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A 54-year-old male patient was admitted to our center on December 2, 2019. The patient presented with chills, numbness, and intermittent claudication in both lower limbs. Fontaine stage was IIa. The specialist physical examination revealed that the patient's pulsation of the left dorsalis pedis artery was weak, and the right dorsalis pedis artery was not palpable. Computed tomographic angiography (CTA) showed signs of filling defect in the proximal lumen of the common iliac, internal and external iliac arteries, and the right anterior tibial artery. They were found to have “continuity interruption” at the end of the abdominal aorta, and irregular stenosis of the local lumen of the bilateral femoral artery, deep femoral artery, popliteal artery, left anterior and posterior tibial artery, and peroneal artery was observed (Figures and ).\nDuring the first hospitalization, the patient refused surgical treatment and was given anticoagulation and vasodilation therapy for one week. After discharge, the patient was treated with antiplatelet aggregation and anticoagulation by oral administration of cilostazol and salgrel axetil. During the medication period, the patient developed ischemic rest pain (Fontaine stage III).\nOn January 8, 2020, the patient was readmitted to the hospital. Specialist physical examination revealed pale skin on both feet, the skin was also warm and cool, and the dorsalis pedis arteries were not palpable. Digital subtraction angiography (DSA) revealed that the right iliac artery was occluded, multiple thrombi were attached to the wall, and the abdominal aorta was occluded from the right renal artery level ().\nThrough three operations that were performed at our center, we successfully opened the abdominal aorta and bilateral iliac arteries to improve the symptoms of lower limb ischemia. During the first procedure, we punctured the right femoral artery, successfully passed through the occlusive segment of the right iliac artery, and placed the end tip of the 5F 20cm UNIFUSE thrombolytic catheter into the abdominal aorta at the level of the right renal artery. Thrombolysis with 200000u urokinase was continuously given after operation. Two days later, we performed a second DSA operation. Thrombolytic catheter angiography showed that the right external iliac artery was partially developed, but the distal abdominal aorta was not seen. The possibility of blockage was considered at the tip end of the thrombolytic catheter, and the thrombolysis angiography showed that the thrombus in the proximal renal abdominal aorta was dissolved more than before. Replacement of 5F 20 cm UNIFUSE thrombolysis catheter continued thrombolysis for 24 hours (). During the third procedure, we used thrombectomy combined with covered endovascular reconstruction of the aortic bifurcation (CERAB). The angiography of the right femoral artery showed a large filling defect below the opening of the renal artery of the abdominal aorta, the development of the right iliac artery was unobstructed, and the left iliac artery was not seen (). The 14 mm balloon was placed above the renal artery, and the balloon was filled and pulled to the end of the abdominal aorta to move the filling defect downward (). The 4F Fogarty balloon embolization tube was implanted into the end of the abdominal aorta, the arterial embolus was removed about 10 cm, and the thrombus was distally removed. The embolus of the left iliac artery was removed by the same method. The pulsation of bilateral femoral arteries was palpable after thrombectomy. The lower segment of the abdominal aorta was implanted with Medtronic ENDURANT EN LW 1616C95EE self-expanding covered stent, and the upper end was located below the opening of the right renal artery. Angiography showed that the blood flow in the stent of the abdominal aorta was unobstructed, and there was no contrast medium extravasation and filling defect. Two self-expanding covered stents (Bard Fluency 8 mm∗80 mm) were implanted in both common iliac arteries, where the proximal end of the two stents was located in the abdominal aortic stent, overlapping some 15 mm. Angiography showed that the blood flow in the abdominal aorta and bilateral iliac arteries was unobstructed, and the orifice of the bilateral iliac arteries was narrowed. The stenotic segments of bilateral iliac arteries were dilated with 8 mm balloon, and the angiographic results showed improved blood flow than before (Figures and ). Finally, we placed 4F Fogarty thrombectomy balloon into the right popliteal artery and removed the artery 5 cm embolus. The angiography showed that the blood flow of the right popliteal artery was smooth ().\nAfter the operation, we examined the patient and found that the skin temperature in both feet recovered, and the bilateral dorsalis pedis arteries were palpable. Reexamination of CTA revealed that the abdominal aorta, bilateral common iliac, internal and external iliac arteries, bilateral femoral arteries, deep femoral arteries, and popliteal arteries were functioning well (Figures and ).\nAfter operation, the patient was orally given aspirin, 100 mg qd, Rivassaban 2.5 mg bid []. Also, the blood coagulation function of the patient was monitored; his prothrombin and international standard ratio were in the ideal range. |
A 37-year-old woman, gravida 1-para-0 (BMI = 28), with no significant past neurological or medical history, was hospitalized with a diagnosis of premature rupture of membranes in pregnancy at 39 + 3 weeks of gestation. The patient, after signing informed consent, underwent three attempts of epidural analgesia with a Tuohy needle of 18 Gauge. Suspecting accidental puncture of dura mater, they modified the anesthetic technique and started intravenous administration of Remifentanil. Moreover, induction of labor was carried out, with vaginal delivery after 1 h and 15 min. After 10 h from the initial attempt of epidural anesthesia, the patient begins to complain of symptoms as orthostatic headache and irradiated neck pain. The pain increases progressively, affecting the entire rachis. Consequently, she received treatment with crystalloids (lactated Ringer) in continuous infusion and painkillers (paracetamol), also during the 1st postpartum day.\nOn the second day postpartum, infusion therapy was resumed 12 h after the release of peripheral venous cannula, with substitution of paracetamol with caffeine orally, without apparent benefit. The headache started to appear also in supine position.\nOn the third day postpartum, painful symptoms worsened, with the appearance of vomiting and extreme physical prostration; hemodynamic parameters within the normal limits. On the same day, an epileptic generalized tonic–clonic seizure occurred: an electroencephalogram and an encephalon-skull-CT were performed, but no diagnostic elements emerged.\nOn the fourth day postpartum, a second convulsive episode appeared, following which the patient underwent a cranial encephalic MR, which demonstrated a reduction in the size of periencephalic liquor spaces at the cranial base and in the posterior cranial fossa, associated with caudal dislocation of the encephalic trunk, dilatation of dural venous sinuses, and contrast impregnation of the dura mater, findings compatible with vascular hypotension and bilateral frontal cortical infarctions with bilateral right and temporal dominance for venous stasis thrombosis in relation to the caudal shift of the brain. Consequently, the patient was transferred to the Stroke Unit, where she was treated with oral anticoagulant therapy, then replaced with low molecular weight heparin and antiepileptic therapy (levetiracetam i.v.) and she underwent blood tests, which showed leukocytosis, anemia, and thrombocytosis, in absence of a hereditary thrombophilic state. During the hospitalization, the patient maintains a negative neurological objectivity and reports an improvement of the headache, documented by the NMR images, until disappearance, 10 days after dural puncture.\nTwo months later, a third MRI survey showed a resolution of the picture, without alterations of the brain signal or abnormal impregnations in intra and extra axial, with liquor spaces within the norm. From a clinical point of view, there were significant memory changes and an anxious depressive state, in pharmacological and psychotherapeutic treatment, for which compensation has been received for iatrogenic damage. |
A 62-year-old woman undergoing angiographic bland embolization of the right hepatic artery by interventional radiology service for liver metastases from a previously resected neuroendocrine tumor of the pancreas, which stained positive for serotonin, developed severe chest and epigastric pain radiating to the back during the procedure. She had been pretreated with octreotide. Initial 12-lead electrocardiogram (ECG) revealed sinus rhythm and lateral and inferior ST-segment depression as well as ST elevation in lead aVL (), while initial cardiac biomarkers were elevated (troponin T 0.85 μg/L, peak 1.08 μg/L; CK 652 U/L). The patient had no history of cardiac disease or cardiac risk factors, while significant medical history included hypothyroidism, psoriatic arthritis, and pancreatic neuroendocrine carcinoma resected a year earlier. After CT scan ruled out aortic dissection (a rare complication of abdominal angiography), she was transferred to the Coronary Care Unit and treated for an acute coronary syndrome. As the embolization procedure was performed by the interventional radiologist in a hospital where there is no cardiac catheterization available, the patient was not immediately referred for a coronary angiogram. A subsequent ECG performed after 24 hours () revealed the resolution of ST segment changes.\nEchocardiography was carried out the next day, revealing a normally contracting left ventricular (LV) apex, but akinesis of all basal segments and hypokinesis of the mid-ventricular segments with LV ejection fraction estimated at 40% with no significant valvular dysfunction (, Supplementary Movie I in Supplementary Material available online at ). Coronary angiography revealed normal coronary arteries in keeping with a nonischemic cardiomyopathy, while ventriculography confirmed the echocardiographic findings, showing severe hypokinesis of the antero- and inferobasal segments with normal contractile function of the more distal segments (, Supplementary Movie II). These findings were consistent with reverse-type Takotsubo cardiomyopathy. During her stay in the coronary care unit she had stable hemodynamics and at no point required inotropic or vasopressor support. She was treated medically with combination of ACE inhibitor, beta blocker, and antiplatelet therapies, and repeat echocardiography performed six weeks later showed significant improvement in LV function with ejection fraction in the low normal range at 50%, with very mild residual hypokinesis of the basal segments alone (Supplementary Movie III) and no significant valvular dysfunction, confirming the diagnosis of reverse-type Takotsubo cardiomyopathy. |
A 32 year-old woman with a history of hypothyroidism and pre-eclampsia initially presented to an outside hospital with acute onset dense left hemiplegia, right gaze preference, and left-sided neglect. Her initial National Institute of Health Stroke Scale (NIHSS) was 14 and she had an admission Glasgow Coma Scale (GCS) of 10. A computed tomography (CT) angiogram of her neck revealed complete occlusion of the right cervical internal carotid artery (ICA). She was outside the time window for intravenous thrombolysis; however, she underwent mechanical thrombectomy using a stent retreiver device and aspiration (Penumbra System®, Alameda, CA). Immediately after the procedure, there was a successful restoration of the blood flow to the distal ICA, proximal middle cerebral artery (MCA), and to the anterior cerebral artery (ACA), with residual distal M2 occlusion. She was intubated for the procedure and was extubated in the following days. Her left-sided weakness persisted and a repeat CTA showed re-occlusion of the right cervical ICA. No further intervention was done and she was treated with aspirin and statin for secondary stroke prophylaxis. The stroke was deemed cryptogenic after work-up for a potential source was negative including an echocardiogram which demonstrated a normal ejection fraction, normal left atrial size, and negative bubble study. A workup for prothrombotic and hypercoagulable states were negative as well. Magnetic resonance imaging (MRI) of the brain was done which showed a large area of diffusion restriction with corresponding decreased apparent diffusion coefficient (ADC) and T2 hyperintensity in the right frontal, parietal, temporal lobes and in the basal ganglia with areas of hypointensities on gradient echo sequencing, which suggested infarction in these areas with some areas of hemorrhagic conversion (Figure ).\nSubsequently, she was discharged to an inpatient rehabilitation center. While at the rehabilitation center, about four weeks after her stroke, she developed moderate to severe insidious onset headache. A repeat MRI, done four days after the onset of headache, showed diffusion restriction (with corresponding decreased ADC) and a ring-enhancing lesion in the right basal ganglia which involved part of the previous ischemic stroke. An extensive area of T2 hyperintensity was seen around this lesion consistent with vasogenic edema (Figure ).\nWith a recent invasive procedure along with the MRI findings, the possibility of an abscess was entertained, even though she did not have systemic signs of an infection (afebrile, WBC count 7800/mm3, negative blood cultures). She was empirically started on broad-spectrum antibiotics (vancomycin, cefepime, and metronidazole) and admitted to our institute for further management. On day three of admission to our hospital, she developed a high-grade fever and had an acute deterioration in her mentation that progressed to coma. An MRI was repeated to evaluate for any progression of the disease and to obtain stereotactic images for drainage. In addition to the previously mentioned ring-enhancing lesion, the post-contrast sequences now demonstrated enhancement of the right lateral ventricular wall which was suggestive of ventriculitis (Figure ).\nShe underwent a stereotactic drainage of the lesion, which aspirated purulent material. The patient was continued on broad-spectrum antibiotics. Vancomycin was discontinued after 10 days. Cefepime was switched to ceftriaxone, which along with metronidazole, was continued for a total of six weeks. An extensive laboratory workup was done which did not reveal a potential source of infection or immunocompromised state. Due to the high suspicion for an abscess and the purulent aspirate, a bacterial DNA probe was carried out on the aspirate, which revealed the presence of Fusobacterium necrophorum. Since Fusobacterium necrophorum is the implicated organism in Lemierre's syndrome, a surveillance of signs were carried out on the patient but failed to reveal neck pain or thrombosis of the internal jugular vein (imaged with an ultrasound of the neck). On post-drainage day one, her mental status improved and she progressed to her baseline prior to her discharge from the hospital. |
A 66 year old male with treated lung carcinoma, underwent a routine CT scan. The scan revealed stable findings, including catheter-related thrombosis of the lower third of the LBCV.\nAll studies were performed on GE Lightspeed 64-detector CT scanners. In cases 1-3, a pulmonary embolism protocol was followed. Patients received a bolus of 55 mL of non-ionic contrast followed by 35 mL of saline flush through the antecubital veins of the left arm at a rate of 4.5 mL/s. The scan delay was predetermined by a test bolus (20 mL) using the pulmonary trunk as the region of interest. In case 4, 90 mL of non-ionic contrast were administered intravenously through the left arm at a rate of 2 mL/s using a scan delay of 65 seconds. Scans were obtained using 1.25 mm collimation at 0.9 mm intervals through the thorax in cases 1-3; in case 4, the study was obtained with 5 mm collimation at 4 mm intervals and reconstructed at 1.25 × 1.25 mm.\nThe CT scans of the first three patients showed an extensive network of venous collaterals in the upper mediastinum, particularly around the aortic arch and pericardium. These collateral vessels included pericardial, esophageal, bronchial and mediastinal branches of the left brachiocephalic, left vertebral, left pericardiacophrenic, left internal thoracic or superior intercostal veins. The collateral vessels also communicated with the azygos-hemiazygos, phrenic and, in two cases, pulmonary veins. In the fourth patient, the scan showed a collateral venous network joining the left and right inferior thyroid veins in the upper mediastinum and lower neck that communicated with the paravertebral venous plexus and gave rise to several small mediastinal and pericardial branches.\nAt the union of the pericardium with the ascending aorta and pulmonary trunk, pericardial branches were noted to be in continuity with small vessels distributed along the walls of the intrapericardial segments of the great arteries in all patients (-). These vessels measured 1.5-2.5 mm in diameter and extended to the root of the ascending aorta and base of the pulmonary trunk where they crossed the coronary arteries. The vessels crossing the right coronary artery drained into the right atrial appendage (all cases), and those crossing the left coronary artery drained into the anterior interventricular and left coronary veins (cases 1 and 3). In the second case, tiny collateral vessels on the left could only be traced to the level of the left coronary artery due to image-degrading pulsation artifacts; we assume that these veins also drained into the great cardiac vein (). The collateral vessels that crossed the right coronary artery joined the anterior aspect of the right atrial appendage. In case 1, focal collections of contrast measuring about 5 × 8 mm were noted in the wall of the appendage where the collateral vessels fended ().\nThe unusual course of a collateral vessel from the anterior wall of the ascending aorta is demonstrated in . After descending along the wall of the aorta to the aortic root, the vessel made a loop posteriorly and connected with the anterosuperior wall of the left atrium.\nFewer than four or five intrapericardial vessels communicated with pericardial veins. The vessels, except for a single channel in case 4, ramified and intercommunicated as they descended towards the base of the great arteries. shows the single vessel in case 4; it coursed along the left lateral wall of the ascending aorta and drained into the right atrial appendage after passing under the right coronary artery. A common finding among all patients was the presence of at least one collateral channel along the wall of the ascending aorta located close to the aortopulmonary groove. |
We received written consent from the patient prior to publication of this case.
A 35-year-old multiparous woman with her fifth pregnancy and fourth labor presented with fatigue and shortness of breath for 10 days at 16 weeks of gestation. During the past 13 years, she had delivered vaginally 3 times without any complications. During this pregnancy, she had not been seen by any obstetrician until she had noticed considerable fatigue and dyspnea for 10 days. According to transthoracic echocardiogram, her pulmonary artery pressure (PAP) was 75 mm Hg, and she had a dilated left ventricle (67 mm) and a ventricular septal defect (1.7 mm) with a bidirectional shunt. On examination, she could only sleep in a semi reclining position. Rhythmic cardiac tones with an accentuated pulmonary component of the second heart sound were found on auscultation, with a heart rate of 80 beats per minute, blood pressure of 104/80 mm Hg and oxyhemoglobin saturation of 93% on room air. Electrocardiography showed sinus rhythm, incomplete right bundle branch block, and left anterior branch block.
The patient with severe PH developed right heart failure early in the second trimester and was at a high risk for sudden death. In discussion with her cardiologist, obstetrician and anesthesiologist, based on maternal will, it was decided to chose an artificial abortion by cesarean section under continuous epidural anesthesia with double catheters to avoid development of cardiac failure at 16 weeks and 2 days of gestation.
Invasive arterial blood pressure and central venous pressure (CVP) were administered for hemodynamic monitoring. The patient started with 104/80 mm Hg blood pressure, 80 beats per minute heart rate, 18 mm Hg CVP, and oxyhemoglobin saturation of 97% to 100% with an oxygen mask (6 L/min) before anesthesia. The arterial blood gas analysis showed pH 7.447, PaCO2 27 mm Hg, PaO2 77 mm Hg, SO2 95%, BE −5 mmol/L |
An 11-month-old female baby presented to the Department of Oral Medicine and Radiology with an asymptomatic swelling in the anterior region of the maxilla. Her parents noticed the swelling at the age of 6 months, which was slowly progressive initially and started growing rapidly for the last 2 months, thus preventing the baby from feeding. There was no history of medication during pregnancy and no relevant medical history was present. Growth and development of the infant were normal for her age. On clinical examination, a non-pigmented, non-ulcerative, non-pedunculated swelling of size 4 cm × 3 cm was seen in the pre-maxilla region with facial asymmetry []. The swelling obliterated the gingivolabial sulcus pushing the upper lip forward and upwards with flaring of the lower part of the nose. The mucosa overlying the swelling was intact and both the primary central incisors (51, 61) were displaced apart []. On palpation, the swelling was non-tender, firm in consistency and non-fluctuant. There were no visible or palpable pulsations. Radiographically, the lesion appeared as a mixed radiolucent and radioopacity with ill-defined and irregular borders suggestive of both osteolytic and osteogenic reaction []. Routine laboratory investigations were within normal limits, except for urine levels of vanillyl mandelic acid (VMA) that were elevated, suggesting that the tumor is of a neuralcrest origin. The pre-operative diagnosis was made as MNTI based on the fine needle aspiration cytology report showing dual population of small neuroblast-like cells and large melanin-containing epithelial cells. An incisional biopsy of the tumor was performed under general anesthesia and the specimen was sent for histopathology. Microscopically [], the specimen showed an intraosseous lesion that eroded the bone and was covered by intact overlying mucosa. Bone destruction was also seen at the periphery of the tumor while the center of the lesion did not contain any bone fragments. The background of the tumor was fibrotic and contained irregular islands of tumor cells. There appear to be two populations of tumor cells, the large melanotic (pigmented) type and the small (non-pigmented) type. On higher magnifications [], the small cells had morphologic features of primitive neuroepithelial cells while the larger cells had features of melanocytes. Based on the above histopathologic findings, the lesion was finally diagnosed as MNTI. A conservative surgical excision of the tumor was performed under general anesthesia along with curettage of the surrounding normal bone and extraction of primary central incisors. The patient's recovery was uneventful, without any post-operative complications. There was no recurrence of the tumor in the 2-year follow-up period. |
A 56 year old male presented with complaint of acute onset painless diminution of vision in the right eye of 3 days duration followed by similar complaint in left eye after 1 day which deteriorated over the next 2 days. This was associated with headache and pain on ocular movements. There was a history of being bitten by a stray dog 8 days before the visual symptoms, and the patient had received three injections of chick embryo cell anti-rabies vaccine (Rabipur, Hoeshst Marion Roussel) on day 0, 3 and 7 after the dog bite. He developed these symptoms after the third injection. There was no history of any other ocular or systemic problems.\nThe general physical examination was within normal limits. The best-corrected visual acuity was 6/60 in the right and 6/24 in the left eye. IOP was 16 mm of Hg by applanation tonometry. Anterior segment examination using slitlamp was normal. Fundoscopy using + 90 D revealed hyperemic optic disc with blurred margins and disc edema. Vessels were tortuous and macular star was present in the nasal part of macula of both the eyes (Fig ). Pupils were sluggishly reacting in both the eyes. Goldmann perimetry revealed centrocaecal scotoma in both eyes which was more dense in the right eye as compared to the left.\nThe routine blood investigations were all within normal limits. The aerobic and aerobic cultures were sterile. The VDRL test and indirect haemagglutination test for syphillis and toxoplasmosis were normal. Serology for bartonella was negative. Mantoux test showed a reading of 8 mm. and the chest X-ray was normal. Magnetic resonance imaging (MRI) brain and optic nerves revealed slight signal enhancement of both the optic nerves with slight increase in optic nerve thickness on axial T2-weighted magnetic resonance imaging. No other significant abnormality was seen in MRI of brain.\nA diagnosis of bilateral neuroretinitis was made. As the dog which had bitten the patient was kept under observation and was alive after one week, further injections of antirabies vaccine were stopped. The patient was given three doses of pulse dexamethasone 100 mg in 150 ml of 5% dextrose. The visual acuity improved to 6/6 in both the eyes over the next two weeks. The disc oedema start resolving within one week but macular exudates took 4 months to resolve. |
A 38 year old female, housewife, came to our outpatient department with chief complaints of pain and swelling over right knee since 6 months. Pain was mild and activity related to start with and progressed to moderate and continuous. There was no history of precedent trauma, no history of fever, weight loss and anorexia. Patient had not solicited any treatment for this. On examination, there was diffuse bony swelling with crunchy ‘feel’ over right femoral condyles more localized to left side, extending to popliteal fossa. Overlying skin and muscles were free and mobile. Patient was unable to squat and walk painfree with knee ROM halted to 50 degree flexion but no fixed flexion deformity and full terminal extension possible.\nOn plain radiographs, a distinct expansile radiolytic lesion was found in lateral femoral condyle, with articular step off , but no cortical breach on medial, lateral or posterior side, with thinned out nonsclerotic margin, graded as Campanacci Grade 2 (). MRI was done, which reaffirmed heterogeneous hyperintense lesion measuring 55x41 mm in epimetaphyseal region of lateral femoral condyle, with no soft tissue involvement(&). A confirmatory biopsy was taken with all precautions for future procedure. Biopsy track was marked so as to include in approach during resection.\nOn confirmation of GCT in biopsy, a marginal resection and curettage with articular reconstruction and PMMA cementing for skeletal support was planned. Under spinal anesthesia, with tourniquet, an anterolateral parapatellar approach was taken, with view of future need of knee arthroplasty. After adequate visualisation of tumor extent and articular margin, marking was done for wide resection (&). Surgical mops were placed before opening the marrow, to avoid surrounding tissue contamination. Cortical breakout of lateral femoral wall occurred midway and curettage was done till the extent of viable bleeding bone. Cauterization of margins was carried out followed by denaturation of margins using 5 % Phenol soaked mop(&). Articular margin of lateral knee joint was reconstructed after elevation of subchondral bone and fixation using 1.8 mm threaded K wires (). Bone defect was again curetted and prepared for cementing. PMMA cement, dual dose, was prepared using vacuum mixing technique, and the bone defect was filled with PMMA cement(). Threaded K wires were cut short, with ends for future retrieval. Closure was done in layers after tourniquet release and adequate hemostasis control.\nPatient was advised immediate ROM exercises on postoperative day 1 and allowed Full weight bearing with support. Patient could gain 90 degree flexion of knee with 10 degree terminal extension lag and Straight leg Raise possible (-).\nAfter 3 months, Postoperative radiology was done to see status of distal femur(). Chest xrays were taken every 3 months. Patient was followed for 2 years and has shown no signs of recurrence, cement subsidence or K wire loosening (Figure 15-17). |
A 59-year-old Japanese woman with hypertension and diabetes mellitus was admitted to our hospital to undergo pelvic surgery. She had a presacral mass that was incidentally detected on CT before cholecystectomy for cholelithiasis, and adenocarcinoma was detected in the mass via endoscopic ultrasound-guided fine needle aspiration through the rectum in a prior hospital. Complete blood count, liver function, basic metabolic panel, and urinalysis were unremarkable. CEA, CA19-9, and CA125 were within their normal ranges.\nA pelvic radiograph revealed a scimitar sacrum with a right-sided defect []. Contrast-enhanced CT revealed two presacral masses in the pelvis. The upper mass had cystic and solid components containing subtle calcification, and the second mass was a simple cyst through an enlarged neural foramen []. Pelvic MRI illustrated the upper mass with cystic and solid components with various intensities on T1- and T2-weighted images (T2WI). The other presacral cystic lesion displayed high intensity on T2WI, and it had no mural nodule, which was suggestive of an anterior meningocele []. The patient was diagnosed with a malignant tumor associated with CS preoperatively, and she underwent surgical resection of the presacral mass in the pelvis. The tumor was completely resected, although the lower cystic lesion was accidentally ruptured because of adhesion.\nPathologic examination confirmed that the cystic and solid tumor contained adenocarcinoma and neuroendocrine tumor (NET) components (G2) []. The resected tumor included many cysts covered by stratified columnar epithelium, smooth muscle tissues, and a glomus tumor. Subtle fat tissue was present in the resected tumor, although it was not detected on preoperative MRI. Histologically, the tumor contained several different tissue types, suggesting a teratomatous nature. Most cysts inside the tumor that exhibited varied intensity on MRI included necrotic tissues. Adenocarcinoma was found in the cyst-like structure, and it was not distinguishable from other cysts on preoperative image examinations []. The adenocarcinoma component exhibited ductal and papillary structures, and a diagnosis of moderately differentiated adenocarcinoma was rendered. In the NET component, which was close to the adenocarcinoma lesion, immunohistochemical staining was positive for synaptophysin and chromogranin A. Residual NET tissue was observed along the surgical margins, and postoperative adjuvant radiotherapy (50.4 Gy/28 fr) was administered without adjuvant chemotherapy. After 8 months of follow-up, contrast-enhanced CT and pelvic MRI revealed no tumor recurrence, but genetic testing has not been performed in line with the patient's preference. |
A 76-year-old man was referred to our hospital for having lower back pain for 5 months, which was suspected to be L2/3 spondylitis on magnetic resonance imaging (MRI). He had previously undergone cervical laminoplasty for ossification of the posterior longitudinal ligament and diffuse idiopathic skeletal hyperostosis 19 years earlier. He had a history of hypertension and diabetes. Although a history of BCG vaccination was unknown, he had no previous history of tuberculosis infection. He had also undergone transurethral resection of a bladder tumor (TUR-Bt) and had been treated with intravesical mitomycin C (MMC) for the bladder cancer 1.5 years earlier. Four months later, he again underwent TUR-Bt, received intravesical MMC, and started BCG therapy for the recurrence of bladder cancer. After the sixth course of intravesical BCG therapy, he was aware of systemic weakness and loss of appetite, and was unable to walk. He appeared to have septic shock and therefore was treated in the intensive care unit. In spite of systemic analyses, the source of his infection and the causative bacteria could not be identified. He was subsequently diagnosed with hypercytokinemia caused by BCG therapy. He showed clinical improvement without the administration of antituberculosis drugs, and was discharged 9 months before he came to our hospital.\nExcept for a temperature of 37.2 °C, his vital signs were within normal limits. Although there were no motor and sensory disturbances in the legs, he was unable to walk owing to lower back pain. Physical examination demonstrated vertebral tenderness at the L2/3 level. Laboratory analysis demonstrated a normal white blood cell (WBC) count of 6300 /μL, a high erythrocyte sedimentation rate (ESR) of 53 mm/h, and a high C-reactive protein (CRP) level of 2.7 mg/dL. There were no other abnormal laboratory findings regarding anemic changes, kidney function, or liver function. The patient did not receive tuberculin skin testing.\nThere were no notable findings on electrocardiogram or chest X-ray. X-ray of the lumbar spine displayed collapsed endplates of L2/3. Sagittal T1-weighted MRI displayed a decreased signal in the L2/3 disc and the vertebral bodies (Fig. ). Sagittal T2-weighted MRI displayed an increased signal in the L2/3 disc and fluid collection in the anterior part of the vertebral bodies (Fig. ). Axial T2-weighted MRI displayed an increased signal around the posterior area of the vertebral bodies, which extended into the left epidural space and reached the peripheral muscle tissue and the area near the aorta (Fig. ).\nOn the second day of hospitalization, the patient underwent an L2/3 disc biopsy, but the general bacteria culture was negative, and the smear was negative for Ziehl-Neelsen staining. The patient’s blood culture was negative, and the result of T-SPOT.TB® (T-SPOT), which is a type of interferon-gamma release assay (IGRA), were also negative; the number of spots for both 6-kDa early secretory antigenic target (ESAT-6) and 10-kDa culture filtrate antigen (CFP-10) was 5 or less. The causative bacteria remained unidentified and therefore he was started on empirical therapy with intravenous ceftriaxone. On the fifth day of hospitalization, a plain computed tomography scan, which was performed for systemic examination, showed soft tissue development in the adjacent abdominal aorta at the L3 level (Fig. ), which was suspected to be an infectious aortic aneurysm. A 2-stage operation was planned for the spondylitis with adjacent infectious aortic aneurysm, to prevent rupture of the infectious aortic aneurysm, obtain spine stabilization, drain the abscess and make a diagnosis. On the tenth day in hospital, because the risk of rupture was considered to be low, the patient underwent an L2/3 laminectomy followed by posterior fixation using percutaneous pedicle screws at T12, L1, L4, and L5 as the first stage. Specimen cultures of the lumbar vertebrae, yellow ligament, necrotic tissue, etc., were negative for general bacteria, specimen smears were also negative for Ziehl-Neelsen staining, and there were no pathological findings of caseating granuloma or necrosis. On the fiftieth day of hospitalization, because the infection had been controlled but the inflammatory response was sustained, he underwent replacement of the aneurysm with a synthetic graft by vascular surgeons, and lesion curettage and L2/3 anterior interbody fusion by iliac bone transplantation as the second stage. Spinal surgery was performed in the transabdominal approach owing to the risk of rupture. On pathological analyses, the L2/3 intervertebral disc, vertebral bone, and tissue surrounding the vertebral bone and aorta showed caseating granuloma and necrosis with multinucleated giant cells and epithelioid cells upon hematoxylin-eosin staining, and positive bacilli upon Ziehl-Neelsen staining. The tuberculosis-polymerase chain reaction (Tb-PCR) result of the tissue was also positive, using COBAS® TaqMan® MTB Test, which is a real-time PCR system targeting the 16S rRNA gene region of Mycobacterium tuberculosis complex DNA. Owing to the patient’s history of BCG therapy, negative T-SPOT, pathological findings, and positive Tb-PCR, the pathogenic bacteria of the spondylitis was considered to be BCG. We then started multidrug therapy with antituberculosis drugs, including isoniazid (INH), rifampin (RFP), and ethambutol (EB), because BCG is typically resistant to pyrazinamide (PZA). PCR-based genomic deletion analysis was performed using the specimens to distinguish BCG from the other M. tuberculosis complexes. Specifically, multiplex PCR was performed utilizing region of difference 1 (RD1), which is present in the DNA of other M. tuberculosis complexes but is deleted in the DNA of BCG []. Primers ET1, ET2 and ET3 bind and amplify a 190-bp region in BCG, whereas a 160-bp region is amplified in the other M. tuberculosis complexes, as observed by electrophoresis on an agarose gel. A clinical isolate sample from our patient was identified as BCG with a deletion in RD1 (Fig. ). Specimen cultures from the first-stage and second-stage operation were later identified as members of the M. tuberculosis complex using a mycobacteria growth indicator tube. After therapeutic intervention, the patient’s WBC count, ESR level, and CRP level were improved and MRI displayed no signs of active infection in the spine, epidural space, peripheral muscle tissue, or aorta. On the ninetieth day after hospitalization, the patient was discharged from our hospital and transferred to a different hospital for physical rehabilitation. |
A male patient aged 31 was admitted to the Neurological Department complaining of burning pains and numbness of the distal parts of the legs, and difficulty in walking due to severe pain. His temperature rose to 39°C and symptoms of nasal congestion appeared. Four days after the onset he was hospitalized with the diagnosis of Guillain-Barre Syndrome. However two weeks after admission his general conditions deteriorated with increasing neurological abnormalities. His temperature suddenly increased again to 39°C. Pain burning and numbness increased in the legs and the same symptoms appeared in the hands. He complained of asymmetrical weakness of the distal parts of the legs and small hand muscles, worse on the left. Marked oedema of feet and toes appeared which spread over the lower legs. There was a change of skin of feet toes and lower legs.\nThe pulse was 96, the blood pressure increased to 160/90 mmHg. The patient was normally orientated in place and time. Cranial nerves were normal. Chest and abdomen were normal. The flexors of the neck were spared. All the small muscles of the hand were severely atrophied and a moderate atrophy of the distal part of forearms was seen. Muscular strength was measured by MRC scale (grade 1-5): shoulder girdle, deltoid, upper arms and extensors of the wrists, grade 5; extensors digitorum communis and flexors of the wrists, grade 3; interossei volaris and dorsalis and lumbricales, grade 1-2, with both side involved, more clearly on the left. Muscle tone in the upper limbs was decreased. Deep reflexes of the arms were reduced. Abdominal reflexes were absent. There was a moderate atrophy of the thigh and a severe atrophy of shin muscles. Proximal muscle power including hip and knee flexion and extension was normal. There was no movement of the ankle and toes on the left, and power was reduced to 1-2 grades on the right. Muscle tone and knee reflexes were reduced. The ankle jerks and plantar responses were absent on both sides.\nThere was dysesthesia, hyperpathia and hyperesthesia of all kinds of sensation in the toes, feet and lower third of the lower legs. Lasègue sign was positive. Palpation of ulnar, radial and especially peroneal communis, tibial and dorsal pedis nerves was painful. Palpation of arm and leg muscles was painless. Dense oedema of the lower legs and feet worsen on the left. Purple cyanosis and hemorrhagic foci were seen on the skin of the feet, lower legs, back of the hands and distal part of the left arm. There were necrotic changes of the distal phalanges of the first and third toes of the left foot.\nBlood analysis showed a decrease of Hb (to 117 g/L) and erythrocytes (to 3.5 x 10/1), moderate leukocytosis up to 9.3 x 10/1. ESV increased to 69 mm/h. The blood levels of sugar, bilirubin, creatine, urea, cholesterol, serum protein, potassium, calcium and phosphorus were within the normal limits; alpha-1 and alpha-2 globulins were increased up the normal levels. Blood levels of ALT and AST were increased up to 97 U/I (normal, less than 43 U/I) and 156 U/I (normal, less than 34 U); CK was greatly increased up to 2190 U/I (normal below 190 U/I). There was proteinuria 0.39 g/L.\nECG showed a sinus tachycardia of 110 per min. Fibrogastroscopy was normal. Repeated chest X-ray radiographies showed signs of slight oedema of the lungs and possible pericarditis. Ultrasonography showed slight hepatosplenomegaly.\nNeedle EMG (gastrocnemius, tibialis anterior, extensor digitorum communis and abductor pollicis brevis muscles) showed many fibrillation and single/moderate fasciculation potentials, with positive sharp waves at rest. Many long duration and increased amplitude of MUPs appeared on minimal voluntary contraction. On maximum contraction a discrete pattern was evident. 25-33% of the action potentials were polyphasic, suggesting a neurogenic process.\nOn electric stimulation of the saphenous and afferent fibres of median nerves the motor responses were absent. Sensory right ulnar nerve conduction velocities were greatly decreased in distal and mildly slowed in proximal parts (14 and 44 m/sec, respectively). Terminal latency was markedly increased up to 7.9 msec.\nMotor nerve conduction velocities in the distal parts of right and left ulnar nerves were mildly slowed (44 and 38 m/sec, respectively), but in the proximal parts of ulnar and in the distal and proximal parts of the median nerves were within normal limits. The terminal latencies were increased (in median nerves 6.6 and 4.3 m/sec, and in the ulnar nerves 3/7 and 4 m/sec). The compound muscle action potentials (CMAPs) were markedly decreased in amplitude on stimulation of the ulnar (1 and 0.1 mV) and median (0.5 and 1.2 mV) nerves. On stimulation of the right peroneal nerve the very lower amplitude (0.7 mV) response was seen only in the tibial anterior muscle. Motor conduction velocity was greatly decreased to 24 m/sec (below fibular head-popliteal fossa).\nLeft motor ulnar nerve conduction showed partial conduction block between the elbow and wrist. The amplitude of the CMAPs was reduced by 79% and the area by 88%. The criterion for partial conduction block was a 50% or greater reduction in amplitude and a 40% or greater reduction in negative peak area of surface-recorded CMAPs obtained with proximal compared with distal stimulation of ulnar nerve, in the absence of increased duration of CMAPs more than 20%, according ().\nThese ENMG findings showed mainly axonal sensorimotor polyneuropathy with different degrees of affection of some nerves, more clearly seen in the lower limbs with partial motor conduction block in the left ulnar nerve (see ).\nThe biopsy (stain H.E. and Van Gieson) performed at the right gastrocnemius muscle showed various changes of the majority of epimysial, perimysial and endomysial arteries of medium and small sizes. Simultaneous atrophy of almost all muscle fibres with knots and chains of dark sarcolemmal striations were invisible and sarcolemmal nuclei disoriented. In the transverse section, in some regions uniform small fibers with degenerative changes, dark cluster nuclei and mononuclear infiltration were also seen. In other regions of the sections increased variation of the diameter of muscle fibres, loss of few fibers, focal necrosis of some muscles with phagocytosis, increased of connection tissue in the endomysium and cellular infiltration were evident. The skin biopsy from right lower leg, at the site of muscle biopsy, showed smoothing of the papillar layer and oedema with inflammatory mononuclear infiltration in the arterial and venous walls ().\nCross section of biopsy of right gastrocnemius muscle (formol-calcium, Van Gieson stain) ().\nThe muscle biopsy findings were compatible with productive inflammatory vasculitis that resembled polyarteriitis nodosa with secondary neural muscular atrophy and nonspecific myositis.\nThe patient was treated with intravenous infusions of dexamethasone 150 mg daily for 5 days, then prednisolone – 500 mg daily – for 5 days and later with prednisolone, 90 mg/day per os in association with intravenous infusion of cyclophosphamide, 200 mg/day for 15 days, followed by cyclophosphamide, 150 mg/day per os, and plasmapharesis.\nSensory and motor disturbances decreased and the patient began walking with a cane. The skin swelling decreased, ESR and leucocytosis fell, but a pronounced trophic and vegetative vascular disorders persisted in toes. The necrosis of the toes gradually progressed to dry gangrene. The patient was transferred to a surgical department; further he developed a progressive heart, lung, liver and kidney insufficiency, despite a moderate regression of the neurological symptoms. The patient suddenly died of thromboembolism of the pulmonary artery one year after the onset of the disease. The autopsy confirmed the diagnosis of polyarteritis nodosa. |
We report the case of a 58-year-old female who was admitted to the Medicine Department of S.C.B Medical College and Hospital, Cuttack in june 2010 because of generalized lymphadenopathy and vomiting. Her peripheral blood smear showed leukocytosis with a WBC of 93,500/microliters, and the bone marrow picture revealed a predominance of blast cells. The blasts were negative for peroxidase, alpha-naphthyl butyrate esterase and PAS. A diagnosis of AML, M0 was made, based on the FAB criteria. She was treated with 3+7 induction therapy, where daunorubicin was given in a dose of 60mg/m2/day for 3 days and cytarabine in a dose of 100 mg/ m2/day for 7 days. Post induction remission chemotherapy bone marrow evaluation confirmed the achievement of complete haematological remission. Subsequently she received high dose cytosine arabinoside in a dose of 3 gm/m2/dose. After 9 days of this high dose chemotherapy she developed mucosal toxicities and high fever with cough during the hospital course. This febrile episode was associated with neutropenia with absolute neutrophil count 500 mm3. So empirical antimicrobial therapy with cefepime and amikacin was started. Although there was a slight decrease in her temperature and moderate improvement in her general condition, she developed sudden tachypnea, dyspnea, fever, bilateral pulmonary infiltrates and acute respiratory distress on the sixteenth day for which she had to be put in a ventillator. Blood, urine and nasopharyngeal swab cultures were negative. Radiological investigations revealed the presence of bilateral consolidations. Pulmonary CT scan was performed which showed bilateral diffuse pulmonary infiltrations suggesting fungal or bacterial pneumonia. There was also no sign of pericardial involvement. Diagnosis of invasive pulmonary aspergillosis (IPA) was done on the basis of isolation of an Aspergillus species from the sputum and from bronchoalveolar lavage samples. The admission electrocardiogram (ECG) had revealed normal sinus rhythm. Basing on the laboratory diagnosis she was administered a single loading dose of 70-mg of caspofungin as intravenous infusion over a period of 1 h. But 45 minutes after the start of the infusion, She developed bradycardia and hypotension which did not respond to frequent fluid boluses. The ECG showed complete atrioventricular (AV) block. In spite of cardiopulmonary resuscitation, the patient died of complete AV block on the same day. |
A 72-year-old man was referred to our cardiovascular center with previous history of hypertension for 5 years and a 2-month history of worsening dyspnea on exertion that motivated him to seek medical attention. Atrial fibrillation was detected during a recent medical check up at a local clinic, and the patient was referred to our hospital and admitted for closer examination. The laboratory data was unremarkable. His electrocardiogram revealed new-onset atrial fibrillation compared with previous data. A chest X-ray film revealed no particular anomalies. Two-dimensional transthoracic echocardiography (2D TTE) showed 1.04×0.87 cm nodular mass attached to the aortic valve. The echogenic mass originated from the ventricular side of the non-coronary cusp of the aortic valve (). Based on these findings, a papillary fibroelastoma of the aortic valve was suspected. Because of his old age and atrial fibrillation, scheduled surgery was performed to avoid systemic embolization. After aortic cross clamp, the aorta was opened. Inspection of the aortic root revealed a normal structure. A mass was found on the ventriclular side of the non-coronary cusp. The mass was successfully excised from the aortic valve (). The tumor measured 1.5 cm and had a smooth surface with a spherical shape. The aortic valve was repaired rather than replaced after tumor resection. Intra-operatory transesophageal echocardiography showed normal left ventricular outflow without regurgitation and no residual mass.\nHistopathological examination of the tumor revealed a benign soft tissue tumor mass. The margin of the mass was irregular but was easily distinguishable from the normal valve tissue. The tumor consisted of plump gray purplish myxoid stroma and slender and long spindle stromal cells. The stromal cells had eosinophilic scanty cytoplasms and dark small nuclei. Fibrocollagenous bands of aortic valve tissue were also noted in the myxoid stroma (Hematoxylin and eosin; ×200) (). The final pathologic diagnosis was a myxoma of the aortic valve. The patient's postoperative convalescence was uncomplicated and he has discharged on aspirin with regularly scheduled follow-up. The post operative echocardiogram showed normal aortic valve function. No residual tumor was evident on the postoperative TTE 7 months after surgery. |
A 19-year-old male patient complained of shoulder pain with no trauma history. He reported that the pain started about 6 months before, with progressive worsening. Pain was present in elevation and abduction, especially above 90°. The pain caused him to interrupt his physical activities, although he did not notice worsening during bodybuilding.\nA winged scapula was identified in physical exam, with scapulothoracic grade III dyskinesia, according to Kibler et al. []. He presented infraspinatus atrophy, complete range of motion, preserved upper limb strength, and discrete paraesthesia at the region of the medial border of the right scapula, with no other signs. There were no clinical signs suggestive of rotator cuff injury or glenohumeral instability ().\nThe imaging exams did not show significant changes. Shoulder MRI showed no rotator cuff lesion, labral lesions, cysts, or other soft tissue involvement. The cervical spine MRI did not show cervical discopathy or syringomyelia. Electroneuromyography with evoked potential of the scapular girdle evidenced diffuse axonal involvement of the long thoracic nerve, without other alterations.\nThe patient was then referred to the physiotherapy service, where he initiated a program of shoulder girdle rehabilitation focused on analgesia and passive mobilization. During ten weeks, he remained under the care of physiotherapists twice a week, but he did not notice an improvement in the pain. When he returned to the orthopedic clinic, he was informed about the possibility of surgical treatment.\nThe patient insisted on conservative treatment. We recommended strengthening of the shoulder girdle and swimming. During three months, he practiced swimming three times a week under the guidance of a physical education professional with experience in athlete training. In order to strengthen the periscapular muscles, he tried to practice the four classic styles of swimming, using floats in the lower limbs and increasing the demand on the upper limbs. Progressively, he noticed an improvement in his pain.\nThe swimming program consists of a 60-minute pool training three times a week, with increasing distances. The front crawl, breaststroke, and backstroke were alternated during training. The main set was a target mile, split in 200 m lengths with 1-minute rest (in a 25 m pool) and alternating strokes.\nHe returned after 90 days, free of pain. He presented dynamic stabilization of the scapula during elevation, and dyskinesia was no longer perceived. The force remained unchanged, but atrophy was no longer identified. The patient was satisfied with the progress made and was encouraged to stay in the muscle strengthening program in aquatic activities (). |
A 19-year-old male fell onto his left wrist and left elbow while working on a roof, reporting immediate pain and swelling in his left wrist and elbow. On examination, there was significant soft tissue swelling with an obvious deformity in the left wrist and left elbow; skin and neurovascular functions were intact. Initial radiographs requested in the emergency department showed a comminuted intraarticular left distal radius fracture with mild dorsal angulation (Figures –) and a posterior dislocation of the left elbow with a displaced fracture fragment along the anterolateral aspect of the elbow (Figures and ). In addition, there was a mildly displaced ulnar styloid fracture and nondisplaced scaphoid wrist fracture. Subsequently, a CT and an MRI was requested, showing evidence of a coronoid process fracture as well as high-grade to complete tears of the lateral ulnar collateral and radial collateral ligaments. High-grade partial-thickness tears of the common flexor tendon were also seen. Due to the severity of the injury, the radiologist report could not rule out injury to the interosseous membrane and required clinical correlation. The elbow was reduced with the assistance of a C-arm followed by a closed reduction of the left wrist. The left upper extremity was stabilized with a double sugar tong splint with IO molding and ulnar deviation prior to the surgical fixation.\nSurgical treatment was managed on the third day of admission. A dorsal spanning plate was used to reduce the distal radius (Figures –). A Kaplan approach was used to perform an arthrotomy on the unstable elbow and reduce the radial head. Intraoperatively, hypermobility at the level of the distal radioulnar joint (DRUJ) and axial and coronal instability of the forearm was observed, confirming intraosseous membrane damage and an Essex-Lopresti injury [, ]. The lateral collateral ligament (LCL) was repaired, and a left internal joint stabilizer (IJS) was placed (Skeletal Dynamics, Miami, FL) (Figures and ). Good concentric flexion and extension of the elbow and radial head were achieved. A posterior splint was placed in the elbow with the wrist in neutral position to protect from any instability with passive range of motion being allowed.\nAt 6 weeks of follow-up, the patient presented to the clinic with minimal pain but diminished ROM about the elbow, forearm, wrist, and hand. He was able to flex and extend about the elbow from 30 to 90 degrees, pronate-supinate to 45 and 45 degrees, and there was limited flexion/extension about the thumb, index, and middle fingers. At 3 months, the spanning plate of the left wrist was removed and tenolysis of the extensor pollicis longus was performed due to adherence to the dorsal spanning plate (Figures –). Tenolysis of the adjacent extensors, including the digital extensors and wrist extensors, was also done, increasing flexion and extension of the wrist to 30 degrees in both planes. The IJS and dorsal base plate of the left elbow were removed, proceeding with an arthrotomy of the radiocapitellar joint to improve extension from 20 degrees to full extension (Figures –). Flexion also improved from 120 to 125 degrees. |
A 73-year-old right handed male presented to emergency room with the sudden onset of right hemiplegia and dysarthria. He was diagnosed as having intracranial hemorrhage of the left basal ganglia and temporal lobe. After 3 weeks, he was offered rehabilitation services for the auditory problem of being unable to differentiate voices, music, and environmental sounds. His past medical history included hypertension and right basal ganglia intracranial hemorrhage 13 years previous. There has been no definite sequelae of previous stroke. In the neuroimaging studies 2 weeks after the onset of the stroke, a non-contrast computed tomography (CT) scan and magnetic resonance imaging (MRI) showed intracranial hemorrhage of the left basal ganglia and temporal lobe area and a single photon emission computed tomography (SPECT) showed a moderate to severe perfusion decrease and impaired vascular reserve in left temporal lobe ().\nHe was an alert patient but, he did not follow oral commands or respond to environmental sounds. His verbal repetition and writing to dictation were severely defective with phonemic substitutions. Neuropsychological evaluations were performed 3 weeks after the onset of stroke. He obtained a score of 4/30 on the Mini Mental State Exam of Korea (MMSE-K) with verbal communication. He was diagnosed as having cognitive dysfunction. But he could comprehend written and gestural communication. A language assessment using the Korean Version of the Western Aphasia Battery (K-WAB) revealed that he had significant deficits in auditory comprehension, repetition, and writing to dictation, despite the presence of good reading, fluent speech, and spontaneous writing, without evidence of aphasia. He could speak complete sentences without paraphasia. The Token test was 0% for verbal communication but 20.9% (13 items out of 62) for written communication. He came close to be misdiagnosed as Wernicke's aphasia but, his language disorder was different from Wernicke's aphasia for naming, reading and spontaneous writing. He obtained a score of 21/30 on MMSE-K with written communication. Therefore, he was regarded as not having severe cognitive dysfunction for his age and educational status. He could not differentiate people's voices, music, and environmental sounds. Everything sounded like a disagreeable noise, a "buzzing", including pure tone signals. Standard pure tone threshold audiometry was conducted. The standard pure tone threshold audiometry showed normal findings (). Also brainstem auditory-evoked potentials (BAEP) exhibited normal bilateral latencies (). The visuo-cognitive and visuo-spatial functions were preserved. The results of evaluation studies are summarized in .\nHe could hear sounds but had difficulty in identifying words and environmental sounds such as: male and female voices, music, a baby crying, laughing sounds, an animal crying (cat, dog, cow, horse, pig, bird), instrumental sounds (piano, clocks, telephone ring), wind, a wave, and honk. Therefore, he was diagnosed as having generalized auditory agnosia with intracranial hemorrhage in the left basal ganglia and temporal lobe. He had speech therapy for variable sound differentiation and lip-reading with a listening program to foster communication skills. Six months after the recent stroke, a follow-up K-WAB and audiometry were performed and there was no significant interval improvement with the previous exam. Gradually he regained the ability to communicate and understand almost all the words used in daily life by trained lip-reading. |
Patient A, a 30-year-old-woman, was presented to our Neurology Department with loss of power and sensory disorders in both arms and legs. The patient suffered from signs of asthma and regularly abused alcohol, however, she never experienced sensory disorders before. Her symptoms have begun 9 days ago and started with tingling sensations from the abdomen to the toes. Five days later, she had coordination problems in both hands, combined with tingling sensations continuing to her neck. She also showed Lhermitte sign while flexing her neck.\nPhysical examination showed a well-oriented, moderately cared woman in a wheelchair, from which she could stand up only with the help of others. The cranial nerves were intact, power in both her arms was normal, but power in both her legs was Medical Research Council (MRC) scale 4 out of 5.\nGnostic sensitivity was reduced in both arms and legs, with the right side being more affected than the left side. Vibration sensitivity was absent in both her legs. Her reflexes on the arms and legs were symmetrically elevated, and plantar reflexes were according to Babinski on both sides. Her movements were atactic and unstable.\nThe patient was recently institutionalized for alcohol abuse and was smoking a pack of cigarettes a day. She admitted using 50 laughing gas whippets a day for the last 2 months. Unfortunately, the patient has not given her motive considering the use of this amount of laughing gas whippets. She was hospitalized and received further treatment at the Neurology Department.\nGiven her clinical state, our patient had both deficits in her corticospinal tract and the posterior funiculus on both sides, mostly localized in the cervical part of the spinal cord. A cervicothoracic MRI was performed, which showed a hyperintensity in the cervical spinal cord on T2-weighted pictures ( and ). These anomalies, combined with the frequent use of laughing gas, indicated a vitamin B12 deficiency–induced myelopathy. Laboratory findings showed a low serum vitamin B12, alongside both raised homocysteine and methylmalonic acid values, demonstrating the symptomatic vitamin B12 deficiency ().\nThe diagnosis of laughing gas–induced combined spinal cord degeneration was evident by these findings; intramuscular injections with 1000 µg vitamin B12 twice per week were started. The laughing gas use was discussed with the patient, alongside the necessity to stop at once.\nDuring hospitalization, her symptoms were gradually improving, but her walking was still quite atactic. After 4 weeks, she was referred to medical rehabilitation; after 1 month, she was fully recovered. |
A 34-year-old female with no significant past medical history presented to our clinic after experiencing a left second metatarsal stress fracture (Figure ). One year prior, while running errands around town, she suddenly felt a sharp pain in her left midfoot and promptly consulted an orthopedic surgeon who placed her in a boot. Six months later, after experiencing minimal improvement in her pain, a different orthopedic surgeon performed an open reduction and internal fixation by injecting 1 mL of bone cement into the diaphysis of the second metatarsal.\nOver the next six months, she noticed no meaningful improvement in her pain. At this point, she presented to our clinic for a third opinion. During our initial visit with her, she stated that her left foot felt different than her right at baseline.\nOn physical exam, there was no gross deformity of her left lower extremity. The skin was intact with a healed incision over the dorsal midfoot, and there was point tenderness to palpation over the second metatarsal. Active and passive range of motion of the ankle and transverse tarsal joint was full and painless. Strength was 5/5 in dorsiflexion, plantarflexion, inversion, and eversion. Sensation to light touch was intact, Achilles reflex was present, and dorsalis pedis and posterior tibialis pulses were palpable.\nLaboratory work revealed an elevated erythrocyte sedimentation rate of 36 (reference range: 0–20) and C-reactive protein of 34.74 (reference range: 0–10.9). Plain radiographs and a computed tomography (CT) scan of the left foot showed diffuse sclerotic changes and cement within the left second metatarsal (Figures , ). Magnetic resonance imaging (MRI) showed diffuse edema of the left second metatarsal with a non-displaced fracture line (Figure ).\nAll treatment options were discussed with the patient and she agreed with undergoing operative fixation. In the operating room, cultures and a bone biopsy of the left second metatarsal were taken. After performing an osteotomy, curettage was performed to remove the injected cement. Open reduction and internal fixation was performed utilizing a plate and calcaneal bone graft (Figure ). The patient was discharged home on the same day with adequate pain control and a bone stimulator. X-rays taken at two weeks post-revision surgery are shown in Figure .\nAt one-month follow-up, her incision was healing well without signs of infection and she had no complaints of pain. At her most recent appointment—three months post-revision surgery—she again reported no pain and good functional recovery with physical therapy. CT scan at three months post-revision surgery showed appropriate alignment of the healing second metatarsal with intact hardware (Figure ). |
A 40-year-old gentleman presented with history of insidious onset headache for 6 months and diminution of vision for 2 months. The headache was holocranial, continuous and of mild to moderate intensity. It was not associated with nausea, vomiting, photo or phonophobia. Since 2 months, he noticed progressive diminution of vision in both eyes. He did not have seizures, altered sensorium or any focal deficit. There was no history of fever. He was previously healthy and was not on any medications. On examination, blood pressure was 120/80 mmHg, general and systemic examination was normal. On neurologic examination, there was optic atrophy in both eyes. Visual acuity in the right eye was 6/60 and there was absent perception of light in the left eye. Rest of the neurologic examination was normal. There were no signs of meningism. A diagnosis of raised intracranial pressure with post-papilloedema optic atrophy was made. A differential diagnosis of chronic meningitis, idiopathic intracranial hypertension and chronic venous sinus thrombosis was made. A computed tomogram (CT) scan of the brain [] with contrast showed mild communicating hydrocephalus. There was no mass lesion, cerebral edema or meningial enhancement. Magnetic resonance venogram (MRV) showed near-complete thrombosis of the right transverse sinus and partial thrombosis of the left transverse and the sigmoid sinus []. Cerebrospinal fluid (CSF) manometry demonstrated opening pressure of 30 cm of CSF in the lateral decubitus position. Twenty of CSF was drained and the pressure reduced to 15 cm. The CSF did not show any cells, while proteins were 5 mg/dL and sugar was 99 mg/dL (blood sugar of 130 mg/dL). India ink preparation and cryptococcal antigen were negative. Post CSF drainage, the headache promptly decreased in intensity. Two days later, the headache recurred. CSF manometry was repeated, where the opening pressure was 27 cm. CSF was drained and pressure was reduced to 15 cm of H2O. There was relief of headache for the next 2 days. Two days later, the headache recurred and CSF manometry (pressure of 30 cm) with drainage of CSF was repeated for the third time, which resulted in prompt relief of headache. In view of persistently elevated CSF pressure and presentation similar to IIH, a thecoperitoneal (TP) shunt was performed. The patient had relief of headache following TP shunt. Post TP shunt, he was started on low-molecular heparin followed by warfarin for venous sinus thrombosis. He did not have headache for the next 4 weeks but there was no improvement in vision. After 4 weeks, the headache recurred and was severe in intensity. CT scan of the brain did not show any significant change. In view of failure of TP shunt to relieve headache and persistence of hydrocephalus, a ventriculoperitonial (VP) shunt was placed. Post VP shunt, there was relief of headache and reduction in ventricular size but no improvement in vision. Six months later, he is free of headache although there is no improvement in vision. He is on oral anticoagulation for chronic venous sinus thrombosis. |
A 55-year-old female with a history of renal cell carcinoma of the left kidney metastatic to the bony pelvis, lungs, mediastinum, and spleen presented to the emergency department with shortness of breath, pleuritic chest pain, and left scapular pain. She presented to the same emergency department one week prior with pleuritic chest pain but was discharged home after pulmonary embolism was ruled out.\nShe was diagnosed with renal cell carcinoma of the left kidney five years prior after presenting with gross hematuria. At that time, she underwent left radical nephrectomy. One year later, she developed a metastatic lesion in the bony pelvis for which she underwent radiation therapy. She as treated with pazopanib for two years with stable disease but stopped due to gastro-intestinal toxicity. Therapy was switched to nivolumab, which was discontinued after six months due to grade four pancreatitis and grade two rash. Eight months prior to her current presentation, she underwent radiation treatment to metastatic lesions in the left pubic symphysis and spleen. The patient initiated therapy with cabozantinib, a tyrosine-kinase-inhibitor used to treat renal cell carcinoma, three months prior to her current presentation.\nOn physical examination, she was wheezing in all lung fields and hypoxemic requiring supplemental oxygen. She had prior 12-pack-year smoking history but no formal diagnosis of chronic obstructive pulmonary disease (COPD). A chest x-ray revealed a small left pleural effusion and left basilar atelectasis. Laboratory workup, including complete blood count, renal and hepatic panels, and troponin, was unremarkable. An electrocardiogram (ECG) revealed sinus tachycardia without signs of ischemia. CT was not repeated due to her negative CT angiogram one-week prior. Given radicular and left scapular pain, an MRI of the spine was done, which revealed no pathologic metastases in the thoracic or lumbar spine but did reveal a new sacral lesion. Given her progressive stridor, she underwent laryngoscopy, which revealed a normal upper airway. A bronchoscopy showed significant trachea-bronchomalacia and thick purulent secretions in the left upper lobe, lingula, and right upper lobe.\nTwo days after admission, repeat chest X-ray revealed near complete opacification of left lung and large pleural effusion, a remarkably different radiograph from admission (Figure ).\nSubsequent CT chest revealed a large left pleural effusion with partial loculation as well as partial atelectasis of the left upper lobe and complete atelectasis of the left lower lobe. A right perihilar metastasis and perisplenic metastases were reported. The study was negative for pulmonary thromboembolism.\nThoracentesis revealed cloudy straw colored exudative effusion. A four French pigtail catheter was placed. Approximately 400 milliliters of yellow-green fluid was immediately drained. Pleural fluid studies revealed a white blood cell count of 33,000/μL (97% neutrophils), pH of 6.44, LDH of 4760 U/L, and an amylase of 394 U/L. She was started on vancomycin, cefepime, and metronidazole for presumed empyema. Pleural fluid cultures showed heavy growth of lactobacillus species, heavy growth of anaerobic gram negative cocci, and moderate growth of Candida krusei. Antimicrobial therapy was subsequently narrowed to ertapenem and anidulafungin. Given lack of improvement and continued significant chest tube output over the following week, further CT imaging was obtained, revealing a gastro-pleural fistula (via the left diaphragm and superior posterolateral stomach) with associated complex pleural effusion containing contrast material and gas (Figure ). This process abutted the known splenic metastases.\nAn esophagogastroduodenoscopy (EGD) revealed a 1.5-cm fistula in the posterolateral stomach that opened to the pleural space (Figure ).\nEndoscopic suturing was attempted to close the fistula with limited success (partial closure noted on imaging, with methylene blue dye taken via mouth visualized in the chest tube drainage catheter on water seal; Figure ).\nFor complete closure, the authors attempted a novel approach utilizing a venting gastrostomy tube and chest tube to water seal to facilitate closure of the fistula over the ensuing six weeks. Enteral feeding via jejunostomy tube to aid closure of the fistula was employed. The patient was continued on ertapenem and anidulafungin. She was also initiated on a proton pump inhibitor. She was discharged to a rehabilitation facility with plans to repeat imaging and methylene blue swallow in six weeks.\nUnfortunately, CT scans after six weeks showed that the fistula remained patent. A second attempt was made at endoscopic closure, which was again unsuccessful. One month later, during a hospitalization for electrolyte abnormalities, the patient decided to pursue elective surgical repair of the fistula in hopes of regaining the ability to resume normal oral intake. Four months after her initial presentation, she underwent laparoscopic surgery for fistula repair. The surgeon visualized extensive radiation fibrosis involving the stomach, spleen and retro-peritoneum. Given these findings and to avoid splenic bleeding, they pursued a conservative surgery whereby they stapled the stomach to ligate the gastro-pleural fistula anatomically. This approach is novel and was successful in our patient. A fluoroscopic upper GI series with oral contrast three days after surgery demonstrated no leakage of contrast outside of the GI tract or into the pleural space, and CT five days after surgery revealed no evidence of communication between the stomach and pleural space (Figure ).\nShe tolerated an oral diet. Gastrostomy tube, jejunostomy tube, and chest tube were removed without complication. |
An 85-year-old female patient with prior history of hypertension, was urgently admitted to an outside institution with symptoms of progressive dyspnoea and malaise of ∼1 week duration. The patient later revealed all symptoms being preceded by an episode of persistent substernal chest discomfort that she managed at home without contacting her primary physician. In the emergency department, she was found with electrocardiographic evidence of a recent acute myocardial infarction (Q in precordial leads V1–V4 with ST elevations and T-wave inversions). She was diagnosed with an anterior ST-elevation myocardial infarction, pretreated with aspirin 325 mg, and clopidogrel 600 mg and urgently underwent coronary intervention with a drug-eluting stent to an occluded intermediate artery. The rest of the coronary arteries were without stenoses. Echocardiography 24-h later showed akinetic anterior and lateral wall with an estimated ejection fraction of 40%. After initial clinical improvement, the patient abruptly deteriorated on the third day with onset of acute pulmonary oedema and cardiogenic shock. At this time, the electrocardiogram did not show any dynamic changes of the ST-T segments or any arrhythmias. Repeat coronary angiography was without any further stenosis and re-evaluation with echocardiography revealed new severe mitral regurgitation and no other abnormalities that could lead to shock, such as pericardial effusion—tamponade or ventricular septal defect (VSD). Clinical examination at the time revealed an elderly female in moderate distress, requiring supplemental oxygen (60% FiO2) for maintenance of a saturation of 92–93%, with orthopnoea, jugular venous distension, and bilateral inspiratory crackles on lung auscultation. There was a two out of six systolic apical murmur, without evident gallop rhythm and mild peripheral oedema. The patient was transferred to our institution for further management and possible surgery. Upon transfer, she was in New York Heart Association (NYHA) Class IV with persistent pulmonary oedema despite high doses of intravenous furosemide (180 mg/24 h), on high dose supplemental oxygen (60% venturi mask), and on dobutamine infusion at 3 μg/kg/min. A transoesophageal echo exam (TOE) revealed partial rupture of the anterolateral PM with flail anterior (A1 and A2 scallops) and posterior leaflet (P1 scallop) (, , Videos S1 and S2). This PM rupture and flail mitral valve produced severe acute mitral regurgitation, pulmonary oedema, and cardiogenic shock. The patient was evaluated by the heart team and was considered at prohibitive surgical risk [log EuroSCORE 43% (85-years-old female, critical preoperative state/cardiogenic shock, EF 30–50%, recent myocardial infarction) and STS score 13%]. Indeed, she was in intractable heart failure, despite maximal intravenous diuretics, with dyspnoea at rest, radiographic appearance of bilateral pulmonary oedema, and in need of intra-aortic balloon counterpulsation for stabilization. She was thus referred for emergency transcatheter mitral valve edge-to-edge repair with MitraClip (Abbott Vascular, Santa Clara, CA, USA), (, , Video S3). Two clips were implanted at the central and lateral part of the valve. The first clip was implanted at the A2–P2 scallop towards the lateral aspect of the valve. Due to the significant remaining regurgitation coming from the flail anterolateral commissure (, Video S4), a second clip was implanted lateral to the first within the commissure. The final result after implantation of these two clips was with an effective reduction of mitral regurgitation from torrential to mild-moderate, while creating a single orifice valve (‘zipping’ of the lateral commissure) with an estimated (with use of three-dimensional echocardiographic multiplanar reconstruction) residual valve area of 2.1 cm2 and a mean transmitral gradient of 6 mmHg (, , Video S5). Subsequent to the MitraClip procedure, the patient was weaned off both intra-aortic balloon counterpulsation and inotropic support (dobutamine and dopamine infusions). The patient was also switched to oral diuretics and had reduced supplemental oxygen demands. The post-procedural course was uneventful. She was discharged home a week later on standard heart failure therapy including metoprolol, ramipril, furosemide, spironolactone, and ivabradine. At the 20-month follow-up, she remains in stable clinical condition in NYHA class II, and no further interventions were required on the mitral valve. Echocardiography revealed moderate mitral regurgitation and a mean transmitral gradient of 6 mmHg. |
A 40-year-old gravida five, para four woman presented for workup and management of abnormal uterine bleeding. Her past medical history was significant only for hypertension and anemia. On review of her social history, she admitted to drinking six packs of beer on the weekends but denied further substance use. She denied previous treatments for her bleeding including any previous intrauterine device usage.\nUltrasonography revealed a 7 cm fundal fibroid with otherwise normal pelvic anatomy. She was initially offered medical management of her bleeding. She declined any medical treatment and strongly desired definitive surgical treatment. She then underwent a total vaginal hysterectomy with adnexal conservation. Due to the large size of the uterus, a myomectomy was performed to facilitate vaginal removal. Her postoperative hospital course was relatively uncomplicated and she was discharged home on postoperative day three.\nOn postoperative day ten, she presented to the Emergency Department (ED) for fever, worsening abdominal pain, and new onset of nausea and vomiting. In the Emergency Department, she was tachycardic and tachypneic but afebrile. Her exam was significant for abdominal tenderness to minimal palpation, vaginal cuff erythema, and significant tenderness to palpation of the vaginal cuff. Lab work showed an elevated white blood cell count. She was admitted for management of presumed pelvic infection.\nA CT of the abdomen and pelvis was obtained and showed a 6.2 x 9.7 cm pelvic abscess adjacent to the vaginal cuff (Figures and ).\nInterventional Radiology placed a drain into the abscess and the patient was started on IV piperacillin/tazobactam. She was transitioned to oral amoxicillin/clavulanate potassium after four days on intravenous antibiotics and her drain was removed on hospital day 5. Vaginal wound cultures remained pending; however, due to continued clinical improvement on the oral antibiotic regimen, she was discharged home on hospital day 5 with a two-week course of amoxicillin/clavulanate potassium.\nThe patient then returned for her outpatient visit approximately one week later. The results of the vaginal wound cultures revealed a large growth of Actinomyces meyeri. The patient's case was discussed with an Infectious Disease (ID) specialist who recommended an additional two-week course of amoxicillin/clavulanate potassium.\nThe patient then returned to the ED on postoperative day 25 for pleuritic chest pain with mild cough but denied gynecologic complaints. She reported compliance with the oral amoxicillin/clavulanate potassium regimen. Exam and lab work were unremarkable. A chest X-ray showed left basilar heterogeneous opacities, likely subsegmental atelectasis. A CT angiogram was obtained due to concern for a possible pulmonary embolism (PE). The imaging was negative for a PE; however, it was concerning for possible pneumonia. The patient was discharged home with a five-day course of levofloxacin for treatment of pneumonia.\nOn postoperative day 27, the patient represented to the ED with worsening shortness of air and chest pain. Again, she reported compliance with her antibiotic prescriptions. Exam and lab work were again unremarkable. A repeat chest X-ray showed a slight progression of right basilar heterogeneous opacities thought to be infectious. Her antibiotic regimen was again discussed with ID specialists and an intravenous antibiotic regimen was felt preferable to an oral antibiotic course. She then completed an outpatient two-week course of IV ampicillin/sulbactam as recommended.\nOn postoperative day 37, a repeat CT of the abdomen and pelvis showed near complete resolution of the previous pelvic abscess. HIV testing was obtained and returned negative result. She reported significant improvement of her symptoms. She was placed on a six-month course of oral amoxicillin per ID recommendations with plans for continued follow-up in their clinic, as well as with gynecology. She has not shown any signs of recurrent infection after approximately 1 year of follow-up. |
A 75-year-old woman was admitted to our intensive cardiac care unit (ICCU) because of recurrent VA episodes complicated by acute pulmonary oedema. The diagnosis of VA was firstly made in 2013, when she underwent coronary angiography after experiencing several rest angina episodes, especially at night and in the early morning. The angiogram excluded the presence of obstructive coronary atherosclerosis. After ergonovine administration, left circumflex coronary artery spasm was induced (Timeline table), and it was associated with chest pain and electrocardiographic changes with characteristics similar to those of spontaneous episodes. Since then, the patient continued to experience rare (one/year) and responsive to sublingual nitrates VA episodes while she was on chronic verapamil (240 mg/day) and isosorbide mononitrate (50 mg/day). In the 5 months preceding the index hospitalization, the patient had five more severe VA attacks, associated with acute heart failure, requiring hospitalization. She was then transferred to our ICCU after a new VA episode complicated by acute pulmonary oedema and haemodynamic instability.\nAfter clinical stabilization with intravenous calcium antagonists, nitroglycerine, and diuretics, coronary angiography was repeated and confirmed the absence of any obstructive vessels disease. Spontaneous coronary spasm was not observed due to achieved clinical stability and concomitant vasodilator therapy. In the following days, the patient experienced further abrupt episodes of VA. During them, the electrocardiogram showed new left bundle-branch block (LBBB), with concomitant echocardiographic evidence of posterior and anterolateral wall akinesia, severe mitral regurgitation (MR) secondary to posterior leaflet tethering, with reduction of left ventricular ejection fraction from 65% to 35% and increase in pulmonary systolic arterial pressure from 40 mmHg to 60 mmHg (). Despite maximal tolerated intravenous therapy (verapamil 3 γ/kg/min and nitroglycerine 0.125 γ/kg/min), the patient had once again a severe VA attack characterized by acute pulmonary oedema (O2 saturation from 98% to 87%) and cardiogenic shock (systolic arterial pressure decreased from 110 mmHg to 65 mmHg, blood lactate levels raised to 5 mmol/L). Non-invasive ventilation was immediately started (pressure support 10 cmH2O, positive end-expiration pressure 5 cmH2O, and O2 inspired fraction 80%) and current intravenous therapy was discontinued because of critical arterial hypotension and lack of coronary vasospasm improvement. Then, an intravenous infusion of levosimendan at a dose of 0.05 γ/kg/min was started without an initial bolus and it was continued at the same infusion rate for 48 h. Few hours after the beginning of levosimendan infusion, a progressive clinical improvement was documented, with a marked reduction of respiratory work and symptoms, restoration of normal arterial pressure and peripheral perfusion. The levosimendan infusion was well tolerated, without symptomatic hypotension requiring vasoconstrictors. In parallel, recovery to baseline electrocardiogram (incomplete LBBB with mild ST-segment depression and negative T-wave in leads D1 and aVL) was observed along with normalization of wall motion abnormalities and return to baseline MR (from severe to mild grade) at echocardiographic evaluation (Figure and ). After 15 days, the patient was discharged with the same initial medical therapy (aspirin 100 mg/day, verapamil 120 mg b.i.d., and mononitrate isosorbide 50 mg/day), and at 3-month follow-up, she was free of recurrences of VA while continuing oral calcium channel blockers and nitrates. |
A 44-year-old woman with a history of metastatic triple negative breast cancer and lung metastases presented with a six-month history of recurrent haemoptysis. She had no other significant medical history. She was initially managed for her right breast cancer with a wide local incision and adjuvant chemoradiotherapy in 2014; however, her malignancy recurred two years later. She had positive margins on subsequent right mastectomy and proceeded to excision of the right pectoralis major and overlying dermis. Six months later she was found to have bilateral pulmonary metastases and underwent initial diagnostic bronchoscopy identifying a bleeding mass in the medial segment of the right middle lobe (RB5), which was subsequently treated with topical adrenaline and biopsied – confirming metastatic disease. Her malignancy progressed despite palliative chemotherapy with epirubicin and cyclophosphamide, during which time she developed worsening haemoptysis of ~1/2 cup (~120 mL) daily. A multidisciplinary decision was then made to perform therapeutic bronchoscopy due to excessive distress caused to the patient because of haemoptysis. She underwent bronchoscopy using a therapeutic video bronchoscope (Olympus BF-TH190, Olympus Corporation, Tokyo, Japan) introduced via a rigid bronchoscope, which provided secure airway access. Endobronchial survey revealed the source of bleeding in the distal right middle lobe, although the actual bleeding source was not directly visible. A volume of 2 mL of TISSEEL was injected into the right middle lobe bronchus via a catheter followed by deployment of a size 6 Spiration (Redmond, WA, USA) IBV to add stability and prevent expectoration (Fig. ). A further 1 mL of TISSEEL was then applied over the valve (Fig. ). The procedure abolished the patient's haemoptysis instantly.\nTwo weeks later the patient developed recurrent haemoptysis; however, repeat bronchoscopy showed a different source of bleeding in the right lower lobe, with the existing combination TISSEEL and IBV still in place in right middle lobe and maintaining haemostasis. To control the new bleeding TISSEEL was injected in the right lower lobe bronchus distal to the opening of RB6, followed by deployment of a size 9 IBV. Further injection of TISSEEL was then applied and haemostasis was achieved. Unfortunately the patient was found to have brain metastases and died of her malignancy 10 weeks later, without recurrence of haemoptysis. |
A 72-year-old woman presented with a 6-month history of cervical pain radiating to the left arm. Her past medical history was not relevant. Clinical examination was consistent with C6 radiculopathy but without sensory or motor involvement. MRI and a cervical scan confirmed cervical disc protrusion and narrowing of the left foramina at the C5–C6 spinal segment.\nThe patient was first unsuccessfully treated with analgesics and non-steroidal anti-inflammatory drugs, so a CT-guided transforaminal cervical injection was discussed. This procedure was performed according to the current medical guidelines (). Immediately after the procedure the patient complained of general weakness followed by loss of consciousness and stiffness of the four extremities. She was immediately transferred to the emergency department.\nUpon her arrival at the emergency department, her blood pressure was 230 mmHg, heart rate was 100 bpm, oxygen saturation was 90% on room air and body temperature was 37.5°C.\nThe initial neurological examination showed a Glasgow coma score of 3/15 (E1V1M1), total quadriplegia and spasticity of the four extremities. Cranial nerve examination showed oculomotor palsy and skew deviation. The rest of the examination was normal. A complete blood count, electrolytes, blood gases and renal function tests were normal. The patient was intubated to protect the airway and further imaging tests were performed to rule out a cerebrovascular accident.\nCT angiography including the supra-aortic trunks was normal, but MRI showed acute bilateral ischaemic lesions in the thalamic nuclei, microlesions in the two posterior cerebral territories and bilateral lesions in the brainstem and cerebellar hemispheres (). These lesions as well as perilesional oedema were noted on the FLAIR sequence ( and ). The electroencephalogram (EEG) showed slowing of the baseline rhythm and absence of epileptic activity.\nThe presence of ischaemic stroke was confirmed. The patient was transferred to the intensive care unit where transoesophageal echocardiography was performed. The suspicion of cardioembolic stroke was formally excluded and cardiac monitoring showed no arrhythmias. No clinical improvement was observed after 72 hours of hospitalization so life-sustaining treatment was stopped, leading to the death of the patient. |
A 33-year-old Caucasian male was transported to the emergency department after a low-speed motorbike accident. He had a free medical record with occasional tobacco use. On presentation, the patient had normal vital signs: Blood pressure: 133/91 mmHg, heart rate: 72bpm, peripheral oxygen saturation (SpO2): 100 %, Temperature 36,8 °C. He complained of deep right upper quadrant and right flank at rest and right upper limb pain, without evidence of ecchymosis or lacerations on his chest or abdomen. Some superficial abrasions were visible on his right elbow and his right lower quadrant. Physical examination revealed mild tenderness to deep palpation in his right upper quadrant and right flank.\nThere were no abnormal signs on his blood analysis, metabolic or coagulation panel. Urinalysis revealed mild microscopic hematuria (20–26 erythrocytes per field vision). Chest, spine and pelvis plain radiography was negative for fractures or any other abnormal findings. A focused assessment sonography for trauma (FAST) performed by a clinical radiologist in the Emergency Department revealed no evidence of trauma. An abdominal contrast-enhanced computed tomography scan (CE-CT) was performed due to persistence of pain and high clinical suspicion. The CT revealed round enlargement of the right adrenal (4.2 × 2.7 cm) with increased density (60-70HU), periadrenal fat stranding and right diaphragmatic crus thickening without extravasation of the intravascular contrast. No injuries from other organs were spotted and the diagnosis of an isolated right adrenal hematoma (AH) was made (). As a result, he was admitted to the clinic for preservative treatment and further monitoring. He had stable vitals and body temperature throughout his hospitalization period and gradually improving pain after intravenous analgesia. He was discharged with oral antibiotics and analgesia for 6 days. A follow-up MRI scheduled in 7 days, showed no size reduction and acute AH characters with signs of possible active hemorrhage (), whereas, close affiliation of the structures posterior to the injured adrenal gland shows compression and chance of shape of the adrenal between the liver and the vertebrae (). The patient was free of symptoms and his urinalysis, metabolic panel and adrenocorticotropic hormone (ACTH) stimulation test performed were normal. New follow-up with abdominal MRI scheduled after 60 days revealed partial resolution of the AH, while the lesion that remained had elements of a chronic hematoma (). |
The patient is a 46-year-old right-handed female with a past medical history of hypertension (HTN), hyperlipidemia (HLD), diabetes mellitus type two (DM2), obesity, and hemorrhagic stroke who was transferred from an outside facility to be evaluated for CNS vasculitis. She was admitted to this outside facility for a four-week period prior to being transferred to the primary facility for further evaluation over a subsequent 23-day period. Total duration of hospitalization at both the facilities was close to 7.5 weeks. Approximately one week into the initial four-week admission, her family found that she was very lethargic with diminished responsiveness and pronounced difficulty speaking. In the emergency room (ER), her blood pressure was measured at 243/129 mmHg with a blood glucose value greater than 400 mg/dL. She was started on aggressive antihypertensive therapy and underwent a series of diagnostic tests. Dual antiplatelet therapy (DAPT) consisting of aspirin and clopidogrel was initiated in combination with high-dose atorvastatin. With respect to her lethargy and fluctuating cognition, there was concern that she may be experiencing complex partial seizures, so lacosamide was also started.\nA baseline computed tomography (CT) scan of the head without contrast showed multiple indeterminate lacunar infarcts involving the head of the right caudate nucleus and left corona radiata. The same day, a magnetic resonance imaging (MRI) was performed and elicited similar findings with the addition of bilateral punctate infarcts of the left thalamus, right periventricular white matter, and right centrum semiovale. Magnetic resonance angiography (MRA) done on the following day showed high-grade stenosis of the left middle cerebral artery (MCA), in addition to markedly diminished caliber of the right MCA and high-grade stenosis involving the left posterior inferior cerebellar artery (PICA). Bilateral carotid ultrasounds showed very mild plaques. An angiogram exhibited an occluded left posterior cerebral artery (PCA) distally and was also suggestive of advanced intracranial atherosclerosis (more so than would be expected in CNS vasculitis). There was no evident change from day two to day six of this hospital course. A spinal tap performed at the end of the first week demonstrated elevated protein and IgG synthesis rate (16.4), which was concerning for CNS vasculitis. Appreciating the contrast between the imaging and spinal tap findings, CNS vasculitis could not be ruled out. The patient was started on intravenous (IV) corticosteroids briefly, however, the medication was discontinued due to worsening hyperglycemia that was progressively difficult to control. Near the end of the third week of hospitalization, a repeat MRI showed a new small stroke in the left subcortical parietal white matter.\nThe patient was transferred to the primary facility after this initial month of hospitalization, at which time the patient had a National Institutes of Health Stroke Score (NIHSS) of seven. She was alert and oriented to person only and able to follow simple commands. Significant findings on subsequent blood testing revealed leukocytosis (12.2), elevated absolute neutrophil count (ANC) at 11.3, hyperglycemia (314 mg/dL), HbA1c of 9.6%, mildly elevated erythrocyte sedimentation rate (ESR) at 36, positive herpes simplex virus type one (HSV1), and the presence of IgG and hepatitis B core antibody (HBcAb). Workup for hypercoagulable state was negative for Factor V Leiden and antithrombin deficiencies, though notably protein C was elevated. A repeat spinal tap on hospital day one showed elevated levels of protein (122), but also demonstrated an elevation in myelin basic protein (6.09). Otherwise, the patient was afebrile and hemodynamically stable on admission. On hospital day two, rheumatology was consulted. In order to confirm the suspected diagnosis of CNS vasculitis, the specialist recommended a leptomeningeal biopsy and IV corticosteroids in the interim. Although angiography is very sensitive, it is nonspecific as it cannot distinguish between vasculitis and reversible cerebral vasoconstriction syndrome (RCVS). Consequently, the angiography that the patient had undergone earlier in her hospital course could not provide us with a definitive diagnosis, thus warranting the biopsy of the brain.\nA baseline transthoracic echocardiogram (TTE) obtained on hospital day three revealed a left ventricular ejection fraction (LVEF) of 57 +/-5 percent with mild dilation of the left atrial cavity. Repeat imaging showed much of the same findings, however, radiology recommended further workup for underlying CNS vasculitis. Over hospital day four to six, the working diagnosis was “multifocal bihemispheric strokes with no clear etiology with an encephalopathic process.” The patient’s cardiovascular risk factors continued to be treated and monitored (lipids, blood pressure, and sugars), and a more extensive rheumatological workup was ordered. Continuous electroencephalogram (EEG) monitoring on hospital day eight also showed evidence of diffuse encephalopathy although there were no epileptiform changes or seizures recorded. Over the second week of hospitalization, a new left cerebellar infarct was detected on MRI, at which time steroids were tapered down and a transesophageal echocardiogram (TEE) was ordered. The results of this echocardiogram were unchanged in comparison to the baseline TTE. There was no thrombus detected in the left atrium, ruling out cardioembolic etiology of the new stroke. The CT studies of the chest and abdomen were negative for any findings pertinent to the patient’s chief complaint.\nDuring the third week of hospitalization, a repeat head CT without contrast revealed additional recent infarcts. A four-vessel angiogram showed 50% stenosis in the petrous and cavernous segments of the left internal carotid artery (ICA), a completely occluded M1 segment of the left MCA, and multiple alternating foci of narrowing within the M2 and M3 branches of the right MCA as well as the P2 and P3 branches of the PCA. In consideration of these findings and given the fact that there is a considerable overlap between the imaging appearance of vasculitis and atherosclerotic disease, neither diagnosis could be excluded. A second rheumatology consult recommended that a leptomeningeal biopsy be considered prior to starting cyclophosphamide, effectively ruling in CNS vasculitis versus ischemic stroke. The neurosurgery team agreed to conduct the biopsy of the meninges and brain. However, after discussing the details of the procedure with the patient's family, her family decided against her having the procedure due to the risks associated with brain surgery and the debilitating neurologic deficits already suffered by the patient. |
A 64-year-old white woman presented with neck swelling, shortness of breath and neck pain in November 2011. Three months prior, she had been diagnosed with an unprovoked deep venous thrombosis of the left leg and thigh and initiated on warfarin for anticoagulation. The patient worked in a chair factory. She had quit her 30-year habit of smoking one pack a day in 1994.\nMedical history was significant for deep venous thrombosis and pulmonary embolism in the postpartum period of her fourth pregnancy in 1971. Family history was significant as both the patient’s mother and a daughter died of breast cancer, the latter with LC. There was ovarian cancer, brain cancer, lung cancer and non-Hodgkin lymphoma in her extended family. Physical examination was remarkable only for mild edema of the left arm.\nA computed tomography (CT) scan of the neck and chest showed thrombosed right internal jugular and brachiocephalic veins. A focal area of consolidation was located in the medial segment of the right middle lobe. There were segmental pulmonary emboli and a mild right pleural effusion. Pleural fluid analysis and biopsy of the right scapular lesion showed malignant cells consistent with adenocarcinoma. An EGFR mutation, exon 19 deletion, was detected in the pleural fluid sample. There was no evidence of a T790M or other mutation.\nA CT-positron emission tomography (PET) scan () showed a 2.3-cm mass in the anterior right lung showing increased metabolic activity with a standardized uptake value (SUV) of 3.8. Multiple metabolically active lymph nodes were located in the mediastinum, left supraclavicular region and retroperitoneum. Multiple bone metastases were detected in the ribs, sacrum and the right scapula. A right scapular lesion had an SUV of 10.6. Magnetic resonance imaging (MRI) of the brain did not show any evidence of parenchymal or leptomeningeal metastatic disease.\nThe patient started oral erlotinib, 150 mg daily, and subcutaneous denosumab, 120 mg monthly, in addition to daily subcutaneous low molecular weight heparin. The patient rapidly became asymptomatic and significant decrease in the lung mass, improvement of pleural effusion and resolution of pulmonary emboli were noted on chest CT 3 months after diagnosis.\nIn July 2013, 18 months after diagnosis, the patient began experiencing episodes of confusion, headaches, bizarre sensations in the head and difficulties with short-term memory. An MRI of the brain showed patchy areas of leptomeningeal enhancement (). A cerebrospinal fluid (CSF) study showed mild decrease in CSF glucose, normal protein level and the presence of multiple mononuclear cells consistent with adenocarcinoma. A CT-PET scan showed evidence of response to therapy with decrease in size of the lung mass to 6 mm without increased metabolic activity, resolution of pleural effusion and sclerosis of bone metastasis without increased metabolic activity.\nA diagnosis of LC was made and treatment options including intrathecal chemotherapy and cranial or craniospinal radiation were discussed. The patient refused these treatments mainly because of the death of her daughter who had LC related to an Ommaya reservoir for treatment of breast cancer several years ago. The patient was placed on pulsed erlotinib therapy with dose adjustments depending on toxicity. The patient derived her own optimal dosage of 600 to 750 mg daily for 3 days on and 2 days off resulting in a total dose of 2,550 mg per week. The patient noticed complete resolution of neurological symptoms in about a week and has remained symptom free at the time of this report more than 12 months after diagnosis of LC. The patient is symptomatic only in terms of grade 1 dermatologic toxicity and grade 1 diarrhea secondary to erlotinib. Results of spinal fluid analysis continue to show adenocarcinoma cells; however, multiple CSF samples failed to provide enough cells for molecular analysis.\nCT-PET scan in March 2014 did not show any evidence of increased metabolic activity. Craniospinal MRI in April 2014 () did not show any evidence of metastatic disease or leptomeningeal enhancement. |
A sixteen year-old male patient referred to our institute with post-auricular discharging sinus on his right side associated with swelling and redness around it, examination revealed that the patient had aural atresia dating since birth and microtia with acute mastoiditis that led to sinus formation on the right side, the left ear was completely normal. Two weeks before presentation, magnetic resonance imaging (MRI) was done in a private hospital due to the patient has complained of pain in right side of his head and face, it showed a soft tissue mass in the right infratemporal fossa (Figures ), surgical excision was advised but the parents of the patient have refused at that time. On presentation, Computed tomography (CT) was done for the patient as a routine radiologic evaluation for mastoiditis and it proved the diagnosis (Figures ). Pure tone audiometry showed conductive hearing loss with an air-bone gap of 60 dB on the right side and normal hearing of left ear. Surgical interference was carried out that had been started with post-auricular incision and excision of the unhealthy skin around the sinus, then cortical mastoidectomy was done, after identification of the lateral semicircular canal; the middle ear was entered via an atticoantrostomy approach, a soft tissue mass was seen filling the middle ear cavity, the mass was keratinous and eroding the hypotympanum and extended to outside the middle ear space. On retraction of the auricle antero-inferiorly, the large keratinous mass appeared in the infratemporal fossa that was anterior to the tempromandibular joint and inferior to the zygomatic arch which was eroded partially (Figures ). On manipulations of the middle ear mass, we noticed movement of the infratemporal fossa mass denoting that both were one mass. The mass was removed completely with no need to widen the exposure. After removal of the mass, no ossicles were found except the stapes, also there was erosion in the floor and anterior bony wall of the middle ear space with exposure of the fibrous capsule of the tempromandibular joint, the defect was about 2 mm in diameter. Reconstruction of the external auditory canal was done through removal of the bone lateral to the middle ear space with meatoplasty of the cartilaginous portion, then a temporalis fascia graft that has been harvested early was used to create the tympanic membrane; it was placed on the head of the stapes. A split thickness skin graft that has been harvested from the right thigh was used to line the newly created external auditory canal and was sutured to edges of newly created meatus. The wound had been closed in the usual fashion with post-auricular drain and packing the newly created external auditory canal. No intra-operative or post-operative complications were reported, the pack was removed in the 10th post-operative day and re-packing of the external canal was done weekly for six weeks. Histopathological examination of the mass confirmed the diagnosis of cholesteatoma, also to confirm the existence of squamous epithelial cells; immunohistochemical staining with involucrin was performed on paraffin-embedded sections of the mass, using the avidin-biotin-peroxidase complex immunoperoxidase technique [], involucrin positivity was seen. Follow-up of the patient for 2 years and 3 months showed a patent, infection free external auditory canal with an air-bone gap of 35db. One year after the operation; MRI was done and it showed no residual or recurrent cholesteatoma. |
An 85-year-old Hispanic man presented to our glaucoma service for a second opinion after 2 years of treatment by an outside glaucoma and retina specialist for presumed uncontrolled neovascular glaucoma of the left eye. Despite the use of maximum topical medical therapy and intravitreal anti-VEGF injections, the glaucoma remained uncontrolled.\nVision in the left eye was no light perception with an intraocular pressure of 54 mm Hg. There was 3+ conjunctival injection with scleral pigmentation noted at 9 o'clock, diffuse microcystic corneal edema, 1+ anterior chamber cell, and a fixed and dilated pupil with ectropion uveae between 2 and 4 o'clock and dark iris pigmentation from 2 to 9 o'clock. Pigment was seen on the anterior surface of the posterior chamber intraocular lens, and the vitreous was filled with loose, floating, pigmented cells (Fig. ). Gonioscopy of the left eye revealed dense pigment 360° in the angle, while the right eye gonioscopy examination was normal. The patient was referred to ocular oncology, where he underwent a B-scan and ultrasound biomicroscopy, which revealed some irregular iris thickening at 3 o'clock but no definite focal mass (Fig. ). Because of the marked degree of pigment deposition throughout the eye and a failure to identify a clear intraocular neoplasm, the patient was sent for full body dermatologic evaluation and whole-body PET scan to exclude metastatic cutaneous melanoma. No evidence of cutaneous melanoma was found, and the patient was diagnosed clinically with likely DIM. Enucleation was recommended.\nThe enucleated eye was filled with pigment in the anterior and posterior segments (Fig. ). Histopathological examination showed a diffuse iris ring-type melanoma involving the ciliary body, iris root, and approximately 75% of the anterior chamber angle structures (Fig. ). On transillumination at the time of fresh tumor retrieval, there was no well-defined mass. There were irregular shadows in the anterior portion of the ciliary body and iris root (Fig. ). The thin pigmented membrane over the intraocular lens was composed by pure melanoma cells and the trabecular meshwork was hyperpigmented (Fig. ). The tumor was composed mainly of spindle B-type cells extending in a sheath-like pattern over the iris with epithelioid-type cells seen at the tumor margins forming nests and invading the trabecular meshwork, Schlemm's canal, and focally into aqueous veins (Fig. ). The vitreous had tumor seeds of pigmented melanoma cells that lined the inner limiting membrane of the retina (Fig. ) and the inner limiting membrane of the optic nerve. The optic nerve was markedly cupped and atrophic with Schnabel's degeneration with focal invasion by a few single epithelioid melanoma cells into the nerve head and lamina cribrosa (Fig. ). Gene expression profiling (GEP) revealed a class 2, Prame-negative molecular signature. The patient continues to undergo routine metastatic surveillance and currently, at 17 months' follow-up, has no radiographic evidence of metastatic uveal melanoma. |
A 65-year-old man was admitted with diffuse abdominal pain and distention. The pain was constant and dull, and started 2 hours prior to admission with no obvious inducible factor. The patient’s past medical history was unremarkable except mild weight loss during the recent 5 months. Physical examination revealed a palpable abdominal mass in the left upper abdominal quadrant with tenderness, and the patient’s hemodynamic condition was unstable with systolic arterial blood pressure only 85 mmHg and heart rate 110 beats/minute. After initial fast fluid resuscitation (1,000 ml hetastarch 130/0.4 NaCl), the hemodynamic status of the patient became stable. Peripheral blood count revealed anemia (hemoglobin 10.0 g/dl) and thrombocytopenia (platelets 90 × 109/l), but it did not reach disseminated intravascular coagulopathy. Results of other laboratory examinations had not been reported to us before the operation due to the emergency. Abdominal ultrasonography showed an enlarged spleen filled with irregular nodules, multiple hepatic solid lesions and moderate accumulation of peritoneal fluid. Abdominal paracentesis was performed with blood fluid aspirated from the peritoneal cavity. These findings were further confirmed by an abdominal computed tomography scan (Figure\n), demonstrating a heterogeneous, low-density signal within the splenic parenchyma, with variable degrees of contrast enhancement as well as intra-abdominal hemorrhage originating most possibly from the spleen. Meanwhile, there were multiple low-density lesions with enhanced rim in the liver.\nLaparotomy revealed a huge spleen actively bleeding and an abnormal liver with several metastatic foci. We did observe a fissure of the capsule of the spleen with a size of 6 cm (length) × 2.5 cm (depth) in the lower pole of spleen. Two liters of blood had been collected from the peritoneal cavity. Splenectomy was performed. During the procedure, the patient received 4 units of concentrated red blood cells and 2 units of fresh frozen plasma.\nThe spleen was 1,650 g in weight, 19 cm × 16 cm × 11 cm in size, with nodular appearance and bleeding. The pathology diagnosis of the excised spleen was angiosarcoma originating from the spleen (Figure\n). Immunohistochemical staining was positive for vimentin (Figure\n), CD31 (Figure\n), CD34 (Figure\n), factor VIII (Figure\n), a1ACT (Figure\n) and SMA (Figure\n), and was negative for lysozyme (Figure\n). The Ki-67 proliferation index was less than 10% (Figure\n). |
A 46-year-old man from Papua New Guinea underwent colonoscopy for investigation for intermittent rectal bleeding. There were no associated weight loss or altered bowel habits; however, he had a history of hyperplastic polyps on a previous colonoscopy 7 years prior. There was also a family history of colorectal cancer, which affected his father and paternal aunt in their 60s and his maternal uncle in his 40s. He had a 30-pack-year smoking history, but aside from cluster headaches, had no other medical or surgical history and was not on any regular medications.\nHis colonoscopy revealed multiple polyps, 12 in total, throughout the colon. They were serrated or hyperplastic in appearance, ranged from 4 to 10 mm, were sessile and all were removed via cold snare. In the caecum, there was a 10 × 10 mm hard submucosal lesion. This differed in appearance to two smaller yellow submucosal lesions nearby, which were soft, and appeared to be lipomatous. Deep biopsies were taken of this hard submucosal lesion in the caecum. It was not amendable to endoscopic mucosal resection due to risk of perforation.\nHe was referred by his gastroenterologist to a colorectal surgeon for consideration of excisional biopsy. On computed tomography (CT) of abdomen and pelvis, there was no evidence of metastasis or advanced local spread of tumour. Deep biopsies of the caecal lesion were normal, and other polyps were hyperplastic. He was suspected of having a hyperplastic polyposis syndrome. Differentials for the caecal lesion, however, included gastrointestinal stromal tumour (GIST), lipoma or carcinoid tumour. Serum serotonin, chromogranin A as well as 24-h urinary 5-HIAA were all within normal limits. After discussion with the patient, a plan for laparoscopic right hemicolectomy was made.\nIntraoperatively, caecal polyps were noted, including firm nodules. There were no complications from his surgery, and his post-operative stay was unremarkable. He was discharged home after opening bowels and tolerating a light diet.\nHistopathology of the right hemicolon found five firm nodules in the caecum and an additional seven polyps in the ascending colon macroscopically. Microscopically, a total of eight lesions in the caecum and ascending colon were found to be GCTs, varying in size from 1 to 7 mm. They were submucosal with intact covering mucosa ( and ). Tumour cells had periodic acid-Schiff (PAS)-positive granular cytoplasm and small nucleoli (). Immunohistochemical staining was positive for S-100 () and inhibin. Twenty-four lymph nodes identified showed no abnormality. Spindling was seen in some tumours in this case, but overall, there are less than three worrisome features, and as such, these GCTs were considered atypical. Other polypoid lesions identified included two hyperplastic polyps and a submucosal lipoma.\nHe remained well at 3-week follow-up post-surgery. |
A 50-year-old woman was brought to the psychiatric outpatient department with history of psychomotor agitation and behavioural problems for the last one and a half months. The personal and family psychiatric history was unremarkable. Initially, the history was taken from the patient's son, who reported that in the last one and a half months the patient had shown behavioural changes, wandering behaviour, and verbal and physical aggression. There was a recent decline in the personal and social functioning. At first psychiatric evaluation, the patient was conscious but confused, not oriented in time, partly oriented towards the place, and seemed uncooperative. Her speech was spontaneous but irrelevant and nongoal directed. Due to her psychomotor agitation, higher mental functions could not be assessed. The patient was admitted, and a provisional diagnosis of a manic episode was kept based on the clinical findings of over-talkativeness, overfamiliarity, agitation, and aggression. Routine blood tests and ECG showed no pathological alterations. To manage the psychiatric symptoms, the patient was started on risperidone (titrated up to 3 mg/day) and diazepam (titrated up to 10 mg/day). Detailed evaluation in the ward revealed memory impairment for the last 3 months. During hospitalization, the patient's condition deteriorated. Perseveration and hallucinatory behavior became evident. A detailed neuropsychological assessment could not be done as the patient was uncooperative. According to the clinical picture, negative psychopathologic history, the rapid progression of memory impairment, and worsening of psychiatric symptoms, an organic substrate for the psychiatric symptoms was hypothesized. Because there was a rapid evolution of symptoms, it was fairly possible to exclude common forms of dementia, such as Alzheimer's disease and frontotemporal dementia. Prion disease was kept as one of the differential diagnoses because of the rapid evolution of symptoms.[] Neurological symptoms eventually became prominent, with the evolution of apraxia and focal neurological deficits (pyramidal/extrapyramidal signs and akinetic mutism). There was rigidity in all the extremities and hyper-reflexic deep tendon reflexes. MRI brain showed bilaterally symmetrical T2-weighted and fluid attenuated inversion recovery (FLAIR) hyperintensity along the cortex, bilateral globus pallidi, and posteromedial thalami, as well as diffusion restriction and hockey stick appearance of thalami [Figures and ].\nEEG revealed bilateral periodic sharp and short interval diffuse discharges with generalized delta slowing. Cerebrospinal fluid (CSF) analysis was not done as the diagnosis was achieved with the help of neuroimaging and EEG findings. There was worsening of psychiatric symptoms despite treatment and the patient was lost to follow-up. |
A 65-year-old man was referred by the otolaryngology department to our outpatient clinic due to sudden swelling and mild pain around the right eye. On examination, the patient exhibited what appeared to be severe edema encompassing the upper and lower lids of the right eye (). Crepitus was clearly audible on palpation of the eyelids. An attempt to open the lids was unsuccessful. Visual acuity and intraocular pressure could not be measured due to extreme lid swelling. The patient reported that he had undergone transnasal endoscopic nasal polypectomy through the right nostril 2 days earlier. He said he had been instructed not to cough or strain after the endoscopic nasal surgery and the sudden swelling occurred immediately after severe coughing and straining. We suspected that the sinus wall was weakened due to his endoscopic surgery and the increased pressure caused by straining had forced air in the nose into the periorbital area. B-mode ultrasonography showed trapped air in the periorbital area ().\nConsidering the patient’s anxiety, the severity of periorbital emphysema, inability to conduct a full ophthalmologic examination, and the risk of complications such as compressive optic neuropathy, the patient was re-evaluated for a surgical intervention. After consultation, it was decided to evacuate the air using a 21 gauge needle inserted in the subcutaneous tissue of the upper and lower lids. In sterile conditions, the eye area was cleaned with 10% povidone-iodine. A 21-gauge needle was passed through the skin and subcutaneous tissue of the upper and lower lids parallel to the tarsus about 1.5 cm from the lid margin. Evacuation of subcutaneous air was evident from a significant reduction in lid swelling during the procedure (). The patient’s vital signs were stable and the procedure was concluded. He was discharged with systemic antibiotics (cefuroxime axetil 500 mg twice daily) and moxifloxacin drops four times daily.\nOn follow-up examination the next day, the periorbital emphysema was substantially reduced and the globe could be examined (, ). He had full visual acuity in both eyes; intraocular pressure was 17 mmHg in the right eye and 16 mmHg in the left eye. Dilated fundus examination was normal. No restriction in eye movements was observed. Follow-up examinations at 1 week and 1 month revealed no pathological findings. |
A 42-year-old male patient reported to us with a chief complaint of pain, swelling, and pus discharge from left upper jaw. History revealed trauma due to blunt injury over the left cheek bone 3 weeks back. Medical history was nonsignificant. On clinical examination, there was a preseptal orbital cellulitis of lower eyelid []. Intraorally, there was a draining abscess in the left maxillary buccal vestibule in the canine–premolar region. Clinically, there was no displaced fracture of the maxilla or zygomatic bone. Noncontrast computerized tomographic scans showed an undisplaced fracture of the left orbital floor with intraorbital air foci, hyperdensity (HU 35–40) in the maxillary, ethmoid and sphenoid sinuses, and soft tissue swelling in the left infraorbital and maxillary region []. The patient was admitted and routine blood investigations and pus culture and sensitivity tests were ordered. The tests reported a random blood glucose level of 498 mg/dl, leucocytosis with neutrophilia, and increased ketone bodies in the urine. Pus culture showed a growth of Klebsiella pneumonia stains. After 10 days, on intraoral examination, a necrotic and mobile bony segment of the left maxilla in the canine to molar segment was noted along with a palatal abscess in the first molar region and mobile teeth. Surgical debridement of the maxillary sinus region and lateral wall of the nose along with the extraction of involved teeth were done and multiple pieces of blackish escharotic bone were recovered [Figures and ]. Histopathology of the tissues using hematoxylin–eosin stain confirmed mucormycosis of the maxilla []. Intravenous injections of lyophilized amphotericin B (AmB) 50 mg were given for 6 weeks with monitoring of blood urea and creatinine levels as the drug is nephrotoxic. After 3 months of healing and complete epithelization, an interim obturator with a nasal and antral bulb was fabricated to prevent oronasal regurgitation. The patient has been disease free for the follow-up period of 1 year []. |
A 47-year-old female presented with a lump in the left breast in September 2009. She had no history of pain or nipple discharge. There was no significant past medical history.\nSonography demonstrated an irregular, spiculated, 1.7 cm hypoechoic nodule in the upper inner quadrant of the left breast, accompanied by skin thickening and edematous change (). A core needle biopsy identified an atypical angiomatous lesion, and the diagnosis of angiosarcoma was confirmed by positive results for factor VIII-related antigen and CD34. Because the patient had no history of previous radiation therapy or surgery, we classified the tumor as a primary angiosarcoma.\nOn dynamic magnetic resonance imaging (MRI), the mass exhibited early rapid enhancement with plateau. The MRI also detected lymph node enlargement in the left axilla ().\nA positron emission tomography (PET)/computed tomography (CT) scan confirmed an area increased fluorodeoxyglucose uptake in the upper inner quadrant of the affected breast with faint uptake by the lymph nodes in the left axilla, but no other metastatic lesions were found.\nThe patient underwent a modified radical mastectomy in October 2009. Grossly, there is a 3.0 × 1.5 × 1.4 cm sized ill defined dark brown hemorrhagic spongy mass in the upper outer quadrant of breast. Microscopically, the mass composed of epithelioid areas, spindle areas and necrosis. The epithelioid areas are made up of large rounded cells of high nuclear grade which are arranged in sheets or rudimentary vascular channels (). The spindle areas show vasoformative growth with complex anastomosing channels (). These neoplastic channels are irregular in shape, freely intercommunicating with one another in a sinusoidal fashion. The mass shows high mitotic count (14/10 HPF), high nuclear grade and necrosis, the diagnosis is angiosarcoma, grade 3 in World Health Organization classification. There is no lymph node metastasis. On immunohistochemical staining, the tumor was negative for estrogen receptor, progesterone receptor and HER2. The tumor was positive for VEGFR-2 and CD-34 ().\nThe patient was treated with adjuvant chemotherapy of doxorubicin (60 mg/m2) and ifosfamide (5,000 mg/m2) every 3 weeks for six cycles from November 2009 until February 2010.\nFollowing this, the patient underwent radiation therapy on the left chest wall with 50 Gy for 5.5 weeks. CT, and bone scan were normal. A biopsy was done in the left axilla. Histological diagnosis was lymphangioendothelioma, which was benign. We decided on follow-up 6 months later due to the possibility of local recurrence.\nIn January 2011, the patient returned complaining of dyspnea that had started 1 week prior. A chest X-ray showed a large pleural effusion in the left side (). Chest CT verified a massive pleural effusion with complete atelectasis in the left lung. A chest tube was placed to drain the effusion and 3,600 mL of dark, sanguineous fluid was emptied. Pleural biopsy and fluid cytology were done. On histological examination, there was no definite malignant lesion but a marked proliferation of vascular endothelial cells, which was thought to be a reactive change.\nDuring this admission, the patient's initial platelet count was normal, but she developed a gradual onset of thrombocytopenia that persisted until discharge (2 months later) despite platelet transfusions. Effusion decreased gradually and chest tube was removed, but the patient's condition was not yet improved (). She left the hospital arbitrarily.\nThe patient presented again with dyspnea and hemoptysis in April 2011. Chest X-ray and CT showed pulmonary hemorrhage in the left side and pleural effusion in the right side (). A chest tube was placed to drain the effusion and sanguineous output was noted. The patient was anemic and again remained thrombocytopenic in spite of transfusion. The skin lesion had been observed in left chest wall (). Biopsy was done and histological examination established the diagnosis of recurrent grade 3 angiosarcoma. Persistent hemoptysis and pleural effusion were not improved.\nAfter diagnosis of recurrent angiosarcoma, we tried combination treatment with bevacizumab and paclitaxel with informed consent. After the first treatment, the patient's respiratory symptoms improved, but after five days, her respiratory status again deteriorated. The patient died of respiratory failure secondary to pulmonary hemorrhage. |
A 28-year-old woman presented at an emergency department on a weekend night at a rural hospital following dog bites sustained to her lips. There were two bites, one that lacerated the lower lip that was still connected to the face by a small, lateral pedicle and one that amputated more than half of the upper lip. The defect crossed the philtrum medially and extended laterally to the nasolabial fold (). No other injuries were afflicted to the patient. The patient was referred to the Sahlgrenska University Hospital and arrived at the Department of Plastic Surgery 90 min after the trauma. The amputate, measuring 4.5 × 3 cm, had been kept in a hypothermic state (). Emergency replantation surgery was initiated. Peroperatively, a 1 mm superior labial artery was identified laterally on the amputate and was microsurgically anastomosed to the proximal stump of the superior labial artery using 10–0 S and T sutures. The ischemia time was approximately 4 h and 45 min. Even after reperfusion, no veins suitable for anastomosis could be identified. One nerve was identified, but it was to damage to repair. The lip was sutured in three layers: the mucosa, the orbicularis oris muscle, and finally, the skin, which was loosely sutured to allow bleeding and lessen the risk of hematoma. The lip rapidly turned blue as a sign of venous congestion. A leech (Hirudo medicinalis) was applied to the lip in order to achieve venous drainage and it immediately regained adequate skin color and became less swollen (). The lower lip was also sutured in three layers and was adequately vascularized through the right inferior labial artery during the whole procedure. During first night, leeches were applied every 45 min, and thereafter, the interval was extended, guided by the color of the lip; day 2 every 90 min, day 3–5 every 2 h, day 6–10 every 3 h, day 11–12 every 4 h. On the 13th day, the lip did not need leech therapy and the patient could be discharged on day 14. Anticoagulant therapy was provided using subcutaneous injection of dalteparin 5000 U/day for 14 days. The constant bleeding from the lip during leech therapy necessitated several blood transfusions to a total of seven units.\nBroad spectrum antibiotics (meropenem 0.5 g three times daily) was used for 10 days in order to prevent infections from bacteria associated with dog bites and bacteria of the leeches normal flora (e.g. Aeromonas hydrophila).[]\nNutrition was achieved by clinifeeding tube during the first three days but was converted to parenteral nutrition due to severe nausea and vomiting. On the 8th day, the patient could ingest sufficient amounts per oral route.\nThe patient received a tracheostomy preoperatively in order to secure the airway due to face swelling and the continuous bleeding from the lip which to some extent went into the mouth and throat.\nAfter discharge, the follow-up was uneventful. At one-year follow-up, the patient had good oral continence, good lip motion, no problems with articulation, but the sensitivity for heat/cold and pin prick was still poor (). The lip still has a slight edema, which is slowly subsiding. |
A 32 year old Asian lady underwent excision surgery of a left frontal meningioma under Neurosurgery and was started on phenytoin at a dose of dose of 300 mg / day along with dexamethasone in the perioperative period. Dexamethasone was discontinued after 2 days and she was discharged from hospital with normal blood parameters including liver function tests 6 days after the surgery. While at home she developed fever and skin rash 12 days from the date of surgery. Phenytoin was promptly discontinued and it was replaced with Leviteracetam by the neurosurgeon but this was also discontinued 2 days later because of the rash becoming more widespread. She was then referred to the Neurology department 3 weeks after the onset of rash for further management.\nOn examination she had extensive maculopapular rash all over the body involving the face, trunk, upper and lower limbs. She had cervical and posterior auricular lymphadenopathy, jaundice, and splenomegaly. She denied any history of exposure to any other drug, recreational drug abuse, alcohol or blood transfusion.\nLab investigation including complete blood count showed absolute eosinophilia 14.9% (Normal-0 to 7%), marked impairment of liver function []. Investigations for other causes of hepatic disease including Serum copper, serum ceruloplasmin, screening for collagen disease, viral markers for hepatitis were all negative (HAV, HBV, HCV).\nEpstein Barr virus IgM and IgG antibodies were both positive. Cytomegalovirus Ab-IgG positive and IgM- negative\nSerum ammonia, serum amylase, D Dimers were negative.\nUrine R/M –no cells, Protein -1. Urine, throat, blood cultures were negative.\nDrug hypersensitivity due to phenytoin was diagnosed and she was started on oral prednisolone 1 mg /Kg. However there was no improvement and the rashes were becoming more florid and liver function tests deteriorated further. Investigations 7 days after admission showed persistent increase of liver enzymes. Despite stopping phenytoin and treatment with prednisolone (1 mg /Kg) the rash was becoming more diffuse and serial LFT revealed increasing enzyme levels. Dermatology consultation confirmed the case as DRESS syndrome (Drug rash, Eosinophilia, Systemic symptoms) syndrome based on fever (39 to 40°c) drug rash, facial edema, eosinophilia, systemic manifestations of lymphadenopathy, splenomegaly, liver and renal impairment.\nIVIG (0.4 Gm/ Kg) was added to prednisolone as there was no improvement even after 7 days of steroid treatment. After 5 days of I.V. Immunoglobulin (0.4 Gram/Kg) clinical improvement was evident, the rash subsided and a rapid decline of liver enzymes was observed [ and ]. Blood parameters returned to normal 3 weeks after starting IVIG and 6 weeks after the onset of the rash. In the meantime she was started on Topiramate which she tolerated well and was discharged from the hospital with tapering dose of prednisolone. On follow up till 2 months she was asymptomatic and well. |
A 49-year-old female was brought to the ED for severe abdominal pain that she has experienced over the past 2 days. She described the pain in the epigastric region, which was constant and was radiating to the left the shoulder and was associated with multiple episodes of vomiting without blood. She also revealed that she had fever, cough and shortness of breath for past 1 week for which she self-isolated thinking that she had a coronavirus infection. However, test for coronavirus infection was not performed. Regarding her past medical history, she was diabetic and hypertensive for which she was on medication but had a poor compliance. Upon surgical records, an emergency open appendectomy was done 10 years back. She was non-alcoholic and non-moker. At presentation, she appeared pale, diaphoretic and was in respiratory distress. Physical examination revealed a temperature of 36.3°C, with a heart rate of 130 beats/min, a blood pressure of 130/80 mm of Hg, a respiratory rate of 24 breath/minute and body mass index 29.2 kg/m2. There was no sign of jaundice. On abdominal examination, abdomen was overtly distended with generalized tenderness with no signs of peritonitis. On rectal examination, tone was normal with stool stained in the finger without palpable mass. The remainder of her systemic examination findings was unremarkable. Laboratory findings consistent with acute severe pancreatitis include an elevated amylase, triglyceride and bilirubin. In addition, an increased in white blood cell count with neutrophila, low hemoglobin level and low albumin were noted (). An arterial blood gas analysis was normal except for mildly raised a lactate level of 2.0.\nRegarding her management at emergency, an aggressive fluid resuscitation with compound sodium lactate 3 litres over half an hour was started. A foley’s catheterization followed by nasogastric tube (NGT) was inserted for gastric decompression. An immediate collection of 900 ml of non-bilious clear fluid was drained. The Rapid Diagnostic Test for COVID 19 infection was sent which showed positive. Then, she was transferred to the COVID-Intensive Care Unit (ICU) for ongoing fluid resuscitation and further management. The PCR test for COVID-19 was sent from ED which showed positive later that day. A broad-spectrum intravenous antibiotics, intravenous analgesics was started. A contrast-enhanced computed tomography (CECT) scan was performed to find out the pathology. Abdominal contrast-enhanced CT showed small indistinct area of hypo-density and hypo-enhancement in the uncinate process with extensive peri-pancreatic inflammation around the pancreas suggestive of acute necrotizing pancreatitis (ANP) ().\nThus, the provisional diagnosis of severe Acute Necrotizing Pancreatitis (ANP) was made. Despite all supportive care, her mental status worsened and her clinical status continued to decline. Laboratory parameters showed progressive thrombocytopenia, anemia, hypotension, acute kidney injury (AKI) and metabolic acidosis with respiratory compensation. She had progressive abdominal distension with decreased bowel sounds. The intra-abdominal pressure measured from intra-vesical pressure showed increased (23 mmHg) which leads to the diagnosis of Abdominal Compartment Syndrome (ACS). Thus, a gastrointestinal prokinetic agent and neuromuscular blockade were prescribed. An NGT was inserted to drain the intraluminal content. However, she did not show any improvement with medical treatment; the decompressive laparotomy with a midline incision was planned the following day. Intra-operatively, more than 2 l of ascitic fluid which was brownish to red color was evacuated from the abdominal cavity. There was saponification throughout the mesentery of the transverse colon, and one of the larger areas of saponification was actively draining brownish black pancreatic fluid from the lesser sac. Other findings included severe retroperitoneal edema and an edematous pancreas, with blackish discoloration at the head of pancreas leading the diagnosis of acute hemorrhagic necrotizing pancreatitis ().\nIntra-operatively, an extended adhesiolysis was performed with opening of gastrocolic ligament and followed by necrotic debridement, and drainage placement. Two radiopaque 28 F abdominal drains were placed in the lesser sac and at the base of the mesentery of the transverse colon and one at the pelvis. The abdomen was left open and was skin opposed with retention sutures (). The patient underwent post-operative resuscitation in the ICU.\nDuring the post-operative period, the patient became progressively difficult to ventilate with very poor pulmonary compliance. She was transfused four pints of whole blood during her stay in ICU. Tidal volume was less than 250 ml on a driving pressure of 15 and a PEEP of 15. PaO2 remained in the 65 s despite FiO2 of 1.0. She was unresponsive to fluid challenge and required an infusion of 45 mcg/min of noradrenaline and 2 mcg/min of vasopressin in order to maintain an adequate mean arterial pressure. Gradually patient developed AKI, being oliguric (<10 ml/h) with a potassium of 6.5 mmol/l. Her blood parameters showed sepsis with WBC count of 3200 × 109. Despite correction of ACS and active intensive therapy, the patient’s condition did not improve and she died 2 days after laparotomy as a result of multi-organ failure. |
A 66-year-old Japanese man who had no past medical or medication history complained of gross hematuria and visited a nearby hospital in October 2013. He had no habit of drinking alcohol or smoking tobacco. He was diagnosed as having a right renal tumor and underwent right nephrectomy laparoscopically. The pathological diagnosis was right renal cell carcinoma (RCC), clear cell carcinoma, pT1bN0M0, v1 (Fig. ). One and half years later, lymph node swelling was detected at hepatic portal region and he underwent lymphadenectomy. The pathological diagnosis was a metastasis from RCC. Two years after diagnosis, he was suspected of lung metastases and started treatment with interferon α. Three years later, the multiple lung metastases grew larger and were determined as progression despite interferon α therapy. At this point, he was referred to our hospital in October 2016. There were no abnormalities on physical examination and his vital signs were normal. He started treatment with sunitinib 50 mg/day on a schedule of 4 weeks on treatment and 2 weeks off; however, adverse events including grade 3 thrombocytopenia (platelet count, 49,000/μL), gum swelling, and hoarseness became intolerable 2 weeks after starting sunitinib. Four weeks after cessation of sunitinib 50 mg/day, he was started on a dose of sunitinib 25 mg/day on a schedule of 2 weeks on and 1 week off. Computed tomography (CT) findings in January 2017 revealed that his lung metastases had shrunk; however, he continued to experience the same adverse events. Therefore, the dose of sunitinib was further reduced to 12.5 mg/day on a schedule of 2 weeks on and 1 week off. CT findings in May 2017 revealed new metastases in the pleura, diaphragm, and the right paracolic gutter (Fig. a, b). As a result, the treatment was changed from sunitinib to axitinib and he started treatment with axitinib at 10 mg/day; however, adverse events including gum swelling, dysphonia, hypertension, diarrhea, and thrombocytopenia became intolerable (Fig. ). Two weeks after cessation of the drug, the dose of axitinib was gradually reduced from 6 mg/day to 4 mg/day. CT findings in September 2017 revealed the metastases had diminished in size and lung metastases were maintained at a diminished size (Fig. c, d); however, the adverse events could not be controlled and he discontinued axitinib treatment. His adverse events improved after discontinuation of axitinib; however, CT findings in December 2017 revealed the size of metastases had increased again (Fig. e, f). Consequently, he was started on fourth-line therapy with nivolumab (3 mg/kg every 2 weeks) and did not experience any adverse events. However, after he had received eight cycles of nivolumab, his metastatic lesions had grown, peritoneal dissemination appeared in his pelvic region, and pleural effusion appeared (Fig. g, h), so nivolumab was discontinued. After giving a detailed explanation of treatment options to our patient, he decided to rechallenge with axitinib 4 mg/day. However, adverse events including gum swelling and dysphonia became intolerable. After that, the dose of axitinib was reduced to 2 mg/day, and he experienced relief of adverse symptoms except for hoarseness. CT findings in August 2018 revealed metastases in lungs, pleura, diaphragm, and the right paracolic gutter had diminished in size (Fig. i, j). He has been continuously receiving a low dose of axitinib at 2 mg/day for 10 months with metastases maintained at reduced size. |
A 56-year-old woman who suffered from headache and vomiting was admitted as an emergent case. The sudden onset of severe pain in the occipital area started 2 weeks before admission and got worse over 2 days. On arriving, the patient was alert and oriented. She had a history of uncontrolled hypertension, and her presenting blood pressure was 170/90 mm Hg. Intravenous hydralazine to lower blood pressure was administered. When the patient was referred for neurological opinion, she denied photophobia, sensory change or motor weakness. A further evaluation was notable for a normal neurologic examination including cranial nerves and no neck stiffness. No abnormality of ocular motility was observed. The laboratory results for coagulation profiles and tumor markers were within normal limits.\nCT showed an 11 mm-sized, noncalcified, and faintly-enhancing mass in the pineal region without ventricular enlargement (). No abnormal finding in the cerebral vasculatures was detected on the reconstructed three-dimensional CT angiography. The pineal lesion appeared at MR images as ovoid, thin-walled, and well-defined cyst. The cyst was hypointense on T1-weighted and hyperintense on T2-weighted sequence. There was a blood-fluid interface within the cyst on gradient echo sequence (). No tumor or vascular malformation in the pineal region was visualized on enhanced MR studies. An imaging diagnosis of intracystic hemorrhage of PC was made by staff neuroradiologists without the need for clinical information about patient's history and findings.\nSince it was unlikely that the cyst would compress the cerebral aqueduct, the patient was scheduled for managing the PC apoplexy conservatively. Her symptoms had completely resolved with use of pain medication and antiemetic drugs. The patient was discharged home at the ninth day of hospitalization with normalized blood pressure. The close observation and regular follow-up with MRI were conducted. At the 6-month follow-up, MR scans revealed the stabilized cyst with resolution of blood products. No recurrent mass or other complication was detected on MR series taken 15 years later after the apoplexy (). The patient was still clinically asymptomatic at the last outpatient visit.\nThe IRB exempted informed consent due to its retrospective nature and minimal risk for harm to the patient, and this report was conducted according to the guidelines of the Declaration of Helsinki for biomedical research. |
A 6 years old boy, ASA 1 (American Society of Anesthesiologists physical status 1) was referred to a maxillofacial surgeon in November 2012, for the evaluation of a solitary growth of the posterior right maxilla.\nOral examination revealed a reddish, well-circumscribed and firm nodule that measured in its largest diameter about 2 cm. The mother of the patient noticed the lesion about a month before the initial examination. Main complaint upon presentation was bleeding from the area of the lesion while brushing the teeth.\nIn order to establish better differential diagnosis and to undercover whether the lesion originates in the bone (meaning a central epicenter), causing bone destruction or whether it is a lesion of the soft tissue alone, the patient was sent to perform imaging. A Panoramic X-ray (Fig. ) and cone beam computed tomography (CBCT) failed to show any pathological change of the right maxilla.\nOwing to the obscure nature of the lesion and the young age of the patient, an incisional biopsy was performed immediately under local anesthesia and sent urgently to the pathological department (one of the considered and alarming differential diagnosis being Lymphoma and Leukemia). After receiving the pathological report based on the incisional biopsy (diagnosis of MPC), the patient underwent excisional biopsy of the lesion under general anesthesia, and a diagnosis of MPC was once again established based on histological and immunohistochemical analysis (Figs. , , ). The incisional and excisional biopsy were performed by the Principal Investigator (Prof. Abu El-Naaj, an Oral and Maxillofacial Surgeon). Types of suture that were used (incisional and excisional biopsy, respectively) were Silk 3*0 and Vicryl 4*0, single interrupted. The excisional biopsy procedure was performed under general anesthesia in order to relieve the patient’s anxiety and also in order to ensure the excision of the lesion and closure of the site are performed under the best medical conditions and maximal control. The operation itself lasted about an hour. Postoperatively, the patient received antibiotics (amoxicillin) and analgesics.\nNo further treatment was necessary as the lesion was fully excised.\nAfter the final diagnosis was established and the postoperative healing was uneventful (Fig. a), the patient remained under close follow up (Fig. b). During the first year the patient was examined every 3 months and later on, once a year. During each follow up session, clinical examination was performed. Once a year, a panoramic Xray was performed.\nTo present date, 8 years postoperatively, no recurrence was detected. |
A 45-year-old male, right-dominant, Caucasian patient had a right primary anterior shoulder dislocation after a car accident. Due to burns at the left flank of about 9% of the body surface area caused by the car accident, the patient underwent several skin reconstructions. He had no previous shoulder pathology or other illnesses. The shoulder was reduced in an outside trauma clinic followed by conservative treatment. At 4 months after trauma, the patient presented initially in our institution with posttraumatic stiffness (passive range of motion (ROM): forward flexion 40°, abduction 30°, external rotation 0°), decreased sensation of the fingers 4 and 5, impaired strength with atrophic interosseous muscles, and palpatory distinct, painful ossifications in the axilla, and conjoint tendons. Electroneurography showed evidence for isolated proximal functional disorder of the ulnar nerve with all other nerves intact. We discussed the therapeutic options and first decided to try and exhaust conservative treatment. 9 months after the trauma, the patient presented with deteriorating symptoms and with constantly palpable and painful HO in the axilla and around the conjoint tendons. Plane radiographs and magnet resonance imaging (MRI) showed unaltered HO (). The shoulder stiffness had worsened with a fixed internal rotation position of the arm (passive ROM: forward flexion 35°, abduction 35°, external rotation -15°). The impaired sensation of the fingers 4 and 5 as well as the reduced strength of the interosseous muscles remained unaltered. Consequently, a surgical intervention was indicated. Preoperative, radiation was applied for recurrence prophylaxis. Computer tomography (CT) diagnostics revealed pronounced HO throughout the axillary recess, partially encasing neurovascular structures, conjoint tendons, and the subscapularis muscle (). Surgical procedure using a deltopectoral and transaxillary approach included resection of HO, neurolysis of the brachial plexus including the axillary artery, tenolysis of the conjoint tendons, and tenotomy of the long head of the biceps. The tendons of the pectoralis major, pectoralis minor, and latissimus dorsi were temporarily detached for complete removal of the HO and neurolysis. The whole ventral side of the subscapularis muscle appeared ossificated, which could explain the fixed internal rotation position of the shoulder. After HO removal (), flexion and abduction improved intraoperatively. In addition, external rotation improved from -15° to 40°. Postoperatively, the patient was treated with indomethacin 150 mg for 4 weeks for recurrence prophylaxis and painkillers. Physiotherapy was initiated on the first postoperative day with no restriction in ROM but limited strength for 6 weeks. 9 months after surgery, the patient reported an improvement of both pain (preoperative VAS 7 to postoperative VAS 0) and function (preoperative Constant score 18 points to postoperative Constant score 72 points). Also, neuropathy of the ulnar nerve was regressive with only slight numbness of the fourth finger. |
A 56-year-old male patient with a height of 166.6 cm and a weight of 59.9 kg was referred from the gastroenterology department for right chest pain caused by hepatocellular carcinoma and right chest fifth rib metastasis. The degree of pain was NRS 6/10, and it was characterized as a spontaneous splitting pain. Transarterial chemoembolization (TACE) chemotherapy had been performed on the patient for liver cancer for one year. Five months before visiting the cancer pain unit at our hospital, metastasis to the right chest fifth rib had been discovered, and the patient had received radiation therapy. In the subsequent MRI and bone scan of the thoracic vertebrae, right fifth rib metastasis with extraosseous mass formation had been observed. The patient had the underlying diseases of hepatitis B and liver cirrhosis and, at the time of referral, had been using Ultracet Tab™ (acetaminophen 325 mg tramadol hydrochloride 37.5 mg, Janssen Korea LTD, Korea) 2 tab tid for pain control. However, the right chest pain had gradually worsened and been causing sleep disturbance. Hence, a thoracic epidural injection had been performed during hospitalization, but 3 days after the procedure, the pain had worsened, with the NRS score increasing again to 6/10 from 2/10; thus scrambler therapy (MC5-A Calmare®) was planned for the patient. The patient complained of chest pain in the right fifth rib area, so the scrambler electrodes were attached to 2 normal sensory areas to the left and right of the right fifth rib pain area for treatment. As the 10 sessions of treatment progressed, it was clear that the pain area was gradually contracting, and the location of the electrodes was adjusted accordingly for treatment. After 2 sessions of scrambler therapy, the NRS score decreased to 2/10, and the treatment effect continued until the next morning. The administration of Ultracet Tab™ was stopped accordingly. After 10 sessions of scrambler therapy, the NRS score has been maintained at 2/10, and the pain area maintained at about 80% reduced state, for 2 months. |
A healthy 16-year-old female, with no known comorbidities, presented to the Outpatient Department with severe perineal pain. The mechanism of injury was a fall from height in which the patient sustained blunt trauma to the perineum. The patient reported immediate onset of severe pain associated with a progressive swelling of the vulva and the presence of blood. This was associated with the gradual onset of headache and symptoms of presyncope, including generalized weakness, nausea, and lightheadedness. The patient denied any history of recent coitus or vaginal instrumentation. The patient was privately counseled on sexual assault, which she also denied. The patient was questioned regarding signs or symptoms suggestive of bleeding diathesis. The past medical history was noncontributory and the patient reported no known allergies or current medications. On physical examination the patient had an ashen appearance with pale sclera and mild respiratory distress. The heart rate was 115 bpm with a manual blood pressure of 100/60 mmHg and an oral temperature of 37.1°C. Her urine was clear with no evidence of macroscopic hematuria.\nExamination of the perineum revealed a 10 × 8 cm swelling of the left vulva with right deviation of the right labia and clotted blood over the left labia minora (Fig. ). There was no active bleeding per vagina. Basic laboratory investigations revealed a Hb of 10.2 g/dL, with a normal WBC and platelet count. The INR/PT was within normal parameters. Initial resuscitation consisted of 1500 mL IV bolus of crystalloid solution. Despite the infusion, the patient remained hypotensive and tachycardic. Given the degree of swelling and accompanying hemodynamic instability, the patient was consented for surgical intervention. Intraoperative, a large hematoma was evacuated through a medial incision of the left labia majora. A single, actively bleeding vessel was identified and suture ligated. The cavity was irrigated and inspected for hemostasis, which was subsequently closed with 1–0 chromic catgut (Fig. ). Prior to the close of the case, a Foley catheter was inserted in the bladder. The patient had an uneventful postoperative course with prompt removal of urinary catheter and discharge on postoperative day three. The patient was seen in follow-up at 4 weeks and the vulva appeared symmetric with the swelling completely resolved. The patient had no complaints. |
A 17-year-old male patient presents to the emergency department with a two-hour history of sudden onset of left-sided chest pain, exacerbated by breathing, with no radiation to other sites, and associated with shortness of breath. The patient reported no recent trauma and his past medical history was unremarkable. On admission, his vital signs revealed temperature of 34.5°C, blood pressure of 80/40 mmHg, heart rate of 125 beats/min, and respiratory rate of 24 breaths/min with an oxygen saturation of 98% on room air. At physical examination, the patient was dyspneic and presented important painful distress. Cardiopulmonary examination was significant for muffled heart sounds on mitral focus and abolished breathing sounds in the left hemithorax. Electrocardiography (ECG) showed normal sinus rhythm, and initial laboratory results were notable only for a hemoglobin level of 8 mg/dl. Chest X-ray (CXR) evidenced a left-sided hydropneumothorax ().\nA computed tomography (CT) scan of the chest revealed left lung collapse with significant deviation of the mediastinum to the right side (). A thoracentesis confirmed the presence of blood in the pleural cavity. A chest tube was then inserted in the 5th intercostal space with drainage of air and 2000 ml of blood. Two units of packed red blood cells were administered and the patient's vital signs stabilized as well as his symptoms relieved. The patient was referred to a tertiary cardiopulmonary service for definitive treatment evaluation.\nWithin the next hours till presentation to our service, additional 800 ml of blood were drained. New CXR revealed adequate lung reexpansion after tube insertion (), and the thoracic surgery team decided not to proceed to emergency thoracotomy. The patient was admitted, and over the next 24 hours, another 200 ml of blood flowed out from the chest tube. One more unit of packed red blood cells was transfused and signs of hemodynamic instability did not reoccur during hospitalization. Owing to the persistent bleeding, the medical team chose to indicate video-assisted thoracic surgery (VATS) to accomplish definitive hemostasis of the bleeding source.\nOn the third hospitalization day, VATS was performed, after induction of general anesthesia, with camera inserted through the already existing thoracostomy tube incision and confection of a portal in the 4th intercostal space. Exploration of the pleural cavity revealed a large amount of retained blood clot with pleural adhesion exhibiting signs of recent bleeding. A small bullae was found in the apex of the left upper lobe, near the site where a pleural adhesion ruptured (Figures and ). Complete removal of clots, irrigation of the pleural cavity, electrocauterization of the bleeding adhesion, bullectomy, and abrasive pleurodesis were effectuated. A new port was made in the 7th intercostal space for chest tube insertion and the previous one was closed.\nHistopathology showed reactive eosinophilic pleuritis consistent with previous pneumothorax, and no morphological evidence of malignancy or granulomas was present.\nThe postoperative course was uneventful, and the chest tube was removed on the 7th postoperative day. The patient remained stable and was discharged on the 10th postoperative day.\nSeven days after hospital discharge, the patient presents asymptomatic to our service for an outpatient follow-up appointment. However, CXR showed radiologic signs of pneumothorax on the right hemithorax, which was then confirmed by a CT scan of the chest (). The patient underwent new thoracoscopy with bullectomy and pleurodesis and was discharged 2 days after the procedure. The patient continued to be reevaluated and no more complications were observed at a 2-month follow-up. |
A 51-year-old woman underwent hysterectomy and pelvic lymph node dissection for uterine cancer when she was 48 years old, and lymphedema developed in the left leg soon after the operation. She had one episode of cellulitis. Despite wearing elastic stockings, lymphedema worsened, and she visited our institution at the age of 49 years. She was diagnosed with lymphedema based on lymphoscintigraphic finding. There was a development of collateral lymphatic vessels and dermal backflow in bilateral lower leg (See figure, Supplemental Digital Content 1, which displays lymphoscintigraphic findings. Collateral lymphatic vessels were observed in the bilateral lower legs. Lymphatic function in the left thigh was impaired, ). She had no allergies or other pertinent medical histories.\nLVA was performed at the age of 50 years. The postoperative course was uneventful, and lymphedema improved. However, lymphedema worsened again at 1 year postoperatively after taking a long flight, although she wears elastic stockings daily. A second LVA was planned (Fig. ).\nPreoperative indocyanine green (ICG) lymphography showed a linear pattern in the right leg. Dermal backflow was observed in the left thigh and lower leg. There was no linear pattern in the area (left lower leg) where the lymphatic thrombus was found afterward. Preoperative echography showed 2 hypoechoic circles measuring about 0.5 mm in diameter that did not collapse with pressure from the probe, although the veins collapsed with pressure (Fig. ). Compared with lymphatic vessels, veins usually collapse more easily under pressure, because the inner pressure of the lymphatic vessels is higher than that of the veins. In this case, the 2 circles did not collapse under pressure, and we surmised that the inner pressure prevented collapse. The hypoechoic circles extended proximal and distally and did not have flow with on color Doppler mode.\nDuring LVA, we identified 2 parallel white vessels beneath the superficial fascia. Two vessels were in close contact. We diagnosed these as lymphatic vessels because of the location and appearance and the fact that they ran in parallel, which is not usually observed with other vessels or nerves. When we incised the vessels, white material was extruded (Fig. ). A diagnosis of lymphatic thrombosis was made, and we concluded that the vessels did not collapse with pressure from the probe during echography because of thrombus. Intraoperative echography revealed the same findings, that is, a hypoplastic circle without collapse by probe pressure, 15 cm distal from the incision, which indicated that there was lymphatic thrombus in at least 15 cm. We ligated the lymphatic vessels, closed the wound at this site, and performed LVA at other sites (4 sites in the left and 1 site in the right leg). Though the postoperative course was uneventful, the patient’s lymphedema did not improve postoperatively. This may be partially because the patient gained weight after LVA, and there is another possibility that postoperative thrombus formed within the anastomosis site had harmful effect for lymphedema, although it is difficult to confirm.\nHistopathological examination showed a thickened smooth muscle layer (tunica media) in the lymphatic vessels (See figure, Supplemental Digital Content 2, which displays histopathological findings of the lymphatic vessel and the lymphatic thrombus. (a) The lymphatic vessel (a) and the lymphatic thrombus (b) (×20, H&E). Thickened smooth muscle layer (tunica media) in the lymphatic vessels is observed. (c) The lymphatic vessel (×100, D2-40). The endothelial cells of the vessel were negative for D2-40, ). Fibrous thickening of the tunica intima was observed, and the inner lumen was narrow. The inner layer of the vessel was negative for D2-40, which is a marker to stain the lymphatic endothelial cells. In the thrombus, hyperplasty of fibroblasts and organization were found (Fig. ). We did not observe hyperplasty of the lymphatic endothelial cells, which are positive for D2-40 within the thrombus. |
A 29-year-old woman without the history of oral contraceptives use presented with hoarseness, dysphagia, vomiting, and headache. Later physical examination in our neurology department also revealed paralysis of the right CNs IX to XII. Contrast-enhanced computed tomography (CT) of the head and neck revealed an interruption of flow in the right sigmoid and transverse sinuses, with no apparent tumor on contrast-enhanced CT scan. Coagulation profile including the prothrombin time, activated partial thromboplastin time was normal. She was diagnosed with Collet-Sicard syndrome caused by thrombosis of the sigmoid and transverse sinuses. She was treated with anticoagulant therapy, and the hoarseness and paralysis of the accessory nerve improved, but recanalization of the sigmoid sinus and transverse sinus was not identified. The hoarseness later worsened again, when she was 31 years old. In another hospital, contrast-enhanced CT scan revealed a tumor in the jugular foramen extending into the deep cervical region and medially displacing the internal carotid artery. The patient was referred to our hospital for further examination of this tumor. The tumor was investigated using contrast-enhanced CT and magnetic resonance imaging. It showed hypointensity on T1-weighted MRI and iso-to hyperintensity on T2-weighted MRI. The smooth tumor rim was enhanced on contrast-enhanced MRI. On a contrast-enhanced CT scan, the tumor displayed slightly heterogenous enhancement. She was diagnosed with jugular foramen schwannoma (Kaye's Type C) causing thrombosis of the sigmoid and transverse sinuses. No personal or family history of tuberous sclesosis was reported.\nAt the one-year follow-up, however, her tumor had grown rapidly, and we found that the tumor had started to surround the internal carotid artery (). This clinical course was thought to be atypical for a jugular foramen schwannoma. We decided to perform a tissue biopsy of the tumor in both the jugular foramen and the deep cervical area. In open biopsy, the jugular bulb and internal jugular vein were filled with the tumor. Histological sections showed a proliferation of epithelioid cells with enlarged nuclei and eosinophilic or clear cytoplasm, arranged in sheets or nested patterns with narrow blood vessels. Immunohistochemically, the tissue tumor cells were positive for HMB-45, Melan-A, and α-smooth muscle actin (α-SMA), but negative for desmin, AE1/AE3, and S-100 protein. Pathological analysis revealed a perivascular epithelioid cell tumor. (A tumor size of >5 cm with a mitotic index of 2 (counts per 10 high-power fields, HPF) and vascular invasion was identified. Based on these features, a diagnosis of PEComa with malignant potential was made. After discussing treatment options with otorhinolaryngologists, radiologists, and an oncologist, we decided to perform surgical treatment first. preoperatively, the superficial temporal artery-middle cerebral artery bypass was performed. We applied a postauricular transtemporal approach with anterior facial nerve rerouting to remove the tumor sacrificing the internal carotid artery involved by the tumor (). However, it resulted in incomplete tumor resection, because the cardiac arrest and bradycardia was found during the removal of the tumor around the inferior petrosal sinus and anterior condylar confluence. The CNs IX to XI were sacrificed because these structures were invaded and it was extremely difficult to separate these structures from the tumor. Post-operative dysphagia, horseness and accessory nerve palsy were found. Six months after surgery, radiotherapy was added to treat the residual tumor volume. After one year follow up, there is no evidence of the regrowth of the residual tumor and metastasis.\nWritten informed consent for the current study was obtained before obtaining the patient's data. |
One month before visiting our pain clinic, a 68-year-old female patient with vesicles and pain on the left side around the navel was diagnosed with herpes zoster and received treatment at a local hospital. Although the skin rash improved, the patient, at times, felt squeezing sensations on her left flank. When the patient was referred to the pain clinic, the patient had a squeezing pain and scars of vesicles on the T10 dermatome level on the left side and her numeric rating scale (NRS) was 5 (out of 10). She felt the squeezing pain several times for approximately one minute each time. Aside from a history of hypertension, there was no other unusual medical history or a history of prior surgeries. She was 152 cm tall and 51.2 kg in weight. Laboratory examinations showed no signs of bacteria or viral infection. Complete blood cell count, prothrombin time and activated partial thrombin time tests showed normal results, and there were no abnormal results in the serologic tests (HBs Ag/Ab, HIV Ag/Ab, quantitive VDRL).\nAfter diagnosis of postherpetic neuralgia, the patient received an epidural block once a week for a total of six weeks. We performed the epidural block with 8 ml of 0.8% mepivacaine with the paramedian approach to T10-T11 with the 'loss of resistance' method. Out of six total procedures, only the first procedure included 20 mg of triamcinolone with local anesthetics, and the rest were composed of local anesthetics only. Any signs or symptoms suggestive of dural puncture, such as cerebrospinal fluid (CSF) aspiration through the block needle, headache, paresthesia or sensory impairment were not observed during each epidural block. Pregabalin 150 mg and amitriptyline 5 mg were also prescribed for twice a day. After each epidural block, a pain free period continued for two or three days but she felt numbness like prior to the procedure. Additionally, the squeezing pain reappeared but the pain intensity was reduced to NRS 3. Thus, we expected full recovery by repetitive epidural blocks and by an additional root block or transforaminal epidural block.\nTwo days after the sixth epidural block, the patient was admitted to the department of neurology, through the emergency department, because the patient felt continuous dizziness and motor weakness in both lower extremities. Five days prior to receiving the sixth epidural block, the patient began to feel slightly dizzy. However, the continual dizziness became extreme by the time the patient was admitted to the hospital. Motor weakness of the lower extremities was felt by the patient two days after the final epidural block. In addition, the patient had symptoms of nausea and vomiting. The patient also could not stand or walk alone. The patient's body temperature was 38.3℃ and she did not have any abnormalities on her skin. Physical examination showed a grade III motor weakness in both of the lower extremities, but the deep tendon reflex and cerebellar function tests were normal. Sensory impairment was not observed. She was referred to our pain clinic, because a complication during an epidural block was considered by neurologists. We reviewed the sixth epidural block, examining for any evidence of dural puncture, aspiration of blood and other abnormal events, but there were none. However, in order to be certain, we performed Magnetic Resonance Imaging (MRI) and CSF tests on the first day of admission. The MRI showed no signs of hematoma, abscess, or arachnoiditis (). No bacteria were detected upon CSF analysis. Considering the results, we were able to exclude the possibility of an epidural block complication. The CSF analysis results showed, however, a mild increase in the WBC count (49,000 count/mm3) and a protein level of 97 mg/dl. Virus serology test of the blood and the CSF showed negative anti-IgG and negative anti-IgM antibodies for herpes simplex virus (HSV) and herpes zoster. Two days after the patient was admitted, the patient complained of urinary difficulty and a Foley catheter was inserted, resulting in discharge of 1,200 ml of urine through the catheter. The patient felt squeezing pain on the T10 dermatome level on the left side during her entire stay at the hospital.\nConsidering her clinical progressions and the increase in protein level in the CSF analysis, herpes zoster myelitis was diagnosed by the neurologist and appropriate treatments commenced. Three days after the patient was admitted, the patient was given intravenous acyclovir 1.6 g and dexamethasone 20 mg daily for two weeks. The patient also received exercise rehabilitation treatment while she was on the medication. After receiving treatment for one week, the patient was able to walk but had difficulty in standing up. The patient showed gradual improvement and four weeks later made a full recovery in motor function. The patient's urinary difficulty improved through urinary training and she fully recovered in eight weeks. |
We evaluated a 9-year-old boy from Onitsha, Nigeria, who presented with a 5-month history of left thigh swelling which progressively increased in size and became painful. He had had no preceding similar swelling or constitutional symptoms. However, about 2 months earlier, he was said to have been treated over a period of 1 month by a traditional bone setter for a presumed left femur fracture sustained in a pedestrian injury. Physical examination revealed a differentially warm, firm, slightly tender mass measuring about 6 cm × 4 cm in the anterolateral part of the distal left thigh. It was not attached to the overlying skin but partly fixed to the underlying structures, and the range of motion of the knee joint was reduced.\nX-ray report showed a reduction in the corticomedullary differentiation of the distal metaphysis of the femur with soft-tissue swelling, preserved fat plane, spiculated periosteal reaction, cortical erosion, and cauterization []. Computed tomography (CT) scan report showed a reduced corticomedullary differentiation in the distal metaphysis of the femur with a sunray periosteal reaction with cortical erosion and a hypodense avascular soft-tissue mass in the distal femur []. Both X-ray and CT scan reports were in keeping with Ewing's sarcoma.\nThe patient was HIV negative and he had a packed cell volume of 35% and total white cell count of 11.4 × 109/l with differentials of 89% neutrophils and 11% lymphocytes. Intraoperative findings when an open biopsy was done included a fibrocystic mass containing brownish fluid situated on, but not attached to the anterolateral portion of the distal femur. Histology report from the biopsy showed numerous histiocytes and a few multinucleated giant cells. Many of the histiocytes have eccentrically located nuclei with cytoplasmic vacuoles containing figure-of-eight-shaped bodies. Similar bodies were also seen in the giant cells and also extracellularly []. The fungal bodies were also identified using periodic acid–Schiff-staining technique [].\nPostoperatively, he had a course of analgesics and antibiotics. When the lesion was confirmed to be of fungal origin, he was commenced on oral itraconazole (200 mg) daily. The surgical incision healed uneventfully, and the patient was discharged home 2 weeks later on oral itraconazole, which he took for a year. Follow-up in our hospital continued for the next 2 years and included clinical examination of the patient and x-rays of the affected femoral bone []. Subsequently, follow-up by telephone communication with the father was used to inquire if any swelling like the initial one treated has developed. There has been none. |
The fifth patient is a 43-year-old female who was diagnosed with diabetes at the age of 13 years. At the age of 21 years, her first pregnancy was complicated by an intrauterine fetal death. At the age of 23 years, a healthy daughter was born with a planned caesarean section. She developed diabetic retinopathy and nephropathy at the age of 28 and 29 years, respectively. The patient suffered from hypertrophic cardiomyopathy, leading to heart failure at the age of 35 years. Two years later, haemodialysis was started. In the same year, the patient received a kidney transplant from her brother complicated by a non-ST segment elevation myocardial infarction acute coronary syndrome 3 days later. One month later, creatinine levels were increasing, showing signs of transplant rejection for which treatment with intravenous methylprednisolone was started. During the treatment, a second acute coronary syndrome occurred. Later she was admitted to the hospital because of sepsis caused by a Staphylococcus aureus infection. After treatment with flucoxacillin, creatinine levels increased and treatment with methylprednisolone was started again. Kidney function was not improving, and for that reason she was treated with anti-thymocyte globulin (ATG) four times. During this treatment, the patient suffered from a third acute coronary syndrome. A year after the ATG treatment, the immunosuppressive treatment was switched from triple therapy to sirolimus and azathioprine. At the age of 40 years, 3 years after transplantation, the m.3243A>G mutation was found as part of a family survey, after her niece was diagnosed with the same mutation. The heteroplasmy level in leucocytes was 16%. Further genetic investigation within the family showed that the patient’s sister and mother of the niece also carried the m.3243A>G mutation. Almost 5 years after transplantation, the patient has stable moderate kidney function (Modification of Diet in Renal Disease estimated glomerular filtration rate 17 mL/min/1.73 m2) without any signs of proteinuria. |
A 40 year old male had a history of single chamber permanent pacemaker implantation (VVIR) in 2001 for complete heart block. As the patient was young, basic investigations were done at that time to exclude auto - immune and infiltrative diseases. Tests for Anti nuclear antibody (ANA), anti phosoholipid antibodies (APLA) and Rheumatoid factor were negative. In 2008, he was detected to have ERI (Emergency Replacement Indicated) and re-implantation was done with the change of pulse generator and pacing lead as lead impedance was high. The old lead was left in situ. After 4 years, he presented in our department with gradually progressive dyspnoea, bilateral pedal oedema and abdominal distension. At the time of presentation, he was in NYHA II functional class. On examination, the patient had pulse rate of 90/ min, blood pressure of 100/60 and normal heart sounds. General physical examination showed bilateral pedal oedema and icterus. The chest examination was normal. Abdominal examination showed distended superficial abdominal veins flowing centrifugally from umbilicus, tender hepatomegaly and shifting dullness, which was indicative of ascites. ECG showed paced rhythum. Chest radiograph revealed pacemaker in situ with its lead at right ventricular (RV) apex and another functionless lead, also at RV apex. 2D trans-thoracic echocardiography showed normal biventricular functions with paradoxical inter ventricular septal motion. Pacemaker lead was seen across tricuspid valve, positioned at RV apex along with another functionless lead at RV apex. This functionless lead had a small loop in right atrium (RA) close to the opening of inferior vena cava (IVC) into RA. A small mass was seen at this site over the lead with an accompanying thrombus at the mouth of IVC which was completely obliterating it (). Similar findings were confirmed with three dimensional echocardiography (). Colour Doppler also showed no flow from IVC to RA, instead increased flow was seen from superior vena cava to RA. A huge conglomeration of veins was seen around IVC which were collaterals, diverting blood from IVC to RA. Ultrasonography (USG) of abdomen showed hepatomegaly with dilated hepatic veins, dilated portal vein with a diameter of 12 mm, and moderate ascites. IVC venography showed no flow from IVC to RA, with flow diverting from IVC along collaterals to RA (). So, a diagnosis of 'thrombus over the functionless lead along with IVC thrombosis, leading to portal hypertension' was made. Basic laboratory investigations were done for excluding thrombophillic disorders. Levels of Protein C, protein S and antithrombin III were normal; reports for Factor V Leiden mutation and Prothrombin gene mutation were negative. USG abdomen and pelvis did not show any abnormality favouring intra-abdominal malignancy leading to increased thrombogenicity. After this, the patient was put on anti-coagulation. There was no improvement with anti-coagulation and patient was thereafter referred to surgeon for retrieval of thrombus and functionless pacemaker lead. |
A 12 year-old girl weighing 41kg was posted for Botox (botulinum) injections and casting for spasticity of the right upper limb. She gave history of having some heart disease since birth for which multiple operations had been done. On going through her past medical records, it was found that she was born with complex congenital heart disease, having dextro-transposition of great arteries,double outlet right ventricle,pulmonary stenosis, and a large ventricular septal defect. She underwent a right subclavian to right pulmonary artery shunt shortly after birth, a Glenn operation at 4 months of age, and an extracardiac Fontan operation (total cavopulmonary conduit) at 5 years of age. She was on chronic therapy with warfarin. At age 10, she was hospitalized with a cerebrovascular accident for a month. Computed tomography (CT) brain showed left middle cerebral artery (MCA) thrombosis due to which she had developed right hemiparesis. The hemiparesis improved gradually and she developed spasticity of the right upper and lower limbs. She had developed pedal edema a few months earlier, due to an element of protein losing enteropathy, which was treated with diuretics and high protein diet. She gave history of no cardiovascular (CVS) complaints at the time of admission, and was regular in her studies and daily activities. She was on treatment with tablet warfarin 5mg and tablet furosemide 20mg once a day.\nOn examination, she was conscious, oriented, well nourished. She was afebrile, pulse was 82 beats/minute and regular, blood pressure was 102/66mm Hg. A median sternotomy scar was present. CVS examination showed a regular heart rate, and a grade 2 systolic murmur. She had spasticity of the right upper limb more than the lower limb. Rest of the systemic examination was unremarkable. Investigations revealed haemoglobin of 11.2g/dL, total and differential white blood cell counts were normal. Renal function tests, serum electrolytes, liver function tests including serum proteins were within normal limits. Prothrombin time was 22s, with an INR of 2.1. Electrocardiogram (ECG) showed sinus rhythm. Echo showed patent functioning cavopulmonary shunts, mild ventricular systolic dysfunction, no thrombi/vegetations.\nA cardiology opinion was taken. She was advised to withhold warfarin and switch over to a low-molecular weight (LMW) heparin 5 days prior to surgery. She was started on enoxaparin 40 mg subcutaneous twice a day. Infective endocarditis prophylaxis was also advised. Enoxaparin was withheld 12 hours prior to surgery.\nOn the night prior to surgery, patient was given tablet alprazolam 0.25mg. Infective endocarditis prophylaxis was given with IV ceftriaxone 1g 1 hour prior to the procedure. It was decided to give general anaesthesia since the patient was very apprehensive and insisted on it. Patient was taken to the operation theatre, monitors were attached (ECG, pulse oximeter, capnograph, non-invasive blood pressure monitor). An infusion of lactated Ringer's solution was started and she was preloaded with around 200ml. Patient was premedicated with IV ondansetron 4mg. Preoxygenation was done for 5 minutes and anaesthesia was induced with IV fentanyl 80 mcg and propofol 80mg IV slowly and maintained with sevoflurane 0.6-0.8% in 50% nitrous oxide in oxygen, on spontaneous ventilation with intermittent assist. Blood pressure was maintained within 20% of the baseline value. Fluids were given to maintain blood pressure. End-tidal CO2 was maintained between 30 and 35mm Hg. Patient received 300ml Ringer's lactate intraoperatively, and the procedure lasted 30minutes. At the end of surgery, a 50mg diclofenac per rectal suppository was given for post op analgesia. Once awake, responding to oral commands and stable, patient was shifted to the recovery room for monitoring. Patient was comfortable, and her vitals were stable. She was shifted to the ward after an hour and had an uneventful post operative period. She was discharged the next day. |
A 45-year-old female Asian nurse with a history of well-controlled hyperthyroidism presented at the emergency room with a sudden onset of dysarthria and paralysis at the weekend. She complained of feeling ill a week before the first visit but had been able to go about her daily life and work. Physical examination revealed a fever over 38.0 °C, altered mental status (13 points on Glasgow coma scale), drop of the right mouth, and paralysis of the right upper and lower extremity. She had not received dental or surgical procedures for one year before the visit.\npresents our patient’s laboratory data at the first visit. While hemoglobin and platelet count were slightly low, white blood cell count was normal (). The prothrombin time (PT) was slightly prolonged, and the activated partial thromboplastin time (APTT) was within the normal range. Renal and liver function tests were normal. Lactate dehydrogenase and C-reactive protein levels were elevated. The leukocyte fraction and the other coagulation tests could not be carried out in our hospital due to it being the weekend. Unfortunately, the clinical laboratory technicians who usually observe blood cells professionally were not available during the weekend.\nComputed tomography (CT) of the head did not show any abnormal findings including an early CT sign. The magnetic resonance imaging (MRI) detected a high-signal-intensity change in her left brain, which indicated M1 occlusion (A,B). Electrocardiography showed normal sinus rhythm. An echocardiogram revealed a vegetation-like structure on her atrial valve. We strongly suspected infectious endocarditis (IE) complicated by cerebral embolism. Therefore, a blood culture was taken immediately, and empirical antibiotics were administered. Anticoagulation therapy was commenced concurrently, and our neurosurgeons decided to perform an endovascular thrombectomy. The blood clot was successfully removed (C insert), leading to rapid and complete reperfusion (C). Soon after, the level of her consciousness improved, and she was admitted into the department of neurosurgery at our hospital.\nHowever, six hours after the endovascular thrombectomy, the level of her consciousness worsened markedly again, and computed tomography revealed massive intracranial hemorrhage around the stroke area. We judged the clinical diagnosis of brain death on the next day, and she died on the fourth hospital day. Regardless of suspecting IE, no bacteria were found in the thrombus obtained from the cerebral artery, and these blood cultures were negative. At the time of her death, leukemia cells were first reported in the peripheral blood () after the weekend. The abnormal leukocytes had abundant azure granules, strong nuclear irregularities, and many Auer bodies (, arrow) in the peripheral blood. A pathological autopsy was performed shortly after death with written informed consent from the patient’s family. |
A 36-year-old Brazilian male patient was admitted to the hospital with a palpable lump in his right breast, located at the junction of the upper quadrants of the right breast (Fig. ). On physical examination, the lesion appeared firm with irregular margins. Axillary lymphadenopathy was negative and there were no palpable supraclavicular nodes. On breast imaging, ultrasonography showed a hypoechoic mass with partially defined contours measuring 4.0 × 3.0 cm, located at the upper region of the right pectoralis major muscle at the 12 o’clock position with muscle infiltration (Fig. ). Histological examination of core biopsy samples revealed a malignant tumor. Preoperative exams, such as X-rays and chest CT scan, abdominal US did not show any signs of disease. Radical mastectomy was then performed, due to pectoralis major muscle infiltration, consisting in removal of the breast along with the major and minor pectoralis muscles. Biopsy of the sentinel lymph node was performed. Gross examination revealed a solid tumor measuring 3.7 × 3.5 cm with a yellowish-tan cut surface and local foci of hemorrhage. Histopathology showed intravascular papillary proliferation of endothelial cells, spindle cell areas and necrosis, atypia and prominent mitotic figures, consistent with the diagnosis of high-grade angiosarcoma with areas of infiltration of the pectoralis major muscle (HE staining, magnification of 400×) (Fig. ). Histopathology also demonstrated a surgical specimen with clear margins, absence of angiolymphatic and perineural invasion, in addition to sentinel lymph node free of metastasis. Immunohistochemical study revealed a tumor positive for CD31 marker (Fig. ), confirming the vascular nature of the tumor. At the two-week follow-up of the surgical procedure, adequate wound healing was observed, without any evidence of the disease. The patient was transferred to the clinical oncology department, where he presented with severe headache and seizures after the second cycle of adjuvant chemotherapy with paclitaxel. Magnetic resonance imaging of the brain was ordered, revealing a right frontal parasagittal lesion, measuring 1.3 × 1.1 cm with a hemorrhagic component and perilesional edema, suggestive of brain metastasis. The disease progressed rapidly, culminating in the patient’s death at 20 days after the onset of neurological symptoms. |
An 8 days old male neonate was born to an Asian mother through vaginal delivery at 37 weeks of gestation, weighed 2,380 g, and had APGAR scores of 9 and 10 at 1 and 5 min, respectively. He was admitted to our hospital with a 2 days history of fever of up to 39°C but did not have respiratory or gastrointestinal symptoms. The infant's family denied any medical history and TB contact. His physical examination at admission documented smooth respiration, clear breathing sound, and no hepatosplenomegaly. The complete blood count indicated a total white blood cell count of 17,500/μL with 69% neutrophils, 20% lymphocytes, 9% monocytes, and 2% eosinophils. The C-reactive protein level was 7.3 mg/dL. The findings of the cerebrospinal fluid (CSF) analysis were normal. Bacterial cultures of the blood, urine, and CSF were negative. Intravenous antibiotics, namely cefotaxime and ampicillin, were administered after admission on the basis of suspicion of neonatal fever. Despite the administration of the antimicrobial combination therapy, the fever persisted and the neonate developed abdominal distension when he was 12 days old. Abdominal radiography exhibited nonspecific dilated bowel loops. Because no improvement in the condition of the patient was observed after changing antibiotics, infection caused by some virus and other atypical pathogen, including Mycobacterium tuberculosis, was considered. Tests for herpes simplex virus, Epstein–Barr virus, cytomegalovirus, hepatitis B virus, rubella, Chlamydia trachomatis, and Toxoplasma gondii were all negative. The repeat C-reactive protein level was elevated to 14.4 mg/dL. Coagulopathy with 323.7 μg/mL of abnormal fibrin degradation product and more than 20 mg/L of D-dimer were also noted. Antibiotics were switched to vancomycin and ceftazidime empirically. Chest radiography displayed only increased right lung field infiltration when the infant was 12 days old (), and chest computed tomography (CT) imaging exhibited a large amount of right pleural effusion with mild inflammatory changes in the right lower lobe when the infant was 15 days old (). Pleural effusion drainage was suggested but refused by his parents at that time. Gastric lavages for acid-fast staining and culture were examined when the infant was 20 days old after his parents agreed to further testing, and one of the three acid-fast stains of gastric lavages yielded few acid-fast bacilli. Repeat chest and abdomen CT imaging performed when the infant was 24 days old indicated patchy consolidation in the right upper lung, multiple new nodules in both the lungs, moderate pleural effusion, and multiple low-density nodules in the spleen and hepatic hilar region without hepatomegaly (). Subsequently, pigtail catheter insertion for pleural effusion drainage was performed. The findings of pleural fluid analysis indicated a total white blood cell count of 10,800/μL with 6% neutrophils, 57% lymphocytes, and 37% mesothelial cells; a total protein level of 4.6 g/dL, a lactic dehydrogenase level of 250 IU/L, and a glucose level of 164 mg/dL. TB infection was strongly suspected. The neonate was administered isoniazid (15 mg/kg/day), rifampicin (15 mg/kg/day), and pyrazinamide (20 mg/kg/day) when he was 24 days old. After initiating anti-TB treatment, the neonate's symptoms and signs subsided gradually. Finally, both gastric lavage and pleural effusion cultures showed M. tuberculosis complex.\nThe neonate's mother was 33 years old, gravida 1, para 1. Her Group B streptococcus test was negative. She had been healthy with no previous medical history and TB contact history; however, she developed a mild dry cough 1 week after delivery, experienced persistent general weakness, and was admitted to our medical intensive care unit because of altered mental status 24 days postpartum. Laboratory examinations indicated leukocytosis, thrombocytopenia, coagulopathy, acute hepatic failure, and acute renal failure. The HIV serology test was negative. A chest X-ray exhibited a miliary TB pattern (). A chest CT image displayed diffuse interlobular and intralobular septal thickening with ground-glass opacities (). Because her neonate was highly suspected to have TB infection at that time, acid-fast staining and TB polymerase chain reaction (PCR) of the sputum were performed. Both tests were strongly positive. The mother was administered anti-TB therapy immediately, but she died 3 days after hospitalization. M. tuberculosis infection was confirmed through sputum culture. |
A 44-year-old Caucasian man was working under a car when the vehicle’s transmission system fell on his chest, squeezing his torso between the heavy item and the ground. After an unknown time, he was found in an unconscious state by a relative, who called for medical aid. It was estimated that at least one hour elapsed before our patient received medical care.\nOn arrival to our emergency department, our patient had a gasping breath without foreign bodies in his oronasal cavities, palpable regular pulses with a rate of 130 beats per minute and an arterial pressure of 80/40mmHg. On pulse oxymetry he had a saturation of 80% on room air. His Glasgow Coma Scale score was 8 (absent eye opening, unintelligible voice responses and limp withdrawal to painful stimuli), his papillae were isochoric and light reflexes were bilaterally present. Because of his altered consciousness and impending respiratory failure, our patient was urgently intubated and put under controlled mechanical ventilation.\nThe rest of the physical examination revealed that his face, the front part of his neck and the upper part of his chest were congested, edematous and covered with numerous petechiae, especially on the conjunctivae and the periorbital skin. In a later bedside ophthalmologic examination, mild bilateral periorbital swelling, severe bilateral subconjunctival hemorrhages, chemosis, mild exophthalmos and mild optic disc edema were observed. Ecchymotic bruises were also noted on the back part of his neck and the upper part of both shoulders. His tympanic membranes were clear and there were no mucosal hemorrhages of his upper airways.\nAbsence of breathing sounds over both lung apices in combination with palpable subcutaneous emphysema over his neck pointed towards the existence of bilateral pneumothorax. Moreover, bloody fluid was drained through the endotracheal tube, indicating possible lung contusions. The physical examination of his heart and abdomen was unremarkable and electrocardiogram was normal. Thoracic X-ray examination revealed bilateral pneumothorax and multiple rib fractures (Figure ). In this respect, bilateral tube thoracostomies were inserted, draining air and blood and eliciting major improvement in his hemodynamic parameters. In subsequent X-rays, bilateral lung opacities were evident, which were consistent with the clinical suspicion of lung contusions. Fiberoptic bronchoscopy was not performed due to the bilateral pneumothorax. Subsequently, our patient was transferred to our intensive care unit (ICU). Arterial blood gases on admission to our ICU were: pH 7.246; partial pressure of carbon dioxide: 58.3mmHg; partial pressure of oxygen: 441mmHg; bicarbonate: 21.9mEq/L; oxygen saturation: 99.9%; and lactate: 1.1mmol/L while our patient was ventilated with a frequency of 15 breaths/min; tidal volume: 700mL; positive end-expiratory pressure: 5cmH2O; and fraction of inspired oxygen: 100%. His Acute Physiology and Chronic Health Evaluation II score was 14, while his past medical history was noted to be non-significant.\nFurther work-up included radiological evaluation of his spine and limbs, which was unremarkable, a normal echocardiography, and head, neck, chest and abdomen computed tomography (CT). On the CT scan, a mild brain edema without signs of hemorrhage was observed, while CT of his chest revealed bilateral hemopneumothorax and sizeable bilateral lung contusions, particularly on his right lung (Figure ).\nSerum biochemistry included elevated levels (ten times above the upper limits of normal) of creatine phosphokinase, lactic dehydrogenase, aspartate aminotransferase and alanine aminotransferase. A urine analysis was normal (Table ).\nIn the ICU, our patient was ventilated with volume-control mode, with a tidal volume of 7mL/kg, frequency 10 to 12 per minute, positive end-expiratory pressure not exceeding 5cmH2O and a gas mixture that was quickly tapered to a fraction of inspired oxygen of 40%. The unimpeded ventilation, the swift restoration of hypercapnia, hypoxia and hemodynamics, the spontaneous containment of the tracheobronchial hemorrhage, the rapid radiographic improvement in the following days (disappearance of the opacities) and the quick recovery, simply by placing thoracostomies tubes, made the event of a potential rupture of major bronchial or arterial branch less plausible. Therefore, we did not proceed to further invasive diagnostic procedures, such as bronchoscopy, which would have added no more information towards the appropriate management of our patient and would even pose some risks.\nFluid resuscitation with crystalloids was copious, in order to prevent renal complications of a potential traumatic rhabdomyolysis. Special care for the brain edema was taken with mannitol administration and frequent neurologic assessments. Regarding his respiratory function, our patient improved swiftly, resulting in an uneventful extubation on the second day of ICU hospitalization. However, his neurologic status lagged behind, as he remained disoriented and agitated until the fourth day. Facial and thoracic petechiae gradually faded within the next three days. Serum aspartate aminotransferase, alanine aminotransferase, creatine phosphokinase and lactic dehydrogenase levels decreased to normal on the seventh day and our patient was discharged from the ICU and transferred to the thoracic surgical ward. |
A 47-year-old man with a 20-year history of chronic hepatitis B was hospitalized with the complaint of yellow urine and skin for 5 months. The patient had been treated with lamivudine in combination with adefovir for 1 year and discontinued treatment of his own 10 months ago. Five months ago, he developed the symptoms of yellow urine and skin and followed by being diagnosed as decompensated hepatitis cirrhosis B and treated with entecavir. During entecavir treatment, he had a total bilirubin concentration of 200−350 μmol/L, prothrombin time of 20−31 s, and a Child-Pugh score of >10. The bilirubin level was persistently high despite repeat (up to four times) artificial liver support therapy. Meanwhile, during the course of treatment, the patient showed hepatic encephalopathy, a large amount of ascites, and hyponatremia with numerous serum sodium concentrations as low as 115 mmol/L. He received large dosage of diuretic, lower salt intake (less than 2 grams per day), water restriction to less than 500 mL per day, and one time paracentesis, but the symptom was not relieved. The patient was treated with tolvaptan at an initial dose of 15 mg qd. After therapy, he had a urine volume of 6100 mL and became obviously thirsty, although the serum sodium concentration showed significant recovery. The tolvaptan dosage was decreased to 7.5 mg qd, but he remained thirsty. For the large volume of urine and the obvious thirst, the dosage of diuretic was decreased once but resumed soon because of increasing ascites. Finally, the dosage of tolvaptan was maintained at 5 mg qd, and the patient's serum sodium concentration was consistently 128 mmol/L and daily urine volume was between 3000 and 4000 mL (see ). During the course of therapy, the condition of patient was markedly improved, as indicated by the continuously decreased level of total bilirubin. After 1 month of treatment with tolvaptan, the drug was stopped and the serum sodium concentration was maintained at 130 mmol/L. Coused drugs were entecavir tablet, ademetionine injection, zolpidem tartrate tablet, lactulose, magnesium isoglycyrrhizinate injection, albumin injection, piperacillin-tazobactam injection, and ornithine aspartate injection during the therapy of tolvaptan. The patient was discharged from the hospital with a total bilirubin concentration as low as 94 μmol/L and a serum sodium concentration of 135 mmol/L. From that time, the patient stopped diuretics therapy and just used entecavir. At the 3-month follow-up, total bilirubin concentration was only 38 μmol/L and serum sodium concentration was 137 mmol/L. B ultrasound showed small amount of ascites. At the 1-year follow-up, the liver function of the patient was normal and B ultrasound showed no ascites at all. |
Our patient was a 61-year-old male. He was admitted into the cardiac care unit (CCU) of our hospital because of more than 16-h precordial pain. Sixteen hours ago, the precordial pain with no obvious cause occurred. The pain was persistent and accompanied by a radiation pain in left shoulder. There were no other concomitant symptoms, including chest tightness, panic, fever, nausea and vomiting, dizziness, headache, etc. There was no significant relief in the pain after taking “SUXIAO JIUXIN PILL.” Then, the patient called an ambulance and went to the local hospital. After the electrocardiogram examination, the local hospital considered the diagnosis as acute myocardial infarction, and oral doses of aspirin and clopidogrel were prescribed. For further diagnosis and treatment, the patient was transferred to the emergency department of our hospital. Electrocardiogram (ECG) examination showed that ST segment elevated in the lead of AVR and V1 and depressed in other leads. The laboratory examination showed that troponin was elevated, so he was admitted to the CCU of our hospital. Asking patients and his family members carefully, the patient underwent coronary angiography in our hospital 11 years ago, showing that 40% stenosis occurred in the distal segment of the left main coronary artery (LM); the stenosis of the proximal segment of LAD was 40%; long lesions in the proximal and middle left circumflex branch (LCX), the narrowest part of LCX was about 80%, and the distal segment of LCX was about 70%; nearly 70% stenosis occurred in the obtuse marginal branch 1 (OM1); the proximal segment of the RCA was completely occluded and the distal segment was visualized through the left coronary collateral branch. After evaluation, CABG was performed on him in our hospital. The patient had a history of hypertension for 11 years, and the blood pressure was as high as 180/110 mmHg. He took felodipine-sustained release tablets and irbesartan to control blood pressure and said that his blood pressure was well-controlled. He also had a history of diabetes for 11 years, and took Metformin and Acarbose for treatment, and his daily blood glucose was controlled at 7–8 mmol/L. He denied a history of chronic obstructive pulmonary disease (COPD), cerebrovascular disease, and other chronic diseases. The patient had 30 years of smoking history, about 20 cigarettes per day. At present, he had quit smoking for 11 years and drunk occasionally. Other family members had no similar clinical manifestations. Past medical history and family history revealed nothing significant.\nOn admission, his body temperature was 36.4°C, the pulse was 96 beats per minute, his respiratory rate was 18 times per minute, his blood pressure was 150/85 mmHg (1 mmHg = 0.133 kPa). Physical examination showed that the patient was in a poor state of mind and spirit. The patient had a mild coarse breath sounds on auscultation and obvious moist rales could be heard. The heart boundary (cardiac dullness) was not big. The heart rate was 96 beats per minute. The heart sounds were normal, and the rhythm was regular. There was no pathological murmur in each valve auscultation area. No edema was found in both lower limbs. There was no distension of jugular vein and no sign of hepatic jugular vein reflux.\nLaboratory examination: the results of myocardial enzyme spectrum showed that cardiac troponin I (cTnI) was 3.8 μg/L (reference value was 0–0.023 μg/L), creatine kinase isoenzyme (CK-MB) was 38 μg/L (reference value was 0–7.2 μg/L), NT-pro BNP was 5,530 pg/ml (reference value was 0–300 μg/L); liver function revealed an ALT level of 11 μ/L, an aspartate aminotransferase (AST) level of 28 μ/L, an albumin (ALB) level of 40 g/L; renal function showed an serum urea nitrogen (BUN) level of 5.2 mmol/L, a serum creatinine (Cr) level of 48 μmol/L; blood sugar (BS) was 9.37 mmol/L; blood biochemistry revealed a total cholesterol (TC) level of 3.7 mmol/L, a low-density lipoprotein (LDL-C) level of 2.98 mmol/L, a sodium (Na+) level of 133 mmol/L, a potassium (K+) level of 3.98 mmol/L. Blood routine showed that the white blood cell count (WBC) was 10.60 × 109/L (reference value was 125–350 × 109/L), the proportion of neutrophils (NEU%) was 81.40%; ESR was 106 mm/h; CRP was 107 mg/L; other examinations were in the normal range. The 12-lead electrocardiogram showed normal sinus rhythm, ST segment depression in lead V4–V6, T wave flattening or inversion in chest lead and limb lead, abnormal Q wave in lead III and AVF (). Echocardiography revealed multiple abnormalities: (i) enlargement of the left heart, with a left atrium diameter of 45 mm and a left ventricular diameter of 48 mm; (ii) the inferior wall of left ventricle became thinner, and the motion almost disappeared; (iii) the activity of myocardium near the apex of interventricular septum decreased. The estimated left ventricular ejection fraction (LVEF) was 45%; (iv) moderate pulmonary hypertension; (v) mild-to-moderate mitral regurgitation, mild-to-moderate tricuspid regurgitation. No obvious abnormality of heart and lung was found in chest X-ray at the bedside.\nBased on the main symptoms of the patient, typical ECG manifestations and changes of myocardial enzyme spectrum, and previous CABG operation history, the main diagnosis of the patient was acute non-ST segment elevation myocardial infarction (NSTEMI); the following diagnoses were made: (i) acute non-ST segment elevation myocardial infarction; (ii) hypertension (Grade 3, very high risk); (iii) Type 2 diabetes. Aspirin and clopidogrel were used to antiplatelet therapy, low molecular weight heparin (LMWH) was used to anti-coagulation, atorvastatin, and ezetimibe were used to decrease the serum lipid level. We used metoprolol, isosorbide mononitrate-sustained release tablets, and trimetazidine to relieve the symptom of angina pectoris; “metoprolol, valsartan, and amlodipine” were used to reduce blood pressure and improve ventricular remodeling; creatine phosphate was used to nourish the myocardium, nicorandil to improve microcirculation of coronary artery. Furosemide, spironolactone, and nesiritide can improve cardiac function, Dapagliptin, acarbose, and metformin were used to treat diabetes. After treatment, the condition of the patient was more stable than before. So, the patient underwent coronary angiography, showing that 50% stenosis occurred in the LM; the left anterior descending branch is small, with a stenosis of about 80% in the proximal segment and irregular in the middle and distal segments. The most severe stenosis is about 70%, and it can be seen that the distal part of the lad is retrograde to the right Corus artery; in LCX, the wall of proximal segment was irregular, the lesions in middle and distal segments were longer, and the most severe stenosis was about 90%; the wall of OM was irregular, and local stenosis was 40%; RCA was occluded from the proximal segment; the LCX bridge was unobstructed.\nWe reexamined the echocardiography of the patients and found: (i) it was in accordance with the echocardiographic findings of myocardial infarction; (ii) enlargement of the left heart, with a left atrium diameter of 41 mm and a left ventricular diameter of 45 mm; (iii) segmental dyskinesia of left ventricular wall and decreased left ventricular diastolic function was found. The LVEF was 51%; (iv) mild pulmonary hypertension; (v) mild mitral and tricuspid regurgitation ().\nThe reexamination of cTnI was 0.01 μg/L (reference value was 0–0.023 μg/L), NT-pro BNP was 4,380 pg/ml (reference value was 0–300 μg/L); ESR was 109 mm/h; CRP was 107 mg/L. After the above positive treatment, the symptoms of precordial pain were significantly improved; the indexes related to myocardial enzyme spectrum were reduced to normal. The renal function and urine volume were acceptable. But the decrease of NT-pro BNP was not obvious; CRP and ESR were significantly increased during the treatment. We considered that pulmonary infection, tumor, and rheumatic disease could lead to significant increase of CRP and ESR. For this reason, we took chest X-ray at the bedside, and the inflammatory indicators were redetected. However, the results of Chest X-ray and the inflammatory indicators showed no signs of acute pulmonary infection or pulmonary tuberculosis. Therefore, the hypothesis that the significant increase of CRP and ESR was due to pulmonary infection was excluded. At the same time, male tumor markers and CT examination showed no obvious abnormality, so the increase of CRP and ESR caused by tumor was also excluded.\nBased on this, we conducted a careful physical examination of the patients. Palpation of bilateral radial artery showed that the pulsation of bilateral radial artery was inconsistent, the pulsation of dorsalis pedis artery was not touched, and vascular murmur could be heard by auscultation of carotid artery. Further detection of the blood pressure of the limbs: the left upper limb was 137/64 mmHg, the right upper limb was 132/52 mmHg, the left lower limb was 90/23 mmHg, and the right lower limb was 113/28 mmHg. We can find that the lower limb blood pressure is significantly lower than the upper limb blood pressure. According to the results of blood pressure and physical examination of limbs, and the increase of ESR, CRP, and other inflammatory indicators, we considered that the patient may be TA. Therefore, we improved CT angiography (CTA) examination; the results showed aortic atherosclerosis and multiple stenosis of its branches, and some showed moderate-to-severe stenosis, bilateral renal arteriosclerosis and multiple stenosis (). And the results of carotid artery ultrasound showed stenosis in the initial segment of left internal carotid artery, left external carotid artery, and right external carotid artery ().\nWe asked rheumatologists for consultation. According to the 1990 American College of Rheumatology (ACR) criteria, the consultation opinion was TA, so we added the diagnosis of TA. The ACR criteria for TA consist of (1) age of the onset before 40 years old; (2) claudication of an extremity; (3) decreased brachial artery pulse; (4) a difference of more than 10-mmHg systolic pressure between two limbs; (5) a bruit over subclavian arteries or the aorta; and (6) angiographic evidence of narrowing or occlusion of the aorta, its primary branches, or large arteries in the proximal upper or lower extremities. If three of the above six items are met, it can be diagnosed as TA (). Intravenous dexamethasone 10 mg daily, calcium carbonate D3, calcitriol capsules, and other oral drug treatments were tailored to treat TA. After 4 days of treatment, Hs-CRP was decreased to 11.67 mg/L, and ESR was decreased to 91. mm/h. The inflammatory index was significantly descended, and NT-pro BNP decreased to 3,100 pg/ml. |
A 63-year-old African American woman presented to the Plastic Surgery clinic from her nursing home with a 2-year history of a painful distal forearm mass. The lesion was initially thought to be a keloid from a stab wound more than 20 years ago; however, she decided to have it evaluated because of a recent increase in size and intermittent bleeding. She denied any numbness, tingling, or weakness of the hand. Her neurovascular examination had normal findings. Clinically, the mass was located on the volar aspect of the mid-forearm, was pink and ulcerated, and 3 × 3 × 4 cm in size (). There was no associated supracondylar or axillary lymphadenopathy.\nBecause of the unique characteristics of the mass, we further evaluated the lesion with a magnetic resonance image of the right forearm. This image demonstrated a 3.8 × 2.1 × 4.1-cm mass in the subcutaneous tissue without invasion into the fascia or muscle (). The lesion was concerning for malignancy, so she was referred to a surgical oncologist for excisional biopsy. One month later, the mass was excised en bloc with careful dissection of the deep margin to maintain the fascia. We used a 6 × 4-cm elliptical incision, and this defect was closed primarily using suprafascial flaps for a tension-free closure.\nOn final pathology, the entire specimen was 6 × 4 × 0.8 cm. It was found to be an invasive nodular melanoma with positive, deep, and radial margins. This case was discussed at our multidisciplinary tumor board with plans of positron emission tomographic (PET) scan, reexcision, and sentinel lymph node biopsy.\nBefore her postoperative follow-up appointment, she underwent a PET scan, which unfortunately demonstrated a hyperactive node in the right axilla as well as a concerning left breast lesion. At her follow-up appointment, we offered a wide local excision with sentinel lymph node biopsy; however, the patient refused to have further surgery. On physical examination, there was a small, red, scaly lesion in the middle of the surgical site concerning for rapid recurrence. We referred her to a breast surgeon for workup of the left breast lesion, which ultimately was found to be ER/PR+ invasive lobular breast cancer. The patient was scheduled for close follow-up in both clinics but had not made her appointments. After reviewing the medical records, the patient refused to have any further surgical procedure at this time. |
This report deals with a 48-year-old man with a 29-year history of systemic sclerosis, who had a 6-month history of painful ulcers on the inner and outer left ankle and on the left hallux. He already had a history of leg and digital ulcers 4 years earlier who responded quite well to intravenous administration of prostanoids. Besides recurrent ulcers he suffered from sclerodactyly, severe Raynaud phenomenon for which he underwent a sympathectomy in the past, and severe esophageal dysmotility with secondary anorexia. There was no evidence for pulmonal or cardiac involvement. Laboratory evaluation showed antinuclear antibody titer of 1 : 1280 with U1-RNP >240 U/mL and RNP-70 of 104 U/mL. Anti-cardiolipin antibodies were negative. Ankle-branchial index showed no signs of arterial insufficiency. In January 2011 the patient developed a painful, sharply margined necrotic ulcer with purple edges on the left outer ankle () and an ulcer on the left hallux (). Intensive local treatments with amniotic membrane transplantation, keratinocyte grafts, and silver foam dressings were unsatisfactory. Use of a dihydropyridine-type calcium antagonist, aspirin, and simvastatin [] was unsuccessful. In April 2011 he developed a new ulcer on the left inner ankle (). Bacterial superinfection occurred and many antibiotic treatments were necessary. Because of further deterioration intravenous administration of prostanoids in combination with subcutaneous administration of low molecular weight heparin [] was started in May 2011. Only the ulcus on the left hallux responded partially (). The leg ulcers however did not show any improvement (). Bosentan 62.5 mg twice a day was started and after 4 weeks elevated to a dose of 125 mg twice a day which was tolerated well. Local treatment was continued with silver foam dressings. After 4 months the ulcers got shallow and smaller and showed good granulating tissue and reepithelialization at the wound edges (Figures and ). After 6 months the leg wounds showed further healing (Figures and ), the ulcer on the hallux closed completely (), and the patient was free of pain. |
A 71-year-old woman was transported to the emergency department of another hospital because of consciousness disorder. Enhanced computed tomography (CT) showed an amount of free fluid in the peritoneal cavity mainly around the right upper abdomen without free air. Ultrasonography identified cholecystolithiasis. Paracentesis revealed intra-abdominal bilious fluid with high levels of total bilirubin (21.6 mg/dL) and amylase (8697 U/L) on biochemical examination. She was diagnosed as having severe peritonitis with septic shock and was transferred to our hospital for emergency surgery and intensive care management.\nShe had no history of past abdominal operations, and other past medical history included choledocholithiasis and pancreatitis. Hematological investigations on admission revealed coagulopathy, renal dysfunction, and circulatory insufficiency (Table ), which indicated septic disseminated intravascular coagulation. Significantly high levels of serum transaminases, bilirubin, and pancreatic enzymes suggested a condition associated with biliary tract disease. As shown in Fig. , enhanced CT revealed that the supraduodenal CBD was markedly dilated similar to a diverticulum (arrow), and the bile duct wall had a partial defect in continuity (arrowhead). Moreover, the common channel of the pancreaticobiliary duct was long at 9.3 mm in length and seemed to be joined outside the muscular layer of the duodenal papilla (arrow) on the coronal CT view (Fig. ). Eventually, we diagnosed biliary panperitonitis due to the spontaneous CBD perforation accompanied with congenital biliary dilatation and pancreaticobiliary maljunction. As her general condition improved following adequate primary resuscitation, she was able to undergo an emergency laparotomy to cure her septic peritonitis.\nIntraoperative findings revealed a large amount of bilious ascites along with edematous omentum. No perforation was apparent either in the gallbladder or the gastrointestinal tract. A necrotic diverticulum-like change with bile leakage was present on the supraduodenal part of the CBD. After the necrotic lesion was removed, a 2.5 × 1 cm perforation was found on the anterolateral wall of the CBD, below the junction of the common hepatic duct and cystic duct (Fig. a, b). Intraoperative cholangioscopy revealed an impacted stone in the major duodenal papilla (Fig. c), but the stone could not be removed easily intraoperatively. The cholecystectomy was performed. The gallbladder showed edematous changes due to inflammation, but was easily dissected. Thorough peritoneal lavage was performed, and the CBD perforation was resolved with a T-tube inserted through the perforation (Fig. d). Based on the preoperative examination and intraoperative findings, the patient was diagnosed as having a perforation of the CBD caused by a combination of congenital biliary dilatation with pancreaticobiliary maljunction, type II by Todani’s classification, and gallstone cholangitis/pancreatitis. Hematoxylin and eosin staining revealed that the resected specimen of the CBD wall was so destroyed that the muscular layer lacked continuity (Fig. a). Furthermore, immunohistochemical examination with anti-desmin antibody did not show the presence of smooth muscle in the tissue (Fig. b). Gallbladder wall revealed that dilated Rokitansky–Aschoff sinuses, mild muscular hyperplasia, and lymphocytic infiltration with hematoxylin–eosin staining.\nThe patient remained unstable and required inotropic agents, artificial respirator support and continuous hemodiafiltration in the intensive care unit until postoperative day 10. Furthermore, multiple additional drainage, and administration of antibacterial and antifungal agents were required for the refractory intra-abdominal and retroperitoneal abscesses. On day 16, active bleeding was observed at the abdominal wall around the T-tube, and hemostasis was achieved by transcatheter arterial embolization. On day 55, T-tube cholangiography revealed that the impacting CBD stone had disappeared naturally and, thus, the long common channel of the pancreaticobiliary duct and the diagnosed pancreaticobiliary maljunction could be observed (Fig. ). After removal of the T-tube on day 136, she was transferred to the hospital for recuperation on day 152. |
A 35-year-old man presented at a local hospital with epigastric pain. Esophagogastroduodenoscopy (EGD) showed that an IVC filter strut had penetrated the third portion of the duodenum (arrow, Fig. ), and this was confirmed by computed tomography (CT) (arrow, Fig. ). In order to retrieve the IVC filter, the patient was referred to our department. He had a history of testicular cancer with para-aorta lymph node metastasis. Left renal vein thrombosis developed because of neoadjuvant chemotherapy before RPLND, and anticoagulants were administered before RPLND. Three years previously, he had undergone left orchiectomy, retro-mediastinal lymph node dissection, and RPLND at the previous hospital. The left common iliac vein was intraoperatively damaged during RPLND. Because the previous surgeon was worried about the high incidence of postoperative DVT and PTE, anticoagulant therapy was continued after RPLND. However, because DVT developed in the left common iliac vein after the initial surgery, a retrievable IVC filter (ALN, France) was placed in the IVC caudal to the renal vein to prevent PTE, and the patient had been receiving anticoagulant therapy. Because follow-up CT after IVC filter placement showed that DVT persisted at the left common iliac vein despite anticoagulant therapy, the IVC filter could not be retrieved at the previous hospital.\nEnhanced CT also revealed that DVT remained in the left common iliac vein. Because ultrasound examination showed organized DVT, an IVC filter was considered unnecessary. An endovascular approach was considered unfeasible for retrieval because two of the filter struts had penetrated the duodenal wall. An extensive discussion with an internal medicine specialist was performed. Because DVT remained with no remarkable changes in CT images for 3 years and the incidence of PTE caused by DVT in the left common iliac vein would be low, we chose surgical treatment for this patient in order to prevent bleeding at the duodenum.\nThe IVC filter was retrieved through cavotomy, and the duodenal penetration site was repaired using intraoperative EGD clipping. The operation lasted 5 h and 54 min, and the intraoperative bleeding volume was 1172 mL. Because it was not possible to mobilize the duodenum due to adhesions resulting from the previous surgery, the IVC at the sites caudal to the renal vein could not be explored. However, a mesenteric incision caudal to the third portion of the duodenum enabled encircling and taping of the IVC (Fig. ). After clamping the IVC cranial and caudal to the duodenum, a 5-cm vertical incision was made on the IVC cranial to the duodenum and the IVC filter was retrieved (Fig. a). Although the head of the IVC filter had penetrated into the IVC intima, we were able to bluntly peel the filter head from the intima. The IVC incision was closed using a continuous 5-0 Prolene suture (arrow, Fig. b). The IVC clamping time was 22 min. Intraoperative EGD revealed no bleeding at the duodenal penetration site (Fig. ). To prevent duodenal bleeding or perforation, the penetration site was repaired by EGD clipping. Retrieved IVC filter was presented as (Fig. ). The patient did not develop any postoperative complications and was discharged on postoperative day 16. |
We present a case of a 43-year-old G2P2, married, cis-gender female who presented on testosterone orally and topical dihydrotestosterone to her clitoris to increase in size. The patient did not suffer from gender dysphoria and had a healthy social and sexual relationship with her husband. The patient was cleared by our center’s psychologist and deemed mentally competent to make her decisions regarding surgery and consent. She also completed the Arizona Sexual Experiences Scale (ASEX) for an objective evaluation following surgery []. The patient desired conversion of her enlarged clitoris into a micropenis or metoidioplasty without scrotoplasty or urethral lengthening, in addition to vaginal tightening at the introitus for self-reported vaginal laxity. Her desire was to have mobile skin completely around her enlarged clitoris as a penis would. Upon physical examination it was noted that her clitoris and labia minora tissues were hypertrophic and a relaxed vagina existed whereby four digits readily passed in the vaginal canal ().\nAfter informed consent was obtained, a general anesthetic was used and the patient was placed in the lithotomy position. The markings were performed and local anesthetic with epinephrine was used. The clitoral suspensory ligament at the apex of the clitoral hood was dissected with loupe magnification avoiding injury to the two clitoral nerves and this was released allowing lengthening of the clitoris. An inferior wedge resection of the labia minora with circumferential wrap of the clitoral body for creation of a micropenis () was then performed and a diamond perineoplasty to narrow the vaginal introitus so that only two digits would be readily inserted into the vaginal canal. ().\nThe patient was discharged following surgery and not allowed to resume vaginal intercourse for 2 months. She was allowed to exercise at 6 weeks. She had no complications and her ASEX Score after surgery was 5 at 5 months.\nAt 1 year she had resumed penetrative vaginal intercourse with orgasm at 3 months, and had improved erogenous sensation in her now micropenis, in addition to improvement in her ASEX score to 5 when previously it was 12. (Average score for adults (patients and controls) without clinical sexual dysfunction is 14 in women and 10 in men). The patient also began using a clitoris suction device after the 1 year to further enlarge its size (). |
A 58-year-old Caucasian male presented with acute kidney injury (AKI) and proteinuria in June 2016. He had originally been diagnosed with melanoma along the right flank in the 1980s that was treated with wide excisional removal. He was followed closely for over three decades with no signs of metastatic disease until he was noted to have a mass near his prior melanoma excisional scar in summer 2014. A biopsy at that time revealed an epithelioid neoplasm with morphology and phenotype highly suggestive of melanoma that was positive for the BRAF V600E mutation. A Positron Emission Tomography/Computed Tomography (PET/CT) at that time showed numerous bilateral pulmonary nodules, and a subsequent right upper lobe wedge resection did confirm metastatic melanoma. In May 2015, he was initiated on ipilimumab (3 mg/kg) and nivolumab (1 mg/kg) every 3 weeks for management of his metastatic melanoma. His course was complicated by grade 3 dermatitis, colitis, and hepatitis, which were treated with courses of prednisone. Therapy was ultimately discontinued in October 2015, after receiving a total of three cycles intermittently, as a result of worsening hepatitis confirmed by liver biopsy. Because of persistent bilateral lung metastases and left pleural metastases, the patient was initiated on dabrafenib 150 mg twice daily and trametinib 2 mg twice daily in December 2015, which he remained on until his presentation to nephrology in June of 2016. He had no personal or family history of chronic kidney disease (CKD). He denied exposure to nephrotoxic agents. He was only taking amlodipine 10 mg for hypertension and had been receiving immunotherapy with dabrafenib and trametinib since December 2015. He denied tobacco use, illicit drug use, and toxic environmental exposure. His blood pressure was 172/89 mm/Hg and had 2+ edema bilaterally. The remainder of his physical examination was normal, and laboratory results indicated a creatinine level of 2.4 mg/dL with a urinary protein-to-creatinine ratio of 2 g/g. His urinalysis showed dysmorphic erythrocytes and red blood cell casts. Renal ultrasonography was normal. Serologic testing was negative for Antineutrophil cytoplasmic autoantibody (ANCA), PR-3, Myeloperoxidase (MPO), and Anti-glomerular basement membrane (anti-GBM) antibody. Complement levels were normal. A renal biopsy was performed in September of 2016 showing focal crescentic (2 of 15 glomeruli with cellular crescents), proliferative, and sclerosing glomerulonephritis with diffuse linear staining of glomerular capillary loops dominant for IgG (3+), IgA (2+), kappa (2+), and lambda (1+) minimal changes (Fig. ). Ultrastructural examination of three glomeruli demonstrated areas with open capillary loops and preserved foot processes. Other areas demonstrated diffuse effacement of foot processes with variable thickening and wrinkling of glomerular basement membranes. No immune complex disease or tubuloreticular structures were identified.\nRepeat anti-GBM testing remained negative, and the patient’s creatinine eventually rose to a peak of 3.8’mg/dL. He had no signs or symptoms of lung hemorrhage. Dabrafenib and trametinib were discontinued, and he was subsequently initiated on oral cyclophosphamide (2 mg/kg/day) and pulse intravenous methylprednisolone (1000 mg daily for 3 consecutive days) followed by 1 mg/kg/day of prednisone. Serum creatinine improved to 2.5 mg/dL, and the active urinary sediment resolved. Immunosuppression with cyclophosphamide was discontinued after 4 months of therapy, and he was weaned off prednisone by 6 months.\nThe patient was off all immunotherapy for his malignancy, and his renal function remained relatively stable over the ensuing 12 months. By June 2018, a PET/CT of the chest showed evidence of metastatic melanoma to the left upper and medial lobes of the lung. The patient was placed back on nivolumab, which seemed to stabilize his oncologic disease. Unfortunately, 4 weeks after reinitiating nivolumab, his creatinine jumped from 2.8 to 5.8 mg/dL and home hemodialysis was initiated. The patient remains stable on home hemodialysis and was more recently taken off nivolumab and placed back on dabrafenib and trametinib for progressive metastatic pulmonary disease, which has led to significant symptom control. |
A 37-year-old woman (informed consent was obtained) was diagnosed with calculous cholecystitis on abdominal sonography. There was no bile duct stricture or stone on magnetic resonance cholangiopancreatography. Her body mass index (BMI) was 30.2 kg/m2, and she had no significant medical history except for 2 cesarean deliveries. She underwent robotic cholecystectomy with the bikini-line incision technique in which ports are placed as described in (8-mm port at the right, middle, and left lower quadrants) []. The operative time was 75 min, with no drainage or intraoperative complications. After the specimen was retrieved from the mid-lower incision site, no fascial closure was performed. The patient was discharged 2 days postoperatively, after an uneventful recovery. Later on the day of discharge, the patient presented to the emergency room with complaints of left incision site pain, a distended abdomen, and 2 episodes of vomiting since discharge. Physical examination showed tenderness in the left lower quadrant with a 3-cm palpable mass above the 8-mm port-site scar. There was no sign of peritonitis. All hematological and biochemical workups were within normal limits. Abdominal radiographs showed no sign of obstruction in the small and large bowel loops. Computed tomography of the abdomen revealed small bowel obstructions secondary to dilated fluid-filled herniated loops through abdominal fascial and peritoneal layers at the 8-mm port site (). After 5 days of conservative treatment and failure to resolve the mechanical ileus, the patient underwent emergency diagnostic laparoscopy. The herniated loop was twisted and obstructed within the fascial defect (). The loop was delivered after the defective fascial and peritoneal layers were carefully dissected. Gangrenous changes were present at the part of the loop adherent to the defective area. The gangrenous part of the small bowel loop was kept under a warm saline-soaked sponge while the patient was given 100% oxygen for approximately 5 min. However, the color and peristalsis of the gut loop did not significantly improve. Therefore, bowel resection and anastomosis of the ischemic area were performed through a small laparotomy incision near the umbilicus. The abdominal herniation site was closed in layers, allowing proper anatomical repair of the fascia and peritoneum. The patient remained in good condition during the postoperative period and was discharged without further complications. |
A 35-year-old male with a history of seizure disorder, traumatic brain injury, and cannabis abuse presented to the emergency department with multiple gunshot wounds to the lower abdomen and left hip. The patient was emergently taken to the operating room for an exploratory laparotomy. He required a small bowel resection with re-anastomosis and was found to have multiple enterotomies to the mid-jejunum, a defect to the greater omentum, and a small left retroperitoneal hematoma, which were repaired appropriately. The patient required significant colloid and crystalloid resuscitation for hypovolemic shock. Consequently, he developed hyperkalemia from transfusion protocol and an acute kidney injury from shock and renal hypoperfusion. The patient was treated conservatively with calcium gluconate, insulin, and intravenous fluids. He was later treated with loop diuretics resulting in the resolution of his hyperkalemia and acute kidney injury. He received two doses of 1 g of calcium gluconate intravenously in the left antecubital fossa. A few hours later, he developed a necrotic lesion with numerous surrounding bullous lesions (Figure ). Compartment syndrome and deep vein thrombosis were ruled out. Initially, cellulitis was suspected but the bullae led to suspicion for immunological disease. A punch biopsy of the skin was performed which showed pauci-inflammatory subepidermal blisters (Figure ). Direct immunofluorescence was negative for immune deposits, which ruled out autoimmune bullous disease. This finding was attributed to calcium gluconate extravasation causing local calcinosis cutis.\nThe patient required wound care and debridement at the lesion. However, his course became complicated as he was discharged from the hospital when his other ailments improved and he was nonadherent with his therapy at home. He presented to the primary care clinic three weeks later with wound progression and slough formation with eschar (Figure ). He required another debridement and was reassigned to the wound care clinic (Figure ). An excisional skin biopsy was performed which showed gangrenous necrosis with signs of acute and chronic inflammation (Figure ). The wound eventually improved, but this adverse reaction to calcium gluconate led to a more complicated and lengthy hospital course than predicted by his original admission for a gunshot wound. |
A 22-years-old woman came to our hospital with chief complaints of slurring of speech, hemiparesis, disorientation, confusion, and restless movement of the right half of the body. The restless movement of the right half of the body was followed by weakness of the left half of the body, which was insidious in onset, progressive in nature. According to the patient party, she was unable to perform normal daily activities with normal conversation five days ago. The weakness of the left half of the body first appeared in the left lower limb, followed by worsening of that of the left upper limb. The patient developed slurring of speech and global aphasia later. There was a history of fever for 20 days, which was intermittent; the maximum temperature recorded was 103 degrees F associated with chills, rigor and excessive sweating. The patient also complained of multiple episodes of vomiting. History of clenching of teeth, up rolling of eyes, and abnormal body movements was also present.\nThe chest examination was normal. Cardiac rhythm and rate were normal with no murmurs. The abdomen was flat, soft, and non-tender with normal liver and spleen. Muscle strength of the right upper and lower limb was normal, but in the left upper and lower limb power was 0/5. Thalamic contracture of the right hand was also seen. All deep tendon reflexes were normal and plantar response was bilateral extensor. Signs of meningeal irritation were present. A computed tomography (CT) of the brain revealed infarct in the anterior limb of the right internal capsule and left the lentiform nucleus, bilateral lateral third ventricles including the 4th ventricle, were dilated. That signified communicating hydrocephalus and infarct on the genu of the corpus callosum on the left side ().\nCerebrospinal fluid (CSF) from lumbar puncture revealed specific gravity of 1.015, white cell count (WBC) of 3 cells/cumm/mm3, all lymphocytes, a protein level of 398.7mg/dl, a glucose level of 17mg/dl a serum glucose level of 130mg/dl and LDH of 467U/I. CSF fluid adenosine deaminase was 273mg/l. CSF was negative for bacterial culture, and KOH stain came negative. Her fasting blood glucose level was 226mg/dl; a known case of diabetes. The initial chest film showed no definite active lung lesion. Serology tests for Human Immunodeficiency Virus, Hepatitis B and C virus were negative. Moreover, liver and renal function tests were normal.\nShe developed hyponatremia 130meq/l and hypokalemia 3.4meq/l during the course of the disease which was corrected by IV fluid and KCI. Her liver function tests were normal. Based on clinical grounds and preliminary cerebrospinal fluid (CSF) laboratory results, antitubercular therapy was initiated. She was also started on dexamethasone and a combination of meropenem, vancomycin, acyclovir, and aspirin. |
A previously healthy 39-year-old woman was referred to our hospital because of a cystic lesion in the liver demonstrated by abdominal ultrasonography (US). Laboratory studies, including liver function tests, and tumor markers were also within the normal limits. Serological markers for hepatitis B or C viral infection were undetectable. Abdominal US revealed a well demarcated, heterogeneously low-echoic mass 170 mm in diameter in right lobe of the liver. Abdominal computed tomography (CT) during hepatic arteriography (CTHA) revealed early ring enhancement in the peripheral area in the arterial phase and slight internal heterogeneous enhancement in the delayed phase (Figures and ). Magnetic resonance imaging (MRI) showed that the tumor had low signal intensity on T1-weighted images and some foci of high signal intensity on T2-weighted images. Gadolinium ethoxybenzyl (Gd-EOB) MRI revealed no uptake in the corresponding area (Figures , , and ). Abdominal angiography demonstrated a large avascular region in the liver corresponding to the tumor, although no typical features of cavernous hemangioma were evident (). 18-Fluorodeoxyglucose positron emission tomography (FDG-PET) revealed no abnormal FDG uptake. With these radiological findings, malignant liver tumor could not be excluded, such as biliary cystadenocarcinoma, cholangiocarcinoma, mesenchymal tumors, and hepatocellular carcinoma associated with cystic formation.\nThe patient underwent posterior sectionectomy. Intraoperative examination revealed a relatively soft dark red tumor (); the resected specimen weighed 1.1 kg and measured as 170×100×80 mm. The cut surface of the tumor revealed a white, solid, and cystic mass that was elastic, soft, and homogeneous with a yellowish area considered to be myxoid degeneration (). Histological examination showed that the tumor mostly consisted of sclerotic area and cavernous hemangioma area is partly observed (). Sclerotic area presents diffuse fibrosis () and the typical histology of cavernous hemangioma was confirmed in some parts. In addition, marked increase and dilation of medium sized veins with cavernous form were frequently noted in the surrounding areas of tumor (). The increased and dilated veins show positivity of CD31 immunostaining being a marker of endothelium (). The pathologic features were consistent with sclerosing hemangioma. The postoperative course was uneventful, and the patient was discharged on postoperative day 10. |
A 69-year-old man with upper abdominal pain was referred to our hospital in August 2006 by a local physician. Upper gastrointestinal endoscopy revealed a Borrmann type II-like tumor in the posterior wall of the lower part of the stomach (). The biopsy specimen revealed well-differentiated adenocarcinoma, and immunohistological staining showed that tumor cells were positive for alpha-fetoprotein-producing (AFP). An upper gastrointestinal series radiography showed an elevated lesion with a central depression () in the lower part of the stomach. Laboratory examination revealed mildly elevated levels of aspartate aminotransferase (AST 67 IU/L), lactate dehydrogenase (834 IU/L), and alkaline phosphatase (565 IU/L). Serum level of carcinoembryonic antigen and CA19-9 was normal, but that of AFP was markedly increased (160,000 ng/ml).\nAbdominal contrast-enhanced computed tomography (CECT) revealed a 14 cm long hypovascular tumor in the left lobe of the liver (). On the basis of these findings, we diagnosed AFPGC with synchronous liver metastasis.\nOur initial treatment plan was to perform a distal gastrectomy and an extended left lobectomy of the liver. However, another liver metastasis was found in the right lobe of the liver by the intraoperative sonography. We limited the surgery to distal gastrectomy with D2 lymph node dissection and Billroth I reconstruction in August 2006. The resected specimen was determined to be a 2.8 cm long Borrmann type II-like tumor ().\nHistopathological examination of the gastric tumor revealed a well-differentiated adenocarcinoma with neoplastic growth of hepatocyte-like cells (). The level of invasion was submucosal and 3 of 35 dissected lymph nodes were positive for metastasis. Intraoperative biopsy of the liver tumor showed features similar to those of the gastric tumor (). Immunohistological staining showed that tumor cells in both the gastric tumor () and the biopsy specimen from the liver tumor () were positive for AFP.\nAfter surgery, we placed a catheter into the proper hepatic artery for HIA. Chemotherapy using oral tegafur/gimeracil/oteracil potassium (TS-1, Taiho Co. Ltd., Tokyo, Japan) (120 mg/body/day from days 1 to 21)/HIA-cisplatin (CDDP) (30 mg/body/day on days 8 and 21, every 28 days) was started at day 15 after the surgery. After 4 courses, CECT revealed that the size of the metastatic tumor in the left lobe had decreased to 53.6% of the peak (), and the small liver metastasis had disappeared. The serum AFP level had decreased to 2,910 ng/ml.\nSix months after the surgery, the serum level of AFP began to increase, that is why we performed a left lobectomy. The resected specimen showed atrophic change in the left lobe and an odd-shaped soft liver tumor (). On cut sections, the tumor was yellowish and highly denatured (). Microscopic findings revealed degenerative changes and necrosis in almost all sections of the tumor. Viable cells were similar in structure to the microscopic findings of the primary gastric cancer. After liver resection, 9 courses of adjuvant oral TS-1/HIA-CDDP and oral TS-1/CPT-11 by drip infusion of vein (div) were administered.\nTwenty-five months after the first surgery, we performed a partial lung resection because of new metastasis to the lung (). Four months after partial lung resection, radiotherapy was performed to treat 2 new lung metastases. We obtained complete response transiently, but another new lung metastasis and para-aortic lymph node metastases appeared immediately. To treat these metastatic lesions, we performed chemotherapy sequentially as follows: 6 courses of paclitaxel (PTX) (div); 2 courses of 5'-Doxifluridine (DFUR) (oral)/Docetaxel (DOC) (div)/CDDP (div); 5 courses of DFUR (oral)/DOC (div); 8 courses of intravenous injection of 5-fluorouracil (5FU)/Adriamycin/Mitomycin-C (FAM); 2 courses of TS-1 (oral)/CPT-11 (div); and 6 courses of PTX (div) ().\nIn spite of these combination chemotherapies, metastatic lesions became difficult to control at 54 months after the first surgery, and the serum AFP level rose to 84,434 ng/ml. Liver metastases () and mediastinal lymph node metastasis enlarged gradually (). The patient complained of respiratory distress resulting from bronchial oppression by enlarged lymph nodes. We could not use trastuzumab because immunohistochemical analysis revealed that human epidermal growth factor receptor type 2 was not expressed in either the primary lesion or the metastatic liver tumor. We proposed best supportive care, but the patient wanted to try a molecular-targeted drug. The administration of oral sorafenib tosylate (sorafenib) was begun at a dose of 800 mg/body/day using of a private import. After 2 months, his serum level of AFP dropped from 100,000 to 59,198 ng/ml. CECT revealed marked attenuation of vascularity at the liver metastasis, but a decrease in tumor size was not observed (). The mediastinal lymph nodes shrunk in size by 16.2% (). The patient's respiratory distress was completely alleviated, and stable disease was observed for a period of 4 months. The only adverse events observed were grade 1 hoarseness, increased AST, and hand-foot syndrome. The patient eventually died from cancer cachexia at 60 months after the first surgery. |
An 85-year-old woman presented to the emergency room with progressive dyspnea for 4 wk with abdominal distension and lower extremity swelling for 2 wk. Past medical history was significant for hypertension and atrial fibrillation, which were being managed with low dose metoprolol succinate. Physical examination revealed moderate respiratory distress and irregularly irregular rhythm with the heart rate ranging 70 to 90 beats/min. Rales were noted over the both lower lung fields, with raised jugular venous pressure, tender hepatomegaly, abdominal distension and bilateral lower extremity edema. The cardiac exam was notable for harsh grade III/IV holosystolic murmur that was loudest at the apex. A bruit was heard in the posterior abdomen loudest at the right costovertebral angle. However no palpable thrill was felt. The electrocardiogram showed atrial fibrillation with the ventricular rate of 80 however without ST/T wave changes. Laboratory analysis revealed elevated b-type Natriuretic Peptide (BNP) of 4497 pg/mL (reference range < 100 pg/mL).\nTransthoracic echocardiogram, performed as part of workup for heart failure, showed moderate concentric left ventricular hypertrophy with preserved left ventricular ejection fraction of 55%-60%. However, the patient was noted to have right heart failure with dilated right atrium and ventricle. The presence of bruit in the posterior abdomen prompted an ultrasound (USG) that revealed a peripherally calcified large anechoic structure within in the right kidney with the color flow on color Doppler imaging concerning for a large renal artery aneurysm and/or a vascular malformation. Computed tomography (CT) angiogram done for further evaluation showed aneurysmally dilated renal artery and vein (Figure and C). The distal renal artery had a peripherally calcified focal aneurysm (Figure ) with early enhancement of renal vein suggesting an AV fistula. Besides severe dilatation of IVC, hepatic veins, and gonadal veins were seen due to compensatory volume overload while right side cardiac chambers were enlarged (Video 1 and Figure ). The patient was deemed to be a poor surgical candidate, and the decision was made to manage by endovascular occlusion. The interpolar branch of renal artery was catheterized that showed a large aneurysm with immediate opacification of the renal vein. It was decided to completely obliterate the aneurysm using large stainless steel coils. After using 22 stainless steel coils, the angiogram image showed a faint visualization of the IVC (Figure ). It was then decided to reinforce the obliteration of fistula by occluding the feeding distal renal artery by 12 mm Amplatzer vascular plug II (AGA Medical Corporation, Plymouth, MN). Following the deployment of the AVP II, there was no opacification of the venous system (Figure and C). Her symptoms were improved and and the clinical exam showed resolution of jugular venous distention, decreased abdominal distension and lower extremity edema. The ascites resolved on follow-up CT (Figure ) and the BNP decreased to 296 pg/mL at 6 mo. |
A five-year-old developmentally normal boy was hospitalized with fever on and off for more than three months. Fever was found to be high-grade and was associated with painless and clear discharge from both ears. There had been discharge offrank pus from his right ear with pain and impaired hearing in the week before hospitalization. He had received both oral and topical medications (comprising fluoroquinolones and aminoglycosides) for his ailment from several practitioners without any respite to his symptoms. He had been immunized appropriately.\nOn examination, he was found to be febrile, toxic, and undernourished with wasting. His submandibular, cervical, and axillary lymphnodes were found to be significantly enlarged bilaterally. His left upper deep cervical lymph nodes were found to be matted. There were multiple healed perforations in both tympanic membranes with granulation tissue, furuncle, and pus discharge from his right ear. The pus was swabbed and sent for microbiological examination. A fine needle aspiration cytology of the left upper deep cervical node revealed caseating granulomas suggestive of tuberculosis. A chest X-ray which was obtained showed miliary mottling []. Tuberculin test elicited no response, and HIV I and II ELISA were also found to be negative. The history had to be reviewed again for possible contact with a known tuberculosis case but was found to be negative.\nAs the child was toxic with high-grade fever and chills, a complete blood count was ordered which showed neutrophilic leukocytosis. The ear swab culture grew S.aureus and smear for acid-fast bacillus (AFB) and culture was reported to be negative (probably because the child had been on aminoglycoside ear drops for variable periods before presentation). The child was treated with appropriate antistaphylococcal antibiotic based on culture and sensitivity of the organism. In view of the chronic symptoms, chronic and painless clear aural discharge initially and miliary mottling on chest X-ray, a diagnosis of miliary tuberculosis with otitis media was made and antitubercular therapy (ATT) had to be added to his treatment. With the above treatment, fever and earache subsided, and pus discharge reduced from his right ear. However, he continued to have clear discharge from both ears for which he required frequent toileting. He was discharged after two weeks of hospitalization on ATT. On follow-up at four weeks, he continued to be afebrile, gained weight, his ear discharge reduced considerably, and his lymph nodes regressed in size. Hearing assessment was not done as he reported a subjective improvement in hearing. |
A 45 year old female with the diagnosis of 10 years' progression of definite () bilateral type I Menière's Disease, according to the Lopez-Escamez classification (), with recurrent vertigo and hearing loss attacks symptomatically controlled with sulpiride (50 mg) on demand, was referred to the cochlear implants unit of a tertiary hospital center as a possible candidate for cochlear implantation, because of profound hearing loss (1997 BIAP) in the left ear and fluctuating moderate-severe (1997 BIAP) hearing loss in the right ear. The most recent vertigo episode occurred 10 days prior to her visit to the cochlear implant unit. No visual impairment or other alterations were found on otoneurotologic examination; normal ocular movements, normal smooth pursuit, and saccadic movement, absence of ocular misalignment on skew deviation test and no spontaneous nystagmus were also found, despite the recency of the last vertigo attack. Other neurological examinations and standard cerebral MRI were also normal for this patient.\nIn addition to the same tests as for case I, because of the bilateral MD diagnosis, the patient had received 3 months prior a 3 Tesla MRI examination of the inner ear using the HYDROPS MRI sequence (): this sequence is based on the digital subtraction of images produced by the different time diffusion of gadolinium along the inner ear fluids. This MRI showed bilateral cochlear and vestibular endolymphatic hydrops with left side hydrops predominating, as can be observed in Figure .\nThe instrumental vestibular testing was performed by the same senior neurotologist using a vHIT ICS Impulse™ USB hardware version with software version 4.1: this device was a different unit from the device used in case I. Figure shows enhanced vHIT eye velocity responses for both sides, with an aVOR gain value of 1.14 on right horizontal canal function test and 1.05 for left side. In this case, the vHIT calibration was repeated four times with similar enhanced gain values obtained, and a fifth calibration was done with the default system calibration parameters, also yielding similar aVOR gain values. The VVOR test was also performed on this patient using the vHIT ICS Impulse™ device, finding an enhanced eye velocity response during the VVOR test with a measured VVOR gain () value of 1.39 for left side and 1.35 for right side.\nFor both cases presented in this paper, the patients gave written consent to publish the results obtained from their clinical examinations and instrumental tests. |
To our knowledge, this is the first reported case of an index finger MCP joint dislocation surgically treated by a lateral approach.\nThe authors describe a case of a 16-year-old male who suffered a fall onto his outstretched right hand during a soccer game. The patient presented to the ER with pain and deformity of the index finger MCP joint. Volarly, the prominence of the second metacarpal head was evident ().\nRadiographs confirmed a dorsal index finger MCP joint dislocation and showed a small dorsal osteochondral fragment (Figures and ).\nAfter multiple unsuccessful reduction attempts under ring block by different physicians, the patient was referred to surgery.\nUnder general anesthesia, a lateral surgical approach () was performed on the MCP joint. A straight longitudinal incision was made over the lateral aspect of the MCP joint; the volar neurovascular bundle and the dorsal branch of the digital nerve were identified and retracted with Farabeufs.\nInterposition of the volar plate () preventing the reduction was observed. Applying gentle traction and flexion, the MCP joint was reduced, and proximal volar plate reinsertion with a 4-0 Vicryl suture was performed.\nThe posterior joint capsule was identified and split longitudinally, above the collateral ligament. Once adequately exposed, a small osteochondral fragment was found (). Reduction and retrograde fixation of the osteochondral fragment with a 1.7 mm screw were performed, burying the screw head in the cartilage.\nThe joint capsule, subcutaneous layer, and skin were closed using appropriate sutures. Reduction was confirmed by intraoperative fluoroscopy.\nThe patient was placed in a volar splint with approximately 45° of flexion and discharged on postoperative day zero without any complications.\nImmobilization was removed by week 3. Radiographic control revealed joint congruence, and the patient was encouraged to actively mobilize the finger.\nAt week 6, the fracture was consolidated (Figures and ). The joint was painless and presented slight stiffness (ROM 0-70°). The patient could return to competition with protective syndactyly.\nOne year postoperative, there was no pain, growth disturbance, or joint stiffness, with full ROM of the index finger. |
We present the case of a 34-year-old male with a past medical history of hypertension, hypercholesteremia, anxiety, acid reflux, and irritable bowel syndrome who presented with the complaints of severe cramping abdominal pain associated with nausea, vomiting, bright red blood per rectum, and constipation. The patient denied having similar symptoms in the past or any history of abdominal surgery. He had an upper gastrointestinal series 10 years ago, which was unremarkable except for a hiatal hernia. He is a body builder and admitted to the use of AAS. He was 1500 mg of testosterone/nandrolone, via intramuscular injection, per week for 3 months. 3 weeks prior to the presentation, he abruptly decreased the dose to 500 mg/week.\nOn presentation, he was found to be diaphoretic, in mild distress, with tachycardia at 100 beats/min and tenderness in the suprapubic region with no guarding, rebound tenderness or palpable masses. Laboratory studies were normal apart from an increased white blood cell count at 15.0, increased hemoglobin at 17.9 and increased hematocrit at 51.2%. He had mild changes in sodium (134), as well as blood urea nitrogen (24). He weighed 265 pounds, with a body mass index of 40.3. A computer tomography (CT) scan of the abdomen demonstrated mild diffuse nonspecific colonic wall thickening, with two separate areas of apparent short segment small bowel intussusception without evidence of obstruction [Figures –]. In addition, a small umbilical hernia containing fat was visualized. Our patient was subsequently medically managed, with pain control and hydration. A colonoscopy completed the day after admission demonstrated edema and erythema in the distal ileum and colon, most densely starting in the distal transverse colon and increasing within the rectum. There were no ulcers, polyps, tumors or sites of hemorrhage noted. Biopsies were taken from each section of the colon. The subsequent pathological evaluation indicated acute and chronic colitis from the descending colon forward and changes suggesting ischemic colitis in the rectum [Figures –].\nA follow-up CT scan was completed on the third day of admission but demonstrated no definitive intussusceptions []. There was no longer any overt colitis evidenced though there may have been an underlying inflammatory process based on submucosal lucency in the transverse colon. With the apparent resolution of the small bowel intussusceptions with conservative medical management, accompanied by a resolution of the patient's pain, he was discharged home. |
A 15-year-old female patient was admitted to the Orthopedics and Traumatology Department with the complaint of a mass on the left upper back for five years. The patient complained of the inability to sleep in a supine position, pain with shoulder motion, and cosmetic discomfort for two years. She reported that there had been no growth in the size of the mass in the last three years. The patient did not have a history of trauma, family history, a history of osteochondroma in another region, weight loss, systemic signs, or any other known disease. On examination, a hard, oval solid mass was palpable with a size of about 6×4 cm on the superior-medial dorsum of the scapula (Figure ). The mass was fixed, and the margins of the mass were prominent and they moved with the scapula. Abduction and extension of the shoulder were restricted and painful. There was no neurologic deficit in the upper limb. X-rays of the left shoulder revealed a bony mass arising from the dorsal aspect of the left scapula (Figure ). A pedunculated mass was detected with a computed tomography (CT) scan on the dorsal aspect of the scapula (Figure ). The patient was informed, and surgery for excisional biopsy was planned.\nUnder general anesthesia, the patient was placed in the prone position. After cleaning the operation field with iodine, a sterile drape was applied. An incision was made over the mass. The soft tissues were dissected carefully and the borders of the mass were exposed (Figure ). The peduncle was identified, and the mass was excised using an osteotome. The specimen was sent for histopathologic examination (Figure ). The histopathologic investigation confirmed the diagnosis of non-malignant transformation osteochondroma. The cartilaginous cap was 2 mm. Non-steroidal anti-inflammatory drugs were ordered for pain relief. On the postoperative third day, the patient was discharged without any complications. After the operation, an arm sling was recommended for two weeks. Shoulder exercises were initiated after suture removal. At one-year follow-up, the patient’s complaints had completely regressed and no recurrence was observed. The patient has given informed consent for the case report to be published. |
A 14-year-old previously healthy girl presented 2 weeks after an uncomplicated laparoscopic appendectomy for non-perforated acute appendicitis in a regional hospital. The girl complained of gradually reducing urinary frequency to twice per day and prolonged hesitancy. The micturition stream was initially weak and slow before becoming interrupted. Straining did not produce stronger urinary stream. She had never suffered from urinary tract infections (UTIs) or constipation and opened her bowels daily.\nFollowing an episode of acute cystitis 2 months later, she completely lost her ability to void. She was put on indwelling Foley urinary catheter, and her cystitis was successfully treated with antibiotics. After every attempt to remove the urinary catheter, she had to be catheterized again with 300 to 1200 mL of urine volume registered. She noted loss of urge to urinate and felt only dull pain in suprapubic region and right iliac fossa on extreme bladder distention. The girl was kept on indwelling urethral urinary catheter and referred to a tertiary center to determine the etiology of her urinary retention.\nShe was examined with normal clinical findings and no obvious pathology on abdominal and pelvic ultrasound scan (USS). A pediatric neurologist found nothing abnormal, and magnetic resonance imaging (MRI) of the brain and spine, electromyography (EMG) of the lower extremity, somatosensory-evoked potentials (SEP) of tibial nerve, electroencephalogram (EEG), and lumbar puncture were with no pathology. On USS, the gynecologist described multiple follicular cysts on ovaries bilaterally and found no pathology explaining her urinary retention.\nOur pediatric urologist performed an examination under general anesthesia including a free calibration of the urethra up to 26F followed by normal findings on cystoscopy. Videourodynamic study (VUDS) showed an asensitive and hypotonic bladder. The bladder filling had to be stopped at 360 mL due to the patient's discomfort. Maximum intravesical pressure achieved 11 cmH\n2\nO. When pulling the urodynamic catheter out of the bladder manually, the maximum urethral pressure measured was 120 cmH\n2\nO. On vesicocystourethrogram (VCUG), there was no vesicoureteral reflux, a smooth bladder wall and closed bladder neck (\n).\nPsychologic and psychiatric evaluation identified no major problem. During the following 2 years of repeated admissions to several regional and university hospitals, many of the tests described above were repeated, including an MRI of brain and spine with identical conclusions.\nClean intermittent catheterization (CIC) was recommended to the patient. However, because of poor tolerance of CIC due to frequent macroscopic hematuria and pain, a suprapubic catheter was placed. Thereafter, she suffered recurrent symptomatic afebrile UTIs caused by multi-resistant bacterial strains, e.g.,\nKlebsiella\n,\nPseudomonas\n, or\nEscherichia\n. Finally, after 2 years, based on the history, symptoms, and urodynamic findings, she was diagnosed with Fowler's syndrome (FS).\nFor the treatment of FS, the patient was indicated for S3 neurostimulation. The implantation of two Medtronic S3 neurostimulators, type Interstim II, bilaterally in the upper gluteal region was performed under general anesthesia in two phases. The first phase was a transcutaneous implantation of the electrodes into S3 foramina and their connection to externalized neurostimulators. The first procedure took 30 minutes. As the patient restored her voiding completely back to normal when switching on the neurostimulators and experienced no side effects, she could undergo the second phase 4 weeks later—permanent subcutaneous implantation of the neurostimulators (\n). The second procedure took 15 minutes under general anesthesia.\nWith a transcutaneous remote control, she was able to modify the intensity of stimulating current to avoid any discomfort (\n). On the last follow-up, 4 months after the implantation, she voided four to six times per day with post-void residuals up to 50 mL on USS. Unfortunately, she suffered two prolonged episodes of burning on micturition even after the operation. On both occasions, she was diagnosed with acute cystitis by\nE. coli\n107 that was treated with antibiotics after sensitivity testing. |
We recently observed a healthy 64-year-old Iranian man who presented with numerous asymptomatic multilobular, oval-to-round, well-defined 0.5 to 1.5 cm cystic lesions, with central umbilication (central black eschar) over the upper portion of chest []. The lesions were dome shaped and umbilicated. They were soft on palpation and could be depressed only by firm pressure. No scale or epidermal changes were apparent. By squeezing, a yellow greasy material was pushed off from the lesions. He was not taking any medications.\nOn further questioning, the patient had been treated one week ago with leeches, under the supervision of a general practitioner, for presumed infection. This protocol had led to the inflammation and bulging of the lesions. The punctum of the leech was observed in the center of all the lesions []. On physical examination five dermal nodules ranging from 1 to 6 cm in diameter were observed on the chest []. Review of systems produced negative results. The patient had no pain or any other symptom. A skin examination did not reveal any other abnormalities. There were no palpable lymph nodes. A routine blood test was within normal limits.\nThere was no history of similar lesions in any other family member. There was no history of trauma. The patient was not using any topical or systemic medication. Two weeks before his visit, he had history of leech therapy under the supervision of a general practitioner.\nHis medical history was significant for leech therapy of lesions five days previously.\nWe made the diagnosis of inflammatory epidermoid cyst, giant comedone, and leech bite on the basis of the constellation of clinical features. The patient was treated with oral ciprofloxacin, at a dose of 2 g daily, and 2% topical erythromycin solution, based on smear testing that showed gram-negative colonization, probably Aeromonas Hydrophila. The inflammatory component of the cutaneous lesions resolved ten days after administration of ciprofloxacin and withdrawal of leech therapy. Despite improvement, evidence of the cystic lesions persisted.\nHe was followed up for another two weeks and after disappearance of the inflammation, with the patient under local anesthesia, the well-circumscribed mass was completely evacuated with a sharp curette and comedone extractor. The patient was subsequently lost to follow-up. |
63-year-old woman who had suffered from the right medial knee pain for 5 years and was not responsive to conservative treatment was admitted to our clinics. 30° varus-valgus stress X-ray indicated that the patient had an intact MCL and LCL. After the detailed physical examination and reviewing of X-ray images, it was decided that UKR would be the most suitable option for the patient with anteromedial knee osteoarthritis. After spinal anesthesia application and sedation, the UKR surgery was performed with a standard minimal invasive midline vertical incision and a medial parapatellar approach; the patella was removed laterally but not dislocated or everted. The patient received a medial partial knee implant with a mobile-bearing insert (medium size with 4 mm thickness; Oxford®, Zimmer Biomet Inc., Warsaw, IN, USA). Following the UKR surgery (), weight bearing was allowed as tolerated by the patient and a standard postoperative physiotherapy was started on the first postoperative day. The patient was discharged at postop 2nd day when she met the following criteria: independent ability to get dressed, to get in and out of the bed, and to sit and rise from a chair/toilet; independence in personal care; and mobilization with crutches. After discharge, a home-based exercise program was given to the patient. At postoperative follow-up, our patient acquired a full knee RoM in the postop 1st month and returned to independent daily activities without any external support in the postop 3rd month.\nAt postoperative 1st year after first UKR application, the patient fell down while getting on a public bus; this caused that the right knee of the patient was exposed to the valgus force vector. After that moment, the patient heard a pop sound and felt an incredible pain that prohibited the flexion and/or extension of the medial side of the right knee. And then she was admitted to our emergency department. The first evaluation was performed, and the patient was diagnosed with a grade 3 MCL rupture and the UKR insert dislocation (). Having completed the preoperative preparations, the patient was operated on the same day. After anesthetic administration, a surgery with a standard minimal invasive midline vertical incision and a medial parapatellar approach (to a previous incision site) was performed to change the mobile-bearing insert with the same size (medium-sized mobile-bearing insert with 4 mm thickness; Oxford®, Zimmer Biomet Inc., Warsaw, IN, USA). After having changed the mobile-bearing insert, the MCL structures were repaired and anchored to its femoral origin with a 5 mm titanium anchor. Following the surgery, weight bearing and full RoM with a hinged knee brace were allowed as tolerated by patient and a standard postoperative physiotherapy was started on the first postoperative day. Crutches were recommended for 2 to 3 weeks to enable the patient to regain a normal gait. The brace was used continuously for 4 weeks and thereafter during the day for 2 weeks. After the physiotherapy program administration, the patient was discharged at postop 1st day.\nThe patients were evaluated regarding pain intensity (Numeric Pain Rating Scale (NPRS)), active range of motion (RoM), and quality of life (Short-Form 12 Health Survey (SF-12 Health Survey)). Functional capacity was evaluated using the Iowa Level of Assistance Scale (ILAS), Iowa Ambulation Velocity Scale (IAVS), Hospital for Special Surgery (HSS) knee score, and Timed Up and Go (TUG) test. Rehabilitation program and outcome evaluation were conducted by one clinical physiotherapist at preoperative period (before the first UKR application), at discharge (postop 2nd day after the first UKR surgery), and at postop 2nd year (after 2 years from the MCL repair and the insert change). The evaluation results are shown in . |
A 46-year-old female was referred for TN and hypothyroidism due to autoimmune thyroiditis. This patient gave informed consent for this case report. Her thyroid US showed bilateral small TNs, <10 mm. Her left TN measured 4.8 mm × 4 mm × 4 mm []. This TN had worrisome US features including irregular margins. Her neck US visualized a small left paratracheal (level 6) lymph node (LN) measuring 5.6 mm × 3 mm × 4.4 mm that appeared partially cystic []. In addition, there was an enlarged LN measuring 17 mm × 4 mm × 5 mm with possible microcalcifications [] in the left lateral neck posterior to the jugular vein []. Shear wave elastography (SWE) velocity measurements of the LN were 6.59 m/s and 5.32 m/s []. The US used was a LOGIQ E9 (GE Ultrasound, Milwaukee, WI, USA).\nThe patient underwent FNAB of the 4.8 mm left TN due to irregular margins. Her left neck level 6 LN was also biopsied due to complex, cystic nature of the node. The FNAB cytopathology for both lesions was read as malignant (Bethesda Category VI). The left lateral LN could not be sampled due to the proximity of the jugular vein.\nThe patient underwent total thyroidectomy with central and left lateral neck dissection at a tertiary care center. Her surgical pathology report indicated multifocal bilateral papillary thyroid carcinoma. The largest focus in the right lobe was 3 mm, and the largest focus in the left lobe was 5 mm, corresponding with her biopsied TN in the left lower pole. Overall, 18 LNs were resected, and one positive LN in the left paratracheal area was reported. The location of this LN corresponded with her FNAB finding. Her surgical pathology did not identify any metastatic LN in the left lateral neck.\nSeven weeks after her surgery, while on a low iodine diet for 2 weeks, she underwent Thyrogen-stimulated I-123 scanning with single-photon emission computed tomography (CT). There was no abnormal activity in the left neck. She was immediately treated with 100 mci of I-131. Her postablation scan also showed no abnormal activity in the central or lateral neck.\nSix months after her initial surgery, her thyroglobulin (TG) level was 4 ng/mL; however, her TG antibody was elevated at 1.9 IU/mL (normal range <0.9 IU/mL). TG and TG antibody were measured with radioimmunoassay by the Beckman Coulter Methodology by Lab Corp (LabCorp Esoterix Inc, Calabasas Hills, CA).\nHer post-surgical neck ultrasound again demonstrated the 17 mm × 4 mm × 5 mm LN with possible microcalcifications in the left lateral neck, posterior jugular area. This LN could not be accessed for FNAB due to jugular vein proximity. The patient had a magnetic resonance imaging study of the neck soft tissue that did not report any abnormal findings and only indicated multiple benign-appearing small LNs. After considerable debate, the patient underwent a lateral neck exploration to remove the target LN. The pathology of the second neck dissection showed one of the five LNs involved by metastatic carcinoma. |
In June 2001, a 76 years old male was referred to our institution hospital with clinical signs and symptoms of acute urinary retention, complaining of voiding difficulties during the previous 2 months. On catheterisation, 600 ml of urine could be rescued. Physical examination was unremarkable, except from digital rectal examination which disclosed an uneven swollen and enlarged prostate gland, of stony-hard consistency, with an irregular capsule profile. The patient had been healthy until the time of our observation. No history of radiation and hormonal therapy could be highlighted. Hence, a complete diagnostic work-up was planned.\nSerum PSA and PAP showed in-range values. Transrectal ultrasound demonstrated hypoechoic lesions in the left peripheral zone of the prostate gland. Excretion urogram, urine cytology and urethro-cystoscopy were negative for malignancy, while bladder neck was slightly elevated on cystoscopy.\nAn abdominal and pelvic computed tomography (CT) scan was then performed, disclosing an irregularly enlarged prostate with a peripheral hypodense mass within, compressing the base of the bladder and disrupting the prostatic anatomy. No enlarged lymphnodes could be detected at any abdominal site.\nOn magnetic resonance imaging (MRI), the boundary between the transition zone and the peripheral zone of the prostate gland was unclear and the signal intensity had decreased in the left peripheral zone on T2-weighted images. Extra-capsular disease could be documented in at least 2 sites of the prostatic profile. No osseous spread could be observed at total body bone scan.\nSextant transrectal ultrasound-guided needle biopsy of the prostate was subsequently performed, with histological examination demonstrating nests and sheets of moderately differentiated squamous carcinomatous cells characterised by intercellular bridges. Focal areas presented with evidence of keratin pearl formation. No squamous metaplasia or transitional cell or adenocarcinomatous components could be observed.\nImmunohistochemically, the PSCC component was non reactive for PSA, PAP and cytokeratin 7 and 20, but stained positive for high molecular weight cytokeratin (Figure ,). Considering all the data mentioned above, a diagnosis of AJCC-UICC Stage III (cT3aN0M0) prostate PSCC was then made.\nThe patient was given 3 courses of chemotherapy (CT) with cisplatin (CDDP) 75 mg/m2 on day 1 and continous infusion 5-fluorouracil (5-FU) 750 mg/m2 on day 1 to 5.\nHe subsequently underwent a full-course of radiation therapy (RT) using linear accelerator (18 MV photons), with a conventional four-field box technique up to 46 Gy to the whole pelvis followed by a boost dose to the prostate bed of 20 Gy and by an adjunctive dose of 6 Gy to the prostate gland (72 Gy in total). The patient was treated in supine position, with a six-field conformal field arrangement, delivered using three-dimensional conformal RT. Treatment was, generally, well tolerated, with no acute toxicity recorded.\nAfter therapy had been completed, the patient was periodically examinated, with a clinical, radiological and serologic follow-up. He remained free from disease for 5 years, finally relapsing locally within the pelvis and subsequently dying of acute renal failure due to bilateral uretero-hydro-nephrosis in June 2006. |
A 76-year-old man was referred to our hospital for having lower back pain for 5 months, which was suspected to be L2/3 spondylitis on magnetic resonance imaging (MRI). He had previously undergone cervical laminoplasty for ossification of the posterior longitudinal ligament and diffuse idiopathic skeletal hyperostosis 19 years earlier. He had a history of hypertension and diabetes. Although a history of BCG vaccination was unknown, he had no previous history of tuberculosis infection. He had also undergone transurethral resection of a bladder tumor (TUR-Bt) and had been treated with intravesical mitomycin C (MMC) for the bladder cancer 1.5 years earlier. Four months later, he again underwent TUR-Bt, received intravesical MMC, and started BCG therapy for the recurrence of bladder cancer. After the sixth course of intravesical BCG therapy, he was aware of systemic weakness and loss of appetite, and was unable to walk. He appeared to have septic shock and therefore was treated in the intensive care unit. In spite of systemic analyses, the source of his infection and the causative bacteria could not be identified. He was subsequently diagnosed with hypercytokinemia caused by BCG therapy. He showed clinical improvement without the administration of antituberculosis drugs, and was discharged 9 months before he came to our hospital.\nExcept for a temperature of 37.2 °C, his vital signs were within normal limits. Although there were no motor and sensory disturbances in the legs, he was unable to walk owing to lower back pain. Physical examination demonstrated vertebral tenderness at the L2/3 level. Laboratory analysis demonstrated a normal white blood cell (WBC) count of 6300 /μL, a high erythrocyte sedimentation rate (ESR) of 53 mm/h, and a high C-reactive protein (CRP) level of 2.7 mg/dL. There were no other abnormal laboratory findings regarding anemic changes, kidney function, or liver function. The patient did not receive tuberculin skin testing.\nThere were no notable findings on electrocardiogram or chest X-ray. X-ray of the lumbar spine displayed collapsed endplates of L2/3. Sagittal T1-weighted MRI displayed a decreased signal in the L2/3 disc and the vertebral bodies (Fig. ). Sagittal T2-weighted MRI displayed an increased signal in the L2/3 disc and fluid collection in the anterior part of the vertebral bodies (Fig. ). Axial T2-weighted MRI displayed an increased signal around the posterior area of the vertebral bodies, which extended into the left epidural space and reached the peripheral muscle tissue and the area near the aorta (Fig. ).\nOn the second day of hospitalization, the patient underwent an L2/3 disc biopsy, but the general bacteria culture was negative, and the smear was negative for Ziehl-Neelsen staining. The patient’s blood culture was negative, and the result of T-SPOT.TB® (T-SPOT), which is a type of interferon-gamma release assay (IGRA), were also negative; the number of spots for both 6-kDa early secretory antigenic target (ESAT-6) and 10-kDa culture filtrate antigen (CFP-10) was 5 or less. The causative bacteria remained unidentified and therefore he was started on empirical therapy with intravenous ceftriaxone. On the fifth day of hospitalization, a plain computed tomography scan, which was performed for systemic examination, showed soft tissue development in the adjacent abdominal aorta at the L3 level (Fig. ), which was suspected to be an infectious aortic aneurysm. A 2-stage operation was planned for the spondylitis with adjacent infectious aortic aneurysm, to prevent rupture of the infectious aortic aneurysm, obtain spine stabilization, drain the abscess and make a diagnosis. On the tenth day in hospital, because the risk of rupture was considered to be low, the patient underwent an L2/3 laminectomy followed by posterior fixation using percutaneous pedicle screws at T12, L1, L4, and L5 as the first stage. Specimen cultures of the lumbar vertebrae, yellow ligament, necrotic tissue, etc., were negative for general bacteria, specimen smears were also negative for Ziehl-Neelsen staining, and there were no pathological findings of caseating granuloma or necrosis. On the fiftieth day of hospitalization, because the infection had been controlled but the inflammatory response was sustained, he underwent replacement of the aneurysm with a synthetic graft by vascular surgeons, and lesion curettage and L2/3 anterior interbody fusion by iliac bone transplantation as the second stage. Spinal surgery was performed in the transabdominal approach owing to the risk of rupture. On pathological analyses, the L2/3 intervertebral disc, vertebral bone, and tissue surrounding the vertebral bone and aorta showed caseating granuloma and necrosis with multinucleated giant cells and epithelioid cells upon hematoxylin-eosin staining, and positive bacilli upon Ziehl-Neelsen staining. The tuberculosis-polymerase chain reaction (Tb-PCR) result of the tissue was also positive, using COBAS® TaqMan® MTB Test, which is a real-time PCR system targeting the 16S rRNA gene region of Mycobacterium tuberculosis complex DNA. Owing to the patient’s history of BCG therapy, negative T-SPOT, pathological findings, and positive Tb-PCR, the pathogenic bacteria of the spondylitis was considered to be BCG. We then started multidrug therapy with antituberculosis drugs, including isoniazid (INH), rifampin (RFP), and ethambutol (EB), because BCG is typically resistant to pyrazinamide (PZA). PCR-based genomic deletion analysis was performed using the specimens to distinguish BCG from the other M. tuberculosis complexes. Specifically, multiplex PCR was performed utilizing region of difference 1 (RD1), which is present in the DNA of other M. tuberculosis complexes but is deleted in the DNA of BCG []. Primers ET1, ET2 and ET3 bind and amplify a 190-bp region in BCG, whereas a 160-bp region is amplified in the other M. tuberculosis complexes, as observed by electrophoresis on an agarose gel. A clinical isolate sample from our patient was identified as BCG with a deletion in RD1 (Fig. ). Specimen cultures from the first-stage and second-stage operation were later identified as members of the M. tuberculosis complex using a mycobacteria growth indicator tube. After therapeutic intervention, the patient’s WBC count, ESR level, and CRP level were improved and MRI displayed no signs of active infection in the spine, epidural space, peripheral muscle tissue, or aorta. On the ninetieth day after hospitalization, the patient was discharged from our hospital and transferred to a different hospital for physical rehabilitation. |
A 63-year-old white man received primary radiation therapy for a T2N0 SCC of the supraglottic larynx in October 2006. He suffered a recurrence and underwent a total laryngectomy and neck dissection in February 2007. He was staged as a recurrent T3N0 SCC of the larynx. He underwent anterior lateral thigh flap reconstruction to close the laryngo-pharyngeal defect. He had partial loss of the flap and development of a pharyngo-cutaneous fistula. He had multiple secondary procedures to cover the carotid artery and local flap procedures to close the fistula. He was gastrostomy tube dependent and aspirated secretions that led to recurrent and frequent hospitalizations. Repeat barium swallow confirmed a pharyngo-tracheal fistula. Since he failed multiple surgeries to primarily close the defect, including placement of a salivary bypass tube, an esophageal removable stent was requested in an attempt to close the fistula. Upper endoscopy was performed with a 4.9-mm thin endoscope, which revealed a skin flap with hair starting at approximately 13 cm from the incisors. At above 18 cm from the incisors, a fistula was seen, containing some granulation tissue. The remaining part of the esophagus was normal distal to that area. Under fluoroscopic guidance, a fully covered removable esophageal stent (18 × 100 mm ALIMAXX-E Alveolus, Inc., Charlotte, NC) was placed. The stent was deployed over Savary wire under fluoroscopy guidance. Four centimeters of the stent was proximal to the leak and the 6 cm were distal. Repeat endoscopy showed the stent in a proper position covering the fistula and extending 14-24 cm from the incisors. Contrast readily passed through stent into the intrathoracic esophagus. There was no filling and thus complete closure of previously identified tracheoesophageal fistula occurred. No other leaks were observed. The patient tolerated liquid challenge very well prior to being discharged home after the same-day procedure. The patient, however, complained of “sore throat” upon cervical flexion, which was treated symptomatically with narcotics. The stent was replaced twice at 3-4 month intervals. While the fistula is reducing in size, it remains patent. The patient reports symptomatic satisfaction with his liquid diet and is able to tolerate the stent well. |
We present the case of a 65 year-old lady referred to us from a rural hospital where she was treated with thrombolytic therapy for a presumed acute anterior myocardial infarct. She had presented with central chest pain radiating down her left arm with ECG findings of 2 mm ST elevation in V2 and V3. There was no recent history of psychological stress although she had been on antidepressant drugs for two years. Four hours after thrombolysis she developed acute pulmonary oedema and a new systolic murmur. At this stage it was presumed she had acute mitral regurgitation secondary to a ruptured papillary muscle, ischaemic dysfunction or an acute ventricular septal defect.\nShe was transferred on heparin and glyceryl trinitrate infusion, in acute pulmonary oedema (confirmed by chest X-ray), with a systolic blood pressure of 110 mmHg, and heart rate of 130/minute. Urgent echocardiogram showed severe mitral regurgitation (see Figure ). Her mitral valve annulus was slightly widened at 3.2 cm and subvalvular structures were intact. She also had left ventricular apical ballooning (LVAB) and systolic anterior motion (SAM) of the mitral valve leaflet with septal contact – left ventricular outflow tract gradient (LVOT) 60–70 mmHg (see Figures and ). Coronary angiography fourteen hours after her presentation showed no obstructive coronary lesions. Ventriculogram revealed akinesis of the anterolateral wall and apex of her left ventricle, more extensive than any single coronary territory, and grade 4/4 mitral regurgitation (see Figures and ).\nShe had an intra-aortic balloon pump (IABP) inserted and was transferred to theatre shortly thereafter. She was in cardiogenic shock, with severe MR and SAM still prominent, despite the IABP. We elected to replace her mitral valve, and at operation, the mitral valve was mildly myxomatous but there were no structural abnormalities, chordae and papillary muscles being intact. She had a mechanical mitral valve replacement (MVR) with a 29 mm St Jude valve. The anterior leaflet was excised and PTFE (gore-tex CV4) was used to reconstruct the subvalvular apparatus. The posterior leaflet was plicated to the annulus. Post-operatively there was no LV outflow obstruction and ventricular function improved to within normal limits by the 10th post-operative day. She recovered well. |
A 2-year-old girl presented to our emergency room in cyanotic spells. She was a known case of TOF and midline thoracic defect for 3 months of age. The child had to be intubated in the emergency room considering severe desaturation not responding to usual measures. There was a visible midline cardiac pulsation on the chest wall with an overlying central raphe [] without any palpable sternum.\nEchocardiography showed large malaligned subaortic ventricular septal defect (VSD), severe infundibular and valvular pulmonary stenosis, confluent- and adequate-sized branch pulmonary arteries, right aortic arch, and bilateral superior vena cavae. A computed tomography scan of the thorax was done to rule out any other associated malformations. The scan confirmed a complete CS []. The child underwent cardiac catheterization study to confirm intracardiac anatomy and to rule out aortopulmonary collaterals.\nA combined staged surgical procedure in the form of intracardiac repair followed by chest wall reconstruction was planned. Meanwhile, the child was kept ventilated.\nThe patient underwent Dacron patch closure of VSD, infundibular resection, pulmonary valvectomy, and transannular autologous pericardial patch augmentation of the right ventricular outflow tract. Branch pulmonary arteries were adequate and confluent. A conscious decision was taken by the team members to postpone the sternal reconstruction by 24 h in view of a major intracardiac procedure having been performed. Sternal reconstruction was done subsequent morning. We performed bilateral sliding chondrotomies of the first four ribs and local advancement of the pectoralis major muscle to close the defect primarily with No. 2 stainless steel wires [Figure and ]. The child was electively ventilated and extubated on the 4th postoperative day. Her hospital stay was prolonged (20 days) due to the need for intense chest physiotherapy and oxygen dependency. The patient was discharged with stable sternum without any deformity. She is doing well on the last follow-up as well, 1 year after the procedure. |
A 53-year-old male with no medical history presented to the emergency room (ER) complaining of recent onset fatigue along with episodes of fever, abdominal pain, and oliguria. Gross clinical examination on admission revealed hypotension, tachycardia, fever, and diffuse abdominal tenderness (more prominent in the right hypochondrium). Laboratory workup revealed elevated inflammation markers, thrombocytopenia, and acute kidney injury. The patient was admitted with an initial diagnosis of intra-abdominal infection and empiric antimicrobial therapy was commenced despite the absence of specific findings on abdominal computed tomography (CT). On Day 3 he suddenly developed right-sided hemiparesis due to cerebral infarct as shown on brain magnetic resonance imaging (MRI). In search of the aetiology of the brain infarct thorough clinical examination revealed a holosystolic apical murmur along with upper extremities petechiae, splinter haemorrhages of the nail beds, and non-tender erythematous macules on the palms recognized as Janeway lesions (). Based on these findings IE was strongly suspected. Same day transthoracic (TTE) and transoesophageal echocardiography (TOE) demonstrated large vegetations (maximum diameter of the largest vegetation ∼2.4 cm—) on the mitral leaflet that resulted in severe valve regurgitation (severe valve deformation with multiple regurgitation jets, systolic flow reversal in the pulmonary veins, vena contracta ∼6 mm of the largest mitral regurgitation jet) further supporting this hypothesis ( and Video 1). Infective endocarditis was confirmed when blood cultures grew Staphylococcus\naureus. Proper antibiotic treatment was started. The patient remained haemodynamically stable, with mild congestive heart failure symptoms controlled with low-dose diuretics. He was offered urgent surgical treatment to avoid further embolization which he denied. The rest of his clinical course was uncomplicated and after 6 weeks of antimicrobial therapy he underwent replacement of his mitral valve with a mechanical prosthesis due to persistent severe mitral regurgitation. Patient was seen in the clinic 6 months after discharge. He was doing well and a TTE showed a well-functioning prosthetic mitral valve. |
A 77-year-old man presented for evaluation of a pruritic rash on both buttocks and thighs in October 2016. He has a history of hypogonadism and receives insertion of testosterone pellets (Testopel, Endo Pharmaceuticals Inc., Malvern, PA) every six months. His first insertion was 18 months earlier in April 2015; he did not develop any skin lesions after the initial insertion. However, within 28 days after each subsequent insertion (October 2015, April 2016, October 2016, and April 2017) he developed a pruritic rash on his buttocks and thighs. Skin lesions appeared within four weeks after the second treatment of 12 testosterone pellets; each pellet was 75 mg for a total dose of 900 mg. He received an intramuscular injection of triamcinolone (corticosteroid) from his primary care physician to treat the initial and subsequent episodes, which promptly treated the first and second reactions. He then presented four weeks after his fourth injection. A cutaneous examination showed erythematous plaques and patches on the buttocks. Similar lesions were on his thighs (Figures -).\nA microscopic examination of the skin biopsy showed orthokeratosis and mild acanthosis of the epidermis. Neither spongiosis nor elongation of the rete ridges were prominent. There was vacuolar alteration of the epidermal basal layer. An inflammatory infiltrate of lymphocytes was present not only around the blood vessels in the upper dermis, but also at the junctions between the epidermis and the dermis. In summary, the changes are those of a vacuolar interface dermatitis (Figures -).\nCorrelation of history, clinical morphology, and pathology established the diagnosis of testosterone pellet associated dermatitis. Topical treatment with a high potency corticosteroid cream (clobetasol propionate 0.05% twice daily) was initiated and the skin lesions completely resolved within a week.\nThe likelihood of re-occurrence of the dermatitis with subsequent testosterone pellet therapy was discussed with the patient. However, he insisted on continuing treatment of his hypogonadism with the same medication. A decreased dose of 750 mg (ten 75 mg testosterone pellets) was inserted six months later in April 2017. Within 28 days, he developed similar cutaneous plaques and patches, of lesser severity, on the same location of his buttocks and thighs (Figures -).\nThe biopsy of the new skin lesion showed the same pathologic changes as his previous skin biopsy. The skin lesions completely resolved within a week after initiating treatment with clobetasol propionate 0.05% cream. |
A 31-year-old African-American man underwent orthotopic liver transplantation 10 years ago for primary sclerosing cholangitis (PSC). He had moderate acute cellular rejection five months after the transplant but had no history of any transplant-related infections. He presented to the emergency department with a one-week history of lower abdominal pain and dysuria. His past medical history was pertinent for ulcerative colitis and type 2 diabetes mellitus. He had been hospitalized multiple times in the past year for abdominal pain. Extensive investigation had shown partial small bowel obstruction caused by intussusception that was treated conservatively, in addition to chronic elevation of total bilirubin, alkaline phosphatase, and transaminases attributed to possible recurrence of PSC in the hepatic graft. At the time of suspicion of possible PSC recurrence, his prednisone dosage had been increased and later tapered when liver function studies improved. He had last been admitted to the hospital 2 weeks earlier for diarrhea and fever, diagnosed with Clostridium difficile infection (CDI), and discharged 10 days prior with oral vancomycin. At the time of current presentation, his immunosuppression regimen included tacrolimus 1 mg twice daily, mycophenolate mofetil 500 mg twice daily, and prednisone 5 mg daily. He reported temporal insertion of indwelling urinary catheters during some of his previous hospital admissions for management of small bowel obstruction. Otherwise, he denied any intravenous (IV) drug use, history of urethral instrumentation, or chronic indwelling urinary catheters. His vital signs on admission revealed blood pressure 134/68 mmHg, heart rate 80 beats per minute, 20 respirations per minute, and temperature 98.5°F. On physical exam, his abdomen was soft with mild tenderness to palpation in the left lower quadrant. Rectal exam revealed a normal-sized, painless prostate with fluctuation at the right base. Laboratory investigation showed white blood cell count of 22,600/μL (94% neutrophils, 2% lymphocytes, 1% bands), hemoglobin 12.3 g/dL, and platelet count 309,000/μL. Biochemistry tests revealed creatinine 1.2 mg/dL, alanine aminotransferase 199 U/L, aspartate aminotransferase 157 U/L, alkaline phosphatase 719 U/L, and total bilirubin 18.3 mg/dL (direct bilirubin 15.6 mg/dL), which were unchanged compared to previous laboratory results. Human immunodeficiency virus (HIV) antibody was negative. Urinalysis showed 11–20 white blood cells/high power field (HPF), 0–2 red blood cells/HPF, few bacteria, and no casts. Contrast-enhanced computed tomography (CT) of the abdomen and pelvis revealed multiple stable areas of intussusception and a 3.0 cm × 1.9 cm prostatic abscess (Figures and ). We empirically treated for infection with IV ceftriaxone and vancomycin. MRSA was isolated from both urine (<10,000 CFU/mL) and 2 sets of blood cultures (aerobic and anaerobic bottles) obtained upon admission. Cultures were sensitive to daptomycin, clindamycin, gentamycin, rifampin, tetracycline, levofloxacin, trimethoprim-sulfamethoxazole, and vancomycin (MIC < 0.5). Both transthoracic and transesophageal echocardiography were negative for vegetations. Based on his recent hospitalizations, he was diagnosed with health-care associated MRSA bacteremia. Detailed review of his hospital records revealed that colonization with MRSA had been detected 2 weeks prior in a nasal swab sample and treated accordingly with topical chlorhexidine and mupirocin. A final diagnosis of MRSA prostatic abscess with bacteremia was made. Due to his immunosuppression and bacteremia, it was decided that he was not a candidate for oral antibiotics. IV vancomycin was continued while ceftriaxone was stopped. We opted for conservative management in this case, which showed rapid improvement of symptoms and inflammatory response. Therefore, abscess aspiration was not deemed necessary. We also continued oral vancomycin to complete 14 days of treatment for CDI. The same dosage of immunosuppressant medications was continued, with the exception of tacrolimus, which was held due to acute kidney injury. Leukocytosis resolved at day 3 while symptoms started to improve at day 5. Repeat blood cultures at day 4 were negative. Urine cultures failed to grow any organism at day 11 of active therapy. His creatinine rose to 1.6 mg/dL and IV vancomycin was switched to daptomycin on day 12 of treatment. He was discharged from the hospital at day 22, with instructions to continue daptomycin for a total of 6 weeks of antibiotics. During a follow-up clinic appointment at 8 weeks after completion of antibiotics, his symptoms were resolved, cultures were negative, and repeat CT of the abdomen and pelvis showed complete resolution of the prostatic abscess (Figures and ). |
A 16-year-old Hispanic male with a past medical history of bronchial asthma presented with his caretaker to our institution with complaints of intermittent abdominal pain, wet cough, and poor weight gain since infancy. The patient also had associated foul-smelling, floating stool with inability to gain weight despite a good appetite. He had no history of gastrointestinal disease, multiple respiratory infections, or severe asthma attacks. He also complained of a recent development of symmetric wrinkling with fissures and swelling of his hands’ palmar skin. His caretaker explained noticing the wrinkling during infancy; however, it was not until the start of his new job at a carwash when it became more prominent. Symptoms occurred within 3-5 minutes of water exposure and resolved within minutes after terminating water contact with no residual side effects. He could not associate worsening of wrinkling with water temperature or various bodies of water. There was never itchiness, pain, or burning sensation present with wrinkling. The patient mentioned having dry skin and increased sweating in his palms since infancy but had never been diagnosed with a dermatological condition. He also denied any family member experiencing similar symptoms.\nOn the physical examination, the patient’s height and weight were below the third percentile based on age, with his body mass index (BMI) at the 23rd percentile. He also presented with neurodevelopmental delay closer to the age of 11-12 years. Upon mouth inspection, oral mucosa was dry with gingival inflammation and halitosis present. Breath sounds on bilateral lung fields were clear; yet, clubbing was appreciated bilaterally in his upper extremities. The abdomen was mildly distended with normal bowel sounds and non-tender to palpation on all quadrants. The patient was asked to wet his hands on the examination room sink for 5 minutes. Increased thickness and wrinkling with associated blanching and edema was seen on bilateral palms and interdigital webs with sparing of the dorsal surface (Figure , B). No erythema, warmth, or desquamation was appreciated. Based on his history and physical examination findings, a chest high-resolution computerized tomography was ordered, which showed mild bilateral bronchiectasis. However, no acute or infiltrative processes, such as pleural effusion or pneumonia, were appreciated. A pulmonary function test (PFT) was performed following the American Thoracic Society guidelines []. Baseline forced vital capacity (FVC) was 71% predicted, forced expired volume in 1 second (FEV1) was 55% predicted, and FEV1/FVC ratio was 78%. Post-bronchodilation with albuterol sulfate demonstrated an FVC of 69% predicted, FEV1 of 58% predicted, and FEV1/FVC ratio of 84% predicted. Additional laboratories demonstrated deficiency in all fat-soluble vitamins (ADEK). A sweat test was performed, exhibiting a concentration of 101 mEq/L (normal range: <60 mEq/L). Subsequently, a sample was sent for CFTR genetic testing, and the patient was diagnosed with two pathogenic heterozygous CF mutations in c.1519_1521del (p.Ile507del) and c.1521_1523 (p.Phe508del). Skin symptoms were attributed to CF-related aquagenic wrinkling of the palms.\nThe patient was initiated on specialized vitamins (ADEK) and mineral supplements for CF as well as elexacaftor-tezacaftor-ivacaftor (100 mg/50 mg/75 mg). Airway clearance breathing treatments were also prescribed for long-term pulmonary management. As AWP symptoms did not hinder the patient’s quality of life, he elected to use barrier protective methods at work and with water exposure in general. On his 16-week follow-up post elexacaftor-tezacaftor-ivacaftor initiation, PFTs improved to the following baseline FVC of 102% predicted, FEV1 of 102% predicted, and FEV1/FVC ratio of 101% predicted. After 24 weeks of starting treatment with elexacaftor-tezacaftor-ivacaftor, the patient reported reduced gastrointestinal symptoms and increased weight reaching the 55th percentile. Patient also began exercising and reported increased energy levels, exercise time, and endurance. During his follow-up appointment, his hands were submerged in water for 5 minutes with significantly decreased wrinkling and no fissuring present (Figure ). The patient mentioned an increase in severity of wrinkling at the beginning of treatment with gradual improvement. He was encouraged to continue with his current treatment with close follow-up to monitor for improvement or adverse effects from medication. |
In November 2008 an Italian, Caucasian, 81-year-old male, a retired mason, presented to our outpatient clinic with a 1-year history of an enlarging subcutaneous mass over the left ACJ. The patient denied any previous shoulder trauma, but complained a long history of left shoulder discomfort during daily life activities. The patient had been submitted to four ultrasound-guided aspirations of the cyst and steroid injections by an interventional radiologist. After the final aspiration the patient complained recurrence of the cyst and formation of a fistula draining a slightly viscous serous fluid at the puncture site. On physical examination the mass was 7 × 2.5 cm in size, symmetric, firm, adherent to the superior aspect of the ACJ and non-tender on palpation (). The overlying skin presented a fistula that drained clear synovial-like fluid. There was no associated lymphadenopathy. The active range of motion of the left shoulder was 160° in forward elevation, intrarotation was at the L2-L3 level, extrarotation and abduction of the arm were weaker with respect to the contralateral normal side, despite a normal range of motion.\nPlain X-ray examination of the left shoulder showed ACJ degeneration, with acromioclavicular (AC) space narrowing and osteophyte formation, proximal migration of the humeral head and osteoarthritis of the gleno-humeral joint (). Further MRI evaluation confirmed the clinical diagnosis of a complete rotator cuff tear and observed a large, well-defined subcutaneous cyst in communication with the degenerative ACJ (). Culture of the synovial-like fluid was negative and white blood cell count, erythrocyte sedimentation rate and reactive C-protein were within normal limits.\nIn view of the persistence of the cyst and the draining fistula the we decided to submit the patient to surgery, which confirmed clinical and MRI findings. The cyst and the fistula were completely excised and, given the impossibility to repair the massively torn rotator cuff, to avoid recurrence of the cyst, resection of 1 cm of the lateral clavicle was also performed (). Histological examination was suggestive of a synovial cyst.\nWithin three weeks from surgery function and range of motion of the operated shoulder returned to pre-operative levels. To date, the patient is asymptomatic and with no cyst recurrence. |
Our patient is a 54-year-old female with a past medical history of hypothyroidism and very severe obesity (BMI 48 kg/m2). She underwent laparoscopic gastric sleeve surgery in the year 2012. Results were non-satisfactory in terms of weight loss with a difference of 6 kg/m2 in BMI post-procedure. So after six years, she underwent a laparoscopic biliopancreatic diversion with a duodenal switch. She had an uneventful postoperative recovery period. An upper gastrointestinal (GI) study contrast post-procedure did not reveal any evidence of obstruction or leak. The patient was discharged home two days after the procedure. A few days later, she started experiencing three episodes of nausea with brown-colored vomitus. She was found to be septic, with a heart rate of 110 beats per minute and temperature of 100.2oF. Her white blood cells count was 12/mm3.The source of infection was presumed to be intraabdominal considering her symptoms. Computed tomography (CT) of the abdomen and pelvis showed mildly dilated proximal small bowel loops. The patient was started on empiric antibiotic therapy with ceftriaxone 1 gm intravenous (IV) daily and metronidazole 500 mg IV every eight hours. Symptoms did not improve, so she was taken back to the operating room for diagnostic laparoscopy. Partial small bowel obstruction was noted along with ischemia of a segment of the ileum that was part of the duodenoileostomy due to mesenteric dissection. She underwent an open revision of the small bowel anastomosis with resection and anastomosis for the obstruction revision of the duodenoileostomy. Her hospital stay post-surgery remained uneventful. Diet was advanced gradually throughout the hospital course and a week later, the patient was discharged home with outpatient follow-up. Three weeks after that procedure, she noticed a productive cough with thick, yellow, foul-smelling phlegm and shortness of breath. She saw her primary care physician. A chest X-ray performed showed a right lung infiltrate with a right-sided pleural effusion. She was started on treatment with augmentin 500 mg/125 mg every eight hours. Her symptoms became worse so she came to the emergency room. Her vitals showed blood pressure 129/79 mmHg, heart rate 86 beats per minute, respiratory rate 20 breaths per minute, and temperature 98.6oF. Pulse oxygen saturation was 97% on room air. Mild leukocytosis was evident (white blood cells count 11.4/mm3 with no bands or left shift). A chest CT showed loculated, right-sided hydropneumothorax with almost total collapse of the right lung (Figure ).\nThere was a fistulous connection evident, extending from the surgical anastomosis in the stomach/bowel in the right upper quadrant through the right hemidiaphragm to the right hemithorax. These CT scan findings were new as compared to a CT scan obtained for this patient six months prior to the duodenal switch when she presented to the emergency department for non-specific left-sided chest pain. To analyze the anatomy of the fistula further, an upper gastrointestinal fluoroscopic contrast study was performed that showed a large fistula from the distal stomach prior to the duodenal bulb opening to the right pleural cavity (Figure ).\nConsultations from gastroenterology and cardiothoracic surgery teams were obtained. Chest tube drains were placed with the plan of eventually performing a video-assisted thoracoscopic surgical decortication. Post-procedure CT showed patent chest tubes draining the right pleural cavity. The drained fluid was exudative in nature as per Light’s criteria (fluid lactate dehydrogenase > 12,000 u/L and total protein ratio = 0.7) and culture from the right lung empyema grew Escherichia coli, Klebsiella pneumoniae, Pseudomonas aeruginosa, and Candida albicans. An infectious disease consultation was placed at this time. The patient was started on levofloxacin 750 mg IV daily for two weeks as per the sensitivity result obtained for Escherichia coli, Klebsiella pneumoniae, and Pseudomonas aeruginosa. Micafungin 100 mg IV daily was started for the infection with Candida albicans. This was later switched to Diflucan 400 mg IV daily for a total of two weeks. Repeat cultures from the draining fluid were negative toward the end of the second week.\nFor treatment of the fistula, the patient was transferred to another facility for esophagogastroduodenoscopy (EGD) and possible clipping of the fistula due to the unavailability of that particular service in our hospital. As a result, there was a delay of 16 days from admission to the treatment of the fistula. When the EGD was performed, it showed that there was no anastomotic leak from the previous surgery. No evidence of any stricture was identified at the previous anastomosis. A small fistulous tract was noted in the distal part of the antrum likely secondary to ulcer formation that was noted in very close proximation to the fistula tract. An endoscopic clipping was performed. A post-procedure contrast study performed on the same day revealed complete closure of the fistulous opening. The patient was transferred back to our facility after the procedure. An upper GI contrast study performed two days later showed residual leakage from the distal stomach to the right upper quadrant. A repeat EGD was performed along with fulguration of a fistulous opening with argon beam coagulation and repeat orthoscopic clip application with complete obliteration of the fistula tract. This was confirmed by an upper gastrointestinal contrast study showing no persistent fistulous communication between the post-bulbar duodenum and pleural space (Figure ).\nAfter confirmation of fistula obliteration, a video-assisted thoracoscopic surgery was performed followed by the washout of the right pleural space with the placement of a right-sided chest tube. The patient was discharged home after the procedure and has been followed by multidisciplinary teams on an outpatient basis. |
An 18-year old female patient presented to our office for an evaluation of bilateral Achilles and posterior compartment pain ongoing for approximately 2 years. The pain was dull aching and made worse with exercising- mainly running. She was evaluated by another physician who recommended physical therapy, NSAIDS and night splints. While she responded modestly to physical therapy, she did not experience any relief from the splints or medication.\nOn physical examination, the patient had bilateral tenderness to the superficial posterior compartment roughly at the musculotendinous junction and muscle belly. She also had some swelling noted. On strength testing, the patient had 4/5 muscle strength with dorsiflexion. She had no loss of motion. Radiographs were obtained which showed a mild Haglund's deformity of the bilateral ankles. The patient's magnetic resonance imaging (MRI) revealed some subtalar synovitis and an os trigonum. These later pathologies were only mildly painful and did not contribute or replicate her main posterior leg pain. After reviewing the MRI, we offered the patient a pre and post exertional compartment pressure check, however she refused. Due to her lack of responsiveness to conservative therapy, we elected to perform a superficial and a deep compartment fasciotomy of her right leg and a right subtalar joint arthroscopy with extensive debridement, os trigonum resection, Haglund calcaneal exostectomy, as well as a left posterior superficial and a deep compartment release staged 6 months apart. Since, the right leg was more symptomatic, surgical treatment was first directed to this extremity. Intraoperatively, neither the gastrocnemius nor the soleus muscle showed any signs of ischemia or necrosis.\nImmediately postoperatively, a Bulky Jones dressing was applied. She was allowed to weight bear in a Controlled Ankle Motion (CAM) walker 2 weeks postoperatively. Six months postoperatively, the patient had a full resolution of her symptoms on the right. She was continued to have symptoms on the left and underwent a similar compartment release of the superficial and deep compartments. She was allowed to weight bear immediately in a CAM boot. At 4 weeks after the surgery, she discontinued the boot and was progressed in her activity level. At 6 months after the second surgery, she had 5/5 plantar flexion strength bilaterally. She is currently running with minimal pain and is able to play soccer for her collegiate intramural team. |
A 57-year-old Caucasian male was first seen by a rheumatologist in 1998 for arthralgias and myalgias of his proximal upper extremities. His medical history included obesity and hypertension. There was no family history of rheumatologic disease. He worked as a glazier, and had no substance abuse history. Physical examination showed synovitis of his metacarpophalangeal joints and wrists. Erythrocyte sedimentation rate was >120 mm/hr, and rheumatoid factor was negative. He was diagnosed with seronegative rheumatoid arthritis, and prednisone was initiated at 10 mg daily in 1999. His arthralgias and myalgias responded clinically to the prednisone.\nIn late 1999, because of new symptoms of fatigue, weight loss, and fevers, 10 mg of weekly methotrexate was added to his regimen. An infectious disease consultation was obtained to rule out infection as a cause of his symptoms, and an extensive workup was performed. A chest radiograph was normal, and computed tomography (CT) scans of the abdomen and pelvis were unremarkable. A bone marrow biopsy was normal and cultures for bacteria, mycobacteria, and fungi were negative. Multiple blood cultures for bacteria were negative. With the exception of a persistently elevated sedimentation rate of 133 mm/hr, laboratory results were normal, and no infectious etiology for his symptoms could be found. Because of a lack of response, his methotrexate was discontinued after 3 months of therapy. His weight loss and fevers stopped, and only his fatigue continued.\nIn January 2002, still on oral prednisone, he was diagnosed with degenerative arthritis of both shoulders. His prednisone was increased to 15 mg daily, and azathioprine 100 mg daily was added. In April 2002, his left acromioclavicular joint was injected with a corticosteroid for symptomatic relief. In June 2002, both acromioclavicular joints were injected with corticosteroids. In December 2002, he first noted an erythematous, warm rash that began simultaneously on both shoulders at the sites of his shoulder injections.\nIn April 2003, he transferred his rheumatological care to our institution. His immunosuppressant regimen consisted of prednisone 10 mg daily, azathioprine 100 mg daily, and hydroxychloroquine 200 mg twice daily. He complained of fevers, fatigue, worsening bilateral shoulder pain, and progressive bilateral shoulder rash. In addition, he noted new dyspnea without cough. His shoulder pain now prevented him from carrying objects and performing his job. His left shoulder rash, shown in Figure , was macular, warm, erythematous, non-tender, and blanched with pressure. At its center was a 5-centimeter crusted ulcer with modest serosanguinous drainage, overlying the acromioclavicular joint. The surrounding erythema had progressed to include his entire left breast to the level of the nipple. It extended slightly onto the left arm and upper back, and did not cross the midline. On the right shoulder, there was a 5-centimeter circular patch of mottled erythema and warmth without ulceration or drainage, overlying the right acromioclavicular joint and symmetric with the central focus of the left-sided rash. There was no appreciable lymphadenopathy. Physical examination of the shoulders was limited by pain with both active and passive range of motion in all directions. There was a palpable effusion of the left shoulder, but not the right. Physical examination of his fingers, wrists, elbows, knees, ankles, and feet was otherwise normal with no evidence of synovitis.\nThe consulting rheumatologist concurred with the original diagnosis of seronegative rheumatoid arthritis. However, due to the presence of the unexplained rash, alternative and additional diagnoses were considered, including dermatomyositis, sarcoidosis, cutaneous leukemia or lymphoma, and infection. Erythrocyte sedimentation rate was >140 mm/hr, and C-reactive protein titer was 1:16. Anti-nuclear antibodies, uric acid, rheumatoid factor, C3, and C4 were normal. White blood cell count was 13,400 cells/mm3 with a neutrophilic predominance, hemoglobin was 10.6 g/dL and platelet count was 400,000 cells/mm3. Serum albumin was 2.8 gm/dL. Serum electrolytes, renal function, liver tests, and creatine kinase were normal. A serum electrophoresis showed polyclonal hypergammaglobulinemia, and ferritin was elevated at 356 ng/ml, suggesting chronic inflammation. A chest radiograph was normal. A skin biopsy of the left shoulder cellulitis in July 2003 revealed non-caseating granulomatous inflammation consistent with sarcoidosis. Stains for fungus and acid-fast bacilli (AFB) were negative, but cultures were not performed.\nHe was diagnosed with cutaneous sarcoidosis and treated with azathioprine 100 mg daily, prednisone 40 mg daily, and rofecoxib 25 mg daily. Chest CT revealed subtle reticulonodular parenchymal changes with lower lung zone predominance. Though the CT findings were compatible with the diagnosis of sarcoidosis, the lower lobe involvement and absence of lymphadenopathy were atypical.\nBecause the clinical syndrome was not entirely consistent with sarcoidosis, repeat skin biopsy was performed in September 2003, this time with fungal and AFB cultures in addition to the routine pathology stains. Once again, pathology revealed granulomatous inflammation consistent with sarcoidosis with negative fungal and AFB stains. However, AFB cultures grew mycobacteria after only eight days. The diagnosis of MAI was made by DNA probe, and confirmed by culture. Bacterial and fungal cultures were negative. Serology for HIV was negative.\nAntimicrobial therapy consisting of azithromycin, ciprofloxacin and ethambutol was initiated in December of 2003 by an infectious diseases consultant. Azathioprine was discontinued and corticosteroids were tapered to 7.5 mg po qd, which was the lowest tolerable dose for the patient because of a return of his myalgias and arthralgias. Shoulder plain films and magnetic resonance imaging (MRI) revealed large joint effusions and evidence for avascular necrosis, greater on the left side (Figure ). Joint aspiration of the left shoulder revealed MAI by culture, and surgical drainage and debridement was performed in January 2004. Intraoperative findings included multiple rice bodies within the joint and a large cloudy effusion. Post-operatively, he was prescribed intravenous amikacin, which was discontinued after 2 weeks due to the development of renal insufficiency. In March 2004, because the cutaneous lesions over his shoulders continued to progress despite initiation of antimicrobial therapy and decrease in immunosuppression, ciprofloxacin was changed to moxifloxacin, and rifabutin was added to his regimen.\nThe patient completed 21 months of multi-drug therapy with relief but not complete resolution of his rash and shoulder pain. In September 2005, his azithromycin, moxifloxacin, ethambutol, and rifabutin were discontinued on a trial basis because of cumulative drug toxicities. Unfortunately, fevers returned and the rash over the left anterior shoulder worsened. A repeat aspiration of his left shoulder confirmed persistence of MAI septic arthritis with a sensitive isolate. A follow-up CT scan of the chest showed no progression of his previously seen nodules. Antimycobacterial therapy has been re-initiated, and he has undergone a second debridement of his left shoulder. He has since responded to continued antimicrobial therapy documented by a negative joint aspiration, although he continues to have severe arthritic pain from permanent joint damage.\nOne of two possible mechanisms probably explains the occurrence of MAI in the skin and shoulder. The first possibility is that MAI was introduced directly into the tissue at the time of injection. MAI is ubiquitous in the environment, including water sources, and can contaminate injectable solutions []. Because MAI grows slowly, a six-month lag between injection and clinical presentation, as seen in our patient, is biologically plausible. Hoffman et al estimated that 40% of the 19 patients with septic arthritis due to atypical mycobacteria in his series had had previous joint injection; however, only 1 of the injected patients had MAI []. It is not reported whether any of the 7 MAI septic arthritis patients in Kozin et al had been previously injected or aspirated []. Czachor et al reported a kidney transplant patient who developed MAI septic arthritis months after a diagnostic arthrocentesis for the diagnosis of gout []. In our patient, the temporal relationship between the shoulder injections, the subsequent appearance of simultaneous bilateral MAI skin infections at the site of the injections, and the eventual occurrence of left shoulder MAI septic arthritis, provides strong circumstantial evidence for the causative role of joint injection in this patient. Cellulitis of the overlying skin has not been described in previous reports of MAI septic arthritis [-,].\nThe second possibility is that the patient had undiagnosed disseminated MAI at the time of joint injection, and the trauma of injection caused concentration of the organisms at the injection sites. There is some evidence that this phenomenon occurs in patients with disseminated MAC. Freed et al reported a case of MAI soft tissue infection documented post-mortem at an IV injection site in an HIV- and MAI-infected patient []. Meadows et al reported an abscess due to MAI at the site of an intramuscular injection in an HIV-infected patient []. Prior to intra-articular injection, our patient had been treated with corticosteroids for approximately 3 years as well as short-term methotrexate and azathioprine. While this degree of immunosuppression makes disseminated MAI possible, there is little evidence to support the theory of pre-existing undiagnosed disseminated MAI infection in our patient, though it remains a possibility. Another possibility may be increased susceptibility to mycobacterial disease due to an underlying genetic defect in the IFN-γ response pathway []. |
A 63-year-old male, overweight, with no comorbidities presented for plaque psoriasis primarily localized to the palm and dorsal surface of the hands in May 2015 (PASI 8.5). As topical therapy was ineffective, systemic therapy with cyclosporine was administered up to the end of 2016, but discontinued due to increased systolic blood pressure in the absence of noticeable clinical improvement. In Jan 2017, the patient was administered methotrexate (10 mg/week sc followed by folic acid 24 h later).\nAfter 17 weeks on methotrexate, there was no significant clinical improvement with significant and severe involvement of the dorsal surface of the hands (). Given this, and following appropriate screening exams, the patient was given adalimumab in April 2017 (initial dose of 80 mg, followed by 40 mg every other week starting 1 week after the initial dose). After 6–7 months, gradual, clinically significant improvement in symptoms was observed, which lasted for ~1 year (parameters from routine blood analyses were always within normal ranges, although anti-adalimumab antibodies were not measured). After this time, the efficacy of adalimumab diminished. The plaques on the hands had improved, leaving areas of vitiligo on the patient’s palms, and progressive worsening of symptoms and appearance of plaques on the elbows and knees was seen (). Diagnosis of vitiligo was made based on objective examination, absence of family history and blood workup to exclude other conditions (anemia, hyperthyroidism and diabetes). All these exams were negative, reinforcing a diagnosis of vitiligo. In May 2018, adalimumab was discontinued and the patient was initiated on therapy with secukinumab (300 mg weekly for 4 weeks; 300 mg monthly thereafter). After 6 months of therapy with secukinumab, substantial clinical improvement was observed with a PASI score of 1.8. During this time, the areas of vitiligo underwent gradual repigmentation (). After 1 year of treatment with secukinumab, small plaque areas remained in the flexor site with complete remission in the extensor area along with near complete resolution of areas of depigmentation. |
A 24 year old previously healthy lady was admitted to a local hospital on third day of fever with arthralgia and myalgia without any bleeding manifestations. On examination she was hemodynamically stable with a pulse rate of 80 beats per minute and blood pressure of 110/70 mmHg. There was no postural drop in blood pressure, pleural effusions or ascites to suggest vascular leakage. She did not have symptoms or signs of neurological involvement on admission. Full blood count revealed white blood count (WBC) 3.26 × 109/L with differentials of Neutrophils 2.0 × 109/L, Lymphocytes 1.1 × 109/L and Monocytes 0.1 × 109/L. Hemoglobin (Hb) 13.2 g/dL, platelet count 80 × 109/L and hematocrit (Hct) 38 %.\nTentative diagnosis of dengue fever without fluid leakage was diagnosed as clinical features and investigations are characteristic of the infection. Fluid management and monitoring of vital parameters was started according to National Dengue management guidelines [].\nOn fifth day of fever patient developed signs of fluid leakage. Hematocrit was 46 % which is a >20 % rise from baseline value. Other signs of leakage were right sided pleural effusion and ascites which were confirmed radiologically and low albumin levels (2.4 g/dl). Critical phase monitoring and fluid management was instituted according to the guideline. Patient did not have bleeding manifestations. Thirty one hours into critical phase she complained of severe headache and difficulty in breathing. Examination revealed a drowsy patient with increased rigidity of all four limbs and sinus tachycardia. Reflexes were exaggerated in all four limbs without focal neurological signs or papilledema. No tonic–clonic movements were noted. Blood pressure remained normal. Peripheral oxygen saturation was 92 % on air. Bilateral pupil sizes were 3 mm with slow reaction to light. Possibility of intracranial hemorrhage with brainstem involvement was suspected. Endotracheal intubation and ambu ventilation was initiated as she rapidly developed respiratory depression and deterioration of Glasgow Coma Scale (GCS) to 6/15. Hemodynamic parameters were stable with a satisfactory urine output. She was transferred to a tertiary care level for urgent brain imaging and neurosurgical opinion.\nUrgent non contrast computer tomography (CT) of brain showed multiple sub arachnoid hemorrhages in right frontal, left parietal and occipital lobes. It also showed right sided sub dural hematoma and gross cerebral edema compressing bilateral lateral ventricles, third ventricle and brainstem. (As reported by consultant radiologist) (Fig. ).\nUrgent neurosurgical opinion was taken regarding further management and was decided to manage conservatively considering the critical phase of dengue fever with low platelet counts. Patient was immediately transferred to the intensive care unit for further management. As advised by the neurosurgical team patient was started on intravenous Dexamethasone 8 mg three times daily, Intravenous Vitamin K 10 mg daily, intravenous Tranexamic acid 500 mg three times daily, Phenytoin Sodium 100 mg two times daily and Folic acid 5 mg daily. Intravenous fluids were given according to the critical phase managing guidelines. Ventilatory support was continued.\nAt the time of developing cerebral hemorrhages platelet count was 40 × 109/L and coagulation profile was normal. Patient did not have signs of bleeding from other sites. Alanine Transaminase level (ALT) was 312 IU/L, Aspartate transaminase (AST) was 666 IU, Serum albumin level 2.4 g/dl, serum creatinine was 49 µmol/L. Blood picture did not show any evidence of disseminated intravascular coagulation or thrombotic thrombocytopenic purpura. Due to multiple cerebral hemorrhages it was decided to transfuse six units of platelets when the platelet count was 15 × 109 on seventh day of illness. ABCS (Acidosis, Bleeding, Calcium and Sugar) were monitored and corrected. Hematocrit dropped to 29 from 46 % despite adequate fluid replacement and there was unexplained tachycardia. 5 ml/kg of packed cells were transfused following which the hematocrit normalized. Venous blood gas analysis did not show evidence of metabolic acidosis. Serum ionized calcium was 1.12 mmol/L and it was corrected with intravenous calcium gluconate. Capillary blood sugar level was within the normal range throughout the illness.\nOn sixth day of the illness patient developed polyuria with >500 ml of urine per hour (>12 L per day) with hypernatremia. Serum sodium level was 150 mmol/L, Serum Potassium was 3.0 mmol/L. Urine specific gravity was 1.005 and urine osmolality was 153 mOsm/kg. A diagnosis of cranial diabetes insipidus was made and treated with intra nasal Desmopressin sprays following which polyuria and hyper osmolality settled. Platelet count continued to drop even after completion of critical phase reaching a lowest value of 15 × 109 on 7th day of illness.\nDespite intensive medical therapy patient’s clinical condition deteriorated with appearance of signs of brainstem herniation. Pupils were fixed dilated with no reaction to light. Patient developed per vaginal bleeding and punctures site bleeding. Blood pressure gradually dropped needing ionotropic support. Patient succumbed to illness on day eighth. |
A 60-year-old previously healthy woman was brought to our department by ambulance for sudden onset of upper abdominal pain at night. A blood test showed a low serum hemoglobin level of 10.8 g/dL, and contrast-enhanced abdominal computed tomography (CT) revealed an iso- or hyper-enhancing area mainly ranging from the dorsal aspect of the pancreatic head to the retroperitoneum around the right kidney. This site does not have contrast enhancement, and no free air or extravasation was observed inside (). Since hemoperitoneum was suspected, abdominal angiography was performed. It was difficult to insert a catheter into the celiac artery; hence, vasoconstriction at the origin of the celiac artery was suspected. Arteriography of the superior mesenteric artery revealed retrograde enhancement of the gastroduodenal and dorsal pancreatic arteries. A 7-mm saccular aneurysm was detected in the arc of Bühler (AOB) (). The aneurysm was embolized with a metal coil. The three-dimensional CT on admission marked that stenosis caused by the median arcuate ligament was detected at the origin of the celiac artery (). The patient was diagnosed as having a ruptured aneurysm in the AOB caused by median arcuate ligament syndrome (MALS). After surgery, upper abdominal pain resolved without aggravation of the anemia. Esophagogastroduodenoscopy (EGD) was performed, revealing edematous stricture that was presumably caused by compression exerted from the lateral aspect of the duodenum by a hematoma in the descending portion of the duodenum (). After 2 weeks, EGD revealed that this stricture had resolved, and the patient was discharged on hospital-day 34.\nThe causes of abdominal visceral artery aneurysms include stenosis at the origin of the celiac artery, arteriosclerosis, trauma, infection, pancreatitis, and tumor. In MALS, the median arcuate ligament compresses and constricts the celiac artery. Consequently, the intraluminal pressure differs between the superior mesenteric and celiac arteries, and compensatory blood flow increases in the AOB, which is a direct anastomotic artery between these arteries and in the pancreaticoduodenal artery. This mechanism is considered to induce an increase in arterial blood pressure, which weakens the arterial wall, where an aneurysm develops.\nSymptoms rarely manifest unless complications occur due to the rupture. Hematemesis or hemorrhagic stool and acute abdomen may develop if the rupture occurs in the gastrointestinal tract and in the abdominal cavity, respectively. If the rupture occurs into the retroperitoneal space, the affected individual usually has no symptoms other than anemia. In addition, a statistically significant correlation was found between the range of carotid artery (CA) stenosis and the presence of collateral circulation. Furthermore, ischemia as a result of mesenteric vessel narrowing by the median arcuate ligament may occur more often than indicated by clinical symptoms and described in literature. This patient developed acute abdomen and anemia, and abdominal CT revealed hematoma in the abdominal cavity and retroperitoneal space. Thus, we needed to consider this symdrome in the differential diagnosis. Computed tomography angiography (CTA), magnetic resonance angiography (MRA), Doppler ultrasound (US), and intravascular ultrasound (IVUS) are useful as diagnostic methods. Regarding treatment, with advances in intervention radiology (IVR), non-invasive transcatheter procedures have been established as safe and effective therapeutic strategies. Previous reports have raised the possibility that the development of new aneurysms is low after administration of only IVR as treatment for focal aneurysms without resection of the arcuate ligament. However, careful follow-up still appears to be necessary in consideration of the possible recurrence of aneurysms at other sites.\nIn conclusion, although such cases are rare, if a patient complains of acute upper abdominal pain and imaging shows retroperitoneal hematoma or hemoperitoneum, we should suspect this syndrome and confirm the existence of an aneurysm with angiography. |
A 58-year-old female patient was admitted to hospital on August 3, 2012 having experienced limb numbness for one month’. This study was conducted in accordance with the Declaration of Helsinki and with approval from the Ethics Committee of the Tianjin Third Central Hospital (Tianjin, China). Written informed consent was obtained from the patient.\nOne month prior to hospitalization, the patient suddenly felt numbness and pain at the fingertips of the hands accompanied by palpitations. After four days, the symptoms gradually involved the hands and feet and the patient was conscious of lower limb weakness, although this was not accompanied by posture or gait abnormalities. The patient additionally experienced upper gastrointestinal discomfort, but without nausea or vomiting, and zonesthesia from the double costal margin to the umbilical level. After two weeks the patient exhibited numbness of the face, mouth and the skin at the top of the temple. Following symptom onset, she went to the clinic of the Tianjin Dagang Oilfield Hospital (Tianjin, China), where a brain computed tomography (CT) and magnetic resonance imaging examination of the brain and cervical spinal cord showed no abnormalities. The local hospital prescribed Mecobalamine as treatment, but the symptoms continued to progress. During the illness, the patient exhibited no dry eye or dry mouth symptoms, occasionally showed changes in bowel habit (twice a day or once every two days) and exhibited a weight loss of ~6 kg compared with previously. Twenty days previously the patient had also taken a health care product of an unknown name. The patient’s blood glucose levels had increased for two years but, following diet control, her fasting and postprandial blood glucose levels could be maintained at ~7 mmol/l. The patient had no history of habitation in an epidemic or rural environment and no history of smoking or alcohol abuse. She was unaware of any familial hereditary disease history or similar cases in her family.\nPhysical examination on admission indicated the following characteristics: clear and co-operative mentality, with normal advanced neural activity; prefrontal and bilateral facial pain and a loss of heat sensation; no atrophy in the limb muscles; normal muscle tension; muscle strength grade V; positive tendon reflex of the upper limbs, with no tendon reflex of the lower limbs; negative bilateral Hoffmann reflex, with the bilateral Babinski reflex not being elicited; normal gait and no ataxia. The patient experienced glove- and stocking-type sensations in her hands and feet and a loss of pain and temperature sensation. The patient’s diapason vibration sensation and joint position sense were normal. In addition, her discriminative touch sense was regular, and the internal medical examination revealed no abnormalities. A routine blood test was conducted following admission, as well as tests for liver and kidney function, five types of hepatitis B, syphilis, human immunodeficiency virus, thyroid function, tumor markers, immune components, vitamin B12, folic acid, fasting blood glucose, mercury, lead, manganese, chromium and other toxins. All results were normal. A gastroscopy, chest CT, abdominal B-ultrasound, echocardiography and thyroid B-ultrasound showed no evident abnormalities. On the second day after admission, the electrophysiological examination was performed. The results revealed that the bilateral median, ulnar, right posterior tibial and peroneal nerves exhibited prolonged distal motor latency, the amplitude was reduced, the proximal amplitude was reduced with a normal speed, and the distal sensory nerve did not elicit a positive waveform. The results of the needle electromyography of the abductor digiti minimi and anterior tibial muscles were normal. Following stimulation of the bilateral median, ulnar and posterior tibial nerves there was a normal F wave, and following stimulation of the bilateral posterior tibial nerve there was no H reflex. In addition, the blink reflex showed prolongation of the ipsilateral R1 and R2 and contralateral R2 latency. Finally, the facial nerve motor conduction was normal, suggesting that the damage may have been to the trigeminal primary afferent (– and ). Examination of the cerebrospinal fluid showed the number of cells and glucose and chloride levels to be normal, while the protein levels were increased to 131.7 mg/dl and the oligoclonal band was negative. Following admission, the patient was diagnosed with sensory GBS, and was administered γ globulin at a dosage of 400 mg/kg/day, intravenous immunoglobulin, for five consecutive days, and heteropathy with vitamins B1 and B12, and neurotropin. On August 16 (six weeks after the symptom onset), the review of the neurophysiology showed that the peripheral nerve motor conduction amplitude had recovered, and there were no clear changes in the motor distal latency and sensory conduction results. The needle electromyography results of the right abductor digiti minimi and anterior tibial muscles showed much denervation potential. The blink reflex was significantly improved (– and ). The review of the lumbar puncture showed protein levels to be 96.8 mg/dl on August 17. On August 18, the clinical symptoms had completely remitted, and the patient was discharged. Ten weeks after the onset of symptoms, the review of the neurophysiological results showed that the amplitude of the peripheral nerve motor conduction had further recovered and the distal latency had improved. The sensory conduction results had not changed significantly. The abductor digiti minimi and anterior tibial muscles needle electromyography results returned to normal, and the blink reflex was approximately normal (– and ). |
A 36-year-old Brazilian male patient was admitted to the hospital with a palpable lump in his right breast, located at the junction of the upper quadrants of the right breast (Fig. ). On physical examination, the lesion appeared firm with irregular margins. Axillary lymphadenopathy was negative and there were no palpable supraclavicular nodes. On breast imaging, ultrasonography showed a hypoechoic mass with partially defined contours measuring 4.0 × 3.0 cm, located at the upper region of the right pectoralis major muscle at the 12 o’clock position with muscle infiltration (Fig. ). Histological examination of core biopsy samples revealed a malignant tumor. Preoperative exams, such as X-rays and chest CT scan, abdominal US did not show any signs of disease. Radical mastectomy was then performed, due to pectoralis major muscle infiltration, consisting in removal of the breast along with the major and minor pectoralis muscles. Biopsy of the sentinel lymph node was performed. Gross examination revealed a solid tumor measuring 3.7 × 3.5 cm with a yellowish-tan cut surface and local foci of hemorrhage. Histopathology showed intravascular papillary proliferation of endothelial cells, spindle cell areas and necrosis, atypia and prominent mitotic figures, consistent with the diagnosis of high-grade angiosarcoma with areas of infiltration of the pectoralis major muscle (HE staining, magnification of 400×) (Fig. ). Histopathology also demonstrated a surgical specimen with clear margins, absence of angiolymphatic and perineural invasion, in addition to sentinel lymph node free of metastasis. Immunohistochemical study revealed a tumor positive for CD31 marker (Fig. ), confirming the vascular nature of the tumor. At the two-week follow-up of the surgical procedure, adequate wound healing was observed, without any evidence of the disease. The patient was transferred to the clinical oncology department, where he presented with severe headache and seizures after the second cycle of adjuvant chemotherapy with paclitaxel. Magnetic resonance imaging of the brain was ordered, revealing a right frontal parasagittal lesion, measuring 1.3 × 1.1 cm with a hemorrhagic component and perilesional edema, suggestive of brain metastasis. The disease progressed rapidly, culminating in the patient’s death at 20 days after the onset of neurological symptoms. |
Our patient was an 83-year-old African-American man, who was referred to our institution after originally presenting with difficult Foley placement at a local hospital. His medical history was relevant for benign prostatic hyperplasia, chronic renal insufficiency, arthritis and hypertension. His social history included tobacco use in the remote past. His vital signs and results of a review of systems were unremarkable. Results of a blood investigation showed a significant left shift with 82% segmented neutrophils. Urine analysis revealed cloudy urine, positive for leukocyte esterase, nitrites, small amount of blood and ketones. Microscopic examination of his urine showed 10 to 20 red blood cells (RBC) per high power field (HPF) and a field full of white blood cells (WBC) and bacteria. Our patient underwent cytoscopy after blood oozed out during initial catheter insertion. A complete investigation for hematuria including a computed tomography (CT) scan was performed that revealed a bladder mass. A TUR was undertaken, and histopathology confirmed the mass as being a high-grade bladder carcinoma. A follow-up metastatic investigation including a CT scan of the chest, abdomen, pelvis and bone were negative. Our patient developed increasing pelvic pain and significant hematuria. A radical cystoprostatectomy was subsequently performed revealing a multi-centric, ill-defined urothelial carcinoma (9 × 7 cm) infiltrating the right pericystic soft tissue and encompassing the right ureteral orifice. Our patient tolerated the surgical procedure well. His post-operative course was complicated by right deep venous thrombosis occluding the right common femoral vein. A histopathological examination of the bladder tumor revealed a high-grade papillary urothelial carcinoma (Figure ) completely involving the dome and posterior wall, and partially involving the anterior and right lateral walls. The tumor extended into the perivascular soft tissue and metastatic urothelial carcinoma was identified in a left pelvic lymph node (staging: pT3; pN2; pMx). Additionally, histopathological examination of the prostate revealed a bilateral, multi-centric adenocarcinoma, Gleason 4 + 4 (Figure ) with perineural and lymphatic/vascular invasion. Metastatic prostatic adenocarcinoma was also identified involving the left and right pelvic lymph nodes (staging: pT3a; pN1; pMx). Interestingly, the left pelvic lymph node revealed a focus of both metastatic urothelial and prostatic carcinomas (Figures , ). The presence of two tumor types colliding in the same lymph node was confirmed using immunohistochemical stains, including CK7 and CK20, pan-CK and PSA. Additionally, both the primary tumors were stained with the same panel as an internal control. The focus of metastatic urothelial carcinoma was positive for CK7 (Figure ), pan-CK, and negative for CK20, while prostatic carcinoma was negative for CK7 (Figure ), CK20, and positive for pan-CK. In addition, the metastatic urothelial carcinoma stained negative for PSA (Figure ), while the prostatic carcinoma was positive (Figure ). |
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