Prediction of Neurodevelopment in Infants With Tuberous Sclerosis Complex Using Early EEG Characteristics

被引:27
|
作者
De Ridder, Jessie [1 ]
Lavanga, Mario [2 ]
Verhelle, Birgit [1 ]
Vervisch, Jan [1 ]
Lemmens, Katrien [1 ]
Kotulska, Katarzyna [3 ]
Moavero, Romina [4 ,5 ]
Curatolo, Paolo [4 ]
Weschke, Bernhard [6 ]
Riney, Kate [7 ,8 ]
Feucht, Martha [9 ]
Krsek, Pavel [10 ]
Nabbout, Rima [11 ]
Jansen, Anna C. [12 ]
Wojdan, Konrad [13 ,14 ]
Domanska-Pakiela, Dorota [3 ]
Kaczorowska-Frontczak, Magdalena [3 ]
Hertzberg, Christoph [15 ]
Ferrier, Cyrille H. [16 ]
Samueli, Sharon [9 ]
Benova, Barbora [10 ]
Aronica, Eleonora [17 ,18 ]
Kwiatkowski, David J. [19 ]
Jansen, Floor E. [16 ]
Jozwiak, Sergiusz [3 ,20 ]
Van Huffel, Sabine [12 ]
Lagae, Lieven [1 ]
机构
[1] Univ Leuven KU Leuven, Dept Dev & Regenerat, Pediat Neurol, Leuven, Belgium
[2] Katholieke Univ Leuven, STADIUS Ctr Dynam Syst Signal Proc & Data Analyt, Dept Elect Engn ESAT, Leuven, Belgium
[3] Childrens Mem Hlth Inst, Dept Neurol & Epileptol, Warsaw, Poland
[4] Tor Vergata Univ, Child Neurol & Psychiat Unit, Syst Med Dept, Rome, Italy
[5] Bambino Gesu Pediat Hosp, Neurosci & Neurorehabil Dept, Chid Neurol Unit, Rome, Italy
[6] Charite Univ Med Berlin, Dept Child Neurol, Berlin, Germany
[7] Queensland Childrens Hosp, Neurosci Unit, Brisbane, Qld, Australia
[8] Univ Queensland, Sch Clin Med, Brisbane, Qld, Australia
[9] Med Univ Vienna, Dept Pediat, Vienna, Austria
[10] Charles Univ Prague, Fac Med 2, Motol Univ Hosp, Dept Paediat Neurol, Prague, Czech Republic
[11] Univ Paris 05, Necker Enfants Malad Hosp, Imagine Inst, Dept Pediat Neurol,Reference Ctr Rare Epilepsies, Paris, France
[12] Univ Ziekenhuis Brussel, Pediat Neurol Unit, Brussels, Belgium
[13] Transit Technol, Warsaw, Poland
[14] Warsaw Univ & Technol, Inst Heat Engn, Warsaw, Poland
[15] Vivantes Klinikum Neukoln, Diag & Behandlungszentrum Kinder & Jugendl, Berlin, Germany
[16] Univ Med Ctr Utrecht, Brain Ctr, Dept Child Neurol, Utrecht, Netherlands
[17] Univ Amsterdam, Amsterdam Univ Med Ctr, Dept Neuro Pathol, Amsterdam, Netherlands
[18] Stichting Epilepsie Instellingen Nederland SEIN, Heemstede, Netherlands
[19] Harvard Med Sch, Brigham & Womens Hosp, Boston, MA 02115 USA
[20] Med Univ Warsaw, Dept Child Neurol, Warsaw, Poland
来源
FRONTIERS IN NEUROLOGY | 2020年 / 11卷
基金
欧盟第七框架计划;
关键词
tuberous sclerosis complex (TSC); EEG; biomarker; neurodeveloment; autism (ASD); TAND profile; PRETERM INFANTS; EPILEPSY; ONSET; CHILDREN; SEIZURES; RISK; ABNORMALITIES; AUTISM;
D O I
10.3389/fneur.2020.582891
中图分类号
R74 [神经病学与精神病学];
学科分类号
摘要
Tuberous Sclerosis Complex (TSC) is a multisystem genetic disorder with a high risk of early-onset epilepsy and a high prevalence of neurodevelopmental comorbidities, including intellectual disability and autism spectrum disorder (ASD). Therefore, TSC is an interesting disease model to investigate early biomarkers of neurodevelopmental comorbidities when interventions are favourable. We investigated whether early EEG characteristics can be used to predict neurodevelopment in infants with TSC. The first recorded EEG of 64 infants with TSC, enrolled in the international prospective EPISTOP trial (recorded at a median gestational age 42 4/7 weeks) was first visually assessed. EEG characteristics were correlated with ASD risk based on the ADOS-2 score, and cognitive, language, and motor developmental quotients (Bayley Scales of Infant and Toddler Development III) at the age of 24 months. Quantitative EEG analysis was used to validate the relationship between EEG background abnormalities and ASD risk. An abnormal first EEG (OR = 4.1, p-value = 0.027) and more specifically a dysmature EEG background (OR = 4.6, p-value = 0.017) was associated with a higher probability of ASD traits at the age of 24 months. This association between an early abnormal EEG and ASD risk remained significant in a multivariable model, adjusting for mutation and treatment (adjusted OR = 4.2, p-value = 0.029). A dysmature EEG background was also associated with lower cognitive (p-value = 0.029), language (p-value = 0.001), and motor (p-value = 0.017) developmental quotients at the age of 24 months. Our findings suggest that early EEG characteristics in newborns and infants with TSC can be used to predict neurodevelopmental comorbidities.
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页数:9
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