and he eventually died because the tumor compressed the vital structures at the brain stem. |
A 32-year-old man was brought to our emergency department, having sustained extensive trauma of the right eye and orbit after being hit by a steel pipe for 3 h. On physical examination, the patient exhibited marked periorbital swelling unilaterally and a full-thickness, oblique laceration on the right lower eyelid. Upon retraction of the right eyelid, marked conjunctival edema and hemorrhage were noted, and no globe was found on visual inspection. The patient had a Glasgow coma score of 15 with no evidence of any neurological dysfunction. No cerebrospinal fluid (CSF) leakage was noted. A computed tomography (CT) scan revealed that the slightly compressed, deformed globe had extruded into the anterior cranial fossa through large fractures in the orbital roof, accompanied by cerebral contusion (Fig. a, b). No fractures were noted in the optic canal, but the optic nerve appeared kinked (Fig. b).\nFollowing the diagnosis, the patient was immediately transported to the operating room for globe repositioning and repair of the skull base under general anesthesia. The dura was opened, and the eyeball was found situated at the inferior surface of the frontal lobe. After removal of the thin bone fragments in the skull base, the neurosurgeon made two attempts to push the globe back into the orbit cavity. However, the eyeball could not be repositioned and slowly retruded into the cranial cavity.\nNext, an orbital exploration was performed using a transconjunctival approach. After dissecting the orbital rim through a pre-existing tissue laceration, the globe was noted to have been displaced superiorly with exposure of the sclera. Forceps were used to grasp the residual Tenon’s capsule attached to the eyeball to guide the globe inferiorly, from the orbital roof into the orbital cavity, and then to rotate the eyeball with the cornea facing forward. Care was taken not to abruptly retract the globe in order to avoid incurring any additional injury to the optic nerve.\nThe status of the extraocular muscles was next assessed. The medial and inferior rectus muscles were torn, nearly from the middle of the muscle belly. However, we were unable to find the distal sections of the medial and inferior rectus muscles. On gross examination, we noted no signs of eyeball rupture and the pupil appeared almost normal with slight hyphema.\nAfter removing the globe, CSF leakage was noted. Debridement and irrigation were performed at the wound tract. Some bone fragments were placed back at the bone-defective region. The dura was sutured as naturally as possible. Periosteal flaps with pedicles were then transferred to the anterior cranial fossa to cover the dural defect. Biomedical fibrin glue was injected into the flaps to enable adherence. Next, an artificial duramater was used to reinforce the repair. Finally, the wound was closed and a dressing was applied.\nThe patient received intravenous antibiotics and a high dose of corticoids. Tropical steroid drops were used as anti-inflammatories. One day after the surgery, the patient showed signs of ptosis of the right eye. On lifting the upper eyelid using a cotton swab, he reported seeing signs of hand movement in front of his right eye. On a bedside slit-lamp examination, his cornea was clear, blood clots were present in the anterior chamber with a nonreactive pupil (4.0 mm), and the intraocular pressure was 11 mmHg. A CT scan was performed on the first postoperative day, revealing the correct anatomic positioning (Fig. c). The optic nerve was seen to be continuous (Fig. d). One week postoperatively, the blood clots in the anterior chamber were partially resolved with an increase in visual acuity of 2/20, despite continued ptosis. Three months following surgery, there were no abnormal findings in the anterior or posterior segments, except for slight symblepharon and a nonreactive pupil. The patient has undergone 12 months of follow-up during which his visual acuity increased to 12/20. The globe had slightly deviated to the inferolateral side from the primary position, but infraduction, abduction, and adduction were proven to be possible as shown in the video (Additional file ). However, the ptosis persisted and was associated with complete loss of supraduction. The patient gradually developed moderate enophthalmos. |
A 55-years-old male weighing 60 kg was diagnosed with bilateral vitreous hemorrhage and scheduled for pars plana vitrectomy. He was known hypertensive since 1 year and was managed with oral amlodipine (5 mg) twice a day and losartan (50 mg) once a day. Since 4 months, patient started having progressively increasing loss of vision and memory loss which was followed by sudden onset headache associated with vomiting and subsequently loss of consciousness. Pupils of left eye were dilated and sluggishly reacting while right eye was of normal size and sluggishly reacting. Magnetic resonance imaging (MRI) of head revealed left frontal intracranial hemorrhage with intraventricular extension. Diagnosis of stroke with left frontal bleed was made. He was kept on conservative management with continuation of antihypertensive drugs. The patient's consciousness gradually improved. GCS was E4 M6 V5, motor power was 5/5 with down going planters. He had a full recovery after 1 month. The vision did not improved and there was progressively increasing loss of vision. On further evaluation, it revealed hypertensive bleed in the eye and magnetic resonance angiography was done which could not rule out aneurysmal bleed. He was planned for pars plana viterectomy.\nPreoperatively, patient was conscious and maintaining vitals. His routine investigations including 12 lead electrocardiogram, chest X-ray, hemogram, liver, and kidney function test were normal. His antihypertensive drugs were continued and was premedicated with oral alprazolam (0.5 mg) night before the surgery. He was also administered oral ranitidine (150 mg) night before the surgery and in the morning after the surgery. A fasting status of 6 h for solid food and 2 h of clear fluid was advised.\nIn the operating room, monitors including 5 lead electrocardiogram, non invasive automated blood pressure, and pulse oximeter were attached. After securing intravenous access, intravenous midazolam (1 mg) and fentanyl (120 μg) were administered. Anesthesia was induced with propofol (120 mg) and lungs were ventilated with 3% sevoflurane in nitrous oxide and oxygen (50:50). After achieving neuromuscular blockade with rocuronium (40 mg) and intravenous administration of preservative lidocaine (60 mg), airway was secured with proseal laryngeal mask airway (PLMA). Anesthesia was maintained with sevoflurane in nitrous oxide and oxygen (MAC 1) and boluses of rocuronium (10 mg) and fentanyl (20 μg). After the surgery, the residual neuromuscular blockade was reversed with neostigmine (2.5 mg), glycopyrollate (0.4 mg) and PLMA was removed after adequate respiratory efforts were present. The patient had an uneventful recovery and discharged 3 days later for further follow up in ophthalmology and neurology clinics. |
A 53-year-old male patient presented to the ED with macroscopic haematuria during therapeutic anticoagulation with rivaroxaban after multiple unprovoked DVT’s of the right leg in the medical history and no other known diseases. The patient did not report any recent flight travel, surgeries, PE, or malignant tumours. Family history was negative for malignant tumours. On clinical examination, residual swelling oedema of the right lower limb was observed. For further clarification, an abdominal CT scan showed a staghorn calculus in the left kidney. Despite therapeutic anticoagulation, the patient developed multiple left-sided DVTs. Consecutively, onset of fever and concomitant atrial fibrillation (AF) was reported. To detect possible pulmonary infection, a chest radiograph was performed which showed a transparency reduction in the right upper lobe. A CT scan of the chest confirmed a lung tumour and subsegmental PE. Histological workup of an endobronchial biopsy revealed the presence of a bronchial adenocarcinoma.\nFor exclusion of structural cardiopathy as aetiology for the newly diagnosed tachycardic AF, we performed a TTE, showing ventricular hypertrophy but otherwise no sign of intraventricular masses. Nevertheless, the quality of the examination was limited due to tachycardic AF.\nAfter successful cardioversion into sinus rhythm, abdominal sonography for assessment of possible hepatic metastases revealed an abnormal mass within the right ventricle. Since it was unclear whether the mass was the result of another thrombotic event or a metastasis of the adenocarcinoma, we performed CMR for further clarification. Cardiac magnetic resonance imaging detected a highly mobile intraventricular, multi-lobular mass in the right ventricle measuring 21 mm × 18 mm, partly fixed on the septum and on the moderator bundle (Video 1). The mass showed an isointense signal in T1w and T2w without EGE and LGE, thus indicating thrombotic nature of the mass.\nDue to the complex situation with PE and bronchial adenocarcinoma, surgical removal of the RVT was initially considered but due to cardiopulmonary instability and poor prognosis, finally declined. Therapy was switched to therapeutic dose of unfractionated heparin, under which we observed a progression and development of new bilateral PE. Finally, the anticoagulation was changed to therapeutic dose low molecular weight heparin (Dalteparin), whereafter no further thrombotic events were observed. Follow-up CMR after 3 months showed complete resolution of the RVT. |
An 11-year-old German boy, resident of India, presented with firm, reddish lesions on the scrotum of 2 months duration. The lesions were mildly itchy and had started as a solitary, reddish, raised lesion and later progressed to involve the anterior part of the scrotum, the root of the penis, and the areas adjacent to it, in an annular pattern over a period of 2 months. He was applying a mild topical corticosteroid with partial relief to itching, but no regression of the lesions. There was no history of similar lesions or any itching in family members and no contributory symptoms such as fatigue, fever, headache, or pain. There was no history of preceding insect bites or skin lesions.\nClinical examination revealed intensely erythematous linear plaque, 3-cm in length on the anterior aspect of the scrotum, alongside the median raphe on the left. The plaque progressed along the root of the penis and the region adjacent to it in an annular pattern. A single erythematous nodule was also present on the right side of the median raphe [Figure and ]. There was no regional lymphadenopathy and no other lesions elsewhere on the body. Systemic examination was non contributory. At this point, on the basis of the history and clinical examination, a differential diagnosis of a persistent nodular insect bite reaction, granuloma annulare, lymphocytic infiltrate, or cutaneous sarcoidosis was made. Routine hematological examination was normal. Chest X-ray, Mantoux test, and serum angiotensin converting enzyme (ACE) levels were normal.\nOn histopathological examination a diffuse, dense infiltrate of lymphocytes was seen throughout the dermis, admixed with histiocytes []. The lymphocytes revealed no atypia. Immunohistochemical analysis revealed diffuse staining for CD 20 and variable staining for CD 3 in the small lymphocytes. CD 30 cells were negative [].\nOn further probing, the parents revealed of having taken a camping trip to the Shenandoah Valley in the West Virginia region in southern United States, after which they travelled to Germany where the lesions appeared. After their return to India, 2 months later the lesions had progressed to the current size.\nThis urged us to consider the possibility of borrelial LC, and an enzyme-linked immunosorbent assay (ELISA0 test for Borrelia burgdorferi antibodies was done, which was positive for both IgG and IgM antibodies. This was followed-up by western blotting, wherein the patient tested positive for IgG antibodies (8 of the 10 bands were reactive) and negative for IgM antibodies (1 of the 3 bands were reactive). This was suggestive of an acute persisting infection. The patient was subsequently put on amoxicillin 500 mg, three times daily for 1 month. After 3 weeks, the lesions showed a partial remission, wherein the erythema and infiltration had decreased significantly [Figure and ]. |
A 66-year-old female, mother of two children was admitted to our hospital with a progressive swelling of the abdomen over 3 years, numbness and darkness of both lower limbs for 3 weeks. She had attained menopause 15 years back and there was no history of any post-menopausal bleeding. There was no history of headache, giddiness, or blurring of vision. There was no history of easy bruising or bleeding. There was no history of loss in appetite, general malaise, fever, persistent cough, weight loss, any breathing difficulty, chest pain. There were no neurological or cognitive deficiencies. There was no history of pruritus post-bathing, history of residing in high altitude or history of smoking or taking pan or any addictive substances. She was diagnosed of having hypertension 2 years back and was on treatment for the same. There was no history of diabetes mellitus, bronchial asthma, hypercholesterolemia, coronary artery disease. There was no history of any malignancies or tuberculosis in the past. There was no history of long term intake of any drugs. There was neither history of any malignancies in the family nor any hematological disorders in the family. There was no other relevant past history. She gave history of intake of mixed diet.\nOn examination, she seemed in good health, was moderately built and moderately nourished. She had erythema of the conjunctiva, no icterus, cyanosis, clubbing and edema or generalized lymphadenopathy. Her weight was 59 kg. Her blood volume was estimated to be around 4500 ml. Her blood pressure was 140/90 mm Hg. Her pulse was 84/min and regular. She was afebrile and had a respiratory rate of 12/min. Systemic examination of the heart, lungs and central nervous system revealed nothing abnormal. Abdominal examination revealed an irregular mass arising out of the pelvis and reaching to the level just above the umbilicus. It was firm, dull to percussion and non-tender, with restricted mobility. There was no evidence of ascites. The liver and spleen could not be palpated.\nOn pelvic examination, the pelvis was found to be occupied by the mass and it was about 28 weeks in size. The uterus could not be felt apart from the mass, but, by displacing the whole mass upwards with the abdominal hand, the cervix was felt to move with it and the mass was, therefore, considered to be part of the uterus. The adnexa could not be palpated. Rectal examination confirmed that the pouch of Douglas was completely occupied by the mass. |
A 63-year old woman presented with fever, night sweating, weight loss and generalized lymphadenopathy since 9 months. She had a history of hypertension and hypothyroidism on treatment. The patient had no history of a hematologic illness and no family history of any hematologic disease. The patient had been investigated in different hospitals without any definitive diagnosis. On physical examination: She was stable, both chest and heart were clinically free, generalized lymphadenopathy including cervical, axillary and superficial inguinal lymphadenopathy were noted and abdominal examination showed hepatosplenomegaly. On admission, CBC: WBC 5.2×109/L, hemoglobin 11.2 g/dL, and platelets 286×109/L and no abnormal cells were detected in the peripheral blood smear. Liver function tests were normal. Hepatitis and tumor markers were negative. The work up for tuberculosis and other infectious diseases were negative. Computed tomography (CT) of the chest, abdomen and pelvis revealed generalized lymphadenopathy including mediastinal, abdominal and pelvic lymph nodes. The largest lymph node measured about 2 cm in short axis. The liver and spleen were enlarged; measured about 24 and 16 cm in length respectively; also an extensive illdefined sclerotic lesions throughout the included skeletal system were noted ().\nThe impression of CT scan findings was suggestive of lymphoma/leukemia along with the extensive skeletal involvement. Accordingly, lymph node and liver biopsy were done and later on BM examination was performed.\nLymph node biopsy showed no evidence of malignancy or granuloma, however, large abnormal cells with multilobated, hyperchromatic nuclei and abundant cytoplasm were seen scattered in the lymph nodes. These abnormal cells were proved to be megakaryocytes by morphological assessment and immunohistochemical stains for CD61 and FVIII. Some megakaryocytes showed abnormal morphology and nuclear atypia. In addition, granulocytic precursors were clearly illustrated by myeloperoxidase (MPO) stain, and no erythroid precursors detected (Figure 1B-D). The diagnosis was determined as EMH in the lymph node, primarily consisting of megakaryopoiesis and granulopoiesis. Liver biopsy was negative for malignancy or granuloma and showed grade I inflammation and stage II fibrosis with no evidence of EMH ().\nBased on the lymph node result, the patient underwent bone marrow (BM) examination. It revealed a hypercellular marrow for patient’s age with granulocytic and megakaryocytic proliferation while erythroid precursors were markedly reduced. Megakaryocytes showed an atypical morphology with hyperchromatic and hyperlobated forms (). No morphologic evidence of increase in blast cells or presence of abnormal lymphoid cells. Reticulin stain revealed increased of BM fibrosis, MF-1 on grading system proposed by Thiele et al. Flow cytometry confirmed no evidence of BM involvement by acute leukemia or lymphoma.\nMolecular studies using real-time PCR were positive for JAK2 V617F mutation and negative for BCL-ABL1 fusion gene. Conventional cytogenetic studies showed normal karyotype (46, XX); Fluorescence in situ hybridization studies (FISH) was negative for BCR/ABL1 as well. The final patient’s diagnosis was reported as myeloproliferative neoplasm (MPN), consistent with PMF.\nThe patient’s prognostic risk category according to International Prognostic Scoring System (IPSS) was intermediate-1 due to presence of constitutional symptoms at diagnosis. Initially, he was started on Hydroxyurea 500 mg daily which resulted in mild improvement of symptoms with poor control of lymphadenopathy and splenomegaly despite escalating of the dose to 1 g daily. Eventually Ruxolitinib 10 mg twice daily was administered resulting in a dramatic symptomatic response and marked reduction of spleen size and lymph nodes. Unfortunately, the patient lost the follow up after 3 months of Ruxolitinib treatment. |
Patient 2 was a 65-year-old man who received bilateral MoM THA in 2008. He presented for his follow-up visits at 1 and 5 months postoperative. The components were well aligned and he was pain free (Fig. a). The patient presented again for follow-up at 53 months after the index procedure. On the right side, he reported pain in his thigh for the past 3 months. On the left side, he was asymptomatic. The AP pelvis radiograph suggested periprosthetic lucencies in DeLee [] zone 1 and 2 on the right side and in zone 1 on left side, respectively (Fig. b). The CT of the right hip showed an osteolytic lesion extending 5 × 1 cm in the anteromedial acetabulum. On the left hip, CT demonstrated a lesion of 2.2 × 1 cm in the anterolateral acetabulum. The corresponding MARS MRI which was obtained at the same day did not detect any areas of bone resorption in the acetabulum on either side. The patient was scheduled for bilateral hip revision surgery the following months; however, the surgery was postponed because of signs of cardiac ischemia in the preoperative stress test. After successful stent implantation, hip revision surgery was scheduled 3 months later. On the day of revision surgery, an additional AP pelvis radiograph was obtained which showed a loose and displaced right acetabular component (Fig. c). Considering the anticipated difficulty of the surgery and the medical condition of the patient, only the right hip was revised at this point. Intraoperatively, there was a significant amount of fluid collection, but no clear evidence of abnormal soft tissue proliferation. The acetabular component was loose and associated with a large acetabular defect medially, anteriorly and superiorly. The acetabular osteolysis required bone grafting and placement of a cage with a cemented liner (Contour Cage, Reflection cemented Cup, Oxinium head; Smith & Nephew, Memphis, TN). The postoperative radiographs, showed well-aligned components (Fig. d). |
A 53 years-old male visited to our clinic because of cognitive impairment for two years ago. The patient graduated from university, had worked as an office worker and recently retired. His wife described that, the patient’s memory and ability of judgment had been impaired for 2 years, and his verbal output had been decreased. The patient was not taking any medicines previously. Because of forgetting his medication schedule, he attached his medicine on his calendar. Sometimes he forgot to turn off a gas stove. Although he had never lost his ways, he had trouble with driving and made some car accidents because of misperception. There were also some changes in his behavior. He had turned much introversive instead of sociable previously, and showed indifference towards household matters or works related with their offspring. Inappropriate laughing was observed in serious situations. Without a caregiver, he could not care of his own hygiene or eating. However, there were no psychiatric symptoms such as hallucination or delusion.\nOn the first visit, there was no remarkable finding in examination of motor and cerebellar function. A brain magnetic resonance imaging (MRI) showed no remarkable finding. A detailed neuropsychiatric evaluation (Seoul Neuropsychological Screening Battery) was performed. The results were interpreted using 1.0 standard deviation (16 percentile) of normal Korean population aged 55, as a cut-off value. He showed time disorientation, and impairments in encoding, retention and retrieval of verbal memory, and retrieval of visual memory. The frontal lobe functions including executive functions were deteriorated. Considering the progressive decline in intellectual function affecting memory and other cognitive domain, and interference of activities of daily, he was diagnosed with dementia. At the time, the patient was regarded as early frontotemporal dementia. Therefore, we decided to follow up treatment without any medication.\nSix months later, he complained about gait disturbance with a feeling of right leg dragging. In addition, he had suffered from the postural imbalance, and difficulties in swallowing and articulation. He developed the orthostatic dizziness and urinary difficulties, including frequency, nocturia and residual urine sense, had also developed. On neurological examination, he was alert. Cranial nerve examination revealed moderate dysarthria and mild dysphagia. There was no limitation of ocular movements. There were no abnormalities on tests for optokinetic nystamgus or smooth pursuit eye movement. However, hypermetric saccade in eye movement was observed. Motor power was normal. Resting and postural tremor, rigidity, and bradykinesia were not observed. The sensation of all modalities was normal. Deep tendon reflexes were increased in all extremities, Hoffman’s signs were positive, bilaterally. Babinski’s signs were negative. Limb ataxias were observed in both lower extremities. He was unable to tandem gait. The serological tests for syphilis and viruses including HIV were negative. Gene tests for spinocerebellar ataxia type 1, 2, 3, 6, 7, and 17, and dentatorubropallidoluysian atrophy were all negative. The head up tilt test revealed orthostatic hypotension; systolic blood pressure was decreased 31 mm Hg 3 minutes after standing. The volume of residual urine immediately after urination was 110 mL. The neuropsychological test was followed up to evaluate the progression of cognitive dysfunction. Verbal and visual memories and frontal lobe functions, including inhibitory function and semantic word fluency, were significantly deteriorated compared with previous test 6 months ago (). The results of neuro-psychological testing suggested the possibility of frontotemporal dementia. The follow-up brain MRI has showed newly developed atrophy of cerebellum, pons and middle cerebellar peduncle (). Positron emission tomography (PET) was performed using 18F-fluorodeoxyglucose (FDG), and showed decreased cerebral glucose metabolism on the bilateral bagal ganglia, cerebellum, both parietal lobes, and left posterior cingulate gyrus in visual analysis (). Finally, based on the features of cerebellar ataxia, autonomic dysfunction and pyramidal dysfunction, he was diagnosed with MSA-cerebellar type (MSA-C). He was prescribed with rivastig-mine for dementia and other medications for urinary problems. |
A 55-year-old female, with symptoms related to cholelithiasis, was admitted to our hospital. She reported a 20-day history of intermittent right upper quadrant pain radiating to the back, associated with nausea and bloating sensation. Her family history included father with lung adenocarcinoma. Her personal history included right ovary salpingectomy for endometriosis cyst, viscerolysis for bowel obstruction caused by adhesions, and repair of a median abdominal incisional hernia using polypropylene mesh. A quadrantectomy and sentinel lymph-node biopsy for moderately differentiated invasive ductal carcinoma of the breast G2pT1cN0, with estrogens receptors of 90%, progesterone receptors of 85%, and HER2/neu = 1+ and Ki-67 < 14%, was performed one month before. At the time of admission, she had normal vital parameters and laboratory investigations. At the physical examination, tenderness in epigastric region and in the right upper quadrant was revealed with no evidence of jaundice. The patient showed an abdominal ultrasound and contrast-enhance abdominal computed tomography: lumen of the gallbladder was occupied by numerous stones with a nonthickened wall, no evidence of biliary dilatation was noted, and there was no ascites. The chest X-ray revealed no unusual findings. After a laparoscopic access, we carried out, for the presence of numerous adhesions, an open cholecystectomy. There was no evident locoregional lymphadenopathy.\nThe patient had an uneventful postoperative recovery and was discharged home after two days. On gross inspection, the gallbladder measured 10 cm in length, some stones were found in its lumen, and a polypoid, whitish lesion, in correspondence with the bottom, measuring 1,5 cm was found in its body. Final histologic examination revealed a neuroendocrine small cell carcinoma of the gallbladder. Tumour was seen infiltrating into the muscular layer but not through the serosa of the gallbladder. Resected margins were free from tumor. Two lymph nodes measuring about 4 mm were isolated from the surgical specimen: the analysis of one lymph node revealed focal metastatic deposits of neoplastic cells (), whereas in the other one no deposits of tumor cells were found. Immunohistochemical studies revealed cells strongly positive for chromogranin () and synaptophysin () and negative for thyroid transcription factor-1.\nPostoperative imaging studies did not show residual disease either locally or remotely. Patient was subjected to radiotherapy for breast cancer and six cycles of chemotherapy with cisplatin and etoposide were administered. Eighteen months after surgery, the patient was free from disease. |
A 47-year-old male patient was hospitalized because of severe pain and swelling of the right leg.
A cementless total hip arthroplasty was implanted two and a half years before, due to a previous osteonecrosis of the femoral head. In addition, the patient suffered from noninsulin-dependent diabetes mellitus type 2 and hepatitis B. The computer tomography (CT) showed a large cystic formation ventral to the hip reaching the psoas muscle (). The CRP was elevated 23-fold (CRP = 116 mg/L ) and in the puncture S. lugdunensis was cultured from several samples. CT-assisted drain was placed into the cyst and left for several days. A targeted antibiotic therapy with oxacillin (2 g every 8 h) and rifampicin (600 mg every 12 h) was intravenously started and switched to oral application and maintained for two months. During the antibiotic therapy the cystic lesion was decreasing.
Two months later the patient complained again about pain in the right hip. Plain radiographs showed no signs of loosening of the cup or the femoral stem. The CRP was elevated with 76 mg/L. A skeletal scintigram was performed. The enhancement of the synovia in the blood-pool phase and the increase of bone metabolism in the late phase of the bone scintigraphy were interpreted as a sign of a prosthetic infection. In the puncture of the joint itself S. lugdunensis was cultured as the causative pathogen again. In summary these results showed now a periprosthetic infection of the hip joint.
Therefore a two-staged revision of the prosthesis was performed. In the first stage, the prosthesis was explanted and replaced by a gentamicin-containing bone cement spacer into the acetabulum and a gentamicin-containing sponge into the femur. In an intraoperatively taken wound swab S. lugdunensis was cultured again. An antibiotic therapy was started with doxycycline (100 mg every 12 h) and rifampicin (600 mg every 12 h) and maintained for |
L.W. is a 30 year-old woman with MSS. Her medical history was obtained from her parents, who accompanied her and upon whom she was totally dependent for care. The patient had speech and cognitive impairment that limited the ability to obtain a direct history. The parents described a history of worsening hip pain from progressive, bilateral hip dysplasias. Whereas previously their daughter could ambulate with assistance, she was now incapacitated by relentless pain. As best as they were able to determine, the pain radiated from her hips laterally, down her thighs and provoked regular paroxysms of screaming, crying and guarding.\nPrior to our encounter, this pain had been managed by an orthopedic surgeon at an outside facility, who performed a series of nine ultrasound-guided intra-articular hip steroid injections, over a period of several years. The last one was performed a few months before presenting. None of the patient's previous records were available to us, but the parents relayed that their daughter's response to the injections had begun to wane with each repeat injection. The most recent recommendation from the orthopedist was to perform bilateral hip arthroplasties. The parents were hesitant to pursue this option, in light of their daughter's previous surgical experience, wherein she required an emergency tracheostomy after failed attempts at securing her airway under anesthesia. Because they were told that future surgeries would require an "awake" tracheostomy for airway protection during surgery, they decided to seek alternative, non-surgical treatments for their daughter's hip pain.\nThe rest of the review of systems was unremarkable. Notably, the parents denied any history of bleeding diathesis. Though previous diagnostic imaging was not available at the time of our initial consultation, they were reviewed at a later date, prior to treatment, and revealed dysplasia of both acetabula, and severe osteoarthritis and subluxation of both hip joints.\nOn physical examination, the patient was small in stature (4ft 2in, 65lbs) and had obvious craniofacial abnormalities. The neuromuscular exam was limited due to lack of patient cooperation. The greater trochanters were asymmetrical, with the right side about 2 cm superior compared to the left. Both were easily palpable and visibly appeared to be grossly out of socket. Though muscle tone was good with no flaccidity, the patient's inability to obey commands prevented us from assessing motor or sensory function. Reflex testing revealed patellar and Achilles hyporeflexia.\nBased on the patient's history, physical examination, and radiographic findings, our impression was that the patient's symptoms arose directly from the articular surfaces of her hips, and possibly from the bilateral impingement of her lateral femoral cutaneous nerves, as a result of her inadequately developed acetabula and subluxed femurs. We suggested a series of three fluoroscopically guided intra-articular hip injections with sodium hyaluronate (Hyalgan©), administered weekly. In the event of an unsatisfactory result from the injections, we intended to perform bilateral lateral femoral cutaneous nerve blocks. The patient's parents elected to pursue injection of sodium hyaluronate, and scheduled an appointment for the procedure. |
A boy (3 years old) was admitted to our hospital with a 2-year history of snoring and breathing through the mouth. The patient also had inspiratory dyspnea, especially during sleeping. The out-patient diagnosis was tonsil hypertrophy. Inspiratory dyspnea and three concavity signs were apparent, even when the child was calm. Nasoscopy showed adenoid hypertrophy, accounting for two thirds of the posterior nares. A tumor with a smooth surface, which was lying on the posterior wall between the nasopharynx and oropharynx, was also detected. A computed tomography scan with contrast showed irregular hyperattenuation in the prevertebral space of C1–C5, and it was approximately 2.0 × 4.6 × 3.4 cm in size, with heterogeneous enhancement (). After admission, the patient developed severe dyspnea. Because of the possibility of pulmonary infection, he was immediately transferred to the pediatric intensive care unit of our hospital. He was provided oral tracheal intubation and gastric tube implantation. After anti-infection and symptomatic treatment, his symptoms greatly improved and he underwent surgery. We wished to avoid tracheotomy because it can cause surgical injury and affect quality of life of the child. Because the inspiratory dyspnea was severe, we performed tracheal intubation and operated under a mouth gag. During the operation, there was no clear gap between the tumor and pharynx mucosa. The tumor was excised together with the mucosa of the pharynx. The tumor easily bled. The tumor extended backwards, but there was a distinct gap between the tumor and prevertebral space. A rapid pathological examination during the operation showed a malignant tumor originating from mesenchymal tissue. Preoperative evaluation of lymph nodes was clear. No lymphadenectomy was performed because mesenchymal malignancy had spread mainly by the hematogenous route.\nTumor tissue was subjected to histopathological and immunohistochemical analyses (). The pathological results indicated an MPNST. Immunohistochemistry showed the following positive and negative results: Ki-67 (+10%), cytokeratin pan antibody CK-pan (−), vimentin (−), S-100 (focal+), smooth muscle actin (−), h-caldesmon (−), desmin (−), cluster of differentiation 117 (–), discovered on gastrointestinal stromal tumor 1(part+), cluster of differentiation 34 (−), β-catentin (+/−), cluster of differentiation 56 (−), and neuron-specific enolase (−).\nAn indwelling nasogastric tube was inserted and the patient was allowed to eat on the 13th day after surgery. The patient received radiotherapy 30 days after the operation. The dose at the posterior thickened area of the pharyngeal wall was 5600 cGy/33F and the tumor bed area dose was 4760 cGy/28F. Mild stenosis was found between the nasopharynx and oropharynx 3 years after the operation. The narrow band imaging mode of laryngoscopy showed that the area of the tumor was normal ().\nInformed consent was obtained from the patient’s parents for publication, and this study received approval from the Research Ethics Committee of The First Hospital of Jilin University. |
A 21-year-old woman with a history of a biopsy-proven sporadic desmoid tumor within the posterior left thigh was referred for MRgFUS therapy. The patient had previously been treated with sulindac and celexocib therapy; however, she palpated interval enlargement and increased firmness of the mass while taking these medications. A repeat MRI was performed demonstrating interval growth of the mass which measured up to 13.5 cm in maximum dimension, increased from 11.5 cm 1 year prior. Referral was made to radiation oncology. However, there was concern for significant left ovarian radiation exposure and risk of infertility. Referral was also made to orthopedic oncology. However, the patient was advised that resection would be associated with a significant risk of recurrence and damage to the adjacent neurovascular structures within the posterior thigh, including the sciatic nerve.\nAt this time, the patient was referred for MRgFUS therapy. The tumor measured up to 24 × 8.6 × 8.5 cm in maximum dimension with pretreatment volume of approximately 730 cm3 and was associated with medial displacement of the sciatic nerve. Her symptoms at this time included limiting tolerance for sitting and difficulty with strenuous activities that relied on use of the left hamstring group.\nMRgFUS of the tumor within the thigh was performed under general anesthesia. The patient was positioned in the left lateral decubitus position in order to minimize the risk to the sciatic nerve. MRgFUS was performed with the same MRgFUS system. Because despite the rapid growth of her tumor, the patient was relatively high-functioning; only the central portion of the tumor was targeted. The procedure was performed with 38 treatment sonications. Enhanced safety measures were used as described in case 1. Total energy per sonication ranged between 1062 and 1911 J with an average of 1539 J. Each individual sonication was 20 s long. Temperature varied between 52 and 77 °C with each sonication. Thermal dose volume was 25.7 cm3. Postcontrast imaging immediately following the treatment demonstrated a relatively low non-perfused volume of the central half of the mass, which measured approximately 30% of the target or 15% of the entire mass (Table ). Review of the sonication data revealed that 20/39 treatment sonications did not definitely reach 60 °C, including 10/39 which did not clearly reach 57 °C based on the MR thermometry sequences. Overall, the average temperature was 60.8 °C following each sonication. Review of the imaging data demonstrated a subtle fascial band in the soft tissues which potentially contributed to poor heating during the treatment. Several sonications did demonstrate heating accumulating along this interface.\nThe patient returned after 6 months for a repeat treatment. The upper half of the tumor was targeted. A slight obliquity of the left lateral decubitus position was used in order to shift the location of the previously noted fascial band. The procedure was performed with the same treatment setup, and 117 treatment sonications were performed. Because the procedure tolerated the first procedure well without any evidence of a skin injury, significantly increased energy was also used compared to the prior treatment with the energy per sonication ranging between 1474 and 6026 J with an average of 3861 J. Thermal dose volume measured 74.3 cm3. There was approximately 70% non-enhancing volume within the targeted superior 1/2 of the mass (where there had been the most interval growth following the prior treatment) (Fig. ). Interestingly, despite the increased non-perfused volume, average temperature was slightly lower in this treatment with an average of 59.0 following each sonication. The patient experienced no complications following either treatment. |
A 70-year-old woman was admitted to the Hepatobiliary Department of Tianjin Medical University Cancer Institute and Hospital, with complaints of abdominal satiety combined with non-specific pain in the right upper quadrant, without nausea, vomit, dyspepsia, and weight loss. The patient was diagnosed to have a liver cyst for approximately 40 years, without a history of congenital cysts. Thirteen years ago, the patient was treated by percutaneous transhepatic drainage, which was guided by ultrasonography. Two years after drainage, the hepatic cyst was found to recur during the follow-up. At this time, the cyst was approximately 3 cm in size, as shown by ultrasonography. However, no treatment was administered because the cyst was asymptomatic. During the subsequent interviews, the patient recalled the obscure onset of intermittent seizures that lasted for approximately five months.\nUpon physical examination, a mass was palpated in the patient’s right upper abdomen. Laboratory examination showed that the parameters of routine hematological and biochemical examinations, including tests of liver and renal function, were within normal limits. Similar results were obtained for the expression levels of tumor markers, including the blood carcinogenic embryonic antigen (CEA) and the alpha-fetoprotein (AFP). The hydatid serology test was negative. Abdominal ultrasonography identified a cyst located in the liver measuring 30 cm in diameter. The abdominal contrast-enhanced MRI-scan likewise revealed a large, thick-walled septus and cystic lesion in the liver, measuring 27.9 cm × 17.9 cm × 14 cm, which occupied almost the entire liver. A slight enhancement of the septum was present after the injection of the intravenous contrast agent. The diagnosis of cystadenocarcinoma could not be completely excluded ().\nGiven the history of recurrence and the unpreclusive diagnosis of cystadenocarcinoma, open surgery was scheduled for the patient one week after her admission. At laparotomy, a cystic mass of approximately 30 cm in size was identified and located in the liver. Excision was conducted from the liver capsule between the cyst and the liver parenchyma. Clear fluid was drained from the cyst when the cyst wall was opened. The cystic and septus structure was observed and the bilious fluid was partially drained out. The diagnosis of hepatic cyst with biliary communication was determined based on its macroscopic appearance.\nIntraoperative cholangiography was employed to determine the existence and position of biliary fistula. A silica gel tube was inserted via the cystic duct after temporarily closing the distal end of the common bile duct with a clamp. By injecting the contrast medium (iopromide), we identified the bile ducts that communicated with the cyst. The results showed two extravasations of the contrast medium from the hepatic bile duct branches ().\nWe then performed cystic wall resection and closed the communicated bile ducts by careful suturing. The contrast medium was subsequently reinjected, but leakage from biliary tract was no longer detected. Finally, we used omental tissue to cover the wound surface and close the abdominal cavity.\nPathological examination confirmed the diagnosis of a hepatic cyst (). Laboratory examinations such as the routine hematological, hepatic, and renal functions were within normal limits at one week after the operation. Ultrasonography revealed no abnormalities. The patient was discharged on the 9th postoperative day with no sign of cholangitis or other symptoms. |
A 69-year-old Japanese woman was referred to our department in August 2015, complaining of swelling in the right preauricular region. She had shown a mass lesion of the right lung on a chest x-ray and was referred to the university hospital. Fluorodeoxyglucose-positron emission tomography (FDG-PET) examination showed accumulation indicative of a pulmonary lesion and a temporomandibular joint lesion. The temporomandibular joint lesion had been enlarging but was otherwise asymptomatic. Her medical history included treatment for pulmonary tuberculosis at the age of 13 and surgery to remove breast cancer at the age of 53.\nOn examination, a tender mass in the right preauricular region was palpable. The chin of the mandible was deviated to the right side during mouth opening (mandibular maximum mouth opening, 41 mm). Facial nerve function and mandibular nerve were intact. There was no indication of cervical lymphadenopathy.\nA panoramic radiographic examination showed resorption of the right mandibular condyle to the ramus (Fig. ). Computed tomography (CT) showed destruction of the right mandibular condyle and a large mass lesion with enhanced margin in the masticator space; a cystic lesion was present inside the tumor mass. Three-dimensional CT was useful to understand the bone resorption findings of the mandibular condyle. There was no finding of metastatic cervical lymph node (Fig. ). T1-weighted magnetic resonance imaging showed an enhanced mass lesion in the right masticator space. Because some portions of the mass lesion showed high intensity in T2-weighted images, cystic lesions were suspected to exhibit changes of blood flow or retention of high-protein liquid. Tumor development was not observed in the articular disk of the temporomandibular joint (Fig. ). FDG-PET revealed abnormal FDG uptake in the right submandibular condyle and masticator space.\nA malignant tumor was suspected after analysis by various modalities. Therefore, we performed an incisional biopsy via preauricular incision. Histopathologically, the tumor was largely composed of proliferative, atypical, spindle-shaped cells. Some tumor cells showed increasing mitotic change and extreme atypia (Fig. ). The histopathological findings of biopsy suggested spindle-cell sarcoma. Tumor resection was performed with the patient under general anesthesia via a combined preauricular and transmandibular approach to the masticator space and infratemporal fossa.\nAfter preauricular temporomandibular incision, the superficial temporal fascia and temporal fascia were elevated. The facial nerve (temporal and zygomatic branches) was protected by the fascia (Fig. a).\nThe facial nerve trunk was identified, and its branch was traced according to the conventional method.\nA midline lip-splitting incision was connected to the submandibular incision and accessed the anterior mandibular ramus. The lip-split incision traversed to the periosteum of the mandible; the periosteum dissector was used to elevate the soft tissues of the mandible, and the lateral mandible was exposed. The branch of the facial nerve was traced continuously with the skin flap, and the masseter muscle and deep parotid gland were dissected on the tumor side (Fig. c). The mandible was resected to within ≥ 20 mm from the primary tumor on the anterior of the mandibular ramus. The masseter muscle was divided at the lower edge of the zygomatic arch; further, the temporal muscle was divided horizontally at the height of the zygomatic arch to reach the side of the temporal bone. Excision was performed on the temporal bone to the base of the pterygoid process, and an osteotomy was performed continuously from the maxilla to the pterygoid process. Osteotomy was performed via ultrasonic scalpel. Then, deep excision was performed on the skull base until the foramen ovale was reached. The mandibular branch (VIII) of the trigeminal nerve was resected at the foramen ovale, and bone wax was used to fill the foramen ovale when hemostasis was achieved. The tumor was excised in bulk with the surrounding tissue (Fig. d). Especially, the glenoid fossa was close to the primary tumor site; however, it was excised including the joint disc, and the glenoid and infratemporal fossa were excised, including the periosteum of the skull base. For that reason, the margin was considered complete, and we did not perform the histopathological evaluation intraoperatively.\nThe surgical defect was reconstructed with a free vascularized fibula with skin paddle (Fig. e). The occlusion was performed with intermaxillary wire fixation for 1 week postoperatively. There were no abnormal findings during the postoperative course with complete healing. Right-sided facial nerve dysfunction appeared immediately after surgery.\nThe resected specimen exhibited nearly identical histological findings as observed in the biopsy. Because it involved a periosteal reaction corresponding partially to Codman’s triangle, the tumor was thought to have derived from bone (Fig. ). Immunohistochemical analysis showed positive staining for vimentin, MIB-1 index (40–80%), desmin, α-smooth muscle actin, Bcl-2, neuron-specific enolase, and S-100; it showed negative staining for AE1/AE3, caldesmon, and CD34. Thus, the final diagnosis was osteosarcoma (fibroblastic). The resection margin was negative for tumor. After excision of the mandibular tumor, excision of lung cancer was performed under thoracoscopy by a respiratory surgeon in our hospital. Although we recommended adjuvant treatment after surgery, the patient refused this treatment.\nThere has been no evidence of local recurrence or distant metastasis through 30 months of follow-up. The chin of the mandible deviates to the right side during opening (maximum mouth opening, 40 mm). Centric occlusion has not changed. Facial nerve dysfunction gradually improved and became mild according to House-Brackmann Scale evaluation (Figs. and ). |
A 61-year-old woman presented with dyspnea (New York Heart Association class 2), chest pain and lower limb edema. In past medical history, she was under treatment for hypertension, hyperlipidemia, and acute coronary syndrome (ACS) and she had a history of hospital admission twice for chest pain and ACS. Despite the hospital admissions, cardiopulmonary assessment was not perfectly performed to diagnose the main cause of her chest pain. At the first visit, initial blood pressure was 110/70 mmHg, heart rate: 68/min, respiratory rate in normal range, and body temperature was 37 °C. She did not have any complaint of productive or dry cough, fever, nocturnal dyspnea or orthopnea.\nHer physical examination revealed an obvious systolic machinery murmur, which was best-heard at the second left intercostal space, and the second heart sound was physiologically split suggesting an absence of significant PHTN. Pulses and diastolic murmurs were near to normal. Pulmonary rates and wheezing could not find in any of both lungs, but bilateral pretibial pitting edema was obviously seen. Echocardiogram showed; normal left ventricular chamber size and function, normal size of both atria. Furthermore, an obvious PDA (diameter = 6-7 mm) connecting the proximal of descending aorta to the left to the pulmonary artery was reported in echocardiography ( and ).\nFor more accurate evaluation, a transthoracic echocardiogram (TTE) was performed, and it showed visualized jet flow at main pulmonary artery, indicating the presence of PDA. TTE also showed mild mitral and tricuspid regurgitation and showed mild diastolic dysfunction but preserved left ventricular function (ejection fraction of the left ventricle = 55%). Patients referred for cardiac catheterization and computed tomographic (CT) angiography. CT angiography was revealed a PDA with 5.5-7 mm luminal width interposed between the roof of left pulmonary artery and descending aorta (both at origins) ().\nNo lung congestion and evidence for PHTN was reported by CT angiography (pulmonary capillary wedge pressure: 30 mmHg). Saturation study showed a step up from right pulmonary artery to the right ventricle (72-60 %). Measured pulmonary to systemic blood flow ratio (Qp/Qs) through oxygen saturation was 1.26. Renal dysfunction, thyroid imbalance, and other etiology of edema were ruled out. The patient was treated with aspirin (80 mg/day), Lasix (tablet, 40 mg/day), losartan (tablet, 0.5 twice a day), atorvastatin (tablet, 20 mg/day), nitrocontin (tablet, 2.6 twice a day), and atenolol (tablet, 50 mg daily). After 1 and 3 months follow-up, edema and other symptoms were resolved. |
A 50-year-old male patient with aortic dissection originating just above the aortic valve and extending down to the common iliac arteries (Stanford A, Figure ) underwent immediate surgery with repair of the ascending aorta in moderate hypothermia under cardiopulmonary bypass. The arterial line was inserted in the right axillary artery, the vent was placed in the right upper pulmonary vein, and two-stage venous line was inserted through the right auricula. The intact aortic valve was resuspended. Extracorporeal circulation was suspended after induction of moderate hypothermia (25°C) and the aortic cross-clamp from the ascending aorta was removed. The false lumen was then glued and an open distal anastomosis to a prosthetic graft was constructed. Then, extracorporeal circulation was resumed, systemic circulation was deaired and the patient was warmed.\nEarly on the first postoperative day, CT of the aorta was requested because of marked elevation of lactate-dehydrogenase (129 μkat/l) and signs of acute renal insufficiency (creatinine 292 μmol/l). CT showed good postoperative result in the ascending aorta, but large amount of air in the branches of the superior mesenteric artery up to the arcades was found (Figure ). There was absolutely no air in the portal-venous system including the liver. The bowel loops were not distended, and there were no signs of bowel paralysis.\nAt midnight on the first postoperative day, a biphasic CT scan of the abdomen was requested due to elevated intra-abdominal pressure (18 mmHg). The CT showed distribution of the intra-arterial gas more into the periphery and into the wall of the bowel loops that still did not display signs of ileus (Figure ). The next morning, surgical exploration was performed due to increasing intra-abdominal pressure as a sign of imminent abdominal compartment syndrome. An extensive bowel resection from oral ileum down to the splenic flexure had to be performed due to extensive necrosis of the bowel. Shortly after the operation, the patient became hypotensive with signs of overwhelming vasoparalysis and died on the third postoperative day of multiple organ dysfunction syndrome (MODS) with systemic inflammatory response syndrome (SIRS). |
The patient is a 71-year-old white male who was found to have a 3.5 cm right kidney mass and had been followed by the urology team closely at VA Pittsburgh Healthcare System. Urine cytology was suspicious for malignant cells. He underwent a radical right nephrectomy on February 3, 2014. Pathology showed clear cell RCC. The tumor was located at the lower pole with a size of 4.5 cm (pT1b) and Fuhrman nuclear grade 2. All margins were not involved by carcinoma, and there was no vascular invasion. He had been followed with a regular CT scan every year. He was found to have small bilateral lung metastasis and lymphadenopathy in 2016. The PET scan on April 26, 2016, revealed FDG activity in the lung and hilar and mediastinal lymph nodes. He underwent endobronchial ultrasound biopsy of the mediastinal lymph node which confirmed to be metastatic from clear cell RCC. Due to his comorbidities and mild thrombocytopenia, we started him on lower dose sunitinib at 37.5 mg per oral daily ×4 weeks every 6 weeks in May 2016. In total, he received 7 cycles of sunitinib. He had been followed every 6 weeks in the clinic. He only developed fatigue due to mild hypothyroidism for which he received levothyroxine. During the follow-up, he was found to have worsening thrombocytopenia with platelet counts in the range of 60,000 to 90,000. A follow-up CT scan and PET scan in October 2016 showed improvement of the lung metastasis and lymphadenopathy. He was last seen in the clinic on March 13, 2017.\nHe was admitted on March 29, 2017, due to muscle weakness, fatigue, poor oral intake, and difficulty swallowing for 2 weeks. During admission, his platelet count was found to be 13,000, serum creatinine 2.3, total bilirubin 4, AST/ALT > 2000, INR 2.9, calcium 7.5, creatine phosphokinase (CPK) > 5000, and uric acid 12 (see ). Sunitinib was discontinued on the first day of admission. CT head revealed no evidence of metastatic disease. Chest X-ray did not show evidence of infiltration or effusion. Echocardiogram showed severe global hypokinesia with LVEF of 30–35%. His LVEF was 55% prior to starting on sunitinib. He quickly developed lactic acidosis and acute respiratory failure. In the intensive care unit, he received bicarbonate, high-dose oxygen, furosemide, and treatment for hyperkalemia. Despite all treatment support, he continued to decline. His family chose to deescalate care, and he died on April 1, 2017. |
A 16-year-old Caucasian female was transferred from an outside hospital with 2 weeks of worsening swelling and pain of a left-sided neck mass. The patient experienced fatigue and intermittent low-grade fevers in the 4 months leading to her admission. The symptoms were attributed to a previous diagnosis of Lyme disease which was treated appropriately with a course of doxycycline prior to the onset of neck swelling. In the 2 weeks preceding her admission, she noticed a “lump” on the left side of her neck. The mass started as a small bump under the skin in the anterior neck and grew larger with increased tenderness. In addition, over the week prior to admission, she became febrile, experienced trismus, limited range of motion of the neck, as well as began to experience multiple episodes of abdominal pain and vomiting.\nThe patient sought treatment from several providers prior to presentation with an ultimate diagnosis of sialadenitis for which azithromycin was prescribed. After 2 days of azithromycin, there was no improvement in the size of the mass nor her pain. Initial laboratory investigations at an outside hospital revealed lymphocytosis and elevated liver enzymes. Computed tomography (CT) of the face/neck with intravenous (IV) contrast demonstrated a 4.8 cm by 3.9 cm by 2.5 cm mass interpreted as a branchial cleft cyst anterior to the left sternocleidomastoid. At this time, the patient was transferred to the Cohen Children’s Medical Center of New York for further evaluation and management.\nOn admission, her vital signs were within normal limits and there were no signs of respiratory distress. The left-sided neck mass was described as a 4-cm non-fluctuant, matted mass, located just lateral to the laryngeal prominence extending posteriorly to the subauricular region with associated tenderness but without overlying skin changes. Bilateral tonsillar enlargement (left greater than right), erythema, and exudate were also noted. Her initial laboratory findings are summarized in . Her laboratory testing indicated acute CMV and EBV coinfection. Bartonella serology was negative. Her manual smear demonstrated increased reactive lymphocytes and large granular forms. During the patient’s hospital course, additional imaging included ultrasound of the head/neck which demonstrated a possible necrotic left-sided lymph node with multiple enlarged lymph nodes bilaterally. Upon further evaluation, repeat CT neck/soft tissue demonstrated a 4.2 cm by 4.1 cm by 3.5 cm necrotic lymph node, bilateral cervical chain lymphadenopathy, partially visualized mediastinal, supraclavicular, and axillary lymphadenopathy, as well as a right upper lung opacification. Further imaging to investigate the extent of lymphadenopathy included CT chest, abdomen, and pelvis with contrast, revealing enlarged thoracic and abdominal pelvic nodes, hepatosplenomegaly, moderate bilateral pleural effusions, small pericardial effusion, and abdominopelvic ascites (). To further elucidate the origin of this systemic involvement, the Infectious Disease, Hematology/Oncology, and Otolaryngology subspecialty teams unanimously recommended a tissue biopsy of the large left-sided cervical lymph node demonstrated on CT. Pediatric General Surgery completed guided biopsy of the cervical lymph node which was successfully obtained without any complications. Pending biopsy results, the patient completed a course of clindamycin as an inpatient and was discharged home in stable clinical condition.\nGram stain and culture of the specimen did not reveal any organisms or growth. Similarly, no organisms were visualized under acid fast smear. Flow cytometry was not performed due to insufficient number of viable cells. However, hematopathologic evaluation of the specimen described necrotizing lymphadenitis. |
A 30-year-old lady congenitally deaf and mute presented with history of headache since one month and right LMN facial palsy since last 20 days. Using the sign language, she indicated that at the onset headache was predominantly over the anterior part of head and within few days it became diffuse. It was pressing in character, present throughout the day and associated with vomiting on increased severity. Ten days after the onset of headache she noticed difficulty in closing her right eyelid and three days later deviation of angle of mouth to left. There was no history of fever, ear discharge, weakness of limbs, seizures, diplopia, blurring of vision, ataxia, tinnitus or recent worsening of deafness. Physical examination showed presence of pallor, no organomegaly, external ear, mastoid and eardrum were normal. She was conscious, alert and oriented. Her visual acuity was 6/9 on both sides. Examination of the optic fundi revealed presence of bilateral papilledema. Eye movements and pupillary reactions were normal. She had right LMN facial palsy with absent taste and tearing ipsilaterally with bilateral sensory neural deafness as evidenced by tuning fork tests. Rest of the cranial nerves, motor, sensory and cerebellar examinations were normal. Clinically she was diagnosed as a case of raised intracranial pressure (ICP) with right LMN facial palsy with congenital deaf mutism.\nInvestigations revealed hemoglobin of 7.1 gm%, microcytic hypochromic blood picture with normal white blood cells and platelets. Serum biochemistry and electrolytes were normal. Rheumatoid factor and antinuclear antibodies were negative. Serum antiphospholipid antibodies, homocysteine levels, angiotensin converting enzyme levels and chest X-ray were normal. Computerized tomography (CT) scan of the brain showed posterior empty delta sign in the superior sagittal sinus. Temporal bone and middle ear cavity were normal. MRI and MRV [] showed thrombosis of superior and right lateral sinus, with no parenchymal lesions. Lumbar puncture was normal except with raised opening pressure. Audiometry showed bilateral profound sensory neural hearing loss. Brain stem auditory evoked responses were absent bilaterally. She was diagnosed as a case of CVT (superior and right lateral sinus) with ipsilateral LMN facial palsy with congenital deaf-mutism. She was started on parenteral followed by oral anticoagulation (prothrombin time, international normalization ratio maintained at about two), hematinics, anti edema measures. Over the next one month her symptoms of headache and facial palsy improved and at 4 months after her symptom onset, she was asymptomatic. |
A 55-year-old incarcerated male presented to the emergency room with a two-week history of left-sided scrotal pain and swelling.\nThis patient had a history of prostate cancer and high-grade urothelial bladder cancer. His prostate cancer was diagnosed 15 years prior to the current presentation, managed with radiation, and had since remained stable. The patient's high-grade, high-risk bladder cancer was diagnosed two years prior to current presentation. At the time, the patient initially presented with hematuria. A CT urogram performed at the time revealed a nonspecific bladder mass. Biopsy of the mass confirmed urothelial carcinoma. The patient's therapy course included a transurethral resection of the bladder tumor (TURBT), intravesical mitomycin, interferon alfa-2b, and intravesical BCG therapy. He received a total of seven intravesical injections of 50 mg live BCG per injection (350 mg cumulative dose) over the course of 18 months.\nThe patient's other significant medical history included rheumatoid arthritis, for which the patient was on weekly methotrexate and daily tofacitinib treatment. Both medications were discontinued on admission.\nThe patient developed scrotal pain two weeks prior to admission. He initially presented to the emergency department with this pain. At that time, he was diagnosed with acute bacterial epididymitis and was prescribed a course of ciprofloxacin. He had no improvement in his symptoms despite this treatment. Over the subsequent days, he reported experiencing intermittent chills and night sweats. He denied any penile discharge or any history of a sexually transmitted disease.\nEvaluation of the testicle with ultrasound revealed multiple diffuse nodular areas of hypoechogenicity (Figure , ), as well as marked hypervascularity involving all of the left-sided structures (Figure , ). Routine blood and urine cultures were negative, and mycobacterial blood and urine cultures, as well as bladder biopsy, were pending as of this writing.\nAntituberculous therapy was started based on the following considerations regarding the patient's presentation. The lack of observed response to ciprofloxacin therapy decreased the likelihood of uncomplicated bacterial orchitis. We considered this patient's history of bladder cancer involving multiple BCG treatments and the increased likelihood of BCG orchitis. Possibly, his immunocompromised state secondary to the malignancy, in combination with mitomycin chemotherapy and immunomodulatory rheumatoid arthritis treatment, could have increased his susceptibility to a mycobacterial infection. Finally, as ultrasound findings were characteristic of BCG orchitis, the patient was promptly initiated on a combination therapy with levofloxacin, rifampin, isoniazid, and ethambutol. The patient showed marked improvement in pain control and testicular swelling over the following weeks. |
A 32-year-old woman presented in 2010 to our clinic with history of primary infertility for two years. She had polycystic ovarian syndrome (PCOS) according to the Rotterdam criteria [] based on her history of irregular periods and sonographic polycystic ovarian morphology. The patient was initially given 100 mg Clomiphene citrate for ovulation induction followed by intrauterine insemination. After four failed cycles of ovulation induction/intrauterine insemination, a decision was made to proceed with IVF.\nThe patient underwent stimulation using a combination of 75 IU/day recombinant follicle stimulating hormone with gonadotropin-releasing hormone antagonist. Transvaginal retrieval of 22 oocytes was performed 36 hours after hCG administration (5,000 IU), and 17 oocytes were fertilized. Three days after egg retrieval, the patient presented with acute severe right lower quadrant pain associated with nausea and vomiting. Upon presentation to our hospital emergency department, her vital signs were stable and her abdominal exam demonstrated right lower quadrant tenderness and guarding with no rebound tenderness. Pelvic sonogram () revealed minimal free fluid in the cul-de-sac, right ovary measuring 85 × 54 mm, and left ovary measuring 71 × 53 mm with multiple residual follicles. Doppler ultrasound study was not available at the time of evaluation. Her white blood cell count was 14.4 × 103 cells/μL with 89% neutrophils. An emergent laparoscopy revealed a 10 × 12 cm purple right ovary twisted 180° around the right infundibulopelvic ligament in addition to a mild right hydrosalpinx. The left tube and ovary appeared normal. The right ovary was untwisted and regained its pinkish color within three minutes, consistent with a viable ovary. The patient had an uneventful recovery and was discharged home in stable condition on the same day.\nThe patient was counseled similarly to the patient described in “Case 1.” She also preferred to proceed with fresh embryo transfer. Two blastocysts were transferred on day five under transabdominal ultrasound guidance. Four weeks later, sonogram showed a dichorionic-diamniotic twin gestation of six-week size. She subsequently had a vaginal delivery of a healthy boy and girl at 35 weeks of gestation. |
A 78 year-old woman complained of a gradually progressive gait abnormality, beginning with difficulty initiating steps after rising from a chair. Her symptoms progressed steadily over four years, to include shuffling during forward walking and FoG with both gait initiation and passage through doorways. She felt that her freezing was significantly improved by the use of a rolling walker. She noticed that her right leg tended to freeze more than the left, but denied resting tremor or stiffness. She also denied falls (including backward), other coordination difficulties, autonomic complaints, and numbness or weakness. Upper limb functioning was normal. A previous trial of levodopa (up to 800 mg daily) did not alleviate her symptoms. Medical history included non-insulin dependent diabetes mellitus, uncontrolled hyperlipidemia, and remote 3 pack-year cigarette exposure. She denied exposure to neuroleptics.\nExamination revealed an oriented woman with normal language. Concentration was intact to complex backward spelling; executive function was intact to bedside Luria sequence testing. More detailed testing, however, revealed visuospatial and executive function deficits (clock drawing, box drawing, and Trails Part B), with a MOCA score of 22/30. Cranial nerves were normal. Tone and strength were normal. Resting tremor and bradykinesia were absent, including in the lower extremities. Proprioception was intact in the toes; coordination was normal. The right patellar reflex was brisk with crossed adduction; other reflexes were normal. Unassisted gait was wide based with freezing upon standing and prominent freezing with both straight walking and turning (see Additional file : video segment 1). The right foot was more affected by freezing with a pattern consisting of several small right steps with the right foot followed by one l step with the left. Walking with trekking poles resulted in dramatic improvement of gait and lessening of freezing (see Additional file : video segment 2). Despite significant gait impairments, standing balance in quiet stance was intact with normal postural sway, even on a foam surface.\nObjective measures of gait from body-worn inertial sensors [] showed significant asymmetry of right and left lower leg angular velocity during forward walking, caused by a peculiar pattern of multiple, small right steps followed by a singular left step (“floor scanning”). This phenomenon was only related to this case, and not seen in healthy control subjects, idiopathic PD or vascular parkinsonism (Fig. vs. , , and ). The areas shaded in light blue (Fig. vs. ) highlight this interesting non-reciprocal, floor scanning gait pattern. The red shaded areas highlight separate FoG episodes during turning.\nMRI of the brain showed a lesion in the anterior corpus callosum (Fig. ). It restricted diffusion but did not have an apparent diffusion coefficient (ADC) correlate to suggest acute infarct; nor did it enhance. Additional subcortical white matter changes were present in anterior periventricular and centrum semiovale regions, and were slightly more marked on the left (Fig. and ). MRI of the thoracic spine showed mild degenerative changes only. A DaT scan was not performed. Probabilistic tractography derived from diffusion tensor imaging revealed interhemispheric fiber loss in regions of the corpus callosum responsible for connecting the dorsal premotor (PMd) and pre-supplementary motor areas (pre-SMA) (Fig. ). For representative control data, see Fig. and . |
This 72-year-old man presented with left shoulder and AC joint pain and swelling, which progressively got worse over the past several months. He also had difficulty in raising his arm above his head. Left shoulder pain over the past several months was associated with a palpable mass along the medial aspect of the clavicle. He was a patient with known primary hemochromatosis and was homozygous for the C282Y mutation. This genotype is consistent with hereditary hemochromatosis.\nHe had a past history of total hip replacement 13 years ago. Histologic sections of the articular surface of the femoral head revealed varying degrees of articular cartilage damage ranging from fibrillation to focal ulceration and changes in the subchondral bone, containing focal thinning and hemorrhagic changes in the base of the articular head. Mild intertrabecular fibrosis was also noted. Several areas showed a split in the articular cartilage at the border of the calcified and uncalcified cartilage. However, no refractile material (iron) was seen in the cartilage or in the subchondral bone.\nOn physical examination, he had swelling and pain in the medial aspect of the left clavicle and left AC and SC joint with associated neck stiffness.\nRadiographs showed a focal erosion and linear chondrocalcinosis at the left humeral head and rotator cuff calcific tendinopathy Additional findings included degenerative changes at the AC joint and an old Hill–Sachs deformity.\nMRI showed a 1-cm erosion at the humeral head, which enhanced with contrast. Additionally, there were subscapularis tendinosis, enlargement of anterior glenoid labrum, iron deposit causing blooming artifact at the tendon, joint and labrum, and formation of glenoid labral ovoid mass (GLOM). He also had erosion and inflammation with effusion at the AC joint. Synovial proliferation, inflammation and soft tissue prominence were also seen at the SC joint. Post-gadolinium images showed marked enhancement of the AC and SC joints, as well as the subacromion/subdeltoid bursa due to bursitis. |
A 28-year-old male presented with the chief complaint of fever with chills along with cough and breathlessness for five days. He had a history of being diagnosed with acute myeloid leukemia three months ago, for which he had received chemotherapy cycle of 7+3 with daunorubicin and cytarabine. He had no history of hypertension, diabetes mellitus, tuberculosis or bronchial asthma. On general examination, the patient was febrile with a temperature of 101 °F, pulse of 106 beats per minute, blood pressure of 110/70 mm Hg in right arm supine position; pallor was present and SpO2 was 91 percent on room air. On systemic examination, trachea was centrally placed, there were fine crackles heard in the inframammary region of the chest, a systolic murmur was heard in the left second intercostal space which was functional, the abdomen was soft and non-tender with no hepatosplenomegaly and the patient was conscious and oriented. The patient was admitted for further evaluation and his nasopharyngeal swab for COVID-19 came positive by reverse transcriptase-polymerase chain reaction method. Blood investigations of the patient revealed pancytopenia and raised inflammatory markers with normal renal and liver function test (Table ). HRCT chest was suggestive of bilateral lower lobe ground-glass opacities with an HRCT score of 5/25 and CORAD 6 (Figure ). Patient was started on oxygen support along with remdesivir and steroids (Methylprednisolone 40mg twice Daily). He was given blood transfusion with two units of packed red cells transfusion in view of anemia. The patient was having persistent fever spikes along with lymphocytopenia in the blood picture. Hence, a careful risk-benefit assessment was made and granulocyte colony-stimulating factor was given to the patient for three days. Remdesivir was also given for 10 days. The patient improved clinically and oxygen was tapered. After 13 days of admission, the patient was ultimately discharged in stable condition with no fever spikes and an SpO2 of 95% on room air. |
A 52-year-old female presented to the emergency department with nausea, vomiting, and a severe headache. Computed tomography (CT) demonstrated subarachnoid hemorrhage anterolateral to the medulla. Cerebral diagnostic angiogram revealed a left posterior inferior cerebellar artery aneurysm ( and ). The aneurysm was treated with endovascular embolization but complicated by intraoperative rupture necessitating placement of an external ventricular drain ( and ). Routine cerebrospinal fluid samples were without evidence of infection. The external ventricular drain was removed on post-procedure day six. On post-procedure day seven, she was neurologically intact without evidence of sequelae. She was seen in clinic three weeks after her initial presentation to the emergency department. At that time, she was neurologically intact without complaints.\nShe presented again to our emergency department two weeks later (five weeks after her initial presentation) with nausea, vomiting, fever, hypotension, and a severe headache. Repeat CT was without evidence of hemorrhage (). A lumbar puncture was performed, which revealed a leukocytosis with a neutrophilic predominance. Although cerebrospinal fluid gram stain and cultures remained negative, intravenous vancomycin, cefepime, and acyclovir were started for presumed external ventricular drain-related meningitis. The following day she was transferred to the intensive care unit for respiratory failure, seizure activity, and decerebrate posturing. Electroencephalography was without evidence of epileptiform activity. Magnetic resonance imaging (MRI) fluid-attenuated inversion recovery (FLAIR) sequence on hospital day three demonstrated significant cerebral edema within the basal ganglia, thalamus, hippocampus, midbrain, and pons without evidence of diffusion restriction or contrast enhancement (). Repeat MRI FLAIR on hospital day 10 demonstrated exacerbation of the edematous process ().\nWithout a diagnosis to guide treatment, a brain biopsy was planned. A stereotacticguided biopsy of the right caudate was performed. Microscopic examination revealed infiltration of small vessels with a mixture of small and larger atypical appearing lymphoid cells, with some of the large atypical cells exhibiting parenchymal infiltration ( and ). Immunostaining demonstrated large atypical cells immunoreactive for CD20 suggesting a B-cell population with CD3 reactive T-cells intermixed with the atypical B-cell population ( and ). In situ hybridization assays demonstrated a larger number of atypical cells expressing lambda light chain with a small number of cells expressing kappa light chain ( and ). Final diagnosis of the biopsy specimen was consistent with atypical lymphoid proliferations of the caudate. The patient was started on intravenous immunoglobulin and intravenous steroids for immune-mediated pathology without clinical improvement. Workup for bacterial, fungal, and viral causes including Epstein- Barr, influenza, parainfluenza, herpes simplex, varicella-zoster, western equine encephalitis, eastern equine encephalitis, St. Louis encephalitis, California encephalitis, West Nile virus and rhinovirus were unremarkable. The patient’s cefepime was discontinued as a possible source of cephalosporin encephalopathy without clinical improvement. The patient expired on hospital day 23 (approximately eight weeks from initial presentation). |
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. |
The second case involves a 30-year-old Deaf, Hispanic male who presented to the Emergency Department after his mother reported that the he was behaving oddly and not taking his risperidone. Per reports, the patient was talking to his mother about going places in a UFO and exhibiting disorganized and illogical behaviors. He was subsequently placed under a Baker Act by the emergency room physician who documented that the patient was exhibiting auditory hallucinations. Initially an interpreter was brought to the hospital prior to his admission. Per the ASL-interpreter, the patient stated that he felt “fine and not crazy” and that all of these events are happening because his mother does not “understanding Deaf culture.” He also conveyed that he did not like to take his meds because they interfered with him being able to drink alcohol and caused drowsiness.\nUpon initial psychiatric interview an interpreter was not present as the hospital only agreed to set periods of time for the interpreter. As an effort to communicate, questions were prepared for the patient to answer via written responses. highlights a portion of the questions and answers that were constructed. From the responses he maintained bizarre delusions but denied current SI, HI, or AVH. When the ASL-interpreter arrived, the patient appeared jovial and yearned to express himself. The interpreter stated she had difficulties reading his rapid signing at first and had to have him slow down several times. However she did note that this was a common occurrence when addressing Deaf individuals.\nWith the interpreter's assistance, the patient was answering questions logically with a linear thought process. He reported that he had been diagnosed with schizophrenia as a teenager after having several interpersonal issues with his mother. She is Spanish speaking only and he stated that she has never fully understood how to communicate effectively with him. He had been taking risperidone for several years but was tired of continuing with the medication due to the side effects of drowsiness and weight gain, which he was never able to fully discuss with his psychiatrist. Patient reported he was in an ASL school and learning a career in massage therapy. After meeting a girlfriend there he began to develop a sense of independence that he reported his mother disapproved of. This caused an altercation that he reports his mother misinterpreted which precipitated his admission.\nThe patient continued to express that he was abducted by aliens as a child and could understand their language, but besides this he expressed no other psychotic processes. He was observed for two days without medications and remained calm/cooperative but was unable to participate in most activities due to limitations of the interpreter availability. After a family session was completed the patient was discharged home with plans to follow up with his community psychiatrist. The patients' diagnosis was changed to delusional disorder upon his discharge. |
A 63-year-old Japanese woman was referred to our department because of an abnormal shadow at the left side of her chest wall on computed tomography. She had undergone total hysterectomy and radiotherapy for cervical carcinoma 4 years prior. One year after the first surgery, three metastatic lung nodules appeared at the upper lobe of her right lung, the lower lobe of her right lung, and the lower lobe of her left lung. Wedge resection for upper and lower lobe of her right lung was initially performed via three-port thoracoscopic surgery. Then, wedge resection for the lower lobe of her left lung was performed via eighth intercostal single incisional thoracoscopic surgery. After the surgery, an intrathoracic chest wall mass developed which increased in size gradually. Her gynecologist introduced her to our department for surgical resection of the mass. Her family, including her parents and two sisters, had been healthy and had no inheritable diseases. She had no symptom, drug history, tobacco smoking history, or psychosocial history, and she was a social drinker. She had not received any medications since the mass developed and until admission to our hospital. She had undergone an operation three times as mentioned above and had been a carrier of type B hepatitis.\nAfter her admission to our department, her general condition was good, and there were three operative scars at both sides of her chest and lower abdomen. Her chest sounds were clear and there was no neurological abnormality. She was 151.1 centimeters tall and weighed 49.8 kilograms. Her heart rate was 77/minute, blood pressure was 135/87 mmHg, and body temperature was 36.1 °C. The laboratory findings were white blood cells 5.25 × 103/μL, hemoglobin 12.7 g/dL, and platelets 156 × 103/μL. A liver function test revealed: albumin 4.6 g/dL, aspartate aminotransferase 15 U/L, alanine aminotransferase 13 U/L, and total bilirubin 0.3 mg/dL. A renal function test revealed blood urea nitrogen 13.6 mg/dL and creatinine 0.79 mg/dL. An electrolyte test revealed sodium 143 mEq/L, potassium 3.8 mEq/L, and chlorine 106 mEq/L. A tumor marker test revealed carcinoembryonic antigen 3.4 ng/mL and squamous cell carcinoma antigen 0.80 ng/mL. Another test revealed positive reaction to type B hepatitis surface antigen and C-reactive protein < 0.1 mg/dL. Computed tomography demonstrated a gradually increasing low-density mass measuring 2.0 × 1.8 cm in diameter (Fig. ). Magnetic resonance imaging demonstrated a low-intensity mass in T1-weighted imaging and a high-intensity mass in T2-weighted imaging (Fig. ). The mass was thought to be a singular cyst; however, this type of cyst was rare and the mass was increasing. Therefore, dissemination of cervical carcinoma could not be excluded, and surgical removal of a part or tissue of the mass was performed.\nIn the right lateral position, thoracoscopic excision of the mass was done with two ports (3 mm and 2 cm access ports) by two general thoracic surgeons (Fig. ). First the 3 mm port was set at the sixth intercostal space on the inframammary line. Most of her left lung was attached to her chest wall; therefore, the second port was set above the cyst and lysis of adhesions was done. After the lysis, the cystic mass was found adhering to the upper lobe of her left lung. The adhesion of the mass to her lung was not strong and could be separated without injury to the visceral pleura. Therefore, the mass was thought to derive from the chest wall pleura and was resected by adhesiolysis.\nThe mass was a unilocular cyst containing mucinous fluid. On microscopic examination, the cyst was lined with a single layer of cuboidal epithelium (Fig. ); immunohistochemistry showed positive staining of calretinin and D2-40 (Fig. ). Thus, the cyst was diagnosed as mesothelial cyst derived from the chest wall pleura. Five years after the surgery, our patient had no evidence of cyst or cervical carcinoma on computed tomography. |
A 10-year-old girl visited our affiliated hospital, complaining of pudendal deformity. The patient was born at 39 weeks of gestation by normal delivery as the second child, and the birth weight was 3,144 g. There was no disorder in the course of pregnancy in her mother. Intake of androgenic medicine or the disorders of ovaries and uterus was not observed by a periodic medical examination. No pudendal deformity was clearly observed at birth, but lateral asymmetry of the pudendal region was noticed at about 4 years old. A child care worker pointed out that she pressed her heel to the crotch while sitting on her folded legs. After 5 years old, her mother confirmed that her clitoral hood clearly hypertrophied. After entering primary school, a teacher in charge pointed out that she pressed her crotch to a chair or bar, and the mother told her to stop it, but she repeated this behavior every day. When she strongly wanted to be absent from an overnight school trip at 9 years old, the mother brought her to the Pediatric Department. Various tests were performed suspecting disorders of sex development (DSD). On the first visit, the height was 132 cm and the body weight was 26 kg. The clitoral hood was enlarged. The appearance was similar to the vulva in children with congenital adrenal hyperplasia, and the clitoris size was 8 x 5 mm. Labial fusion or adhesion was not detected, and the urinary tract and vagina were open at the normal positions. No masculinization, such as acne and polytrichosis, was noted (). Intake of androgenic medicine or the disorders of prepuce was not observed. In the blood test, the sex chromosome was 46, XX. The blood count, blood chemistry, and hormonal test were normal (). On abdominal ultrasonography, the uterus and ovaries were present. Abdominal CT and MRI examinations showed no tumorous lesion.\nBased on the above examination and test findings, DSD was considered negative. The patient was diagnosed with clitoral hood enlargement and referred to our department to undergo clitoral hood reduction. For surgery, a longitudinal incision was designed for the dorsal side in order to resect the clitoral hood by cut and try. In the clitoral region, the clitoral hood and corpus cavernosum were dissected through an inverted V-shape incision. The volume of the exposed corpus cavernosum clitoridis was reduced while conserving the neurovascular bundle, following the Marberger method []. The clitoral hood was resected into a triangle shape and used for labial formation. On histopathological examination, lymphedema and venous tasis in a grade consistent with the influence of foreskin excision were observed. No abnormality was noted in the corpus cavernosum. Her postoperative course was uneventful. As of 10 months after surgery, favorable improvement of the appearance was noted (). |
A 30-year-old male with a noncontributory medical history presented for the replacement of missing upper-left first and second premolars and molars, which were extracted 3 years earlier. The results of blood tests, including a complete blood count, chemistry, and clotting profile, were all within normal limits.\nThe initial orthopantomogram () revealed an alveolar bone height resorbed edentulous area in the upper-left posterior maxillary region with pneumatization of the maxillary sinus. The residual bone height was 2.5 mm (class D) between the upper first and second molars, 4.5 mm (class C) in the second molar area, and 7 mm (class B) in the second premolar area. The classification of sinus morphology (classes A through E) was initially proposed by Jensen to aid in the selection of the appropriate grafting material or technique at a specific site []. The alveolar ridge was approximately 8 to 10.5 mm in width, and the occlusal clearance was 6 to 7 mm.\nIn the first stage, the 1st implant was placed at the least alveolar bone height, which was located in the 1st molar area, to obtain maximum lifting of the sinus area and avoid any interference with the Schneiderian membrane in the 2nd stage and prepare the adjacent resorbed sites for further implant placement in the sinus areas. The punch incision was initially preferred (the alveolar ridge was approximately 8 to 10.5 mm in width) but because of the risk of miscalculating the exact alveolar bone height in that critical area, under local anesthesia, a full-thickness flap was elevated using a mid-crestal incision and 2 releasing buccal and palatal incisions of the upper-left first molar. The bone was accessed and marked with a round bur. A 2.2 mm diameter pilot drill was used to the required depth of 0.5 mm below the roof of the sinus. Radiographs were taken with a depth gauge and distance indicator to determine the length of the preparation.\nTo improve the primary stability in cancellous bone, bone condensation through radial reinforcement was attained by a series of bone condensation devices with a tapered tip and an appropriate diameter of 2.2 mm up to 4.2 mm to widen the implant bed (ITI osteotome instruments for bone condensation, Institut Straumann, Basel, Switzerland). Malleting was performed to fracture the bottom of the sinus cavity using a 4.2 mm angled osteotome with a concave tip (ITI instrument for partial sinus floor elevation, Institut Straumann). A change in the resonance during malleting indicated complete osteotomy. The Valsalva test to assess the patency of the Schneiderian membrane was negative throughout the procedure. A 12 mm long, 4.8 mm diameter, standard neck, and nonsubmerged, SLA, screw-type ITI implant (Institut Straumann) was used. Manual and gentle screwing of the implant facilitated lifting of the sinus membrane to the required implant height, and the initial stability was attained. The surgical site was closed using 4-0 silk sutures. The implant position, osteotomized portions of the sinus floor, and the amount of sinus floor elevation were visible on radiographs (). Amoxicillin, 500 mg per 8 h, analgesics, and chlorhexidine mouthwash twice daily for 7 days were prescribed for the patient. The sutures were removed after 10 days, and the patient was followed up once monthly for 3 months.\nThree months after the first implant was placed in the upper-left first molar area, a reformatted flythrough image of the maxillary sinus was obtained using computed tomography (CT) and DentaScan; reformatted cross-sectional images were obtained for the radiographic evaluation of the inferior wall of the maxillary sinus and apical border for the first implant at area of the upper-left first molar after the osteotome sinus floor elevation () to reveal the Schneiderian membrane over the apical border projection of the first inserted implant.\nFour months after the first implant was placed, punch incisions were used to access the underlying alveolus for further implant placement. These sites required more than 5 mm of faciolingual width and little or no site development. A small envelope incision or tissue punch can be made to expose the bone for the osteotome; however, punch incision can be less traumatic and time consuming, with fewer complications and faster soft tissue healing. By including soft tissue thickness and using a surgical guide in the surgical plan, the procedure was accomplished and resulted in better patient comfort and easier surgical care. Three implants were placed on either side of the first implant. (i) A 12 mm long, 4.1 mm diameter, standard neck, nonsubmerged, SLA, screw-type ITI implant was used in the area of the upper-left second premolar. (ii) A 10 mm long, 4.1 mm diameter, standard neck, nonsubmerged, SLA, screw-type ITI implant was used in the area of the upper-left second molar. Both implants were placed using sinus floor elevation with the osteotome technique. (iii) A 12 mm long, 4.1 mm diameter, standard neck, nonsubmerged, SLA, screw-type ITI implant was placed in the upper-left first premolar area away from the maxillary sinus.\nFour months after inserting the implants, radiographs were taken and they showed the implant and surrounding bone under the tented sinus membrane in the upper-left first molar region (). The radiographs also showed the transformation of the residual ridge from classes C and D to classes A and B at the medial and distal sides of the implant, respectively. All the implants were loaded and restored with porcelain-metal retainers. Radiographs taken at 12 and 24 months (Figures and ) showed a stable clinical situation in the area around the apex of the implants. A dome-shaped structure was observed at the sites of the first and second molar areas. Different CT scans, which were obtained after 24 months postoperatively, were used to show the implant, surrounding bone, and new sinus floor using the osteotome sinus floor elevation technique (Figures and ). |
A 59-year-old man was referred to our hospital in August 2007 because of a progression of his chronic low back pain which began at the beginning of May 2007 accompanied by recurrent episodes of fever. The fever occured a few weeks later at the end of May 2007, and because of a temperature of 39°C the general practitioner treated the patient empirically with a 10-day course of amoxicillin/clavulanate. After that, the fever disappeared for a few weeks, but new episodes of fever and pain attacks occurred in July and August 2007. During this time, the C-reactive protein level was rising to 250 mg/l at admission (standard value <5 mg/l). The patient was under immunosuppression with Ciclosporine A and Mycophenolate-Mofetile after renal transplantation 2004, and he got an endovascular repair of an infrarenal aortic aneurysm with a Talent-graft in 2005. At admission, the patient's low back pain and fluctuating fever were interpreted as a sacroileitis. A MRI-scan showed no skeletal pathology but a slight enhancement of the sclerotic infrarenal aorta including an enhancement of the periaortal tissue. So, a CT guided translumbar needle aspiration of the enhancing structure was taken, and the cultures yielded growth of Listeria monocytogenes, whereas the blood cultures remained negative. Therefore, the patient was treated with ampicillin 2 gm q4h intravenously without gentamicin because of his impaired renal function (see ).\nUnder this antibiotic treatment, the CRP-level sank initially, but remained then around 120 mg/l so that gentamicin 1 mg/kg i.v. three times a day was added. At this time, the surgeon refused a surgical approach since the renal transplant was vasculated through the graft and a removal of the graft would have been a high risk for the transplanted organ. After 6 weeks of combined antibiotic therapy a new MRI showed new collections around the vascular graft down to the psoas muscle. A drainage delivered pure pus, the cultures were sterile but an in-house eubacterial PCR (16S ribosomal DNA sequence analysis) was positive for Listeria. So after 2 months of conservative treatment a surgical approach was faced due to lacking improvement. Intraoperatively extensive inflammatory adhesions were found; therefore, the aneurysm with the endograft could not be removed. Only a debridement around the aneurysm and in the psoas muscle could be performed. The Mycophenolate-Mofetil dosing was reduced at that time. Two weeks thereafter and after 8 weeks of combined intravenous antibiotic therapy the patient could be discharged with an oral regimen consisting of 320 mg trimethoprim/1600 mg sulfamethoxazole twice a day. A MRI-scan 6 months later in June 2008 showed no more abscesses but a slight enhancement in the periaortic tissue as well as in the psoas muscle. In January 2009, the CRP achieved a normal level (<5 mg/l), and, in July 2009, there was no more enhancement in the MRI-scan. Nevertheless, we decided to continue the antibiotic treatment for another 2 years (until July 2011) since the patient has a stable function of his renal transplant and since a relapse of the graft infection would be disastrous for the whole situation of the patient. In March 2011, the patient is well without any symptoms. |
A 63-year-old male with a past medical history of non-ischemic cardiomyopathy (ejection fraction 10%-15%), hypertension, chronic kidney disease stage G3A, and severe mitral valve regurgitation status post valve repair initially presented for an upgradation of his biventricular pacemaker to biventricular implantable cardioverter defibrillator (BIV-ICD) and replacement of non-functional right atrial lead. The pacemaker pocket was exposed and identified and the atrial and ventricular leads were pulled back under fluoroscopic guidance. A new left coronary sinus lead was placed. Next, right atrial (RA) and right ventricular (RV) leads were placed and access was obtained via the left coronary sinus as the left subclavian vein was found to be occluded with formation of collaterals. The leads were sutured in fascial planes via silk suture. The pacing and sensing parameters were confirmed. BIV-ICD was placed in the pocket, and the pouch was closed via vicryl sutures. The patient had electrocardiogram (EKG) (Figure ) and chest X-ray (Figure ) performed after the procedure. He had developed small pneumothorax from the procedure, which resolved on its own.\nThe patient was asymptomatic for the next seven months and then he developed increasing shortness of breath. At that point, the EKG (Figure ) revealed loss of appropriate pacing function. It was discovered that the patient kept massaging his pacemaker pocket site in the last seven months but he denied manipulating the device itself. He had also been moving his arm up and down frequently, which would have led all his device leads to be pulled up towards the device in a ratchet-like fashion. The patient was scheduled again for revision of BIV-ICD and on incision of the generator pocket, the left ventricular lead was found coiled up in the pocket beside the generator (Figure ). A new left ventricular lead was placed in the coronary sinus and then access was obtained via the right subclavian vein for right-sided leads. His right atrial and right ventricular leads were also found pulled up. A decision was made to place new right atrial and right ventricular leads. It is to be noted that all his device leads were undamaged and his device was found to be in the pocket in normal orientation. After securing the leads via sutures, the pacing and sensing function was analyzed. The pocket was revised and closed with vicryl sutures and the device was sewn to the pectoral fascia. A chest X-ray (Figure ) and EKG (Figure ) were obtained after the procedure to ensure the absence of pneumothorax and to document the appropriate functioning of the device, respectively. |
A 64-year-old man was transferred to our institution from an outside hospital complaining of chest pain and shortness of breath. Past medical history was significant for myocardial infarction, diabetes, congestive heart failure and prior aorto-bifemoral bypass grafting for leg claudication. On physical exam, a blood pressure could not be measured in either arm. We expected this to be a result of advanced PAD affecting the upper extremities, though the patient denied symptoms including pain and numbness in his arms.\nCoronary angiography showed significant narrowing of the left anterior descending, right coronary and circumflex arteries. A computed-tomography angiogram indicated diffuse atherosclerosis within the aortic arch with near-total occlusion of the innominate and bilateral subclavian arteries. Carotid duplex ultrasound showed that the left common carotid artery was also nearly occluded, but only low-grade stenosis was observed on the right.\nDue to the patient’s clinical picture, we scheduled him for surgery. In the operating room, a cutdown technique was used to mobilize the axillary arteries bilaterally such that a bypass could be performed. The great saphenous vein was simultaneously harvested from the right leg in an endoscopic fashion. Next, a skin incision was made along the medial border of the left sternocleidomastoid muscle and the carotid sheath was entered. Complete occlusion of the common carotid was appreciated just proximal to the bifurcation. The chest was then opened through a median sternotomy and the aorta was found to be heavily calcified. We determined that the ascending aorta was not amenable to bypass grafting and planned to replace it with a Dacron graft. After commencing cardiopulmonary bypass, the patient was cooled to 25°C. A saphenous vein graft (SVG) was anastomosed to the posterolateral branch of the right coronary artery in an end-to-side fashion using 7–0 Prolene. The same graft was looped to the left and sutured side-to-side to a marginal branch of the circumflex artery. A separate SVG was then anastomosed to the left anterior descending artery. Next, the aorta was resected from just above the right coronary ostium to the base of the innominate artery. The aorta was heavily calcified but the valve did not appear to be diseased. The resected portion of the aorta was replaced with a 26-mm Dacron graft using 2–0 Prolene. Once this graft was in place, two openings were made within the graft and the two proximal SVG anastomoses were performed using 5–0 Prolene. A 12-mm bifurcating Dacron graft was then sutured to an opening in the side of the prosthetic ascending aorta with 4–0 Prolene (). The patient was then rewarmed and weaned off of cardiopulmonary bypass without complication. Total time on cardiopulmonary bypass was 99 minutes and the aorta was cross-clamped for 73 minutes.\nThe 6-mm limbs of the bifurcation graft were then pulled through the pleural cavities bilaterally and tunneled to the axillary arteries where they were anastomosed in an end-to-side fashion using 5–0 Prolene. A Doppler stethoscope was used to confirm adequate blood flow to the arms. Next, a 6-mm Dacron graft was anastomosed to the left limb of the bifurcation graft using 5–0 Prolene. Clamps were then placed on the left carotid artery before an opening was made at the bifurcation distal to the occlusion. The 6-mm graft was tunneled through the chest and into the neck where it was anastomosed to the common carotid artery (). Adequate blood flow to both the internal and external carotid arteries was confirmed using a Doppler stethoscope. Operative length was 5 hours and 16 minutes, and the patient’s post-operative course was unremarkable. At 1-month follow-up, all grafts were patent and the patient’s chest pain had resolved. |
The second patient was a 64-year-old man who presented similarly, with 2 weeks of intermittent pyrexia and profuse night sweats. He had a tissue aortic valve replacement for severe aortic stenosis 5 years prior to presentation. He underwent aortic root replacement 2 years later. There were no localizing symptoms. On examination, he had an ejection systolic murmur heard throughout the precordium with a valvular click noted. Examination showed no peripheral stigmata of endocarditis.\nThe ECG showed sinus rhythm and chest X-ray was normal. Full blood count and renal function were normal. Liver enzymes were slightly elevated without jaundice or hypoalbuminaemia. The CRP was elevated (47 mg/L) (<5 mg/L). All blood cultures were negative and a sample for mycobacterial culture was sent. HIV and hepatitis screen were negative. Transthoracic and transoesophageal echocardiography showed no vegetations. Abdominal ultrasound showed a thickened gallbladder wall and one small gallstone. CT thorax, abdomen, and pelvis excluded the presence of malignancy or abscess as the cause of the pyrexia.\nAn FDG PET CT scan was performed which showed a retrosternal soft tissue density/collection involving the pericardium and linear increased uptake was noted in the ascending aorta and root (). This mass was felt to represent an abscess. There were also metabolically active areas in the right upper lobe and focal splenic uptake. The mycobacterium blood culture isolated a Mycobacterium avium intracellular type organism which was identified later as M. chimaera infection, and diagnosis of infective endocarditis secondary to M. chimaera was established.\nPatient 2 was initially treated as endocarditis and started on intravenous flucloxacillin, rifampicin, and gentamicin. Based on the equivocal abdominal ultrasound result, microbiologists briefly advised treating with intravenous piperacillin–tazobactam for suspected cholangitis. There was no right upper quadrant tenderness however and clinical suspicion of cholangitis was low. He continued to spike temperatures prompting the addition of doxycycline to cover for atypical bacterial infection. His temperature settled and he was discharged with infectious diseases team outpatient follow-up in 2 weeks. Over the follow-up period, he reported rigors and night sweats. The diagnosis of M. chimaera infective endocarditis was eventually established based on the results of the PET scan and the mycobacterial blood cultures.\nHe was commenced on rifampicin, ethambutol, and clarithromycin. His temperatures had settled and his night sweats reduced in frequency and intensity. However, his appetite remained poor and he continued to lose weight. On follow-up with the infectious disease consultant, he complained of dysphagia and ongoing weight loss. He also developed an extensive rash over his arms, legs, and back. The infectious disease team felt this was medication-related and decided to stop all antibiotics temporarily until the rash resolved.\nAfter stopping all medications, he continued to feel unwell and shivery with myalgia and a diffuse itchy rash. He became persistently pyrexial and developed diffuse erythroderma. The patient was admitted to hospital, received topical steroids, improved and was discharged after a week. This presentation was discussed in the multidisciplinary infectious disease meeting and diagnosed as DRESS syndrome secondary to clarithromycin. The rash completely resolved after 1 month. The patient was then admitted for reintroduction of antimicrobial therapy for M. chimaera infection and discharged without complications.\nAfter 2 months, the patient developed gradually worsening dysphagia. Swallowing assessment did not show structural abnormality. His barium swallow was normal, and CT scan of the chest, abdomen, and pelvis showed new diffuse parenchymal lung shadowing with new significant splenomegaly. A diagnosis of disseminated M. chimaera infection was then made. An honest and thorough discussion with the patient and family about the situation was held to discuss his poor prognosis and poor response to therapy as well as a ceiling of care. In his case repeat cardiac surgery to remove the infected valve was not feasible as he had undergone two previous cardiac operations; a third operation was felt to be too high a risk. The patient expressed the wish to die at home. After discussion with the palliative care team, a plan for care of the dying person was set up. He died almost 1 year after the initial presentation. |
The patient is a 82-year-old Malawian man, otherwise healthy, who first noted swelling and mass on penis for a 2-week duration. Per report, this mass was localized to the right side of the penile shaft and resembled a blister. The blister was unroofed at a local clinic and the patient was sent home with oral analgesic medications. Over the course of the following week, the patient noted that his penile lesion had burst, and discovered leakage of urine and foul-smelling discharge from the fungating mass. He was transferred to Mzuzu Central Hospital (MCH) which acts as a tertiary referral center for surgical cases.\nUpon arrival to MCH, the patient was hemodynamically stable with no systemic signs of infection. On physical examination, he was noted to have a 3 cm by 3 cm fungating mass on the right aspect of his penile shaft (Fig. ). The patient also had firm, bilateral inguinal lymphadenopathy concerning for malignancy. Both the mass and inguinal areas were moderately tender to palpation. Our preliminary diagnosis at this time was penile cancer versus genital infection. We cleaned the mass and applied a sterile gauze dressing and continued with daily dressing changes. A complete blood count was ordered which revealed a microcytic anemia (hemoglobin 10.1 g/dl) and a normal white blood cell count (5400 cells/ul) without left-shift. He had no other systemic signs of infection, however due to inability to rule-out an infectious etiology, the patient was started on IV ceftriaxone.\nThe patient was taken to major theater later that day for exploration and penile amputation. Spinal anesthesia was induced given that general anesthetic agents were running low and reserved for emergency cases (Fig. ). After the patient’s penis and bilateral groins were prepped and draped, we then turned our attention to the penile mass. A circumferential incision was made through the skin at the base of the penis, away from the lesion itself. A skin flap was then raised circumferentially using electrocautery and blunt dissection. The penis was degloved, then a tourniquet was placed to ensure hemostasis. A penile amputation was performed and the specimen was sent for permanent pathology along with inguinal lymph node. The exposed corpora cavernosa were closed with a running 0-Vicryl suture. The tourniquet was then removed and hemostasis was ensured along the amputated surfaces.\nNext, we moved on to the reconstruction portion of the operation. Scrotal flaps were raised bilaterally by making a vertical incision into the median raphe. A left hydrocele was encountered which was opened, drained, and evaginated using absorbable sutures. The scrotal flaps were then used to perform a complex reconstruction by closing the flaps over the remnant urethra. The urethra was spatulated sharply along the three- and nine-o-clock aspects, then the spatulated opening was sewn to the scrotal flaps using absorbable sutures. A 20-French Foley catheter was gently inserted into the reconstructed urethra avoiding any undue tension. The remainder of the skin flap was closed in multiple layers, and the procedure was completed (Fig. ).\nPostoperatively, the patient was taken to the post-anesthesia care unit for recovery, then transferred to the male surgical ward. He was continued on IV ceftriaxone to complete a seven-day course, which is standard protocol given the poor sterile conditions at MCH. Pain control was achieved with diclofenac and paracetamol. Surgical dressing was removed on the third postoperative date. We noted that the skin flaps had approximately 5% necrosis on the inferior aspect of the reconstructed penis, but otherwise appeared healthy without separated tissue. The patient was discharged on the seventh postoperative date after his family members were taught dressing change techniques. The patient’s Foley catheter was kept in place at discharge to allow for urethral healing without stricture formation, with planned removal at follow-up. Pathology report was not available at time of discharge. |
An 87-year old woman visited our dental hospital for prosthetic treatment. She had residual teeth which could not be used as abutments for the denture. For prosthodontic rehabilitation, removal of remaining teeth and full denture placement were planned. Therefore, she was referred to the department of Oral surgery for extraction of the residual teeth and tori removal prior to prosthetic treatment. She had hypertension and was taking amlodipine. The American Society of Anesthesiologists physical status of the patient was II. Preoperative laboratory findings and the chest X-ray image were normal. The electrocardiogram showed sinus rhythm with right bundle branch block and left anterior fascicular block. However, no significant abnormality was noted on echocardiography. Her skin was thin and inelastic due to aging, with extensive wrinkling in the face.\nGeneral anesthesia was induced for surgery and the patient was monitored. In addition, bispectral index (BIS) monitoring was conducted for checking the depth of anesthesia and the BIS sensor was fixed using surgical drape tape (Loban™2 incise drapes; 3M, St Paul, MN, USA). Surgical tape was attached from the bottom of the ear to the forehead for prevention of betadine drape and water. Approximately one hour after the surgery, she was reversed from the effects of the muscle relaxant using pyridostigmine 10 mg and glycopyrrolate 0.4 mg. After extubation, the surgical tape was removed to detach the BIS sensor and the hair cover. After the surgical tape was removed, skin injury occurred on the left side of her face. Along the site of attachment, 1.5 cm by width and 4 cm by length of her skin peeled off with the surgical tape. In addition, a red bruise appeared around the area of the surgical tape (). Immediately after repositioning the epidermis, antibacterial ointment was applied and covered with non-adhesive foam dressing to maintain a moist wound environment. It was maintained for five days, and a repeat dressing was done during the next visit. The epidermis was retained and secondary healing had initiated in the uncovered area. However, the bruising had worsened (). After realignment of the epidermal margin, ointment was applied and a dressing was placed. She changed her dressing daily at home by herself and visited the clinic a week later. The wound had healed well compared to the first visit at outpatient clinic after surgery. The area of bruising had reduced considerably. No evidence of infection was seen (). There was no problem in the intra-oral surgical site and stitch out was performed. She decided to continue performing the dressing at home by herself. |
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 30-year-old man was primarily presented with a simple spontaneous swelling and pain on the dorsum of the foot. Initially, the internist treated the problem as gout. However, the problem persisted and thus radiography and magnetic resonance imaging (MRI) evaluations were carried out. A unicameral bone cyst (talocalcaneal and talonavicular joints) was suggested () and the patient underwent surgical curettage followed by harvesting autologous bone graft from the iliac crest. As the surgery was carried out at another center, we had no information on the surgical setting, preoperative planning, or the surgeon’s decision on the differential diagnoses of the disease. The pathology report indicated synovial cell sarcoma and subsequently the patient underwent chemoradiotherapy.\nDue to postoperative tumor recurrence, the patient was referred to our center. At this stage, knee amputation was suggested. However, the patient rejected this option and requested another tumor excision attempt. We accepted the request conditioned to amputation if a margin-free tumor excision was not possible. Two weeks later, regretfully, we had to perform another operation for a below-the-knee amputation. After 9 months, during a follow-up visit, a bulge in the pelvis around the previous site of donor graft was observed. Sonography, MRI, and CT-scan evaluations revealed a mass on the ilium at the graft harvesting site of the first surgery (). Another surgery was performed and the mass was excised. Surprisingly, the pathologic study revealed synovial sarcoma (). Immunohistochemistry studies (with CD99, EMA and S100 markers staining) revealed monophasic sarcoma composed of only spindle cell sarcomatous component characterized by a high degree of cellularity, nuclear atypia, and mitotic activity. Spindle cells had distinct lobulation and were arranged in fascicles.\nBased on the above course of events, we deduced the implantation of the primary tumor cells of the foot to the graft harvesting site. We believe that the first surgeon did not consider the possibility of a malignant tumor and used the same surgical devices for both the tumor excision and the graft harvesting. Written informed consent was obtained from the patient for the publication of this case report, including accompanying images. |
In August 1993, a then 45-year-old Indonesian female underwent a modified radical left mastectomy for T1N0M0 breast cancer. Her earlier history revealed keloid formation on shoulders and upper back, although she then denied significant previous traumas. Postoperatively, timed mammographies of the right breast were proposed according to the Guideline on Breast Cancer []. Approximately 1 month following the first mammography in November 1993, keloid tissue started to emerge around the nonoperated right breast, reflecting the position of the rigid breast compression paddle that was used for the mammography. Although she was worried that the lesions would progress, mammography screening was continued annually, indeed leading to worsening of the keloid formation.\nWhen she presented to our outpatient clinic in 2005, a physical examination revealed firm, hyperpigmented, smooth-surfaced lesions on both sides of the thorax (Fig. ). Remarkably, the scars following the mastectomy remained within the confines of the original surgical incision and had not expanded over time. Conversely, keloid formation on the right side was progressive reflecting the repetitive trauma associated with approximately 12 consecutive mammographies over a period of 12 years. As she was convinced of an association between the keloid formation and the mammographies, it was decided to abandon these radiographies. She declined serial MRIs. As a consequence, it was decided to limit oncological control to palpation.\nBecause of the patient's progressive emotional burden associated with the growing keloids she was referred to a dermatologist who advised calendula, Dermatix® silicone gel, and corticosteroid injections, but to no avail. She was advised to seek consultation of a plastic surgeon, but she dismissed this option because of fear for invasive therapy as well as doubts on the success rates of therapies.\nIn 2018, she again visited our surgical department outpatient clinic as keloids had progressed even more over time. On inspection, strands of keloid tissue expanded from the right breast to the left breast causing spontaneous pain (Fig. ). She told us that the localizations of keloid on both her shoulders had possibly developed after insect bites and scathing, at the age of 13 years. At the age of 21, she underwent excision of these shoulder keloids. Unfortunately, widespread recurrence occurred thereafter (Fig. ). Prior to the modified radical left mastectomy, no screening mammography has been performed. Only one diagnostic mammography has been performed 2 weeks prior to the operation without origination of keloid. Later, at the age of 63, a vestibular schwannoma was removed via a retroauricular incision (Fig. ). Her sister and one daughter were also affected by excessive keloid formation following skin injuries (e.g., abrasion, surgical excision). Her other daughter, son, mother, and father were not affected. |
A 55-year-old Vietnamese male with no significant past medical history presents to a local community hospital emergency department because of abdominal pain and distention of two-week duration. The abdominal pain was generalized, described as cramping, present throughout the day, had no association with meals, and was getting progressively worse. The patient also reported nausea and vomiting clear material. He denied any recent fever, chills, night sweats, weight loss, change in bowel habits, sick contacts, and consumption of raw food. In addition, he denied any chest pain, shortness of breath, joint swelling, and skin rash. After immigration from Vietnam 11 years ago, his only travel outside the USA was back to his homeland one year prior to this illness. The patient was not taking any prescribed or over the counter medications or herbal compounds and denied any allergies. His physical examination showed no skin rash or jaundice, cardiopulmonary examination showed no abnormality, and the abdomen was moderately distended, with active bowel sounds, diffuse tenderness without rebound, and moderate ascites. There was no hepatomegaly or abdominal mass.\nIn the emergency department, a complete blood count and comprehensive metabolic panel were significant for an elevated white blood count of 15.400 with 36% eosinophils. Abdominal and pelvis computer tomography (CT) showed moderate ascites with thickening of the gastric antrum and proximal small bowel (). The patient was admitted to the general medical service and placed on bowel rest and intravenous fluid hydration. Further he underwent esophagogastroduodenoscopy (EGD), which demonstrated mild duodenitis and biopsies demonstrated mild nonspecific acute inflammation predominantly lymphocytic. Stool tests were negative for ova and parasites. In addition, a screen for Cryptosporidium, Cyclospora, Isospora, and Sarcocystis did not reveal evidence of recent infection. Over several days, the patient's abdominal pain improved, diet was advanced, and he was discharged home with an empiric trial of albendazole for a presumptive diagnosis of parasitic infection.\nTwo weeks after discharge, the patient was readmitted with worsening abdominal pain. Physical examination showed increased abdomen distention. Repeated blood counts and serum biochemical tests demonstrated an increase in white cell count of 17.100/mL with 71% eosinophils (absolute eosinophil count of 12.141/μl with normal upper limit <450) (). Liver function tests continued to be within normal limits. Serum IgE level was elevated at 548 IU/mL (normal < 180). Repeat stool tests were negative for ova and parasites. Furthermore, immunologic studies for Toxocara, Trichinella, Strongyloides, Filiaria, Schistosoma, Echinococcus, and Cysticerus were negative.\nRepeated EGD was nondiagnostic. Flow cytometry of peripheral blood revealed no myelo- or lymphoproliferative findings. Serum β-2-microglobulin and LDH were 2.2 mg/dL (normal: 0.8–3.0) and 170 U/L (normal: 80–200), respectively. Ultrasound guided abdominal paracentesis showed WBC count of 6600/mL, 95% of which were eosinophils (), LDH 284 mg/dL, albumin 3.2 g/dL (simultaneous serum albumin 4.1 g/dL). In order to exclude small bowel lymphoma, the patient underwent diagnostic laparoscopy with full-thickness biopsy of an inflamed portion of the jejunum. This revealed skipped areas of hyperemia and discoloration involving the small intestine and to a lesser degree the colon in addition to yellow-green ascites (). Histopathological evaluation showed marked eosinophilic infiltration of the muscularis propria and serosa with concomitant mild acute inflammatory reaction (). There was no evidence of malignancy, granuloma, TB, or parasites.\nThe constellation of clinical presentation and histopathological findings were suggestive of eosinophilic gastroenteritis. Subsequently, the patient was started on oral prednisone (20 mg/day). Two weeks later and with noticeable symptomatic improvement, the prednisone was tapered over a two-week period. After completion of steroids, the patient's abdominal pain and physical finding of ascites completely resolved and a peripheral blood count revealed an absolute eosinophil count of 300/μl (nL < 450). Furthermore, IgE level dropped to 105 IU/mL and CT imaging of the abdominal and pelvis showed complete resolution of the ascites and small bowel thickening. Four months have elapsed since treatment and the patient remains asymptomatic on no medications. |
A 52-year-old female with history of hypertension, smoking, alcohol dependence, and coronary vascular disease with recent myocardial infarction was admitted due to acute change in mental status. The patient had a spontaneous intraventricular hemorrhage, and an emergency bedside ventriculostomy was performed. Her hospital course was marked by persistent fevers in spite of multiple negative blood, cerebrospinal fluid (CSF), and urinary cultures. She received empiric levofloxacin intravenously 750 mg daily and vancomycin 1 gram intravenously every 12 hours until day 9 of hospitalization. Her neurologic function steadily improved, and the ventriculostomy was discontinued on day 11. However, her CSF cultures on that day yielded coagulase-negative staphylococcal (CoNS) species. Vancomycin 1 gram and rifampin 600 mg intravenously were administered every 12 hours. On day 15, computerized tomography (CT) of the head demonstrated increased intraventricular hemorrhage requiring reinsertion of an external ventriculostomy. Repeated CSF analysis demonstrated elevated WBC (57 cells/mm3 and protein 149 mg/dL) with decreased glucose (67 mg/dL). CSF cultures yielded CoNS again. Cultures of CSF continued to yield CoNS for the next three days despite the addition of daptomycin intravenously at 10 mg/kg (dosed at actual body weight) daily. As a result, intraventricular daptomycin was added on day 18, 10 mg daily for the first two days and then every other day. Culture of CSF became sterile on day 25 following 7 days of daptomycin intravenous and intraventricular therapy in addition to continued administration of intravenous vancomycin and rifampin. The second ventriculostomy was discontinued on day 38 (as was rifampin administration). Intravenous daptomycin and vancomycin were discontinued on day 55 after 37 days of dual treatment. Mental status progressively improved during this time.\nDaptomycin peak and trough levels in the CSF were measured on day 29, and day 30, respectively (correlating to day 10 following the start of intravenous and day 11 following the start of intraventricular daptomycin). The peak CSF level (following intravenous and intraventricular administration) was 6.30 mcg/mL on day 29 and the trough CSF level was 1.39 mcg/mL on day 30. The serum trough level on day 30 was 20.15 mcg/mL, and the minimum inhibitory concentration (MIC) of CoNS to daptomycin was <1 mcg/mL.\nThe remaining hospital course was complicated with respiratory failure requiring tracheostomy and ventilation support, ventilator-associated pneumonia, sacral pressure ulcer, and Clostridium difficile colitis. The patient was successfully discharged on day 65 to a rehabilitation center. |
A 51-year old male with Type 2 diabetes, taking no medication, monitored his plasma glucose levels from April 24 to May 30, 2003. He also monitored the dirty electricity in his home using a Protek 506 Digital Multimeter connected to a ubiquitous filter () to remove the 60-Hz signal and its harmonics. Measurements were taken in the morning and randomly throughout the day. Low or no readings of dirty electricity were taken in an electromagnetic clean environment far from power lines and cell phone antennas ( upper graph). Three years later, the microsurge meter became available and Case 1 monitored his blood sugar levels once more ( lower graph). This meter provides a digital readout of the absolute changing voltage as a function of time (|dv/dt|, expressed as GS units) for the frequency range 4–100 kHz and with an accuracy of ±5% ().\nshows a positive correlation between dirty electricity and plasma glucose levels taken randomly during the day (upper graph) and first thing in the morning (lower graph). His elevated plasma glucose is unrelated to eating. Working on a computer increases blood sugar, but these values decrease as much as 0.11 mmol/L [2 mg/dL] per minute after moving away from the computer. Blood viscosity decreased as his plasma glucose levels dropped.\nCase 1 also documented rapid changes in blood sugar as he moved from a medical clinic (environment with dirty electricity), to his parked vehicle (no dirty electricity), and back to the medical clinic. His blood sugar levels changed significantly within 20 min. His endocrinologist classified him as pre-diabetic when his blood sugar was tested immediately upon entering the medical clinic and as a Type 2 diabetic after a 20-min wait in the medical clinic. Measurement of blood sugar needs to be done in an electromagnetically clean environment to prevent misdiagnosis and to accurately determine the severity of the disease. |
A 78-year-old male patient was admitted to our hospital due to a soft mass with a rapid growth over 10 d.\nIn 2020, he accidentally noticed a soft mass the size of a pigeon egg on the inner side of his left knee joint with good mobility and no local redness, swelling or pain. He visited the local clinic, and Color Doppler ultrasound examination suggested that the tumor was a pilomatricoma. No further treatment was performed at that time. However, when the tumor increased over 10 d, the patient felt occasional numbness and discomfort in the left lower extremity.\nThe patient was previously diagnosed with a superficial spreading melanoma on his left thigh, which was excised in our hospital in 2015. The postoperative positron emission tomography-computed technology examination showed no evidence of regional lymph node metastasis and distant metastasis, so the subsequent radiotherapy or chemotherapy was not performed.\nThe patient had no relevant family medical history.\nA painless, tough tumor 5 cm × 5 cm × 4 cm in diameter within his left knee joint with a clear boundary, rough surface and hot flushed skin was noted. There were no obvious abnormalities in sensation, blood supply and movement of the left lower limb.\nBefore operation, the patient’s erythrocyte sedimentation rate increased (30 mm/h, normal range: 0-15 mm/h) and C-reactive protein was slightly high (10.3 mg/L, normal range: 0-10 mg/L). Other laboratory examination results were normal, including hematological, coagulation, kidney and liver functions as well as electrolytes.\nColor Doppler ultrasonography (Figure ) revealed a hypoechoic mass with an unclear boundary in the subcutaneous soft tissues of the medial left knee with abundant dotted and band-shaped blood flow signals in and around the lesion. Computed tomography scanning (Figure ) showed a subcutaneous lesion in the left medial femoral area with an average computed tomography value of 34 HU, which was significantly lower than the adjacent soft tissue (62 HU). The skin adjacent to the tumor was slightly thickened, and no obvious signs of bone destruction were observed. Magnetic resonance imaging (MRI) examination (Figure ) revealed a subcutaneous and lobulated tumor, measuring 4.51 cm × 2.75 cm × 3.00 cm in maximum size, irregular in shape but well-circumscribed with a rich blood supply and the absence of a visible entering or exiting nerve. The lesion was heterogeneously hypointense on T1-weighted images (T1WI) (Figure ) and fat-saturated T2-weighted images (T2WI) (Figure ) in the sagittal plane. It also showed heterogeneous enhancement with nonenhanced focal areas on contrast-enhanced T1WI in the sagittal and coronal plane (Figure and ). The hyperintense grid-like fascia on fat-saturated T2WI (Figure ) and enhanced thickened skin (Figure ) on contrast-enhanced T1WI suggested peritumoral edema and the invasion of surrounding soft tissues. |
A 36-year-old woman with 21 weeks of pregnancy was admitted to our hospital for a left renal tumor, which was incidentally detected by ultrasonography in a routine pregnancy examination. Ultrasound showed a confounding echo mass in the middle part of the left kidney, 7.9 cm in size, uneven internal echo, and clear blood flow signal inside. Computed tomography (CT) was not performed, as she was pregnant. Abdominal magnetic resonance imaging (MRI) showed a round mass in the middle part of the left kidney with a maximum diameter of 8.3 cm (). The signal is heterogeneous, with some fluid visible inside the mass. Since angiomyolipoma usually shows hyperechoic rather than confounding echo in ultrasound, renal cancer was considered. The patient denied lower back pain, hematuria, fever, frequency of urination, urgency, pain, and other discomforts. Past medical history was unremarkable. The patient gave birth to a healthy girl 5 years ago. Personal history and family history were not remarkable. After admission, there were no abnormalities in vital signs, blood pressure was 130/80 mmHg, blood routine and biochemical examinations were within the normal range (hemoglobin was 114 g/L, serum creatinine was 47 μmol/L, and potassium was 3.7 mmol/L). Physical examination: There was no tenderness or muscle tension in the abdomen, and the mass was not touched.\nA multidisciplinary team including urologists, gynecologists, pediatrists, anesthesiologists, and radiologists was responsible for the decision making to help the patient and her baby. Considering that the patient is only 36 years old, her strong desire to remove the tumor as well as retain the kidney, and the relatively indolent nature of the RCC, retroperitoneal laparoscopic partial nephrectomy was performed by an experienced surgeon after the risk was explained and informed consent was signed.\nDuring the procedure, the patient was placed in the lateral flank position and underwent general anesthesia with endotracheal intubation. The procedure was performed through a retroperitoneal approach, and the retroperitoneal cavity was formed by blunt dissection and balloon dilation from a small incision located 2 cm above the iliac crest of the midaxillary line. After the establishment of the retroperitoneal space, four trocars were inserted on the left waist between the superior edge of the iliac spine and the inferior border of the rib. The tumor was removed completely, and renal reconstruction was then achieved with a 1/0 self-retaining barbed suture (V-Loc). Although maternal hemodynamic parameters were maintained stable and end-tidal CO2 was monitored below 35 mmHg, pneumoperitoneum pressure was strictly controlled below 12 mmHg to reduce maternal hypercapnia and fetal acidosis throughout the surgery. The operation time was 100 min with a warm ischemia time of 28 min and an estimated blood loss of 150 ml. The patient recovered uneventfully after the operation and was discharged within a week after the surgery. Blood hemoglobin was decreased postoperatively [95 g/L on postoperative day (POD) 1, 99 g/L on POD3] and back to normal on POD7 (121 g/L), while serum creatinine levels were normal throughout the perioperative period (66 μmol/L on POD1, 61 μmol/L on POD3, and 60 μmol/L on POD7). Obstetrics and gynecology consultation monitored the fetus before and throughout the operation.\nThe pathology report revealed an 8.2-cm clear RCC, Fuhrman grade 2, with negative surgical margins (). According to the TNM classification system, it is classified as pT2aN0M0. During the 38th gestational week, a healthy male infant was born. We followed our patient every half year after surgery. Blood hemoglobin, serum creatinine, and thorax–abdominal–pelvic CT scan showed normochromic, normal renal function, and no sign of local recurrence or metastases. After 46 months of follow-up, the patient's baby is in good health and does not have any developmental birth defects. The whole treatment process is shown in . |
A 38-year-old man was transferred to our emergency room for shortness of breath. Two hours before admission, he began to have sudden onset of dyspnea, and was intubated in another hospital. He reported to be healthy before this episode, and systemic review was negative. He did not have a remarkable past medical history, including surgery, allergy or specific medications. However, he had a family history of ischemic stroke; his father died of complication of ischemic stroke at the age of 65 years and his brother had ischemic stroke at the age of 39 years.\nIn the emergency department, he had respiratory distress and tachycardia, with an oxygen saturation of 81% even under the intubated state. The blood pressure fell to an unmeasurable level, and the heart rate was 102 beats/min. There were crackles in both lungs on auscultation. Chest X-ray revealed mediastinal widening and cardiomegaly with bilateral infiltrations. Computed tomography (CT) of the chest showed dilated pulmonary trunk, but there was no evidence of pulmonary thrombus, aortic dissection or lymphadenopathy. The patient was admitted to the intensive care unit with a tentative diagnosis of acute respiratory failure due to severe pneumonia.\nOn hospital day 3, chest X-ray showed decreased infiltration and vital signs were stabilized. However, he developed sudden onset of tachypnea and hypotension on the next day. Arterial blood gas analysis results while breathing ambient air were pH 7.21, PaO2 50.0 mmHg, PaCO2 71.4 mmHg and oxygen saturation 76%. A follow-up chest CT revealed a large thrombus in the right main pulmonary artery and marked dilatation of the pulmonary arteries []. In addition, atrophy of pectoralis and paravertebral muscles was detected []. Transthoracic echocardiogram (TTE) showed pulmonary arterial hypertension (right ventricular systolic pressure 60 mmHg) with D-shaped left ventricle. However, echocardiographic evidence of a thrombus was not identified.\nThe patient received thrombolytic therapy followed by subcutaneous low-molecular weight heparin, and his symptoms improved with the treatment. Screening tests for hypercoagulable state including protein C, protein S and anti-thrombin III were normal. Lupus anticoagulant, anti-cardiolipin antibodies and factor V Leiden mutation tests were also negative.\nDuring the admission, neurologic examinations performed by a consultant neurologist revealed mild ptosis in both eyes, frontal baldness, temporal wasting, elongated face, weakness of distal upper extremities and percussion myotonia. Electromyography showed typical myotonic discharges in proximal and distal extremities, consistent with myotonic dystrophy. The diagnosis of myotonic dystrophy type 1 was subsequently confirmed by an expansion of CTG repeat on chromosome 19. He was discharged to a rehabilitation facility, at which time he was functioning independently with nocturnal non-invasive ventilation. |
A 46-year-old male patient was referred to Dayananda Sagar Dental College and Hospital with a gradually enlarging swelling of the right mandible crossing the midline. The patient had noticed the tumor 5 years before and enlargement of the tumor 4 months prior to admission. Physical examination revealed a swelling on the right mandibular angle area. Intraorally, a bony hard painless swelling was extending from the lower right third molar region to the left canine region with intact overlying mucosa. All the teeth in the lesional area were vital with mobility of the right incisors and molars.\nThe panoramic radiograph revealed a predominantly well defined multilocular radiolucency with sclerotic border and scalloped margins in the right mandibular body region. Antero-posteriorly, it extended from the mesial aspect of third molar region to the mesial aspect of first premolar and superior-inferiorly from the periapical region of the right mandibular posterior teeth to the inferior body of the mandible. Anterior to the multilocular radiolucency, an area of mixed radiopaque/radiolucency was noted extending from the mandibular right second premolar to the mandibular left lateral incisor crossing the midline. The entire lesion measured about 8×4 cm. Root resorption of lower anterior teeth and molars were noticeable (). Axial computed tomograph (CT) revealed a mixed radiolucent-radiopaque mass with well defined borders on the right mandibular body crossing the midline along with buccal cortical plate expansion ().\nBased on the clinical and radiographic findings a presumptive diagnosis of ameloblastoma or odontogenic keratocyst was made. Incisional biopsy was performed from the posterior and anterior regions which were submitted to the department of oral pathology. The biopsy from the posterior region was reported as a unicystic ameloblastoma of mural type and the biopsy from the anterior region was reported as a follicular ameloblastoma with acanthomatous changes. A hemi-mandibulectomy was performed for tumor resection under general anesthesia. The resected specimen measured about 9×4×2 cm (). The tumor was solid with few cystic areas, and the cut surface was slightly brown.\nMultiple histological sections were prepared from the resected mandible representing all the areas and were thoroughly analyzed. The main part of the tumor showed cystic lining of ameloblast-like cells, which were tall columnar, having peripheral palisading and hyperchromatic nuclei. Intra luminal proliferations of ameloblast-like cells were found in plexiform pattern (). The mural part of the tumor mass showed follicular and acanthomatous ameloblastoma, which coexisted with desmoplastic ameloblastoma ( and ). This association of desmoplastic ameloblastoma with follicular and acanthomatous patterns, was consistent with the features of so-called hybrid lesion of ameloblastoma (). The final diagnosis of 'Unicystic Ameloblastoma type 1.2.3 - luminal, intra luminal, and intra mural type' was made. |
A 42-year-old multiparous woman presented with episodes of vague abdominal discomfort predominantly on the left side. Per abdominal examination revealed an ill-defined lump in the abdomen arising out of pelvis stretching beyond the umbilicus more on the left side. On pelvic examination, a pelvic mass that was of around 14 weeks gestational size, immobile, slightly tender, more on the left side was palpable. Her last childbirth was 19 years ago and she had undergone a total hysterectomy for a 20 cm fibroid arising from isthmus of the uterus 6 years back. The hematological and biochemical investigations including CA125 levels were within normal limits. Chest X-ray was normal.\nUltrasonography revealed mixed echogenicity mass in pelvis extending bilaterally, predominantly on left side, bilateral adnexae could not be visualized separately from the mass, the uterus was not seen as she had undergone a total hysterectomy. There was no free fluid in the pouch of Douglas []. The upper abdomen was normal. Possibility of complex ovarian mass most likely cystadenoma of ovary was suggested and the patient was taken up for laparotomy. Peroperatively, a single nodule of 5 cm × 5 cm was also identified in the mesentery. The left ovary was enlarged and homogenous around 10 cm × 14 cm, posteriorly going into the retroperitoneum. Right ovary and tube were healthy looking. Tissue was taken for frozen sections from the retroperitoneum and ovary and possibility of a mesenchymal lesion was suggested. Subsequently, the nodules were excised and bilateral salphingoopherectomy was performed and specimen received in 10% formalin for histopathology. Grossly, the retroperitoneum, left ovary, and mesenteric nodules were gray-white, homogenous in appearance, the cut section showed a whorled gray-white appearance []. The right ovary and bilateral tubes were unremarkable. Several tissue blocks were processed and routinely stained for light microscopy examination.\nHistology revealed similar findings from the retroperitoneum, left ovary, and mesenteric masses; interlacing and intersecting fascicles of smooth muscle cells arranged in whorls and having an abundant eosinophilic fibrillar cytoplasm with focal myxoid and cystic change were appreciated []. Areas of hyalinization were also present. There was no atypia or necrosis. Histomorphology was suggestive of ovarian leiomyoma and leiomyomatosis peritonealis disseminata (LPD). On Masson trichome stain, the smooth muscle cells stained red and the hyalinized areas were blue []. The smooth muscle cells were positive for smooth muscle actin (SMA) on immunohistochemistry []. The smooth muscle cells were also positive for estrogen receptor (ER) and progesterone receptor (PR) which further supported the diagnosis. |
A 70-year-old female patient presented with the complaint of purple discolorations on the dorsum of the right foot, which had been present for 2 years and growing in number. Her past history was remarkable for an operation against colon carcinoma 10 years ago. She had not taken chemotherapy and radiotherapy. She also had goiter for 5 years. Dermatologic examination revealed multiple purple-colored macular lesions with diameters ranging from 0.5 to 1 cm on the dorsum of the right foot. Her serologic examination was negative for HIV-1. Systemic examination and laboratory tests were negative for a finding suggestive of systemic involvement of KS. Biopsies were taken from the lesions with an initial diagnosis of KS.\nThe patient was treated with local excision and cryotherapy for cutaneous Kaposi sarcoma lesions. The patient was followed at six month periods for a total of 11 months. During follow-up, local recurrences were detected in lower and upper extremities and treated with cryotherapy. No systemic involvement or an aggressive course was present during follow-up.\nH&E sections of the lesion revealed a vascular proliferation forming more than 90% of the lesion at the dermis, which had similar features with the first case. The lesion was composed of vascular structures filled by blood, which were lined by flattened endothelium and organized as lobular patterns, some of which included fibrous septa. Some vascular structures were closely spaced from skin adnexae that were surrounded by hemosiderin pigment. At the papillary dermis adjacent to the lesion were immature vascular structures intersecting collagen and extravasated erythrocytes, findings of the early-stage KS. No spindle cell proliferation was evident in the lesion.\nImmunohistochemical examinations of lesions from both cases showed a strong reaction with endothelial markers (CD31, CD34, and D2-40) and HHV-8 (LNA-1) both in endothelia of the cavernous hemangioma-like vascular structures and at the areas of early-stage KS. Endothelia of the mature vascular structures adjacent to the lesions had a positive reaction with Factor VIIIra whereas neoplastic vascular structures were negative. A focal positive reaction was present with actin and desmin at the walls of the cavernous hemangioma-like vascular structures. In the light of clinical, histopathological, and immunohistochemical findings, both lesions were interpreted as classical-type early-stage cutaneous KS, that showed features similar to cavernous hemangioma. |
An 86-year-old man presented with a pulsatile mass in his right leg that had persisted for two months. He had a history of hypertension, diabetes mellitus, hyperuricemia, and atrial fibrillation. He had been hospitalized for acute pancreatitis in the past. However, he did not have a history of relevant episodes of injury or infection. Clinical findings of Behcet, von Recklinghausen, and any other connective tissue diseases were absent.\nA physical examination revealed a pulsatile mass, about the size of a hen egg, on the lateral face of his right leg. All peripheral pulses were normal.\nComputed tomography (CT) angiography revealed a 3.2×5 cm aneurysm of the anterior tibial artery, another two aneurysmal changes (0.8×0.7 cm) on the distal side of the aneurysm, and patent peripheral arteries (). The aneurysm contained no mural thrombosis, and the posterior tibial and peroneal arteries were patent. No other aneurysms were evident.\nSurgery was initiated through a tibialis anterior lateral approach. The incision was made vertically about 10 cm above the aneurysm, and the tibialis anterior muscle and long extensor muscle of the great toe were divided. No adhesions had formed around the aneurysm, and the neurovascular bundle was confirmed. The deep peroneal nerve, located behind the aneurysm, was gently peeled away with careful manipulation. The aneurysm was opened, and the wall was removed. The artery was reconstructed using a reversed greater saphenous vein graft, which was proximally and distally sutured by an end-to-side anastomosis (). Postoperative peripheral pulses were good.\nHistological analysis of the aneurysmal wall revealed fibrous thickening of the intima and medial degeneration with calcified atheroma (). Arteritis or dissection was not observed. Thus, the mass was diagnosed as an atherosclerotic, true aneurysm.\nThe patient was discharged 10 days after surgery with no signs of ischemia or deep peroneal nerve disorder. Six months after surgery, the graft remained patent on CT images (). |
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 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 52-year-old Caucasian woman, gravida IV, para 2, abortus 2, with a 5-year history of uterine leiomyomas, presented in January 1999 with a pelvic mass of 87 mm at its greatest diameter at sonography. In July 2000, the mass had increased in volume, and in January 2001, the patient was admitted to hospital for a total abdominal hysterectomy with a bilateral salpingo-oophorectomy owing to uterine leiomyomas.\nAt laparotomy, the uterus was found to be three times the normal size (14 cm × 14.5 cm). In addition, a mass of about 30 mm in diameter located in the right laterocervical region was resected; this second mass was softer and less resistant to indentation than the leiomyomas inside the uterus. The ovaries appeared to be regular. The histological exam confirmed the presence of multiple leiomyomas of the corpus uteri and a right laterocervical leiomyoma. All of the specimens considered were positive for oestrogen and progesterone receptors. At 2-month follow-up, there was no evidence of disease. More than 10 years before admission, two incomplete abortions had occurred in the first trimester of pregnancy and had been followed by uterine curettages.\nAfter 3 years, the patient presented with abdominal bloating and occasional abdominal pain. A pelvic sonographic examination revealed a mass of 12 cm at its greatest point. In December 2004, magnetic resonance imaging was carried out. This revealed the pelvic mass to be solid and non-homogeneous, as evidenced by the presence of haemorrhagic areas, necrosis and vascular structures; the non-homogeneous nature was reinforced following the injection of contrast medium whereby non-homogeneous high enhancement became apparent. The mass was connected to the right pelvic wall via a vascular supply that appeared to originate from the iliac vessels in the obturator region. The rectum and bladder walls did not appear to be affected by the growth of the mass. A positron emission tomography-computed tomography (PET-CT) total body scan was performed, from which a pelvic localisation with low glucose affinity could be concluded. There were no pulmonary masses on pre-operative chest X-ray.\nIn order to remove the mass, a second abdominal intervention was performed, which revealed no evidence of tumour invasion in the pelvis or abdominal viscera. The mass measured 13 cm × 9 cm × 6 cm, was delimited by serosa, and when cut appeared to be myxoid and with oedematous areas inside.\nHistologically, spindle-shaped smooth muscle cells were present each containing a regular oval nucleus, without mitosis and with a moderate vascularisation and oedematous aspect. The cells were positive for smooth muscle specific actin, and there was a low proliferation index (2% of nucleus being MIB-1 positive). Again, the specimens expressed oestrogen and progesterone receptors (Figures to ). The final diagnosis was leiomyoma.\nIn light of the histological results and the lack of evidence to suggest immediate prescription of radiotherapy or chemotherapy, the decision was taken to withhold treatment and await observation at follow-up.\nA routine chest X-ray the following year, in May 2005, showed a nodular posterior basal density in the right lung of about 4 cm in diameter. The presence of a single nodular mass was confirmed at CT scan of the thorax (Figure ). At this point, a second PET-CT total body scan was performed, showing a pulmonary lesion with a low metabolic gradient, as is consistent with a benign lesion.\nIn June 2005, a right lower lung lobectomy was performed, during the course of which an intra-operative frozen section was also carried out. This failed to identify any malignant elements. A subsequent and more accurate histological examination permitted identification of the mass as a mesenchymal neoplasm with a smooth muscle differentiation, typifying a leiomyoma. Evidence to this effect was as follows: a low mitotic index of less than 1 mitosis per 10 high-power fields (HPFs); a low MIB-1 index of 1%; and a positive immunohistochemistry reaction for oestrogen receptors, progesterone receptors, H-caldesmon and desmins. Moreover, testing was negative for keratins, Bcl2, CD10 and CD99.\nA review of the histological pattern of the original mass and pulmonary mass showed low mitotic indices in both the pelvic mass (2 mitoses per 10 HPFs) and also the pulmonary mass (less than 1 mitosis per 10 HPFs) and that the histology of the masses was similar, typifying a leiomyoma (Figures , and ).\nOne month after the intervention, a course of chemotherapy prophylaxis was commenced using 80 mg per day of megestrol. At the 6-month follow-up, a CT scan of the thorax and abdomen was negative for masses; the 12-month examination was similarly negative. |
A 68-year-old asthmatic male patient presented to our center with 12 days history of melena. He denied any previous episode of melena or hematochezia or bleeding from another site. The patient did not have any other associated symptom, and had no other co-morbidities, or medication use. Upon referral, he was uncomfortable and looked pale. He was vitally unstable, with a blood pressure of 90/60 mmHg and a pulse rate of 120 beats/min. The examination revealed the presence of clotted blood on the anal verge, and some tarry stool on digital rectal examination. The hemoglobin level was 7.7 g/dl, the hematocrit was 22.8, and the blood urea nitrogen was 8 mg/dl. The prothrombin time and the partial thromboplastin time were normal.\nResuscitation was performed with transfusion of 2 units of packed red blood cells and intravenous fluids. He was admitted to the ICU for intensive monitoring. After admission and stabilization, upper and lower endoscopies were performed without demonstrating the bleeding site. They only revealed clotted and red blood throughout the colon.\nTechnetium-labeled red blood cell bleeding scan was done to localize the site of bleeding. This scan showed no evidence of early focal increased uptake in the abdomen to indicate active gastrointestinal bleeding during early images, but in the delayed images, it revealed that there was a focal uptake in the right and transverse colon. After that, capsule endoscopy was also performed without findings. As the angiography became available, the patient underwent selective angiography without findings noted at that time. These tests were inconclusive because they were performed while the episodes of bleeding ceased.\nAfter 8 days of conservative management and negative investigations to define the cause of the bleeding, a sudden drop in hemoglobin level from 10.8 mg/dl to 6.9 mg/dl occurred over 12 h, which mandated operative management. Exploratory laparotomy was performed. Extensive jejunal saccular pouches were found 10 cm distal to duodenojejunal junction extending 1.6 m distally Fig. . The bleeding was difficult to control and the decision to clamp the major branched was performed. Division of the small bowel proximal and distal to the diseased part using gastrointestinal stapler was performed with side to side primary anastomosis Fig. . The specimen was a part of small bowel, 117 cm in length, with congested wall and multiple pouches at the mesenteric site. Opening of the specimen showed normally looking mucosa with active bleeding that stopped after awhile. No polyps or masses were detected. We reviewed the angiography achieve after that and a suspicious shadow reflecting the diverticular outpouching was detected.\nMicroscopic examination of the specimen revealed many diverticula; some of which being true diverticula, while the others are devoid of muscularis propria (false diverticulae). Within the diverticula and in the intervening portions of the bowel wall, there were numerous dilated thick- and thin-walled small blood vessels in the submucosa. Additionally, submucosal intermediate-sized vascular clusters and feeder vessels in the muscularis propria and serosa were present. The overall features were those of small intestinal diverticulosis and arteriovenous malformations. The latter involves the diverticula and intervening portions of the bowel wall Fig. .\nPostoperatively, the patient was doing well, discharged home on day 5 postoperatively, with an uneventful postoperative course. He was followed up 2 years after that without complications. |
A 29-year-old male with a history of right tibial shaft and lateral malleolus fractures following a fall injury underwent open reduction and internal fixation with a locking plate for the fibula fracture and minimally invasive percutaneous osteosynthesis for the tibial shaft fracture at another hospital. Postoperatively, the patient was followed up at our hospital for 1 year due to residential issues, and the removal of the plate was scheduled after confirmation of bone union. According to the previous hospital’s medical records, a 9-hole locking plate (APIS®, Gwangju, Republic of Korea) was used for the distal tibia, with three 5.0-mm locking screws for proximal fixation and six 3.5-mm locking screws for distal fixation. Under spinal anesthesia, a plate removal surgery was performed, following which the fibular plate was easily removed. However, five of the six distal locking screws were damaged, and they could not be removed with a screwdriver. This situation was explained to the patient during the operation, and we could confirm that the patient wanted to remove the plate irrespective of the amount of time required. At the time, a tourniquet was used for approximately 1 h, and screw removal was attempted using a conical extraction screw, but only one of the six screws could be removed. Previously proposed techniques requiring additional skin incisions were not considered because there were multiple damaged screws that could not be removed, these techniques would require extended amount of time for removal, and the surrounding soft tissues could be damaged. Hence, we decided to use a screw extractor (IRWIN®, Huntersville, NC, USA), a non-medical instrument, following sterilization with ethylene oxide. In brief, 6-mm drill bit of the extractor was used, four screw heads were drilled, the locking screws and locking plate were separated, and the plate was removed. Of the remaining four screws, one was removed using a vice grip; the remaining three screws could not be removed with the vice grip owing to a short extruded portion. A hollow reamer could not be used because the three screws were adjacent to each other and the reamer could cause a large bone defect; hence, we created sufficient space around the screws by forming small holes around them with a 1.8-mm K-wire, following which all screws could be removed using the vice grip. After saline irrigation, debridement of the soft tissues contaminated with metal debris was performed. The sizes of the metal debris were found to be relatively large, which facilitated easier debris removal. With 2-h use of a tourniquet, all implants could be removed and the skin could be closed (). Postoperatively, no complications were noted during physical and radiological examinations. Remarkably, no problems were noted at the final outpatient visit at 3 months after discharge; therefore, follow-up was terminated. The patient had not visited an outpatient clinic for more than a year since the last follow-up, thus we thought that he had no specific complications including infection and re-fracture. |
Our patient was a 70-year-old man with severe dysphagia as a result of accidental ingestion of sulfuric acid while intoxicated when he was roughly 30 years old. He was initially treated by subtotal esophagectomy with gastric tube–cervical esophageal anastomosis. Severe dysphagia continued afterwards, and in 2005 (at the age of 58 years) he visited the Department of Surgery of Nakagami Hospital. Despite undergoing endoscopic esophageal dilatation several times every year, his symptoms did not improve, and he underwent contracture release and PMMC flap transfer in 2009 (at the age of 62 years). He also underwent periodic endoscopic esophageal dilatation thereafter (up to five times per year). He was referred from the department of surgery to the department of plastic surgery in May 2016. On examination, there was a surgical scar on his neck, and a scar where the right PMMC flap had been harvested. There was also a scar in the center of his abdomen from the first operation, as shown in . He complained of extreme difficulty in swallowing, leading him to squeeze his neck manually to pass food. Endoscopic examination by an otolaryngologist showed no major problems in his larynx, and it was considered that the food that he consumed entered the gastric tube via the PMMC flap from the right piriform sinus. It was impossible to advance the endoscope forward due to the severe stenosis. Three-dimensional multi-detector CT (MDCT) and VE were performed. MDCT was performed using a 64-slice CT apparatus (Aquilion 64; Toshiba Medical Systems Corp., Otawara, Japan). The technical parameters were 120 kVp, 150 mA, collimation of 64×0.5, a rotation time of 0.5, a slice thickness of 1 mm, and an increment of 1 mm. On VE, the stenosis was about 2–3 cm in length. A contrast swallowing examination also showed a similar result, as shown in . Based on the VE findings, we decided to utilize a patch graft, for which we expected that the luminal mucosa could be used to some extent. Thus, surgery was planned to release the stenosis and replace it with a FJPG.\nIn August 2016, a joint operation took place. A U-shaped skin incision was made in the neck. The transplanted right PMMC flap was dissected. Using a nerve-stimulating device, peeling of the flap and esophageal stricture was performed. Ultimately, the PMMC flap was excised, and the leftover epithelium remained in the lumen. As shown in the preoperative VE image of the right piriform sinus, pinhole-like stenosis was observed.\nThe stenosis was incised and expanded sufficiently with fingers on both the cephalad and caudal sides. The right piriform sinus remained only on the left wall (inner wall), and it was decided to reconstruct the three sides of the front, the rear, and the outer walls with a FJPG. The defect length was about 5 cm, in a spindle shape. The harvested jejunum was split in the longitudinal direction, and was divided into a patch graft and a monitoring segment. The FJPG was transferred in a 90°-transposed fashion. The third jejunal artery and the right transverse cervical artery were end-to-end anastomosed with 9-0 nylon sutures. The third jejunal vein and the right internal jugular vein were end-to-side anastomosed with 9-0 nylon sutures.\nThe flap survived completely. Videofluorography was performed at 3 months postoperatively, and good passage was confirmed, as well as at 2 years postoperatively. There were no complications, such as an abdominal wall hernia. The patient was able to enjoy meals and his body weight increased by 10 kg. Upper gastrointestinal endoscopy was performed at 2 years and 3 months after surgery. Superficial gastritis was found in only a part of the gastric tube, and no evidence of stricture or cancer was seen, as shown in . |
A 75-year-old male with past medical history of paroxysmal atrial fibrillation status post cardiac pacemaker, hypertension presented to urgent care clinic with several weeks of low-grade fever, cough, and shortness of breath. He was discharged on augmentin for possible pneumonia as there was a questionable retrocardiac infiltrate on chest X-ray. Three days later, he presented in the ED with complaints of acute dyspnea in addition to fever, cough, and fatigue for one month. His initial vitals were blood pressure 127/54 mmHg, pulse rate 77 beats per minute, respiratory rate 20 breaths per minute, temperature 100.3°C, and an oxygen saturation of 100% on room air. He was alert and oriented and physical examination was significant for an unexplained murmur. He was admitted on medical floors; lab workup was sent including blood cultures and urine cultures. The infectious disease department was consulted for fever of unknown origin and augmentin was discontinued. The initial differential diagnosis included endocarditis, occult malignancy, and recurrent pulmonary emboli (patient's son recently sustained a pulmonary embolism). On day two of admission, 1/2 sets of blood cultures were positive for Enterococcus faecalis, then two more subsequent sets were positive. The patient was started on ampicillin plus ceftriaxone, gentamicin for six weeks. The organism was later found to be sensitive to rifampin, so this agent was also added. Trans-thoracic echocardiogram was done which showed severe aortic valve regurgitation. Trans-esophageal echocardiogram was also done which showed a 1 cm x 1 cm mass on the aortic valve. When the bacteremia resolved, he was discharged on ampicillin, ceftriaxone, rifampin for six weeks, and gentamicin for two weeks. \nTwo days after discharge, the patient presented again to the ED with a near syncopal event and worsening shortness of breath. Lab work was significant for hypomagnesemia. Brain-natriuretic peptide (BNP) was also elevated and chest radiograph showed small bilateral pleural effusions suggestive of acute pulmonary edema/heart failure. He was treated with Lasix. The following day, the patient had an episode of self-limiting torsades (Figure ).\nHe was given IV magnesium and was started on amiodarone, which was later changed to a lidocaine drip. At this point, he was transferred to the critical care unit and cardiothoracic surgery was consulted for aortic valve replacement and a coronary angiogram was scheduled. Cardiac catheterization showed multi-vessel disease. The patient underwent aortic valve replacement, coronary artery bypass graft for triple-vessel disease, ligation of left atrial appendage, and evacuation of the bilateral pleural effusions. His post-op course was complicated by increased chest tube drainage for which subsequent exploration was done. The patient was given prothrombin complex concentrate and blood products. He required continued intubation and vasopressor support for hemodynamic stability. Eventually the patient was weaned off the vasopressor support, urine output improved but he developed elevated liver enzymes, probably due to hypoperfusion to the liver for an ultrasound did not reveal any biliary obstruction. After a day he again developed hypotension and was restarted on vasopressors and laboratory assessment revealed leukocytosis. Surgery and gastroenterology were consulted for pancolitis evaluation. No intervention was recommended as patient was not stable enough; he was started on fluconazole for Candida (sputum and urine culture grew Candida) and flagyl for possible pancolitis. The patient started to have worsening liver function tests; cholecystostomy tube was placed. The patient’s hospital course continued to decline and realizing his poor prognosis, the family decided to withdraw all care. He subsequently died of cardiopulmonary arrest. |
A 63-year-old male presented to the emergency ear, nose and throat (ENT) clinic complaining of bilateral ear discharge, more pronounced in the right ear, associated otalgia, and hearing loss for several months. The patient had no other medical problems.\nThe patient was treated in primary care with a seven-day course of amoxicillin. Pain and hearing loss persisted and thus the patient was referred to the otology department.\nOn initial examination, a cotton wool bud was found in the right EAC and removed via forceps. Following microsuction of the right and left EACs, evidence of bilateral otitis externa with a right ear canal polyp was seen. The patient was prescribed a 10-day course of betnesol and ciprofloxacin drops.\nOn review two weeks later, the patient continued to complain of a mild degree of hearing loss and associated non-pulsatile tinnitus. On examination, the right EAC was observed to be narrowed and a polyp was still present. The left otitis externa had resolved. An audiogram showed a mixed conductive and sensorineural hearing loss on the right side and mild high-frequency hearing loss on the left. Due to the chronicity of the patient's symptoms, a CT scan of the temporal bones was completed. This demonstrated: chronic bilateral thickening of the EACs, right middle ear inflammatory disease, and subtle ossicular erosions (Figure ). The patient was listed for examination plus biopsy under anaesthesia to better delineate the cause of his ongoing symptoms.\nUnder anaesthesia, thick inflammatory tissue was noted arising from the inferior suture line and anterior external auditory canal wall. The pars tensa was just visible posteriorly. Multiple biopsies were taken and sent for histology and microbiology. The right ear was packed with two pieces of bismuth iodoform paraffin paste (BIPP) packs.\nOn review in the clinic, the packing was removed, revealing that the right EAC was still filled with polypoid material and thick pus. The microbiology results confirmed Pseudomonas aeruginosa and Serratia marcescens. Histology showed a hyperkeratotic and squamoproliferative lesion showing moderate-severe dysplasia. The ear was re-packed with Tri-Adcortyl soaked ribbon gauze (active ingredients: triamcinolone acetonide, neomycin sulphate, gramicidin, and nystatin) and the patient was re-prescribed ciprofloxacin and started on flucloxacillin as per sensitivities and listed for deep biopsies of the right ear under general anaesthesia.\nOn examination, no significant bony erosion was seen. A very thick and inflamed EAC epithelium and a thickened tympanic membrane were observed again. The tympanomeatal epithelium was fully excised down to the bone of the EAC and circumferentially to the edge of the annulus. Histological analysis showed skin hyperkeratosis and parakeratosis with epithelial budding. Focally, deep keratin pearls were identified. The dermis was fibrotic and minimally inflamed. These findings were consistent with regressed/regressing keratoacanthoma.\nOver the next three weeks, the patient's ear discharge settled without further treatment. The patient continued to be under regular review in the outpatient ENT clinic and the initial presenting symptoms did not return over the period of a year. and Subsequently, he was discharged back to primary care. |
A 75-year-old woman with a 60-year history of tobacco use was noted to have a right hilar mass on a routine chest X-ray. Computed tomography (CT) scan and magnetic resonance imaging (MRI) demonstrated a 4.5 cm well defined mass with slightly lobulated margins present between the upper and lower lobes of the right lung posterior to the right hilum (Figure ). The mass abutted the right main bronchus, the bronchus intermedius and the medial pleural surface. Although the visual impression of the mass radiologically suggested a solid mass, attenuation within the mass (approximately 24 Hounsfield Units (HU)) was less than in the aorta containing blood (approximately 35 HU) and skeletal muscle (approximately 46 HU) indicative of a cystic rather than a solid lesion (Figure ). There was no associated adenopathy or obvious compression of the airway or other structures. Positron emission tomography (PET) scan showed increased uptake in the thyroid gland which on follow-up ultrasound and fine-needle aspiration was diagnosed as multinodular colloid goiter and thyroiditis. Additional work-up was negative for metastatic disease. The patient was minimally symptomatic with a mild cough productive of yellow sputum, but with no shortness of breath or evidence of hemoptysis. The pulmonary mass was felt to represent a primary tumor and the patient underwent subsequent thoracotomy with right upper lobectomy and segmentectomy of the right lower lobe. At follow-up 2 years later, she had no evidence of recurrence or metastatic disease.\nOn pathological examination, the specimen consisted of resected right upper lobe of lung with attached superior segment of right lower lobe. The pleural surface showed a retracted, indurated area 4.5 × 3 cm close to the bronchus intermedius of the upper lobe. On sectioning, a 5.5 cm nodular cystic mass with a distinctly mucoid appearance (Figure and ) was seen.\nMicroscopically, the tumor, although situated adjacent, did not have any relationship with the bronchus, was well-demarcated from the surrounding lung, and had a multicystic appearance without any distinct solid areas (Figure ). The cysts contained abundant pale basophilic mucin and the cyst lining showed a spectrum of appearances. In some areas, the lining consisted of simple columnar mucinous epithelium, with frequent goblet cells. The nuclei were regular, basally oriented; without stratification, atypia or mitoses (Figure ). In other areas, there was significant tufting, stratification and hyperchromasia of nuclei coupled with pleomorphism and mitotic activity (Figure ). At the periphery of the tumor, there was evidence of invasion into surrounding parenchyma in a 'lepidic/bronchioloalveolar' pattern. Mucinous tumor cells appeared to line alveoli in a patchy fashion while respecting the alveolar septal framework (Figure ).\nImmunohistochemically, the tumor cells showed strong positivity for Cytokeratin 7 (CK7) (Figure ). Cytokeratin 20 (CK20) was focally positive (Figure ). Thyroid Transcription Factor-1 (TTF-1) and p53 were negative and cell proliferation marker MIB-1 showed a low positivity index (< 10%). |
A 20-year-old female presented to our center with a telangiectatic osteosarcoma of the humerus. There was no history of malignancies within the family.\nMRI examination and open biopsy were performed by the previous physician. At the time the results were thought to be malignant lymphoma. After several diagnostic trials, the patient visited our center to seek advice about her telangiectatic osteosarcoma (A).\nWe observed a circumferential mass on the distal part of the right arm with a slight deformity of the arm, with marked venous engorgmnt and distal edema. The mass was warm and solid on palpation. Function of the right hand was still preserved. From the laboratory findings, there were marked elevation of the alkaline phosphatase and lactate dehydrogenase. From humerus X-ray, there was mixed lesions along the humerus with pathological fracture on the midshaft (A). T2-weighted MRI showed iso-hyperintense and expansile lesions along the humerus (A). For the metastatic workup, chest X-ray showed no coin lesions or metastatic characteristics. Patient also had a PET scan and the result was unremarkable. From the Clinico Pathological Conference (CPC) forum, it was concluded that the diagnosis was osteosarcoma of the right humerus stage IIB according to the Enneking classification.\nPatient took neoadjuvant chemotherapy regimen with Doxorubicin and Cisplatin for three cycles from January to March 2016. At the end of the third cycles, clinical and radiological evaluations were performed. Clinically the mass was not getting bigger compared to before chemotherapy (B). From x-ray, the mixed lesions became more marked compared to the previous x-ray (B). MRI showed that the mass was slightly became larger compared to the previous MRI with no involvement of neurovascular bundle (B).\nSix months after the initial complaint, the surgery was performed. We used extensive deltopectoral approach with anterolateral extension through the proximal part and curved backward to complete the posterior distal humerus and elbow approach. This approach was used because of the previous biopsy tract was in the posterior aspect of the distal humerus. The vascularity, rotator cuff tendons, biceps and triceps muscle, and majority of the nerves (musculocutaneous, radial, median, and ulnar nerves) were spared during the total resection of the humerus. Axillary nerve was sacrificed during the tumor resection. To reconstruct the humerus, both long shoulder hemiarthroplasty and total elbow prostheses were used. These two prostheses were joined using two long one-third tubular plate that worked as the long stem augmentation. Extension cerclage wire was used to make the implant as one unit (A). Finally, the stem was augmented with a bone cement from proximal through the distal. Prolene mesh then was sheathed to the bone cement, and then the preserved rotator cuff tendons and biceps and triceps muscle were sutured back with the Ethibond sutures (B). The resected tumor and humerus were then sent to the lab for histopathologic analysis (C).\nFrom the post-operative x-ray, the modified prosthesis sat well on the shoulder and elbow joint (). Function of the hand was excellent post-operatively. Patient also could immediately flex her elbow. Post-operative histopathological examination showed telangiectatic osteosarcoma with HUVOS IV (). After the wound healed without complication, patient underwent adjuvant chemotherapy. Two months after the surgery, patient could start writing with her right hand without marked difficulties. Further follow up of three years post-operatively, patient already came back to work and were able to perform daily activities without difficulties. Patient’s shoulder abduction and elbow flexion was shown on the pictures (). The MSTS functional score for the upper limb scored 83% which was excellent. There were no post-operative complications and the immunohistochemical workup used to rule out lymphoma (CD20, CD15, CD 30, and CK) would be planned. |
A 47-year-old man was admitted to hospital with a 9 day history of abdominal pain and diarrhoea and an incidental fluctuant swelling on his back. He had a previous medical history of alcoholic liver cirrhosis, anaemia and hypothyroidism. Examination revealed a fluctuant tender swelling on the left side of his back extending to the inferior angle of scapula. His blood tests demonstrated raised inflammatory markers (WCC 12.3, CRP 169, normal LFTs). On CT he was found to have a large left pleural enhancing collection with adjacent rib destruction corresponding to the clinically palpable mass. At that time note was made of incidental mild acute cholecystitis (Fig. ). The patient required rib resection, drainage of his pleural empyema and antibiotics.\nOn day 7 post op he suffered a massive fresh PR bleed. Hb measured 61 g/L having been 103 g/L on admission. There was no evidence of coagulopathy and liver function tests were unremarkable. The patient was treated with blood transfusion, FFP and tranexamic acid. Flexible sigmoidoscopy revealed no cause for fresh PR bleeding. Triple phase CT was performed. The pre-contrast images demonstrated layering of variable density fluid in the gallbladder with the hyperdense fluid also within the colon at the hepatic flexure, which was located adjacent to and inseparable from the gallbladder (Fig. ). On the post-contrast images there was a blush within the gallbladder in keeping with a cystic artery pseudoaneurysm (Fig. ). The patient was transferred to IRU where DSA confirmed the cystic artery pseudoaneurysm (Fig. ). This was embolised with a single 2 × 3 mm Tornado coil (Cook Medical) (Fig. ). Ideally the coil would have been deployed more distally relative to the origin of the cystic artery, however despite prolonged attempts, the microcatheter would not advance distally and so the coil was deployed as demonstrated.\nFollowing embolization, the patient was transferred to ITU but suffered no further episodes of bleeding. Repeat CT scan demonstrated successful embolization of the cystic artery pseudoaneurysm. The patient was stepped down to the ward 2 days later and discharged 1 week after embolization on oral antibiotics for his pleural empyema with a surgical outpatient appointment regarding future cholecystectomy. |
A 69-year-old female with a pertinent medical history of congestive heart failure with preserved ejection fraction, liver cirrhosis, seizure disorder, and rheumatoid arthritis (not on treatment) presented to the emergency department due to worsening shortness of breath. This shortness of breath was worse on exertion, was associated with tiredness, dry intermittent cough several times a day lasting for a few minutes, and an increase in her baseline abdominal distension. The dyspnea had no relation to changes in her position, did not wake her up from sleep, and further denied any day time somnolence or snoring at night. She, however, reported that for the past month her symptoms have progressively gotten worse and hence she came to the ED.\nOn the initial assessment, her vital signs revealed tachycardia and oxygen saturation of 87% for which she was placed on 2 liters of oxygen via nasal cannula. Her physical examination revealed a distended abdomen, crackles were audible at the base of her lungs bilaterally and her lower extremities showed traces of edema bilaterally. Her initial electrocardiogram (EKG) showed sinus tachycardia, her echocardiogram was performed and it revealed normal left ventricular ejection fraction, mild tricuspid regurgitation and severe pulmonary hypertension with the right ventricular systolic pressure estimating to be 66 mmHg. The X-ray done showed bilateral opacities and the CT of her chest (Figure and Figure ) was suggestive of interstitial lung disease with interstitial thickening, bronchiectasis, and honeycombing. The pulmonary team saw the patient, suggested a rheumatologic workup for the patient to further investigate for a cause of the ILD. The following day, her labs came back and her antinuclear antibody (ANA) was found to be elevated with a ratio of 1:2560, her rheumatoid factor was elevated to >650, and her cyclic citrullinated peptide (CCP) antibody was strongly positive to >250 (Table ). These results increased our suspicion of rheumatoid arthritis-associated ILD. The rheumatology team also saw the patient and requested laboratory studies to prepare her for a steroid-sparing agent, to be started as an outpatient. The results revealed that her centromere antibody was elevated as well. The patient was started on prednisone 60 mg daily and bronchodilators. The patient's hypoxia improved, she was discharged and she was recommended to follow up with a pulmonologist and a rheumatologist as an outpatient. |
A 47-year-old woman with a history of known gallstone disease presented to the office with worsening post-prandial right upper abdominal pain radiating to the back, abdominal bloating, and nausea. Her clinical history was significant for hypertension, hyperlipidemia, and tubal ligation. Her vitals were within normal limits, and physical examination showed mild right upper quadrant tenderness without rebound or Murphy’s sign. Laboratory studies including complete blood cell count, liver function tests, and coagulation panel were within normal limits. An ultrasound of the abdomen confirmed the diagnosis of cholelithiasis (Figure , ). There was no indication of an AL on the ultrasound images. After a detailed discussion about the biology, natural history, and management options of gallstone disease, including risks and benefits of operative vs non-operative approaches, a decision was made to proceed with an elective laparoscopic cholecystectomy. During the laparoscopic exploration of the Morrison’s space, an approximately 2 cm ellipsoid structure similar in color and consistency to the liver was found attached to the serosal surface of the anteromedial wall of the gallbladder (Figure , ).\nThis finding was consistent with the diagnosis of an AL with its own vascular pedicle. After achieving the critical view of safety, the cystic duct, cystic artery, and the vascular pedicle of the AL were secured with laparoscopic endoclips and transected. The AL was dissected en block with the gallbladder off the liver bed. The patient tolerated and recovered from the procedure well, and she was discharged to home on the day of surgery without complications.\nIn addition to several choleliths, gross examination of the gallbladder revealed a 1.4 x 0.9 x 0.4 cm encapsulated fragment of brown soft tissue resembling liver tissue on the serosa (Figure ). Histologic examination demonstrated characteristic liver architecture including liver cell cords and sinuses and defined portal tracts containing all three major structures: portal venule, hepatic arteriole, and interlobular bile duct (Figure ). The histologic findings supported the diagnosis of an AL. Of note, the AL was located 0.1 cm from the true liver bed of the gallbladder. |
A 24-year-old Caucasian male with no significant past medical history presented to the emergency room with a complaint of sudden onset double vision and headache with worsening intensity associated with non-bloody emesis for four days. He described his double vision as seeing two images of a single object and unable to move his eyes outward bilaterally. He related his headache as throbbing in character, severe in intensity, and diffusely involving his entire skull with radiation down his neck. He also noted worsening of his headache with cough and Valsalva, associated with pulsatile ringing in the ears.\nVital signs were within normal limits. Neurological examination revealed bilateral abducens nerve palsy. He underwent a dilated fundoscopic examination which did not show any evidence of papilledema. Computed tomography (CT) of the head was done, which showed an extensive thrombus from the proximal portion of the right internal jugular vein extending into the right sigmoid sinus, right transverse sinus, following into the confluence of sinuses and extending into the superior sagittal sinus (Figure ). Magnetic resonance venography (MRV) showed the absence of blood flow in the superior sagittal sinus (Figure ). He underwent a CT of chest/abdomen/pelvis, which did not show any evidence of malignancy. There were no signs of sinusitis, sepsis, head injury, or mechanical precipitants. He has shown no indication of systemic or intracranial infection during this admission; therefore, a lumbar puncture was not done.\nHe was admitted to the hospital for control of his headache and nausea, which improved with intravenous (IV) analgesics and antiemetics. He was started on IV heparin, transitioned to warfarin and enoxaparin during his hospital course, and was subsequently discharged home on warfarin and enoxaparin. He had extensive hypercoagulable workup, which was pending at the time of discharge. The results for factor C, factor S, factor V Leiden, homocysteinemia, and antiphospholipid antibody were negative except for heterozygous mutation of the prothrombin G20210A gene. D-dimer was not checked in our patient, but when elevated, it is supportive of the diagnosis. However, normal levels do not rule out the diagnosis. This is in contrast to other causes of venous thromboembolism where d-dimer tends to have a very high negative predictive value. The patient had a repeat CT of the head with contrast in seven days, which showed dissolving cerebral venous thrombosis (Figure ). His diplopia and headache improved at the time of a repeat CT scan. He was able to move eyes in all directions, still had a constant headache for which was started on topiramate. On a month follow-up, his vision continued to improve and his headache resolved. |
A 19-year-old black female presented with painful lower lip swelling of 2 weeks duration. She was initially seen by a physician whom she claimed had given her anti-allergic medication and painkillers in the form of tablets for 1 week, but the situation worsened and the swelling and pain increased. She was then prescribed intravenous penicillin for another week with no improvement, after which she presented to our hospital for assessment and further management. There was lower lip swelling with discharge from the exfoliated crusted lower lip surface. The condition had started as a small pimple on her chin just below the lower lip on the left side which was painful and increasing in size. She attempted to open it and did not use any antibiotic. After 48 h she became febrile and her lower lip became swollen. She went first to a physician, then presented to our dental hospital. The patient’s medical and family history was non-contributory, and there was no history of trauma, hospitalization or current medication except those prescribed by the physician. She was lactating and had a 6 month old boy.\nThe patient’s oral cavity was examined carefully and good oral hygiene was noted. There were no obvious odontogenic causes or lip paraesthesia. Panoramic radiograph was done to exclude odontogenic causes and was insignificant. A thorough facial examination followed and a small crusted pimple was noticed on the left side of the chin. There was extensive swelling involving the whole lower lip and extending to involve the chin and submental area (Fig. ) with pus discharging from the left side of the chin just beneath the site of the pimple (Fig. ). The patient’s blood investigations were negative for HIV and hepatitis, with a TWBC of (25.8×), random blood glucose of 190 mg/dl and a creatinine of 0.7 mg/dl. She was anemic (10 g/dl), mildly hyponatremic (134 mmol/l) and hypokalemic (2.5 mmol/l). Potassium correction was started within travenous potassium chloride at 10 meq/l over 4 h for 1 day, after which she was shifted to diet supplements. Normal saline 500 ml/day was given for 2 days, as well as ferrous sulphate tablets. Electrolyte and blood levels were back to normal after 6 days (K = 3.5 mmol/l, Na = 137 mmol/l and Hb = 12 g/dl.\nBased on the history and clinical presentation, the diagnosis of necrotizing fasciitis was made and a swab of the site was taken for culture and sensitivity. Treatment started with lower lip debridement and exploration where necrosis of the middle part of the lower lip was noted. A segment of necrotic fascia on the left side of the chin was found and removed with preservation of the overlying skin except over the acne-affected area where skin was removed (Fig. ). Multiple mental and submental incisions were made for drainage. Irrigation with normal saline was done and corrugated rubber drains were inserted and secured. The patient was admitted to the hospital where intravenous fluids and an empirical triple antibiotic regimen were started immediately (Ceftazidime 1Gm/day, Metronidazole 500 mg 8 hourly and Gentamycin 80 mg/day), and continued after culture and sensitivity results revealed no microbial growth. After 3 days, further tissue necrosis was noticed, so a second debridement was done with consequent tissue loss over the middle upper surface of the lower lip. Additionally, irrigation with hydrogen peroxide followed by normal saline wash was performed. Ten days later, dramatic improvement was seen, where only the Gentamycin was stopped and irrigation was continued with normal saline and topical application of Tetracycline ointment for 7 days. The drain was removed on day 19 and the patient was discharged on the 20th day on oral antibiotics for another week. On follow up, the wound showed good healing by secondary intention and there was no need for reconstruction (Fig. ). |
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 68-year-old male was admitted to the clinic. The patient had a history of a fall two weeks prior to the admission to the clinic, although no direct external force had been applied to the head and neck during the fall. The patient complained of numbness in the forearms and hands, tingling sensations and a lack of power when clenching the fists. Through the physical examination, it was observed that the flexion and extension range of the patient’s body and neck were normal and that there was no sign of tenderness. The flexion and extension strength of the patient’s elbow muscle was of Grade III, there was numbness and hyperalgesia in the bilateral forearm and hand, the grip of the patient’s two hands was weak, the bilateral Hoffmann sign was (+) and the lower extremity muscle strength was graded as Class IV. Furthermore, the muscle tone was normal, the tendon reflex was (+++), the patellar clonus was (−) and the ankle clonus was (−). Cervical magnetic resonance imaging (MRI) examination revealed that cervical disc degeneration was apparent at C5–6 level with backward extrusion, and that there were high signal changes within the same level of the spinal cord. Mild dural compression was observed in the cross section (). According to the study by Nagata et al (), the degree of spinal cord compression may be classified into one of four groups: Level 0, no pressure on the spinal cord; Level 1, mild compression on the spinal cord; Level 2, the degree of spinal cord compression is <1/3; Level 3, the degree of spinal cord compression is >1/3.\nIn this case, the degree of spinal cord compression on the MRI imaging was classed as Level 1, i.e. only mild compression; however, T2-weighted (T2W) MRI examination revealed that there were high signal changes in the spinal cord. Therefore, although the clinical manifestations and symptoms indicated damage to the central beam of the spinal cord, this was not consistent with the mild degree of compression. Laboratory examinations revealed that the level of serum VB12 was 173 pg/l, which was below the normal range (211–946 pg/l). It is possible that VB12 deficiency may be the cause of spinal cord injury in this patient in whom there is only a mild degree of spinal cord compression.\nThe patient underwent anterior cervical decompression-fusion and internal fixation (), at the same time as receiving three months of oral VB12 replacement therapy. One week later, the tingling feeling in the patient’s hands was significantly improved and his fist-clenching ability was restored and two weeks later the patient’s muscle strength reached Grade III; three months subsequently, the numb feeling in the forearms and hands of the patient had almost disappeared, the patient’s finger activities had returned to normal and the fist muscle strength had recovered to Class IV. |
A 40-year-old female, with 3-month history of nasal obstruction and tinnitus was admitted in August 2012. Nasopharyngeal endoscopy and biopsy already had been performed in another hospital, showing nonkeratinizing undifferentiated NPC. This histopathologic diagnosis was confirmed in our center. Magnetic resonance imaging (MRI) of the nasopharynx and neck revealed the tumor was confined to the nasopharynx and the bilateral locoregional cervical lymph nodes enlarged with its greatest dimension of 2 cm. Chest computed tomography (CT) scan, ultrasound of abdomen, and whole-body bone scan ruled out distant metastases. So clinical staging was determined to be T1N2M0, IIIA according to American Joint Committee on Cancer TNM Staging System for NPC (7th ed, 2010).\nThe patient was treated with definitive IMRT to 7050 cGy for primary tumor and 6600 cGy for infiltrated regional lymph nodes. Concurrent chemotherapy based on cisplatin and 5-flurorouracil was administrated for 2 cycles and then 2-cycle chemotherapy was given subsequently to consolidate the efficiency with the same regimen. At the end of therapy, she obtained clinical complete response by nasopharynx and neck MRI.\nIn the initial therapy, the patient had undergone abdominal ultrasonography for 4 times, and no hepatic lesions were noted during this period. Nevertheless, when she came to our hospital for 3-month conventional follow-up in April 2013, abdominal sonogram revealed a liver cystic lesion with thin wall and smooth margin of 18 × 16 mm in the right liver lobe, and the lesion was interpreted as a simple liver cyst (Fig. ). Therefore, intense follow-up was suggested. Five months later, the cystic lesion enlarged to be 59 × 46 mm, with thick wall, but no signal of blood flow. Further CT presented a low density and heterogeneous lesion taking irregular wall and incomplete septa with strong contrast enhancement, indicating liver abscess (Fig. ). However, the patient was asymptomatic, with no fever, no right up abdominal pain, and no palpable mass by physical examination. On the contrary, laboratory findings were negative, liver function was normal, the white blood cell count, C-reactive protein, and cancer-related antigen including α-fetoprotein were within the normal limits. Afterward the patient was transferred to another hospital for abscess drainage, and fluid culture was negative, but fine-needle aspiration was not performed. After drainage, the mass shrank significantly to one-third of the original size. However, it returned to previous size by CT 2 months later. Thus, the liver lesion was suspected to be malignant. Work-up examination including chest CT, pelvic MRI, emission CT for bones, gastroscopy, colonoscopy, and brain MRI excluded other lesion that may account for another primary tumor or extra-hepatic distance metastases from NPC. In addition, nasopharynx MRI showed no evidence of local relapse. On November 21, 2013, surgical resection was administrated both for histologic diagnosis and treatment, and the surgical margin was negative. Histopathologic examination definitely confirmed that the metastasis originated from NPC, since the cells of the surgical segment were similar to primary NPC on the morphology and they were positive in Epstein–Barr virus (EBV) encoded RNAs (EBERs) (Fig. ). No adjuvant chemotherapy was done after resection of the liver metastasis.\nHowever, relapse-free survival time lasted only for 8 months. In July 2014, 2 small cystic lesions were found on abdominal CT scan again, which were extremely similar to simple cysts (Fig. ). Further contrast-enhanced ultrasonography provided no sign of “fast in and fast out,” that is, a characteristic appearance in malignant carcinoma. As a result of multidiscipline team discussion, “watch and wait” strategy was recommended. However, the number of hepatic lesions increased to 4, and the size of previous 2 cysts enlarged to be 3 cm within 3 months. The multidiscipline team members reached a consensus that the cystic lesions were metastases from NPC and suggested a palliative systematic chemotherapy. Then the patient received chemotherapy with paclitaxel and cisplatin for 6 cycles. During the first 3 cycles, partial lesions diminished in size, but stayed stable within the later 3 cycles. Nevertheless, the intraliver metastases exhibited significant progress soon after the chemotherapy. Subsequently, she underwent chemotherapy with gemcitabine plus targeted therapy with nimotuzumab, then single navelbine both for 3 cycles, but neither protocols showed notable effects on hepatic lesions. In addition, during the phase of chemotherapy with navelbine, the patient complained of chest pain, an irregular and fixed lump was found on her chest wall, approximately 3 cm in diameter. After local surgical resection was performed, the lump histologically was demonstrated to be an extra-hepatic metastasis from NPC. Considering the patient's good performance status score and grade A liver function according to the Child-pugh grading system, transcatheter hepatic artery chemoembolization (TACE) was administrated for 5 times from December 2015 to April 2016. After the TACE treatment, all the lesions were obviously decreased in size, with the largest metastasis decreasing from 64 × 53 to 33 × 32 cm (Fig. ). Meanwhile, carbohydrate antigen 125 (CA125) decreased from 125 to 60.8 U/mL, and squamous cell carcinoma antigen decreased from 2.8 to 0.9 ng/mL. Then 4 cycles of gemcitabine plus cisplatin were offered to consolidate the clinical effects. In the next 6 months, her liver metastases maintained stable, but lung metastases were noted. In October 2016, the CT showed the liver metastases progressed. At last, she died of liver failure in March 2017. |
A 70-year-old woman presented at our outpatient department with primary glenohumeral osteoarthritis at the right side \n[]. She had osteoarthritis in multiple joints and underwent previously a bilateral total knee replacement. General medical history revealed diverticulitis, mild chronic obstructive pulmonary disease (COPD), and atrial fibrillation. Because of her persisting pain non-responding to conservative treatment, she was scheduled for a resurfacing shoulder prosthesis on the right side. Standard antibiotic prophylaxis of three gifts of 1 g cefazoline in 24 h was used. A deltopectoral approach was used during surgery and the rotator cuff was intact. The osteophytes were removed and the 44 mm uncemented resurfacing shoulder prosthesis (DePuy; Global Cap) was placed \n[]. The glenoid was sclerotic without any bone loss and microfracturing was performed with the chondropick. No wound problems occurred and the patient was discharged 2 days after surgery. The physiotherapist accompanied a standard rehabilitation program with initial restricted passive range of motion. During the first 4 months, the patient was very satisfied with painless shoulder and full range of motion.\nSince this initial period, she suffered progressive shoulder pain. At examination, there was a painful arc and the supraspinatus resistance test was painful, but there were no clinical signs of a cuff rupture. It was thought that rotator cuff tendinitis caused this pain and an arthroscopic subacromial decompression was performed. Preoperative 1 gr of cefazoline was given. During surgery the rotator cuff was intact. At this time, an infection was not considered and therefore the glenohumoral joint was neither inspected nor aspirated. However, the subacromial decompression did not relieve the complaints and the range of motion gradually decreased. At 2 years after the arthroplasty, the patient had pain on the anterior side of the shoulder, mild rest pain, increased pain whilst lifting, and a restricted forward flexion of 90° and external rotation of 50°. No clinical infectious signs were present and the cuff seemed intact. Her erythrocyte sedimentation rate (ESR) was slightly elevated since the first operation (between 30 and 45) with a normal C-reactive protein (CRP). However, the biochemical markers are not completely reliable for an infection due to her diverticulitis. The X-rays showed a well-positioned prosthesis without signs of loosening and some progression in glenoid erosion []. To exclude an infection, a culture of intra-articular fluid (obtained by fine-needle aspiration) was done, which was negative.\nBecause of the persisting pain and signs of progressive of glenoid erosion on plain radiographs, a conversion from hemi to total shoulder prosthesis was performed []. Three times cefozline was given perioperative. Intraoperative inspection of the joint showed induration of the synovium and a sclerotic glenoid with irregular erosions. The cap was macroscopically solidly incorporated in the bone. Routine swabs and tissue samples from the bone and synovium were taken for cultures. The peri- and postoperative course was uneventful with normal wound healing. However, after 10 days, all intraoperative cultures revealed a combination of micro-organisms and antibiotic therapy was started. The Pantoea agglomerans, Staphylococcus epidermidis, and Propionium acnes were found in all four operative cultures.\nThe resected resurfacing prosthesis was sent to a specialized bone lab. It was fixated in buffered formalin at 4°C and the prosthesis was cut along the central stem. After embedding of the halves in polymethyl methacrylate (PMMA), the surfaces were polished, sputter coated with cold, and examined by scanning electron microscope (SEM). Then, surfaces were polished again, acid etched to partially remove the PMMA, and then stained with basic fuchsin and photographed. A microscopic stable prosthesis with full osseointegration was seen. The bone was intact, generally osteoporotic, and in good contact with the coating of the prosthesis. There were no signs of soft tissue interface formation due to loosening or microfractures of the bone trabeculae [Figures –].\nThese findings led to the conclusion that the resurfacing prosthesis failed due to a painful low-grade infection, without loosening of the prosthesis. According to the advice of the microbiologist, prolonged antibiotic regiment was started until the ESR and CRP normalized after 3 months. The rehabilitation period was longer than after the first operation, but uneventful and no wound problems occurred. At the last follow-up, 24 months postoperative, she was satisfied with her shoulder. She had a pain-free active forward flexion of 140°, endorotation of 70°, and exorotation of 30°. There were no clinical or biochemical signs of infection and the X-ray showed a correct position of the prosthesis with no signs of loosening []. |
A 51-year-old Caucasian man with the cardiovascular risk factors of hypertension and smoking was admitted to our hospital because of chest pain and syncope during exercise. He had experienced intermittent chest pain for one week prior to presentation. Upon admission, his blood pressure was 110/70 mmHg and his pulse rate was 70 beats/minute. His physical examination was unremarkable. His routine biochemistry laboratory parameters and full blood cell count were normal. A chest radiograph did not show indications of left-sided cardiac failure. A 12-lead electrocardiogram (ECG) obtained without chest pain in the patient showed normal sinus rhythm at 63 beats/minute without Q waves and with a normal QRS width (Figure ). The echocardiogram showed a normal left ventricular ejection fraction without regional motion abnormalities. The patient was referred for exercise testing using a standard Bruce protocol. The test was stopped at minute 10 of the protocol because the patient was experiencing fatigue, had an increasing heart rate of 148 beats/minute (87% of maximal predicted heart rate), had maximal blood pressure of 183/98 mmHg, and had a workload of 12.8 metabolic equivalents without clinical symptoms or ECG abnormalities. At minute one of the recovery phase, the ECG showed a giant R wave pattern in the inferolateral leads, including an increase of R wave voltage rising to 34 mm, loss of S wave, and merging of the QRS complex with the ST segment, causing a monophasic QRS-ST complex (Figure ). A nodal rhythm was also observed (Figure ). Clinically, the patient experienced chest pain and dizziness. His blood pressure was 183/98 mmHg, and we administered sublingual nitroglycerin, and the patient's ECG gradually returned to normal status (Figure ). In Figure , we show the evolution in lead III during the episode. The maximal level of troponin I measured after the ETT was 0.1 ng/mL (normal 0.0 ng/mL to 0.2 ng/mL). Emergency coronary arteriography showed single coronary vessel disease with severe stenosis in a mid-dominant circumflex coronary artery (Figure ). The patient was successfully treated with coronary percutaneous revascularization (Figure ), and, two days later, he was discharged without further symptoms. |
In May of 2017, a 75-year-old man presented to our hospital, with progressive back pain, radiating pain and numbness of his left lower limb. The patient, having been diagnosed of B3 thymoma for 9 years, received surgical treatments including thymectomy in 2008, and posterior spinal cord decompression, tumor resection and a stabilization procedure due to the spinal metastases of malignant thymoma followed by postoperative radiation in 2011. In the medical journal of his current illness, the patient stated he had been experiencing paroxysmal and severe back pain for approximately 3 months, and he had also experienced a worsening numbness and radiating pain of his left lower limb for approximately 3 months. The pain in his back can reach 6 to 7 points using visual analog scale (VAS) and cannot be alleviated with rest and hot compresses. The patient denied experiencing any other constitutional symptoms. Upon further questioning, he recalled a history of old myocardial infarction since 2003. No pertinent family history was identified, including hypertension and cancer.\nOn physical examination, the patient showed pressure pain and percussion pain in his sacral region decreased sensation to pin-prick and fine-touch of his left lower limb and exhibited 5-/5 strength in his bilateral lower limbs. Deep tendon reflexes revealed normal for knee-jerk and Achilles tendon reflexes bilaterally. Ataxia, cranial nerves, mini-mental, and the rest of the neurological examination showed no abnormalities. Preoperative hemodynamic and cardiovascular assessments included electrocardiogram, echocardiogram, and chest radiography. Preoperative laboratory assessment was conducted, including routine laboratory tests (electrolytes, liver, and kidney function tests, complete blood count), tumor markers, myocardial enzymogram, and screening for myasthenia gravis. The results of the laboratory studies were almost within normal range. X-rays revealed sacral lesions, with high suspicion of spinal soft tissue tumors (Fig. A, B). Spinal magnetic resonance imaging (MRI) was ordered to visualize the metastatic lesions, assess the stability of the vertebral column, and to aid in the formulation of a surgical approach. MRI of the spine showed the density of soft tissue measuring 10 cm × 7.5 cm × 5.7 cm, obvious bony destruction in the sacrum, and spinal cord compression secondary to the giant mass, with increased metastatic marrow infiltration of the sacrum (Fig. A–H). Tumor infiltrated through the sacrum body into the posterior elements, thus extraosseously spread into the bilateral aspects of the epidural space extending posteriorly, resulting in spinal cord compression (Fig. A, B). The bone scanning revealed high intake in the sacral spine, with high suspicion of spinal metastases (Fig. ).\nSubsequently, osteoplasty with cement augmentation was performed to destroy the functional tumor and stabilize the spine under local anesthesia. In brief, percutaneous vertebroplasty at sacrum was performed according to the original surgical plan. For the posterior approach, we used C-arm for perspective positioning, bilateral vertebral lesions of sacrum were identified as surgical targets, and the bilateral sacral pedicle puncture points were located. Then 2% lidocaine was used for local infiltration anesthesia, and the puncture needle was inserted through the cannula. Under the C-arm fluoroscopy, the vertebral lesion was penetrated through the left pedicle of the sacrum first, and bone cement for vertebroplasty was introduced. Under the perspective, the 10.0 mL cement of left side was slowly pushed through the putter, and the biopsy passage was closed. In the same way, the right S1 vertebral pedicle was then punctured to the right S1 vertebral body lesion, and 9.6 mL bone cement was slowly pushed into the right S1 vertebral body lesion through a push rod under fluoroscopy, and the biopsy pathway was closed. Fluoroscopy confirmed the good dispersion of bone cement. The operation was successful and intraoperative bleeding was about 60 mL. Postoperative posteroanterior and lateral radiographs of the spine showed cement augmentation was satisfactory (Fig. A and B). The patient was unwilling to undertake any further treatments and was discharged and monitored on an outpatient basis. The postoperative pathology report confirmed a malignant B3 type thymoma, which was consistent with the primary tumor (Fig. A–D). Pathological analysis was positive for AE1/AE3 indicating epithelial origin from thymus gland. Biopsy samples were negative for chromogranin A, synaptophysin, CD56 (NK-1), TTF-1, CD5, with 30% Ki-67 positive nuclei (Fig. A–F). Consequently, malignant thymoma with sacrum metastases was diagnosed via history taking, laboratory values, imaging results, and pathological studies.\nOne week after the operation, the patient's muscle strength of lower extremities improved to grade V compared to the preoperative status, and the symptoms were relieved significantly. Moreover, VAS score of his back pain improved to 0 to 1 points compared to the preoperative status, 6 to 7 points. Postoperatively, the patient underwent rehabilitation therapy and was discharged and monitored as an outpatient. The postoperative 6-month and 1-year follow-up visit showed no tumor progression and no new symptoms. There were no other complications associated with the operation during the follow-up period. |
A 31-year-old male patient with no known comorbidities was brought to our tertiary care center at Tamaka, Kolar, Karnataka, South India. The patient revealed a history of road traffic accident, due to which he sustained injury to the right lower limb. The patient had been unable to walk since then. He underwent native treatment for about six weeks and was brought to our hospital for further management. On examination, his vitals were stable. Local examination of the right leg showed swelling and tenderness present over the proximal third of the right leg. Range of motion at the right knee joint was 10-30 degrees. Assessment of range of motion at the ankle joint revealed that dorsiflexion was absent. Capillary refill time was normal. All other long bones and joints were found to be clinically normal.\nThe patient was initially managed with intravenous administration of amoxicillin- potassium clavulanate 1.2 g prophylactically. The patient was then temporarily stabilized with an above-knee fiberglass splint. He was sent for preoperative imaging, as shown in Figure . Plain radiograph of the right knee showed split fracture involving the lateral tibial condyle extending into the articular surface, with lateral displacement of fracture fragments, and oblique fracture of fibular head with caudal displacement of fracture fragments. MRI of the right knee showed a high-grade tear in the proximal aspect of the lateral collateral ligament and a grade 2 medial collateral ligament tear. In addition, the patient was counselled regarding expectations and outcomes. After obtaining informed consent, the patient was operated under spinal anesthesia. Open reduction and internal fixation with proximal tibia locking compression plating with autologous bone grafting from the ipsilateral iliac crest (corticocancellous graft) along with lateral meniscectomy and arthrolysis of the knee joint was performed under spinal anesthesia, as shown in Figure . The post-operative period was uneventful.\nHospital course\nPost-operatively, the patient received intravenous amoxicillin-potassium clavulanate 1.2 g twice daily for seven days and amikacin sulfate 500 mg twice daily for five days followed by oral amoxicillin-potassium clavulanate 625 mg twice daily for seven days. Post-operative radiograph of the operated knee joint was taken on post-operative day 2, as shown in Figure . An above-knee slab support was applied, and the patient was advised strict non-weight bearing for six weeks. Culture and sensitivity from surgical site showed staphylococcus epidermidis, which was sensitive to tetracycline and doxycycline. The patient was started on tablet doxycycline 100 mg twice a day for 14 days. All staples were removed on post-operative day 14, and the surgical site was found to be healthy. The patient underwent physiotherapy in the form of muscle stimulation test 15 minutes daily till he recovered from the foot drop. The guarded continuous passive motion was also started for the patient. At discharge, range of motion at the knee joint was 0- to 120-degree flexion after one month of physiotherapy. Tinel sign was positive 1-cm along the common peroneal nerve distribution at the level of the middle third of the leg. The patient had recovered from the foot drop (possibly due to neuropraxia). Partial weight bearing was initiated after six weeks. The patient was allowed full weight bearing at three months. At the last follow-up, six months post-injury, he was walking without any difficulty. |
A 49-year-old woman with no significant past medical history presented to her family physician in 2014 with vague right upper quadrant abdominal pain. Her vital signs were stable, her abdominal exam was unremarkable, and she had no palpable organomegaly. Her hemoglobin, platelet count, white cell count, liver function, and renal function tests were all within normal limits. She underwent abdominal ultrasound, which demonstrated multiple vascular lesions within the liver, with the appearance of hemangiomata. Her abdominal pain had since resolved, but due to the ultrasound findings, she received an MRI of the abdomen. This study demonstrated multiple liver lesions with enhancement patterns consistent with hemangiomata. There was also a 4.6 × 3.1 × 2.8 cm vascular lesion within the body of the spleen that enhanced with features consistent of hemangioma, although it was less bright than the liver hemangiomata. She remained asymptomatic. These lesions were followed with serial MRI studies, and while the liver lesions remained stable in size and enhancement, follow-up imaging at two years demonstrated an interval enlargement in the splenic lesion to 7.1 × 5.0 × 6.3 cm, with four additional new nodules within the spleen (, white arrows). The dominant lesion had heterogenous enhancement compared to the liver hemangiomata (white lesions within the liver in ).\nDue to the increase in size, new nodules, and heterogeneous enhancement, she was referred for surgical resection. She underwent an uncomplicated open splenectomy in September 2016. Gross pathology demonstrated an 8.0 × 5.8 × 4.5 cm lesion with a mottled appearance and focal hemorrhagic tissue which nearly replaced the splenic parenchyma (), which was consistent with preoperative imaging (; white arrows correlate to similar areas in gross specimen in ). Histological assessment demonstrated a highly cellular, atypical complex vascular lesion with rare mitotic figures on H&E staining (). MIB-1 staining illustrates a proliferation index of approximately 10% (). Immunophenotyping with CD31 () and CD34 (not shown) confirmed the presence of vascular endothelium. Taken together, the morphological and immunophenotypic findings are consistent with a diagnosis of epithelioid hemangioendothelioma (EHE). Given this diagnosis, and the previously identified hepatic vascular lesions, she underwent a left hepatic lobectomy and nonanatomic resection of a segment 8 lesion to rule out HEHE in November 2016. Final pathology showed benign hemangiomata in the areas observed on imaging and multifocal, microscopic EHE within the liver parenchyma which was not appreciated on multiple previous imaging studies. Based on these findings, she was referred for liver transplantation. She was carefully evaluated by our multidisciplinary team and referred to medical oncology for consultation. She underwent full body imaging including a CT chest, abdomen, and pelvis, as well as abdominal and pelvis MRI, which did not demonstrate any new lesions or evidence of extrahepatic disease. The collective opinion based on multidisciplinary assessment was to proceed with liver transplantation as the best option for R0 resection. She ultimately underwent liver transplantation in early 2017, with evidence of multifocal HEHE in the explanted liver (). There were no concerning lesions, nodules, or lymph nodes noted at the time of transplant, and no malignant periportal lymph nodes were identified within the explant. She received induction immunosuppression with basiliximab and maintenance therapy with tacrolimus and mycophenolate mofetil, which is the standard of care for most patients in our center. She has had an uncomplicated postoperative course with no evidence of recurrent EHE on follow-up imaging, with more than one year of follow-up to date. |
This 54-year-old female was referred to the Neurofibromatosis outpatient clinic of the Oral and Craniomaxillofacial Surgery Department for treatment of numerous cutaneous tumors that preferentially covered her trunk and extremities. On admission, the patient described the tumors to be painless and disfiguring. Besides the cutaneous tumors the patient sought advice for treatment of a painful region of her left small finger (Figure 1 ). For some time the ulnar side of the distal phalanx of this finger had become very touch-sensitive. There was neither a tumor visible nor any other pathological alteration of the skin. However, the finger could not be examined by palpation. The suspected diagnosis was initially a glomus tumor [] or a non-palpable nodular PNF.\nMRI at 3T revealed numerous, popcorn-like, contrast-absorbing, hyperintense formations on T2-weighted images, up to 1.2 x 1.9 cm² in size located on the palmar side of the left hand (Figure 2 ). These small lesions mainly affected the metacarpal bones II and III. MRI also showed a sharply defined lesion of max. 1 cm in diameter in the subcutaneous layer at the level of the radiocarpal joint and distal to first digit that reached to the tendons of the extensor musculature. The bony structures showed a homogeneous signal. The findings were interpreted as disseminated subcutaneous and cutaneous neurofibromas of the entire left hand. Structures were found on the distal phalanx of the left small finger whose intensity pattern corresponded to that of the other lesions.\nDuring surgery for the excision and vaporization of numerous neurofibromas of the trunk and extremities, the pain sensitive finger region was also explored. After incision of the skin, no tumor was visible. Therefore, a circumscribed subcutaneous excision was taken as a tissue sample and examined histologically.\nThe tissue findings confirmed cutaneous neurofibromas for the numerous nodules excised from the trunk and extremities. Surprisingly, there was no evidence for a glomus tumor in the tissue sample of the finger. Smaller neurofibromas were differentiable in this tissue sample. Within the corium as well as in the subcutaneous adipose tissue there were some clustered Vater-Pacini (VP) corpuscles. Next to the corpuscles several small nerve fibers were present. Diagnosis of VP neuroma was made (Figure 3 ).\nThe nerve fibers were positive for S100 and neurofilament. S100 immunoreactivity was especially strong in the center of the corpuscle. The perineurium of the nerve fascicles and the lamellae of the corpuscles expressed epithelial membrane antigen (EMA).\nThe healing process of the wounds was inconspicuous. The patient noticed that the sensations of pain had diminished significantly in the area of the small finger soon after surgery. |
The patient is a 69-year-old Caucasian female who reported asymmetry of her abdomen in early 2011. An abdominal mass was visualized with magnetic resonance imaging (MRI) (Fig. ). CT-guided biopsy was undertaken, and pathology was consistent with a T2b grade 3 (stage IIB) high-grade soft-tissue sarcoma arising from the lower rectus abdominis muscle. In March 2011, following surgical resection with concurrent interstitial high-dose-rate brachytherapy, she had external beam radiation therapy (XBRT). The patient was observed, and 2 years later a solitary metastatic lesion to the anterior lingula of the left lung was noted on follow-up CT scan. This mass was resected in May 2013. Follow up PET/CT in August and November 2013 revealed no further evidence of disease other than a pulmonary nodule in the apical left upper lobe that was not fluorodeoxyglucose (FDG) avid. After an observational period of nearly 2 years, follow-up CT scan of the chest, abdomen, and pelvis confirmed stability of the apical left upper lobe pulmonary nodule. However, there was development of a pulmonary nodule more caudally in the anterior segment of the left upper lobe concerning for a possible solitary lung metastasis. The anterior segment left upper lobe pulmonary nodule had enlarged on the subsequent, March 2015 PET/CT scan, measuring 1.8 × 1.1 cm and was metabolically active, having a standardized uptake value (SUV) of 5.9, which was consistent with solitary pulmonary metastasis (Fig. ). The patient received a left upper lobectomy in late March 2015. The specimen pathology was found to be metastatic PEComa and was sent for additional genetic testing.\nIn September 2015, CT revealed progression of disease with development of a subaortic lymph node metastasis, in the anterior mediastinum (Fig. ). In this case, surgical resection was deemed to not be a viable option. Therefore, systemic therapeutic strategies were instead considered. mTOR inhibitors were a potential choice of therapy, based on studies indicating some efficacy in this setting, but no clinical trials utilizing these agents were available [, , , , ]. Secondary to elevated expression of PD-L1 on IHC of the tumor, treatment with the PD-1 inhibitor pembrolizumab 2 mg/kg intravenously every 3 weeks was begun in October 2015.\nAfter cycle 21, a little more than 1 year after commencing pembrolizumab therapy, the patient had a PET/CT scan with no metabolically active disease (Fig. ). At this time, the patient’s only complaint was shortness of breath on exercise, which was attributed to the prior lung resection. She continued on pembrolizumab treatment with no evidence of disease until April 2018 for a total of 40 consecutive cycles at which time she was placed under surveillance. She has been disease free off therapy for 19 months. |
A 51-year old woman with a 2-year history of rheumatoid arthritis presented with three weeks history of vertigo and vomiting. Her symptoms worsened progressively and she became unsteady on walking. She also felt episodes of tingling and numbness on the right side of the face. There was no prior history of neurological disease and no family history of multiple sclerosis.\nShe was maintained on Methotrexate 20 mg weekly for the arthritis, however due to inadequate response, subcutaneous Etanercept 50 mg weekly was begun three months prior to her admission with marked improvement in her joint symptoms. She had received the last dose of Etanercept four days prior to hospital admission. Past medical history was also significant for 1-year history of diabetes on Metformin, and hypercholesterolemia on Simvastatin 10 mg. The physical examination revealed a horizontal nystagmus with fast component towards left side and broad-based, ataxic gait with tendency to fall to the left side. Rest of examination was unremarkable and there were no signs of active joint disease.\nComplete blood count, urea, electrolytes, and blood sugar were normal and Electrocardiography revealed occasional ventricular ectopic beats. Autoimmune tests including ANA, Anti-ds DNA and Anti-CCP were negative. Rheumatoid factor also was negative at admission.\nMRI of the brain () revealed multiple rounded foci with iso-intense T1 and hyper-intense FLAIR and T2 signals involving the left side of the medulla, the right middle cerebral peduncle, the body of the corpus callosum and the sub-cortical white matter of both cerebral hemispheres, which were suggestive of demyelinating foci. The patient refused lumbar puncture.\nEtanercept was discontinued immediately. Three days later she felt better, with reduction in intensity of vertigo and disappearance of nystagmus, however her gait remained ataxic.\nIntravenous methylprednisolone pulse therapy was then started and three days later, she made a good recovery and was able to walk unsupported. She was discharged on oral prednisolone 60 mg daily to be tapered gradually and then maintained on Methylprednisolone 4 mg daily. Three months later, she presented with new onset ataxia, double vision and vertigo. Neurological examination revealed ataxic gait, nystagmus with fast component to left side. Repeated MRI of the brain () revealed a newly developed rounded, about 1 cm in size, demyelinating lesion in the right posterior pariental area, with scattered small demyelinating lesions. The patient requested discharge to seek a second opinion abroad. |
In April 2010, a 40-year-old male, with a history of NSCLC (histology, adenocarcinoma) treated from 2008, was referred to the Department of Radiation Oncology, Tommaso Campanella Cancer Center (Catanzaro, Italy) for a lesion in the right frontal lobe measuring 1.4 cm in maximum diameter (). Based on RTOG criteria, the patient was eligible for SRS: Lesion measuring <3 cm in maximum diameter, producing minimal mass effect and ECOG performance status, 0.\nIn May 2010, the patient underwent a stereotactic treatment delivered in a single dose of 24 Gy using a Clinac DBX 600 (Varian Medical Systems Inc., Palo Alto, CA, USA) and was followed up clinically by serial MRI and CT scans. In September 2010, a complete response to the radiotherapy treatment was documented by an MRI of the brain.\nA CT scan revealed a recurrent disease, in the same area as treated previously, 17 months after the irradiation of the metastatic brain tumor (). At this point, the patient was offered a second SRS treatment as an alternative to surgery. Following obtaining consent for re-irradiation, in November 2011, the patient underwent a second session of radiosurgery, which was delivered in the same manner as the initial session, but at a single dose of 15 Gy (). In this case, the dose was selected according to a previous study using the linear quadratic model to derive information on the cumulative biological effective tolerance dose (BEDcumulative) that results from BEDinitial plus BEDre-irradiation().\nA stable disease, associated with no symptoms or signs associated with neurological decline, was documented in the following CT scan and MRI brain controls until July 2012, when a MRI brain scan again showed a lesion measuring 1 cm in diameter in the same region and surrounded by an extensive area of vasogenic edema, compatible with a recurrence of disease.\nTherefore, in August 2012, the patient underwent surgery to remove the right frontal lesion. Histological diagnosis following surgery showed absence of disease and the occurrence of radionecrosis ().\nAt present, the patient is receiving treatment with erlotinib and appears to be in good general condition (ECOG performance status, 0). |
A 74-year-old male was admitted to the Second Xiangya Hospital (Central South University, Changsha, Hunan, China) in October 2005 due to an increased defecation frequency for a duration of more than one year. For one year prior to being admitted, the patient had observed a defecation frequency that had increased from once per day to six-seven times per day without precipitating causes and accompanied by stool reduction. The stool was yellow, but retained its shaped. The patient had no history of a frequent/urgent requirement to urinate, gross hematuria or abdominal pain. The prostate could not be palpated clearly when performing the physical examination, though a huge mass was identified behind the prostate during the rectal examination. The mass was medium-hard without unusual palpable nodules and tenderness. The other physical examinations were normal. Ultrasonography revealed that the boundary of the prostate was unclear and that it was 39×25×23 mm in size. A thick-walled inhomogenous hypoechoic cystic mass (76×77×74 mm), in which the thickness of the cyst wall was uneven, was detected posterior to the prostate. The level of blood prostate specific antigen (PSA) was normal, while the pelvic CT () revealed that the diameters of the mass were 77 mm from top to bottom and 76 mm from right to left. A ring-like subcapsular high-density district was observed in the mass and there was a mixed high and low density and punctate calcification. The mass was not connected to the adjacent tissues and appeared to originate from the prostate. The contrast enhanced CT also demonstrated non-uniform changes. The bladder was filling well and the wall was normal. No swollen lymph nodes were identified in the cavity.\nA laparotomy through an abdomen incision revealed a large, slightly capsulated mass that arose from the seminal vesicle but not the prostate, without connecting to the gastrointestinal tract. The size of the mass was 8×7×7 cm and the capsule boundary was clear, without adherence to the organs around the seminal vesicles. The mass was removed using the no-touch technique. A biopsy and immunophenotype staining were performed. The texture of the mass was moderate and the surface was smooth with a fibrous membrane. Light microscopy examination revealed hemorrhage and necrosis with rich and crowded cells. Karyokinesis was rare and the mitotic index was <10 mitoses per 50 high power fields (HPF). The histopathological diagnosis was of a mesenchymal tumor originating from the seminal vesicle. The immunohistochemical studies revealed that the cells were strongly positive for CD34, CD117, PDGFRA and Vim () and negative for HHF35, smooth muscle actin (SMA), S-100 protein and creatine kinase (CK). These results strongly supported a diagnosis of a low-risk EGIST of the seminal vesicle. A point mutation analysis for a KIT protein mutation was not performed due to its unavailability at the Departments of Urology and Pathology, The Second Xiangya Hospital. The patient received 600 mg imatinib orally, taken once daily with food for 6 months. Currently, the patient is being followed-up and has not experienced a recurrence or metastasis for 6 years. |
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