• Internal medicine · Jan 2023

    Case Reports

    Pulmonary and Intestinal Involvement in a Patient with Myeloperoxidase-specific Antineutrophil Cytoplasmic Antibody-positive Hermansky-Pudlak Syndrome.

    • Takatomo Tokito, Noriho Sakamoto, Hiroshi Ishimoto, Daisuke Okuno, Takuto Miyamura, Atsuko Hara, Takashi Kido, Kazuko Yamamoto, Hiroyuki Yamaguchi, Yasushi Obase, Yuko Akazawa, Ken Okamura, Tamio Suzuki, Yuji Ishimatsu, and Hiroshi Mukae.
    • Department of Respiratory Medicine, Nagasaki University Graduate School of Biomedical Sciences, Japan.
    • Intern. Med. 2023 Jan 1; 62 (1): 103106103-106.

    AbstractA 26-year-old Japanese woman was admitted with a 1-month history of diarrhea, a high fever for a few days, and exacerbation of dyspnea. She was treated with an antifibrotic drug and long-term oxygen therapy for Hermansky-Pudlak syndrome-related pulmonary fibrosis. New ground-glass attenuation appeared on chest computed tomography (CT), and a colon biopsy showed an inflammatory cell accumulation with a high titer of myeloperoxidase (MPO)-specific anti-neutrophil cytoplasmic antibodies (ANCA). Systemic inflammation related to MPO-ANCA titer elevation was suspected. Steroid pulse therapy and intravenous cyclophosphamide improved chest CT findings and diarrhea. Therefore, immunosuppressant treatment should be considered for systemic inflammation related to MPO-ANCA.

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