Jornal de Distúrbios e Terapia da Tireóide
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Familial Non Medullary Thyroid Cancer: Clinic Cases and Review of literature

R. Ciuni, C. Spataro, S. Nicosia, A. Biondi, F. Basile and S. Ciuni

Introduction: Vasculitis is a rare complication of antithyroid drugs reported with propylthiouracil, carbimazole, methimazole and Benzylthiouracil. Benzylthiouracil –induced Vasculitis are often severe forms with renal or pulmonary involvement, which can be life-threatening if left untreated. We describe the clinical course and medical management of 2 cases of severe vasculitis with alveolar hemorrhage and renal involvement occurred in 2 patients with Graves’ disease treated by Benzylthiouracil.

Cases report: A 36 and 33-year-old women with Graves’ disease developed alveolar hemorrhage and acute renal failure after respectively 36 and 144 months of Benzylthiouracil therapy. Kidney biopsy showed pauci-immune crescentic glomerulonephritis in the 2 cases. Anti Neutrophil Cytoplasmic Antibody (ANCA) was positive (P- ANCA in the first patient and c-ANCA in the second patient). The condition of the first patient improved when Benzylthiouracil was withdrawn associated with corticosteroids and immunosuppressive treatment after a follow up of 6 months. However, the second patient died by severe infection after the same treatment.

Conclusion: Benzylthiouracil vasculitis is a serious complication but its prognosis is good if diagnosis and treatment are early. However mortality is related to risk of infection.