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Neurobehavioral Concerns Among Males with Dystrophinopathy Using Population-Based Surveillance Data from the Muscular Dystrophy Surveillance, Tracking, and Research Network.

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Neurobehavioral Concerns Among Males with Dystrophinopathy Using Population-Based Surveillance Data from the Muscular Dystrophy Surveillance, Tracking, and Research Network.

Content Provider	World Health Organization (WHO)-Global Index Medicus
Author	Caspers Conway, Kristin Mathews, Katherine D. Paramsothy, Pangaja Oleszek, Joyce Trout, Christina Zhang, Ying Romitti, Paul A.
Organization	MD STARnet
Spatial Coverage	United States
Description	Country affiliation: India Author Affiliation: Caspers Conway K ( *Department of Epidemiology, College of Public Health, The University of Iowa, Iowa City, IA)
Abstract	OBJECTIVE: To describe the occurrence of selected neurobehavioral concerns among males with a dystrophinopathy and to explore the associations with corticosteroid or supportive device use. METHODS: Medical record abstraction of neurobehavioral concerns was conducted for 857 affected males from 765 families, born since 1982 and followed through 2011, and enrolled in the population-based Muscular Dystrophy Surveillance, Tracking, and Research Network. Cumulative probabilities for attention-deficit hyperactivity disorder (ADHD), behavior problems, and depressed mood were calculated from Kaplan-Meier estimates for the subsample of oldest affected males (n = 765). Hazard ratios (HRs) and 95% confidence intervals (95% CIs) for corticosteroid and supportive device use were estimated from Cox regression models with time-dependent covariates. RESULTS: Of the 857 affected males, 375 (44%) had at least 1 of the 3 selected neurobehavioral concerns; a similar percentage (45%) was found among the 765 oldest affected males. The estimated cumulative probabilities among these oldest affected males were 23% for ADHD, 43% for behavior problems, and 51% for depressed mood. Corticosteroid (HR = 2.35, 95% CI = 1.75-3.16) and mobility device (HR = 1.53, 95% CI = 1.06-2.21) use were associated with behavior problems. Use of a mobility device (HR = 3.53, 95% CI = 2.13-5.85), but not corticosteroids, was associated with depressed mood. ADHD was not significantly associated with corticosteroid or mobility device use. Respiratory assist device use was not examined due to low numbers of users before onset of neurobehavioral concerns. CONCLUSION: Selected neurobehavioral concerns were common among males with a dystrophinopathy. Reported associations highlight the importance of increased monitoring of neurobehavioral concerns as interventions are implemented and disease progresses.
File Format	HTM / HTML
ISSN	0196206X
e-ISSN	15367312
DOI	10.1097/DBP.0000000000000177
Journal	Journal of Developmental & Behavioral Pediatrics
Issue Number	6
Volume Number	36
Language	English
Publisher	Lippincott Williams & Wilkins
Publisher Date	2015-07-01
Publisher Place	United States
Access Restriction	Open
Subject Keyword	Discipline Behavioral Sciences Discipline Pediatrics Adrenal Cortex Hormones Therapeutic Use Attention Deficit Disorder With Hyperactivity Epidemiology Depression Muscular Dystrophy, Duchenne Therapy Problem Behavior Registries Self-help Devices Statistics & Numerical Data Adolescent Etiology Child, Preschool Infant Complications Research Support, U.s. Gov't, P.h.s.
Content Type	Text
Resource Type	Article
Subject	Developmental and Educational Psychology Pediatrics, Perinatology and Child Health Psychiatry and Mental Health

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