ZFIN ID: ZDB-PUB-120815-13
Craniofacial abnormalities result from knock down of nonsyndromic clefting gene, crispld2, in zebrafish
Yuan, Q., Chiquet, B.T., Devault, L., Warman, M.L., Nakamura, Y., Swindell, E.C., and Hecht, J.T.
Date: 2012
Source: Genesis (New York, N.Y. : 2000)   50(12): 871-881 (Journal)
Registered Authors: Swindell, Eric C.
Keywords: cleft lip and palate, CRISPLD2, zebrafish, neural crest cells, craniofacial development
MeSH Terms:
  • Amino Acid Sequence
  • Animals
  • Brain/abnormalities
  • Cell Adhesion Molecules/genetics*
  • Cell Adhesion Molecules/metabolism
  • Cleft Lip/genetics
  • Cleft Palate/genetics
  • Craniofacial Abnormalities/genetics*
  • Gene Knockdown Techniques
  • Glycoproteins/genetics*
  • Glycoproteins/metabolism
  • Jaw/embryology
  • Jaw Abnormalities/genetics
  • Molecular Sequence Data
  • Morpholinos
  • Neural Crest/abnormalities
  • Neural Crest/embryology
  • Palate/abnormalities
  • Palate/embryology
  • Zebrafish
  • Zebrafish Proteins/genetics*
  • Zebrafish Proteins/metabolism
PubMed: 22887593 Full text @ Genesis

Nonsyndromic cleft lip and palate (NSCLP), a common birth defect, affects 4000 newborns in the US each year. Previously, we described an association between CRISPLD2 and NSCLP and showed Crispld2 expression in the murine palate. These results suggested that a perturbation in CRISPLD2 activity affects craniofacial development. Here, we describe crispld2 expression and the phenotypic consequence of its loss of function in zebrafish. crispld2 was expressed at all stages of zebrafish morphogenesis examined and localized to the rostral end by 1-day post fertilization. Morpholino knockdown of crispld2 resulted in significant jaw and palatal abnormalities in a dose dependent manner. Loss of crispld2 caused aberrant patterning of neural crest cells (NCC) suggesting that crispld2 is necessary for normal NCC formation. Altogether, we show that crispld2 plays a significant role in the development of the zebrafish craniofacies and alteration of normal protein levels disturbs palate and jaw formation. These data provide support for a role of CRISPLD2 in NSCLP.