Figure 14

Schematic of dose-dependent defects observed with loss of Dync1h1. Illustrations of 3-dpf zebrfish cone photoreceptors from wild-type, low-dose dync1h1 morphants, and cnb mutants. Wild-type photoreceptors typically showed non-vesiculated outer segments, well organized connecting cilium and basal body, tightly packed mitochondria in the ellipsoid region, Golgi apparatus just apical to the nucleus in the myoid region, and numerous inputs and floating ribbons in the synaptic region. Photoreceptors of dync1h1 morphants and cnb mutants showed a spectrum of phenotypes with the most normal photoreceptors positioned in the central retina and more severely affected photoreceptors at the periphery. Cellular defects included increased incidence of outer segment vesiculation, varying degrees of organelle dispersion and depolarization, and under-development of the synaptic region. OS, outer segment; IS, inner segment; CB cell body; SR, synaptic region; E, ellipsoid region; M, myoid region.