TY - JOUR
T1 - Evolution of electroencephalogram in infants with tuberous sclerosis complex and neurodevelopmental outcome
T2 - a prospective cohort study
AU - EPISTOP Consortium
AU - De Ridder, Jessie
AU - Kotulska, Katarzyna
AU - Curatolo, Paolo
AU - Jansen, Anna C
AU - Aronica, Eleonora
AU - Kwiatkowski, David J
AU - Jansen, Floor E
AU - Jóźwiak, Sergiusz
AU - Lagae, Lieven
N1 - © 2021 Mac Keith Press.
PY - 2022/4
Y1 - 2022/4
N2 - AIM: To describe the evolution of electroencephalogram (EEG) characteristics in infants with tuberous sclerosis complex (TSC) and the relationship with neurodevelopmental outcome at 24 months.METHOD: Eighty-three infants were enrolled in the EPISTOP trial and underwent serial EEG follow-up until the age of 24 months (males n=45, females n=37, median age at enrolment 28d, interquartile range 14-54d). Maturation of the EEG background and epileptiform discharges were compared between the TSC1 and TSC2 variants and between preventive and conventional groups respectively.RESULTS: Children with TSC2 more frequently had a slower posterior dominant rhythm (PDR) at 24 months (51% vs 11%, p=0.002), a higher number of epileptiform foci (median=8 vs 4, p=0.003), and a lower fraction of EEGs without epileptiform discharges (18% vs 61%, p=0.001) at follow-up. A slower PDR at 24 months was significantly associated with lower cognitive (median=70 vs 80, p=0.028) and motor developmental quotients (median=70 vs 79, p=0.008). A higher fraction of EEGs without epileptiform discharges was associated with a lower probability of autism spectrum disorder symptoms (odds ratio=0.092, 95% confidence interval=0.009-0.912, p=0.042) and higher cognitive (p=0.004), language (p=0.002), and motor (p=0.001) developmental quotients at 24 months.INTERPRETATION: TSC2 is associated with more abnormal EEG characteristics compared to TSC1, which are predictive for neurodevelopmental outcome.
AB - AIM: To describe the evolution of electroencephalogram (EEG) characteristics in infants with tuberous sclerosis complex (TSC) and the relationship with neurodevelopmental outcome at 24 months.METHOD: Eighty-three infants were enrolled in the EPISTOP trial and underwent serial EEG follow-up until the age of 24 months (males n=45, females n=37, median age at enrolment 28d, interquartile range 14-54d). Maturation of the EEG background and epileptiform discharges were compared between the TSC1 and TSC2 variants and between preventive and conventional groups respectively.RESULTS: Children with TSC2 more frequently had a slower posterior dominant rhythm (PDR) at 24 months (51% vs 11%, p=0.002), a higher number of epileptiform foci (median=8 vs 4, p=0.003), and a lower fraction of EEGs without epileptiform discharges (18% vs 61%, p=0.001) at follow-up. A slower PDR at 24 months was significantly associated with lower cognitive (median=70 vs 80, p=0.028) and motor developmental quotients (median=70 vs 79, p=0.008). A higher fraction of EEGs without epileptiform discharges was associated with a lower probability of autism spectrum disorder symptoms (odds ratio=0.092, 95% confidence interval=0.009-0.912, p=0.042) and higher cognitive (p=0.004), language (p=0.002), and motor (p=0.001) developmental quotients at 24 months.INTERPRETATION: TSC2 is associated with more abnormal EEG characteristics compared to TSC1, which are predictive for neurodevelopmental outcome.
KW - electroencephalogram
KW - infants
KW - tuberous sclerosis complex
KW - neurodevelopmental outcome
UR - http://www.scopus.com/inward/record.url?scp=85116455796&partnerID=8YFLogxK
U2 - 10.1111/dmcn.15073
DO - 10.1111/dmcn.15073
M3 - Article
C2 - 34601720
SN - 0012-1622
VL - 64
SP - 495
EP - 501
JO - Developmental Medicine and Child Neurology
JF - Developmental Medicine and Child Neurology
IS - 4
ER -