ArticleInvestigative ophthalmology & visual science2026
WDR34 Deficiency Disrupts Retrograde Intraflagellar Transport and Induces Unfolded Protein Response-Driven Inflammation and Retinal Degeneration.
Article in Investigative ophthalmology & visual science, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.
What it found
Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.
The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
0 citing papers in PubMed.
No citing paper in PubMed yet.
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
6 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Purpose: Retinitis pigmentosa (RP) is a hereditary retinal disease characterized by progressive photoreceptor cell (PRC) degeneration. WD repeat domain 34 (WDR34), an intermediate chain of dynein-2, is essential for retrograde intraflagellar transport (IFT). However, the mechanisms by which WDR34 deficiency causes retinal degeneration remain unclear. This study aims to investigate the impact of WDR34 deficiency on retrograde IFT and its contribution to retinal degeneration. Methods: WDR34 deficiency was modeled in vivo via subretinal injection of adeno-associated virus-shRNA-WDR34 and in vitro by CRISPR/Cas9-mediated knockout in 661W cells. Retinal degeneration and IFT defects were assessed by histologic, functional, and ultrastructural analyses. Proteomic analysis followed by in vivo validation was used to investigate the molecular mechanism underlying WDR34-deficient retinal degeneration. Results: WDR34 knockdown induced progressive retinal degeneration characterized by PRC apoptosis, gradual outer nuclear layer thinning, reduced electroretinography responses, and outer segment shortening. WDR34 deficiency impaired retrograde IFT and caused rhodopsin and opsin mislocalization. These alterations induced endoplasmic reticulum stress and unfolded protein response (UPR) activation, activating the IRE1α/TRAF2/NF-κB signaling pathway, ultimately contributing to retinal inflammation and degeneration. Conclusions: WDR34 is crucial for maintaining retrograde IFT in PRCs. WDR34 deficiency disrupts outer segment maintenance and triggers UPR-mediated inflammatory responses and apoptosis, ultimately leading to retinal degeneration. This study reveals a novel mechanistic link among WDR34, retrograde IFT, ciliopathies, and retinal degeneration, providing potential therapeutic insights for ciliopathy-associated RP.
Indexed as
Identifiers
What OpenQuestion holds
Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.