Fuz expression critical for craniofacial and tooth development: Study

Today, during the 89th General Session & Exhibition of the International Association for Dental Research, held in conjunction with the 40th Annual Meeting of the American Association for Dental Research and the 35th Annual Meeting of the Canadian Association for Dental Research, lead researcher Z. Zhang will hold an oral presentation on a study titled "Fuz is Essential for Craniofacial and Tooth Development." Fuz has been identified as a planar cell polarity effector gene and shown to be involved in ciliogenesis.

The objective of this study was to determine if fuz expression was critical for craniofacial and tooth development. In this study, fuz-/- mutant mice and mouse embryo fibroblasts were used to analyze the effect of fuz expression on tooth development and gene expression pathways, respectively. Immunohistochemical, molecular and biochemical assays were used to understand the defects and molecular mechanisms underlying the phenotype of the Fuz-/- mice.

The Fuz-/- mutant mice exhibited massive craniofacial deformities such as a hypoplastic mandible, a complete lack of upper and lower incisor development, malformed molars, hyperplastic Meckel's cartilage and missing eyes and tongue. Fuz has an essential role in ciliogenesis, and the primary cilium has been shown to repress canonical Wnt signaling. The results of the data collected suggests that Fuz is involved in a negative feedback loop of canonical Wnt signalling regulation, and this feedback loop is essential for precise regulation of craniofacial development. Support for this research was provided from grant DE13941 from the National Institute of Dental and Craniofacial Research.