TY - JOUR
T1 - Cognition, ADHD Symptoms, and Functional Impairment in Children and Adolescents With Neurofibromatosis Type 1
AU - Payne, Jonathan M.
AU - Haebich, Kristina M.
AU - MacKenzie, Rachel
AU - Walsh, Karin S.
AU - Hearps, Stephen J.C.
AU - Coghill, David
AU - Barton, Belinda
AU - Pride, Natalie A.
AU - Ullrich, Nicole J.
AU - Tonsgard, James H.
AU - Viskochil, David
AU - Schorry, Elizabeth K.
AU - Klesse, Laura
AU - Fisher, Michael J.
AU - Gutmann, David H.
AU - Rosser, Tena
AU - Packer, Roger J.
AU - Korf, Bruce
AU - Acosta, Maria T.
AU - Bellgrove, Mark A.
AU - North, Kathryn N.
N1 - Funding Information:
https://orcid.org/0000-0001-9565-3845 Payne Jonathan M. 1 2 Haebich Kristina M. 1 MacKenzie Rachel 3 Walsh Karin S. 4 Hearps Stephen J. C. 1 Coghill David 1 2 Barton Belinda 5 6 Pride Natalie A. 5 6 Ullrich Nicole J. 7 Tonsgard James H. 8 Viskochil David 9 Schorry Elizabeth K. 10 Klesse Laura 11 Fisher Michael J. 12 Gutmann David H. 13 Rosser Tena 14 Packer Roger J. 4 Korf Bruce 15 Acosta Maria T. 4 Bellgrove Mark A. 16 North Kathryn N. 1 2 for the NF Clinical Trials Consortium 1 Murdoch Children’s Research Institute, Royal Children’s Hospital, Parkville, Victoria, Australia 2 University of Melbourne, Parkville, Victoria, Australia 3 La Trobe University, Bundoora, Victoria, Australia 4 Children’s National Health System, Washington, DC, USA 5 The Children’s Hospital at Westmead, New South Wales, Australia 6 The University of Sydney, New South Wales, Australia 7 Boston Children’s Hospital, MA, USA 8 The University of Chicago Medicine Comer Children’s Hospital, IL, USA 9 The University of Utah, Salt Lake City, USA 10 Cincinnati Children’s Hospital Medical Center, OH, USA 11 University of Texas Southwestern Medical Center, Dallas, USA 12 Children’s Hospital of Philadelphia, PA, USA 13 Washington University School of Medicine, St Louis, MO, USA 14 Children’s Hospital Los Angeles, CA, USA 15 The University of Alabama at Birmingham, USA 16 Monash University, Clayton, Victoria, Australia Jonathan M. Payne, Murdoch Children’s Research Institute, Royal Children’s Hospital, Flemington Road, Parkville, Victoria 3052, Australia. Email: [email protected] 12 2019 1087054719894384 © The Author(s) 2019 2019 SAGE Publications 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. ADHD neurofibromatosis type 1 cognition executive function functional impairment US Department of Defense Neurofibromatosis Research Program W81XWH-05-1-0615 edited-state corrected-proof typesetter ts1 Author’s Note Kristina M. Haebich is also affiliated with University of Melbourne, Parkville, Victoria, Australia. Declaration of Conflicting Interests The author(s) declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: D.C. reports grants and personal fees from Shire, and personal fees from Eli Lilly, Medice, Novartis, Oxford University Press, Servier, and grants from Vifor. D.H.G. reports a patent Neurofibromatosis Gene with royalties paid, a patent Neurofibromin pathway modulators issued, and a patent Neurofibromin/dopamine signaling biomarker for cognitive and behavioral problems in children with neurofibromatosis type 1 pending. Funding The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the United States Army Medical Research and Materiel Command, Office of the Congressionally Directed Medical Research Programs, Department of Defense Neurofibromatosis Research Program (Grant Number W81XWH-05-1-0615); J.M.P. is supported by a Murdoch Children’s Research Institute Clinician-Scientist Fellowship; M.A.B. is supported by a Senior Research Fellowship from the National Health and Medical Research Council (NHMRC) of Australia. ORCID iD Jonathan M. Payne https://orcid.org/0000-0001-9565-3845 Supplemental Material Supplemental material for this article is available online.
Funding Information:
The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the United States Army Medical Research and Materiel Command, Office of the Congressionally Directed Medical Research Programs, Department of Defense Neurofibromatosis Research Program (Grant Number W81XWH-05-1-0615); J.M.P. is supported by a Murdoch Children?s Research Institute Clinician-Scientist Fellowship; M.A.B. is supported by a Senior Research Fellowship from the National Health and Medical Research Council (NHMRC) of Australia.
Publisher Copyright:
© The Author(s) 2019.
PY - 2021/6
Y1 - 2021/6
N2 - 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.
AB - 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.
KW - ADHD
KW - cognition
KW - executive function
KW - functional impairment
KW - neurofibromatosis type 1
UR - http://www.scopus.com/inward/record.url?scp=85077157238&partnerID=8YFLogxK
U2 - 10.1177/1087054719894384
DO - 10.1177/1087054719894384
M3 - Article
C2 - 31838937
AN - SCOPUS:85077157238
SN - 1087-0547
VL - 25
SP - 1177
EP - 1186
JO - Journal of Attention Disorders
JF - Journal of Attention Disorders
IS - 8
ER -