Skip to main content

Advertisement

SpringerLink
Log in

Subgroup analysis of seizure and cognitive outcome after vagal nerve stimulator implantation in children

  • Original Article
  • Published:
Child's Nervous System Aims and scope Submit manuscript

Abstract

Objective

Vagal nerve stimulator (VNS) implantation at an early age seems to lead to improved quality of life and cognitive outcome. The aim of this analysis is to evaluate whether specific patient or seizure characteristics might lead to better seizure control, cognitive outcome, and higher quality of life in children undergoing VNS implantation.

Methods

Primary outcome measure was reduction in seizure frequency. Secondary outcome measures were epilepsy outcome assessed by McHugh and Engel classifications, reduction in antiepileptic drugs (AED), developmental and cognitive outcome, as well as quality of life assessed through the pediatric quality of life (PEDSQL™) questionnaire and care giver impression (CGI) scale. Forty-five consecutive children undergoing VNS implantation were analyzed for the following subgroups: age (categorized to 1–2 years old, 3–5 years old, 6–12 years old, and 13–18 years old), sex, underlying cause (categorized to idiopathic, encephalitis, stroke, syndromic), duration of preoperative seizures (dichotomized to under or above 89 months, corresponding to the median of the whole cohort), and preoperative seizure frequency (dichotomized to under and above 360 seizures per month).

Results

Encephalitis as the underlying cause for seizures was the only variable significantly associated with higher reduction rate of seizure frequency. Patients with VNS implantation at the age of ≤ 2 years showed a strong association with better developmental and cognitive outcome, as well as quality of life. Shorter duration of preoperative seizures and higher preoperative seizure frequency showed a strong association with better developmental outcome, as well as quality of life. Engel outcome scores were significantly better in patients with epilepsy due to encephalitis (100% Engel I–III). However, patients with epilepsy due to encephalitis showed significantly higher complication rates (71.4%, p = 0.045).

Conclusions

Children suffering from epilepsy due to encephalitis show higher seizure reduction rates after VNS implantation when compared with children suffering from epilepsy due to other causes. Developmental and cognitive outcomes as well as quality of life of children undergoing VNS implantation is strongly associated with shorter duration of preoperative seizures and implantation at a young age.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4

Similar content being viewed by others

References

  1. Eriksson KJ, Koivikko MJ (1997) Prevalence, classification, and severity of epilepsy and epileptic syndromes in children. Epilepsia 38:1275–1282. https://doi.org/10.1111/j.1528-1157.1997.tb00064.x

    Article  CAS  PubMed  Google Scholar 

  2. Ramos-Lizana J, Aguilera-López P, Aguirre-Rodríguez J, Cassinello-García E (2009) Response to sequential treatment schedules in childhood epilepsy. Seizure 18:620–624. https://doi.org/10.1016/j.seizure.2009.07.001

    Article  CAS  PubMed  Google Scholar 

  3. Mohanraj R, Brodie MJ (2006) Diagnosing refractory epilepsy: response to sequential treatment schedules. Eur J Neurol 13:277–282. https://doi.org/10.1111/j.1468-1331.2006.01215.x

    Article  CAS  PubMed  Google Scholar 

  4. Berg AT, Shinnar S, Levy SR, Testa FM, Smith-Rapaport S, Beckerman B (2001) Early development of intractable epilepsy in children: a prospective study. Neurology 56:1445–1452. https://doi.org/10.1212/WNL.56.11.1445

    Article  CAS  PubMed  Google Scholar 

  5. Berg AT, Wusthoff C, Shellhaas RA, Loddenkemper T, Grinspan ZM, Saneto RP, Knupp KG, Patel A, Sullivan JE, Kossoff EH, Chu CJ, Massey S, Valencia I, Keator C, Wirrell EC, Coryell J, Millichap JJ, Gaillard WD (2019) Immediate outcomes in early life epilepsy: a contemporary account. Epilepsy Behav 97:44–50. https://doi.org/10.1016/j.yebeh.2019.05.011

    Article  PubMed  Google Scholar 

  6. Soleman J, Knorr C, Datta AN, Strozzi S, Ramelli GP, Mariani L, Guzman R (2018) Early vagal nerve stimulator implantation in children: personal experience and review of the literature. Childs Nerv Syst 34:893–900. https://doi.org/10.1007/s00381-017-3694-5

    Article  PubMed  Google Scholar 

  7. Soleman J, Stein M, Knorr C, Datta AN, Constantini S, Fried I, Guzman R, Kramer U (2018) Improved quality of life and cognition after early vagal nerve stimulator implantation in children. Epilepsy Behav 88:139–145. https://doi.org/10.1016/j.yebeh.2018.09.014

    Article  PubMed  Google Scholar 

  8. Healy S, Lang J, Te Water Naude J et al (2013) Vagal nerve stimulation in children under 12 years old with medically intractable epilepsy. Childs Nerv Syst 29:2095–2099. https://doi.org/10.1007/s00381-013-2143-3

    Article  CAS  PubMed  Google Scholar 

  9. Elliott RE, Rodgers SD, Bassani L, Morsi A, Geller EB, Carlson C, Devinsky O, Doyle WK (2011) Vagus nerve stimulation for children with treatment-resistant epilepsy: a consecutive series of 141 cases. J Neurosurg Pediatr 7:491–500. https://doi.org/10.3171/2011.2.PEDS10505

    Article  PubMed  Google Scholar 

  10. Alexopoulos AV, Kotagal P, Loddenkemper T, Hammel J, Bingaman WE (2006) Long-term results with vagus nerve stimulation in children with pharmacoresistant epilepsy. Seizure 15:491–503. https://doi.org/10.1016/j.seizure.2006.06.002

    Article  PubMed  Google Scholar 

  11. Thompson EM, Wozniak SE, Roberts CM, Kao A, Anderson VC, Selden NR (2012) Vagus nerve stimulation for partial and generalized epilepsy from infancy to adolescence. J Neurosurg Pediatr 10:200–205. https://doi.org/10.3171/2012.5.PEDS11489

    Article  PubMed  Google Scholar 

  12. Coykendall DS, Gauderer MWL, Blouin RR, Morales A (2010) Vagus nerve stimulation for the management of seizures in children: an 8-year experience. J Pediatr Surg 45:1479–1483. https://doi.org/10.1016/j.jpedsurg.2010.02.066

    Article  PubMed  Google Scholar 

  13. Cersósimo RO, Bartuluchi M, Fortini S, Soraru A, Pomata H, Caraballo RH (2011) Vagus nerve stimulation: effectiveness and tolerability in 64 paediatric patients with refractory epilepsies. Epileptic Disord 13:382–388. https://doi.org/10.1684/epd.2011.0479

    Article  PubMed  Google Scholar 

  14. Colicchio G, Policicchio D, Barbati G, Cesaroni E, Fuggetta F, Meglio M, Papacci F, Rychlicki F, Scerrati M, Zamponi N (2010) Vagal nerve stimulation for drug-resistant epilepsies in different age, aetiology and duration. Childs Nerv Syst 26:811–819. https://doi.org/10.1007/s00381-009-1069-2

    Article  PubMed  Google Scholar 

  15. Orosz I, McCormick D, Zamponi N, Varadkar S, Feucht M, Parain D, Griens R, Vallée L, Boon P, Rittey C, Jayewardene AK, Bunker M, Arzimanoglou A, Lagae L (2014) Vagus nerve stimulation for drug-resistant epilepsy: a European long-term study up to 24 months in 347 children. Epilepsia 55:1576–1584. https://doi.org/10.1111/epi.12762

    Article  PubMed  Google Scholar 

  16. Bodin E, Le Moing A-G, Bourel-Ponchel E et al (2016) Vagus nerve stimulation in the treatment of drug-resistant epilepsy in 29 children. Eur J Paediatr Neurol 20:346–351. https://doi.org/10.1016/j.ejpn.2016.01.011

    Article  PubMed  Google Scholar 

  17. Zamponi N, Rychlicki F, Corpaci L, Cesaroni E, Trignani R (2008) Vagus nerve stimulation (VNS) is effective in treating catastrophic 1 epilepsy in very young children. Neurosurg Rev 31:291–297. https://doi.org/10.1007/s10143-008-0134-8

    Article  PubMed  Google Scholar 

  18. Yu C, Ramgopal S, Libenson M, Abdelmoumen I, Powell C, Remy K, Madsen JR, Rotenberg A, Loddenkemper T (2014) Outcomes of vagal nerve stimulation in a pediatric population: a single center experience. Seizure 23:105–111. https://doi.org/10.1016/j.seizure.2013.10.002

    Article  PubMed  Google Scholar 

  19. Varni JW, Seid M, Rode CA (1999) The PedsQL: measurement model for the pediatric quality of life inventory. Med Care 37:126–139. https://doi.org/10.1097/00005650-199902000-00003

    Article  CAS  Google Scholar 

  20. Tsai J-D, Chang Y-C, Lin L-C, Hung K-L (2016) The neuropsychological outcome of pediatric patients with refractory epilepsy treated with VNS — a 24-month follow-up in Taiwan. Epilepsy Behav 56:95–98. https://doi.org/10.1016/j.yebeh.2015.12.030

    Article  PubMed  Google Scholar 

  21. Menascu S, Kremer U, Schiller Y, Blatt I, Watemberg N, Boxer M, Goldberg H, Korn-Lubetzki I, Steinberg M, Ben-Zeev B (2013) The Israeli retrospective multicenter open-label study evaluating vagus nerve stimulation efficacy in children and adults. Isr Med Assoc J 15:673–677

    PubMed  Google Scholar 

  22. Hallböök T, Lundgren J, Stjernqvist K, Blennow G, Strömblad LG, Rosén I (2005) Vagus nerve stimulation in 15 children with therapy resistant epilepsy; its impact on cognition, quality of life, behaviour and mood. Seizure 14:504–513. https://doi.org/10.1016/j.seizure.2005.08.007

    Article  PubMed  Google Scholar 

  23. Thompson PJ, Duncan JS (2005) Cognitive decline in severe intractable epilepsy. Epilepsia 46:1780–1787. https://doi.org/10.1111/j.1528-1167.2005.00279.x

    Article  PubMed  Google Scholar 

  24. Vendrame M, Alexopoulos AV, Boyer K, Gregas M, Haut J, Lineweaver T, Wyllie E, Loddenkemper T (2009) Longer duration of epilepsy and earlier age at epilepsy onset correlate with impaired cognitive development in infancy. Epilepsy Behav 16:431–435. https://doi.org/10.1016/j.yebeh.2009.08.008

    Article  PubMed  Google Scholar 

  25. Livanova MRI Guidelines for VNS Therapy®. http://omamedical.fi/en/wp-content/uploads/sites/2/2018/09/MRI-Guidelines-for-VNS-Therapy-2018.pdf. Accessed 22 Feb 2020

  26. Englot DJ, Chang EF, Auguste KI (2011) Vagus nerve stimulation for epilepsy: a meta-analysis of efficacy and predictors of response. J Neurosurg 115:1248–1255. https://doi.org/10.3171/2011.7.JNS11977

    Article  PubMed  Google Scholar 

  27. Elliott RE, Morsi A, Kalhorn SP, Marcus J, Sellin J, Kang M, Silverberg A, Rivera E, Geller E, Carlson C, Devinsky O, Doyle WK (2011) Vagus nerve stimulation in 436 consecutive patients with treatment-resistant epilepsy: long-term outcomes and predictors of response. Epilepsy Behav 20:57–63. https://doi.org/10.1016/j.yebeh.2010.10.017

    Article  PubMed  Google Scholar 

  28. Ji T, Yang Z, Liu Q, Liao J, Yin F, Chen Y, Zou L, Li B, Gao Y, Shu X, Huang S, Gao F, Liang J, Lin SF, Peng J, Song S, Wang J, Che C, Sun W, Tian M, Yang L, Hua Y, Hao Y, Cai L, Li L, Jiang Y (2019) Vagus nerve stimulation for pediatric patients with intractable epilepsy between 3 and 6 years of age: study protocol for a double-blind, randomized control trial. Trials 20:44. https://doi.org/10.1186/s13063-018-3087-4

    Article  PubMed  PubMed Central  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Neurosurgery and Division of Pediatric Neurosurgery, Children’s University Hospital of Basel (UKBB), Basel, Switzerland

    Corine Knorr, Ladina Greuter, Raphael Guzman & Jehuda Soleman

  2. Departments of Neurosurgery and Pediatric Neurosurgery, Tel Aviv Medical Center and Dana Children’s Hospital, Tel Aviv University, Tel Aviv, Israel

    Shlomi Constantini, Itzhak Fried & Jehuda Soleman

  3. Department of Pediatric Neurology, Dana Children’s Hospital, Tel Aviv University, Tel Aviv, Israel

    Uri Kremer

  4. Department of Pediatric Neurology, Children’s University Hospital of Basel (UKBB), Basel, Switzerland

    Alexandre N. Datta

  5. Faculty of Medicine, University of Basel, Basel, Switzerland

    Raphael Guzman & Jehuda Soleman

Authors
  1. Corine Knorr
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Ladina Greuter
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Shlomi Constantini
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Itzhak Fried
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Uri Kremer
    View author publications

    You can also search for this author in PubMed Google Scholar

  6. Alexandre N. Datta
    View author publications

    You can also search for this author in PubMed Google Scholar

  7. Raphael Guzman
    View author publications

    You can also search for this author in PubMed Google Scholar

  8. Jehuda Soleman
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Jehuda Soleman.

Ethics declarations

Conflict of interest

The authors report no conflict of interest concerning the materials or methods used in this study or the findings specified in this paper.

Additional information

Publisher’s note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Knorr, C., Greuter, L., Constantini, S. et al. Subgroup analysis of seizure and cognitive outcome after vagal nerve stimulator implantation in children. Childs Nerv Syst 37, 243–252 (2021). https://doi.org/10.1007/s00381-020-04628-0

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00381-020-04628-0

Keywords

Search

Navigation