Intravenous Immunoglobulin Monotherapy for Granulomatous Lymphocytic Interstitial Lung Disease in Common Variable Immunodeficiency

Mizue Hasegawa; Fumikazu Sakai; Asako Okabayashi; Akitoshi Sato; Naoko Yokohori; Hideki Katsura; Chihiro Asano; Toshiko Kamata; Eitetsu Koh; Yasuo Sekine; Kenzo Hiroshima; Takashi Ogura; Tamiko Takemura

doi:10.2169/internalmedicine.7757-16

CASE REPORTS

Intravenous Immunoglobulin Monotherapy for Granulomatous Lymphocytic Interstitial Lung Disease in Common Variable Immunodeficiency

Mizue Hasegawa, Fumikazu Sakai, Asako Okabayashi, Akitoshi Sato, Naoko Yokohori, Hideki Katsura, Chihiro Asano, Toshiko Kamata, Eitetsu Koh, Yasuo Sekine, Kenzo Hiroshima, Takashi Ogura, Tamiko Takemura

Author information

Mizue Hasegawa
Division of Respiratory Medicine, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Department of Diagnostic Radiology, Saitama Medical University International Medical Center, Japan
Fumikazu Sakai
Department of Diagnostic Radiology, Saitama Medical University International Medical Center, Japan
Asako Okabayashi
Division of Respiratory Medicine, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Akitoshi Sato
Division of Respiratory Medicine, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Naoko Yokohori
Division of Respiratory Medicine, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Hideki Katsura
Division of Respiratory Medicine, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Chihiro Asano
Division of Hematology, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Toshiko Kamata
Division of Thoracic Surgery, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Eitetsu Koh
Division of Thoracic Surgery, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Yasuo Sekine
Division of Thoracic Surgery, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Kenzo Hiroshima
Division of Pathology, Tokyo Women's Medical University Yachiyo Medical Center, Japan
Takashi Ogura
Department of Respiratory Medicine, Kanagawa Cardiovascular and Respiratory Center, Japan
Tamiko Takemura
Department of Pathology, Japanese Red Cross Medical Center, Japan

Keywords: granulomatous lymphocytic interstitial lung disease, common variable immunodeficiency, lymphoproliferative disease, hypogammaglobulinemia, intravenous immunoglobulin therapy

JOURNAL OPEN ACCESS

2017 Volume 56 Issue 21 Pages 2899-2902

DOI https://doi.org/10.2169/internalmedicine.7757-16

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Details

Abstract

Common variable immunodeficiency (CVID) is a heterogeneous subset of immunodeficiency disorders. Recurrent bacterial infection is the main feature of CVID, but various non-infectious complications can occur. A 42-year-old woman presented with cough and abnormal chest X-ray shadows. Laboratory tests showed remarkable hypogammaglobulinemia. Computed tomography revealed multiple consolidation and nodules on the bilateral lung fields, systemic lymphadenopathy, and splenomegaly. A surgical lung biopsy specimen provided the final diagnosis of lymphoproliferative disease in CVID, which was grouped under the term granulomatous lymphocytic interstitial lung disease. Interestingly, the lung lesions of this case resolved immediately after the initiation of intravenous immunoglobulin monotherapy.

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