A LINE-1 Mediated Deletion Resulting in Germline Retinoblastoma Predisposition

Erica L Macke, Anthony R Miller, Eileen Stonerock, Randal Olshefski, Kristin N Zajo, Tracy A Bedrosian, Elaine R Mardis, Yassmine M N Akkari, C. Cottrell, K. Schieffer
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Abstract

Retinoblastoma is an ocular cancer associated with genomic variation in the RB1 gene. In individuals with bilateral retinoblastoma, a germline variant in RB1 is identified in virtually all cases. We describe herein an individual with bilateral retinoblastoma for whom multiple clinical lab assays performed by outside commercial laboratories failed to identify a germline RB1 variant. Paired tumor/normal exome sequencing, long-read whole genome sequencing, and long-read isoform sequencing performed on a translational research basis ultimately identified a germline likely de novo LINE-1 mediated deletion resulting in a premature stop of translation of RB1 as the underlying genetic cause of retinoblastoma in this individual. Based on these research findings, the LINE-1 mediated deletion was confirmed via Sanger sequencing in our clinical laboratory and results reported into the patient medical record to allow for appropriate genetic counseling.
LINE-1 介导的缺失导致遗传性视网膜母细胞瘤易感性
视网膜母细胞瘤是一种与RB1基因基因组变异相关的眼癌。在双侧视网膜母细胞瘤患者中,RB1的种系变异几乎在所有病例中都被发现。我们在此描述了一个双侧视网膜母细胞瘤的个体,由外部商业实验室进行的多次临床实验室分析未能识别种系RB1变体。配对肿瘤/正常外显子组测序、长读全基因组测序和长读异构体测序在翻译研究的基础上最终确定了一种生殖细胞可能从头开始的LINE-1介导的缺失,导致RB1翻译过早停止,这是该个体视网膜母细胞瘤的潜在遗传原因。基于这些研究结果,我们的临床实验室通过Sanger测序证实了LINE-1介导的缺失,并将结果报告到患者病历中,以便进行适当的遗传咨询。
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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