The ciliary Frizzled-like receptor Tmem67 regulates canonical Wnt/β-catenin signalling in the developing cerebellum via Hoxb5

Abstract excerpt

Abstract Primary cilia defects result in a group of related pleiotropic malformation syndromes known as ciliopathies, often characterised by cerebellar developmental and foliation defects. Here, we describe the cerebellar anatomical and signalling defects in theTmem67tm1(Dgen)/Hknockout mouse. At mid-gestation,Tmem67mutant cerebella were hypoplastic and had aberrantly high canonical Wnt/β-catenin signalling,...

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