Cases reported "Cardiomyopathies"

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1/227. Evidence of cell-mediated cardiac myocyte injury involved in the heart failure of a patient with progressive systemic sclerosis.

    A 54-year-old woman with progressive systemic sclerosis (PSS) was admitted to hospital because of dyspnea and chest pain. Echocardiogram revealed diffuse hypokinesis of the left ventricle (ejection fraction 24%). methylprednisolone, heparin, and diuretics were administered, without benefit. anemia, thrombocytopenia, and renal dysfunction rapidly progressed, and she died of heart failure on the 14th hospital day. Immunohistochemical study of the myocardial tissue showed mild to moderate cell infiltration, mainly consisting of natural killer (NK) cells, macrophages, cytotoxic T lymphocytes (CTLs), and T helper cells. perforin, a cytolytic factor, was expressed in the infiltrating CTLs and NK cells, indicating that these cells were activated killer cells. Furthermore, human leukocyte antigen classes I and II, intercellular adhesion molecule-1, as well as costimulatory molecules B7-1, B7-2, and CD40, all of which are known not to be expressed in cardiac myocytes under normal conditions, were moderately to strongly expressed in cardiac myocytes. There was no detectable level of enterovirus genomes in the polymerase chain reaction products from the myocardial tissue of this patient. These findings strongly suggest that the infiltrating killer cells recognized cardiac myocytes as target cells and directly damaged them by releasing perforin. Enhanced expression of these antigens may have played an important role in the activation and cytotoxicity of the infiltrating killer cells. Absence of enterovirus genomes in the myocardial tissue may suggest that this autoimmune process is primarily induced by PSS.
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2/227. Sudden death in right ventricular dysplasia with minimal gross abnormalities.

    Arrhythmogenic right ventricular cardiomyopathy is emerging as a relatively common cause of exercise-induced sudden death in the young. The diagnostic criteria at autopsy are, however, not fully established, leading to both over- and underdiagnosis. We report a young man and a young woman dying suddenly of right ventricular dysplasia during exercise, in whom the gross autopsy findings in the right ventricle were minimal or even absent. However, the histologic features in both right and left ventricles were typical of the disease, and consisted of fibrofatty infiltrates with typical myocyte degeneration of the right ventricle and subepicardial regions of the left ventricle. These cases illustrate that microscopic findings are diagnostic and may be present in the absence of gross findings. Marked fat replacement is not essential for the diagnosis of right ventricular dysplasia, and the right ventricle should be extensively sampled histologically in all cases of sudden unexpected death, especially those that are exercise related.
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3/227. Severe cardiac dysrhythmia in patients using bromocriptine postpartum.

    Used worldwide since 1980 for the prevention of breast engorgement in the puerperium, in 1994 bromocriptine mesylate was withdrawn from the American market as an agent suitable for ablactation. The relevant recommendation of the food and Drug Administration rested on case reports that described severe vasospastic reactions among users of the drug. Some patients so affected suffered stroke, intracranial bleeding, cerebral edema, convulsions, myocardial infarction, and puerperal psychosis. More recently, it has been suggested that the side effects of the drug may also include circulatory collapse secondary to cardiac dysrhythmia. This report describes two additional cases in this category. The antepartum clinical evaluation of these women suggested that they were predisposed to arrhythmias.
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4/227. Severe hypertensive sequelae in a child with Seckel syndrome (bird-like dwarfism).

    We report a 19-year-old male with Seckel syndrome (bird-like dwarfism) who presents with malignant hypertension associated with hypertensive nephrosclerosis, dilated cardiomyopathy, and a ruptured cerebral artery aneurysm. Although end-organ injury due to chronic hypertension occurs frequently in adults, no previous reports of renal insufficiency due to hypertension exist in children or adolescents. We speculate that this patient may have been particularly prone to hypertensive end-organ injury due to his extreme short stature.
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5/227. Hereditary cardiac amyloidosis associated with the transthyretin Ile122 mutation in a white man.

    An 83 year old white man with atrial fibrillation was admitted to hospital after a cerebral infarct. echocardiography was characteristic of cardiac amyloid deposition and subsequent tests confirmed amyloidosis of transthyretin (TTR) type, in association with the Ile122 mutation of the TTR gene; this has only been reported previously in african americans in whom it occurs with an allele frequency of 2%. Haplotype analysis did not suggest a different founder than for the African Ile122 mutation. Cardiac amyloidosis should be considered among elderly patients presenting with cardiac failure and/or arrhythmia, particularly if they are resistant to conventional treatment; if confirmed, it should be followed by precise characterisation of amyloid fibril type. The prevalence of autosomal dominant cardiac TTR amyloidosis in elderly white people is unknown but early diagnosis and supportive treatment may prevent complications among affected family members.
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6/227. Mobile intracardiac calcinosis: a new risk of thromboembolism in patients with haemodialysed end stage renal disease.

    Cardiac calcinosis is a common complication of end stage renal disease. A newly observed risk of thromboembolism is reported in four patients with mobile cardiac calcinosis, treated with long term dialysis. Rapidly growing mobile calcification was confirmed by echocardiography. Each patient had an imbalance in serum calcium x inorganic phosphate (Ca x P product >/= 50); this imbalance could not be treated due to the sudden death of the patient or the need for surgical resection to prevent recurrent cerebral thromboembolism. Histological examination revealed intracardiac calcinosis in three cases, and each case showed haemodialysis hypoparathyroidism (intact PTH < 160 pg/ml). thromboembolism in such cases is rare, however it indicates a need for cautious echocardiographic monitoring in end stage renal disease in patients with an uncontrolled Ca x P product.
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7/227. Cardiopulmonary manifestations of Henoch-Schonlein purpura.

    Henoch-Schonlein purpura (HSP) is usually a mild condition involving the skin, gut, joints, and kidneys and has a good prognosis. We present a 63-year-old Hispanic man who had an unusually severe form of HSP with a fatal outcome attributable to vasculitis causing myocardial necrosis. There is only one citation in the literature of HSP-related myocardial vasculitis, which involved the right ventricle and was successfully treated with steroids. Our patient had severe HSP-related myocardial necrosis, tracheobronchitis, and nephritis. The bronchial lesions resolved, presumably because of steroid therapy. This probably is the first case of fatal myocardial necrosis related to HSP. We conclude that HSP can, in some cases, have an aggressive course. It becomes imperative to recognize the involvement of the other organ systems, such as the heart, so that appropriate therapy may be initiated. immunosuppression may have a beneficial effect on extrarenal lesions. Controlled clinical trials are needed to establish the efficacy of such treatment.
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8/227. Calcified left ventricular mass: unusual clinical, echocardiographic, and computed tomographic findings of primary cardiac osteosarcoma.

    Primary cardiac osteosarcomas are rare and usually originate in the left atrium. In contrast, osteosarcomas metastatic to the heart most commonly involve the right cardiac chambers. This case report describes an unusual primary cardiac osteosarcoma, initially observed as a slowly growing, densely calcified mass of the left ventricle with subsequent secondary pulmonary metastasis. Although cardiac tumors may be asymptomatic, this patient had recurrent bouts of ventricular tachyarrhythmia. We describe the clinical, echocardiographic, and radiological observations spanning 6 years and the gross and microscopic features at autopsy.
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9/227. Left ventricular calcification following resuscitation.

    In this paper we report a case of 34-year-old man with a severe septic shock. Because of profound hypotension he was given massive amounts of catecholamines for 10 days. After a short recovery the function of his heart started to deteriorate again and clear calcification around the left ventricle was disclosed by computer tomography. catecholamines are known to induce myocardial injury resulting in a special form of cardiomyopathy with eventual calcification, but there are no previous reports of myocardial calcification to this extent.
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10/227. Demonstration of transient entrainment in monomorphic sustained ventricular tachycardia associated with cardiac sarcoidosis.

    A 49-year-old man was referred for further treatment of sustained monomorphic ventricular tachycardia (VT) associated with cardiac sarcoidosis. During an electrophysiologic study (EP), dl-sotalol suppressed the spontaneous VT and prevented induction of VT. However, when predonisolone treatment was started, monomorphic VT recurred frequently. To terminate the VT, a temporal pacing lead was placed at the apex of the right ventricle, and programmed electrical stimulation was attempted from the lead. During the EP study, 2 different monomorphic VTs were repetitively induced and both types were able to be terminated by rapid ventricular pacing; in one of the VT morphologies, constant and progressive fusion was obvious during the ventricular pacing. Some monomorphic VTs associated with cardiac sarcoidosis are due to reentry with an excitable gap, but the clinical efficacy of EP-guided antiarrhythmic drug treatment seems to be less certain during steroid therapy. In the present case, a defibrillator device was implanted to prevent a possible arrhythmic event.
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