What is it about?
The retina is a highly specialized sensorineural tissue composed of diverse cell types, each with distinct functional roles in vision. Among these, the rod and cone photoreceptors are unique in their possession of large specialized sensory cilia, which are essential for phototransduction, while non-photoreceptor neurons typically exhibit simpler primary cilia. This study identifies Foxn3 as a key transcriptional regulator in establishing and maintaining this dichotomy. Retina-specific Foxn3 ablation in mice leads to ectopic expression of ciliary genes and abnormal ciliogenesis in non-photoreceptor cells, resulting in visual impairments. And Foxn3 directly represses the promoters of ciliary genes and their transactivator genes.
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Why is it important?
Our findings highlight Foxn3 as a key transcriptional repressor that may function to ensure the proper ciliary architecture of retinal neurons by preventing non-photoreceptor neurons from adopting photoreceptor-like ciliary features, provide new insights into the molecular mechanisms of retinal development and ciliopathies such as retinitis pigmentosa, cone‑rod dystrophy and Leber congenital amaurosis, and offer potential therapeutic targets for retinal ciliopathies.
Perspectives
This article represents years of research by graduate students, postdocs and technicians, who, like me, were initially bewildered by the experimental results from mutant mice. It is really rewarding to see our findings published in a prestigious journal. I am especially grateful to my team for their perseverance and agility in adopting emerging technologies to tackle a challenging problem. I hope this work provides a refreshing perspective that will encourage both researchers and clinicians to broaden their consideration of relevant cell types, pathogenesis and therapeutic targets involved in retinal ciliopathies.
Mengqing Xiang
Sun Yat-Sen University
Read the Original
This page is a summary of: Foxn3 is required to suppress aberrant ciliogenesis in nonphotoreceptor retinal neurons, Proceedings of the National Academy of Sciences, July 2025, Proceedings of the National Academy of Sciences,
DOI: 10.1073/pnas.2500871122.
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