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以色列少数民族中 MUTYH 相关息肉病的高发率与罕见的致病种系变异有关。

High prevalence of MUTYH associated polyposis among minority populations in Israel, due to rare founder pathogenic variants.

机构信息

The Genetics Institute, Rambam Health Care Campus, Haifa, Israel.

Raphael Recanati Genetic Institute, Rabin Medical Center, Petach Tikva, Israel; Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel.

出版信息

Dig Liver Dis. 2023 Jul;55(7):880-887. doi: 10.1016/j.dld.2023.01.151. Epub 2023 Feb 4.

Abstract

BACKGROUND

Autosomal recessive conditions are common in consanguineous populations. Since consanguinity is common in the Israeli Arab population, we evaluated the rate of MUTYH polyposis (MAP) among polyposis patients in this population and studied Pathogenic Variants (PVs) spectrum.

METHODS

We reviewed health records of all Arab and Druze polyposis patients referred for counseling during 2013-2020 who fulfilled the Israeli Genetic Society criteria for MUTYH/APC testing, in a tertiary center in Northern Israel and four additional gastro-genetic clinics in Israel.

RESULTS

The Northern cohort included 37 patients from 30 unrelated families; 8(26.6%) carried bi-allelic MUTYH PVs. The major variant p.Glu452del was detected in 6/8 Druze and Muslim families who shared the same haplotype. Other PVs detected in both cohorts included p.Tyr56Ter, p.His57Arg, c.849+3A>C, p.Ala357fs, and p.Tyr151Cys. Among bi-allelic carriers, 88% reported consanguinity, and 100% had positive family history for polyposis or colorectal cancer (CRC). Generally, the age of CRC was 10 years younger than reported in the general MAP population.

CONCLUSIONS

MAP accounted for 27% of polyposis cases in the Arab population of Northern Israel. PVs spectrum is unique, with high frequency of the founder variant p.Glu452del. Our results may inform the genetic testing strategy in the Israeli Arab population.

摘要

背景

常染色体隐性疾病在近亲人群中较为常见。由于以色列阿拉伯人群中存在近亲结婚的情况,我们评估了该人群中多发性息肉病(MAP)的 MUTYH 多态性(MAP)的发生率,并研究了致病变异(PV)谱。

方法

我们回顾了 2013 年至 2020 年间在以色列北部一家三级中心和以色列另外四家胃肠遗传诊所接受咨询的所有阿拉伯和德鲁兹族息肉病患者的健康记录,这些患者符合 MUTYH/APC 检测的以色列遗传学会标准。

结果

北方队列包括 30 个无关家庭的 37 名患者;8(26.6%)人携带双等位基因 MUTYH PVs。在 6/8 名德鲁兹人和穆斯林家庭中检测到主要变体 p.Glu452del,这些家庭共享相同的单倍型。在两个队列中检测到的其他 PV 包括 p.Tyr56Ter、p.His57Arg、c.849+3A>C、p.Ala357fs 和 p.Tyr151Cys。在双等位基因携带者中,88%有近亲结婚的记录,100%有息肉病或结直肠癌(CRC)的阳性家族史。一般来说,CRC 的发病年龄比一般 MAP 人群中报道的年轻 10 岁。

结论

MAP 占以色列北部阿拉伯人群中息肉病病例的 27%。PV 谱是独特的,具有高频率的创始人变体 p.Glu452del。我们的结果可能为以色列阿拉伯人群的基因检测策略提供信息。

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