... -attributable hearing loss. In addition, atp11a mutant zebrafish raised in a standard light cycle have reduced photoreceptor outer segments, the severity of which is lessened when mutant larvae are raised in the dark. Photoreceptors that do remain in homozygous atp11a mutants undergo mitochondrial fission...

...Lalitha Sastry; Johnathan Rylee; Simpla Mahato; Andrew C. Zelhof ABSTRACT Specialized membrane and cortical protein regions are common features of cells and are utilized to isolate differential cellular functions. In Drosophila photoreceptors, the apical membrane domain is defined by two distinct...

...Hitomi Yamashita; Yuka Ochi; Yumi Yamada; Shogo Sasaki; Tatsuya Tago; Takunori Satoh; Akiko K. Satoh ABSTRACT Polarized transport is essential for constructing multiple plasma membrane domains in the cell. Drosophila photoreceptors are an excellent model system to study the mechanisms of polarized...

... pronounced for mouse than human photopigment. G-protein-coupled receptor Melanopsin Optogenetics Photoreceptor Human Frontier Science Program http://dx.doi.org/10.13039/100004412 RGP0034/2014 Medical Research Council http://dx.doi.org/10.13039/501100007155 MR/N012992/1 EP...

...Xiaoming Fang; Andrew A. Peden; Fredericus J. M. van Eeden; Jarema J. Malicki ABSTRACT In vertebrate photoreceptors, opsins are highly concentrated in a morphologically distinct ciliary compartment known as the outer segment (OS). Opsin is synthesized in the cell body and transported to the OS...

...Brittany J. Carr; Paloma Stanar; Orson L. Moritz ABSTRACT Mutations in prominin-1 (prom1) and photoreceptor cadherin (cdhr1) are associated with inherited retinal degenerative disorders but their functions remain unknown. Here, we used CRISPR-Cas9 to generate prom1 -null, cdhr1 -null, and prom1...

...Yuna Otsuka; Takunori Satoh; Nozomi Nakayama; Ryota Inaba; Hitomi Yamashita; Akiko K. Satoh ABSTRACT Rab11 is essential for polarized post-Golgi vesicle trafficking to photosensitive membrane rhabdomeres in Drosophila photoreceptors. Here, we found that Parcas (Pcs), recently shown to have guanine...

...Abhishek Chadha; Stefanie Volland; Natella V. Baliaouri; Elaine M. Tran; David S. Williams ABSTRACT The photoreceptor outer segment is the most elaborate primary cilium, containing large amounts of rhodopsin (RHO) in disk membranes that grow from a connecting cilium. The movement of RHO along...

..., the contour of which is denoted by the dashed line, A,B) stained for Arm (A′,B′) and aPKC (A″,B″). White arrows indicate cno R2 mutant photoreceptors that have delaminated from the retinal neuroepithelium. (C–F) cnoIR clones positively labeled by GFP (blue, C,E) and stained for Arm (C′,E′), Baz (C″), Crb...

... melanogaster photoreceptors, photon absorption by the G-protein-coupled receptor (GPCR) rhodopsin 1 (Rh1; also known as NinaE) triggers its endocytosis through clathrin-mediated endocytosis (CME). We find that CME of Rh1 is regulated by phosphatidylinositol 5 phosphate 4-kinase (PIP4K). Flies lacking PIP4K...

... in the regulation of microtubule functions. Glycylation has so far predominantly been found in motile cilia and flagella, and absence of this modification leads to ciliary disassembly. Here, we demonstrate that the correct functioning of connecting cilia of photoreceptors, which are non-motile sensory cilia...

... in the developing Drosophila compound eye photoreceptors (R cells). As part of normal development, R cells take a turn as professional secretory cells with a massive secretory effort that builds the photosensitive membrane organelle, the rhabdomere. We find rough ER sheets proliferate as rhabdomere biogenesis...

...Nidhi Khattree; Linda M. Ritter; Andrew F. X. Goldberg Summary Vertebrate vision requires photon absorption by photoreceptor outer segments (OSs), structurally elaborate membranous organelles derived from non-motile sensory cilia. The structure and function of OSs depends on a precise stacking...

...Natalia Mora; Isabel Almudi; Berta Alsina; Montserrat Corominas; Florenci Serras Summary In a genome-wide expression profile search for genes required for Drosophila R7 photoreceptor development we found β amyloid protein precursor-like (Appl), the ortholog of human APP , which is a key factor...

...Sidney M. Gospe, III; Sheila A. Baker; Vadim Y. Arshavsky Photoreceptors are among the most metabolically active cells in the body, relying on both oxidative phosphorylation and glycolysis to satisfy their high energy needs. Local glycolysis is thought to be particularly crucial in supporting...

...A. Ashleigh Long; Cecon T. Mahapatra; Elvin A. Woodruff, III; Jeff Rohrbough; Hung-Tat Leung; Shikoh Shino; Lingling An; Rebecca W. Doerge; Mark M. Metzstein; William L. Pak; Kendal Broadie A systematic Drosophila forward genetic screen for photoreceptor synaptic transmission mutants identified...

...Youwen Zhang; Laurie L. Molday; Robert S. Molday; Shanta S. Sarfare; Michael L. Woodruff; Gordon L. Fain; Timothy W. Kraft; Steven J. Pittler Ion flow into the rod photoreceptor outer segment (ROS) is regulated by a member of the cyclic-nucleotide-gated cation-channel family; this channel consists...

... offers an ideal model system to study these processes. Localized actin polymerization is required to constrict the apical surface of epithelial cells of the eye imaginal disc to maintain the refined arrangement of the developing ommatidia. The identity of each photoreceptor cell within the epithelium...

... for the pigmentation abnormalities is well understood, the role of myosin Va in neural function is not. Myosin Va has been found in synaptic terminals in the retina and brain. We report here new physiological evidence for a role of myosin Va in synaptic function. Photoreceptor synapses in neurologically affected...

... two (xrds36 and -35) are more distant relatives. By immunocytochemical analysis of retinal sections, xrds38 is distributed in both rod and cone photoreceptors, while xrds36 and xrds35 are present in rods only. At the EM level, xrds38 is present specifically in the rims and incisures of rod and cone...