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Rituximab Restores IFN-γ-STAT1 Function and Ameliorates Disseminated Mycobacterium avium Infection in a Patient with Anti-Interferon-γ Autoantibody

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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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Authors and Affiliations

  1. Department of Clinical Infectious Diseases, Aichi Medical University Hospital, 1-1 Yazakokarimata, Nagakute, Aichi, 480-1195, Japan

    Yusuke Koizumi, Naoya Nishiyama, Jun Hirai, Nobuhiro Asai, Yuka Yamagishi & Hiroshige Mikamo

  2. Department of Infection Control and Prevention, Aichi Medical University Hospital, Aichi, Japan

    Yusuke Koizumi, Naoya Nishiyama, Jun Hirai, Nobuhiro Asai, Yuka Yamagishi, Hideo Kato, Mao Hagihara, Daisuke Sakanashi, Hiroyuki Suematsu & Hiroshige Mikamo

  3. Department of Respiratory Medicine and Infectious Disease, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Niigata, Japan

    Takuro Sakagami

  4. Department of Respiratory Medicine, National Hospital Organization Higashinagoya Hospital, Nagoya, Aichi, Japan

    Yuta Hayashi & Kenji Ogawa

Authors
  1. Yusuke Koizumi
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  2. Takuro Sakagami
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  3. Naoya Nishiyama
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  4. Jun Hirai
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  5. Yuta Hayashi
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  6. Nobuhiro Asai
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  7. Yuka Yamagishi
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  8. Hideo Kato
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  9. Mao Hagihara
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  10. Daisuke Sakanashi
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  11. Hiroyuki Suematsu
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  12. Kenji Ogawa
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  13. Hiroshige Mikamo
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Corresponding author

Correspondence to Yusuke Koizumi.

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Conflicts of Interest

The authors declare that they have no conflict of interest.

Ethics Approval and Consent to Treatment

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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Cite this article

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

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