TULP1 is a photoreceptor-specific protein essential for retinal function and photoreceptor survival. 1 The protein is exclusively expressed in photoreceptor cells and localized to the outer plexiform layer and inner segments. 1 TULP1 contains a conserved carboxy-terminal tubby domain that selectively binds membrane phosphoinositides, enabling its role as a bipartite bridge coordinating multiple signaling pathways. 2 Functionally, TULP1 is required for normal photoreceptor synapse development and opsin trafficking to photoreceptor cilia. 3 Loss of TULP1 function impairs ciliary structure through downregulation of tektin2, a ciliary microtubule component, and triggers ferroptosis-mediated photoreceptor degeneration. 3 YTHDF1, an m⁶A reader protein, enhances TULP1 mRNA translation efficiency in the retina, highlighting post-transcriptional regulation of TULP1. 4 Mutations in TULP1 cause early-onset severe retinal dystrophy, including Leber congenital amaurosis 15 and retinitis pigmentosa 14. 5 The c.148delG variant produces progressive maculopathy with foveal ellipsoid zone loss predictive of visual decline, suggesting potential therapeutic intervention windows. 6