ArticlePediatric neurology2021
Reliability of the Telemedicine Application of the Gross Motor Function Measure-88 in Patients With Leukodystrophy.
Article in Pediatric neurology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT06747884 (Trial Readiness and Endpoint Assessment in Pediatric Myotonic Dystrophy Extension), which is not on this map. Cited by 13 papers, 1 of them a synthesis that pooled it.
What it found
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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.
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.
Trial Readiness and Endpoint Assessment in Pediatric Myotonic Dystrophy Extension
Who cites it
13 citing papers in PubMed, 1 synthesis or guideline pooled it, 19 citations in OpenAlex.
- Application of the Gross Motor Function Measure in children with conditions other than cerebral palsy: A systematic review.Developmental medicine and child neurology · 2025Pooled it
- Performance outcomes of the PEDI-CAT for assessing functional ability in the population with leukodystrophy.Developmental medicine and child neurology · 2026Article
- Rigorous genetic diagnosis review in natural history studies.Orphanet journal of rare diseases · 2026Article
- Design of a Pediatric Low Motor Function Item Battery in leukodystrophies.Molecular genetics and metabolism · 2026Article
- Functional ability profiles in beta-propeller protein-associated neurodegeneration (BPAN).Molecular genetics and metabolism · 2025Observational
- Telemedicine for Personalized Nutritional Intervention of Rare Diseases: A Narrative Review on Approaches, Impact, and Future Perspectives.Nutrients · 2025Review
- Remote assessment of physical fitness via videoconferencing: a systematic review.BMC sports science, medicine & rehabilitation · 2025Article
- Development of an Infantile GM2 Clinical Rating Scale: Remote Assessment of Clinically Meaningful Health-Related Function.Journal of child neurology · 2024Article
- Analysis of a model for pediatric physical therapy and clinical education via telehealth.Journal of pediatric rehabilitation medicine · 2024Article
- Exploration of Gross Motor Function in Aicardi-Goutières Syndrome.Journal of child neurology · 2023Article
- Gross Motor Function in Pediatric OnsetJournal of child neurology · 2023Article
- Feasibility of Video Consultation for Preterm Neurodevelopmental Follow-up Care During the COVID-19 Pandemic: Cohort Study.JMIR pediatrics and parenting · 2023Article
- Psychometric outcome measures in beta-propeller protein-associated neurodegeneration (BPAN).Molecular genetics and metabolismArticle
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
14 authors at 1 institution in 2 countries.
Funding
Abstract
backgroundLeukodystrophies are a rare class of disorders characterized by severe neuromotor disability. There is a strong need for research regarding the functional status of people with leukodystrophy which is limited by the need for in-person assessments of mobility. The purpose of this study is to assess the reliability of the Gross Motor Function Measure-88 (GMFM-88) using telemedicine compared with standard in-person assessments in patients with leukodystrophy.
methodsA total of 21 subjects with a diagnosis of leukodystrophy (age range = 1.79-52.82 years) were evaluated by in-person and by telemedicine evaluations with the GMFM-88 by physical therapists. Inter-rater reliability was assessed through evaluation of the same subject by two independent raters within a three-week period (n = 10 encounters), and intrarater reliability was assessed through blinded rescoring of video-recorded assessments after a one-week time interval (n = 6 encounters).
resultsRemote assessments were performed by caregivers in all 21 subjects using resources found in the home with remote guidance. There was agreement between all paired in-person and remote measurements (Lin's concordance correlation ≥0.995). The Bland-Altman analysis indicated that the paired differences were within ±5%. Intrarater and inter-rater reliability demonstrated an intraclass correlation coefficient of >0.90.
conclusionsThese results support that remote application of the GMFM-88 is a feasible and reliable approach to assess individuals with leukodystrophy. Telemedicine application of outcome measures may be of particular value in rare diseases and those with severe neurologic disability that impacts the ability to travel.
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Registered trials
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.