与NHEJ1基因突变相关的严重免疫缺陷谱:Cernunnos/XLF缺陷

Ana María Navarro, Gabriela Mantilla, Jorge Andrés Fernández, Mario Fernando Unigarro, Alfonso Suárez, María Claudia Ortega
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摘要

Cernunnos/XLF缺乏症是一种罕见的、严重的联合免疫缺陷,遗传为常染色体隐性模式(OMIM编号:611290),与NHEJ1基因有关。该基因参与DNA非同源末端连接途径,修复哺乳动物细胞DNA双链断裂。临床表现为生长迟缓、小头畸形、三角形脸、反复感染、成纤维细胞对γ电离辐射过度敏感、低γ球蛋白血症;B淋巴细胞和T淋巴细胞亚群计数低,自然杀伤细胞值正常。本手稿旨在提出一个极其罕见的联合免疫缺陷的情况下,在一个20岁的男子与非近亲父母和纯合子变异的NHEJ1基因。鉴于nhej1相关免疫缺陷的患病率较低以及由于缺乏知识而难以诊断,该病例是文献中报道的第五十例,也是哥伦比亚的首例。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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Severe immunodeficiency spectrum associated with NHEJ1 gene mutation: Cernunnos/XLF deficiency

Cernunnos/XLF deficiency is a rare, severe combined immunodeficiency, inherited in an autosomal recessive pattern (OMIM number: 611290), related to the NHEJ1 gene. This gene participates in the DNA non-homologous end-joining pathway, repairing double-strand breaks in the DNA of mammalian cells. The clinical features include growth retardation, microcephaly, triangle-shaped face, recurrent infections, fibroblast's excessive sensitivity to gamma-ionizing radiation, and hypogammaglobulinemia; also, low counts of subpopulations of B and T lymphocytes, with normal values of natural-killer cells. This manuscript aims to present an extremely rare case of combined immunodeficiency in a twenty-years-old man with non-consanguineous parents and a homozygote variant of the NHEJ1 gene. This case is the fiftieth reported in the literature and the first in Colombia, given the low prevalence of NHEJ1-related immunodeficiency and its difficult diagnosis due to scarce knowledge.

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