Social cognition in DMD and BMD dystrophinopathies: a cross-sectional preliminary study

被引:9
作者
Garcia, Irune [1 ]
Martinez, Oscar [1 ]
Lopez-Paz, Juan Francisco [1 ]
Garcia, Maitane [1 ]
Espinosa-Blanco, Patricia [1 ]
Rodriguez, Alicia Aurora [1 ]
Pallares-Sastre, Merce [1 ]
Ruiz de Lazcano, Aitana [1 ]
Amayra, Imanol [1 ]
机构
[1] Univ Deusto, Fac Hlth Sci, Dept Psychol, Neuro E Mot Res Team, Bilbao, Spain
关键词
DMD; BMD; dystrophinopathies; social cognition; general intelligence; DUCHENNE MUSCULAR-DYSTROPHY; NEUROPSYCHOLOGICAL PROFILE; EMOTION RECOGNITION; MANAGEMENT; DIAGNOSIS; CHILDREN; BOYS; MIND; CHILDHOOD; MUTATIONS;
D O I
10.1080/13854046.2023.2202332
中图分类号
B849 [应用心理学];
学科分类号
040203 ;
摘要
Objective: The dystrophinopathies called Duchenne and Becker muscular dystrophies (DMD/BMD) are rare, progressive, incurable, and life-limiting paediatric-onset neuromuscular diseases. These diseases have long been associated with specific neuropsychological deficits. However, the performance of these patients in the social cognition domain has not been properly investigated. Thus, the main objective of this study was to compare the performance on social cognition between DMD/BMD patients and healthy age-matched boys. Method: This cross-sectional study included 20 DMD/BMD children and adolescents and 20 healthy controls. The protocol included the Social Perception Domain of the NEPSY-II, the Reading the Mind in the Eyes Test - Child and Happe's Strange Stories test. General intelligence was controlled to eliminate the possible influence of covariables. All the assessments were performed remotely. Results: Most social cognition tasks were worse in patients with DMD/BMD than in matched healthy controls. These differences remained even after controlling for the general intelligence variable, with the exception of Total Disgust Errors (F = 1.462, p = .234, eta(2)(p)= .038) and Verbal task (F = 1.820, p = .185, eta(2)(p)= .047) scores from the NEPSY-II. Conclusions: This is the first study to demonstrate that the neuropsychological domain of social cognition is impaired in DMD/BMD patients, independent of the level of general intelligence. Screening assessments in DMD/BMD patients should be promoted to allow social cognition difficulties to be detected at an early stage to enhance patients' quality of life and social development.
引用
收藏
页码:219 / 234
页数:16
相关论文
共 76 条
[1]   Advances in imaging of brain abnormalities in neuromuscular disease [J].
Angelini, Corrado ;
Pinzan, Elena .
THERAPEUTIC ADVANCES IN NEUROLOGICAL DISORDERS, 2019, 12
[2]  
[Anonymous], 1991, META ANALYTIC PROCED
[3]   Cognitive and Neurobehavioral Profile in Boys With Duchenne Muscular Dystrophy [J].
Banihani, Rudaina ;
Smile, Sharon ;
Yoon, Grace ;
Dupuis, Annie ;
Mosleh, Maureen ;
Snider, Andrea ;
McAdam, Laura .
JOURNAL OF CHILD NEUROLOGY, 2015, 30 (11) :1472-1482
[4]   The "Reading the Mind in the Eyes" test revised version: A study with normal adults, and adults with Asperger syndrome or high-functioning autism [J].
Baron-Cohen, S ;
Wheelwright, S ;
Hill, J ;
Raste, Y ;
Plumb, I .
JOURNAL OF CHILD PSYCHOLOGY AND PSYCHIATRY, 2001, 42 (02) :241-251
[5]   Longitudinal data of neuropsychological profile in a cohort of Duchenne muscular dystrophy boys without cognitive impairment [J].
Battini, R. ;
Lenzi, S. ;
Lucibello, S. ;
Chieffo, D. ;
Moriconi, F. ;
Cristofani, P. ;
Bulgheroni, S. ;
Cumbo, F. ;
Pane, M. ;
Baranello, G. ;
Alfieri, P. ;
Astrea, G. ;
Cioni, G. ;
Vicari, S. ;
Mercuri, E. .
NEUROMUSCULAR DISORDERS, 2021, 31 (04) :319-327
[6]   Cognitive, Emotional and Psychological Manifestations in Amyotrophic Lateral Sclerosis at Baseline and Overtime: A Review [J].
Benbrika, Soumia ;
Desgranges, Beatrice ;
Eustache, Francis ;
Viader, Fausto .
FRONTIERS IN NEUROSCIENCE, 2019, 13
[7]   Diagnosis and management of Duchenne muscular dystrophy, part 3: primary care, emergency management, psychosocial care, and transitions of care across the lifespan [J].
Birnkrant, David J. ;
Bushby, Katharine ;
Bann, Carla M. ;
Apkon, Susan D. ;
Blackwell, Angela ;
Colvin, Mary K. ;
Cripe, Linda ;
Herron, Adrienne R. ;
Kennedy, Annie ;
Kinnett, Kathi ;
Naprawa, James ;
Noritz, Garey ;
Poysky, James ;
Street, Natalie ;
Trout, Christina J. ;
Weber, David R. ;
Ward, Leanne A. .
LANCET NEUROLOGY, 2018, 17 (05) :445-455
[8]  
Birnkrant DJ, 2018, LANCET NEUROL, V17, P251, DOI 10.1016/S1474-4422(18)30024-3
[9]   Characterization of brain dystrophins absence and impact in dystrophin-deficient Dmdmdx rat model [J].
Caudal, Dorian ;
Francois, Virginie ;
Lafoux, Aude ;
Ledevin, Mireille ;
Anegon, Ignacio ;
Le Guiner, Caroline ;
Larcher, Thibaut ;
Huchet, Corinne .
PLOS ONE, 2020, 15 (03)
[10]   Psychosocial Management of the Patient With Duchenne Muscular Dystrophy [J].
Colvin, Mary K. ;
Poysky, James ;
Kinnett, Kathi ;
Damiani, Mario ;
Gibbons, Melissa ;
Hoskin, Janet ;
Moreland, Scott ;
Trout, Christina J. ;
Weidner, Norbert .
PEDIATRICS, 2018, 142 :S99-S109