Optical coherence tomography in children with inherited retinal disease.

Thomas L Edwards, Jasleen K Jolly, Lauren N Ayton, Brent M Rodda, Jonathan B Ruddle

Journal: Clinical & experimental optometry 2024;107(3):255-266

PMID: 38252959

Abstract

Recent advances have led to therapeutic options becoming available for people with inherited retinal disease. In particular, gene therapy has been shown to hold great promise for slowing vision loss from inherited retinal disease. Recent studies suggest that gene therapy is likely to be most effective when implemented early in the disease process, making consideration of paediatric populations important. It is therefore necessary to have a comprehensive understanding of retinal imaging in children with inherited retinal diseases, in order to monitor disease progression and to determine which early retinal biomarkers may be used as outcome measures in future clinical trials. In addition, as many optometrists will review children with an inherited retinal disease, an understanding of the expected imaging outcomes can improve clinical care. This review focuses on the most common imaging modality used in research assessment of paediatric inherited retinal diseases: optical coherence tomography. Optical coherence tomography findings can be used in both the clinical and research setting. In particular, the review discusses current knowledge of optical coherence tomography findings in eight paediatric inherited retinal diseases - Stargardt disease, Bests disease, Leber's congenital amaurosis, choroideremia, RPGR related retinitis pigmentosa, Usher syndrome, X-linked retinoschisis and, Batten disease.

Address: Vision and Eye Research Institute, Anglia Ruskin University, Cambridge, UK.; Nuffield Laboratory of Ophthalmology, Nuffield Department of Clinical Neurosciences, University of Oxford, Oxford, UK.; Department of Optometry and Vision Sciences, The University of Melbourne, Carlton, Victoria, Australia.; Ophthalmology, Department of Surgery, The University of Melbourne, East Melbourne, Victoria, Australia.; Centre for Eye Research Australia, The Royal Victorian Eye and Ear Hospital, East Melbourne, Victoria, Australia.; Department of Optometry and Vision Sciences, The University of Melbourne, Carlton, Victoria, Australia.; Ophthalmology, Department of Surgery, The University of Melbourne, East Melbourne, Victoria, Australia.; Centre for Eye Research Australia, The Royal Victorian Eye and Ear Hospital, East Melbourne, Victoria, Australia.; Centre for Eye Research Australia, The Royal Victorian Eye and Ear Hospital, East Melbourne, Victoria, Australia.; Department of Ophthalmology, Royal Children's Hospital, Parkville, Victoria, Australia.

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