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着色性干皮病C型(XP-C)患者血细胞中独特的高自发突变负荷。

Uniquely high spontaneous mutational load in blood cells of XP-C patients.

作者信息

Fan-Huang Gordon, Schmidt Elizabeth L, Lee Moonsook, Sun Shixiang, Boull Christina, Maguiness Sheilagh M, Vijg Jan, Niedernhofer Laura J, Maslov Alexander Y

机构信息

Department of Genetics, Albert Einstein College of Medicine, Bronx NY 10461, USA.

Institute on the Biology of Aging and Metabolism, University of Minnesota Medical School.

出版信息

bioRxiv. 2025 Jul 19:2025.07.16.665113. doi: 10.1101/2025.07.16.665113.

Abstract

We discovered a uniquely high spontaneous somatic mutational load in peripheral blood mononuclear cells (PBMCs) of Xeroderma Pigmentosum group C (XP-C) patients, characterized by elevated single nucleotide variants associated with mutation signatures SBS8 and SBS32, as well as an enrichment of single-nucleotide cytosine deletions. This hypermutability was markedly lower in fibroblasts, suggesting a replication-dependent mechanism of mutagenesis due to deficient global genome nucleotide excision repair (GG-NER). Our findings reveal distinct molecular subtypes within XP defined by spontaneous mutational load in normal blood cells and demonstrate that the exceptionally high mutation burden in XP-C leukemia originates from mutations already present prior to malignant transformation.

摘要

我们在着色性干皮病C组(XP-C)患者的外周血单个核细胞(PBMC)中发现了独特的高自发体细胞突变负荷,其特征是与突变特征SBS8和SBS32相关的单核苷酸变异升高,以及单核苷酸胞嘧啶缺失的富集。这种高突变性在成纤维细胞中明显较低,提示由于全球基因组核苷酸切除修复(GG-NER)缺陷导致的复制依赖性诱变机制。我们的研究结果揭示了XP中由正常血细胞中的自发突变负荷定义的不同分子亚型,并证明XP-C白血病中异常高的突变负担源于恶性转化之前就已存在的突变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/d662/12338625/4144d1dcca58/nihpp-2025.07.16.665113v1-f0001.jpg

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