{"title":"Roles of the HUWE1 ubiquitin ligase in nervous system development, function and disease.","authors":"Andrew C Giles, Brock Grill","doi":"10.1186/s13064-020-00143-9","DOIUrl":null,"url":null,"abstract":"<p><p>Huwe1 is a highly conserved member of the HECT E3 ubiquitin ligase family. Here, we explore the growing importance of Huwe1 in nervous system development, function and disease. We discuss extensive progress made in deciphering how Huwe1 regulates neural progenitor proliferation and differentiation, cell migration, and axon development. We highlight recent evidence indicating that Huwe1 regulates inhibitory neurotransmission. In covering these topics, we focus on findings made using both vertebrate and invertebrate in vivo model systems. Finally, we discuss extensive human genetic studies that strongly implicate HUWE1 in intellectual disability, and heighten the importance of continuing to unravel how Huwe1 affects the nervous system.</p>","PeriodicalId":49764,"journal":{"name":"Neural Development","volume":" ","pages":"6"},"PeriodicalIF":4.0000,"publicationDate":"2020-04-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.1186/s13064-020-00143-9","citationCount":"24","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Neural Development","FirstCategoryId":"99","ListUrlMain":"https://doi.org/10.1186/s13064-020-00143-9","RegionNum":3,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"DEVELOPMENTAL BIOLOGY","Score":null,"Total":0}
引用次数: 24
Abstract
Huwe1 is a highly conserved member of the HECT E3 ubiquitin ligase family. Here, we explore the growing importance of Huwe1 in nervous system development, function and disease. We discuss extensive progress made in deciphering how Huwe1 regulates neural progenitor proliferation and differentiation, cell migration, and axon development. We highlight recent evidence indicating that Huwe1 regulates inhibitory neurotransmission. In covering these topics, we focus on findings made using both vertebrate and invertebrate in vivo model systems. Finally, we discuss extensive human genetic studies that strongly implicate HUWE1 in intellectual disability, and heighten the importance of continuing to unravel how Huwe1 affects the nervous system.
期刊介绍：
Neural Development is a peer-reviewed open access, online journal, which features studies that use molecular, cellular, physiological or behavioral methods to provide novel insights into the mechanisms that underlie the formation of the nervous system.
Neural Development aims to discover how the nervous system arises and acquires the abilities to sense the world and control adaptive motor output. The field includes analysis of how progenitor cells form a nervous system during embryogenesis, and how the initially formed neural circuits are shaped by experience during early postnatal life. Some studies use well-established, genetically accessible model systems, but valuable insights are also obtained from less traditional models that provide behavioral or evolutionary insights.
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