• 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

常染色体显性遗传皮肤病在儿童期出现重叠皮损而被发现。

Autosomal dominant genodermatoses in adults being heralded by superimposed skin lesions in children.

机构信息

Department of Dermatology, Medical Center-University of Freiburg, Freiburg, Germany.

出版信息

Am J Med Genet C Semin Med Genet. 2023 Jun;193(2):109-115. doi: 10.1002/ajmg.c.32055. Epub 2023 Jun 8.

DOI:10.1002/ajmg.c.32055
PMID:37288730
Abstract

In autosomal dominant skin disorders, pronounced mosaic involvement may sometimes occur in the neonate, originating in a heterozygous embryo from early loss of heterozygosity, probably during the first week after fertilization. In biallelic phenotypes, such overlaying mosaic involvement may coexist with disseminated mosaicism, for example, in neurofibromatosis or tuberous sclerosis. In other phenotypes, however, classical nonsegmental involvement tends to appear much later, which is why the superimposed mosaic is a heralding feature. In Brooke-Spiegler syndrome (eccrine cylindromatosis), a large pedigree documented a 5-year-old boy with multiple, congenital small eccrine cylindromas along the lines of Blaschko. Disseminated cylindromas were absent because they usually appear in adulthood. ̶ In Hornstein-Knickenberg syndrome, an affected woman had an 8-year-old son with a nevus comedonicus-like lesion exemplifying a forerunner of the syndrome. ("Birt-Hogg-Dubé syndrome" represents a nonsyndromic type of hereditary perifollicular fibromas.) In glomangiomatosis, neonatal superimposed mosaicism is a heralding feature because disseminated lesions appear during puberty or adulthood. Linear porokeratosis is a harbinger of disseminated porokeratosis that develops 30 or 40 years later. ̶ Cases of superimposed linear Darier disease were forerunners of nonsegmental manifestation. ̶ In a case of Hailey-Hailey disease, neonatal mosaic lesions heralded nonsegmental involvement that began 22 years later.

摘要

在常染色体显性皮肤疾病中,杂合子胚胎在受精后第一周内可能由于早期杂合性丢失而出现明显的镶嵌性受累,新生儿中有时会出现这种情况。在双等位基因表型中,这种重叠的镶嵌性受累可能与弥漫性镶嵌性共存,例如神经纤维瘤病或结节性硬化症。然而,在其他表型中,经典的非节段性受累往往出现得更晚,这就是为什么重叠的镶嵌性是一个预示特征。在布鲁克-斯皮格尔综合征(大汗腺瘤病)中,一个大型家系记录了一名 5 岁男孩,他沿着 Blaschko 线有多发性先天性小汗腺圆柱瘤。弥漫性圆柱瘤不存在,因为它们通常在成年后出现。在霍恩斯坦-克尼克伯格综合征中,一名受影响的女性有一个 8 岁的儿子,他有一个类似于粉刺瘤样的病变,这是该综合征的先驱。(“Birt-Hogg-Dubé 综合征”代表一种遗传性毛囊周围纤维瘤的非综合征型。)在血管球瘤病中,新生儿重叠镶嵌性是一个预示特征,因为弥漫性病变在青春期或成年期出现。线状角层松解症是随后出现的弥漫性角层松解症的先兆,后者在 30 或 40 年后发展。重叠的线状 Darier 病病例是节段性表现的先驱。在一例 Hailey-Hailey 病中,新生儿镶嵌性病变预示着随后 22 年开始的非节段性受累。

相似文献

1
Autosomal dominant genodermatoses in adults being heralded by superimposed skin lesions in children.常染色体显性遗传皮肤病在儿童期出现重叠皮损而被发现。
Am J Med Genet C Semin Med Genet. 2023 Jun;193(2):109-115. doi: 10.1002/ajmg.c.32055. Epub 2023 Jun 8.
2
[Segmental type 2 manifestation of autosome dominant skin diseases. Development of a new formal genetic concept].[常染色体显性遗传性皮肤病的节段性2型表现。一种新的正式遗传学概念的发展]
Hautarzt. 2001 Apr;52(4):283-7. doi: 10.1007/s001050051309.
3
The categories of cutaneous mosaicism: A proposed classification.皮肤镶嵌现象的类别:一项提议的分类法。
Am J Med Genet A. 2016 Feb;170A(2):452-459. doi: 10.1002/ajmg.a.37439. Epub 2015 Oct 22.
4
Darier disease with paired segmental manifestation of either excessive or absent involvement: a further step in the concept of twin spotting.具有受累过度或未受累配对节段性表现的 Darier 病:孪生斑概念的进一步拓展。
Dermatology. 2002;205(4):344-7. doi: 10.1159/000066423.
5
Progressive Osseous Heteroplasia is not an Autosomal Dominant Trait but Reflects Superimposed Mosaicism in Different Inactivation Disorders.进行性骨化性异生并非常染色体显性性状,而是反映了不同失活疾病中叠加的镶嵌现象。
Indian Dermatol Online J. 2021 Mar 2;12(2):316-318. doi: 10.4103/idoj.IDOJ_584_20. eCollection 2021 Mar-Apr.
6
Progressive osseous heteroplasia is not a Mendelian trait but a type 2 segmental manifestation of GNAS inactivation disorders: A hypothesis.进行性骨化性纤维发育不良并非孟德尔性状,而是GNAS基因失活疾病的2型节段性表现:一种假说。
Eur J Med Genet. 2016 May;59(5):290-4. doi: 10.1016/j.ejmg.2016.04.001. Epub 2016 Apr 4.
7
Linear porokeratosis superimposed on disseminated superficial actinic porokeratosis: report of two cases exemplifying the concept of type 2 segmental manifestation of autosomal dominant skin disorders.线状汗孔角化症叠加于播散性浅表性光化性汗孔角化症:两例报告,例证常染色体显性遗传性皮肤病2型节段性表现的概念
J Am Acad Dermatol. 1999 Oct;41(4):644-7.
8
Mibelli revisited: a case of type 2 segmental porokeratosis from 1893.米贝利病再探:1893 年报告的 2 型节段性先天性角化不良 1 例。
J Am Acad Dermatol. 2010 Jan;62(1):136-138. doi: 10.1016/j.jaad.2008.12.049. Epub 2009 Jul 25.
9
Allelic loss underlies type 2 segmental Hailey-Hailey disease, providing molecular confirmation of a novel genetic concept.等位基因缺失是2型节段性黑棘皮病的基础,为一种新的遗传学概念提供了分子学证据。
J Clin Invest. 2004 Nov;114(10):1467-74. doi: 10.1172/JCI21791.
10
A case of linear porokeratosis superimposed on disseminated superficial actinic porokeratosis.1例线状汗孔角化症叠加于播散性浅表性光化性汗孔角化症。
Case Rep Dermatol. 2010 Aug 6;2(2):130-4. doi: 10.1159/000319708.

引用本文的文献

1
Zosteriform eruption in an adult male.一名成年男性的带状疱疹样皮疹。
JAAD Case Rep. 2024 Jan 20;45:79-81. doi: 10.1016/j.jdcr.2024.01.009. eCollection 2024 Mar.