TY - JOUR
T1 - Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism
T2 - A case report
AU - Segoe, Sorato
AU - Sada, Ken ei
AU - Hayashi, Keigo
AU - Yamamura, Yuriko
AU - Morishita, Michiko
AU - Watanabe, Haruki
AU - Matsumoto, Yoshinori
AU - Wada, Jun
PY - 2018/12
Y1 - 2018/12
N2 - 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.
AB - 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.
KW - Antineutrophil cytoplasmic antibody
KW - Familial Mediterranean fever
KW - Periodic fever
KW - Pleurisy
KW - Propylthiouracil
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U2 - 10.1097/MD.0000000000013805
DO - 10.1097/MD.0000000000013805
M3 - Article
C2 - 30572542
AN - SCOPUS:85058909639
VL - 97
JO - Medicine; analytical reviews of general medicine, neurology, psychiatry, dermatology, and pediatries
JF - Medicine; analytical reviews of general medicine, neurology, psychiatry, dermatology, and pediatries
SN - 0025-7974
IS - 51
M1 - e13805
ER -