晚期诊断为x连锁无球蛋白血症患者的新型BTK突变。

IF 0.7 Q4 IMMUNOLOGY
Case Reports in Immunology Pub Date : 2023-11-25 eCollection Date: 2023-01-01 DOI:10.1155/2023/6698913
Amanpreet Kalkat, Olivia Humpel, Robert Hostoffer
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引用次数: 0

摘要

x连锁无球蛋白血症(XLA)是一种布鲁顿酪氨酸激酶(BTK)突变的遗传性疾病。B细胞发育和免疫球蛋白产生缺陷导致6月龄时母体IgG丢失后的复发性感染。一名55岁男性,长期接受输液治疗,诊断为常见的可变免疫缺陷,临床表现为皮肤t细胞淋巴瘤,这激发了整体的重复评估。免疫球蛋白水平和淋巴细胞标志物,家族史和基因检测提示XLA和BTK基因的新突变的真正诊断。在BTK的所有五个结构域中都发现了与疾病相关的突变,在已报道的100种遗传改变中,最常引用的是错义变异。BTK蛋白在造血系和浆细胞中表达,T淋巴细胞除外。蛋白质功能的破坏或缺乏BTK会在B前过渡细胞阶段阻止正常的B细胞发育并诱导过早凋亡。我们提出了一个新的半合子BTK c.1492C > G突变引起XLA患者的第一例报告。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Novel BTK Mutation in Patient with Late Diagnosis of X-Linked Agammaglobulinemia.

X-linked agammaglobulinemia (XLA) is a genetic disorder with mutation in Bruton's tyrosine kinase (BTK). Defects in B cell development and immunoglobulin production lead to recurrent infections following loss of maternal IgG at 6 months of age. A 55-year-old male with a longstanding common variable immunodeficiency diagnosis on infusion therapy presented to the clinic with cutaneous T-cell lymphoma, which inspired overall repeat evaluation. Immunoglobulin levels and lymphocyte markers, family history, and genetic testing prompted a true diagnosis of XLA and novel mutation in the BTK gene. Disease-associated mutations have been noted in all five domains of BTK, with missense variants most commonly cited among the 100s of reported genetic alterations. The BTK protein is expressed in hematopoietic lineages and plasma cells, with the exception of T lymphocytes. Disruption in the protein function or absence of BTK halts normal B cell development at the pre-B transitional cell stage and induces premature apoptosis. We present the first reported case of a novel hemizygous BTK c.1492C > G mutation in a patient causing XLA.

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来源期刊
CiteScore
1.90
自引率
0.00%
发文量
14
审稿时长
15 weeks
期刊介绍: Case Reports in Immunology is a peer-reviewed, Open Access journal that publishes case reports and case series related to allergies, immunodeficiencies, autoimmune diseases, immune disorders, cancer immunology and transplantation immunology.
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