Successful Treatment of Granulomatous-lymphocytic Interstitial Lung Disease in a Patient with CTLA-4 Deficiency

Masashi Nishimura; Jun Miyata; Tomomi Tanigaki; Sakika Nomura; Yusuke Serizawa; Syunya Igarashi; Koki Itou; Tomohiro Ohno; Yuhei Kurata; Yoshifumi Kimizuka; Yuji Fujikura; Yujin Sekinaka; Kanako Sekinaka; Susumu Matsukuma; Shigeaki Nonoyama; Akihiko Kawana

doi:10.2169/internalmedicine.0076-22

Successful Treatment of Granulomatous-lymphocytic Interstitial Lung Disease in a Patient with CTLA-4 Deficiency

Masashi Nishimura, Jun Miyata, Tomomi Tanigaki, Sakika Nomura, Yusuke Serizawa, Syunya Igarashi, Koki Itou, Tomohiro Ohno, Yuhei Kurata, Yoshifumi Kimizuka, Yuji Fujikura, Yujin Sekinaka, Kanako Sekinaka, Susumu Matsukuma, Shigeaki Nonoyama, Akihiko Kawana

研究成果: Article › 査読

1 被引用数 (Scopus)

抄録

Common variable immunodeficiency (CVID) causes granulomatous-lymphocytic interstitial lung disease (GLILD) and has a poor prognosis. We herein report a case of GLILD in a 49-year-old woman with CTLA-4 deficiency-associated CVID. The patient presented with dyspnea that had worsened over the past two years. A laboratory examination revealed hypoglobulinemia and pancytopenia. Chest computed tomography showed diffuse infiltrative and granular shadows in the bilateral interstitium. A flow cytometric analysis of blood cells and genetic testing confirmed CTLA-4 deficiency. We performed video-assisted thoracoscopic surgery for the pathological diagnosis of GLILD and to exclude infection and malignancy. Corticosteroid treatment successfully improved the condition of the patient.

本文言語	English
ページ（範囲）	871-875
ページ数	5
ジャーナル	Internal Medicine
巻	62
号	6
DOI	https://doi.org/10.2169/internalmedicine.0076-22
出版ステータス	Published - 2023
外部発表	はい

ASJC Scopus subject areas

内科学

文献へのアクセス

10.2169/internalmedicine.0076-22

他のファイルとリンク

引用スタイル

Nishimura, M., Miyata, J., Tanigaki, T., Nomura, S., Serizawa, Y., Igarashi, S., Itou, K., Ohno, T., Kurata, Y., Kimizuka, Y., Fujikura, Y., Sekinaka, Y., Sekinaka, K., Matsukuma, S., Nonoyama, S., & Kawana, A. (2023). Successful Treatment of Granulomatous-lymphocytic Interstitial Lung Disease in a Patient with CTLA-4 Deficiency. Internal Medicine, 62(6), 871-875. https://doi.org/10.2169/internalmedicine.0076-22

Nishimura, M, Miyata, J, Tanigaki, T, Nomura, S, Serizawa, Y, Igarashi, S, Itou, K, Ohno, T, Kurata, Y, Kimizuka, Y, Fujikura, Y, Sekinaka, Y, Sekinaka, K, Matsukuma, S, Nonoyama, S & Kawana, A 2023, 'Successful Treatment of Granulomatous-lymphocytic Interstitial Lung Disease in a Patient with CTLA-4 Deficiency', Internal Medicine, vol. 62, no. 6, pp. 871-875. https://doi.org/10.2169/internalmedicine.0076-22

@article{275949b6f02f4692b3264c020d23b244,

title = "Successful Treatment of Granulomatous-lymphocytic Interstitial Lung Disease in a Patient with CTLA-4 Deficiency",

abstract = "Common variable immunodeficiency (CVID) causes granulomatous-lymphocytic interstitial lung disease (GLILD) and has a poor prognosis. We herein report a case of GLILD in a 49-year-old woman with CTLA-4 deficiency-associated CVID. The patient presented with dyspnea that had worsened over the past two years. A laboratory examination revealed hypoglobulinemia and pancytopenia. Chest computed tomography showed diffuse infiltrative and granular shadows in the bilateral interstitium. A flow cytometric analysis of blood cells and genetic testing confirmed CTLA-4 deficiency. We performed video-assisted thoracoscopic surgery for the pathological diagnosis of GLILD and to exclude infection and malignancy. Corticosteroid treatment successfully improved the condition of the patient.",

keywords = "CTLA-4 deficiency, common variable immunodeficiency, corticosteroid, granulomatouslymphocytic interstitial lung disease, video-assisted thoracoscopic surgery",

author = "Masashi Nishimura and Jun Miyata and Tomomi Tanigaki and Sakika Nomura and Yusuke Serizawa and Syunya Igarashi and Koki Itou and Tomohiro Ohno and Yuhei Kurata and Yoshifumi Kimizuka and Yuji Fujikura and Yujin Sekinaka and Kanako Sekinaka and Susumu Matsukuma and Shigeaki Nonoyama and Akihiko Kawana",

year = "2023",

doi = "10.2169/internalmedicine.0076-22",

language = "English",

volume = "62",

pages = "871--875",

journal = "Internal Medicine",

issn = "0918-2918",

publisher = "Japanese Society of Internal Medicine",

number = "6",

}

TY - JOUR

T1 - Successful Treatment of Granulomatous-lymphocytic Interstitial Lung Disease in a Patient with CTLA-4 Deficiency

AU - Nishimura, Masashi

AU - Miyata, Jun

AU - Tanigaki, Tomomi

AU - Nomura, Sakika

AU - Serizawa, Yusuke

AU - Igarashi, Syunya

AU - Itou, Koki

AU - Ohno, Tomohiro

AU - Kurata, Yuhei

AU - Kimizuka, Yoshifumi

AU - Fujikura, Yuji

AU - Sekinaka, Yujin

AU - Sekinaka, Kanako

AU - Matsukuma, Susumu

AU - Nonoyama, Shigeaki

AU - Kawana, Akihiko

PY - 2023

Y1 - 2023

N2 - Common variable immunodeficiency (CVID) causes granulomatous-lymphocytic interstitial lung disease (GLILD) and has a poor prognosis. We herein report a case of GLILD in a 49-year-old woman with CTLA-4 deficiency-associated CVID. The patient presented with dyspnea that had worsened over the past two years. A laboratory examination revealed hypoglobulinemia and pancytopenia. Chest computed tomography showed diffuse infiltrative and granular shadows in the bilateral interstitium. A flow cytometric analysis of blood cells and genetic testing confirmed CTLA-4 deficiency. We performed video-assisted thoracoscopic surgery for the pathological diagnosis of GLILD and to exclude infection and malignancy. Corticosteroid treatment successfully improved the condition of the patient.

AB - Common variable immunodeficiency (CVID) causes granulomatous-lymphocytic interstitial lung disease (GLILD) and has a poor prognosis. We herein report a case of GLILD in a 49-year-old woman with CTLA-4 deficiency-associated CVID. The patient presented with dyspnea that had worsened over the past two years. A laboratory examination revealed hypoglobulinemia and pancytopenia. Chest computed tomography showed diffuse infiltrative and granular shadows in the bilateral interstitium. A flow cytometric analysis of blood cells and genetic testing confirmed CTLA-4 deficiency. We performed video-assisted thoracoscopic surgery for the pathological diagnosis of GLILD and to exclude infection and malignancy. Corticosteroid treatment successfully improved the condition of the patient.

KW - CTLA-4 deficiency

KW - common variable immunodeficiency

KW - corticosteroid

KW - granulomatouslymphocytic interstitial lung disease

KW - video-assisted thoracoscopic surgery

UR - http://www.scopus.com/inward/record.url?scp=85150512999&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85150512999&partnerID=8YFLogxK

U2 - 10.2169/internalmedicine.0076-22

DO - 10.2169/internalmedicine.0076-22

M3 - Article

C2 - 35945007

AN - SCOPUS:85150512999

SN - 0918-2918

VL - 62

SP - 871

EP - 875

JO - Internal Medicine

JF - Internal Medicine

IS - 6

ER -

Successful Treatment of Granulomatous-lymphocytic Interstitial Lung Disease in a Patient with CTLA-4 Deficiency

抄録

ASJC Scopus subject areas

文献へのアクセス

他のファイルとリンク

フィンガープリント

引用スタイル