Cognition, ADHD Symptoms, and Functional Impairment in Children and Adolescents With Neurofibromatosis Type 1

被引：39

作者：

Payne, Jonathan M. ^{[1
,2
]}

Haebich, Kristina M. ^{[1
]}

MacKenzie, Rachel ^{[5
]}

Walsh, Karin S. ^{[6
]}

Hearps, Stephen J. C. ^{[1
]}

Coghill, David ^{[1
,3
]}

Barton, Belinda ^{[11
,13
]}

Pride, Natalie A. ^{[12
,14
]}

Ullrich, Nicole J. ^{[15
]}

Tonsgard, James H. ^{[16
]}

Viskochil, David ^{[17
]}

Schorry, Elizabeth K. ^{[18
]}

Klesse, Laura ^{[19
]}

Fisher, Michael J. ^{[20
,21
,22
]}

Gutmann, David H. ^{[23
,24
]}

Rosser, Tena ^{[25
]}

Packer, Roger J. ^{[7
,8
,9
,10
]}

Korf, Bruce ^{[26
,27
,28
]}

Acosta, Maria T. ^{[9
]}

Bellgrove, Mark A. ^{[29
,30
]}

North, Kathryn N. ^{[1
,4
]}

机构：

[1] Royal Childrens Hosp, Murdoch Childrens Res Inst, Flemington Rd, Parkville, Vic 3052, Australia

[2] Univ Melbourne, Dept Pediat, Parkville, Vic, Australia

[3] Univ Melbourne, Dept Pediat, Dev Mental Hlth, Parkville, Vic, Australia

[4] Univ Melbourne, Child Hlth Res, Parkville, Vic, Australia

[5] La Trobe Univ, Bundoora, Vic, Australia

[6] Childrens Natl Hlth Syst, Div Pediat Neuropsychol, Washington, DC USA

[7] Childrens Natl Hlth Syst, Ctr Neurosci & Behav Med, Washington, DC USA

[8] Childrens Natl Hlth Syst, Neurofibromatosis, Washington, DC USA

[9] Childrens Natl Hlth Syst, Gilbert Neurofibromatosis Inst, Washington, DC USA

[10] Childrens Natl Hlth Syst, Brain Tumor Inst, Washington, DC USA

[11] Childrens Hosp Westmead, Childrens Hosp Educ Res Inst, Westmead, NSW, Australia

[12] Childrens Hosp Westmead, Clin Genet & Kids Neurosci Res Ctr, Neurocognit Team, Neuropsychol, Westmead, NSW, Australia

[13] Univ Sydney, Fac Med, Discipline Pediat & Child Hlth, Sydney, NSW, Australia

[14] Univ Sydney, Sydney Med Sch, Sydney, NSW, Australia

[15] Boston Childrens Hosp, Boston, MA USA

[16] Univ Chicago Med, Comer Childrens Hosp, Chicago, IL USA

[17] Univ Utah, NF Clin, Salt Lake City, UT USA

[18] Cincinnati Childrens Hosp Med Ctr, Cincinnati, OH 45229 USA

[19] Univ Texas Southwestern Med Ctr Dallas, Dept Pediat, Dallas, TX 75390 USA

[20] Childrens Hosp Philadelphia, Sect Neuooncol, Philadelphia, PA 19104 USA

[21] Childrens Hosp Philadelphia, Neurooncol Fellowship Program, Philadelphia, PA 19104 USA

[22] Childrens Hosp Philadelphia, Neurofibromatosis Program, Philadelphia, PA 19104 USA

[23] Washington Univ, Sch Med, Neurol Neurosurg Pediat & Genet, St Louis, MO USA

[24] Washington Univ, Sch Med, Res, St Louis, MO USA

[25] Childrens Hosp Los Angeles, Los Angeles, CA 90027 USA

[26] Univ Alabama Birmingham, Med Genet, Birmingham, AL USA

[27] Univ Alabama Birmingham, Dept Genet, Birmingham, AL USA

[28] Univ Alabama Birmingham, Heflin Ctr Genom Sci, Birmingham, AL USA

[29] Monash Univ, Sch Psychol Sci, Cognit Neurosci, Clayton, Vic, Australia

[30] Monash Univ, Sch Psychol Sci, Multidisciplinary Team Studying Biol Basis Attent, Clayton, Vic, Australia

来源：

JOURNAL OF ATTENTION DISORDERS | 2021年 / 25卷 / 08期

基金：

英国医学研究理事会;

关键词：

ADHD; neurofibromatosis type 1; cognition; executive function; functional impairment; ATTENTION-DEFICIT/HYPERACTIVITY DISORDER; QUALITY-OF-LIFE; EXECUTIVE FUNCTION; DYSFUNCTION; LOVASTATIN; VALIDITY; DEFICITS; MODEL; BOYS;

D O I：

10.1177/1087054719894384

中图分类号：

B844 [发展心理学（人类心理学）];

学科分类号：

040202 ;

摘要：

Objective: We examined the contribution of attention and executive cognitive processes to ADHD symptomatology in NF1, as well as the relationships between cognition and ADHD symptoms with functional outcomes. Methods: The study sample consisted of 141 children and adolescents with NF1. Children were administered neuropsychological tests that assessed attention and executive function, from which latent cognitive variables were derived. ADHD symptomatology, adaptive skills, and quality of life (QoL) were assessed using parent-rated questionnaires. Path analyses were conducted to test relationships among cognitive functioning, ADHD symptomatology, and functional outcomes. Results: Significant deficits were observed on all outcome variables. Cognitive variables did not predict ADHD symptomatology. Neither did they predict functional outcomes. However, elevated ADHD symptomatology significantly predicted functional outcomes. Conclusion: Irrespective of cognitive deficits, elevated ADHD symptoms in children with NF1 negatively impact daily functioning and emphasize the importance of interventions aimed at minimizing ADHD symptoms in NF1.

引用

页码：1177 / 1186

页数：10

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