{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,27]],"date-time":"2026-02-27T05:01:48Z","timestamp":1772168508457,"version":"3.50.1"},"reference-count":46,"publisher":"The Company of Biologists","license":[{"start":{"date-parts":[[2020,1,1]],"date-time":"2020-01-01T00:00:00Z","timestamp":1577836800000},"content-version":"am","delay-in-days":0,"URL":"http:\/\/www.biologists.com\/user-licence-1-1\/"}],"funder":[{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"crossref","award":["GNT1105374"],"award-info":[{"award-number":["GNT1105374"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100012789","name":"Veski","doi-asserted-by":"crossref","award":["VIF23"],"award-info":[{"award-number":["VIF23"]}],"id":[{"id":"10.13039\/100012789","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:p>Brain development requires precise regulation of axon outgrowth, guidance and termination by multiple signaling and adhesion molecules. How the expression of these neurodevelopmental regulators is transcriptionally controlled is poorly understood. The Caenorhabditis elegans SMD motor neurons terminate axon outgrowth upon sexual maturity and partially retract their axons during early adulthood. Here we show that C-Terminal Binding Protein-1 (CTBP-1), a transcriptional corepressor, is required for correct SMD axonal development. Loss of CTBP-1 causes multiple defects in SMD axon development: premature outgrowth, defective guidance, delayed termination and absence of retraction. CTBP-1 controls SMD axon guidance by repressing the expression of SAX-7 - a L1 cell adhesion molecule (L1CAM). CTBP-1-regulated repression is crucial as deregulated SAX-7\/L1CAM causes severely aberrant SMD axons. We found that axonal defects caused by deregulated SAX-7\/L1CAM are dependent on a distinct L1CAM, called LAD-2, which itself plays a parallel role in SMD axon guidance. Our results reveal that harmonization of L1CAM expression controls the development and maturation of a single neuron.<\/jats:p>","DOI":"10.1242\/dev.193805","type":"journal-article","created":{"date-parts":[[2020,9,29]],"date-time":"2020-09-29T14:03:02Z","timestamp":1601388182000},"source":"Crossref","is-referenced-by-count":5,"title":["Harmonization of L1CAM expression facilitates axon outgrowth and guidance of a motor neuron"],"prefix":"10.1242","author":[{"given":"Tessa","family":"Sherry","sequence":"first","affiliation":[{"name":"Development and Stem Cells Program, Monash \u00adBiomedicine Discovery Institute and Department of Anatomy and Developmental Biology, Monash University, Melbourne, Victoria 3800, Australia"},{"name":"School of Life and Environmental Sciences, The University of Sydney, Sydney, NSW 2006, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ava","family":"Handley","sequence":"additional","affiliation":[{"name":"Development and Stem Cells Program, Monash \u00adBiomedicine Discovery Institute and Department of Anatomy and Developmental Biology, Monash University, Melbourne, Victoria 3800, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2239-9249","authenticated-orcid":false,"given":"Hannah R.","family":"Nicholas","sequence":"additional","affiliation":[{"name":"School of Life and Environmental Sciences, The University of Sydney, Sydney, NSW 2006, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5515-3608","authenticated-orcid":false,"given":"Roger","family":"Pocock","sequence":"additional","affiliation":[{"name":"Development and Stem Cells Program, Monash \u00adBiomedicine Discovery Institute and Department of Anatomy and Developmental Biology, Monash University, Melbourne, Victoria 3800, Australia"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2020,1,1]]},"reference":[{"key":"2021042700402612800_DEV193805C1","doi-asserted-by":"publisher","first-page":"92","DOI":"10.1016\/j.stemcr.2017.04.032","article-title":"THAP1: role in mouse embryonic stem cell survival and differentiation","volume":"9","author":"Aguilo","year":"2017","journal-title":"Stem Cell Rep."},{"key":"2021042700402612800_DEV193805C2","doi-asserted-by":"publisher","first-page":"686","DOI":"10.1126\/science.1066642","article-title":"Immunoglobulin-domain proteins required for maintenance of ventral nerve cord organization","volume":"295","author":"Aurelio","year":"2002","journal-title":"Science"},{"key":"2021042700402612800_DEV193805C3","doi-asserted-by":"publisher","first-page":"285","DOI":"10.1016\/S0092-8674(03)00267-8","article-title":"Stereotyped pruning of long hippocampal axon branches triggered by retraction inducers of the semaphorin family","volume":"113","author":"Bagri","year":"2003","journal-title":"Cell"},{"key":"2021042700402612800_DEV193805C4","doi-asserted-by":"publisher","first-page":"173","DOI":"10.1007\/s10048-016-0482-4","article-title":"A recurrent de novo CTBP1 mutation is associated with developmental delay, hypotonia, ataxia, and tooth enamel defects","volume":"17","author":"Beck","year":"2016","journal-title":"Neurogenetics"},{"key":"2021042700402612800_DEV193805C5","doi-asserted-by":"publisher","first-page":"129","DOI":"10.1007\/s10048-019-00578-1","article-title":"A pathogenic CtBP1 missense mutation causes altered cofactor binding and transcriptional activity","volume":"20","author":"Beck","year":"2019","journal-title":"Neurogenetics"},{"key":"2021042700402612800_DEV193805C6","doi-asserted-by":"publisher","first-page":"e1002819","DOI":"10.1371\/journal.pgen.1002819","article-title":"The secreted immunoglobulin domain proteins ZIG-5 and ZIG-8 cooperate with L1CAM\/SAX-7 to maintain nervous system integrity","volume":"8","author":"B\u00e9nard","year":"2012","journal-title":"PLoS Genet."},{"key":"2021042700402612800_DEV193805C7","doi-asserted-by":"publisher","first-page":"87","DOI":"10.1016\/S0959-4388(98)80012-3","article-title":"Neural cell recognition molecule L1: from cell biology to human hereditary brain malformations","volume":"8","author":"Br\u00fcmmendorf","year":"1998","journal-title":"Curr. 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