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新型遗传性TAP1基因N端变体与严重临床表现——基因型-表型相关性正在显现?

Novel Inherited N-terminus TAP1 Variants and Severe Clinical Manifestations- Are Genotype-Phenotype Correlations Emerging?

作者信息

Bhattarai Dharmagat, Banday Aaqib Zaffar, Sharda Sheetal, Patra Pratap Kumar, Walter Jolan E, Sullivan Kathleen E

机构信息

Advanced Center for Immunology and Rheumatology, Kathmandu, 44600, Nepal.

Pediatric Clinical Immunology and Rheumatology, Division of Immunology & Pediatric Rheumatology, Advanced Centre for Immunology and Rheumatology, Kathmandu, Nepal.

出版信息

J Clin Immunol. 2025 Jan 3;45(1):63. doi: 10.1007/s10875-024-01856-w.

Abstract

Major histocompatibility complex class I deficiency results from deleterious biallelic variants in TAP1, TAP2, TAPBP, and B2M genes. Only a few patients with variant-curated TAP1 deficiency (TAP1D) have been reported in the literature and the clinical phenotype has been variable with an emphasis on autoimmune and inflammatory complications. We report TAP1D in a Nepalese girl with a severe clinical phenotype with serious viral infections at a very young age. A novel frameshift termination variant near the protein's amino (N-) terminal was found. Variants in exon 1 of the TAP1 gene (as in our case) have not been reported previously. We also perform a brief review of TAP1D that hints at potential genotype-phenotype correlations. However, these findings need to be interpreted with due prudence given the small number of patients with TAP1D reported thus far.

摘要

主要组织相容性复合体I类缺陷是由TAP1、TAP2、TAPBP和B2M基因中的有害双等位基因变异引起的。文献中仅报道了少数经变异整理的TAP1缺陷(TAP1D)患者,其临床表型各不相同,主要表现为自身免疫和炎症并发症。我们报告了一名尼泊尔女孩患有TAP1D,其临床表型严重,在很小的时候就出现了严重的病毒感染。在蛋白质氨基(N)末端附近发现了一个新的移码终止变异。TAP1基因第1外显子中的变异(如我们病例中的情况)此前尚未见报道。我们还对TAP1D进行了简要综述,提示了潜在的基因型-表型相关性。然而,鉴于迄今为止报道的TAP1D患者数量较少,这些发现需要谨慎解读。

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