Neuropsychological factors associated with performance on the rey-osterrieth complex figure test in children with neurofibromatosis type 1

被引:2
作者
Maier, Alice [1 ]
Pride, Natalie A. A. [2 ]
Hearps, Stephen J. C. [1 ,3 ]
Shah, Nijashree [4 ]
Porter, Melanie [4 ]
North, Kathryn N. N. [1 ,5 ]
Payne, Jonathan M. M. [1 ,5 ,6 ]
机构
[1] Royal Childrens Hosp, Murdoch Childrens Res Inst, Melbourne, Australia
[2] Childrens Hosp Westmead, Kids Neurosci Ctr, Sydney, Australia
[3] Univ Melbourne, Fac Med Dent & Hlth Sci, Dept Crit Care, Melbourne, Australia
[4] Macquarie Univ, Sch Psychol, Sydney, Australia
[5] Univ Melbourne, Fac Med Dent & Hlth Sci, Dept Paediat, Melbourne, Australia
[6] Royal Childrens Hosp, Murdoch Childrens Res Inst, Flemington Rd, Parkville, Vic 3052, Australia
关键词
Neurofibromatosis type 1; rey-osterrieth complex figure test; visuospatial skills; executive functioning; genetic conditions; EXECUTIVE FUNCTION; CLINICAL-TRIALS; SCORING SYSTEM; ADOLESCENTS; ATTENTION; DIFFICULTIES; PRESCHOOL; DEFICITS;
D O I
10.1080/09297049.2023.2199975
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Children with neurofibromatosis type 1 (NF1) are at considerable risk for cognitive difficulties, including visuospatial deficits and executive dysfunction. This study aimed to (1) assess the overall performance of children with NF1 on the Rey-Osterrieth Complex Figure Test (RCFT) compared to unaffected siblings and (2) examine neuropsychological predictors of RCFT performance in children with NF1. A retrospective clinical audit was performed on neuropsychological records from a multidisciplinary NF1 Clinic in Australia. We searched for children that had completed an assessment between 2000 and 2015 which included the RCFT and other neuropsychological outcomes in this study. These included the Wechsler Intelligence Scale for Children, Judgment of Line Orientation (JLO), Tower of London test, Conners ADHD Scales, and the Behavioral Rating Inventory of Executive Function (BRIEF). The study population consisted of 191 children with NF1 aged 6-16 years, and 55 unaffected siblings recruited from a separate study. Results revealed that 62% of children with NF1 performed at or below the first percentile on the RCFT copy, which was significantly worse than their unaffected siblings. Visuospatial skills, parent-rated executive abilities, ADHD symptoms, and intellectual skills all predicted poorer performance on the RCFT copy, however the best fitting multiple regression model only contained the JLO, BRIEF Metacognition Index, and chronological age. The JLO emerged as the strongest predictor of RCFT performance. This study provides evidence that visuospatial deficits are a key driver of reduced RCFT performance in NF1 and that executive skills as well as a younger age are also independent predictors of RCFT performance.
引用
收藏
页码:348 / 359
页数:12
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