Year: 2023
Type: article
Abstract: The primary cilium is a signaling organelle with a unique membrane composition maintained by a diffusional barrier residing at the transition zone. Many transition zone proteins, such as the tectonic complex, are linked to preserving ciliary composition but the mechanism remains unknown. To understand tectonic's role, we generate a photoreceptor-specific Tctn1 knockout mouse. Loss of Tctn1 results in the absence of the entire tectonic complex and associated MKS proteins yet has minimal effects on the transition zone structure of rod photoreceptors. We find that the protein composition of the photoreceptor cilium is disrupted as non-resident membrane proteins accumulate in the cilium over time, ultimately resulting in photoreceptor degeneration. We further show that fluorescent rhodopsin moves faster through the transition zone in photoreceptors lacking tectonic, which suggests that the tectonic complex acts as a physical barrier to slow down membrane protein diffusion in the photoreceptor transition zone to ensure proper removal of non-resident membrane proteins.
Source: Nature Communications
Authors: Hanh M. Truong, Kevin O. Cruz-Colón, Jorge Y. Martínez-Márquez, Jason R. Willer, Amanda M. Travis, Sondip K. Biswas, Woo-Kuen Lo, Hanno J. Bolz, Jillian N. Pearring
Language: English
Subfield: Genetics
Domain: Life Sciences
Open Access status: gold
Estimated APC paid: $6,290
Funders: U.S. Department of Health and Human Services, Research to Prevent Blindness, State University of New York, National Institutes of Health, State University of New York Upstate Medical University, National Eye Institute, National Cancer Institute, National Institute of General Medical Sciences, National Institute of Diabetes and Digestive and Kidney Diseases
Awards: University of Michigan Center for Gastrointestinal Research, G2789405364, G2976444577, Cell Junctions and Cell Membranes in the Lens, G3463356597, G3628099039, University of Michigan O'Brien Kidney Translational Core Center, THE STUDY OF CELL JUNCTIONS IN THE OCULAR LENS, G4135767613, Cellular and Molecular Biology at Michigan, G538843489, Leica SP8 Confocal Microscope, G6383387132, E75 IN THE DEVELOPING DROSOPHILA EYE, G7404080673, G9000066216, G9000138755, G9000207620, G9000264677, G9000308391, University of Michigan Vision Research Center, University of Michigan Rogel Cancer Center Support Grant 2023-2028, Understanding Photoreceptor Trafficking Pathways to the Outer Segment
OpenAlex ID: W37704658