Cases reported "Recurrence"

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1/504. A trisomic germ cell line and precocious chromatid segregation leads to recurrent trisomy 21 conception.

    A chromosomally normal 37-year-old woman was referred for preimplantation genetic diagnosis after having several conceptuses with trisomy 21. Segregation of chromosome 21 was assessed in unfertilised meiosis II oocytes and preimplantation embryos from PGD cycles using fluorescent in situ hybridisation (FISH). Of 7 preimplantation embryos, 5 were chromosomally abnormal with 4 having trisomy 21 and one being tetraploid. Of 4 oocytes, 3 had an abnormal chromosomal constitution with either an extra chromosome 21 or an extra chromatid 21. In one oocyte an extra chromatid 21 was detected in both the metaphase II complement and the first polar body providing the first direct evidence of a maternal trisomic germ cell line. Moreover, this result shows that the extra chromosome 21 can precociously divide into its two chromatids at the first meiotic division.
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2/504. Primary subcutaneous mucormycosis (zygomycosis): a case report.

    A case of mycormycosis presenting primarily as a subcutaneous mass of the left leg in an immunocompetent individual is described. The mass that was diagnosed initially as a non-specific foreign body granulomatous process recurred a year later. Histopathological examination of the primary and recurrent lesions revealed partly degenerated hyphae associated with acute necrotizing and chronic granulomatous inflammation. Histomorphological features of primary subcutaneous mucormycosis without predisposing factors have not been previously reported.
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3/504. Acute disseminated encephalomyelitis in childhood: report of 10 cases.

    We report 10 children with the diagnosis of acute disseminated encephalomyelitis. diagnosis was based on clinical and radiologic findings, and after acute encephalitis was excluded by negative culture and antibody results. The most common presenting symptom was ataxia, followed by optic neuropathy, cranial nerve palsy, convulsions, motor dysfunction, and loss of consciousness. Brain magnetic resonance imaging showing bilateral symmetrical hyper-intense lesions of the same age in brain stem, subcortical white matter, thalamus, basal ganglia, or cerebellum was the mainstay of the diagnosis. The presence of a preceding event (either an infection or vaccination) was present in 8 of 10 patients. Brain computed tomographic scans were abnormal in 3 of 10, and electroencephalogram was normal in all patients. High-dose corticosteroids were given to six patients, one received low-dose steroids, and the other three had symptomatic follow-up. Those who relapsed were mainly from the symptomatic follow-up group. Only one patient (the youngest) receiving high-dose methylprednisolone relapsed. Therefore, early high-dose steroid treatment seems to be the most effective treatment in acute disseminated encephalomyelitis and can prevent relapses.
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4/504. T lymphocyte subsets and cytokine production by graft-infiltrating cells in FSGS recurrence post-transplantation.

    BACKGROUND: Focal segmental glomerulosclerosis (FSGS) aetiology remains undefined although a derangement of lymphocytes and monocytes macrophages, at least, has been strongly suspected. We report the graft-infiltrating phenotypes and their cytokine production in a case of FSGS recurrence post-transplantation. methods: The kidney transplant recipient suffered immediate FSGS recurrence. Aspiration biopsies were done at the first and second week post-surgery and were analysed by flow cytometry. The cytokine analysis was done on aspiration sample culture supernatants and serum by enzyme-linked immunosorbent assay. RESULTS: High expression of CD3CD69, CD3CD71 and CD4CD29 was found on infiltrating lymphocytes. biopsy cultures pointed to a Th0/Th1 pattern of cytokine production as well as significant synthesis of transforming growth factor-beta1. Interestingly, monocyte chemokines were absent. CONCLUSION: We report evidence of intragraft lymphocyte activation in the early days of FSGS recurrence. Aspiration biopsy cultures showed failure of cyclosporin A to inhibit interleukin-2 (IL-2) production by infiltrating lymphocytes. If our findings are confirmed in similar patients, a trial with anti-IL-2-receptor antibody could be warranted.
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5/504. Cryosupernatant plasma exchange in the treatment of antiphospholipid antibody syndrome with lupus nephritis.

    We report a case of a 22-year-old female with antiphospholipid antibody syndrome (APS) associated with systemic lupus erythematosus in whom cryosupernatant plasma exchange was effective and improved both the refractory venous thrombosis in her legs and relapsing thrombocytopenia. A renal biopsy specimen showed not only features of active lupus nephritis but also renal arteriolar thrombosis which is considered to be a type of thrombotic microangiopathy (TMA). Because a pathological role of unusually large von willebrand factor (vWF) multimers has been reported in patients with TMA including thrombotic thrombocytopenic purpura, plasma exchange using replacement with cryosupernatant, which is free of unusually large vWF multimers, is likely to be an option of treatment modality for patients with refractory and chronic relapsing APS manifesting TMA.
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6/504. Treatment of cutaneous sarcoidosis using phonophoresis.

    sarcoidosis is a multiple-system disorder of unknown origin characterized histologically by epithelioid granulomas with little or no necrosis. We describe a 32-year-old female patient with a history of systemic sarcoidosis and common variable immunodeficiency with recurrent, multiple, soft, erythematous and violaceous nodules on the back of her left hand. Her lesions responded to phonophoresis after unsuccessful treatment with topical and intralesional corticosteroids. However, nodules appeared on other parts of her body after phonophoresis was stopped, which suggests that phonophoresis had a localized rather than systemic effect.
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7/504. Long-term administration of cyclosporin A to HCV-antibody-positive patients with dermatologic diseases.

    BACKGROUND: cyclosporine A (CYA) is an immunosuppressive agent which is being used in the treatment of an increasingly wide range of dermatologic diseases, but its use has been avoided in carriers of hepatitis c virus (HCV). methods: We administered small doses of CYA (maximum, 3 mg/kg/day) for a long time to treat dermatologic diseases in one HCV-antibody-positive patient with no HCV-rna in the blood, one patient with a small amount of HCV-rna in the blood, and two patients with large amounts of HCV-rna in the blood. RESULTS: skin lesions improved in all patients, but recurred upon complete or partial withdrawal of CYA. In the absence of HCV-rna in the blood, or when only a small quantity of HCV-rna was present in the blood, HCV-rna load showed no apparent change. In one patient with a large blood HCV-rna load, CYA dosage reduction was followed by increases in alanine aminotransferase (ALT) levels and decreases in blood HCV-rna. Aggravation of hepatitis due to immunologic reactivation was suspected in this patient. CONCLUSIONS: The reduction of CYA dosage is a key element in the use of this agent for cutaneous diseases.
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8/504. Asymptomatic pancreatitis associated with clozapine.

    Besides the well-known adverse effects of clozapine, such as granulocytopenia, tiredness and hypersalivation, acute pancreatitis is known to be a very rare complication of the drug. In the literature a total of five case reports have been published so far. We report a case of asymptomatic pancreatitis subsequent to clozapine treatment at therapeutic doses in a 38-year-old male patient with chronic paranoid-hallucinatory schizophrenia. The patient was rehospitalized after an acute exacerbation of the psychosis subsequent to an attempt to change medication on an outpatient basis. Treatment with clozapine was initiated again. During phases of progressively increasing the clozapine dose, serum levels of amylase and lipase were increased; after maintaining daily doses of clozapine of 300 mg and/or 600 mg the pancreatic enzymes normalized quickly within a few days. The patient did not report any pancreas-related complaints, nor did specific diagnostic studies produce any indicative result, only a minor thickening of the head and body of the pancreas in the ultrasound. It is assumed that the phenomenon of subclinical, asymptomatic pancreatitis during increasing dosage of clozapine occurs more often than previously supposed. The monitoring of serum amylase levels during slow increase in clozapine is recommended; if leukocytosis or eosinophilia is present, the possibility of even a subclinical and asymptomatic pancreatitis should be considered.
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9/504. Symptoms of recurrent intrathecal baclofen withdrawal resulting from drug delivery failure: a case report.

    A 24-yr-old, completely (T8) paraplegic male patient presenting with severe spasticity had a drug administration device implanted in April 1991 for continuous intrathecal administration of baclofen. After a period of remarkable improvement in both the spasticity level and his quality of life, the patient experienced several short-lasting episodes of increased spasticity, with severe spasms. Among the possible causes of these deleterious episodes were microcrystalluria, obstipation, a decubitus ulcer, a foreign body in the buttocks, drug tolerance to baclofen, electromagnetic interference, and erroneous filling and programing of the pump. The catheter was the most common source of intrathecal baclofen withdrawal symptoms and had to be changed four times in 5 yr. Intrathecal baclofen administered through an implantable drug administration device is a highly effective but complex and expensive procedure that requires careful patient selection and close monitoring by highly qualified and well-trained health professional. Withdrawal symptoms may be related to noncompliance on the part of the patient, erroneous filling or programing of the pump, depletion of the battery, random component failure, concomitant illness, drug tolerance, or advancement of the disease itself. When failure of the device is suspected, substitution with oral baclofen is recommended until a full work-up is performed to determine the defect.
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10/504. Successful treatment of adolescent pemphigus vulgaris by immunoadsorption method.

    A 15-year-old girl with pemphigus vulgaris did not respond to oral administration of prednisolone at 45 mg/day. The skin and oral mucous membrane lesions recurred after effective treatments with methylprednisolone pulse therapy and combination therapy with prednisolone and cyclosporine. The finally successful treatment involved eleven cycles of immunoadsorption using a tryptophan column and administration of a moderate dose of prednisolone. serum gamma-globulin level and anti-intercellular antibody titer decreased from 1.08 g/dl to 0.5 g/dl and 1:320 to 1:20, respectively. She has been well controlled with 21.5 mg/day prednisolone for 8 months after the final adsorption. Considering the physical, mental and social situation of adolescent student patients, immunoadsorption is a highly preferable choice among a variety of treatment modalities for pemphigus vulgaris because it makes the term of hospitalization shorter and avoids undesirable side effects from initial high dose corticosteroids.
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