Observational studyActa diabetologica2017
Retinal thickness as a marker of disease progression in longitudinal observation of patients with Wolfram syndrome.
Observational study in Acta diabetologica, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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The trial behind it
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Who cites it
10 citing papers in PubMed, 25 citations in OpenAlex.
- Wolfram Syndrome Type I Case Report and Review-Focus on Early Diagnosis and Genetic Variants.Medicina (Kaunas, Lithuania) · 2024Review
- Wolfram Syndrome 1: A Pediatrician's and Pediatric Endocrinologist's Perspective.International journal of molecular sciences · 2023Review
- Longitudinal Changes in Vision and Retinal Morphology in Wolfram Syndrome.American journal of ophthalmology · 2022Article
- Longitudinal progression of diabetes mellitus in Wolfram syndrome: The Washington University Wolfram Research Clinic experience.Pediatric diabetes · 2022Observational
- Serum microRNA as indicators of Wolfram syndrome's progression in neuroimaging studies.BMJ open diabetes research & care · 2020Article
- Multiple Retinal Anomalies in Wfs1-Deficient Mice.Diagnostics (Basel, Switzerland) · 2020Article
- Novel mutations and the ophthalmologic characters in Chinese patients with Wolfram Syndrome.Orphanet journal of rare diseases · 2019Article
- [Structural endpoints for glaucoma studies].Der Ophthalmologe : Zeitschrift der Deutschen Ophthalmologischen Gesellschaft · 2019Review
- Measurement of corneal thickness, optic nerve sheath diameter and retinal nerve fiber layer as potential new non-invasive methods in assessing a risk of cerebral edema in type 1 diabetes in children.Acta diabetologica · 2018Article
- Visual pathway function and structure in Wolfram syndrome: patient age, variation and progression.BMJ open ophthalmology · 2018Article
Corrections and comments
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Authors and funding
7 authors at 2 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
aimsWolfram syndrome (WFS) is a recessively inherited monogenic form of diabetes coexisting with optic atrophy and neurodegenerative disorders with no currently recognized markers of disease progression. The aim of the study was to evaluate retinal parameters by using optical coherence tomography (OCT) in WFS patients after 2 years of follow-up and analysis of the parameters in relation to visual acuity.
methodsOCT parameters and visual acuity were measured in 12 WFS patients and 31 individuals with type 1 diabetes.
resultsTotal thickness of the retinal nerve fiber layer (RNFL), average retinal thickness and total retinal volume decreased in comparison with previous OCT examination. Significant decreases were noted for RNFL (average difference -17.92 µm 95% CI -30.74 to -0.10; p = 0.0157), macular average thickness (average difference -5.38 µm 95% CI -10.63 to -2.36; p = 0.0067) and total retinal volume (average difference -0.15 mm
conclusionsOCT measures structural parameters and can precede visual acuity loss. The OCT study in WFS patients should be performed longitudinally, and serial retinal examinations may be helpful as a potential end point for future clinical trials.
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