Optic vesicle morphogenesis requires primary cilia.
Dev Biol
; 462(2): 119-128, 2020 06 15.
Article
en En
| MEDLINE
| ID: mdl-32169553
ABSTRACT
Arl13b is a gene known to regulate ciliogenesis. Functional alterations in this gene's activity have been associated with Joubert syndrome. We found that in Arl13 null mouse embryos the orientation of the optic cup is inverted, such that the lens is abnormally surrounded by an inverted optic cup whose retina pigmented epithelium is oddly facing the surface ectoderm. Loss of Arl13b leads to the disruption of optic vesicle's patterning and expansion of ventral fates. We show that this phenotype is consequence of miss-regulation of Sonic hedgehog (Shh) signaling and demonstrate that the Arl13b-/- eye phenotype can be rescued by deletion of Gli2, a downstream effector of the Shh pathway. This work identified an unexpected role of primary cilia during the morphogenetic movements required for the formation of the eye.
Palabras clave
Texto completo:
1
Colección:
01-internacional
Base de datos:
MEDLINE
Asunto principal:
Cilios
/
Factores de Ribosilacion-ADP
/
Ojo
Tipo de estudio:
Prognostic_studies
Idioma:
En
Revista:
Dev Biol
Año:
2020
Tipo del documento:
Article
País de afiliación:
Estados Unidos