ArticleAmerican journal of ophthalmology2026
Central Retinal Sensitivity Decline in RPGR-Related Retinal Phenotypes.
Article in American journal of ophthalmology, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.
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Who cites it
2 citing papers in PubMed.
- Inflammatory Markers and Disease Progression in Moderate-Stage Retinitis Pigmentosa: Longitudinal Analysis from the Retinitis Pigmentosa Progression and Inflammatory Marker Registry Study.Ophthalmology science · 2026Article
- Functional Outcomes and Ocular Safety of Subretinal AAV-RPGR Gene Therapy for RPGR-Associated X-Linked Retinitis Pigmentosa: A Systematic Review and Meta-Analysis.Journal of clinical medicine · 2026Review
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Authors and funding
5 authors.
Funding
No grant is acknowledged in the PubMed record.
Abstract
purposeTo characterize the natural history of central retinal sensitivity decline in RPGR-associated retinopathy and to evaluate and quantify phenotypic differences in disease progression using microperimetry-derived metrics.
designA retrospective cohort study. SUBJECTS: 50 patients with genetically confirmed RPGR mutations (36 rod-cone, 14 cone-rod dystrophy [CORD], contributing 357 microperimetry assessments) were recruited.
methodsCentral retinal function was assessed using the standard 68-point MAIA microperimeter grid pattern after 20 minutes of dark adaptation and without dilation. Mean sensitivity across the central 4 (MS4) and 16 (MS16) loci were extracted and analyzed post hoc. Longitudinal decline rates were modeled using generalized linear mixed models, and survival analyses were conducted using Cox proportional hazards models with random intercepts and robust clustering to account for repeated measures and intra-subject correlation.
main outcome measuresThe rate of retinal sensitivity decline was measured.
resultsCORD patients exhibited significantly faster rates of central retinal sensitivity decline. The annual MS16 decline rate was 10.8% (95% CI: 6.6, 14.8) in CORD vs 5.1% (95% CI: 2.9, 7.1) in rod-cone patients (P = .02). MS4 declined by 14.9% (95% CI: 10.2, 19.4) annually in CORD compared to 4.1% (95% CI: 1.2, 6.8) in rod-cone (P < .001). The median survival age to total loss of sensitivity for MS16 was 25.1 years in CORD and 33.1 years in rod-cone (P = .01), and for MS4, 27.2 years and 33.3 years, respectively (P = .02). Hazard ratios demonstrated an 8.2-fold (MS16) and 7.2-fold (MS4) increased instantaneous risk of central vision loss in CORD compared with rod-cone patients.
conclusionsThis study reveals significantly different rates of functional decline between RPGR phenotypes, with CORD patients at markedly higher risk of early central vision loss. These findings support the use of microperimetry as a sensitive outcome measure and provide critical survival data for clinical trial design, patient counseling, and therapeutic prioritization. Patients with cone-dominated phenotypes may benefit from earlier intervention.
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