|ZFIN ID: ZDB-PUB-200617-10|
Mitf-family transcription factor function is required within cranial neural crest cells to promote choroid fissure closure
Sinagoga, K.L., Larimer-Picciani, A.M., George, S.M., Spencer, S.A., Lister, J.A., Gross, J.M.
|Source:||Development (Cambridge, England) 147(21): (Journal)|
|Registered Authors:||Gross, Jeffrey, Lister, James A.|
|Keywords:||Choroid fissure, Coloboma, Mitf, Neural crest, Zebrafish|
|PubMed:||32541011 Full text @ Development|
Sinagoga, K.L., Larimer-Picciani, A.M., George, S.M., Spencer, S.A., Lister, J.A., Gross, J.M. (2020) Mitf-family transcription factor function is required within cranial neural crest cells to promote choroid fissure closure. Development (Cambridge, England). 147(21):.
ABSTRACTA critical step in eye development is 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. While 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, MITF was shown to be mutated in a subset of human coloboma patients, but how MITF functions during CF closure is unknown. To address this question, zebrafish with mutations in mitfa and tfec, two members of the Mitf-family of transcription factors, were analyzed and their functions during CF closure determined. mitfa;tfec mutants possess severe colobomas and our data demonstrate that Mitf activity is required within cranial neural crest cells (cNCCs) during CF closure. In the absence of Mitf function, cNCC migration and localization in the optic cup are perturbed. These data shed light on the cellular mechanisms underlying colobomas in patients with MITF mutations and identify a novel role for Mitf function in cNCCs during CF closure.
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