Wechsler Scale Intelligence Testing in Males with Dystrophinopathies: A Review and Meta-Analysis.

Autor:	Weerkamp PMM; Expert Centre for Neurological and Developmental Learning Disabilities, Kempenhaeghe, Sterkselseweg 65, 5591 VE Heeze, The Netherlands.; School for Mental Health and Neuroscience, Maastricht University, Universiteitssingel 40, UNS40, 6229 ER Maastricht, The Netherlands., Mol EM; Expert Centre for Neurological and Developmental Learning Disabilities, Kempenhaeghe, Sterkselseweg 65, 5591 VE Heeze, The Netherlands.; Klimmendaal Academy, Klimmendaal Rehabilitation Specialists, Heijenoordseweg 5, 6813 GG Arnhem, The Netherlands., Sweere DJJ; Expert Centre for Neurological and Developmental Learning Disabilities, Kempenhaeghe, Sterkselseweg 65, 5591 VE Heeze, The Netherlands.; School for Mental Health and Neuroscience, Maastricht University, Universiteitssingel 40, UNS40, 6229 ER Maastricht, The Netherlands., Schrans DGM; Mutsaersstichting, Postweg 88, 5915 HB Venlo, The Netherlands., Vermeulen RJ; School for Mental Health and Neuroscience, Maastricht University, Universiteitssingel 40, UNS40, 6229 ER Maastricht, The Netherlands.; Department of Neurology, Maastricht University Medical Centre, 6229 ER Maastricht, The Netherlands., Klinkenberg S; Expert Centre for Neurological and Developmental Learning Disabilities, Kempenhaeghe, Sterkselseweg 65, 5591 VE Heeze, The Netherlands.; School for Mental Health and Neuroscience, Maastricht University, Universiteitssingel 40, UNS40, 6229 ER Maastricht, The Netherlands.; Department of Neurology, Maastricht University Medical Centre, 6229 ER Maastricht, The Netherlands., Hurks PPM; School for Mental Health and Neuroscience, Maastricht University, Universiteitssingel 40, UNS40, 6229 ER Maastricht, The Netherlands.; Department of Neurology, Maastricht University Medical Centre, 6229 ER Maastricht, The Netherlands., Hendriksen JGM; Expert Centre for Neurological and Developmental Learning Disabilities, Kempenhaeghe, Sterkselseweg 65, 5591 VE Heeze, The Netherlands.; School for Mental Health and Neuroscience, Maastricht University, Universiteitssingel 40, UNS40, 6229 ER Maastricht, The Netherlands.
Jazyk:	angličtina
Zdroj:	Brain sciences [Brain Sci] 2022 Nov 14; Vol. 12 (11). Date of Electronic Publication: 2022 Nov 14.
DOI:	10.3390/brainsci12111544
Abstrakt:	Background: Intelligence scores in males with Duchenne Muscular Dystrophy (DMD) and Becker Muscular Dystrophy (BMD) remain a major issue in clinical practice. We performed a literature review and meta-analysis to further delineate the intellectual functioning of dystrophinopathies. Method: Published, peer-reviewed articles assessing intelligence, using Wechsler Scales, of males with DMD or BMD were searched from 1960 to 2022. Meta-analysis with random-effects models was conducted, assessing weighted, mean effect sizes of full-scale IQ (FSIQ) scores relative to normative data (Mean = 100, Standard Deviation = 15). Post hoc we analysed differences between performance and verbal intelligence scores. Results: 43 studies were included, reporting data on 1472 males with dystrophinopathies; with FSIQ scores available for 1234 DMD (k = 32) and 101 BMD (k = 7). DMD males score, on average, one standard deviation below average (FSIQ = 84.76) and significantly lower than BMD (FSIQ = 92.11). Compared to a previous meta-analysis published in 2001, we find, on average, significantly higher FSIQ scores in DMD. Conclusion: Males with Duchenne have, on average, significantly lower FSIQ scores than BMD males and the general population. Clinicians must consider lower intelligence in dystrophinopathies to ensure good clinical practice.
Databáze:	MEDLINE
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