{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T16:27:47Z","timestamp":1787070467392,"version":"build-2736575974"},"reference-count":66,"publisher":"Oxford University Press (OUP)","issue":"17","license":[{"start":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T00:00:00Z","timestamp":1787011200000},"content-version":"vor","delay-in-days":229,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"name":"Baylor College of Medicine Seed Funding"},{"name":"Clifford Elder White Graham Endowed Research Fund"},{"DOI":"10.13039\/100002243","name":"National Ataxia Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002243","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,8,10]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Spinocerebellar ataxia type 1 is a progressive neurodegenerative disorder caused by polyglutamine expansion in ATXN1, yet the normal physiological roles of ATXN1 and its paralog ATXN1L remain incompletely understood. To define these roles, we generated the first zebrafish knockouts (KOs) of the three ataxin-1 family genes, atxn1a, atxn1b, and atxn1l, using CRISPR\/Cas9 and performed phenotypic and transcriptomic analyses. All KOs exhibited reduced early survival and mild larval growth deficits. Behavioral assays revealed both shared and paralog-specific effects: atxn1a mutants displayed a reproducible light-dependent locomotor deficit, whereas atxn1b and atxn1l mutants showed generalized hypoactivity. Adult behavioral assessment revealed a gradient of phenotypic severity, with atxn1a KOs displaying the earliest and most pronounced alterations in vertical tank exploration and the greatest impairment in swim-tunnel performance. RNA-seq at 5\u00a0days post-fertilization identified extensive transcriptional alterations, including both shared and gene-specific differentially expressed genes associated with neural differentiation, microglial\/immune cell migration, and immune signaling. Co-expression network analysis further identified distinct KO-associated gene modules, including a phototransduction-enriched module strongly correlated with atxn1a KO status, providing a potential mechanistic link to its light-dependent locomotor phenotype. Together, these findings define conserved and specialized roles of ATXN1-family genes in locomotor behavior, neurodevelopment, retinal function, and immune-related processes.<\/jats:p>","DOI":"10.1093\/hmg\/ddag078","type":"journal-article","created":{"date-parts":[[2026,8,4]],"date-time":"2026-08-04T11:44:58Z","timestamp":1785843898000},"source":"Crossref","is-referenced-by-count":0,"title":["Zebrafish knockout models of\n                    <i>atxn1a, atxn1b<\/i>\n                    , and\n                    <i>atxn1l<\/i>\n                    reveal distinct and shared phenotypic and transcriptomic alterations"],"prefix":"10.1093","volume":"35","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5795-6674","authenticated-orcid":false,"given":"Anwarul","family":"Karim","sequence":"first","affiliation":[{"name":"Department of Neurology, Baylor College of Medicine , Houston, TX 77030 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pramuk","family":"Keerthisinghe","sequence":"additional","affiliation":[{"name":"Department of Neurology, Baylor College of Medicine , Houston, TX 77030 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sreeja","family":"Sarasamma","sequence":"additional","affiliation":[{"name":"Department of Neurology, Baylor College of Medicine , Houston, TX 77030 ,","place":["United States"]},{"name":"Department of Fisheries and Wildlife, Michigan State University , East Lansing, MI 48824 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nicholas A","family":"Ciaburri","sequence":"additional","affiliation":[{"name":"Department of Neurology, Baylor College of Medicine , Houston, TX 77030 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mabel Guerra","family":"Giraldez","sequence":"additional","affiliation":[{"name":"Department of Neurology, Baylor College of Medicine , Houston, TX 77030 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kylan","family":"Naidoo","sequence":"additional","affiliation":[{"name":"Department of Neurology, Baylor College of Medicine , Houston, TX 77030 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James P","family":"Orengo","sequence":"additional","affiliation":[{"name":"Department of Neurology, Baylor College of Medicine , Houston, TX 77030 ,","place":["United States"]},{"name":"Department of Neuroscience, Baylor College of Medicine , Houston, TX 77030 ,","place":["United States"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2026,8,18]]},"reference":[{"key":"2026081811260448100_ref1","doi-asserted-by":"publisher","first-page":"29","DOI":"10.1016\/1043-4682(95)90012-8","article-title":"Spinocerebellar ataxia type 1","volume":"6","author":"Zoghbi","year":"1995","journal-title":"Semin Cell Biol"},{"key":"2026081811260448100_ref2","doi-asserted-by":"publisher","first-page":"4015","DOI":"10.1007\/s00018-020-03520-z","article-title":"Pathogenic mechanisms underlying spinocerebellar ataxia type 1","volume":"77","author":"Tejwani","year":"2020","journal-title":"Cell Mol Life Sci"},{"key":"2026081811260448100_ref3","doi-asserted-by":"publisher","first-page":"404","DOI":"10.1007\/s00018-022-04419-7","article-title":"The extra-cerebellar effects of spinocerebellar ataxia type 1 (SCA1): looking beyond the cerebellum","volume":"79","author":"Olmos","year":"2022","journal-title":"Cell Mol Life Sci"},{"key":"2026081811260448100_ref4","doi-asserted-by":"publisher","first-page":"1335","DOI":"10.1016\/j.cell.2006.11.038","article-title":"ATAXIN-1 interacts with the repressor Capicua in its native complex to cause SCA1 neuropathology","volume":"127","author":"Lam","year":"2006","journal-title":"Cell"},{"key":"2026081811260448100_ref5","doi-asserted-by":"publisher","first-page":"333","DOI":"10.1016\/j.omtm.2022.04.004","article-title":"Combined overexpression of ATXN1L and mutant ATXN1 knockdown by AAV rescue motor phenotypes and gene signatures in SCA1 mice","volume":"25","author":"Carrell","year":"2022","journal-title":"Mol Ther - Methods Clin Dev"},{"key":"2026081811260448100_ref6","doi-asserted-by":"publisher","first-page":"e1001021","DOI":"10.1371\/journal.pgen.1001021","article-title":"Partial loss of Ataxin-1 function contributes to transcriptional dysregulation in spinocerebellar ataxia type 1 pathogenesis","volume":"6","author":"Crespo-Barreto","year":"2010","journal-title":"PLoS Genet"},{"key":"2026081811260448100_ref7","doi-asserted-by":"publisher","first-page":"3339","DOI":"10.1038\/sj.emboj.7600785","article-title":"Boat, an AXH domain protein, suppresses the cytotoxicity of mutant ataxin-1","volume":"24","author":"Mizutani","year":"2005","journal-title":"EMBO J"},{"key":"2026081811260448100_ref8","doi-asserted-by":"publisher","first-page":"1159","DOI":"10.1016\/j.cell.2019.07.043","article-title":"Loss of Ataxin-1 potentiates Alzheimer\u2019s pathogenesis by elevating cerebral BACE1 transcription","volume":"178","author":"Suh","year":"2019","journal-title":"Cell"},{"key":"2026081811260448100_ref9","doi-asserted-by":"publisher","first-page":"eaav7188","DOI":"10.1126\/science.aav7188","article-title":"Multiple sclerosis genomic map implicates peripheral immune cells and microglia in susceptibility","volume":"365","author":"International Multiple Sclerosis Genetics Consortium","year":"2019","journal-title":"Science"},{"key":"2026081811260448100_ref10","doi-asserted-by":"publisher","first-page":"23742","DOI":"10.1073\/pnas.2003798117","article-title":"Ataxin-1 regulates B cell function and the severity of autoimmune experimental encephalomyelitis","volume":"117","author":"Didonna","year":"2020","journal-title":"Proc Natl Acad Sci"},{"key":"2026081811260448100_ref11","doi-asserted-by":"publisher","first-page":"127","DOI":"10.1186\/s12974-025-03450-2","article-title":"An expanded polyglutamine in ATAXIN1 results in a loss-of-function that exacerbates severity of multiple sclerosis in an EAE mouse model","volume":"22","author":"Talukdar","year":"2025","journal-title":"J Neuroinflammation"},{"key":"2026081811260448100_ref12","doi-asserted-by":"publisher","first-page":"2122","DOI":"10.1093\/hmg\/ddm162","article-title":"Down-regulation of the dopamine receptor D2 in mice lacking ataxin 1","volume":"16","author":"Goold","year":"2007","journal-title":"Hum Mol Genet"},{"key":"2026081811260448100_ref13","doi-asserted-by":"publisher","first-page":"5508","DOI":"10.1523\/JNEUROSCI.18-14-05508.1998","article-title":"Mice lacking Ataxin-1 display learning deficits and decreased hippocampal paired-pulse facilitation","volume":"18","author":"Matilla","year":"1998","journal-title":"J Neurosci"},{"key":"2026081811260448100_ref14","doi-asserted-by":"publisher","first-page":"4021","DOI":"10.1093\/hmg\/ddw242","article-title":"Ataxin-1 regulates the cerebellar bioenergetics proteome through the GSK3\u03b2-mTOR pathway which is altered in spinocerebellar ataxia type 1 (SCA1)","volume":"25","author":"S\u00e1nchez","year":"2016","journal-title":"Hum Mol Genet"},{"key":"2026081811260448100_ref15","doi-asserted-by":"publisher","first-page":"746","DOI":"10.1016\/j.devcel.2011.08.017","article-title":"ATXN1 protein family and CIC regulate extracellular matrix remodeling and lung alveolarization","volume":"21","author":"Lee","year":"2011","journal-title":"Dev Cell"},{"key":"2026081811260448100_ref16","doi-asserted-by":"publisher","first-page":"788","DOI":"10.3390\/biom13050788","article-title":"Therapeutic strategies for spinocerebellar ataxia type 1","volume":"13","author":"Kerkhof","year":"2023","journal-title":"Biomolecules"},{"key":"2026081811260448100_ref17","doi-asserted-by":"publisher","first-page":"940484","DOI":"10.3389\/fnmol.2022.940484","article-title":"Zebrafish as a model organism for neurodegenerative disease","volume":"15","author":"Chia","year":"2022","journal-title":"Front Mol Neurosci"},{"key":"2026081811260448100_ref18","doi-asserted-by":"publisher","first-page":"45","DOI":"10.1038\/s41420-018-0109-7","article-title":"Zebrafish: an emerging real-time model system to study Alzheimer\u2019s disease and neurospecific drug discovery","volume":"4","author":"Saleem","year":"2018","journal-title":"Cell Death Discov"},{"key":"2026081811260448100_ref19","doi-asserted-by":"publisher","first-page":"1322","DOI":"10.3390\/biology12101322","article-title":"Zebrafish models of rare neurological diseases like spinocerebellar ataxias (SCAs): advantages and limitations","volume":"12","author":"Sarasamma","year":"2023","journal-title":"Biology"},{"key":"2026081811260448100_ref20","doi-asserted-by":"publisher","first-page":"W216","DOI":"10.1093\/nar\/gkac194","article-title":"DAVID: a web server for functional enrichment analysis and functional annotation of gene lists (2021 update)","volume":"50","author":"Sherman","year":"2022","journal-title":"Nucleic Acids Res"},{"key":"2026081811260448100_ref21","doi-asserted-by":"publisher","first-page":"e70028","DOI":"10.1002\/mef2.70028","article-title":"Zebrafish as a versatile model organism: from tanks to treatment","volume":"4","author":"Srivastava","year":"2025","journal-title":"MedComm \u2013 Future Med"},{"key":"2026081811260448100_ref22","doi-asserted-by":"publisher","first-page":"313","DOI":"10.1080\/01677060802399976","article-title":"Characterization of the zebrafish atxn1\/axh gene family","volume":"23","author":"Carlson","year":"2009","journal-title":"J Neurogenet"},{"key":"2026081811260448100_ref23","doi-asserted-by":"publisher","first-page":"11348","DOI":"10.3390\/ijms222111348","article-title":"Structural analysis and spatiotemporal expression of Atxn1 genes in zebrafish embryos and larvae","volume":"22","author":"Vauti","year":"2021","journal-title":"Int J Mol Sci"},{"key":"2026081811260448100_ref24","doi-asserted-by":"publisher","first-page":"7351","DOI":"10.3390\/ijms22147351","article-title":"Genetic modeling of the neurodegenerative disease spinocerebellar ataxia type 1 in zebrafish","volume":"22","author":"Elsaey","year":"2021","journal-title":"Int J Mol Sci"},{"key":"2026081811260448100_ref25","doi-asserted-by":"publisher","first-page":"e0266491","DOI":"10.1371\/journal.pone.0266491","article-title":"Characterization of locomotor phenotypes in zebrafish larvae requires testing under both light and dark conditions","volume":"17","author":"Tuz-Sasik","year":"2022","journal-title":"PLoS One"},{"key":"2026081811260448100_ref26","doi-asserted-by":"publisher","first-page":"104374","DOI":"10.1016\/j.xpro.2026.104374","article-title":"Protocol for quantitative analysis of adult zebrafish swimming behavior using DeepLabCut","volume":"7","author":"Keerthisinghe","year":"2026","journal-title":"STAR Protoc"},{"key":"2026081811260448100_ref27","doi-asserted-by":"publisher","first-page":"1281","DOI":"10.1038\/s41593-018-0209-y","article-title":"DeepLabCut: markerless pose estimation of user-defined body parts with deep learning","volume":"21","author":"Mathis","year":"2018","journal-title":"Nat Neurosci"},{"key":"2026081811260448100_ref28","doi-asserted-by":"publisher","first-page":"W83","DOI":"10.1093\/nar\/gkae410","article-title":"The galaxy platform for accessible, reproducible, and collaborative data analyses: 2024 update","volume":"52","author":"Community","year":"2024","journal-title":"Nucleic Acids Res"},{"key":"2026081811260448100_ref29","doi-asserted-by":"publisher","first-page":"44","DOI":"10.1038\/nprot.2008.211","article-title":"Systematic and integrative analysis of large gene lists using DAVID bioinformatics resources","volume":"4","author":"Huang","year":"2009","journal-title":"Nat Protoc"},{"key":"2026081811260448100_ref30","first-page":"1","article-title":"Emerging roles of 5-lipoxygenase phosphorylation in inflammation and cell death","volume":"2019","author":"Sun","year":"2019","journal-title":"Oxidative Med Cell Longev"},{"key":"2026081811260448100_ref31","doi-asserted-by":"publisher","first-page":"3028","DOI":"10.1016\/j.devcel.2023.11.001","article-title":"Single-cell analysis of shared signatures and transcriptional diversity during zebrafish development","volume":"58","author":"Sur","year":"2023","journal-title":"Dev Cell"},{"key":"2026081811260448100_ref32","doi-asserted-by":"publisher","first-page":"107570","DOI":"10.1016\/j.jbc.2024.107570","article-title":"Her4.3+ radial glial cells maintain the brain vascular network through activation of Wnt signaling","volume":"300","author":"Wang","year":"2024","journal-title":"J Biol Chem"},{"key":"2026081811260448100_ref33","doi-asserted-by":"publisher","first-page":"3156","DOI":"10.1242\/dev.147256","article-title":"Hes5 regulates the transition timing of neurogenesis and gliogenesis in mammalian neocortical development","volume":"144","author":"Bansod","year":"2017","journal-title":"Development"},{"key":"2026081811260448100_ref34","doi-asserted-by":"publisher","DOI":"10.1093\/bioadv\/vbae065","article-title":"danRerLib: a python package for zebrafish transcriptomics","volume":"4","author":"Schwartz","year":"2024","journal-title":"Bioinforma Adv"},{"key":"2026081811260448100_ref35","doi-asserted-by":"publisher","first-page":"366","DOI":"10.1038\/s41420-022-01155-6","article-title":"dock8 deficiency attenuates microglia colonization in early zebrafish larvae","volume":"8","author":"Wu","year":"2022","journal-title":"Cell Death Discov"},{"key":"2026081811260448100_ref36","doi-asserted-by":"publisher","first-page":"1812","DOI":"10.1002\/cne.25542","article-title":"Insulin signaling promotes neurogenesis in the brain of adult zebrafish","volume":"531","author":"Gence","year":"2023","journal-title":"J Comp Neurol"},{"key":"2026081811260448100_ref37","doi-asserted-by":"publisher","first-page":"7084","DOI":"10.1523\/JNEUROSCI.23-18-07084.2003","article-title":"Insulin receptor Substrate-2 deficiency impairs brain growth and promotes tau phosphorylation","volume":"23","author":"Schubert","year":"2003","journal-title":"J Neurosci"},{"key":"2026081811260448100_ref38","doi-asserted-by":"publisher","first-page":"110","DOI":"10.1016\/j.scr.2018.10.005","article-title":"SOX4 inhibits oligodendrocyte differentiation of embryonic neural stem cells in vitro by inducing Hes5 expression","volume":"33","author":"Braccioli","year":"2018","journal-title":"Stem Cell Res"},{"key":"2026081811260448100_ref39","doi-asserted-by":"publisher","first-page":"5495","DOI":"10.1523\/JNEUROSCI.1384-07.2007","article-title":"Prolonged glial expression of Sox4 in the CNS leads to architectural cerebellar defects and ataxia","volume":"27","author":"Hoser","year":"2007","journal-title":"J Neurosci"},{"key":"2026081811260448100_ref40","doi-asserted-by":"publisher","first-page":"103975","DOI":"10.1016\/j.compbiomed.2020.103975","article-title":"Advantages of CEMiTool for gene co-expression analysis of RNA-seq data","volume":"125","author":"Cheng","year":"2020","journal-title":"Comput Biol Med"},{"key":"2026081811260448100_ref41","doi-asserted-by":"publisher","first-page":"56","DOI":"10.1186\/s12859-018-2053-1","article-title":"CEMiTool: a Bioconductor package for performing comprehensive modular co-expression analyses","volume":"19","author":"Russo","year":"2018","journal-title":"BMC Bioinformatics"},{"key":"2026081811260448100_ref42","doi-asserted-by":"publisher","first-page":"D638","DOI":"10.1093\/nar\/gkac1000","article-title":"The STRING database in 2023: protein\u2013protein association networks and functional enrichment analyses for any sequenced genome of interest","volume":"51","author":"Szklarczyk","year":"2023","journal-title":"Nucleic Acids Res"},{"key":"2026081811260448100_ref43","doi-asserted-by":"publisher","first-page":"1377","DOI":"10.1007\/s00424-021-02562-x","article-title":"Photoreceptor phosphodiesterase (PDE6): activation and inactivation mechanisms during visual transduction in rods and cones","volume":"473","author":"Cote","year":"2021","journal-title":"Pfl\u00fcg Arch - Eur J Physiol"},{"key":"2026081811260448100_ref44","doi-asserted-by":"publisher","first-page":"39828","DOI":"10.1074\/jbc.M110.170068","article-title":"Rod phosphodiesterase-6 PDE6A and PDE6B subunits are enzymatically equivalent","volume":"285","author":"Muradov","year":"2010","journal-title":"J Biol Chem"},{"key":"2026081811260448100_ref45","doi-asserted-by":"publisher","first-page":"221","DOI":"10.1038\/ng0793-221","article-title":"Expansion of an unstable trinucleotide CAG repeat in spinocerebellar ataxia type 1","volume":"4","author":"Orr","year":"1993","journal-title":"Nat Genet"},{"key":"2026081811260448100_ref46","doi-asserted-by":"publisher","first-page":"fcaa064","DOI":"10.1093\/braincomms\/fcaa064","article-title":"ATXN1 repeat expansions confer risk for amyotrophic lateral sclerosis and contribute to TDP-43 mislocalization","volume":"2","author":"Tazelaar","year":"2020","journal-title":"Brain Commun"},{"key":"2026081811260448100_ref47","doi-asserted-by":"publisher","DOI":"10.1111\/febs.70328","article-title":"Conditional deletion of the multiple sclerosis susceptibility gene ATXN1 identifies cell-autonomous effects in the B-cell compartment","volume":"293","author":"Carver","year":"2025","journal-title":"FEBS J"},{"key":"2026081811260448100_ref48","doi-asserted-by":"publisher","first-page":"358","DOI":"10.1111\/imcb.12622","article-title":"Ataxin-1 controls the expression of specific noncoding RNAs in B cells upon autoimmune demyelination","volume":"101","author":"Ma","year":"2023","journal-title":"Immunol Cell Biol"},{"key":"2026081811260448100_ref49","doi-asserted-by":"publisher","first-page":"999","DOI":"10.1007\/s13311-019-00763-y","article-title":"Moving towards therapy in SCA1: insights from molecular mechanisms, identification of novel targets, and planning for human trials","volume":"16","author":"Srinivasan","year":"2019","journal-title":"Neurotherapeutics"},{"key":"2026081811260448100_ref50","doi-asserted-by":"publisher","first-page":"bio059229","DOI":"10.1242\/bio.059229","article-title":"Zebrafish embryos hatch early in response to chemical and mechanical indicators of predation risk, resulting in underdeveloped swimming ability of hatchling larvae","volume":"11","author":"Wisenden","year":"2022","journal-title":"Biol Open"},{"key":"2026081811260448100_ref51","doi-asserted-by":"publisher","first-page":"88","DOI":"10.1080\/01677063.2017.1358268","article-title":"Neurotoxicity of cGMP in the vertebrate retina: from the initial research on rd mutant mice to zebrafish genetic approaches","volume":"31","author":"Iribarne","year":"2017","journal-title":"J Neurogenet"},{"key":"2026081811260448100_ref52","doi-asserted-by":"publisher","first-page":"105230","DOI":"10.1016\/j.beproc.2025.105230","article-title":"Understanding (and appreciating) behavioral complexity of zebrafish novel tank assays","volume":"230","author":"Golushko","year":"2025","journal-title":"Behav Process"},{"key":"2026081811260448100_ref53","doi-asserted-by":"publisher","first-page":"161","DOI":"10.1016\/j.it.2015.01.003","article-title":"Nitric oxide synthase in innate and adaptive immunity: an update","volume":"36","author":"Bogdan","year":"2015","journal-title":"Trends Immunol"},{"key":"2026081811260448100_ref54","doi-asserted-by":"publisher","first-page":"753","DOI":"10.3390\/antiox12030753","article-title":"Nitric oxide\/nitric oxide synthase system in the pathogenesis of neurodegenerative disorders\u2014an overview","volume":"12","author":"Iova","year":"2023","journal-title":"Antioxidants"},{"key":"2026081811260448100_ref55","doi-asserted-by":"publisher","first-page":"710","DOI":"10.3389\/fimmu.2019.00710","article-title":"The iNOS activity during an immune response controls the CNS pathology in experimental autoimmune encephalomyelitis","volume":"10","author":"Sonar","year":"2019","journal-title":"Front Immunol"},{"key":"2026081811260448100_ref56","doi-asserted-by":"publisher","first-page":"12867","DOI":"10.1073\/pnas.0601075103","article-title":"NO synthase 2 (NOS2) deletion promotes multiple pathologies in a mouse model of Alzheimer's disease","volume":"103","author":"Colton","year":"2006","journal-title":"Proc Natl Acad Sci"},{"key":"2026081811260448100_ref57","doi-asserted-by":"publisher","first-page":"571","DOI":"10.3233\/JAD-2008-15405","article-title":"The effects of NOS2 gene deletion on mice expressing mutated human A\u03b2PP","volume":"15","author":"Colton","year":"2008","journal-title":"J Alzheimer's Dis"},{"key":"2026081811260448100_ref58","doi-asserted-by":"publisher","first-page":"7","DOI":"10.1186\/s13041-023-00996-1","article-title":"Genetic deletion of nitric oxide synthase 2 ameliorates Parkinson\u2019s disease pathology and neuroinflammation in a transgenic mouse model of synucleinopathy","volume":"16","author":"Kim","year":"2023","journal-title":"Mol Brain"},{"key":"2026081811260448100_ref59","volume-title":"The Zebrafish Book. A Guide for the Laboratory Use of Zebrafish (Danio Rerio). The Zebrafish Book. A Guide for the Laboratory Use of Zebrafish (Danio Rerio)","author":"Westerfield","year":"2000","edition":"4"},{"key":"2026081811260448100_ref60","doi-asserted-by":"publisher","first-page":"W401","DOI":"10.1093\/nar\/gku410","article-title":"CHOPCHOP: a CRISPR\/Cas9 and TALEN web tool for genome editing","volume":"42","author":"Montague","year":"2014","journal-title":"Nucleic Acids Res"},{"key":"2026081811260448100_ref61","doi-asserted-by":"publisher","first-page":"1874","DOI":"10.12688\/f1000research.21142.2","article-title":"Falco: high-speed FastQC emulation for quality control of sequencing data","volume":"8","author":"De Sena Brandine","year":"2021","journal-title":"F1000Research"},{"key":"2026081811260448100_ref62","doi-asserted-by":"publisher","first-page":"10","DOI":"10.14806\/ej.17.1.200","article-title":"Cutadapt removes adapter sequences from high-throughput sequencing reads","volume":"17","author":"Martin","year":"2011","journal-title":"EMBnet.journal"},{"key":"2026081811260448100_ref63","doi-asserted-by":"publisher","first-page":"15","DOI":"10.1093\/bioinformatics\/bts635","article-title":"STAR: ultrafast universal RNA-seq aligner","volume":"29","author":"Dobin","year":"2013","journal-title":"Bioinformatics"},{"key":"2026081811260448100_ref64","doi-asserted-by":"publisher","first-page":"923","DOI":"10.1093\/bioinformatics\/btt656","article-title":"featureCounts: an efficient general purpose program for assigning sequence reads to genomic features","volume":"30","author":"Liao","year":"2014","journal-title":"Bioinformatics"},{"key":"2026081811260448100_ref65","doi-asserted-by":"publisher","first-page":"550","DOI":"10.1186\/s13059-014-0550-8","article-title":"Moderated estimation of fold change and dispersion for RNA-seq data with DESeq2","volume":"15","author":"Love","year":"2014","journal-title":"Genome Biol"},{"key":"2026081811260448100_ref66","doi-asserted-by":"publisher","first-page":"W191","DOI":"10.1093\/nar\/gkz369","article-title":"G:profiler: a web server for functional enrichment analysis and conversions of gene lists (2019 update)","volume":"47","author":"Raudvere","year":"2019","journal-title":"Nucleic Acids Res"}],"container-title":["Human Molecular Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/hmg\/article-pdf\/35\/17\/ddag078\/70685560\/ddag078.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/hmg\/article-pdf\/35\/17\/ddag078\/70685560\/ddag078.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T15:27:31Z","timestamp":1787066851000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/hmg\/article\/doi\/10.1093\/hmg\/ddag078\/8763892"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026]]},"references-count":66,"journal-issue":{"issue":"17","published-print":{"date-parts":[[2026,8,10]]}},"URL":"https:\/\/doi.org\/10.1093\/hmg\/ddag078","relation":{},"ISSN":["0964-6906","1460-2083"],"issn-type":[{"value":"0964-6906","type":"print"},{"value":"1460-2083","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2026]]},"published":{"date-parts":[[2026]]},"article-number":"ddag078"}}