Evidence map›Paper›PMID 41091173›Full record

Observational studyGraefe's archive for clinical and experimental ophthalmology = Albrecht von Graefes Archiv fur klinische und experimentelle Ophthalmologie2025

Characterising microstructural retinal changes in children with inherited retinal dystrophies - a retrospective observational cross-sectional study.

Jonathon Holland, Brent Rodda, Jonathan B Ruddle, Marianne Ef Piano, Elisse J Higginbotham, Lauren N Ayton, Jasleen K Jolly

Abstract readObservational Study
In one paragraph

Observational study in Graefe's archive for clinical and experimental ophthalmology = Albrecht von Graefes Archiv fur klinische und experimentelle Ophthalmologie, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
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1 · What the graph read from it

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.

2 · The registry

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.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors.

Jonathon HollandDepartment of Clinical Neurosciences, University of Cambridge, Bay 13, Clifford Albutt Building, Cambridge Biomedical Campus, Cambridge, CB2 0AH, UK.
Brent RoddaDepartment of Optometry and Vision Sciences, The University of Melbourne, Melbourne, Australia.
Jonathan B RuddleDepartment of Ophthalmology, Royal Children's Hospital, Melbourne, VIC, Australia.
Marianne Ef PianoDepartment of Optometry and Vision Sciences, The University of Melbourne, Melbourne, Australia.
Elisse J HigginbothamNational Vision Research Institute, Australian College of Optometry, Carlton, Australia.
Lauren N Ayton *Department of Optometry and Vision Sciences, The University of Melbourne, Melbourne, Australia.
Jasleen K Jolly *Department of Optometry and Vision Sciences, The University of Melbourne, Melbourne, Australia. jasleen.jolly@unimelb.edu.au.ORCID http://orcid.org/0000-0001-9878-4621

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

purposeTrials exploring novel treatments for inherited retinal diseases (IRDs) have utilised microstructural retinal changes for outcome measures, but such changes in children are incompletely described. We aimed to characterise the retinal microstructural in children with IRDs versus controls using optical coherence tomography (OCT).

methodsCross-sectional study of retrospective OCT data from 51 children (91 eyes) with IRDs attending the Melbourne Children's Eye Clinic and 64 controls (126 eyes) recruited prospectively at Australian College of Optometry clinics. OCT was performed using a Nidek RS-3000 with volume scans in each participant eye. Image quality was manually reviewed. Semi-automated layer segmentation was performed and thickness based on eccentricity relative to the central fovea position was calculated to standardise measurements between participants.

resultsInner retinal layer thickness was significantly increased in IRD groups versus controls (rod-cone p < 0.0001; cone-rod p < 0.0001; macula p < 0.0001; cone p < 0.0001). This was not observed for photoreceptor complex thickness where instead thinning versus controls was seen in cone-rod disorders (rod-cone p = 0.89; cone-rod p < 0.0001; macula p = 0.21; cone p = 0.17). Total thickness was significantly reduced in cone and cone-rod group versus controls, but not in other IRD types (rod-cone p = 0.25; cone-rod p < 0.0001; macula p = 0.74; cones p = 0.004).

conclusionOur findings suggest retinal remodelling takes place in childhood-onset IRDs. Clinical trials in IRDs should consider remodelling when evaluating treatment effects. Future studies on the longitudinal natural history of retinal microstructural changes in childhood IRDs are required to address the impact of factors including IRD type, sex, age and disease duration.

Indexed as

Retinal DystrophiesTomography, Optical CoherenceVisual AcuityAdolescentChildChild, PreschoolCross-Sectional StudiesFemaleFollow-Up StudiesHumansMaleRetrospective StudiesChildEye diseases, hereditaryHumansOptical coherence tomographyRetinaRetinal dystrophies

Identifiers

PMID41091173
PMCPMC12886368

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Registered trials

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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.