Cheng Hei-Shun, Chiu Pui-Hing, Wong Charles, Tong Chun-Wai, Miu Pui-Ling Flora
Department of Medicine Pamela Youde Nethersole Eastern Hospital Chai Wan Hong Kong.
Respirol Case Rep. 2023 Aug 9;11(9):e01207. doi: 10.1002/rcr2.1207. eCollection 2023 Sep.
Adult-onset immunodeficiency (AOID) syndrome due to the presence of anti-interferon gamma antibody (AIGA) is characterized by multiple opportunistic infections. We report a case of a 65-year old healthy woman who suffered from and Mycobacterium intracellulare co-infection with clinical presentation mimicking metastatic lung cancer. She presented with chronic cough and weight loss. Her positron emission tomography scan showed a right upper lobe mass, mediastinal lymphadenopathy and multiple bone lesions. Acid fast bacilli culture of the lung mass and mediastinal lymph node revealed Mycobacterium intracellulare and she improved with prolonged antibiotic. Relapse of disseminated Mycobacterium intracellulare infection occurred 15 months post-treatment and AIGA was positive with functional neutralizing activity on downstream immune pathway. AOID syndrome secondary to AIGA was diagnosed. This case illustrated the importance of high index of suspicion of AOID syndrome and the difficulty of early diagnosis. Further studies on its predictive factors and AIGA-targeted treatment modalities are urgently needed.
由于存在抗干扰素γ抗体(AIGA)导致的成人起病免疫缺陷(AOID)综合征的特征为多种机会性感染。我们报告一例65岁健康女性病例,她患有胞内分枝杆菌合并感染,临床表现酷似转移性肺癌。她表现为慢性咳嗽和体重减轻。她的正电子发射断层扫描显示右上叶肿块、纵隔淋巴结肿大和多处骨病变。肺部肿块和纵隔淋巴结的抗酸杆菌培养显示为胞内分枝杆菌,经长期抗生素治疗后病情好转。治疗后15个月发生播散性胞内分枝杆菌感染复发,AIGA呈阳性,对下游免疫途径具有功能性中和活性。诊断为继发于AIGA的AOID综合征。该病例说明了高度怀疑AOID综合征的重要性以及早期诊断的困难。迫切需要对其预测因素和以AIGA为靶点的治疗方式进行进一步研究。