ZFIN ID: ZDB-PUB-150825-57
Crispld2 is required for neural crest cell migration and cell viability during zebrafish craniofacial development
Swindell, E.C., Yuan, Q., Maili, L.E., Tandon, B., Wagner, D.S., Hecht, J.T.
Date: 2015
Source: Genesis (New York, N.Y. : 2000)   53(10): 660-7 (Journal)
Registered Authors: Swindell, Eric C., Wagner, Daniel
Keywords: Birth Defects, Early Development, Migration, Neural Crest, Other
MeSH Terms:
  • Animals
  • Apoptosis/genetics
  • Body Patterning/genetics
  • Cell Differentiation/genetics
  • Cell Movement/genetics*
  • Cell Survival/genetics
  • Fluorescent Antibody Technique
  • Gene Expression Regulation, Developmental
  • Gene Knockdown Techniques
  • Glycoproteins/genetics*
  • Glycoproteins/metabolism
  • Head/embryology
  • Humans
  • Microscopy, Confocal
  • Neural Crest/cytology
  • Neural Crest/embryology
  • Neural Crest/metabolism*
  • RNA, Messenger/genetics
  • RNA, Messenger/metabolism
  • Skull/embryology
  • Skull/metabolism
  • Time Factors
  • Time-Lapse Imaging
  • Zebrafish/embryology
  • Zebrafish/genetics*
  • Zebrafish/metabolism
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/metabolism
PubMed: 26297922 Full text @ Genesis
The CAP superfamily member, CRISPLD2, has previously been shown to be associated with nonsyndromic cleft lip and palate (NSCL/P) in human populations and to be essential for normal craniofacial development in the zebrafish. Additionally, in rodent models, CRISPLD2 has been shown to play a role in normal lung and kidney development. However, the specific role of CRISPLD2 during these developmental processes has yet to be determined. In this study, we demonstrate that Crispld2 protein localizes to the orofacial region of the zebrafish embryo and knockdown of crispld2 results in abnormal migration of neural crest cells during both early and late time points. We also show an increase in cell death after crispld2 knockdown as well as an increase in apoptotic marker genes. Our data suggests that Crispld2 modulates the migration, differentiation and/or survival of NCCs during early craniofacial development. These results indicate an important role for Crispld2 in neural crest cell migration during craniofacial development and suggests involvement of Crispld2 in cell viability during formation of the orofacies. This article is protected by copyright. All rights reserved.