It is intriguing however, that despite the dramatic ciliary and Shh phenotypes in in vitro cell culture models, the in vivo role of Sdccag8 in modulating Hh signaling appears to be restricted to a few tissues—the developing rib cage and limb buds, as our analysis of Sdccag8gt/gt mice did not reveal any other gross abnormalities that are frequently associated with impaired Hh signaling, such as exencephaly, hydrocephalus or cleft palate. This evidence concerns the gene SDCCAG8 and Hydrocephalus.