Plastic Surgery Research Council
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PSRC 60th Annual Meeting

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Analysis Of The Role Of Wls In Craniofacial Development
Lucie J. Rochard, PhD, Yawei Kong, PhD, Kenta C. Kawasaki, N/A, Michael Grimaldi, n/a, Tatiana Hoyos, MD, Eric C. Liao, MD, PhD.
Massachussets General Hospital, Boston, MA, USA.

Background: Wnt signaling is a critical pathway regulating craniofacial development, where dysregulation leads to orofacial clefts. Intracellular trafficking and secretion of wnt ligands is chaperoned by wntless (wls). In human, the WLS gene is on the short arm of the chromosome 1 (1p31.3) and several deletions of this region associated with craniofacial malformations have been reported. We hypothesize that the wls gene functions to modulate wnt signaling important in morphogenesis of the craniofacial skeleton.
Material and Methods:
Gene expression analysis by whole mount in situ hybridization (WISH) of wls was performed in zebrafish, across embryonic timepoints, in wildtype and mutant embryos. Phenotypic analysis of the wls mutant was also carried out, by WISH to examine markers of neural crest and craniofacial development, and with Alcian blue stain to delineate the skeletal structures.
Results:
Gene expression analysis of wls via WISH during embryogenesis detected transcripts in the presumptive neural tissue, in the pharyngeal arches, and later in the palate. In the oropharynx, the wls expression domain corresponded to that of wnt9a. Further, the wls mutants exhibited dysmorphology of skeletal structures in the head. Each element of jaws appeared shorter and the neurocranium was also smaller in all direction, revealing a problem in extension mechanisms during craniofacial morphogenesis.
Conclusions: This study describes the requirement of wls in craniofacial development. Our group has previously defined the role of wnt9a, frzb and fzd7a in craniofacial morphogenesis, to participate in regulating convergence and extension morphogenetic mechanisms in formation of the palate, and the formation of the lower jaw. Our studies of wls take advantage of this body of work to elucidate the role of wls and wnt signaling pathway in palatogenesis. Our future experiments will focus on the studies of the secretion of wnt9a in wls mutants and regulation of wls by other signaling pathways that regulate craniofacial development.


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