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[1例经小唾液腺活检证实对度普利尤单抗有反应的IgG4相关性疾病]

[A CASE OF IgG4-RELATED DISEASE WITH CONFIRMED RESPONSE TO DUPILUMAB BY SMALL SALIVARY GLAND BIOPSY].

作者信息

Yoshinoya Kiyokazu, Watanabe Satoshi, Nakazima Saki, Honda Hidehiro, Harada Yoshinori, Kudo Keita, Okuda Miyuki, Ohshima Shiro

机构信息

Sennanshinge Clinic.

Department of Medical Oncology, NHO Osaka Minami Medical Center.

出版信息

Arerugi. 2024;73(9):1158-1162. doi: 10.15036/arerugi.73.1158.

Abstract

A 71-year-old man was referred to our department due to a cough that occurred one year after surgery for papillary duodenal cancer. We clinically diagnosed the patient with bronchial asthma, with an increase in peripheral blood eosinophil count, exhaled NO, and IgE and obstructive ventilation disorders based on pulmonary function tests. Fluticasone-vilanterol was introduced, but there was little improvement in the cough. We suspected bronchial asthma complicated by IgG4-related disease due to the high serum IgG4 levels and diagnosed the patient with IgG4-related disease through a pancreatic tissue stain of a previously resected duodenal papillectomy section. When dupilumab was initiated for bronchial asthma complicated by IgG4-related disease, the cough resolved. In addition, serum IgG4 levels decreased after the initiation of dupilumab treatment, and a decrease in IgG4-positive plasma cells was observed on small salivary gland biopsy. Thus, the treatment of inhaled steroid-resistant bronchial asthma with dupilumab can also improve IgG4-related disease, and we confirmed a decrease in IgG4-positive plasma cells in the small salivary glands.

摘要

一名71岁男性因在十二指肠乳头癌手术后一年出现咳嗽而转诊至我科。基于肺功能测试,我们临床诊断该患者患有支气管哮喘,外周血嗜酸性粒细胞计数、呼出一氧化氮、免疫球蛋白E升高以及存在阻塞性通气障碍。开始使用氟替卡松-维兰特罗治疗,但咳嗽改善甚微。由于血清免疫球蛋白G4(IgG4)水平升高,我们怀疑支气管哮喘合并IgG4相关疾病,并通过对先前切除的十二指肠乳头切除术切片进行胰腺组织染色诊断该患者患有IgG4相关疾病。当开始使用度普利尤单抗治疗合并IgG4相关疾病的支气管哮喘时,咳嗽消失。此外,开始度普利尤单抗治疗后血清IgG4水平下降,小唾液腺活检观察到IgG4阳性浆细胞减少。因此,用度普利尤单抗治疗吸入性类固醇抵抗的支气管哮喘也可改善IgG4相关疾病,并且我们证实小唾液腺中IgG4阳性浆细胞减少。

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