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一名患有遗传性γ-干扰素受体缺陷的儿童发生致命性播散性耻垢分枝杆菌感染。

Fatal disseminated Mycobacterium smegmatis infection in a child with inherited interferon gamma receptor deficiency.

作者信息

Pierre-Audigier C, Jouanguy E, Lamhamedi S, Altare F, Rauzier J, Vincent V, Canioni D, Emile J F, Fischer A, Blanche S, Gaillard J L, Casanova J L

机构信息

INSERM U429 and Service de Microbiologie, d'Anatomie Pathologique, and d'Immunologie Pédiatrique, Hôpital Necker-Enfants Malades, Paris, France.

出版信息

Clin Infect Dis. 1997 May;24(5):982-4. doi: 10.1093/clinids/24.5.982.

Abstract

Mycobacterium smegmatis is a common environmental mycobacterium that was first identified in 1884, yet is a rare pathogen in humans. The few M. smegmatis infections reported to date have been localized and have occurred in association with a primary lesion in otherwise immunocompetent individuals. To our knowledge, no case of disseminated M. smegmatis infection has ever been reported, even in patients with severe immune deficiencies. We report a case of disseminated mycobacterial infection that was diagnosed in a 3-year-old girl. The pathogen was not identified as M. smegmatis until the patient was 6 years old. Her condition gradually worsened, and she died when she was 8 years old despite appropriate antimycobacterial therapy. No other opportunistic infections were documented. Immunological investigations revealed an inherited interferon gamma receptor 1 deficiency. This report identifies M. smegmatis as a new opportunistic agent that may be responsible for disseminated disease in immunocompromised individuals.

摘要

耻垢分枝杆菌是一种常见的环境分枝杆菌,于1884年首次被鉴定出来,但在人类中是一种罕见的病原体。迄今为止报告的少数耻垢分枝杆菌感染病例均为局限性感染,且发生在免疫功能正常的个体中,并伴有原发性病变。据我们所知,即使在严重免疫缺陷的患者中,也从未报告过播散性耻垢分枝杆菌感染病例。我们报告一例在一名3岁女孩中诊断出的播散性分枝杆菌感染病例。直到患者6岁时,病原体才被鉴定为耻垢分枝杆菌。她的病情逐渐恶化,尽管接受了适当的抗分枝杆菌治疗,仍在8岁时死亡。未记录到其他机会性感染。免疫学调查显示存在遗传性干扰素γ受体1缺陷。本报告将耻垢分枝杆菌确定为一种新的机会性病原体,可能导致免疫功能低下个体发生播散性疾病。

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