Pilot Validation of the Tuberous Sclerosis-Associated Neuropsychiatric Disorders (TAND) Checklist
psychosocial; children; Pediatrics; cognition; behavior; Neurosciences & Neurology; mental health; autism; gene; recommendations; academic; attention deficits; complex consensus conference; diagnostic-criteria; identification; neuropsychology; population; sample; TAND; tuberous sclerosis complex; behavioral problems; tuberous sclerosis; tool development; scale development
BACKGROUND: Tuberous sclerosis complex is a multisystem disorder that includes a range of tuberous sclerosis associated neuropsychiatric disorders (TAND). The lifetime prevalence rates of TAND are very high; yet surveys suggest that the majority of individuals with tuberous sclerosis never receive appropriate assessment or treatment for TAND. To aid systematic enquiry, a TAND Checklist was developed. Here, we performed pilot validation of the TAND Checklist. METHOD: Mixed methods were used across two stages. In stage 1, we gathered feedback on the Checklist from tuberous sclerosis "expert professionals" and "expert parents and caregivers." The aim was to examine face and content validity. Stage 2 involved the administration of the refined TAND Checklist to 20 parents of individuals with tuberous sclerosis concurrently with four widely used validated rating scales, to examine external validity and obtain qualitative feedback on face-to-face administration of the TAND Checklist. RESULTS: Twenty professionals and 62 parents and caregivers from 28 countries participated in the pilot. The TAND Checklist demonstrated good face and content validity with high overall mean and median scores. Qualitative analysis highlighted concerns about the likely use of the TAND Checklist, suggesting that family members and individuals with tuberous sclerosis should drive usage. Stage 2 results showed moderate-to-very good external validity across TAND domain and key subdomains. Internal consistency of domains and subdomains was acceptable to very good. Ninety-three percent of all participants (93%) reported four or more lifetime TAND behavioral difficulties. CONCLUSION: The pilot validation suggested that the TAND Checklist could provide a useful screening tool in clinical settings.
Leclezio L; Jansen A; Whittemore V H; de Vries P J
Pediatric Neurology
2015
Article information provided for research and reference use only. PedPalASCNET does not hold any rights over the resource listed here. All rights are retained by the journal listed under publisher and/or the creator(s).
<a href="http://doi.org/10.1016/j.pediatrneurol.2014.10.006" target="_blank" rel="noreferrer noopener">10.1016/j.pediatrneurol.2014.10.006</a>
Behavior problems in children with tuberous sclerosis complex and parental stress
cancer; families; adolescents; Neurosciences & Neurology; seizure; attention; epilepsy; disorders; autism; Psychiatry; hyperactivity; autism spectrum disorder; diagnostic-criteria; population; tuberous sclerosis complex; Behavioral Sciences; mental; behavioral problems; frequency; infantile spasms; methylphenidate; mixed seizure disorder; parenting stress; retardation; tsc1; tuberous sclerosis; trajectory; characteristics
Behavioral problems are common in children with tuberous sclerosis complex (TSC) and can be challenging to manage at home. Standardized measures were used to assess behavior in 99 pediatric patients with TSC and to evaluate parenting stress in their parents. About 40% of the pediatric patients presented clinically significant behavioral problems, most frequently involving symptoms of autism spectrum disorder, inattention, and hyperactivity. Higher seizure frequency, mixed seizure disorder, and low intellectual functioning placed the patient at significant risk for behavior problems. Almost 50% of participating parents reported experiencing clinically significant parenting stress, which was associated with specific characteristics of the child, including the presence of current seizures, a history of psychiatric diagnosis, low intelligence, and behavioral problems. Clinicians should be aware that behavioral problems are prominent in children with TSC. Referrals for behavioral intervention and monitoring of parental stress should be included in the medical management of children with TSC. (C) 2008 Elsevier Inc. All rights reserved.
Kopp C M C; Muzykewicz D A; Staley B A; Thiele E A; Pulsifer M B
Epilepsy and Behavior
2008
Article information provided for research and reference use only. PedPalASCNET does not hold any rights over the resource listed here. All rights are retained by the journal listed under publisher and/or the creator(s).
<a href="http://doi.org/10.1016/j.yebeh.2008.05.010" target="_blank" rel="noreferrer noopener">10.1016/j.yebeh.2008.05.010</a>