TY - JOUR
T1 - Sleep disorders and neuropsychiatric disorders in a pediatric sample of tuberous sclerosis complex
T2 - a questionnaire-based study
AU - Moavero, Romina
AU - Voci, Alessandra
AU - La Briola, Francesca
AU - Matricardi, Sara
AU - Toldo, Irene
AU - Mancardi, Maria Margherita
AU - Negrin, Susanna
AU - Messana, Tullio
AU - Mazzone, Luigi
AU - Valeriani, Massimiliano
AU - Curatolo, Paolo
AU - Bruni, Oliviero
N1 - Copyright © 2021 The Author(s). Published by Elsevier B.V. All rights reserved.
PY - 2022/1
Y1 - 2022/1
N2 - Objective and background: Sleep disorders (SD) are very common in childhood, especially in certain genetic syndromes. Tuberous Sclerosis Complex (TSC) is a genetic syndromesassociated with a high rate of SD, although these are still under-recognized. The aim of this study was to assess the prevalence of SD in TSC, and to evaluate the relationship between sleep, epilepsy and TSC-associated neuropsychiatric disorders (TAND). Methods: We administered the Sleep Disturbance Scale for Children (SDSC) and the Epworth Sleepiness Scale for Children and Adolescents (ESS-CHAD) to parents of 177 children with TSC referring to different Italian centers. We also collected information on epilepsy and TAND. Results: SDSC score was positive in 59.3% of patients, being positive in 30.4% of patients without and in 63.6% of those with epilepsy (p = 0.005). However, in a multivariate logistic model considering antiseizure medications and nocturnal seizures, epilepsy ceased to be a significant risk factor for positive SDSC (OR = 2.4; p = 0.17). As for TAND, SDSC was positive in 67.9% of patients with and in 32.5% of those without TAND (p < 0.001). After adding in a multivariate logistic model active epilepsy, age, and pharmacotherapies, TAND continued to be a significant risk factor for positive SDSC (p = 0.01, OR = 1.11). Conclusions: Our results revealed a high prevalence of SD in children with TSC. Epilepsy didn't increase the risk for SD, while a very strong association was found with TAND. An early detection of SD is of utmost importance in order to plan an individualized treatment, that in some cases might also ameliorate behavior and attention.
AB - Objective and background: Sleep disorders (SD) are very common in childhood, especially in certain genetic syndromes. Tuberous Sclerosis Complex (TSC) is a genetic syndromesassociated with a high rate of SD, although these are still under-recognized. The aim of this study was to assess the prevalence of SD in TSC, and to evaluate the relationship between sleep, epilepsy and TSC-associated neuropsychiatric disorders (TAND). Methods: We administered the Sleep Disturbance Scale for Children (SDSC) and the Epworth Sleepiness Scale for Children and Adolescents (ESS-CHAD) to parents of 177 children with TSC referring to different Italian centers. We also collected information on epilepsy and TAND. Results: SDSC score was positive in 59.3% of patients, being positive in 30.4% of patients without and in 63.6% of those with epilepsy (p = 0.005). However, in a multivariate logistic model considering antiseizure medications and nocturnal seizures, epilepsy ceased to be a significant risk factor for positive SDSC (OR = 2.4; p = 0.17). As for TAND, SDSC was positive in 67.9% of patients with and in 32.5% of those without TAND (p < 0.001). After adding in a multivariate logistic model active epilepsy, age, and pharmacotherapies, TAND continued to be a significant risk factor for positive SDSC (p = 0.01, OR = 1.11). Conclusions: Our results revealed a high prevalence of SD in children with TSC. Epilepsy didn't increase the risk for SD, while a very strong association was found with TAND. An early detection of SD is of utmost importance in order to plan an individualized treatment, that in some cases might also ameliorate behavior and attention.
KW - Autism
KW - Epilepsy
KW - Neuropsychiatric disorders
KW - Sleep
KW - Sleep disorders
KW - Tuberous sclerosis
UR - http://www.scopus.com/inward/record.url?scp=85121213183&partnerID=8YFLogxK
U2 - 10.1016/j.sleep.2021.11.010
DO - 10.1016/j.sleep.2021.11.010
M3 - Journal article
C2 - 34915263
SN - 1389-9457
VL - 89
SP - 65
EP - 70
JO - Sleep Medicine
JF - Sleep Medicine
ER -