Open AccessAntineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism
Author(s) -
Sorato Segoe,
Ken Ei Sada,
Keigo Hayashi,
Yuriko Yamamura,
Michiko Morishita,
Hirotaka Watanabe,
Yoshinori Matsumoto,
Jun Wada
Publication year - 2018
Publication title -
medicine
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 0.59
H-Index - 148
eISSN - 1536-5964
pISSN - 0025-7974
DOI - 10.1097/md.0000000000013805
Subject(s) - medicine , familial mediterranean fever , serositis , colchicine , vasculitis , propylthiouracil , anti neutrophil cytoplasmic antibody , mefv , pleurisy , dermatology , gastroenterology , immunology , gene mutation , arthritis , pleural effusion , gene , mutation , genetics , disease , thyroid , biology
Rationale: Familial Mediterranean fever (FMF) is a genetic autoinflammatory disorder characterized by serositis and recurrent fever. Previous reports identified patients with antineutrophil cytoplasmic antibody (ANCA)-positive FMF, but vasculitis symptoms were not reported. Patient concerns: We report the case of a 44-year-old man with numbness. He had a history of 3 episodes of pleurisy and was being treated with propylthiouracil for hyperthyroidism. Because he was ANCA-positive, we suspected drug-induced ANCA-associated vasculitis and propylthiouracil was discontinued. However, his numbness was not ameliorated, and he again developed high fever with pleurisy. Diagnosis: Diagnosis of FMF was finally made, and genetic analysis revealed compound heterozygous mutations in exon 2 of the familial Mediterranean fever gene (L110P/E148Q). Interventions: The patient was treated with 0.5 mg/day of colchicine. Outcomes: His numbness improved, and fever has not recurred. Lessons: Appearance of ANCA and development of vasculitis should be considered in a clinical course of FMF with hyperthyroidism.