拉丁美洲嗜铬细胞瘤和副神经节瘤的个性化治疗:遗传学视角。

Felipe Freitas-Castro, Madson Q Almeida
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引用次数: 0

摘要

辉铬细胞瘤和副神经节瘤(PPGLs)是一种罕见的神经内分泌肿瘤,具有临床异质性,与遗传性疾病高度相关,约占病例的 30%。中国和欧洲患者在 PPGLs 的表现和遗传病因方面存在差异。与其他队列(中国 21%、欧洲 31% 和癌症基因组图谱计划队列 27%)相比,巴西患者的种系遗传诊断频率明显更高(50%)。有趣的是,与其他队列(3%-9%)相比,种系SDHB基因缺陷在巴西PPGL患者(17%)中也更为普遍。在巴西PPGLs患者中,SDHB外显子1缺失导致的SDHB致病/可能致病变异约占50%,这是由于创始效应所致。种系 SDHB 外显子 1 缺失占巴西(可能也包括拉丁美洲)患者种系驱动因子的 10%。因此,SDHB 1 号外显子缺失的单一诊断 PCR 可能对拉丁美洲 PPGL 患者的基因检测和咨询临床实践非常有用。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Personalized management for phaeochromocytomas and paragangliomas in Latin America: A genetic perspective.

Phaeochromocytomas and paragangliomas (PPGLs) are rare neuroendocrine tumors with clinical heterogeneity and a high association with hereditary disease, affecting approximately 30 % of the cases. Differences in the presentation and genetic etiologies of PPGLs have been demonstrated between Chinese and European patients. The frequency of germline genetic diagnosis was remarkably higher in Brazilian patients (∼50 %) compared with other cohorts (Chinese 21 %, European 31 %, and The Cancer Genome Atlas Program cohort 27 %). Interestingly, germline SDHB genetic defects were also more prevalent in Brazilian patients (17 %) with PPGLs when compared with other cohorts (3-9 %). The SDHB exon 1 deletion was responsible for approximately 50 % of the SDHB pathogenic/likely pathogenic variants in Brazilian patients with PPGLs due to a founder effect. The germline SDHB exon 1 deletion represents ∼10 % of the germline drivers in Brazilian patients (and possibly in Latin America). Therefore, a single diagnostic PCR for the SDHB exon 1 deletion might be very useful in clinical practice for genetic testing and counseling of patients with PPGLs in Latin America.

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