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The mutational constraint spectrum quantified from variation in 141,456 humans.从 141456 名人类个体的变异中量化的突变约束谱。
Nature. 2020 May;581(7809):434-443. doi: 10.1038/s41586-020-2308-7. Epub 2020 May 27.
3
Conformational Dynamics from Ambiguous Zinc Coordination in the RanBP2-Type Zinc Finger of RBM5.RanBP2 型锌指结构中锌配位的不明确导致构象动态变化。
J Mol Biol. 2020 Jun 26;432(14):4127-4138. doi: 10.1016/j.jmb.2020.05.012. Epub 2020 May 22.
4
Estimating the Effect of Single-Point Mutations on Protein Thermodynamic Stability and Analyzing the Mutation Landscape of the p53 Protein.估计单点突变对蛋白质热力学稳定性的影响,并分析 p53 蛋白的突变景观。
J Chem Inf Model. 2020 Jun 22;60(6):3315-3323. doi: 10.1021/acs.jcim.0c00256. Epub 2020 May 21.
5
Recombination may occur in the absence of transcription in the immunoglobulin heavy chain recombination centre.在免疫球蛋白重链重组中心,转录可能发生在重组发生之前。
Nucleic Acids Res. 2020 Apr 17;48(7):3553-3566. doi: 10.1093/nar/gkaa108.
6
How mouse RAG recombinase avoids DNA transposition.鼠 RAG 重组酶如何避免 DNA 转座。
Nat Struct Mol Biol. 2020 Feb;27(2):127-133. doi: 10.1038/s41594-019-0366-z. Epub 2020 Feb 3.
7
Cutting antiparallel DNA strands in a single active site.在单个活性位点切割反平行 DNA 链。
Nat Struct Mol Biol. 2020 Feb;27(2):119-126. doi: 10.1038/s41594-019-0363-2. Epub 2020 Feb 3.
8
Human Inborn Errors of Immunity: 2019 Update on the Classification from the International Union of Immunological Societies Expert Committee.人类先天性免疫缺陷:国际免疫学联盟专家委员会 2019 年分类更新。
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10
Combined Immunodeficiency With Late-Onset Progressive Hypogammaglobulinemia and Normal B Cell Count in a Patient With RAG2 Deficiency.一名RAG2缺陷患者合并联合免疫缺陷、迟发性进行性低丙种球蛋白血症且B细胞计数正常
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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.

DOI:10.1007/s10875-021-01052-0
PMID:33954879
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8319791/
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。