• 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

一名Netherton综合征患儿的新型SPINK5突变及皮下免疫球蛋白替代治疗成功案例

A novel SPINK5 mutation and successful subcutaneous immunoglobulin replacement therapy in a child with Netherton syndrome.

作者信息

Zelieskova Maria, Banovcin Peter, Kozar Marek, Kozarova Andrea, Nudzajova Zuzana, Jesenak Milos

机构信息

Department of Paediatrics, Centre for Primary Immunodeficiencies, Jessenius Faculty of Medicine of Comenius University in Bratislava, University Hospital in Martin, Martin, Slovakia.

Department of Neonatology, Jessenius Faculty of Medicine of Comenius University in Bratislava, University Hospital in Martin, Martin, Slovakia.

出版信息

Pediatr Dermatol. 2020 Nov;37(6):1202-1204. doi: 10.1111/pde.14318. Epub 2020 Aug 7.

DOI:10.1111/pde.14318
PMID:32767583
Abstract

We report a 2-year-old patient with Netherton syndrome presenting with generalized exfoliative erythroderma, ichthyosiform dermatitis, trichorrhexis invaginata, hypernatremic dehydration, failure to thrive, and recurrent respiratory infections. Molecular analysis of SPINK5 identified a novel mutation (c.1530CA). Our case report also verifies and supports the safety and efficacy of subcutaneous immunoglobulin substitution in chronic generalized skin disorders associated with primary immunodeficiencies such as Netherton syndrome.

摘要

我们报告了一名患有Netherton综合征的2岁患者,其表现为全身性剥脱性红皮病、鱼鳞病样皮炎、套叠性脆发症、高钠血症性脱水、生长发育迟缓以及反复呼吸道感染。对SPINK5的分子分析鉴定出一种新的突变(c.1530C>A)。我们的病例报告还验证并支持了皮下免疫球蛋白替代疗法在与原发性免疫缺陷相关的慢性全身性皮肤疾病(如Netherton综合征)中的安全性和有效性。

相似文献

1
A novel SPINK5 mutation and successful subcutaneous immunoglobulin replacement therapy in a child with Netherton syndrome.一名Netherton综合征患儿的新型SPINK5突变及皮下免疫球蛋白替代治疗成功案例
Pediatr Dermatol. 2020 Nov;37(6):1202-1204. doi: 10.1111/pde.14318. Epub 2020 Aug 7.
2
A Novel SPINK5 Gene Mutation Associated with Netherton Syndrome in an Omani Patient.一种与阿曼患者的 Netherton 综合征相关的新型 SPINK5 基因突变。
Sultan Qaboos Univ Med J. 2021 Nov;21(4):652-656. doi: 10.18295/squmj.4.2021.047. Epub 2021 Nov 25.
3
[Netherton syndrome].[ Netherton综合征 ]
Actas Dermosifiliogr. 2006 Jun;97(5):348-50. doi: 10.1016/s0001-7310(06)73417-2.
4
Netherton Syndrome in a Mother and Her Two Children. Netherton 综合征在一位母亲及其两个孩子中的表现。
S D Med. 2022 Dec;75(12):554-556.
5
A new SPINK5 mutation in a patient with Netherton syndrome: a case report.一名Netherton综合征患者的新型SPINK5突变:病例报告
Pediatr Dermatol. 2012 Jul-Aug;29(4):521-2. doi: 10.1111/j.1525-1470.2011.01525.x. Epub 2011 Jun 22.
6
Netherton syndrome: report of identical twins presenting with severe atopic dermatitis.Netherton综合征:一对患有严重特应性皮炎的同卵双胞胎的病例报告。
Eur J Pediatr. 2006 Sep;165(9):594-7. doi: 10.1007/s00431-006-0141-0. Epub 2006 May 3.
7
Two siblings affected by Netherton/Comèl syndrome. Diagnostic pathology and description of a new SPINK5 variant.两名受Netherton/Comèl综合征影响的兄弟姐妹。新的SPINK5变异体的诊断病理学及描述
Dermatol Online J. 2019 Jul 15;25(7):13030/qt0881q3sk.
8
Netherton syndrome: A neonatal case with respiratory insufficiency.Netherton综合征:一例伴有呼吸功能不全的新生儿病例。
Arch Argent Pediatr. 2018 Aug 1;116(4):e609-e611. doi: 10.5546/aap.2018.eng.e609.
9
Comèl-Netherton syndrome defined as primary immunodeficiency.科梅耳-内瑟顿综合征被定义为原发性免疫缺陷。
J Allergy Clin Immunol. 2009 Sep;124(3):536-43. doi: 10.1016/j.jaci.2009.06.009. Epub 2009 Aug 14.
10
Netherton syndrome caused by compound heterozygous mutation, c.80A>G mutation in SPINK5 and large-sized genomic deletion mutation, and successful treatment of intravenous immunoglobulin. Netherton 综合征由 SPINK5 中的复合杂合突变 c.80A>G 及大片段基因缺失突变引起,经静脉注射免疫球蛋白治疗后有效。
Mol Genet Genomic Med. 2021 Mar;9(3):e1600. doi: 10.1002/mgg3.1600. Epub 2021 Jan 16.

引用本文的文献

1
Hyper IgE Syndromes: Understanding, Management, and Future Perspectives: A Narrative Review.高免疫球蛋白E综合征:理解、管理与未来展望:一篇叙述性综述
Health Sci Rep. 2025 Mar 19;8(3):e70497. doi: 10.1002/hsr2.70497. eCollection 2025 Mar.
2
T2-driven manifestations of inborn errors of immunity.免疫缺陷病的T2驱动表现。
J Allergy Clin Immunol. 2024 Aug;154(2):245-254. doi: 10.1016/j.jaci.2024.05.007. Epub 2024 May 17.
3
Congenital Ichthyosis: A Practical Clinical Guide on Current Treatments and Future Perspectives.先天性鱼鳞病:当前治疗方法与未来展望实用临床指南
Clin Cosmet Investig Dermatol. 2023 Sep 11;16:2473-2479. doi: 10.2147/CCID.S388608. eCollection 2023.
4
Biological treatments for pediatric Netherton syndrome.儿童 Netherton 综合征的生物治疗。
Front Pediatr. 2022 Dec 23;10:1074243. doi: 10.3389/fped.2022.1074243. eCollection 2022.
5
A novel mutation in gene underlies a case of atypical Netherton syndrome.一个基因中的新型突变是一例非典型 Netherton 综合征的病因。
Front Genet. 2022 Sep 9;13:943264. doi: 10.3389/fgene.2022.943264. eCollection 2022.
6
Outcomes of Systemic Treatment in Children and Adults With Netherton Syndrome: A Systematic Review.先天性板层状鱼鳞病患儿和成人系统性治疗的结局:系统评价。
Front Immunol. 2022 Mar 30;13:864449. doi: 10.3389/fimmu.2022.864449. eCollection 2022.
7
A novel SPINK5 donor splice site variant in a child with Netherton syndrome.一个患有 Netherton 综合征的儿童中新型 SPINK5 供体位点剪接变异。
Mol Genet Genomic Med. 2021 Mar;9(3):e1611. doi: 10.1002/mgg3.1611. Epub 2021 Feb 3.
8
Netherton syndrome caused by compound heterozygous mutation, c.80A>G mutation in SPINK5 and large-sized genomic deletion mutation, and successful treatment of intravenous immunoglobulin. Netherton 综合征由 SPINK5 中的复合杂合突变 c.80A>G 及大片段基因缺失突变引起,经静脉注射免疫球蛋白治疗后有效。
Mol Genet Genomic Med. 2021 Mar;9(3):e1600. doi: 10.1002/mgg3.1600. Epub 2021 Jan 16.