遗传性血管性水肿的诊断和治疗现状及未来发展。

IF 3.8 4区 医学 Q1 DERMATOLOGY
Andreas Recke
{"title":"遗传性血管性水肿的诊断和治疗现状及未来发展。","authors":"Andreas Recke","doi":"10.1111/ddg.15889","DOIUrl":null,"url":null,"abstract":"<p><p>Hereditary angioedema (HAE) is a rare hereditary disease characterized by edema, which can be life-threatening in case of swelling in the larynx. The most common form of HAE is caused by a mutation of the SERPING1 gene and is characterized by a deficiency (type I) or loss of function (type II) of the C1 inhibitor (C1-INH), leading to excessive production of bradykinin. In contrast, the HAE-nC1-INH entity is associated with a normal C1-INH protein and is caused by mutations in other genes. Because HAE is a rare and often underdiagnosed disease, it may take years from symptom onset to diagnosis. The angioedema attacks cause suffering and affect both the ability to work and quality of life (QoL). The treatment of HAE includes attack treatment (on-demand), short-term prophylaxis (e.g., before medical procedures), and long-term prophylaxis. Four first-line treatment options for long-term prophylaxis are available, effectively preventing attacks and supporting the guideline goal of complete disease control. Further treatment options, including CRISPR/Cas9-based gene therapy, are under development and promise to provide individually tailored treatment for patients. This review aims to provide an overview of the clinical presentation, diagnosis, and treatment of HAE.</p>","PeriodicalId":14758,"journal":{"name":"Journal Der Deutschen Dermatologischen Gesellschaft","volume":" ","pages":""},"PeriodicalIF":3.8000,"publicationDate":"2025-09-04","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"Status quo and future developments in the diagnosis and treatment of hereditary angioedema.\",\"authors\":\"Andreas Recke\",\"doi\":\"10.1111/ddg.15889\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Hereditary angioedema (HAE) is a rare hereditary disease characterized by edema, which can be life-threatening in case of swelling in the larynx. The most common form of HAE is caused by a mutation of the SERPING1 gene and is characterized by a deficiency (type I) or loss of function (type II) of the C1 inhibitor (C1-INH), leading to excessive production of bradykinin. In contrast, the HAE-nC1-INH entity is associated with a normal C1-INH protein and is caused by mutations in other genes. Because HAE is a rare and often underdiagnosed disease, it may take years from symptom onset to diagnosis. The angioedema attacks cause suffering and affect both the ability to work and quality of life (QoL). The treatment of HAE includes attack treatment (on-demand), short-term prophylaxis (e.g., before medical procedures), and long-term prophylaxis. Four first-line treatment options for long-term prophylaxis are available, effectively preventing attacks and supporting the guideline goal of complete disease control. Further treatment options, including CRISPR/Cas9-based gene therapy, are under development and promise to provide individually tailored treatment for patients. This review aims to provide an overview of the clinical presentation, diagnosis, and treatment of HAE.</p>\",\"PeriodicalId\":14758,\"journal\":{\"name\":\"Journal Der Deutschen Dermatologischen Gesellschaft\",\"volume\":\" \",\"pages\":\"\"},\"PeriodicalIF\":3.8000,\"publicationDate\":\"2025-09-04\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal Der Deutschen Dermatologischen Gesellschaft\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1111/ddg.15889\",\"RegionNum\":4,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q1\",\"JCRName\":\"DERMATOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal Der Deutschen Dermatologischen Gesellschaft","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1111/ddg.15889","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"DERMATOLOGY","Score":null,"Total":0}
引用次数: 0

摘要

遗传性血管性水肿(HAE)是一种罕见的以水肿为特征的遗传性疾病,在喉部肿胀的情况下可危及生命。最常见的HAE是由SERPING1基因突变引起的,其特征是C1抑制剂(C1- inh)缺乏(I型)或功能丧失(II型),导致缓激肽过量产生。相反,HAE-nC1-INH实体与正常的C1-INH蛋白相关,并由其他基因突变引起。由于HAE是一种罕见且常被误诊的疾病,从症状发作到诊断可能需要数年时间。血管性水肿发作造成痛苦,影响工作能力和生活质量(QoL)。HAE的治疗包括发作性治疗(按需)、短期预防(如医疗程序前)和长期预防。目前有四种一线治疗方案可用于长期预防,有效地预防疾病发作,并支持完全控制疾病的指导目标。进一步的治疗方案,包括基于CRISPR/ cas9的基因治疗,正在开发中,并有望为患者提供个性化的治疗。本文综述了HAE的临床表现、诊断和治疗。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Status quo and future developments in the diagnosis and treatment of hereditary angioedema.

Hereditary angioedema (HAE) is a rare hereditary disease characterized by edema, which can be life-threatening in case of swelling in the larynx. The most common form of HAE is caused by a mutation of the SERPING1 gene and is characterized by a deficiency (type I) or loss of function (type II) of the C1 inhibitor (C1-INH), leading to excessive production of bradykinin. In contrast, the HAE-nC1-INH entity is associated with a normal C1-INH protein and is caused by mutations in other genes. Because HAE is a rare and often underdiagnosed disease, it may take years from symptom onset to diagnosis. The angioedema attacks cause suffering and affect both the ability to work and quality of life (QoL). The treatment of HAE includes attack treatment (on-demand), short-term prophylaxis (e.g., before medical procedures), and long-term prophylaxis. Four first-line treatment options for long-term prophylaxis are available, effectively preventing attacks and supporting the guideline goal of complete disease control. Further treatment options, including CRISPR/Cas9-based gene therapy, are under development and promise to provide individually tailored treatment for patients. This review aims to provide an overview of the clinical presentation, diagnosis, and treatment of HAE.

求助全文
通过发布文献求助,成功后即可免费获取论文全文。 去求助
来源期刊
CiteScore
3.50
自引率
25.00%
发文量
406
审稿时长
1 months
期刊介绍: The JDDG publishes scientific papers from a wide range of disciplines, such as dermatovenereology, allergology, phlebology, dermatosurgery, dermatooncology, and dermatohistopathology. Also in JDDG: information on medical training, continuing education, a calendar of events, book reviews and society announcements. Papers can be submitted in German or English language. In the print version, all articles are published in German. In the online version, all key articles are published in English.
×
引用
GB/T 7714-2015
复制
MLA
复制
APA
复制
导出至
BibTeX EndNote RefMan NoteFirst NoteExpress
×
提示
您的信息不完整,为了账户安全,请先补充。
现在去补充
×
提示
您因"违规操作"
具体请查看互助需知
我知道了
×
提示
确定
请完成安全验证×
copy
已复制链接
快去分享给好友吧!
我知道了
右上角分享
点击右上角分享
0
联系我们:info@booksci.cn Book学术提供免费学术资源搜索服务,方便国内外学者检索中英文文献。致力于提供最便捷和优质的服务体验。 Copyright © 2023 布克学术 All rights reserved.
京ICP备2023020795号-1
ghs 京公网安备 11010802042870号
Book学术文献互助
Book学术文献互助群
群 号:604180095
Book学术官方微信