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1/16. A case of cytophagic histiocytic panniculitis associated with exertional rhabdomyolysis.

    An 18-year-old man who suffered from panniculitis involving the entire left lower limb after exertional rhabdomyolysis is reported. A high fever (>39 degrees C) and leukocytosis (>20,000/microL) persisted for 1 week, and his general status deteriorated rapidly into pre-disseminated intravascular coagulation, complicated by pleural effusion and prolonged clotting time. His condition was dramatically improved by steroid pulse therapy and he has remained in good health for the 20 months since discharge. Histologic examination of subcutaneous tissue from the swollen left lower limb revealed pleomorphic small, medium or large lymphocytes, macrophages and neutrophils infiltrating the edematous subcutaneous adipose tissue in a lobular panniculitis-like pattern. The majority of inflammatory cells were T lymphocytes, with equal proportions of CD4 and CD8 cells. As polymerase chain reaction did not show bands suggesting T cell receptor gamma gene rearrangement, the proliferation of T lymphocytes was considered to be polyclonal. The T lymphocytes also expressed Fas ligand, suggesting the involvement of Fas-mediated cytotoxicity. This case may represent a new category of cytophagic histiocytic panniculitis induced by exertional rhabdomyolysis.
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2/16. Arthropathy, skin and bone lesions in pancreatic disease.

    A patient with a history of alcoholism and pancreatic calcification, developed subcutaneous fat necrosis and an arthropathy, associated with a pancreatic pseudocyst and accompanied by an elevation of serum lipase and amylase. The illness was complicated by bacteremia and destructive bone lesions. A clinical distinction between osteomyelitis and medullary fat necrosis proved difficult. infection of bone was demonstrated at one site but did not exclude medullary fat necrosis elsewhere.
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keywords = bone
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3/16. A case of Weber-Christian disease with collapsing glomerulopathy.

    We report a case of Weber-Christian disease confirmed by skin biopsy in a patient who presented with collapsing glomerulopathy and lipophagic interstitial nephritis. On renal biopsy, glomerular visceral epithelial cells, tubular cells, and interstitial macrophages were loaded with inclusions that were morphologically consistent with oxidized lipoproteins, suggesting that lipids derived from the panniculitis may have an etiopathogenic role.
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4/16. adult lipophagic atrophic panniculitis.

    We report two patients with chronic, recurrent, nodular panniculitis lesions that later developed areas of lipoatrophy. Histologically, there was a consistent lobular lipophagic replacement of fat cells with lipophagic giant cells. The clinical appearance was that of tender, erythematous, superficial or subcutaneous, symmetrical nodules and plaques of 1-2 weeks' duration. The lesions could occur with episodes of fever. One patient had hepatomegaly and the other had an increased sedimentation rate and leucocytosis. The histology and the clinical pattern of the panniculitus syndrome resembled those of lipophagic lipoatrophy of childhood. This is a panniculitis of unknown cause in which the principal inflammatory cell response in the subcutaneous tissue is the macrophage.
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5/16. An intracranial mass lesion in systemic xanthogranulomatosis: case report.

    The authors describe a 42-year-old woman with systemic xanthogranulomatosis and bilateral intraorbital tumors, who subsequently developed multiple lesions of the intracranial dura mater, spinal cord, retroperitoneum, pericardium, and mediastinum. Systemic xanthogranulomatosis is histologically similar to systemic Weber-Christian disease, except for the absence of subcutaneous panniculitis. Immunohistochemical studies suggest that this clinical entity can be differentiated from histiocytosis X, because foamy cells in systemic xanthogranulomatosis demonstrate macrophages but not T-zone histiocyte markers. Differentiation of this disease from other intracranial xanthogranulomas and treatment are discussed.
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6/16. Weber-Christian panniculitis with systemic cytophagic histiocytosis.

    A 49-year-old woman suffered from Weber-Christian panniculitis with a typically periodic course. Subsequently, the attacks of the disease developed to a severe state: high fever, endotoxinemia, pancytopenia, and clotting disorder, in addition to the cutaneous manifestations. One and a half years after the outbreak of the disease the patient died in a septic shock. At autopsy all three stages of Weber-Christian panniculitis were found. In addition, an immense proliferation of benign cytophagic histiocytes could be observed in the bone marrow, spleen, lymph nodes and, less distinctly, in the fatty and interstitial tissues of the visceral organs. As a nosologic entity, the Weber-Christian disease is frequently questioned. Some authors consider the lobular, histiocytic, cytophagic panniculitis a unique syndrome. On the other hand, it must be emphasized that benign, cytophagic histiocytosis may exist associated with infections or may accompany different diseases. Furthermore, the Weber-Christian panniculitis is clinically and morphologically well defined.
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7/16. Cytophagic panniculitis.

    A 60 year old woman developed generalised erythema nodosum-like lesions together with hectic fever, lymphadenopathy, and hepatitis. Biopsies revealed lobular panniculitis with a benign histiocytic infiltrate and prominent phagocytosis in subcutaneous sites, lymph nodes, and bone marrow. All immunological, serological, and culture studies were negative, apart from throat isolation of herpes simplex. The patient responded to high-dose corticosteroids. The case illustrates the differential diagnosis of lobular panniculitis, with histiocytic infiltrate and cytophagocytosis. These combined features are consistent with the recently described syndromes of cytophagic panniculitis and virus-associated hemophagocytic syndrome.
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ranking = 0.00086079231876145
keywords = bone
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8/16. Weber-Christian disease. Analysis of 15 cases and review of the literature.

    We report 15 patients encountered over 13 years who presented with inflammation of subcutaneous fat and were given clinical and pathologic diagnoses of Weber--Christian disease (WCD). Prominent clinical features included female predominance, lower extremity nodules, fevers, arthritis/arthralgias, and myalgias. Notable laboratory features were elevated erythrocyte sedimentation rate, anemia, leukopenia, and hypocomplementemia, frequently with circulating 7S IgM or immune complexes at times of active symptoms. Histologic findings were lobular--together with frequent septal--panniculitis, fat-laden macrophages, variable cellular infiltrates, necrosis, and occasional vasculitis. Follow-up revealed the death of 2 patients and disease stabilization or improvement in 13 patients. Six patients developed features of other diseases (factitial disease, erythema nodosum, acute myelogenous leukemia, rheumatoid arthritis, systemic lupus erythematosus, and sarcoid) and a seventh may have had erythema induratum. We suggest that classic WCD, as originally described, reflects an increasingly recognized spectrum of panniculitides. These are syndromes of diverse etiology that share many clinical, inflammatory, and immunologic features.
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9/16. Cytophagic histiocytic panniculitis. Systemic histiocytosis presenting as chronic, nonhealing, ulcerative skin lesions.

    Systemic histiocytosis was found in a patient who presented with chronic, nodular, and ulcerative skin lesions. The patient's hospital course was complicated by persistent fever, thrombocytopenia, severe neutropenia, and coagulation abnormalities. Treatment consisted only of splenectomy and supportive care. Postoperatively, the patient's skin lesions and the fever, pancytopenia, and coagulopathy resolved. Marked proliferation of histologically benign macrophages was observed in dermal, splenic, hepatic, and lymphoid tissues; leukophagocytosis and erythrophagocytosis were clearly demonstrated. This illness is most consistent with cytophagic histiocytic panniculitis, a newly described syndrome.
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10/16. Cytophagic histiocytic panniculitis with fever, cytopenia, liver failure, and terminal hemorrhagic diathesis.

    We have seen five adult patients with a clinical picture of recurrent histiocytic, cytophagic panniculitis, cytopenia, abnormal liver function tests, and a terminal, febrile bleeding diathesis. Originally thought to have Weber-Christian disease, these patients, we believe, represent a unique syndrome: lobular, histiocytic, cytophagic panniculitis. Erythrophagocytosis and cytophagocytosis are readily observed, but the cells do not show malignant features. histiocytosis can be found at times in the bone marrow, lymph nodes, liver, spleen, and serosal tissues, as well as in the skin and subcutaneous tissues. The terminal hemorrhage in these patients is characterized by features of pancytopenia, liver failure, and intravascular coagulation. This disease may be separated from malignant histiocytosis by the chronic course, the primary involvement of the adipose tissue, and the benign histiocytes in the infiltrate. It has some similarities to other regional histiocytoses such as sinus histiocytosis, intestinal histiocytosis, and splenic histiocytosis.
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keywords = bone
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