{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T17:12:35Z","timestamp":1774977155313,"version":"3.50.1"},"reference-count":92,"publisher":"The Company of Biologists","issue":"3","license":[{"start":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T00:00:00Z","timestamp":1774915200000},"content-version":"vor","delay-in-days":30,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"crossref","award":["R21NS120079-01"],"award-info":[{"award-number":["R21NS120079-01"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"crossref","award":["R01NS116354-01"],"award-info":[{"award-number":["R01NS116354-01"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/100007703","name":"North Carolina State University","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100007703","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,3,1]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>CHARGE syndrome is a developmental disorder that affects 1 in 10,000 births, and patients exhibit both physical and behavioral characteristics. De novo variants in chromodomain helicase DNA binding protein 7 (CHD7) cause 67% of CHARGE syndrome cases. CHD7 is a DNA-binding chromatin remodeler with thousands of predicted binding sites in the genome, making it challenging to define molecular pathways linking loss of CHD7 to CHARGE syndrome phenotypes. To address this problem, here, we used a previously characterized zebrafish model of CHARGE syndrome to generate transcriptomic and proteomic datasets from larval zebrafish head tissue at two developmental time points. By integrating these datasets with differential expression, pathway and upstream regulator analyses, we identified multiple consistently dysregulated pathways and defined a set of candidate genes that link loss of chd7 with disease-related phenotypes. Finally, we identified that CRISPR\/Cas9-mediated knockdown of capgb, nefla or rdh5 phenocopies behavioral defects seen in chd7 mutants, functionally validating the roles of these genes. Our data provide a resource for further investigation of molecular mediators of CHD7 and a template to reveal functionally relevant therapeutic targets to alleviate specific aspects of CHARGE syndrome.<\/jats:p>","DOI":"10.1242\/dmm.052592","type":"journal-article","created":{"date-parts":[[2026,2,10]],"date-time":"2026-02-10T08:25:38Z","timestamp":1770711938000},"source":"Crossref","is-referenced-by-count":1,"title":["Multi-omic analyses identify molecular targets of Chd7 that contribute to CHARGE syndrome model phenotypes"],"prefix":"10.1242","volume":"19","author":[{"given":"Melody B.","family":"Hancock","sequence":"first","affiliation":[{"name":"North Carolina State University Department of Biological Sciences, , Raleigh, NC 27607 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Dana R.","family":"Ruby","sequence":"additional","affiliation":[{"name":"North Carolina State University Department of Biological Sciences, , Raleigh, NC 27607 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Rachael A.","family":"Bieler","sequence":"additional","affiliation":[{"name":"North Carolina State University Department of Biological Sciences, , Raleigh, NC 27607 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"D. Chris","family":"Cole","sequence":"additional","affiliation":[{"name":"North Carolina State University Department of Biological Sciences, , Raleigh, NC 27607 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8087-6181","authenticated-orcid":false,"given":"Kurt C.","family":"Marsden","sequence":"additional","affiliation":[{"name":"North Carolina State University Department of Biological Sciences, , Raleigh, NC 27607 ,","place":["USA"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"237","published-online":{"date-parts":[[2026,3,31]]},"reference":[{"key":"2026033103184781500_DMM052592C1","doi-asserted-by":"publisher","first-page":"1267","DOI":"10.1006\/bbrc.1993.2614","article-title":"Selective expression of the glutamate receptor channel delta 2 subunit in cerebellar Purkinje cells","volume":"197","author":"Araki","year":"1993","journal-title":"Biochem. Biophys. Res. Commun."},{"key":"2026033103184781500_DMM052592C2","doi-asserted-by":"publisher","first-page":"103661","DOI":"10.1016\/j.ejmg.2019.04.018","article-title":"Chemical screens in a zebrafish model of CHARGE syndrome identifies small molecules that ameliorate disease-like phenotypes in embryo","volume":"63","author":"Asad","year":"2020","journal-title":"Eur. J. Med. Genet."},{"key":"2026033103184781500_DMM052592C3","doi-asserted-by":"publisher","first-page":"3539","DOI":"10.1093\/hmg\/ddw198","article-title":"Rescue of neural crest-derived phenotypes in a zebrafish CHARGE model by Sox10 downregulation","volume":"25","author":"Asad","year":"2016","journal-title":"Hum. Mol. Genet."},{"key":"2026033103184781500_DMM052592C4","doi-asserted-by":"publisher","first-page":"1172933","DOI":"10.3389\/fcell.2023.1172933","article-title":"Vestibular physiology and function in zebrafish","volume":"11","author":"Baeza-Loya","year":"2023","journal-title":"Front. Cell Dev. Biol."},{"key":"2026033103184781500_DMM052592C5","doi-asserted-by":"publisher","first-page":"958","DOI":"10.1038\/nature08733","article-title":"CHD7 cooperates with PBAF to control multipotent neural crest formation","volume":"463","author":"Bajpai","year":"2010","journal-title":"Nature"},{"key":"2026033103184781500_DMM052592C6","doi-asserted-by":"publisher","first-page":"57","DOI":"10.1016\/j.ydbio.2013.07.026","article-title":"Knockdown of fbxl10\/kdm2bb rescues chd7 morphant phenotype in a zebrafish model of CHARGE syndrome","volume":"382","author":"Balow","year":"2013","journal-title":"Dev. Biol."},{"key":"2026033103184781500_DMM052592C7","doi-asserted-by":"publisher","first-page":"600","DOI":"10.1016\/j.tig.2015.05.009","article-title":"Functional insights into chromatin remodelling from studies on CHARGE syndrome","volume":"31","author":"Basson","year":"2015","journal-title":"Trends Genet."},{"key":"2026033103184781500_DMM052592C8","doi-asserted-by":"publisher","first-page":"2858","DOI":"10.1093\/hmg\/ddq189","article-title":"CHD8 interacts with CHD7, a protein which is mutated in CHARGE syndrome","volume":"19","author":"Batsukh","year":"2010","journal-title":"Hum. Mol. Genet."},{"key":"2026033103184781500_DMM052592C9","doi-asserted-by":"publisher","first-page":"171","DOI":"10.1038\/ejhg.2009.158","article-title":"Study of smell and reproductive organs in a mouse model for CHARGE syndrome","volume":"18","author":"Bergman","year":"2010","journal-title":"Eur. J. Hum. Genet."},{"key":"2026033103184781500_DMM052592C10","doi-asserted-by":"publisher","first-page":"159","DOI":"10.1177\/000992289803700302","article-title":"CHARGE association: an update and review for the primary pediatrician","volume":"37","author":"Blake","year":"1998","journal-title":"Clin. Pediatr."},{"key":"2026033103184781500_DMM052592C11","doi-asserted-by":"publisher","first-page":"151","DOI":"10.1177\/000992280504400207","article-title":"Adolescent and adult issues in CHARGE syndrome","volume":"44","author":"Blake","year":"2005","journal-title":"Clin. Pediatr."},{"key":"2026033103184781500_DMM052592C12","doi-asserted-by":"publisher","first-page":"3463","DOI":"10.1093\/hmg\/ddi375","article-title":"Multiple mutations in mouse Chd7 provide models for CHARGE syndrome","volume":"14","author":"Bosman","year":"2005","journal-title":"Hum. Mol. Genet."},{"key":"2026033103184781500_DMM052592C13","doi-asserted-by":"publisher","first-page":"795","DOI":"10.1016\/S0092-8674(00)80590-5","article-title":"Slit proteins bind Robo receptors and have an evolutionarily conserved role in repulsive axon guidance","volume":"96","author":"Brose","year":"1999","journal-title":"Cell"},{"key":"2026033103184781500_DMM052592C14","doi-asserted-by":"publisher","first-page":"2526","DOI":"10.1242\/jeb.003939","article-title":"Modulation of locomotor activity in larval zebrafish during light adaptation","volume":"210","author":"Burgess","year":"2007","journal-title":"J. Exp. Biol."},{"key":"2026033103184781500_DMM052592C90","doi-asserted-by":"publisher","first-page":"i884","DOI":"10.1093\/bioinformatics\/bty560","article-title":"fastp: an ultra-fast all-in-one FASTQ preprocessor","volume":"34","author":"Chen","year":"2018","journal-title":"Bioinformatics"},{"key":"2026033103184781500_DMM052592C15","doi-asserted-by":"publisher","first-page":"1047","DOI":"10.1016\/j.cell.2007.11.028","article-title":"Calcium signaling","volume":"131","author":"Clapham","year":"2007","journal-title":"Cell"},{"key":"2026033103184781500_DMM052592C16","doi-asserted-by":"publisher","first-page":"1175","DOI":"10.1242\/dev.126.6.1175","article-title":"The Drosophila kismet gene is related to chromatin-remodeling factors and is required for both segmentation and segment identity","volume":"126","author":"Daubresse","year":"1999","journal-title":"Development"},{"key":"2026033103184781500_DMM052592C91","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":"2026033103184781500_DMM052592C17","doi-asserted-by":"publisher","first-page":"86","DOI":"10.3389\/fnana.2017.00086","article-title":"Cerebellar vermis and midbrain hypoplasia upon conditional deletion of Chd7 from the embryonic mid-hindbrain region","volume":"11","author":"Donovan","year":"2017","journal-title":"Front. Neuroanat."},{"key":"2026033103184781500_DMM052592C18","doi-asserted-by":"publisher","first-page":"23","DOI":"10.1016\/0165-5876(95)01188-H","article-title":"Clinical manifestations of CHARGE Association","volume":"33","author":"Edwards","year":"1995","journal-title":"Int. J. Pediatr. Otorhinolaryngol."},{"key":"2026033103184781500_DMM052592C19","doi-asserted-by":"publisher","first-page":"e56883","DOI":"10.7554\/eLife.56883","article-title":"KDM5A mutations identified in autism spectrum disorder using forward genetics","volume":"9","author":"El Hayek","year":"2020","journal-title":"eLife"},{"key":"2026033103184781500_DMM052592C20","doi-asserted-by":"publisher","first-page":"a004242","DOI":"10.1101\/cshperspect.a004242","article-title":"Calcium signaling in cardiac myocytes","volume":"3","author":"Fearnley","year":"2011","journal-title":"Cold Spring Harbor Perspect. Biol."},{"key":"2026033103184781500_DMM052592C21","doi-asserted-by":"publisher","first-page":"62","DOI":"10.1016\/j.stem.2013.05.002","article-title":"The chromatin remodeler CHD7 regulates adult neurogenesis via activation of SoxC transcription factors","volume":"13","author":"Feng","year":"2013","journal-title":"Cell Stem Cell"},{"key":"2026033103184781500_DMM052592C22","doi-asserted-by":"publisher","first-page":"14758","DOI":"10.1038\/ncomms14758","article-title":"Chd7 is indispensable for mammalian brain development through activation of a neuronal differentiation programme","volume":"8","author":"Feng","year":"2017","journal-title":"Nat. Commun."},{"key":"2026033103184781500_DMM052592C23","doi-asserted-by":"publisher","first-page":"7923","DOI":"10.1167\/iovs.15-18069","article-title":"Mouse models for the dissection of CHD7 functions in eye development and the molecular basis for ocular defects in CHARGE syndrome","volume":"56","author":"Gage","year":"2015","journal-title":"Invest. Ophthalmol. Vis. Sci."},{"key":"2026033103184781500_DMM052592C24","doi-asserted-by":"publisher","first-page":"e2311720121","DOI":"10.1073\/pnas.2311720121","article-title":"CHD7 and SOX2 act in a common gene regulatory network during mammalian semicircular canal and cochlear development","volume":"121","author":"Gao","year":"2024","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2026033103184781500_DMM052592C25","doi-asserted-by":"publisher","first-page":"e113494","DOI":"10.1371\/journal.pone.0113494","article-title":"Kismet positively regulates glutamate receptor localization and synaptic transmission at the Drosophila neuromuscular junction","volume":"9","author":"Ghosh","year":"2014","journal-title":"PLoS ONE"},{"key":"2026033103184781500_DMM052592C26","doi-asserted-by":"publisher","first-page":"e43","DOI":"10.1002\/imt2.43","article-title":"Complex heatmap visualization","volume":"1","author":"Gu","year":"2022","journal-title":"iMeta"},{"key":"2026033103184781500_DMM052592C27","doi-asserted-by":"publisher","first-page":"2847","DOI":"10.1093\/bioinformatics\/btw313","article-title":"Complex heatmaps reveal patterns and correlations in multidimensional genomic data","volume":"32","author":"Gu","year":"2016","journal-title":"Bioinformatics"},{"key":"2026033103184781500_DMM052592C28","doi-asserted-by":"publisher","first-page":"22425","DOI":"10.1038\/s41598-022-23614-x","article-title":"The autism risk factor CHD8 is a chromatin activator in human neurons and functionally dependent on the ERK-MAPK pathway effector ELK1","volume":"12","author":"Haddad Derafshi","year":"2022","journal-title":"Sci. Rep."},{"key":"2026033103184781500_DMM052592C29","first-page":"41","article-title":"Challenging Behavior in CHARGE Syndrome","volume-title":"Mental Health Aspects of Developmental Disabilities","author":"Hartshorne","year":"2004"},{"key":"2026033103184781500_DMM052592C30","doi-asserted-by":"publisher","first-page":"257","DOI":"10.1002\/ajmg.a.30545","article-title":"Autistic like behavior in CHARGE syndrome","volume":"133A","author":"Hartshorne","year":"2005","journal-title":"Am. J. Med. Genet. A"},{"key":"2026033103184781500_DMM052592C31","doi-asserted-by":"publisher","first-page":"431","DOI":"10.1002\/ajmg.c.31588","article-title":"Behavior in CHARGE syndrome","volume":"175","author":"Hartshorne","year":"2017","journal-title":"Am. J. Med. Genet. C Semin. Med. Genet."},{"key":"2026033103184781500_DMM052592C32","doi-asserted-by":"publisher","first-page":"678","DOI":"10.1038\/nn.4258","article-title":"Chd7 cooperates with Sox10 and regulates the onset of CNS myelination and remyelination","volume":"19","author":"He","year":"2016","journal-title":"Nat. Neurosci."},{"key":"2026033103184781500_DMM052592C33","doi-asserted-by":"publisher","first-page":"839862","DOI":"10.3389\/fcvm.2022.839862","article-title":"A novel missense variant in actin binding domain of MYH7 is associated with left ventricular noncompaction","volume":"9","author":"Hesaraki","year":"2022","journal-title":"Front. Cardiovasc. Med."},{"key":"2026033103184781500_DMM052592C34","doi-asserted-by":"publisher","first-page":"e12839","DOI":"10.1111\/gbb.12839","article-title":"Morphological and sensorimotor phenotypes in a zebrafish CHARGE syndrome model are domain-dependent","volume":"22","author":"Hodorovich","year":"2023","journal-title":"Genes Brain Behav."},{"key":"2026033103184781500_DMM052592C35","doi-asserted-by":"publisher","first-page":"1","DOI":"10.1089\/zeb.2023.0052","article-title":"Effects of 4 testing arena sizes and 11 types of embryo media on sensorimotor behaviors in wild-type and chd7 mutant zebrafish larvae","volume":"21","author":"Hodorovich","year":"2024","journal-title":"Zebrafish"},{"key":"2026033103184781500_DMM052592C36","doi-asserted-by":"publisher","first-page":"4094","DOI":"10.1038\/s41380-025-02990-6","article-title":"Mutation of CHD7 impairs the output of neuroepithelium transition that is reversed by the inhibition of EZH2","volume":"30","author":"Huang","year":"2025","journal-title":"Mol. Psychiatry"},{"key":"2026033103184781500_DMM052592C37","doi-asserted-by":"publisher","first-page":"3139","DOI":"10.1242\/dev.047894","article-title":"The ATP-dependent chromatin remodeling enzyme CHD7 regulates pro-neural gene expression and neurogenesis in the inner ear","volume":"137","author":"Hurd","year":"2010","journal-title":"Development"},{"key":"2026033103184781500_DMM052592C38","doi-asserted-by":"publisher","first-page":"e50958","DOI":"10.15252\/embr.202050958","article-title":"Chromatin remodeller CHD7 is required for GABAergic neuron development by promoting PAQR3 expression","volume":"22","author":"Jamadagni","year":"2021","journal-title":"EMBO Rep."},{"key":"2026033103184781500_DMM052592C39","doi-asserted-by":"publisher","first-page":"32456","DOI":"10.1074\/jbc.M104949200","article-title":"Characterization of a dehydrogenase activity responsible for oxidation of 11-cis-retinol in the retinal pigment epithelium of mice with a disrupted RDH5 gene. A model for the human hereditary disease fundus albipunctatus","volume":"276","author":"Jang","year":"2001","journal-title":"J. Biol. Chem."},{"key":"2026033103184781500_DMM052592C40","doi-asserted-by":"publisher","first-page":"17946","DOI":"10.1016\/S0021-9258(18)38255-3","article-title":"Purification and expression of gCap39. An intracellular and secreted Ca2(+)-dependent actin-binding protein enriched in mononuclear phagocytes","volume":"265","author":"Johnston","year":"1990","journal-title":"J. Biol. Chem."},{"key":"2026033103184781500_DMM052592C41","doi-asserted-by":"publisher","first-page":"196","DOI":"10.1002\/stem.1822","article-title":"CHD7 maintains neural stem cell quiescence and prevents premature stem cell depletion in the adult hippocampus","volume":"33","author":"Jones","year":"2015","journal-title":"Stem Cells"},{"key":"2026033103184781500_DMM052592C42","doi-asserted-by":"publisher","first-page":"603","DOI":"10.1038\/nn.2791","article-title":"D-serine regulates cerebellar LTD and motor coordination through the \u03b42 glutamate receptor","volume":"14","author":"Kakegawa","year":"2011","journal-title":"Nat. Neurosci."},{"key":"2026033103184781500_DMM052592C43","doi-asserted-by":"publisher","first-page":"994412","DOI":"10.3389\/fcell.2022.994412","article-title":"Eyes on CHARGE syndrome: roles of CHD7 in ocular development","volume":"10","author":"Krueger","year":"2022","journal-title":"Front. Cell Dev. Biol."},{"key":"2026033103184781500_DMM052592C44","doi-asserted-by":"publisher","first-page":"109299","DOI":"10.1016\/j.exer.2022.109299","article-title":"Chromatin remodeler Chd7 regulates photoreceptor development and outer segment length","volume":"226","author":"Krueger","year":"2023","journal-title":"Exp. Eye Res."},{"key":"2026033103184781500_DMM052592C45","doi-asserted-by":"publisher","first-page":"1095","DOI":"10.3390\/genes5041095","article-title":"Altered actions of memantine and NMDA-induced currents in a new Grid2-deleted mouse line","volume":"5","author":"Kumagai","year":"2014","journal-title":"Genes"},{"key":"2026033103184781500_DMM052592C46","doi-asserted-by":"publisher","first-page":"1909","DOI":"10.1093\/hmg\/ddp112","article-title":"Defects in neural stem cell proliferation and olfaction in Chd7 deficient mice indicate a mechanism for hyposmia in human CHARGE syndrome","volume":"18","author":"Layman","year":"2009","journal-title":"Hum. Mol. Genet."},{"key":"2026033103184781500_DMM052592C47","doi-asserted-by":"publisher","first-page":"638674","DOI":"10.3389\/fcell.2021.638674","article-title":"Semaphorin regulation by the chromatin remodeler CHD7: An emerging genetic interaction shaping neural cells and neural crest in development and cancer","volume":"9","author":"Lettieri","year":"2021","journal-title":"Front. Cell Dev. Biol."},{"key":"2026033103184781500_DMM052592C48","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":"2026033103184781500_DMM052592C49","doi-asserted-by":"publisher","first-page":"849","DOI":"10.18240\/ijo.2023.06.03","article-title":"All-trans retinoic acid regulates the expression of MMP-2 and TGF-\u03b22 via RDH5 in retinal pigment epithelium cells","volume":"16","author":"Mao","year":"2023","journal-title":"Int. J. Ophthalmol."},{"key":"2026033103184781500_DMM052592C50","doi-asserted-by":"publisher","first-page":"363","DOI":"10.1126\/science.1185152","article-title":"Cbln1 is a ligand for an orphan glutamate receptor delta2, a bidirectional synapse organizer","volume":"328","author":"Matsuda","year":"2010","journal-title":"Science"},{"key":"2026033103184781500_DMM052592C51","doi-asserted-by":"publisher","first-page":"434","DOI":"10.1093\/hmg\/ddt435","article-title":"CHD7 and retinoic acid signaling cooperate to regulate neural stem cell and inner ear development in mouse models of CHARGE syndrome","volume":"23","author":"Micucci","year":"2014","journal-title":"Hum. Mol. Genet."},{"key":"2026033103184781500_DMM052592C52","doi-asserted-by":"publisher","first-page":"49","DOI":"10.1001\/archotol.1993.01880130051006","article-title":"Ear-nose-throat abnormalities in the CHARGE association","volume":"119","author":"Morgan","year":"1993","journal-title":"Arch. Otolaryngol. Head Neck Surg."},{"key":"2026033103184781500_DMM052592C53","doi-asserted-by":"publisher","first-page":"14116","DOI":"10.1073\/pnas.0703718104","article-title":"Ionotropic glutamate-like receptor delta2 binds D-serine and glycine","volume":"104","author":"Naur","year":"2007","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2026033103184781500_DMM052592C54","doi-asserted-by":"publisher","first-page":"2229","DOI":"10.1534\/genetics.106.065862","article-title":"Correlation of mRNA expression and protein abundance affected by multiple sequence features related to translational efficiency in Desulfovibrio vulgaris: a quantitative analysis","volume":"174","author":"Nie","year":"2006","journal-title":"Genetics"},{"key":"2026033103184781500_DMM052592C55","doi-asserted-by":"publisher","first-page":"e23306","DOI":"10.1002\/dvg.23306","article-title":"Hox genes: downstream \u201ceffectors\u201d of retinoic acid signaling in vertebrate embryogenesis","volume":"57","author":"Nolte","year":"2019","journal-title":"Genesis"},{"key":"2026033103184781500_DMM052592C56","doi-asserted-by":"publisher","first-page":"e21114","DOI":"10.7554\/eLife.21114","article-title":"CHARGE syndrome modeling using patient-iPSCs reveals defective migration of neural crest cells harboring CHD7 mutations","volume":"6","author":"Okuno","year":"2017","journal-title":"eLife"},{"key":"2026033103184781500_DMM052592C57","doi-asserted-by":"publisher","first-page":"300","DOI":"10.1016\/j.brainresbull.2016.07.013","article-title":"Actin filament-microtubule interactions in axon initiation and branching","volume":"126","author":"Pacheco","year":"2016","journal-title":"Brain Res. Bull."},{"key":"2026033103184781500_DMM052592C58","doi-asserted-by":"publisher","first-page":"223","DOI":"10.1016\/S0022-3476(81)80454-4","article-title":"Coloboma, congenital heart disease, and choanal atresia with multiple anomalies: CHARGE association","volume":"99","author":"Pagon","year":"1981","journal-title":"J. Pediatr."},{"key":"2026033103184781500_DMM052592C59","doi-asserted-by":"publisher","first-page":"e31650","DOI":"10.1371\/journal.pone.0031650","article-title":"Role of Chd7 in zebrafish: a model for CHARGE syndrome","volume":"7","author":"Patten","year":"2012","journal-title":"PLoS ONE"},{"key":"2026033103184781500_DMM052592C60","doi-asserted-by":"publisher","first-page":"82","DOI":"10.1016\/j.ydbio.2015.06.017","article-title":"A critical role for the chromatin remodeller CHD7 in anterior mesoderm during cardiovascular development","volume":"405","author":"Payne","year":"2015","journal-title":"Dev. Biol."},{"key":"2026033103184781500_DMM052592C61","doi-asserted-by":"publisher","first-page":"5621","DOI":"10.1210\/jc.2004-2474","article-title":"CHARGE syndrome includes hypogonadotropic hypogonadism and abnormal olfactory bulb development","volume":"90","author":"Pinto","year":"2005","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"2026033103184781500_DMM052592C62","doi-asserted-by":"publisher","first-page":"5702","DOI":"10.1038\/s41467-021-25846-3","article-title":"CHARGE syndrome protein CHD7 regulates epigenomic activation of enhancers in granule cell precursors and gyrification of the cerebellum","volume":"12","author":"Reddy","year":"2021","journal-title":"Nat. Commun."},{"key":"2026033103184781500_DMM052592C63","doi-asserted-by":"publisher","first-page":"a004259","DOI":"10.1101\/cshperspect.a004259","article-title":"Calcium signaling in neuronal development","volume":"3","author":"Rosenberg","year":"2011","journal-title":"Cold Spring Harbor Perspect. Biol."},{"key":"2026033103184781500_DMM052592C64","doi-asserted-by":"publisher","first-page":"211","DOI":"10.1136\/jmg.2005.036160","article-title":"Phenotypic spectrum of CHARGE syndrome in fetuses with CHD7 truncating mutations correlates with expression during human development","volume":"43","author":"Sanlaville","year":"2006","journal-title":"J. Med. Genet."},{"key":"2026033103184781500_DMM052592C65","doi-asserted-by":"publisher","first-page":"590","DOI":"10.1101\/gr.086983.108","article-title":"Genomic distribution of CHD7 on chromatin tracks H3K4 methylation patterns","volume":"19","author":"Schnetz","year":"2009","journal-title":"Genome Res."},{"key":"2026033103184781500_DMM052592C66","doi-asserted-by":"publisher","first-page":"e1001023","DOI":"10.1371\/journal.pgen.1001023","article-title":"CHD7 targets active gene enhancer elements to modulate ES cell-specific gene expression","volume":"6","author":"Schnetz","year":"2010","journal-title":"PLoS Genet."},{"key":"2026033103184781500_DMM052592C67","doi-asserted-by":"publisher","first-page":"4396","DOI":"10.1093\/hmg\/ddu156","article-title":"CHARGE and Kabuki syndromes: a phenotypic and molecular link","volume":"23","author":"Schulz","year":"2014","journal-title":"Hum. Mol. Genet."},{"key":"2026033103184781500_DMM052592C68","doi-asserted-by":"publisher","first-page":"997","DOI":"10.1007\/s00439-014-1444-2","article-title":"CHD7, the gene mutated in CHARGE syndrome, regulates genes involved in neural crest cell guidance","volume":"133","author":"Schulz","year":"2014","journal-title":"Hum. Genet."},{"key":"2026033103184781500_DMM052592C69","doi-asserted-by":"publisher","first-page":"1825","DOI":"10.1242\/dev.00413","article-title":"RhoA and Rac1 GTPases mediate the dynamic rearrangement of actin in peripheral glia","volume":"130","author":"Sepp","year":"2003","journal-title":"Development"},{"key":"2026033103184781500_DMM052592C70","doi-asserted-by":"publisher","first-page":"101866","DOI":"10.1016\/j.nicl.2019.101866","article-title":"Quantitative brain morphological analysis in CHARGE syndrome","volume":"23","author":"Shiohama","year":"2019","journal-title":"NeuroImage. Clin."},{"key":"2026033103184781500_DMM052592C71","doi-asserted-by":"publisher","first-page":"2089","DOI":"10.1093\/cvr\/cvad059","article-title":"CHARGE syndrome-associated CHD7 acts at ISL1-regulated enhancers to modulate second heart field gene expression","volume":"119","author":"Stathopoulou","year":"2023","journal-title":"Cardiovasc. Res."},{"key":"2026033103184781500_DMM052592C92","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":"2026033103184781500_DMM052592C72","doi-asserted-by":"publisher","first-page":"15975","DOI":"10.1073\/pnas.0603883103","article-title":"Galectin-1 is essential in tumor angiogenesis and is a target for antiangiogenesis therapy","volume":"103","author":"Thijssen","year":"2006","journal-title":"Proc. Natl. Acad. Sci. USA"},{"key":"2026033103184781500_DMM052592C74","doi-asserted-by":"publisher","first-page":"244","DOI":"10.1083\/jcb.86.1.244","article-title":"The organization of actin filaments in the stereocilia of cochlear hair cells","volume":"86","author":"Tilney","year":"1980","journal-title":"J. Cell Biol."},{"key":"2026033103184781500_DMM052592C75","doi-asserted-by":"publisher","first-page":"4443","DOI":"10.1093\/hmg\/ddv180","article-title":"Kabuki syndrome genes KMT2D and KDM6A: functional analyses demonstrate critical roles in craniofacial, heart and brain development","volume":"24","author":"Van Laarhoven","year":"2015","journal-title":"Hum. Mol. Genet."},{"key":"2026033103184781500_DMM052592C76","doi-asserted-by":"publisher","first-page":"306","DOI":"10.1002\/ajmg.a.30559","article-title":"Updated diagnostic criteria for CHARGE syndrome: a proposal","volume":"133A","author":"Verloes","year":"2005","journal-title":"Am. J. Med. Genet. A"},{"key":"2026033103184781500_DMM052592C77","doi-asserted-by":"publisher","first-page":"471","DOI":"10.1007\/s10882-007-9064-6","article-title":"Psychiatric diagnoses and psychotropic medications in CHARGE syndrome: a pediatric survey","volume":"19","author":"Wachtel","year":"2007","journal-title":"J. Dev. Phys. Disabil."},{"key":"2026033103184781500_DMM052592C78","doi-asserted-by":"publisher","first-page":"11208","DOI":"10.1002\/jcp.27771","article-title":"NEFLb impairs early nervous system development via regulation of neuron apoptosis in zebrafish","volume":"234","author":"Wang","year":"2019","journal-title":"J. Cell. Physiol."},{"key":"2026033103184781500_DMM052592C79","doi-asserted-by":"publisher","first-page":"e585","DOI":"10.1002\/cpz1.585","article-title":"Exploring epigenomic datasets by ChIPseeker","volume":"2","author":"Wang","year":"2022","journal-title":"Curr. Protoc."},{"key":"2026033103184781500_DMM052592C80","doi-asserted-by":"publisher","first-page":"1133","DOI":"10.3390\/genes12081133","article-title":"The mechanisms of CHD8 in neurodevelopment and autism spectrum disorders","volume":"12","author":"Weissberg","year":"2021","journal-title":"Genes"},{"key":"2026033103184781500_DMM052592C81","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.c.31595","article-title":"Distinct cerebellar foliation anomalies in a CHD7 haploinsufficient mouse model of CHARGE syndrome","volume":"175","author":"Whittaker","year":"2017","journal-title":"Am. J. Med. Genet. C Semin. Med. Genet."},{"key":"2026033103184781500_DMM052592C82","doi-asserted-by":"publisher","first-page":"874","DOI":"10.1172\/JCI83408","article-title":"The chromatin remodeling factor CHD7 controls cerebellar development by regulating reelin expression","volume":"127","author":"Whittaker","year":"2017","journal-title":"J. Clin. Invest."},{"key":"2026033103184781500_DMM052592C93","doi-asserted-by":"crossref","DOI":"10.1007\/978-3-319-24277-4","volume-title":"ggplot2: Elegant Graphics for Data Analysis","author":"Wickham","year":"2016"},{"key":"2026033103184781500_DMM052592C83","doi-asserted-by":"publisher","first-page":"e3002786","DOI":"10.1371\/journal.pbio.3002786","article-title":"Chromatin remodeller Chd7 is developmentally regulated in the neural crest by tissue-specific transcription factors","volume":"22","author":"Williams","year":"2024","journal-title":"PLoS Biol."},{"key":"2026033103184781500_DMM052592C84","doi-asserted-by":"publisher","first-page":"188","DOI":"10.1038\/9707","article-title":"Mutations in the gene encoding 11-cis retinol dehydrogenase cause delayed dark adaptation and fundus albipunctatus","volume":"22","author":"Yamamoto","year":"1999","journal-title":"Nat. Genet."},{"key":"2026033103184781500_DMM052592C85","doi-asserted-by":"publisher","first-page":"17445","DOI":"10.1038\/s41598-020-74537-4","article-title":"CHD7 promotes neural progenitor differentiation in embryonic stem cells via altered chromatin accessibility and nascent gene expression","volume":"10","author":"Yao","year":"2020","journal-title":"Sci. Rep."},{"key":"2026033103184781500_DMM052592C86","doi-asserted-by":"publisher","first-page":"a018309","DOI":"10.1101\/cshperspect.a018309","article-title":"Neurofilaments and neurofilament proteins in health and disease","volume":"9","author":"Yuan","year":"2017","journal-title":"Cold Spring Harbor Perspect. Biol."},{"key":"2026033103184781500_DMM052592C87","doi-asserted-by":"publisher","first-page":"220","DOI":"10.1038\/nature12141","article-title":"De novo mutations in histone-modifying genes in congenital heart disease","volume":"498","author":"Zaidi","year":"2013","journal-title":"Nature"},{"key":"2026033103184781500_DMM052592C88","doi-asserted-by":"publisher","first-page":"674","DOI":"10.1002\/ajmg.a.33323","article-title":"Molecular and phenotypic aspects of CHD7 mutation in CHARGE syndrome","volume":"152A","author":"Zentner","year":"2010","journal-title":"Am. J. Med. Genet. A"},{"key":"2026033103184781500_DMM052592C89","doi-asserted-by":"publisher","first-page":"707","DOI":"10.1016\/j.cell.2013.03.030","article-title":"Integrated systems approach identifies genetic nodes and networks in late-onset Alzheimer's disease","volume":"153","author":"Zhang","year":"2013","journal-title":"Cell"}],"container-title":["Disease Models &amp; Mechanisms"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.052592\/3684456\/dmm052592.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.052592\/3692571\/dmm052592.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/journals.biologists.com\/dmm\/article-pdf\/doi\/10.1242\/dmm.052592\/3692571\/dmm052592.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,31]],"date-time":"2026-03-31T07:18:54Z","timestamp":1774941534000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.biologists.com\/dmm\/article\/19\/3\/dmm052592\/371215\/Multi-omic-analyses-identify-molecular-targets-of"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,3,1]]},"references-count":92,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2026,3,1]]}},"URL":"https:\/\/doi.org\/10.1242\/dmm.052592","relation":{},"ISSN":["1754-8403","1754-8411"],"issn-type":[{"value":"1754-8403","type":"print"},{"value":"1754-8411","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,3,1]]},"article-number":"dmm052592"}}