Cases reported "Disease Progression"

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11/53. Echocardiographical demonstration of a progressively expanding left ventricular aneurysm preceded by endomyocardial tearing.

    A 70-year-old woman with acute myocardial infarction (AMI) had a narrow necked left ventricular (LV) aneurysm and pericardial effusion. Although there had been no obvious sign of pseudoaneurysm at the first operation on the 13th day after onset, LV volume increased so dramatically that dyspnea on mild exertion was induced only 2 months after the onset of AMI. She underwent Dor's operation for the expanded LV aneurysm. The histological findings of the resected tissue, which were fibrotic epicardial lesion with small myocyte islands, indicated a true aneurysm. The ultrasound manifestation of a narrow necked aneurysm with abrupt thinning of the myocardium at the hinge point may be a valuable predictor of free wall rupture in the early phase and severely progressive LV remodeling in the late phase. Such aneurysms need to be considered as high risk.
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12/53. A unique case of the rapid onset of a large cystic hygroma in the adult.

    Cystic hygroma is an uncommon lymphatic tumor seen rarely in adults, with less than 100 cases reported in the literature. The etiology and pathophysiology of this lesion is still in question. The majority of cystic hygromas occur in the head and neck, particularly in the posterior triangle. Although cystic hygromas tend to enlarge progressively over a span of weeks or months, relatively rapid enlargement over a span of days has been described. We present the unique case of an adult woman who experienced sudden onset of a large cystic hygroma in the neck without history of antecedent swelling, infection, or trauma. Successful surgical removal of the hygroma was performed. A brief review of the literature is presented.
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13/53. Severe osteopenia in a young boy with Kostmann's congenital neutropenia treated with granulocyte colony-stimulating factor: suggested therapeutic approach.

    Kostmann's syndrome is a congenital disorder that causes an impairment of myeloid differentiation in the bone marrow characterized by severe neutropenia, which can be treated with recombinant human granulocyte colony-stimulating factor (G-CSF). We present the case of a 13-year-old boy with Kostmann's syndrome who was treated with recombinant human G-CSF from age 3.5 years. His growth and development was normal, although complicated by intermittent infections. Bone mineral density (BMD) measurement revealed severe osteopenia at the spine and hips (lumbar spine BMD 0.486 g/cm(2); Z score -3.6), and he was referred to the Endocrine Service. Relevant laboratory evaluation showed a pretreatment ionized calcium level at the upper limit of normal (1.28 mmol/L; range: 1.13-1.32 mmol/L), suppressed intact parathyroid hormone (iPTH) level (12 pg/mL; range: 10-65 pg/mL), and a low 1,25-dihydroxy vitamin d level (21 pg/mL; range: 24-65 pg/mL). He had evidence of increased bone turnover evidenced by elevated urinary deoxypyridinoline (DPD) cross-links (46.9 nmol/mmol creatinine; range: 2-34 nmol/mmol creatinine) and a simultaneous increase in markers of bone formation with elevated osteocalcin level (200 ng/mL; normal: 20-80 ng/mL) and alkaline phosphatase level (236 IU/mL; normal: 38-126 IU/mL). Because of clinical concern for his skeletal health, bisphosphonate therapy with intravenous pamidronate was initiated. One month after treatment, the iPTH and DPD cross-links were in the normal range (54 pg/mL and 17.7 nmol/mmol creatinine, respectively) and the 1,25-dihydroxy vitamin d level was elevated (111 pg/mL). Four months after treatment, there was a striking increase in BMD at the lumbar spine ( 30.86%), femoral necks (left, 20.02%; right, 17.98%), and total hips (left, 18.40%; right, 15.94%). Seven months after bisphosphonate therapy, his biochemical parameters showed a return toward pretreatment levels with increasing urinary DPD cross-links (28.7 nmol/mmol creatinine) and decreasing iPTH (26 pg/mL). However, the BMD continued to increase (8 months posttreatment), but the magnitude of the increment was attenuated (lumbar-spine, 4.8%; left total hip, 1.2% and right total hip 2.4%), relative to BMD at 4 months. Eight months after the initial treatment, his iPTH was suppressed at 14 pg/mL and he again received pamidronate (at a lower dose); 3 months later, he had an additional increase in BMD (lumbar spine 7.4%, left total hip 3.9%, right total hip 2.7%), relative to the previous study. We hypothesize that prolonged administration of G-CSF as treatment for Kostmann's syndrome is associated with increased bone resorption, mediated by osteoclast activation and leading to bone loss. In children, the resulting osteopenia can be successfully managed with antisreorptive bisphosphonate therapy with significant improvement in bone density. Measurements of biochemical parameters of bone turnover can be used to monitor the magnitude and duration of the therapeutic response and the need for BMD reassessment and, perhaps, retreatment.
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14/53. Malignant transformation of multiple dermal cylindromas.

    Cylindromas are benign tumours arising as small, solitary, slow-growing nodules on the head and neck. Multiple cylindromas may form a 'turban tumour' in the autosomal dominant Brooke-Spiegler syndrome. We report two unusual cases of multiple cylindromas with transformation into cylindrocarcinomas. The first patient, a 63-year-old white woman, developed a cylindrocarcinoma on pre-existing multiple cylindromas on her right shoulder. Eight months after resection she developed a lymph node metastasis in the right axilla. The second patient, a 68-year-old white woman, presented with multiple cylindromas of the scalp. One of these transformed into a cylindrocarcinoma, infiltrating the dura mater, with local recurrence 2 years after incomplete resection and postoperative radiation.
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15/53. Metastatic testicular seminoma--a case report.

    We present the case of a 42-year-old male who presented with a hot, tender swelling in the left supraclavicular fossa. He was pyrexial on presentation with a mildly elevated leucocyte count of 12.4x10(9)/l. Clinical examination, including full ear, nose and throat assessment, proved unremarkable. The medical history revealed that 2 years earlier the patient had been diagnosed with a testicular seminoma for which he underwent a right inguinal orchidectomy and abdominal radiotherapy. CT scan highlighted a 6 cm para-laryngeal mass, of mixed attenuation, with an adjacent region of inflammation. overall appearance was suggestive of an infective mass. Ultrasound-guided fine needle aspiration cytology revealed a metastatic seminomatous deposit. Imaging of the chest and abdomen revealed this as the only site of metastasis. He is currently undergoing chemotherapy, and is responding well. We review the pathology of testicular seminoma. This case highlights the myriad of pathologies that may present as a lump in the neck.
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16/53. The evolution of lemierre syndrome: report of 2 cases and review of the literature.

    lemierre syndrome (postanginal septicemia) is caused by an acute oropharyngeal infection with secondary septic thrombophlebitis of the internal jugular vein and frequent metastatic infections. A high degree of clinical suspicion is necessary for diagnosis. fusobacterium necrophorum is the usual etiologic agent. The disease progresses in several steps. The first stage is the primary infection, which is usually a pharyngitis (87.1% of cases). This is followed by local invasion of the lateral pharyngeal space and IJV septic thrombophlebitis (documented in 71.5% of cases), and finally, the occurrence of metastatic complications (present in 90% of cases at the time of diagnosis). A sore throat is the most common symptom during the primary infection (82.5% of cases). During invasion of the lateral pharyngeal space and IJV septic thrombophlebitis, a swollen and/or tender neck is the most common finding (52.2% of patients) and should be considered a red flag in patients with current or recent pharyngitis. The most common site of metastatic infection is the lungs (79.8% of cases). In contrast to the preantibiotic era, cavitating pneumonia and septic arthritis are now uncommon. Most patients (82.5%) had fever at some stage during the course of the disease. Gastrointestinal complaints such as abdominal pain, nausea, and vomiting were common (49.5% of cases). An elevated white blood cell count occurred in 75.2% of cases. hyperbilirubinemia with slight elevation of liver enzyme levels occurred in one-third of patients, but frank jaundice was uncommon, in contrast to its high frequency reported in the preantibiotic era. We conclude that, most likely as a consequence of widespread antibiotic use for pharyngeal infections, the typical course of the disease has changed since Lemierre's original description. The typical triad in our series was: pharyngitis, a tender/swollen neck, and noncavitating pulmonary infiltrates. The previous classical description of severe sepsis with cavitating pneumonia and septic arthritis was not commonly seen in our review. mortality was low in our series (6.4%), but significant morbidity occurred, which was likely preventable by early diagnosis and treatment. The pathophysiology, natural history, diagnostic methods for internal jugular vein thrombosis, and management are discussed.
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17/53. The nested variant of urothelial carcinoma: a rare but important bladder neoplasm with aggressive behavior. Three case reports and a review of the literature.

    OBJECTIVE: To describe our experience with the nested variant of urothelial carcinoma (UC-NV) of the bladder, by characterization of the clinical picture and the prognostic implications of this rare form of bladder neoplasm. MATERIALS AND methods: Three cases of UC-NV of the bladder treated in our institutions were revised and data compared with previously published case-reports. RESULTS: Three patients presented with advanced muscle-invasive UC-NV, of which two had lymph node metastasis at cystoprostatectomy. The histopathology in the latter two cases showed the same picture in the lymph node metastasis as in the primary tumor with nests of tumor cells with mild-moderate atypia. In all three cases the tumor involved a ureteric orifice or the bladder neck. CONCLUSION: UC-NV is a rare but important histopathologic entity. It has a poor prognosis. At early stage, tumors might be difficult to differentiate from benign conditions and awareness of the condition is of outermost importance.
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18/53. Anticoagulation and spontaneous retropharyngeal hematoma.

    A 57-year-old man treated with Coumadin for an episode of paroxysmal atrial fibrillation presented to the Emergency Department with progressive dysphagia and odynophagia. physical examination revealed a large right-sided lateral and posterior pharyngeal wall mass. Laboratory studies were significant for an international normalized ratio (INR) of 5.4 with a white blood cell count of 11,600/muL and a hematocrit of 33.2%. A lateral soft tissue radiograph and computed tomography (CT) scan of the neck demonstrated a retropharyngeal hematoma compromising the airway. Rapid reversal of the coagulopathy was achieved with fresh frozen plasma and vitamin k. patients with space-occupying retropharyngeal masses present a significant management dilemma. The choice between observation, intubation, or a surgical airway intervention must rely upon knowledge of the natural course and high rate of airway occlusion when faced with a patient who presents with a retropharyngeal hematoma. Once symptoms of airway compromise manifest, the airway must be expeditiously secured before complete obstruction occurs. This diagnosis should be sought when an anticoagulated patient presents with upper airway symptoms.
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19/53. Central poorly differentiated adenocarcinoma of the maxilla: report of a case.

    Central adenocarcinoma of the jaws is an extremely rare malignant tumor. We reported a case of poorly differentiated adenocarcinoma occurring intraosseously in the maxilla. A 62-year-old male was referred to our hospital because of swelling of the palate. MRI showed a central tumorous lesion in the maxilla. He underwent maxillectomy combined with neck and parapharyngeal dissection. Histologic examinations of the surgical specimen revealed poorly differentiated adenocaricinoma showing a positive reaction for PAS, CEA and cytokeratin. He underwent adjuvant chemotherapy with cis-platinum diamminodichloride, 5-fluorouracil, and pirarubicin, but he died of multiple distant metastases 7 months after the surgery.
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20/53. EBER-1 positive diffuse large cell lymphoma presenting as lupus nephritis.

    Approximately one-third of membranous glomerulonephritis (MGN) cases in adults are associated with systemic diseases, including systemic lupus erythematosus (SLE) or malignancies. Malignancy-associated glomerulonephritis is rarely found in non-Hodgkin's lymphoma (NHL). Epstein-Barr virus (EBV) has been postulated to contribute to the pathogenesis of both SLE and NHL. We described a 37-year-old woman with nephrotic syndrome who presented with clinical features of SLE and renal-biopsy revealed lupus MGN. The patient also suffered from concomitant progressive lymphadenopathy and NHL (diffuse large B-cell type) was demonstrated by neck lymph node biopsy. Serologic studies demonstrated EBV infection and specific EBV antigens were present on lymph node and metastatic sites. We offer a discussion regarding the complex relationships between SLE, NHL, MGN and EBV.
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