Hedgehog signaling regulates cell motility and optic fissure and stalk formation during vertebrate eye morphogenesis.

ABSTRACT: Establishment of precise three-dimensional tissue structure is vital for organ function. In the visual system, optic fissure and stalk morphogenesis is a crucial yet poorly understood process, disruptions of which can lead to coloboma, a birth defect causing visual impairment. Here, we use four-dimensional imaging, cell tracking, and molecular genetics in zebrafish to define the cell movements underlying normal optic fissure and stalk formation. We determine how these events are disrupted in a coloboma model in which the Hedgehog (Hh) receptor ptch2 is lost, resulting in overactive Hh signaling. In the ptch2 mutant, cells exhibit defective motile behaviors and morphology. Cells that should contribute to the fissure do not arrive at their correct position, and instead contribu

SUBMITTER: Gordon HB 

PROVIDER: S-EPMC6262791 | biostudies-literature | 2018 Nov

REPOSITORIES: biostudies-literature