Ontology highlight
ABSTRACT:
SUBMITTER: Grimes DT
PROVIDER: S-EPMC5574193 | biostudies-other | 2016 Jun
REPOSITORIES: biostudies-other
Science (New York, N.Y.) 20160601 6291
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 multiple independent cilia motility genes yields IS phenotypes. We define a finite developm ...[more]