RRC ID 2481
Author Deflorian G, Tiso N, Ferretti E, Meyer D, Blasi F, Bortolussi M, Argenton F.
Title Prep1.1 has essential genetic functions in hindbrain development and cranial neural crest cell differentiation.
Journal Development
Abstract In this study we analysed the function of the Meinox gene prep1.1 during zebrafish development. Meinox proteins form heterotrimeric complexes with Hox and Pbx members, increasing the DNA binding specificity of Hox proteins in vitro and in vivo. However, a role for a specific Meinox protein in the regulation of Hox activity in vivo has not been demonstrated. In situ hybridization showed that prep1.1 is expressed maternally and ubiquitously up to 24 hours post-fertilization (hpf), and restricted to the head from 48 hpf onwards. Morpholino-induced prep1.1 loss-of-function caused significant apoptosis in the CNS. Hindbrain segmentation and patterning was affected severely, as revealed by either loss or defective expression of several hindbrain markers (foxb1.2/mariposa, krox20, pax2.1 and pax6.1), including anteriorly expressed Hox genes (hoxb1a, hoxa2 and hoxb2), the impaired migration of facial nerve motor neurons, and the lack of reticulospinal neurons (RSNs) except Mauthner cells. Furthermore, the heads of prep1.1 morphants lacked all pharyngeal cartilages. This was not caused by the absence of neural crest cells or their impaired migration into the pharyngeal arches, as shown by expression of dlx2 and snail1, but by the inability of these cells to differentiate into chondroblasts. Our results indicate that prep1.1 has a unique genetic function in craniofacial chondrogenesis and, acting as a member of Meinox-Pbc-Hox trimers, it plays an essential role in hindbrain development.
Volume 131(3)
Pages 613-27
Published 2004-2
DOI 10.1242/dev.00948
PII dev.00948
PMID 14711874
MeSH Animals Branchial Region / embryology Branchial Region / metabolism Cartilage / abnormalities Cartilage / metabolism Cell Differentiation / physiology* Cell Nucleus / metabolism DNA-Binding Proteins* Embryo, Nonmammalian / metabolism* Gastrula / metabolism Head / abnormalities Head / physiology Homeodomain Proteins / genetics* Homeodomain Proteins / metabolism Neural Crest / embryology* Neural Crest / metabolism Protein Transport / physiology Rhombencephalon / embryology* Rhombencephalon / metabolism Transcription Factors Zebrafish / embryology Zebrafish / metabolism Zebrafish Proteins / metabolism
IF 5.763
Times Cited 41
Zebrafish ?