A morpholino-based screen to identify novel genes involved in craniofacial morphogenesis

Vida Senkus Melvin, Weiguo Feng, Laura Hernandez-Lagunas, Kristin Bruk Artinger, Trevor J. Williams

Research output: Contribution to journalArticlepeer-review

47 Scopus citations


Background: The regulatory mechanisms underpinning facial development are conserved between diverse species. Therefore, results from model systems provide insight into the genetic causes of human craniofacial defects. Previously, we generated a comprehensive dataset examining gene expression during development and fusion of the mouse facial prominences. Here, we used this resource to identify genes that have dynamic expression patterns in the facial prominences, but for which only limited information exists concerning developmental function. Results: This set of ~80 genes was used for a high-throughput functional analysis in the zebrafish system using Morpholino gene knockdown technology. This screen revealed three classes of cranial cartilage phenotypes depending upon whether knockdown of the gene affected the neurocranium, viscerocranium, or both. The targeted genes that produced consistent phenotypes encoded proteins linked to transcription (meis1, meis2a, tshz2, vgll4l), signaling (pkdcc, vlk, macc1, wu:fb16h09), and extracellular matrix function (smoc2). The majority of these phenotypes were not altered by reduction of p53 levels, demonstrating that both p53-dependent and -independent mechanisms were involved in the craniofacial abnormalities. Conclusions: This Morpholino-based screen highlights new genes involved in development of the zebrafish craniofacial skeleton with wider relevance to formation of the face in other species, particularly mouse and human. Developmental Dynamics 242:817-831, 2013.

Original languageEnglish (US)
Pages (from-to)817-831
Number of pages15
JournalDevelopmental Dynamics
Issue number7
StatePublished - Jul 2013
Externally publishedYes


  • Branchial arches
  • Craniofacial development
  • Morpholino
  • Neural crest derived cartilages
  • Zebrafish


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