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在中国一个遗传性头颈部副神经节瘤家系中鉴定出致病性SDHD突变:对遗传咨询和管理的意义。

Identification of a pathogenic SDHD mutation in a Chinese family with hereditary head and neck paraganglioma: implications for genetic counseling and management.

作者信息

Wang Pu, Gao Liming, Zhang Wenyang, Guo Rui, Xia Yin

机构信息

Department of Otorhinolaryngology Head and Neck Surgery, Beijing Tiantan Hospital, Capital Medical University, No. 119, South Fourth Ring Road West, Fengtai District, Beijing, 100070, China.

出版信息

World J Surg Oncol. 2025 Jan 3;23(1):4. doi: 10.1186/s12957-024-03641-w.

Abstract

BACKGROUND

This study aims to identify a pathogenic SDHD mutation associated with hereditary head and neck paraganglioma (HNPGL) in a Chinese family and to explore its implications for genetic counseling.

METHODS

The study involved a family with 15 members spanning three generations. A 31-year-old patient (II-4) was diagnosed with a left parotid gland tumor and a right carotid body tumor, while both the father and elder sister had right carotid body tumors, and the third sister had bilateral carotid body tumors. Whole exome sequencing and Sanger sequencing were employed to identify candidate pathogenic variants. Genetic counseling was conducted for third-generation descendants to assess the likelihood of carrying the mutation and to guide future diagnosis and treatment.

RESULTS

A nonsense mutation in the SDHD gene (NM_001276503:exon2:c.C64T: p.R22X) was identified in the patient and three other affected family members. Genetic counseling for the third generation revealed that only one child (III-4) carried the pathogenic mutation inherited from the patient's third sister.

CONCLUSION

We identified a pathogenic mutation in SDHD in a Chinese HNPGL family, which is the second reported case of its kind. Our genetic counseling analysis for the third generation provided important information for the family and guidance for future diagnosis and treatment.

摘要

背景

本研究旨在鉴定一个中国家族中与遗传性头颈部副神经节瘤(HNPGL)相关的致病性SDHD突变,并探讨其在遗传咨询中的意义。

方法

该研究涉及一个三代共15名成员的家族。一名31岁患者(II-4)被诊断患有左侧腮腺肿瘤和右侧颈动脉体瘤,其父亲和姐姐均患有右侧颈动脉体瘤,三姐患有双侧颈动脉体瘤。采用全外显子组测序和桑格测序来鉴定候选致病性变异。对第三代后代进行遗传咨询,以评估携带该突变的可能性,并指导未来的诊断和治疗。

结果

在该患者及其他三名受影响的家族成员中鉴定出SDHD基因的一个无义突变(NM_001276503:exon2:c.C64T:p.R22X)。对第三代的遗传咨询显示,只有一名儿童(III-4)携带从患者三姐遗传而来的致病性突变。

结论

我们在一个中国HNPGL家族中鉴定出SDHD的一个致病性突变,这是该类报道的第二例。我们对第三代的遗传咨询分析为该家族提供了重要信息,并为未来的诊断和治疗提供了指导。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8ea/11697643/18d8d1d6a649/12957_2024_3641_Fig1_HTML.jpg

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