ArticleInvestigative ophthalmology & visual science2026
Rod-Cone Dystrophy Related WDR34 Is Essential for Ciliary Integrity and Survival of Mammalian Photoreceptor Cells.
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. Cited by 2 papers.
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
2 citing papers in PubMed.
- WDR34 Deficiency Disrupts Retrograde Intraflagellar Transport and Induces Unfolded Protein Response-Driven Inflammation and Retinal Degeneration.Investigative ophthalmology & visual science · 2026Article
- The roles of cytoplasmic dynein complex in various ocular disorders.Molecular medicine (Cambridge, Mass.) · 2026Review
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
9 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Purpose: The photoreceptor outer segment is a highly specialized ciliary structure essential for phototransduction, rendering photoreceptors especially vulnerable to ciliary dysfunction. WDR34, a key component of the retrograde intraflagellar transport machinery, has been implicated in rod-cone dystrophy. However, the pathogenic mechanisms linking WDR34 deficiency to photoreceptor degeneration remain elusive. In this study we aim to investigate the in vivo function of Wdr34 in the photoreceptor cells using conditional knockout allele of Wdr34. Methods: We generated retina-specific Wdr34 knockout mice using rod-specific and cone-specific drivers to investigate the in vivo roles of WDR34 in photoreceptor maintenance. Results: Wdr34 deficiency in rod photoreceptors resulted in progressive rod cell degeneration accompanied by a marked decline in scotopic electroretinography (ERG) responses. Similarly, cone-specific Wdr34 ablation led to impaired photopic ERG responses and subsequent cone photoreceptor death. Transcriptomic profiling of Wdr34-deficient retinas revealed broad differential gene expression changes, with significant enrichment in axonemal integrity and microtubule-based transport. Notably, subretinal delivery of an adeno-associated virus vector expressing WDR34 significantly preserved rod photoreceptor structure and function, underscoring the therapeutic potential of WDR34 gene supplementation. Conclusions: Our findings establish WDR34 as a critical factor for photoreceptor survival and function, emphasizing its role in the pathogenesis of WDR34-associated retinal degeneration. Moreover, this study demonstrates that WDR34-targeted gene therapy can effectively delay photoreceptor loss, highlighting a promising treatment strategy for patients with WDR34-related retinal disease.
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.