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Pediatric Neurology

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Pilot Study of Neurodevelopmental Impact of Early Epilepsy Surgery in Tuberous Sclerosis Complex - 23/07/20

Doi : 10.1016/j.pediatrneurol.2020.04.002 
Leslie E. Grayson, MD a, †, Jurriaan M. Peters, MD, PhD b, †, Tarrant McPherson, MA c, Darcy A. Krueger, MD, PhD d, Mustafa Sahin, MD, PhD e, Joyce Y. Wu, MD f, Hope A. Northrup, MD g, Brenda Porter, MD h, Gary R. Cutter, PhD c, Sarah E. O'Kelley, PhD i, Jessica Krefting, RN a, Scellig S. Stone, MD, PhD j, Joseph R. Madsen, MD j, Aria Fallah, MD, MS k, Jeffrey P. Blount, MD l, Howard L. Weiner, MD m, E. Martina Bebin, MD, MPA a, ⁎
on behalf of the

TACERN Study Group

Simon K. Warfield, PhD, Monisha Goyal, MD, Deborah A. Pearson, PhD, Marian E. Williams, PhD, Ellen Hanson, PhD, Nicole Bing, PsyD, Bridget Kent, MA, CCC-SLP, Rajna Filip-Dhima, MS, Kira Dies, ScM, CGC, Stephanie Bruns, Benoit Scherrer, PhD, Donna S. Murray, PhD, Steven L. Roberds, PhD, Jamie Capal

a Department of Neurology, University of Alabama at Birmingham, Birmingham, Alabama 
b Localization Laboratory, Division of Epilepsy and Clinical Neurophysiology, Department of Neurology, Boston Children's Hospital and Harvard Medical School, Boston, Massachusetts 
c Department of Biostatistics, University of Alabama at Birmingham, Birmingham, Alabama 
d Department of Neurology, Cincinnati Children's Hospital Medical Center, and Department of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, Ohio 
e Department of Neurology and the F.M. Kirby Neurobiology Center, Boston Children's Hospital and Harvard Medical School, Boston, Massachusetts 
f Division of Pediatric Neurology, UCLA Mattel Children's Hospital, David Geffen School of Medicine, University of California Los Angeles, Los Angeles, California 
g Department of Pediatrics, McGovern Medical School, University of Texas Health Science Center at Houston, Houston, Texas 
h Department of Neurology, The Stanford University Medical Center, Stanford, California 
i Department of Psychology, University of Alabama at Birmingham, Birmingham, Alabama 
j Division of Pediatric Neurosurgery, Department of Neurosurgery, Boston Children's Hospital and Harvard Medical School, Boston, Massachusetts 
k Department of Neurosurgery, Division of Pediatric Neurosurgery, University of California Los Angeles Mattel Children's Hospital, David Geffen School of Medicine, University of California Los Angeles, Los Angeles, California 
l Department of Neurosurgery, Division of Pediatric Neurosurgery, Children's Hospital of Alabama and University of Alabama at Birmingham, Birmingham, Alabama 
m Division of Pediatric Neurosurgery, Department of Surgery, Texas Children's Hospital and Department of Neurosurgery, Baylor College of Medicine, Houston, Texas 

∗Communications should be addressed to: Bebin; Department of Neurology; CIRC 312; 1530 3rd Ave S; Birmingham, AL 35294-3280.Department of NeurologyCIRC 3121530 3rd Ave SBirminghamAL35294-3280

Abstract

Background

To determine if early epilepsy surgery mitigates detrimental effects of refractory epilepsy on development, we investigated surgical and neurodevelopmental outcomes in children with tuberous sclerosis complex who underwent surgery before age two years.

Methods

Prospective multicenter observational study of 160 children with tuberous sclerosis complex. Surgical outcome was determined for the seizure type targeted by surgery. We obtained Vineland Adaptive Behavior Scales, Second Edition (Vineland-II); Mullen Scales of Early Learning; and Preschool Language Scales, Fifth Edition, at age three, six, nine, 12, 18, 24, and 36 months. Surgical cases were compared with children without seizures, with controlled seizures, and with medically refractory seizures.

Results

Nineteen children underwent surgery (median age 17 months, range 3.7 to 21.3), and mean follow-up was 22.8 months (range 12 to 48). Surgical outcomes were favorable in 12 (63%, Engel I-II) and poor in seven (37%, Engel III-IV). Nine (47%) had new or ongoing seizures distinct from those surgically targeted. All children with seizures demonstrated longitudinal decline or attenuated gains in neurodevelopment, the surgical group scoring the lowest. Favorable surgical outcome was associated with increased Mullen Scales of Early Learning receptive and expressive language subscores compared with the medically refractory seizure group. A nonsignificant but consistent pattern of improvement with surgery was seen in all tested domains.

Conclusions

These pilot data show neurodevelopmental gains in some domains following epilepsy surgery. A properly powered, prospective multicenter observational study of early epilepsy surgery is needed, using both surgical and developmental outcome metrics.

Le texte complet de cet article est disponible en PDF.

Keywords : Children, Epilepsy, Tuberous sclerosis complex, Epilepsy surgery


Plan


 Funding: Research reported in this publication was supported by the National Institute of Neurological Disorders And Stroke of the National Institutes of Health (NINDS) and Eunice Kennedy Shriver National Institute of Child Health & Human Development (NICHD) under Award Number U01NS082320. The content is solely the responsibility of the authors and does not necessarily represent the official views of the National Institutes of Health.
 Author contributions: See Appendix 1.
 Conflicts of interest: The authors report no conflicts of interest relevant to the manuscript.


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Vol 109

P. 39-46 - août 2020 Retour au numéro
Article précédent Article précédent
  • The Efficacy and Safety of Rapid Cycling Vagus Nerve Stimulation in Children With Intractable Epilepsy
  • Husam Kayyali, Sherouk Abdelmoity, Lalit Bansal, Christian Kaufman, Kyle Smith, Erin Fecske, Kailash Pawar, Ara Hall, Megan Gustafson, Ayman Abdelmoity, Ahmed Abdelmoity
| Article suivant Article suivant
  • Intracranial Pseudoaneurysm Caused by Cerebral Paragonimiasis in Pediatric Patients
  • Lusheng Li, Yuting Zhang, Jin Zhu, Xuan Zhai, Jinhua Cai, Ling He, Ping Liang

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