Abstract
Background: Dominant Optic Atrophy (DOA) causes slowly progressive visual decline usually beginning in childhood. As new therapies come to clinical trial, the choice of biomarkers to be used as clinical trial endpoints has become a critical question to be addressed. Methods: We undertook a systematic review and meta-analysis of studies reporting longitudinal data of any biomarker in DOA patients. Results: In total, seven studies were included in the systematic review, and four presented paired results compatible with meta-analysis. Visual acuity was the only biomarker found with reported longitudinal data. Of the included studies in the meta-analysis, the rate of yearly visual acuity decline (0.022 LogMAR/year., 95% CI: −0.008 to 0.052) was not significantly different from zero (Z = 1.4, p = 0.155). Conclusion: Quantifying this slow rate of visual decline has implications for future study design and suggests that further natural history studies examining alternative biomarkers in DOA are warranted.
| Original language | English |
|---|---|
| Pages (from-to) | 652-659 |
| Number of pages | 8 |
| Journal | Clinical and Experimental Ophthalmology |
| Volume | 53 |
| Issue number | 6 |
| Early online date | 7 May 2025 |
| DOIs | |
| Publication status | Published - Aug 2025 |
| Externally published | Yes |
Bibliographical note
Copyright the Author(s) 2025. Version archived for private and non-commercial use with the permission of the author/s and according to publisher conditions. For further rights please contact the publisher.Keywords
- autosomal dominant optic atrophy
- biomarkers
- review
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