biostudies-literatureSinagoga KLNEI NIH HHSNCATS NIH HHSNational Institutes of Healthdev187047https://www.ebi.ac.uk/biostudies/studies/S-EPMC7375471biostudies-literatureUnknown147(21)A crucial step in eye development is the closure of the choroid fissure (CF), a transient structure in the ventral optic cup through which vasculature enters the eye and ganglion cell axons exit. Although many factors have been identified that function during CF closure, the molecular and cellular mechanisms mediating this process remain poorly understood. Failure of CF closure results in colobomas. Recently, <i>MITF</i> was shown to be mutated in a subset of individuals with colobomas, but how MITF functions during CF closure is unknown. To address this issue, zebrafish with mutations in <i>mitfa</i> and <i>tfec</i>, two members of the Mitf family of transcription factors, were analyzed and their functions during CF closure determined. <i>mitfa;tfec</i> mutants possess severe colobomas anDevelopment (Cambridge, England)Mitf-family transcription factor function is required within cranial neural crest cells to promote choroid fissure closure.PMC7375471UL1 TR000058P30 EY008098T32 EY017271R01 EY018005R01-EY18005Lister JAGross JMSpencer SASinagoga KLLarimer-Picciani AMGeorge SMfalseMitf-family transcription factor function is required within cranial neural crest cells to promote choroid fissure closure.A crucial step in eye development is the closure of the choroid fissure (CF), a transient structure in the ventral optic cup through which vasculature enters the eye and ganglion cell axons exit. Although many factors have been identified that function during CF closure, the molecular and cellular mechanisms mediating this process remain poorly understood. Failure of CF closure results in colobomas. Recently, <i>MITF</i> was shown to be mutated in a subset of individuals with colobomas, but how MITF functions during CF closure is unknown. To address this issue, zebrafish with mutations in <i>mitfa</i> and <i>tfec</i>, two members of the Mitf family of transcription factors, were analyzed and their functions during CF closure determined. <i>mitfa;tfec</i> mutants possess severe colobomas an2020-01-01T00:00:00Z2020 Jul2026-05-07T16:58:31.781Z2022-02-10T19:11:48.079ZS-EPMC73754713254101110.1242/dev.187047