韩国嗜铬细胞瘤和副神经节瘤患者的基因突变和表型关联:一项多中心研究和文献综述。

IF 4 2区 医学 Q1 MEDICAL LABORATORY TECHNOLOGY Annals of Laboratory Medicine Pub Date : 2024-11-01 Epub Date: 2024-07-29 DOI:10.3343/alm.2023.0376
Kwan Hoon Jo, Jaewoong Lee, Jaeeun Yoo, Hoon Seok Kim, Eun Sook Kim, Je Ho Han, Yi Sun Jang, Jae-Seung Yun, Jang Won Son, Soon Jib Yoo, Seung Hwan Lee, Dong Jun Lim, Hyuk-Sang Kwon, Seungok Lee, Sungdae Moon, Myungshin Kim
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引用次数: 0

摘要

建议对所有嗜铬细胞瘤和副神经节瘤(PPGL)患者进行基因检测,以确定基因型与表型之间的关联。我们利用新一代测序技术(NGS),针对 38 个 PPGL 相关基因(包括韩国 PPGL 工作组推荐的基因),对韩国六家大学医院的 59 名 PPGL 患者的种系突变进行了调查。在 13 例患者(22%)中发现了种系突变,受 4 个基因影响:RET、NF1、VHL和SDHD。种系突变与 PPGL 家族史、较小的肿瘤大小以及其他类型肿瘤的存在有明显关联。我们利用文献综述中发现的 95 例韩国 PPGL 胚系突变病例和我们队列中的 13 例病例,描述了基因型与表型的相关性。在RET(密码子631和634)、VHL(157和167)和SDHB(131和253)的特定密码子中发现了突变热点。NF1 的突变情况各不相同,表明不存在共同的热点。这些发现突显了针对韩国 PPGL 患者推荐的 NGS 面板的有效性,以及基因检测在确定临床管理和个性化治疗策略方面的重要性。
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Germline Mutations and Phenotypic Associations in Korean Patients With Pheochromocytoma and Paraganglioma: A Multicenter Study and Literature Review.

Genetic testing is recommended for all patients with pheochromocytomas and paragangliomas (PPGL) to establish genotype-phenotype associations. We investigated germline mutations in 59 patients with PPGL at six Korean university hospitals using next-generation sequencing (NGS) targeting 38 PPGL-associated genes, including those recommended by the Korean PPGL Task Force. Germline mutations were identified in 13 patients (22%), and affected four genes: RET, NF1, VHL, and SDHD. Germline mutations were significantly associated with a family history of PPGL, smaller tumor size, and the presence of other types of tumors. Using 95 Korean PPGL cases with germline mutations identified through a literature review and 13 cases from our cohort, we characterized genotype-phenotype correlations. Mutation hotspots were identified in specific codons of RET (codons 631 and 634), VHL (157 and 167), and SDHB (131 and 253). NF1 mutations varied, indicating the absence of common hotspots. These findings highlight the efficacy of the recommended NGS panel for Korean patients with PPGL and the importance of genetic testing in establishing clinical management and personalized therapeutic strategies.

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来源期刊
Annals of Laboratory Medicine
Annals of Laboratory Medicine MEDICAL LABORATORY TECHNOLOGY-
CiteScore
8.30
自引率
12.20%
发文量
100
审稿时长
6-12 weeks
期刊介绍: Annals of Laboratory Medicine is the official journal of Korean Society for Laboratory Medicine. The journal title has been recently changed from the Korean Journal of Laboratory Medicine (ISSN, 1598-6535) from the January issue of 2012. The JCR 2017 Impact factor of Ann Lab Med was 1.916.
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