ArticleOphthalmology2014
Macular function and morphologic features in juvenile stargardt disease: longitudinal study.
Article in Ophthalmology, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 31 papers.
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Who cites it
31 citing papers in PubMed, 62 citations in OpenAlex.
- Longitudinal Changes of Fundus Autofluorescence and Correlation With Visual Acuity in ABCA4-Associated Stargardt Disease.Investigative ophthalmology & visual science · 2026Article
- Perspectives on the Current and Future State of Artificial Intelligence in Medical Genetics.American journal of medical genetics. Part A · 2025Review
- The Natural History of Retinal Sensitivity Loss in Diabetic Macular Ischemia over One Year Evaluated by Microperimetry.Journal of clinical medicine · 2024Article
- Article
- Development of oculomotor control throughout childhood: A multicenter and multiethnic study.Journal of vision · 2022Article
- Clinical Observation and Genotype-Phenotype Analysis of ABCA4- Related Hereditary Retinal Degeneration before Gene Therapy.Current gene therapy · 2022Article
- An Overview of the Genetics ofGenes · 2021Review
- Fundus-controlled perimetry (microperimetry): Application as outcome measure in clinical trials.Progress in retinal and eye research · 2021Review
- Correlation of Morphology and Function of Flecks Using Short-Wave Fundus Autofluorescence and Microperimetry in Patients With Stargardt Disease.Translational vision science & technology · 2021Article
- Inferred retinal sensitivity in recessive Stargardt disease using machine learning.Scientific reports · 2021Article
- Clinical spectrum, genetic complexity and therapeutic approaches for retinal disease caused by ABCA4 mutations.Progress in retinal and eye research · 2020Review
- Longitudinal Microperimetric Changes of Macular Sensitivity in Stargardt Disease After 12 Months: ProgStar Report No. 13.JAMA ophthalmology · 2020Article
- Prospective Cohort Study of Childhood-Onset Stargardt Disease: Fundus Autofluorescence Imaging, Progression, Comparison with Adult-Onset Disease, and Disease Symmetry.American journal of ophthalmology · 2020Article
- Performance of a Defect-Mapping Microperimetry Approach for Characterizing Progressive Changes in Deep Scotomas.Translational vision science & technology · 2019Article
- Demonstration of anatomical development of the human macula within the first 5 years of life using handheld OCT.International ophthalmology · 2019Observational
- Inverted internal limiting membrane-flap technique for large macular hole: a microperimetric study.International journal of retina and vitreous · 2019Article
- Macular hyperpigmentary changes inInternational journal of retina and vitreous · 2019Article
- Cross-Sectional and Longitudinal Assessment of Retinal Sensitivity in Patients With Childhood-Onset Stargardt Disease.Translational vision science & technology · 2018Article
- Longitudinal Changes of Fixation Location and Stability Within 12 Months in Stargardt Disease: ProgStar Report No. 12.American journal of ophthalmology · 2018Article
- Peripheral Visual Fields in ABCA4 Stargardt Disease and Correlation With Disease Extent on Ultra-widefield Fundus Autofluorescence.American journal of ophthalmology · 2017Article
Corrections and comments
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Authors and funding
7 authors at 2 institutions in 1 country.
Funding
Abstract
purposeTo evaluate disease progression in a cohort of patients with a clinical and genetic diagnosis of Stargardt disease.
designLongitudinal cohort study.
participantsA total of 56 selected patients with a clinical and molecular diagnosis of Stargardt disease, an early age of onset, and a median follow-up length of 2 years.
methodsPatients underwent routine examination, including full-field electroretinography, microperimetry, and optical coherence tomography.
main outcome measuresBest-corrected visual acuity (BCVA), mean retinal sensitivity, fixation stability, preferred retinal locus, inner segment/outer segment (IS/OS) junction loss, and atrophic lesion area.
resultsA total of 56 patients with a mean age at disease onset of 15.3 years (range, 3-28 years), a mean disease duration of 12.1 years, and a mean age at baseline of 27.4 years were analyzed. The median BCVA was 20/200 in both eyes. Optical coherence tomography parameters (IS/OS alteration and retinal pigment epithelium lesion area) were obtained in only 49 patients because the signal quality was poor in the remaining 7 patients. Optical coherence tomography revealed a mean retinal pigment epithelium lesion area of 2.6 mm(2), preserved foveal IS/OS in 4.1% of patients, loss of foveal IS/OS in 59.2% of patients, and extensive loss of macular IS/OS in 36.7% of patients. Microperimetric findings showed a reduced macular sensitivity (mean, 10 decibels [dB]) and an unstable fixation in half of the patient cohort. The longitudinal analysis showed a significant progressive reduction of BCVA and macular sensitivity (at an estimated rate of 0.04 decimals and 1.19 dB/year, respectively) associated with a significant enlargement of retinal pigment epithelium lesion area (0.282 mm(2)/year). No significant changes in ophthalmoscopic findings and electroretinographic responses were detected.
conclusionsThis study highlights the importance of microperimetry and optical coherence tomography in monitoring patients with Stargardt disease. Quantifying the decline of visual functionality and detecting morphologic macular changes prove useful in evaluating disease progression over a short-term follow-up and should be taken into account for the design of future clinical trials of gene therapy to treat retinal dystrophy.
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