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遗传性转甲状腺素蛋白淀粉样变性热点区域的基因检测及奠基者突变证据

Genetic testing and evidence of a founder mutation in a hotspot for hereditary transthyretin amyloidosis.

作者信息

Ventayol-Guirado Marc, Cisneros-Barroso Eugenia, Ribot-Sanso Maria Antonia, Gonzalez-Moreno Juan, Losada Ines, Ripoll-Vera Tomas, Pons Jaume, Fortuny Elena, Bosch Teresa, Figuerola Antonio, Descals Cristina, Montala Joan Carles, Alvarez-Rubio Jorge, Hernandez-Rodriguez Jessica, Lustre-Rodriguez Jose, Llull-Alberti Maria Victoria, Jimenez-Barcelo Juan Antonio, Asensio-Landa Victor Jose, Torres-Juan Laura, Martinez-Lopez Iciar, Buades-Reines Juan, Heine-Suñer Damian

机构信息

Genomics of Health Research Group, Health Research Institute of the Balearic Islands (IdISBa), Palma, Balearic Islands, Spain.

Balearic Research Group in Genetic Cardiopathies, Sudden Death and TTR Amyloidosis. Health Research Institute of the Balearic Islands (IdISBa), Palma, Balearic Islands, Spain.

出版信息

Sci Rep. 2025 Aug 14;15(1):29773. doi: 10.1038/s41598-025-14707-4.

Abstract

Variant transthyretin (ATTRv) amyloidosis is a rare, inherited disorder caused by mutations in the TTR gene, leading to amyloid fibril deposition. The Balearic Islands are a known endemic focus for the NP_000362.1:p.Val50Met (V30M) variant, one of the most prevalent pathogenic mutations. We analysed 23 years of genetic testing data from the Balearic Islands' public health system to determine the prevalence and distribution of ATTR pathogenic variants, with a focus on V30M. A total of 1,478 individuals underwent genetic testing, with 319 positive tests for ATTRv: 308 carried the V30M variant (96.4%) and 11 carried other pathogenic variants. The V30M prevalence was highest in Mallorca and Menorca (1 in 2,900 and 4,700, respectively), with lower rates in Ibiza and Formentera. The co-occurrence of V30M and NP_000362.1:p.Gly26Ser (G6S) on the same chromosome in 93% of V30M carriers suggests a common origin. This study becomes the first registry of ATTRv in the Balearic Islands, aiming to raise awareness among clinicians across all areas and services while enabling more accurate diagnostics, informed genetic counselling, and targeted clinical follow-ups. We also postulate the Balearic Islands as a major global focus for the V30M variant, with a distinct genetic profile suggesting a unique founder effect within the region.

摘要

变异型转甲状腺素蛋白(ATTRv)淀粉样变性是一种罕见的遗传性疾病,由TTR基因突变引起,导致淀粉样纤维沉积。巴利阿里群岛是NP_000362.1:p.Val50Met(V30M)变异型的已知地方性聚集区,该变异型是最常见的致病突变之一。我们分析了巴利阿里群岛公共卫生系统23年的基因检测数据,以确定ATTR致病变异型的患病率和分布情况,重点关注V30M。共有1478人接受了基因检测,其中319人ATTRv检测呈阳性:308人携带V30M变异型(96.4%),11人携带其他致病变异型。V30M患病率在马略卡岛和梅诺卡岛最高(分别为1/2900和1/4700),伊维萨岛和福门特拉岛的患病率较低。93%的V30M携带者中,V30M和NP_000362.1:p.Gly26Ser(G6S)在同一条染色体上同时出现,这表明它们有共同的起源。本研究成为巴利阿里群岛首个ATTRv登记册,旨在提高所有领域和服务的临床医生的认识,同时实现更准确的诊断、提供充分信息的遗传咨询和有针对性的临床随访。我们还推测巴利阿里群岛是V30M变异型的主要全球聚集区,其独特的基因特征表明该地区存在独特的奠基者效应。

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