{"doi":"10.1002/dvdy.194","title":"Shootin‐1 is required for nervous system development in zebrafish","abstract":"<jats:title>Abstract</jats:title><jats:sec><jats:title>Background</jats:title><jats:p>Semaphorin6A (Sema6A) and its PlexinA2 (PlxnA2) receptor canonically function as repulsive axon guidance cues. To understand downstream signaling mechanisms, we performed a microarray screen and identified the “clutch molecule” <jats:italic>shootin‐1</jats:italic> (<jats:italic>shtn‐1)</jats:italic> as a transcriptionally repressed target. Shtn‐1 is a key proponent of cell migration and neuronal polarization and must be regulated during nervous system development. The mechanisms of Shtn‐1 regulation and the phenotypic consequences of loss of repression are poorly understood.</jats:p></jats:sec><jats:sec><jats:title>Results</jats:title><jats:p>We demonstrate <jats:italic>shtn‐1</jats:italic> overexpression results in impaired migration of the optic vesicles, lack of retinal pigmented epithelium, and pathfinding errors of retinotectal projections. We also observed patterning defects in the peripheral nervous system. Importantly, these phenotypes were rescued by overexpressing PlxnA2.</jats:p></jats:sec><jats:sec><jats:title>Conclusions</jats:title><jats:p>We demonstrate a functional role for repression of <jats:italic>shtn‐1</jats:italic> by PlxnA2 in development of the eyes and peripheral nervous system in zebrafish. These results demonstrate that careful regulation of <jats:italic>shtn‐1</jats:italic> is critical for development of the nervous system.</jats:p></jats:sec>","journal":"Developmental Dynamics","year":2020,"id":658005,"datarank":0.33738732449774245,"base_score":1.6094379124341003,"endowment":1.6094379124341003,"self_citation_contribution":0.24141568686511508,"citation_network_contribution":0.09597163763262734,"self_endowment_contribution":0.24141568686511508,"citer_contribution":0.09597163763262734,"corpus_percentile":null,"corpus_rank":null,"citation_count":4,"citer_count":4,"citers_with_citation_signal":4,"citers_with_endowment":4,"datacite_reuse_total":0,"is_dataset":false,"is_dataset_confidence":null,"is_data_producer":false,"deposit_databanks":null,"is_oa":false,"file_count":0,"downloads":0,"has_version_chain":false,"published_date":null,"fair_score":null,"fair_percentile":null,"algorithm_id":"datarank_citation_only_1hop_v6","ranking_scope":"data_only","authors":[{"id":789153,"name":"Helaina R. Stergas","orcid":null,"position":1,"is_corresponding":false},{"id":1717677,"name":"Simon O. Bupp‐Chickering","orcid":null,"position":2,"is_corresponding":false},{"id":369667,"name":"Alicia M. Ebert","orcid":"0000-0002-1112-2009","position":3,"is_corresponding":false},{"id":1359119,"name":"Sarah E. Emerson","orcid":"0000-0001-9587-3784","position":0,"is_corresponding":false}],"reference_count":0,"raw_metadata":{"has_enrichment":true,"resolved":true,"title":"Shootin‐1 is required for nervous system development in zebrafish","abstract":"<jats:title>Abstract</jats:title><jats:sec><jats:title>Background</jats:title><jats:p>Semaphorin6A (Sema6A) and its PlexinA2 (PlxnA2) receptor canonically function as repulsive axon guidance cues. To understand downstream signaling mechanisms, we performed a microarray screen and identified the “clutch molecule” <jats:italic>shootin‐1</jats:italic> (<jats:italic>shtn‐1)</jats:italic> as a transcriptionally repressed target. Shtn‐1 is a key proponent of cell migration and neuronal polarization and must be regulated during nervous system development. The mechanisms of Shtn‐1 regulation and the phenotypic consequences of loss of repression are poorly understood.</jats:p></jats:sec><jats:sec><jats:title>Results</jats:title><jats:p>We demonstrate <jats:italic>shtn‐1</jats:italic> overexpression results in impaired migration of the optic vesicles, lack of retinal pigmented epithelium, and pathfinding errors of retinotectal projections. We also observed patterning defects in the peripheral nervous system. Importantly, these phenotypes were rescued by overexpressing PlxnA2.</jats:p></jats:sec><jats:sec><jats:title>Conclusions</jats:title><jats:p>We demonstrate a functional role for repression of <jats:italic>shtn‐1</jats:italic> by PlxnA2 in development of the eyes and peripheral nervous system in zebrafish. These results demonstrate that careful regulation of <jats:italic>shtn‐1</jats:italic> is critical for development of the nervous system.</jats:p></jats:sec>","is_dataset_classified":null,"base_score":1.6094379124341003,"endowment":1.6094379124341003,"datacite_reuse_total":0,"file_count":0,"downloads":0,"views":0,"has_version_chain":false,"is_dataset":false,"is_oa":false,"pmid":"32406957","pmcid":null,"openalex_id":"https://openalex.org/W3024526760","authors":[],"funders":[{"funder_name":"National Science Foundation","grant_id":"IOS1456846","title":null}],"total_grants":1,"fwci":0.0,"citation_percentile":0.04402916,"influential_citations":0,"citation_trend":[{"year":2024,"count":4}],"oa_status":"closed","license":"http://onlinelibrary.wiley.com/termsAndConditions#am","oa_locations":[{"url":"https://api.wiley.com/onlinelibrary/tdm/v1/articles/10.1002%2Fdvdy.194","host_type":"publisher"},{"url":"https://onlinelibrary.wiley.com/doi/pdf/10.1002/dvdy.194","host_type":"publisher"},{"url":"https://onlinelibrary.wiley.com/doi/full-xml/10.1002/dvdy.194","host_type":"publisher"},{"url":"https://anatomypubs.onlinelibrary.wiley.com/doi/am-pdf/10.1002/dvdy.194","host_type":"publisher"},{"url":"https://anatomypubs.onlinelibrary.wiley.com/doi/pdf/10.1002/dvdy.194","host_type":"publisher"},{"url":"https://doi.org/10.1002/dvdy.194","host_type":"journal"},{"url":"https://pubmed.ncbi.nlm.nih.gov/32406957","host_type":"repository"}],"fields_of_study":["Axon Guidance and Neuronal Signaling","Developmental Biology and Gene Regulation","Hippo pathway signaling and YAP/TAZ","Animals","Axons","Body Patterning","Cell Movement","Cytoskeletal Proteins","Gene Expression Regulation, Developmental","Humans","Motor Neurons","Nerve Tissue Proteins","Nervous System","Neurons","Peripheral Nervous System","Phenotype","Receptors, Cell Surface","Retinal Pigment Epithelium","Semaphorins","Zebrafish","Zebrafish Proteins"],"mesh_terms":["Animals","Axons","Cell Movement","Cytoskeletal Proteins","Humans","Motor Neurons","Nerve Tissue Proteins","Nervous System","Neurons","Phenotype","Receptors, Cell Surface","Zebrafish","Peripheral Nervous System","Gene Expression Regulation, Developmental","Body Patterning","Zebrafish Proteins","Semaphorins","Retinal Pigment Epithelium"],"keywords":["Biology","Zebrafish","Nervous system","Computational biology","Cell biology","Neuroscience","Evolutionary biology","Genetics","Gene","Retina","Cytoskeleton","Migration","Microarray","Plexina2","Semaphorin6a"],"sdg_mappings":[],"linked_datasets":[],"clinical_trials":[],"software_tools":[],"database_accessions":[],"source":"live","citation_network_status":"fetched"},"created_at":"2026-08-12T03:00:04.802229Z","pmid":null,"pmcid":null,"fwci":null,"citation_percentile":null,"influential_citations":0,"oa_status":null,"license":null,"views":0,"total_file_size_bytes":0,"version_count":0,"fair_f":null,"fair_a":null,"fair_i":null,"fair_r":null,"fair_zscore":null,"fair_rationale":null,"fair_model":null,"fair_agent_version":null,"fair_fulltext_source":null,"fair_has_llm":null,"fair_computed_at":null,"clinical_trials":[],"software_tools":[],"db_accessions":[],"linked_datasets":[],"topics":[]}