Article
Zebrafish models of idiopathic scoliosis link cerebrospinal fluid flow defects to spine curvature.
Science (New York, N.Y.) - 10 Jun 2016
Grimes D T, Boswell C W, Morante N F C, Henkelman R M, Burdine R D, Ciruna B
Abstract excerpt
Idiopathic scoliosis (IS) affects 3% of children worldwide, yet the mechanisms underlying this spinal deformity remain unknown. Here we show that ptk7 mutant zebrafish, a faithful developmental model of IS, exhibit defects in ependymal cell cilia development and cerebrospinal fluid (CSF) flow. Transgenic reintroduction of Ptk7 in motile ciliated lineages prevents scoliosis in ptk7 mutants, and mutation of...
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