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Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism: A case report

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 repo...

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Autores principales: Segoe, Sorato, Sada, Ken-ei, Hayashi, Keigo, Yamamura, Yuriko, Morishita, Michiko, Watanabe, Haruki, Matsumoto, Yoshinori, Wada, Jun
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wolters Kluwer Health 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6319784/
https://www.ncbi.nlm.nih.gov/pubmed/30572542
http://dx.doi.org/10.1097/MD.0000000000013805
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author Segoe, Sorato
Sada, Ken-ei
Hayashi, Keigo
Yamamura, Yuriko
Morishita, Michiko
Watanabe, Haruki
Matsumoto, Yoshinori
Wada, Jun
author_facet Segoe, Sorato
Sada, Ken-ei
Hayashi, Keigo
Yamamura, Yuriko
Morishita, Michiko
Watanabe, Haruki
Matsumoto, Yoshinori
Wada, Jun
author_sort Segoe, Sorato
collection PubMed
description 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.
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spelling pubmed-63197842019-01-24 Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism: A case report Segoe, Sorato Sada, Ken-ei Hayashi, Keigo Yamamura, Yuriko Morishita, Michiko Watanabe, Haruki Matsumoto, Yoshinori Wada, Jun Medicine (Baltimore) Research Article 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. Wolters Kluwer Health 2018-12-21 /pmc/articles/PMC6319784/ /pubmed/30572542 http://dx.doi.org/10.1097/MD.0000000000013805 Text en Copyright © 2018 the Author(s). Published by Wolters Kluwer Health, Inc. http://creativecommons.org/licenses/by/4.0 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. http://creativecommons.org/licenses/by/4.0
spellingShingle Research Article
Segoe, Sorato
Sada, Ken-ei
Hayashi, Keigo
Yamamura, Yuriko
Morishita, Michiko
Watanabe, Haruki
Matsumoto, Yoshinori
Wada, Jun
Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism: A case report
title Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism: A case report
title_full Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism: A case report
title_fullStr Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism: A case report
title_full_unstemmed Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism: A case report
title_short Antineutrophil cytoplasmic antibody-positive familial Mediterranean fever and hyperthyroidism: A case report
title_sort antineutrophil cytoplasmic antibody-positive familial mediterranean fever and hyperthyroidism: a case report
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6319784/
https://www.ncbi.nlm.nih.gov/pubmed/30572542
http://dx.doi.org/10.1097/MD.0000000000013805
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