Sakki, H;
Bowman, R;
Sargent, J;
Kukadia, R;
Dale, N;
(2021)
Visual function subtyping in children with early‐onset cerebral visual impairment.
Developmental Medicine and Child Neurology
, 63
(3)
pp. 303-312.
10.1111/dmcn.14710.
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Abstract
Aim: To develop a data‐driven subgrouping method to identify and profile subtypes of early‐onset childhood cerebral visual impairment (CVI). / Method: Sixty‐three children with suspected or diagnosed congenital CVI were recruited (28 males, 35 females, median age=8y, range=5–16y). Cognitive, basic, and higher‐order vision functions were assessed and quality of life, functional vision questionnaire, neurodevelopmental, and ophthalmological data were collected. Cluster analysis and other statistical analyses were undertaken to determine and validate the subgrouping. / Results: Forty‐three participants completing the full test battery were included in cluster analysis, revealing two subgroups. Group A1 (n=15) showed selective visual perception and visuomotor deficits. Group A2 (n=28) showed more severe and broader visual perception and visuomotor deficits, and variable visual acuity. A third, lower‐functioning group, Group B (n=20), was differentiated and showed significant visual acuity reduction compared with Group A (p<0.001, V=0.69). External validation showed significant cooccurring ophthalmological (e.g. strabismus p<0.001, V=0.59) and motor impairment differences (χ2=16.26, p<0.001, V=0.51) between the three groups. All groups had lowered parent‐reported quality of life and everyday functional vision. / Interpretation: Statistical analyses revealed three subgroups with differentiated vision function characteristics on a gradient of severity. The subgrouping method provides the first steps in developing a novel classification system to underpin future clinical diagnostics and profiling of early‐onset CVI. / What this paper adds: Three data‐driven subgroups of vision function deficits were identified. A similar severity gradient was shown in cooccurring cognitive and neurodevelopmental deficits. Reported quality of life and functional vision difficulties were low across all groups.
| Type: | Article |
|---|---|
| Title: | Visual function subtyping in children with early‐onset cerebral visual impairment |
| Open access status: | An open access version is available from UCL Discovery |
| DOI: | 10.1111/dmcn.14710 |
| Publisher version: | https://doi.org/10.1111/dmcn.14710 |
| Language: | English |
| Additional information: | Copyright © 2020 The Authors. Developmental Medicine & Child Neurology published by John Wiley & Sons Ltd on behalf of Mac Keith Press. This is an open access article under the terms of the Creative Commons Attribution‐NonCommercial‐NoDerivs License (http://creativecommons.org/licenses/by-nc-nd/4.0/), which permits use and distribution in any medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made. |
| UCL classification: | UCL UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health |
| URI: | https://discovery-pp.ucl.ac.uk/id/eprint/10124251 |
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