Abstract
A 67-year-old Japanese female with back pain and severe cachexia visited our hospital. The diagnosis was disseminated Mycobacterium avium complex infection (dMAC) with multiple bone involvement. Anti-mycobacterial chemotherapy was started, but fever persisted and dislocation of cervical vertebrae has made her bedridden. Because anti-interferon (IFN)-γ autoantibody was positive, four doses of rituximab 375 mg/m2, every 7 day, were administered. Soon after treatment, progression of osteolytic lesions and wasting has stopped. We proved that rituximab has recovered IFN-γ signaling as shown by IFN-γ-induced STAT1 phosphorylation. It can be a promising option for dMAC cases with anti-IFN-γ autoantibody.
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Acknowledgements
Yusuke Koizumi, Yuka Yamagishi, Kenji Ogawa, and Hiroshige Mikamo wrote the paper. Takuro Sakagami performed IFN-γ and STAT1 assay. Yusuke Koizumi, Naoya Nishiyama, Yuta Hayashi, Hideo Kato, Mao Hagihara, and Nobuhiro Asai made the diagnosis, clinical decisions, and treatment. Daisuke Sakanashi and Hiroyuki Suematsu performed the cultures and microbiological examinations.
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Informed consent was obtained from the patient after verbal and written information provision. Permission for rituximab off label use was provided by the Institutional Ethics Committee at Aichi Medical University Hospital. Therefore, all the procedures have been performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments.
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Koizumi, Y., Sakagami, T., Nishiyama, N. et al. Rituximab Restores IFN-γ-STAT1 Function and Ameliorates Disseminated Mycobacterium avium Infection in a Patient with Anti-Interferon-γ Autoantibody. J Clin Immunol 37, 644–649 (2017). https://doi.org/10.1007/s10875-017-0425-3
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DOI: https://doi.org/10.1007/s10875-017-0425-3