Proximity labeling proteomics maps radial glial ciliary proteins across the developing telencephalon
Year: 2026
Type: article
Abstract: Primary cilia in radial glia act as signaling hubs essential for brain development, yet their molecular roles remain poorly understood. Here, using proximity-labeling-mediated in vivo proteomics, we systematically mapped ciliary proteins in the developing telencephalon. Our dataset reveals region-specific ciliary composition across the dorsal and ventral telencephalon. We identified and validated ribosome proteins and translational machinery components in radial glial cilia. Further, we uncovered ciliary roles for neurodevelopmental disorder-associated proteins, including MARCKS, a key regulator of radial glial polarity, and CKAP2L, a protein linked to Filippi syndrome. Functional studies show that MARCKS contributes to ciliogenesis and CKAP2L regulates neurogenesis by modulating Hedgehog signaling. These findings highlight previously unrecognized mechanisms by which primary cilia modulate brain formation. Our in vivo ciliary proteomic dataset provides a unique resource for understanding ciliary functions in brain development and developmental disorders.
Source: Cell Reports
Authors: Xiaoliang Liu, Oscar T. Gutierrez, Sabyasachi Baboo, Eva Cai, Gurleen Kaur, Yazan Al-Issa, Jolene K. Diedrich, John R. Yates, Xuecai Ge
Language: English
Subfield: Genetics
Domain: Life Sciences
Open Access status: gold
Estimated APC paid: $5,620
Awards: MRI: Acquisition of an Upright Laser Scanning Confocal Microscope to Advance Research and Education at the University of California, Merced, G2293390060, G3183594643, CAREER: Mechanistic studies of cilium proteins in cell signaling, Mechanistic study of Hedgehog signaling in the cilium, G6336487826, Regulation of Hedgehog Signaling by new cilium proteins, Regulation of the Hedgehog Pathway by new cilium proteins in Development and Disease, G963911763
OpenAlex ID: W7160512819