O-glycosylation in development of zebrafish

Abstract

Zebrafish are a useful vertebrate model for the analysis of development and organogenesis. As zebrafish are highly similar to humans at the molecular and developmental levels, they are also regarded as an excellent system for modeling human diseases. Concerning analysis of zebrafish glycosylation, the recent development in live imaging technology by metabolic labeling of glycans with azide-modified sugars and glycomic studies of embryos have provided information on the cell-surface expression patterns and structures of O-glycans in zebrafish. Moreover, a reverse genetic approach using antisense morpholino oligonucleotides in zebrafish, in combination with genome-wide association study of human patients, led to identification of a number of genes that are involved in O-glycosylation, mutations of which give rise to altered development and physiology. In particular, it is notable that several genes responsible for α-ystroglycan glycosylation were identified, suggesting their involvement in congenital muscular dystrophies. This essay highlights the contribution of zebrafish to elucidation of the structures, expression, and functions of O-glycans and describes notable examples that identified genes that are physiologically important for O-glycosylation.