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1/101. Gastrointestinal zygomycotic infection caused by Basidiobolus ranarum: case report and review.

    Basidiobolus species are filamentous fungi belonging to the order entomophthorales. Unlike other zygomycetes, Basidiobolus species have been mainly associated with a tropical form of subcutaneous zygomycosis in otherwise healthy individuals. Visceral disease caused by this pathogen is rare, but cases of gastrointestinal infection with Basidiobolus ranarum have been reported worldwide. In many of these reports, the inflammatory disease of the colon has been confused with Crohn's disease. We report the third case of B. ranarum gastrointestinal infection in the united states, which was initially treated as inflammatory bowel disease.
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2/101. Successful intradialytic parenteral nutrition after abdominal "Catastrophes" in chronically hemodialysed patients.

    OBJECTIVE: To assess the therapeutic contribution of intradialytic parenteral nutrition (IDPN) in four acutely ill, hypercatabolic, hemodialysed patients. All underwent major surgery, complicated by infection and malnutrition. DESIGN: A retrospective clinical study. SETTING: An in-center hemodialysis unit, at a tertiary referral hospital. patients: Patient 1: a young woman, with a good renal transplant. Developed gastric lymphoma, which required gastrectomy. After cessation of immunosuppression, "lost" her kidney and returned to hemodialysis. Received IDPN for 4 months and recovered well from severe malnourishment. Patient 2: an elderly, malnourished man, on continuous ambulatory peritoneal dialysis (CAPD). Developed biliary peritonitis and bacteremia. In a 3-month period, the patient had four operations. Maintained on IDPN for 4 months. Patient 3: a young and obese man, who suffered from life-threatening staphylococcal aureus peritonitis, resulting in widespread bowel adhesions. Underwent repeated aspirations of purulent ascites, laparoscopy, and explorative laparotomy. IDPN was administered for 4 months and stopped on the patient's request. Patient 4: a young man, who after cadaveric renal transplantation remained hospitalized for 6 months because of acute rejection and peritoneal and retroperitoneal abscesses. Had major surgery performed seven times. Received IDPN for 6 months, and is now well. RESULTS: All four patients benefited from 4 to 6 months of IDPN, as an integral part of intensive supportive and nutritional treatment. weight loss was halted, as patient appetite returned and oral nutrition became adequate. Estimated daily protein intake reached 1.2 g/kg, while caloric intake rose to nearly 30 kcal/kg/d (Table 3). Mean serum albumin levels increased from 25.5 g/L /- 0.9 g/L to 38.0 g/L /- 1.5 g/L. No adverse side effects were seen from IDPN. CONCLUSION: IDPN is a worthwhile part of treatments used in the catabolic, postoperative hemodialysed patient. It is safe and efficient when used over a 6-month period in trying to attenuate existing, or worsening malnutrition in these patients. It should be commenced at an early stage in these patients, after attempts at oral nutritional support have been deemed inadequate.
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3/101. Gastrointestinal basidiobolomycosis. Morphologic findings in a cluster of six cases.

    We describe the histopathologic features of 6 cases of gastrointestinal basidiobolomycosis examined at 4 Phoenix, AZ, area hospitals during the last 4 years. Resected stomach and intestinal specimens were characterized by marked mural thickening with fibrosis, prominent tissue eosinophil infiltration and palisading granulomatous inflammation around pale fungal hyphae. In 2 cases, there was colonic perforation. Basidiobolus ranarum hyphae (associated with spore-like spherules in 4 cases) were identified within tissue sections; the irregularly branched, thin-walled, occasionally septated hyphae were typically surrounded by a thick eosinophilic cuff (Splendore-Hoeppli phenomenon). Although the histologic features of B ranarum are well described in the skin and subcutaneous tissue, gastrointestinal involvement has presented considerable diagnostic difficulty. Before the occurrence of this cluster of cases, intra-abdominal B ranarum infection has been reported only rarely.
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4/101. Gastrointestinal mucormycosis complicated by arterio-enteric fistula in a patient with non-Hodgkin's lymphoma.

    Gastrointestinal mucormycosis is a rare, often fatal, systemic infection found predominantly in immunocompromised patients. We report a case of gastrointestinal mucormycosis in a 53-year-old female with non-Hodgkin's lymphoma. Following her first course of chemotherapy, bowel obstruction developed as a result of mucormycosis. Despite treatment with antifungal therapy, she required a laparotomy owing to severe haemorrhage caused by mucormycosal invasion of her iliac artery. With continued antifungal treatment and further chemotherapy, she ultimately underwent reversal of her Hartmann's procedure and remains disease-free.
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5/101. Acute meningococcaemia complicated by late onset gastrointestinal vasculitis.

    Extra-meningeal and non-cutaneous manifestations of meningococcal infection are well recognized but rare. Herein we report a little recognized complication of meningococcaemia, namely late-onset gastrointestinal vasculitis. In the case presented, the prostacycline analogue iloprost was used in an attempt to minimize incipient digital gangrene ina patient with evidence of protracted immunological phenomena. In addition, diclofenac was used to treat severe arthralgia. Withdrawal of iloprost on day 19 was associated with abdominal pain and peritonism. laparotomy demonstrated ischaemia and ulceration in the caecum, and histology revealed full thickness mucosal ulceration with a prominent vasculitic process. It is postulated that these findings were immunologically mediated, with possible rebound vasoconstriction following withdrawal of iloprost. It is also possible that NSAID use contributed to the findings via cyto-toxicity to mucosal cells.
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6/101. Acute ophthalmoparesis (without ataxia) associated with anti-GQ1b IgG antibody: clinical features.

    OBJECTIVE: To examine the clinical features of acute ophthalmoparesis (AO) (without ataxia) associated with anti-GQ1b immunoglobulin g (IgG) antibody. DESIGN: Retrospective observational case series. PARTICIPANTS: Twenty-one subjects with AO (without ataxia) who had anti-GQ1b IgG. methods: Clinical features of 21 subjects with AO were analyzed. RESULTS: Seventeen had symptoms of antecedent infection. Gaze limitation was bilateral in 16 subjects and unilateral in five, indicative that laterality does not always negate AO. Nine subjects showed abducens paresis, and two limitation of abduction and adduction. Eight, who initially had bilateral abducens palsy, subsequently had impairment of adduction and vertical movement. These showed that bilateral abducens palsy followed by oculomotor nerve involvement is characteristic of AO. Muscle stretch reflexes were normal in nine subjects, hypoactive in eight, absent in three, and brisk in one. Distal paresthesias were present in seven subjects. Acellular cerebrospinal fluid (CSF) associated with raised protein concentration was detected in three. CONCLUSIONS: Antecedent infectious symptoms, characteristic limitation of ocular movement, areflexia, distal paresthesias, and CSF albuminocytologic dissociation are useful markers for diagnosing AO as well as anti-GQ1b IgG. AO can be considered a mild form of miller fisher syndrome or a regional variant of guillain-barre syndrome.
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7/101. Fatal upper and lower gastrointestinal cytomegalovirus disease following autologous peripheral blood stem cell transplantation.

    Although the life-threatening cytomegalovirus (CMV) disease is a well known complication following allogeneic hematopoietic stem cell transplantation (HSCT), it has been considered infrequent after autologous peripheral blood stem cell transplantation (PBSCT). On the other hand, the massive involvement of the gastrointestinal (GI) tract as the primary site of fatal CMV disease is particularly rare after autologous PBSCT. We present the case of a woman who suffered from CMV disease after high-dose busulphan/melphalan/thiotepa (BuMelTT) and autologous PBSCT. The primary site of infection was the GI tract, which was extensively affected. During the fifth week post-transplant the patient started with epigastralgia, diarrhea, fever, GI bleeding, and thrombocytopenia, and she died on day 52. Another case of fatal CMV disease among the few patients treated with BuMelTT has been recently reported, which suggests that the immunodeficiency associated with that regimen can be as intense as that occurring after allogeneic BMT.
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8/101. Gastrointestinal histoplasmosis in patients with AIDS: case report and review.

    histoplasmosis is the most common endemic mycosis in individuals with AIDS, occurring in 2%-5% of this population. infection is more likely to be disseminated than in immunocompetent individuals and generally presents insidiously with nonspecific symptoms. The gastrointestinal tract is involved in 70%-90% of cases of disseminated histoplasmosis, yet gastrointestinal histoplasmosis per se is infrequently encountered in patients with AIDS. The diagnosis of gastrointestinal histoplasmosis is often not suspected, particularly in areas of nonendemicity, and a delay in diagnosis may lead to increased morbidity and risk of death. Since antifungal therapy improves outcome for >80% of AIDS patients with histoplasmosis, it is essential that caregivers be aware of the varied presentations of gastrointestinal histoplasmosis in order to diagnose and to treat this potentially life-threatening infection effectively.
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9/101. Neonatal diabetes mellitus, enteropathy, thrombocytopenia, and endocrinopathy: Further evidence for an X-linked lethal syndrome.

    We describe an unusual family with a fatal genetic syndrome of neonatal diabetes mellitus (DM), enteropathy, endocrinopathy, and severe infections with variable thrombocytopenia. All affected individuals are male; X-linked inheritance is likely. The most common clinical features are neonatal DM, inanition, and enteropathy; a variety of other autoimmune phenomena are less frequent. Clinical variability within and among families is common, including lack of one or more cardinal features. The syndrome is usually fatal, but survival is sometimes possible with immunosuppressive therapy. Clinical variability and frequent new mutations may contribute to poor recognition and underreporting of similar cases.
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10/101. Gastrointestinal basidiobolomycosis in arizona: clinical and epidemiological characteristics and review of the literature.

    Gastrointestinal basidiobolomycosis (GIB) is an unusual fungal infection that is rarely reported in the medical literature. From April 1994 through May 1999, 7 cases of GIB occurred in arizona, 4 from December 1998 through May 1999. We reviewed the clinical characteristics of the patients and conducted a case-control study to generate hypotheses about potential risk factors. All patients had histopathologic signs characteristic of basidiobolomycosis. Five patients were male (median age, 52 years; range, 37--59 years) and had a history of diabetes mellitus (in 3 patients), peptic ulcer disease (in 2), or pica (in 1). All patients underwent partial or complete surgical resection of the infected portions of their gastrointestinal tracts, and all received itraconazole postoperatively for a median of 10 months (range, 3--19 months). Potential risk factors included prior ranitidine use and longer residence in arizona. GIB is a newly emerging infection that causes substantial morbidity and diagnostic confusion. Further studies are needed to better define its risk factors and treatment.
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