Format

Send to

Choose Destination
Epilepsy Behav. 2017 May;70(Pt A):245-252. doi: 10.1016/j.yebeh.2017.02.007. Epub 2017 Apr 28.

Influence of seizures on early development in tuberous sclerosis complex.

Author information

1
Department of Neurology MLC 2015, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229, USA. Electronic address: Jamie.capal@cchmc.org.
2
Seccion de Neuropediatria, Hospital Infantil Universitario Nino Jesus, Madrid, Spain.
3
Department of Neurology MLC 2015, Cincinnati Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229, USA.
4
Department of Developmental and Behavioral Pediatrics, Cincinnati Children's Hospital Medical Center, 3430 Burnet Avenue, Cincinnati, OH 45229, USA; Autism Speaks Inc, 85 Devonshire St, Boston, MA 02109, USA.
5
Department of Developmental and Behavioral Pediatrics, Cincinnati Children's Hospital Medical Center, 3430 Burnet Avenue, Cincinnati, OH 45229, USA.
6
McGovern Medical School, The University of Texas Health Science Center at Houston, 6431 Fannin Street, Houston, TX 77030, USA.
7
Department of Neurology, Boston Children's Hospital, 300 Longwood Avenue, Boston, MA 02115, USA.

Abstract

OBJECTIVE:

Epilepsy is commonly seen in Tuberous Sclerosis Complex (TSC). The relationship between seizures and developmental outcomes has been reported, but few studies have examined this relationship in a prospective, longitudinal manner. The objective of the study was to evaluate the relationship between seizures and early development in TSC.

METHODS:

Analysis of 130 patients ages 0-36months with TSC participating in the TSC Autism Center of Excellence Network, a large multicenter, prospective observational study evaluating biomarkers predictive of autism spectrum disorder (ASD), was performed. Infants were evaluated longitudinally with standardized evaluations, including cognitive, adaptive, and autism-specific measures. Seizure history was collected continuously throughout, including seizure type and frequency.

RESULTS:

Data were analyzed at 6, 12, 18, and 24months of age. Patients without a history of seizures performed better on all developmental assessments at all time points compared to patients with a history of seizures and exhibited normal development at 24months. Patients with a history of seizures not only performed worse, but developmental progress lagged behind the group without seizures. All patients with a history of infantile spasms performed worse on all developmental assessments at 12, 18, and 24months. Higher seizure frequency correlated with poorer outcomes on developmental testing at all time points, but particularly at 12months and beyond. Patients with higher seizure frequency during infancy continued to perform worse developmentally through 24months. A logistic model looking at the individual impact of infantile spasms, seizure frequency, and age of seizure onset as predictors of developmental delay revealed that age of seizure onset was the most important factor in determining developmental outcome.

CONCLUSIONS:

Results of this study further define the relationship between seizures and developmental outcomes in young children with TSC. Early seizure onset in infants with TSC negatively impacts very early neurodevelopment, which persists through 24months of age.

KEYWORDS:

Autism spectrum disorder; Cognitive development; Epilepsy; Infantile spasms; Seizures; Tuberous Sclerosis Complex

PMID:
28457992
PMCID:
PMC5497719
DOI:
10.1016/j.yebeh.2017.02.007
[Indexed for MEDLINE]
Free PMC Article

Supplemental Content

Full text links

Icon for Elsevier Science Icon for PubMed Central
Loading ...
Support Center