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RAG1/2 基因突变患者的临床表现、突变分析和免疫表型:来自墨西哥的首个病例系列并描述两种新突变

Clinical Manifestations, Mutational Analysis, and Immunological Phenotype in Patients with RAG1/2 Mutations: First Cases Series from Mexico and Description of Two Novel Mutations.

机构信息

Laboratorio de Inmunodeficiencias, Instituto Nacional de Pediatría, Mexico City, Mexico.

Centro de Investigación en Dinámica Celular, Universidad Autónoma del Estado de Morelos, Cuernavaca, Mexico.

出版信息

J Clin Immunol. 2021 Aug;41(6):1291-1302. doi: 10.1007/s10875-021-01052-0. Epub 2021 May 5.

Abstract

Mutations in recombinase activating genes 1 and 2 (RAG1/2) result in human severe combined immunodeficiency (SCID). The products of these genes are essential for V(D)J rearrangement of the antigen receptors during lymphocyte development. Mutations resulting in null-recombination activity in RAG1 or RAG2 are associated with the most severe clinical and immunological phenotypes, whereas patients with hypomorphic mutations may develop leaky SCID, including Omenn syndrome (OS). A group of previously unrecognized clinical phenotypes associated with granulomata and/or autoimmunity have been described as a consequence of hypomorphic mutations. Here, we present six patients from unrelated families with missense variants in RAG1 or RAG2. Phenotypes observed in these patients ranged from OS to severe mycobacterial infections and granulomatous disease. Moreover, we report the first evidence of two variants that had not been associated with immunodeficiency. This study represents the first case series of RAG1- or RAG2-deficient patients from Mexico and Latin America.

摘要

重组激活基因 1 和 2(RAG1/2)的突变导致人类严重联合免疫缺陷(SCID)。这些基因的产物对于淋巴细胞发育过程中抗原受体的 V(D)J 重排是必不可少的。导致 RAG1 或 RAG2 无重组活性的突变与最严重的临床和免疫表型相关,而具有功能减弱突变的患者可能会发展为渗漏性 SCID,包括 Omenn 综合征(OS)。一组以前未被识别的与肉芽肿和/或自身免疫相关的临床表型已被描述为功能减弱突变的结果。在这里,我们介绍了来自无关家庭的 6 名 RAG1 或 RAG2 错义变异患者。这些患者的表型范围从 OS 到严重的分枝杆菌感染和肉芽肿性疾病。此外,我们报告了首例与免疫缺陷无关的两种变异的证据。本研究代表了来自墨西哥和拉丁美洲的 RAG1 或 RAG2 缺陷患者的首个病例系列。

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