Wang Li, Du Ran, Han Lin, Yang Rui, Li Yingxue
Department of Gynecology & Obstetrics, Liaocheng People's Hospital, School of Medicine, Liaocheng University, PR China.
Department of Pathology, Liaocheng People's Hospital, PR China.
Transl Oncol. 2024 Jul;45:101963. doi: 10.1016/j.tranon.2024.101963. Epub 2024 Apr 25.
This study presents a detailed analysis of the clinical and genetic characteristics of uterine leiomyoma associated with Hereditary Leiomyomatosis and Renal Cell Cancer (HLRCC), combined with exploration of family history, pathology, and management procedures, supported by thorough evidence collection.
Blood samples were collected from the proband, and the pathogenic variant was verified using Sanger sequencing. A comprehensive review of family history, FH deficiency pathology, FH and 2SC immunohistochemistry staining was conducted. Functional evidence was derived from clinical and genetic information, supplemented by a literature collection and mutation was reclassified based on ACMG/AMP guidelines.
The study successfully identifies a novel missense mutation (c.1240A>G; p.Lys414Glu) in exon 9 of FH, with no prior reports in existing databases. The patient's phenotype and family history, coupled with evidence collected from the literature, contribute to the preliminary determination of the variant as likely pathogenic. We also emphasize that the importance of combining FH-deficient morphology and immunohistochemical staining with 2SC for enhanced sensitivity.
This research adds a novel missense mutation to the repertoire of FH gene variants, emphasizing its likely pathogenic nature based on a multidimensional analysis of phenotype, family history, and literature evidence. The findings enhance our understanding of the genetic landscape associated with FH and underscore the importance of thorough characterization for accurate variant classification.
本研究对与遗传性平滑肌瘤病和肾细胞癌(HLRCC)相关的子宫平滑肌瘤的临床和遗传特征进行了详细分析,并结合家族史、病理学和管理程序进行探索,同时进行了全面的证据收集。
采集先证者的血液样本,使用桑格测序法验证致病变异。对家族史、FH缺乏病理学、FH和2SC免疫组织化学染色进行了全面回顾。功能证据来自临床和遗传信息,并辅以文献收集,根据ACMG/AMP指南对突变进行重新分类。
该研究成功在FH基因第9外显子中鉴定出一个新的错义突变(c.1240A>G;p.Lys414Glu),现有数据库中无先前报道。患者的表型和家族史,以及从文献中收集的证据,有助于初步确定该变异可能具有致病性。我们还强调了将FH缺乏形态学和免疫组织化学染色与2SC相结合以提高敏感性的重要性。
本研究为FH基因变异库增添了一个新的错义突变,基于对表型、家族史和文献证据的多维度分析强调了其可能的致病性。这些发现增进了我们对与FH相关的遗传格局的理解,并强调了进行全面特征描述以准确进行变异分类的重要性。