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Fetal Brain Magnetic Resonance Imaging Findings Predict Neurodevelopment in Children with Tuberous Sclerosis Complex

HM. Hulshof, EMH. Slot, M. Lequin, D. Breuillard, N. Boddaert, S. Jozwiak, K. Kotulska, K. Riney, M. Feucht, S. Samueli, T. Scholl, P. Krsek, B. Benova, KPJ. Braun, FE. Jansen, R. Nabbout, EPISTOP consortium

. 2021 ; 233 (-) : 156-162.e2. [pub] 20210226

Language English Country United States

Document type Journal Article, Multicenter Study, Research Support, Non-U.S. Gov't

Persistent link   https://www.medvik.cz/link/bmc21025678

OBJECTIVE: To correlate fetal brain magnetic resonance imaging (MRI) findings with epilepsy characteristics and neurodevelopment at 2 years of age in children with tuberous sclerosis complex (TSC) to improve prenatal counseling. STUDY DESIGN: This retrospective cohort study was performed in a collaboration between centers of the EPISTOP consortium. We included children with definite TSC, fetal MRIs, and available follow-up data at 2 years of age. A pediatric neuroradiologist masked to the patient's clinical characteristics evaluated all fetal MRIs. MRIs were categorized for each of the 10 brain lobes as score 0: no (sub)cortical lesions or doubt; score 1: a single small lesion; score 2: more than one small lesion or at least one large lesion (>5 mm). Neurologic manifestations were correlated to lesion sum scores. RESULTS: Forty-one children were included. Median gestational age at MRI was 33.3 weeks; (sub)cortical lesions were detected in 97.6%. Mean lesion sum score was 4.5. At 2 years, 58.5% of patients had epilepsy and 22% had drug-resistant epilepsy. Cognitive, language, and motor development were delayed in 38%, 81%, and 50% of patients, respectively. Autism spectrum disorder (ASD) was diagnosed in 20.5%. Fetal MRI lesion sum scores were significantly associated with cognitive and motor development, and with ASD diagnosis, but not with epilepsy characteristics. CONCLUSIONS: Fetal cerebral lesion scores correlate with neurodevelopment and ASD at 2 years in children with TSC.

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$a Fetal Brain Magnetic Resonance Imaging Findings Predict Neurodevelopment in Children with Tuberous Sclerosis Complex / $c HM. Hulshof, EMH. Slot, M. Lequin, D. Breuillard, N. Boddaert, S. Jozwiak, K. Kotulska, K. Riney, M. Feucht, S. Samueli, T. Scholl, P. Krsek, B. Benova, KPJ. Braun, FE. Jansen, R. Nabbout, EPISTOP consortium
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$a OBJECTIVE: To correlate fetal brain magnetic resonance imaging (MRI) findings with epilepsy characteristics and neurodevelopment at 2 years of age in children with tuberous sclerosis complex (TSC) to improve prenatal counseling. STUDY DESIGN: This retrospective cohort study was performed in a collaboration between centers of the EPISTOP consortium. We included children with definite TSC, fetal MRIs, and available follow-up data at 2 years of age. A pediatric neuroradiologist masked to the patient's clinical characteristics evaluated all fetal MRIs. MRIs were categorized for each of the 10 brain lobes as score 0: no (sub)cortical lesions or doubt; score 1: a single small lesion; score 2: more than one small lesion or at least one large lesion (>5 mm). Neurologic manifestations were correlated to lesion sum scores. RESULTS: Forty-one children were included. Median gestational age at MRI was 33.3 weeks; (sub)cortical lesions were detected in 97.6%. Mean lesion sum score was 4.5. At 2 years, 58.5% of patients had epilepsy and 22% had drug-resistant epilepsy. Cognitive, language, and motor development were delayed in 38%, 81%, and 50% of patients, respectively. Autism spectrum disorder (ASD) was diagnosed in 20.5%. Fetal MRI lesion sum scores were significantly associated with cognitive and motor development, and with ASD diagnosis, but not with epilepsy characteristics. CONCLUSIONS: Fetal cerebral lesion scores correlate with neurodevelopment and ASD at 2 years in children with TSC.
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$a Slot, Emma M H $u Department of Pediatric Neurology, University Medical Center, Utrecht Brain Center, Utrecht, the Netherlands (Member of the European Reference Network EpiCARE)
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$a Lequin, Maarten $u Department of Radiology, University Medical Center, Utrecht, the Netherlands
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$a Breuillard, Delphine $u Department of Pediatric Neurology, Necker Enfants Maladies Hospital, Paris, France (Member of the European Reference Network EpiCARE)
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$a Boddaert, Nathalie $u Department of Pediatric Radiology, Necker Enfants Maladies Hospital, Paris, France
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$a Jozwiak, Sergiusz $u Instytut Pomnik-Centrum Zdrowia Dziecka, The Children's Memorial Health Institute, Warsaw, Poland (Member of the European Reference Network EpiCARE); Department of Child Neurology, Medical University of Warsaw, Warsaw, Poland
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$a Kotulska, Katarzyna $u Instytut Pomnik-Centrum Zdrowia Dziecka, The Children's Memorial Health Institute, Warsaw, Poland (Member of the European Reference Network EpiCARE)
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$a Riney, Kate $u Neurosciences Unit, Queensland Children's Hospital/University of Queensland School of Medicine, Brisbane, Australia
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$a Feucht, Martha $u Department of Pediatric and Adolescent Medicine, Medical University of Vienna, Vienna, Austria (Member of the European Reference Network EpiCARE)
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$a Samueli, Sharon $u Department of Pediatric and Adolescent Medicine, Medical University of Vienna, Vienna, Austria (Member of the European Reference Network EpiCARE)
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$a Scholl, Theresa $u Department of Pediatric and Adolescent Medicine, Medical University of Vienna, Vienna, Austria (Member of the European Reference Network EpiCARE)
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$a Krsek, Pavel $u Department of Pediatric Neurology, Motol University Hospital, Prague, Czech Republic
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$a Benova, Barbora $u Department of Pediatric Neurology, Motol University Hospital, Prague, Czech Republic
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$a Braun, Kees P J $u Department of Pediatric Neurology, University Medical Center, Utrecht Brain Center, Utrecht, the Netherlands (Member of the European Reference Network EpiCARE)
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$a Jansen, Floor E $u Department of Pediatric Neurology, University Medical Center, Utrecht Brain Center, Utrecht, the Netherlands (Member of the European Reference Network EpiCARE)
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