Thymoma with immunodeficiency, combined diffuse panbronchiolitis, and latent autoimmune diabetes in adults- case report and systematic review

被引:2
作者
Xu, Yijiao [1 ]
Wang, Lumin [1 ]
Chen, Zhisheng [1 ]
Zhang, Qingwei [1 ]
Shen, Yun [1 ,2 ]
Ye, Yanrong [1 ,2 ]
Liu, Jiaxin [1 ]
Zhang, Huijun [1 ,2 ]
机构
[1] Fudan Univ, Zhongshan Hosp Xiamen, Xiamen 361015, Peoples R China
[2] Fudan Univ, Zhongshan Hosp, Shanghai 200032, Peoples R China
关键词
Good's syndrome; Diffuse panbronchiolitis; LADA (latent autoimmune diabetes in adult diabetes); Thymoma; Immunodeficiency; GOODS-SYNDROME; PATIENT; ONSET; ASSOCIATION; DISTURBANCES; MYASTHENIA; INFECTION;
D O I
10.1016/j.jtauto.2023.100230
中图分类号
R392 [医学免疫学]; Q939.91 [免疫学];
学科分类号
100102 ;
摘要
Thymoma with Immunodeficiency (Good's Syndrome, GS) is a rare association between thymoma and immunodeficiency, first described over 60 years ago. Patients with GS typically present with thymomas, reduced or absent B cells in the peripheral blood, hypogammaglobulinemia, and defects in cell-mediated immunity. We report the case of a 67-year-old woman diagnosed with GS following the development of a progressive, severe, refractory pulmonary infection and diffuse panbronchiolitis (DPB). She also had diabetes, characterized by anti-glutamic acid decarboxylase antibody positivity, leading to a diagnosis of latent autoimmune diabetes in adults (LADA). A thorough review of existing literature revealed that GS is often confirmed after multiple episodes of opportunistic infections or autoimmune diseases post-thymoma surgery. Due to their immunodeficiency, GS patients frequently suffer from recurrent infections over extended periods, and some succumb to severe in-fections. Regular immunoglobulin infusions may be effective in treating GS.
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页数:6
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