TY - JOUR
T1 - Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism
T2 - A case report
AU - Segoe, Sorato
AU - Sada, Kenei
AU - Hayashi, Keigo
AU - Yamamura, Yuriko
AU - Morishita, Michiko
AU - Watanabe, Haruki
AU - Matsumoto, Yoshinori
AU - Wada, Jun
N1 - Funding Information:
Patient consent: Written informed consent for this case report was obtained from the patient. J.W. received a speaker honorarium from Daiichi Sankyo, MSD, Tanabe Mitsubishi, and Taisho Toyama, and receives grant support from Baxter, Dainippon Sumitomo, Ono, and Teijin Pharma. K.S. received a speaker honorarium from Chugai. Y.M. receives grant support from Mochida, Astellas, Kissei, Taisho, and Kobayashi Pharma. The other authors have no conflicts of interest to disclose. aOkayama University Medical School, bDepartment of Nephrology, Rheumatology, Endocrinology and Metabolism, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan. ∗Correspondence: Ken-ei Sada, Okayama University, Okayama 700-8558, Japan (e-mail: sadakenn@okayama-u.ac.jp).
Publisher Copyright:
Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc. This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
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 -