• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

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.

DOI:10.1186/s12957-024-03641-w
PMID:39754145
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11697643/
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/11697643/67a0ddfa60cd/12957_2024_3641_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8ea/11697643/18d8d1d6a649/12957_2024_3641_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8ea/11697643/9560cfede26b/12957_2024_3641_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8ea/11697643/67a0ddfa60cd/12957_2024_3641_Fig3_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8ea/11697643/18d8d1d6a649/12957_2024_3641_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8ea/11697643/9560cfede26b/12957_2024_3641_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f8ea/11697643/67a0ddfa60cd/12957_2024_3641_Fig3_HTML.jpg

相似文献

1
Identification of a pathogenic SDHD mutation in a Chinese family with hereditary head and neck paraganglioma: implications for genetic counseling and management.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。
World J Surg Oncol. 2025 Jan 3;23(1):4. doi: 10.1186/s12957-024-03641-w.
2
Hereditary paraganglioma due to the SDHD M1I mutation in a second Chinese family: a founder effect?第二个中国家系中因SDHD M1I突变导致的遗传性副神经节瘤:奠基者效应?
Laryngoscope. 2003 Jun;113(6):1055-8. doi: 10.1097/00005537-200306000-00026.
3
The spectrum of SDHD mutations in Russian patients with head and neck paraganglioma.俄罗斯头颈部副神经节瘤患者中SDHD基因突变谱
Int J Neurosci. 2018 Dec;128(12):1174-1179. doi: 10.1080/00207454.2018.1503181. Epub 2018 Oct 30.
4
Germline mutations and genotype-phenotype associations in head and neck paraganglioma patients with negative family history in China.中国无家族史的头颈部副神经节瘤患者的胚系突变及基因型-表型关联
Eur J Med Genet. 2015 Sep;58(9):433-8. doi: 10.1016/j.ejmg.2015.05.008. Epub 2015 Jun 19.
5
SDHx gene detection and clinical Phenotypic analysis of multiple paraganglioma in the head and neck.头颈部多发副神经节瘤的SDHx基因检测及临床表型分析
Laryngoscope. 2019 Feb;129(2):E67-E71. doi: 10.1002/lary.27509. Epub 2018 Nov 28.
6
Case report of a man with multiple paragangliomas and pathogenic germline variants in both NF1 and SDHD.一名患有多种副神经节瘤并携带 NF1 和 SDHD 种系变异的男性病例报告。
Cancer Genet. 2021 Aug;256-257:110-114. doi: 10.1016/j.cancergen.2021.05.008. Epub 2021 May 25.
7
A novel succinate dehydrogenase subunit B germline variant associated with head and neck paraganglioma in a Dutch kindred: A family-based study.一个与荷兰家系头颈部副神经节瘤相关的新型琥珀酸脱氢酶亚基 B 种系变异:基于家系的研究。
Clin Otolaryngol. 2018 Jun;43(3):841-845. doi: 10.1111/coa.13059. Epub 2018 Jan 25.
8
Malignant phenotype and two SDHD mutations in a family with paraganglioma syndrome type 1.1型副神经节瘤综合征家族中的恶性表型和两个SDHD突变
Genet Res (Camb). 2015 Mar 30;97:e3. doi: 10.1017/S0016672315000063.
9
The R22X mutation of the SDHD gene in hereditary paraganglioma abolishes the enzymatic activity of complex II in the mitochondrial respiratory chain and activates the hypoxia pathway.遗传性副神经节瘤中SDHD基因的R22X突变消除了线粒体呼吸链中复合物II的酶活性,并激活了缺氧途径。
Am J Hum Genet. 2001 Dec;69(6):1186-97. doi: 10.1086/324413. Epub 2001 Oct 16.
10
Evaluation of SDHB, SDHD and VHL gene susceptibility testing in the assessment of individuals with non-syndromic phaeochromocytoma, paraganglioma and head and neck paraganglioma.评估非综合征性嗜铬细胞瘤、副神经节瘤和头颈部副神经节瘤个体中 SDHB、SDHD 和 VHL 基因易感性检测的价值。
Clin Endocrinol (Oxf). 2013 Jun;78(6):898-906. doi: 10.1111/cen.12074. Epub 2013 Apr 6.

本文引用的文献

1
Clinical management and outcome of head and neck paragangliomas (HNPGLs): A single centre retrospective study.头颈部副神经节瘤(HNPGLs)的临床管理和预后:单中心回顾性研究。
Clin Endocrinol (Oxf). 2024 Sep;101(3):243-248. doi: 10.1111/cen.15070. Epub 2024 May 2.
2
Inhibition of Succinate Dehydrogenase by Pesticides (SDHIs) and Energy Metabolism.抑制琥珀酸脱氢酶的杀虫剂(SDHIs)和能量代谢。
Int J Mol Sci. 2023 Feb 17;24(4):4045. doi: 10.3390/ijms24044045.
3
Paraganglioma of the Head and Neck: A Review.头颈部副神经节瘤:综述。
Endocr Pract. 2023 Feb;29(2):141-147. doi: 10.1016/j.eprac.2022.10.002. Epub 2022 Oct 15.
4
Update from the 5th Edition of the World Health Organization Classification of Head and Neck Tumors: Overview of the 2022 WHO Classification of Head and Neck Neuroendocrine Neoplasms.第五版世界卫生组织头颈部肿瘤分类更新:2022 年世界卫生组织头颈部神经内分泌肿瘤分类概述。
Head Neck Pathol. 2022 Mar;16(1):123-142. doi: 10.1007/s12105-022-01435-8. Epub 2022 Mar 21.
5
Paragangliomas of the head and neck.头颈部副神经节瘤
J Oral Pathol Med. 2022 Nov;51(10):897-903. doi: 10.1111/jop.13286. Epub 2022 Feb 23.
6
SDHC phaeochromocytoma and paraganglioma: A UK-wide case series.SDHC相关性嗜铬细胞瘤和副神经节瘤:一项全英国病例系列研究。
Clin Endocrinol (Oxf). 2022 Apr;96(4):499-512. doi: 10.1111/cen.14594. Epub 2021 Sep 24.
7
The Impact Of Gene (SDH) Mutations In Renal Cell Carcinoma (RCC): A Systematic Review.基因(SDH)突变在肾细胞癌(RCC)中的影响:一项系统综述。
Onco Targets Ther. 2019 Sep 26;12:7929-7940. doi: 10.2147/OTT.S207460. eCollection 2019.
8
Variant type is associated with disease characteristics in SDHB, SDHC and SDHD-linked phaeochromocytoma-paraganglioma.变异类型与 SDHB、SDHC 和 SDHD 相关的嗜铬细胞瘤-副神经节瘤的疾病特征有关。
J Med Genet. 2020 Feb;57(2):96-103. doi: 10.1136/jmedgenet-2019-106214. Epub 2019 Sep 6.
9
A systematic review on the genetic analysis of paragangliomas: primarily focused on head and neck paragangliomas.一篇关于副神经节瘤遗传分析的系统综述:主要侧重于头颈部副神经节瘤。
Neoplasma. 2019 Jun 29;66(5):671-680. doi: 10.4149/neo_2018_181208N933. Print 2019 Sep.
10
Genetics and imaging of pheochromocytomas and paragangliomas: current update.嗜铬细胞瘤和副神经节瘤的遗传学和影像学:最新研究进展。
Abdom Radiol (NY). 2020 Apr;45(4):928-944. doi: 10.1007/s00261-019-02044-w.