• 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

魏斯-克鲁什卡综合征的七种新变体及表型扩展

Seven Novel Variants of Weiss-Kruszka Syndrome and Phenotype Expansion.

作者信息

Hau Anna, Baxter Anne, Chandler Kate, Fennell Andrew, Hsieh Tzung-Chien, Krawitz Peter M, Pinner Jason, Goel Himanshu

机构信息

Hunter Genetics, Hunter New England Health Service, Newcastle, Australia.

Manchester Centre for Genomic Medicine, Manchester University NHS Foundation Trust, Saint Mary's Hospital, Manchester, UK.

出版信息

Am J Med Genet A. 2025 Feb;197(2):e63856. doi: 10.1002/ajmg.a.63856. Epub 2024 Sep 17.

DOI:10.1002/ajmg.a.63856
PMID:39287049
Abstract

Weiss-Kruszka syndrome (WKS) is a rare genetic disorder characterized by metopic ridging, ptosis, arched eyebrows, down slanting palpebral fissures, abnormalities in the corpus callosum, cardiac malformations, and variable neurodevelopmental delay. To date, 32 individuals with a diagnosis of WKS have been reported in the literature. The syndrome is caused by a heterozygous pathogenic variant in the ZNF462 gene or a deletion of the 9p31.2 region involving ZNF462. There is significant phenotypic heterogeneity and intrafamilial variability among these patients. Our study reviewed nine patients from seven unrelated families and identified seven novel heterozygous ZNF462 variants through exome sequencing. GestaltMatcher analysis of our cohort's facial images, alongside previously published images of ZNF462 patients, demonstrated a high degree of facial similarity. Further longitudinal research is needed to delineate this rare condition's long-term health implications and adult-onset features.

摘要

魏斯-克鲁什卡综合征(WKS)是一种罕见的遗传性疾病,其特征包括额缝隆起、上睑下垂、眉弓高耸、睑裂向下倾斜、胼胝体异常、心脏畸形以及不同程度的神经发育迟缓。迄今为止,文献中已报道了32例诊断为WKS的个体。该综合征由ZNF462基因的杂合致病性变异或涉及ZNF462的9p31.2区域缺失引起。这些患者之间存在显著的表型异质性和家族内变异性。我们的研究回顾了来自七个无关家族的九名患者,并通过外显子组测序鉴定出七个新的ZNF462杂合变异。对我们队列的面部图像进行的GestaltMatcher分析,以及之前发表的ZNF462患者的图像,显示出高度的面部相似性。需要进一步的纵向研究来描述这种罕见疾病对长期健康的影响和成人发病特征。

相似文献

1
Seven Novel Variants of Weiss-Kruszka Syndrome and Phenotype Expansion.魏斯-克鲁什卡综合征的七种新变体及表型扩展
Am J Med Genet A. 2025 Feb;197(2):e63856. doi: 10.1002/ajmg.a.63856. Epub 2024 Sep 17.
2
Phenotypic spectrum in Weiss-Kruszka syndrome caused by ZNF462 variants: Three new patients and literature review.Weiss-Kruszka 综合征表型谱:ZNF462 变异所致三例新病例及文献复习。
Eur J Med Genet. 2024 Oct;71:104964. doi: 10.1016/j.ejmg.2024.104964. Epub 2024 Jul 26.
3
Analysis of clinical phenotypes and genetic variations in two pedigrees affected with Weiss-Kruszka syndrome.分析两例 Weiss-Kruszka 综合征家系的临床表型和遗传变异。
BMC Med Genomics. 2024 Nov 5;17(1):261. doi: 10.1186/s12920-024-02035-x.
4
A novel mutation in the gene c.3306dup; p.(Gln1103Thrfs*10) is associated to Weiss-Kruszka syndrome. A case report.基因 c.3306dup;p.(Gln1103Thrfs*10) 中的一个新突变与 Weiss-Kruszka 综合征相关。病例报告。
Acta Clin Belg. 2022 Feb;77(1):118-121. doi: 10.1080/17843286.2020.1780391. Epub 2020 Jun 16.
5
Phenotype delineation of ZNF462 related syndrome.ZNF462 相关综合征的表型描绘。
Am J Med Genet A. 2019 Oct;179(10):2075-2082. doi: 10.1002/ajmg.a.61306. Epub 2019 Jul 30.
6
Further characterization of the 9q31 microdeletion phenotype; delineation of a common region of overlap containing ZNF462.进一步描述 9q31 微缺失表型;描绘包含 ZNF462 的重叠常见区域。
Mol Genet Genomic Med. 2023 Mar;11(3):e2116. doi: 10.1002/mgg3.2116. Epub 2022 Dec 3.
7
Empty Sella Syndrome Associated with Growth Hormone Deficiency: the First Case Report of Weiss-Kruszka Syndrome.空蝶鞍综合征合并生长激素缺乏:Weiss-Kruszka 综合征首例报告。
J Korean Med Sci. 2021 May 10;36(18):e133. doi: 10.3346/jkms.2021.36.e133.
8
Acute lymphoblastic leukemia in a child with Weiss-Kruszka syndrome: Casual or causal association?儿童魏斯-克鲁斯卡综合征伴急性淋巴细胞白血病:偶然关联还是因果关联?
Eur J Med Genet. 2022 Apr;65(4):104457. doi: 10.1016/j.ejmg.2022.104457. Epub 2022 Feb 16.
9
Haploinsufficiency of ZNF462 is associated with craniofacial anomalies, corpus callosum dysgenesis, ptosis, and developmental delay.ZNF462单倍剂量不足与颅面异常、胼胝体发育不全、上睑下垂和发育迟缓有关。
Eur J Hum Genet. 2017 Aug;25(8):946-951. doi: 10.1038/ejhg.2017.86. Epub 2017 May 17.
10
Kallmann syndrome in a patient with Weiss-Kruszka syndrome and a de novo deletion in 9q31.2.一名患有魏斯-克鲁什卡综合征且9q31.2存在新发缺失的患者的卡尔曼综合征。
Eur J Endocrinol. 2021 May 21;185(1):57-66. doi: 10.1530/EJE-20-1387.

引用本文的文献

1
First Report of a Novel Variant Linked to Weiss-Kruszka Syndrome and Congenital Diaphragmatic Hernia: Insights into Potential Additional Malformations.与魏斯-克鲁什卡综合征和先天性膈疝相关的新型变异的首次报告:对潜在其他畸形的见解
Mol Syndromol. 2025 May 2:1-6. doi: 10.1159/000546167.