A case of development of autoimmune pulmonary alveolar proteinosis during the treatment of hypersensitivity pneumonitis

Maki Asami-Noyama; Kosuke Ito; Misa Harada; Yukari Hisamoto; Yoshie Kunihiro; Eiji Ikeda; Tasuku Yamamoto; Junki Suizu; Ayumi Fukatsu; Syuichiro Ohata; Yoriyuki Murata; Keiji Oishi; Yoshikazu Yamaji; Nobutaka Edakuni; Tomoyuki Kakugawa; Tsunahiko Hirano; Kazuto Matsunaga

doi:10.1016/j.rmcr.2023.101862

A case of development of autoimmune pulmonary alveolar proteinosis during the treatment of hypersensitivity pneumonitis

Respir Med Case Rep. 2023 May 16:44:101862. doi: 10.1016/j.rmcr.2023.101862. eCollection 2023.

Authors

Maki Asami-Noyama¹, Kosuke Ito², Misa Harada², Yukari Hisamoto¹, Yoshie Kunihiro³, Eiji Ikeda⁴, Tasuku Yamamoto¹, Junki Suizu², Ayumi Fukatsu¹, Syuichiro Ohata¹, Yoriyuki Murata¹, Keiji Oishi¹, Yoshikazu Yamaji¹, Nobutaka Edakuni¹, Tomoyuki Kakugawa⁵, Tsunahiko Hirano¹, Kazuto Matsunaga¹

Affiliations

¹ Department of Respiratory Medicine and Infectious Disease, Graduate School of Medicine, Yamaguchi University, Ube, Japan.
² Department of Respiratory Medicine, National Hospital Organization Yamaguchi-Ube Medical Center, Ube, Japan.
³ Department of Radiology, Graduate School of Medicine, Yamaguchi University, Ube, Japan.
⁴ Department of Pathology, Graduate School of Medicine, Yamaguchi University, Ube, Japan.
⁵ Department of Pulmonology and Gerontology, Graduate School of Medicine, Yamaguchi University, Ube, Japan.

Abstract

Herein, we report the case of a 73-year-old woman with an occupational history of plaster grinding who developed autoimmune pulmonary alveolar proteinosis (PAP) during the treatment of fibrotic hypersensitivity pneumonitis with steroids and immunosuppressive drugs. Based on the changes in computed tomography imaging findings, poor response to steroid therapy, and markedly elevated KL-6 levels, PAP was suspected and diagnosed by bronchoscopy. Repeated segmental bronchoalveolar lavage under high-flow nasal cannula oxygen therapy resulted in slight improvement. Steroids and immunosuppressive treatments for other interstitial lung diseases may cause PAP or exacerbate latent PAP.

Keywords: Anti-GM-CSF antibody; Autoimmune pulmonary alveolar proteinosis; Fibrotic hypersensitivity pneumonia; Segmental bronchoalveolar lavage.

Publication types

Case Reports