Retinal detachment

Lin, Jonathan B.; Narayanan, Raja; Philippakis, Elise; Yonekawa, Yoshihiro; Apte, Rajendra S.

doi:10.1038/s41572-024-00501-5

Cited by 7 publications

(1 citation statement)

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“…The vertebrate retina is a layered structure with a wide variety of cells that form distinct circuits working parallel to produce a complex visual output. [ 1 ] The structure of the retina is conserved across vertebrates and includes three major layers: the outmost retinal pigment epithelial (RPE) layer, the photoreceptor‐containing outer nuclear layer (ONL), and the inner nuclear layer (INL) which contains Müller cells and bipolar cells. This intricate arrangement is highly prone to damage, leading to disruptions in retinal homeostasis and contributing to an array of retinal diseases, such as diabetic retinopathy, retinitis pigmentosa, and age‐related macular degeneration.…”

Section: Introductionmentioning

confidence: 99%

KIF11 UFMylation Maintains Photoreceptor Cilium Integrity and Retinal Homeostasis

Ran,

Guo,

Zhang

et al. 2024

Advanced Science

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The photoreceptor cilium is vital for maintaining the structure and function of the retina. However, the molecular mechanisms underlying the photoreceptor cilium integrity and retinal homeostasis are largely unknown. Herein, it is shown that kinesin family member 11 (KIF11) localizes at the transition zone (connecting cilium) of the photoreceptor and plays a crucial role in orchestrating the cilium integrity. KIF11 depletion causes malformations of both the photoreceptor ciliary axoneme and membranous discs, resulting in photoreceptor degeneration and the accumulation of drusen‐like deposits throughout the retina. Mechanistic studies show that the stability of KIF11 is regulated by an interplay between its UFMylation and ubiquitination; UFMylation of KIF11 at lysine 953 inhibits its ubiquitination by synoviolin 1 and thereby prevents its proteasomal degradation. The lysine 953‐to‐arginine mutant of KIF11 is more stable than wild‐type KIF11 and also more effective in reversing the ciliary and retinal defects induced by KIF11 depletion. These findings identify a critical role for KIF11 UFMylation in the maintenance of photoreceptor cilium integrity and retinal homeostasis.

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Section: Introductionmentioning

confidence: 99%