重新评估TNFRSF13B变异对抗体缺乏的贡献。

Journal of human immunity Pub Date : 2025-11-03 Epub Date: 2025-08-19 DOI:10.70962/jhi.20250016
Hassan Abolhassani, Andres Caballero-Oteyza, Mingyu Yang, Michele Proietti, Samaneh Delavari, Patrick Maffucci, Alejandro A Schäffer, Bertrand Boisson, Jean-Laurent Casanova, Nima Rezaei, Qiang Pan-Hammarström, Charlotte Cunningham-Rundles, Lennart Hammarström, Bodo Grimbacher
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引用次数: 0

摘要

主要抗体缺乏症(PAD)是人类先天性免疫缺陷(IEI)最普遍的形式。PAD的特点是反复的细菌感染、免疫失调和免疫球蛋白产生受损。在大约20%的病例中,可以确定PAD的单基因病因。大约10%的患者携带肿瘤坏死因子受体超家族成员13B基因(TNFRSF13B)杂合突变,该基因编码B细胞表面蛋白TACI。TNFRSF13B的杂合变异并不足以引起PAD,因为大约1%的健康人群携带这些变异之一。为了确定这些个体中免疫缺陷的其他遗传因素,我们检查了161名具有罕见破坏性TNFRSF13B变异的PAD患者的外显子组。我们确定了(i) TNFRSF13B的双等位基因突变,(ii) hla ii类标记(DPA1*03),以及(iii)已知b细胞相关基因中的多个单核苷酸多态性,作为额外的遗传风险因素。此外,其他已知IEI基因的致病性突变出现在16%的杂合子TNFRSF13B变异患者中。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Re-evaluation of the contribution of TNFRSF13B variants to antibody deficiency.

Predominantly antibody deficiency (PAD) is the most prevalent form of human inborn errors of immunity (IEI). PAD is characterized by recurrent bacterial infections, immune dysregulation, and impaired immunoglobulin production. A monogenic cause of PAD can be identified in about 20% of cases. Approximately 10% of patients carry heterozygous mutations in the tumor necrosis factor receptor superfamily member 13B gene (TNFRSF13B), encoding the B cell surface protein TACI. Heterozygous variants in TNFRSF13B are not sufficient to cause PAD, as approximately 1% of the healthy population carries one of these variants. To identify additional genetic contributors to the immune defect in these individuals, we examined the exomes of 161 PAD patients with rare-damaging variants in TNFRSF13B. We identified (i) biallelic mutations in TNFRSF13B, (ii) the HLA-class II marker (DPA1*03), and (iii) multiple single nucleotide polymorphisms in known B-cell related genes, as additional genetic risk factors. Moreover, pathogenic mutations in other known IEI genes were presented in 16% of patients with heterozygous TNFRSF13B variants.

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