Cases reported "Granuloma"

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1/112. Intravenous injection of talc-containing drugs intended for oral use. A cause of pulmonary granulomatosis and pulmonary hypertension.

    Clinical and morphologic features are described in two patients known to have repeatedly injected intravenously talc-containing drugs intended for oral use. In one patient severe pulmonary hypertension developed; the talc granulomas in him were located predominantly within the pulmonary arteries. The second patient had normal pulmonary arterial pressures, and the talc granulomas in him were located predominantly in the pulmonary interstitium. Of 19 previously described patients with pulmonary talc granulomas, 12 had morphologic evidence of pulmonary hypertension (in three of severe degree); in each, talc granulomas were located predominantly within the pulmonary arteries. In those without signs of pulmonary hypertension, granulomas were located predominantly in the pulmonary interstitium. Why there are differences in the distribution of the talc granulomas is unclear. It is clear, however, as demonstrated by one of our patients, that severe pulmonary hypertension may be a consequence of intravenous injection of drugs intended for oral use.
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2/112. mesalamine-induced granulomatous hepatitis.

    A 42-yr-old man with ulcerative colitis was admitted for investigation of prolonged fever associated with cholestatic liver tests. Endoscopic retrograde cholangiopancreatography demonstrated a normal biliary tree, and liver biopsy showed granulomata. A clinical diagnosis of drug-induced granulomatous hepatitis was established as the symptoms disappeared after cessation of mesalamine therapy and recurred on rechallenge. Although the differential diagnosis of fever and hepatitis in patients with inflammatory bowel disease is wide, in this case mesalamine is the most likely cause.
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3/112. Erdheim Chester disease: a rare cause of knee and leg pain.

    A case of Erdheim Chester disease in a 51-year-old Turkish patient is described. Erdheim Chester disease is a rare form of lipoid granulomatosis. knee and leg pain are the most common symptoms, and physicians working in orthopaedics and traumatology are the first to be consulted. Our patient demonstrated a typical bilateral, symmetric sclerosis of the metaphyseal region of long bones of the lower extremity, histologic examination revealed foamy, lipid-loaded histiocytes. The patient also suffered from arterial hypertension, diabetes insipidus and exophthalmos of the left eye. The diagnosis was confirmed by a bone biopsy, and the patient was treated with non-steroidal anti-inflammatory drugs, corticosteroids and vincristine.
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4/112. A case of sarcoidosis and sarcoid granuloma, papillary carcinoma, and Graves' disease in the thyroid gland.

    sarcoidosis is a systemic chronic granulomatous disease of unknown etiology most commonly affecting young females. The disease was first described in the thyroid gland in 1938. Our patient, a 27-year-old male with known sarcoidosis, was referred to the National University Hospital for acute symptoms of thyrotoxicosis (weight loss of 6 kg, tremor, thyroid enlargement, and tachycardia). Laboratory findings showed suppressed serum thyrotropin (TSH, <0.03 mU/L [0.5-4.20]), increased total thyroxine (T4) (223 nmol/L, [60-140]), and triiodothyronine (T3) (8.5 nmol/L, [1.5-2.7]). Furthermore, Tc-99m pertechnetate scintigraphy disclosed diffuse accumulation of the isotope confirming the diagnosis of Graves' disease. During the next 18 months of antithyroid treatment (thiamazole, Thycapzol) hyperthyroidism was difficult to control, the thyroid gland gradually enlarged, and surgery was recommended. Initially, the patient declined surgery but after an additional 18 months, he accepted surgery. During the 36-month period of antithyroid drug treatment TSH was suppressed (<0.01 mU/L) and T3 often elevated despite high doses of thiamazole. Total thyroidectomy was performed, and histologic examination of the removed thyroid tissue confirmed the diagnosis of Graves' disease and also the presence of sarcoid granuloma and metastatic papillary adenocarcinoma with spread to neck lymph nodes. Four months later, a modified radical neck dissection was performed with removal of neck lymph nodes followed by external radiation therapy (2 Gy x 32 fractions to the neck). The concomitant presence of sarcoidosis, papillary carcinoma, and Graves' disease in a thyroid gland, to our knowledge, has not previously been described in the literature.
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5/112. Scopulariopsosis and hypersensitivity pneumonitis in an addict.

    Granulomatosis caused by fungal spores of a soil saprophyte is a newly recognized pulmonary complication of intravenous drug addiction. Brown, non-budding spores were histologically identified in necrotic tissue, inside giant cells of sarcoidlike granulomata, and in the vicinity of focal angiitic lesions. The fungus was identified by culture as the dematiaceous scopulariopsis brumptii. Cultural and histopathologic studies of lung biopsy specimens established the diagnosis. We showed precipitating antibodies to fungal antigen in the serum, prepared from the patient's isolate. Similar granulomatous pulmonary lesions were experimentally produced in mice by a single intravenous injection of spores of S. brumptii. The spores remained viable but did not show evidence of growth in the animal's tissue. Precipitating antibodies to fungal antigen and immediate wheal and late necrotizing type of skin reactions were shown in the challenged mice. The studies support the notion that pulmonary hypersensitivity to fungal spores was mediated by an Arthus'-type phenomenon.
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6/112. bone marrow granulomas possibly associated with amiodarone.

    amiodarone is a class III antiarrhythmic agent that is effective in treating different types of cardiac dysrhythmias. It was approved only for treatment of life-threatening ventricular dysrhythmias refractory to other therapy; however, its use for atrial dysrhythmias such as atrial fibrillation is well accepted. Adverse effects associated with amiodarone include pulmonary, hepatic, thyroid, ocular, and neurologic toxicities. Our patient experienced intermittent fever, night sweats, and fatigue while taking the drug for treatment of atrial fibrillation. bone marrow biopsy showed granuloma formation after 17 months of therapy with amiodarone. amiodarone was discontinued due to significant hypotension and shortness of breath. To our knowledge, this is the third case report of granuloma formation in bone marrow possibly associated with this agent.
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7/112. sarcoidosis and systemic vasculitis.

    BACKGROUND: systemic vasculitis is an unusual complication of sarcoidosis. Over a 10-year period, the authors have provided care for six patients who had features of both sarcoidosis and vasculitis. Vasculitis could not be attributed to other causes. OBJECTIVES: To report six patients (five children) who had sarcoidosis and systemic vasculitis and compare our experience with previous literature. To better delineate the clinical spectrum of sarcoid vasculitis and its response to therapy. methods: Retrospective analysis and a medline literature review of sarcoid and concurrent vasculitis from 1966. RESULTS: Our six patients had systemic illnesses that included fever, peripheral adenopathy, hilar adenopathy, rash, pulmonary parenchymal disease, musculoskeletal symptoms, and scleritis or iridocyclitis. Biopsies revealed features compatible with the diagnosis of sarcoidosis or necrotizing sarcoid granulomata in either skin, lymph node, lung, synovium, bone, bone marrow, liver, trachea, or sclera. Arteriography showed features of large vessel vasculitis in three patients, all of whom were African American, whereas patients with small vessel vasculitis were white. Prior reports of sarcoid and vasculitis included 14 adults, of whom half had predominantly small vessel disease, and half had medium- or large-sized vessel disease. Eight previously reported children included seven with primarily large vessel sarcoid vasculitis. Racial background was noted in 15 reported cases and included whites (6), african americans (5), and Asians (4). Among the authors' six patients, four improved when treated with prednisone alone. However, relapses occurred when the drug was tapered or withdrawn. CONCLUSIONS: sarcoidosis may be complicated by systemic vasculitis that can affect small- to large-caliber vessels. Sarcoid vasculitis can mimic hypersensitivity vasculitis, polyarteritis nodosa, microscopic polyangiitis, or Takayasu's arteritis. African American and Asian patients are disproportionately represented among cases with large vessel involvement. Corticosteroid and cytotoxic therapy is palliative for all forms of sarcoid vasculitis. However, relapses and morbidity from disease and treatment is common.
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8/112. Granulomatous tubulointerstitial nephritis in the renal allograft.

    Granulomatous tubulointerstitial nephritis has rarely been described in renal allografts. Of 1,574 renal allograft tissue specimens obtained from 514 patients in the period 1993 to 1998, we report three cases (0.6%) with interstitial nephritis containing multiple noncaseating granulomas. biopsy specimen 1 was obtained from a 44-year-old woman with a 6-day history of systemic candida albicans infection and showed multiple granulomas containing budding yeasts. biopsy specimen 2 was from a 33-year-old man who presented with miliary spread of mycobacterium tuberculosis 12 days before the allograft biopsy. biopsy specimen 3 was from a 23-year-old woman who presented with escherichia coli urinary infection and bacteremia that was treated with antibiotics for 10 days before the biopsy. Granulomatous inflammation in reponse to infectious agents or drugs in immunosuppressed kidney transplant recipients can rarely give rise to allograft interstitial nephritis that is distinct from acute rejection. To our knowledge, there are no prior reports of granulomatous tubulointerstitial nephritis associated with C albicans and E coli infection or antibiotic therapy in human renal allografts.
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9/112. Acute granulomatous interstitial nephritis and colitis in anticonvulsant hypersensitivity syndrome associated with lamotrigine treatment.

    We present the case of a 17-year-old woman with a history of bipolar disorder, who developed a clinical syndrome manifested by fever, lymphadenopathy, skin rash, diarrhea, and acute renal failure requiring dialysis after the use of lamotrigine. Renal biopsy showed acute interstitial nephritis (AIN) with focal granulomas. Similarly, colonic biopsy specimens showed colitis and ileitis with non-necrotizing epithelioid granulomas. The patient had a complete recovery after withdrawal of the medication and steroid treatment. Although lamotrigine has been previously implicated in the development of anticonvulsant hypersensitivity syndrome, there have been no previous reports of acute granulomatous interstitial nephritis or colitis associated with the use of this drug.
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10/112. Interstitial granulomatous drug reaction with a histological pattern of interstitial granulomatous dermatitis.

    The interstitial granulomatous drug reaction (IGDR) is a novel drug-associated entity, characterized by violaceous plaques with a predilection for skin fold areas. light microscopically, it resembles the incipient diffuse interstitial phase of granuloma annulare. Differentiating light microscopic features include the absence of complete collagen necrobiosis, the presence of interface dermatitis, and variable lymphoid atypia. The lack of vasculitis rules out the extravascular necrotizing granuloma (Winkelmann granuloma) associated with systemic disease. The differential diagnosis with interstitial granulomatous dermatitis with arthritis as defined by Ackerman et al. has not been studied until now. Our aim was to determine the histologic criteria allowing us to differentiate IGDR without interface dermatitis and lymphoid atypia from interstitial granulomatous dermatitis. We report three patients with IGDR triggered, in two cases by respectively angiotensin convertin enzyme (ACE) inhibitors and furosemide, and in one case by the association of an ACE inhibitor, furosemide, and fluindione. Histologic examination showed a histological pattern of interstitial granulomatous dermatitis. We found a dense, diffuse histiocytic infiltrate distributed interstitially and in palisaded array within the reticular dermis. eosinophils and some neutrophils were scattered throughout the infiltrate. In some tiny foci, enveloped by histiocytes, thick collagen bundles associated with basophilic nuclear debris or "flame figures" were seen. Vasculitis, interface dermatitis, or lymphoid atypia were absent. Our study allowed us to expand the histological spectrum of IGDR including a histological pattern similar to interstitial granulomatous dermatitis. The lack of degenerated collagen could be a subtle clue in favor of interstitial granulomatous dermatitis triggered by a drug.
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