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- W1966358413 abstract "A 38-year old woman presented with progressive metastatic World Health Organization (WHO) subtype B1 thymoma after previous cisplatin-based chemotherapy, mediastinal radiation and pemetrexed. She enrolled on a phase II trial of cixutumumab, an insulin-like growth factor-1 receptor antibody (20 mg/kg, intravenously q 3 weeks).1After two doses, a 29% reduction in the sum of target lesions was observed. Three weeks after the third dose, she presented with fatigue, dyspnea, dysphagia and dysphonia. She had grade 4 proximal muscle weakness of extremities and neck with generalized hyporeflexia. Investigations (Table 1) revealed severe multi-organ dysfunction with polymyositis, fascitis, myocarditis, hepatitis, hypogammaglobulinemia and possible bulbar myasthenic involvement. A day later, she developed worsening of bulbar symptoms and respiratory failure necessitating non-invasive ventilation. She was treated with immunoglobulin and methylprednisolone and responded rapidly with marked improvement in respiratory status, muscle strength, pulmonary function and biochemical parameters (Figure 1A). Peripheral blood lymphocytes and anti-interferon (IFN)-α antibodies were elevated, but CD19+ and CD20+ mature B cells were absent. CD4+, CD8+, CD4−/CD8−, mature naive CD4+ and CD8+ T cells were increased with an inverted CD4/CD8 ratio.Figure 1Trends in aspartate aminotransferase (AST), Alanine transaminase (ALT), Creatine phosphokinase (CPK), troponin I (A), hemoglobin and platelet count (B). Arrows indicate time points at which cixutumumab was administered.Table 1Results of pertinent diagnostic tests at presentationShe received three more doses of cixutumumab before disease progression. During steroid taper, she was diagnosed with pure red cell aplasia (PRCA) and amegakaryocytic thrombocytopenia based on hypoproliferative anemia (hemoglobin 10 g/dl), thrombocytopenia (platelet count 13,000/ul) and cellular marrow with absent megakaryocytes and severe erythroid hypoplasia. Hematological parameters normalized on cyclosporine A (Figure 1B) and thymoma remained stable." @default.
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- W1966358413 date "2015-02-01" @default.
- W1966358413 modified "2023-10-16" @default.
- W1966358413 title "Multiorgan Autoimmune Manifestations Associated with Thymoma" @default.
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- W1966358413 doi "https://doi.org/10.1097/jto.0000000000000324" @default.
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