Vigabatrin therapy for Epilepsy in children with Tuberous Sclerosis Complex: an analysis of 25 cases in mainland China_Journal of Epilepsy

Authors：

 ZHANGLili ¹ , ZHOUHao ¹ , LUTong ¹ , 李春培 ¹ , ZHENGJing ¹ , ZHOUShuizhen ¹ ,  WANGYi ^1,2

1. Children′s Hospital of Fudan University, Shanghai 201102, China;
2. Collaborative Innovation Center for Brain Science, Fudan University，Shanghai 200032, China;

Corresponding author：

WANGYi, Email: yiwang@shmu.edu.cn

Keywords：

Tuberous Sclerosis Complex; Vigabatrin; Therapeutic effect; Children

DOI：

10.7507/2096-0247.20160020

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Objective To explore the efficiency of Vigabatrin for epilepsy in children with Tuberous Sclerosis Complex, and to further research the risk factors related to the outcome after adjunctive use of Vigabatrin. Methods 25 children with TSC and epilepsy treated with Vigabatrin at Children′s Hospital of Fudan University between 2013 and 2015 were included. Clinical characteristics and the effectiveness of other antiepileptic drugs were extracted from the follow-up data. The prevalence of visual field defect was analyzed among the cases. And correlations were made between the responses to Vigabatrin in groups. Results 25 cases, 15 male (60%). 18 cases had response to VGB-adjuvant therapy. Children with epilepsy onset at greater than six months of age were most likely to demonstrateagood response to VGB treatment. And the poorly response of cases showed that 4 had TSC1 mutation. And among the 25 cases, one child had the visual filed defect. Conclusions Vigabatrin as adjunctive therapy showed certain effect in controlling epilepsy in TSC cases, especially infantile spasms and some partial epilepsy. But the side effect of visual filed defect should be cautious. Age-appropriate visual field testing is recommended at baseline and then repeated at intervals in patients exposed to long term Vigabatrin therapy.

Citation： ZHANGLili, ZHOUHao, LUTong, ZHENGJing, ZHOUShuizhen, WANGYi. Vigabatrin therapy for Epilepsy in children with Tuberous Sclerosis Complex: an analysis of 25 cases in mainland China. Journal of Epilepsy, 2016, 2(2): 109-113. doi: 10.7507/2096-0247.20160020 Copy

1.	Chu-Shore CJ, Major P, Camposano S, et al. The natural history of epilepsy in tuberous sclerosis complex. Epilepsia, 2010, 51(7): 1236-1241.
2.	Willmore LJ, Abelson MB, Ben-Menachem E, et al. Vigabatrin: 2008 update. Epilepsia, 2009, 50(2): 163-173.
3.	Greiner HM, Lynch ER, Fordyce S, et al. Vigabatrin for childhood partial-onset epilepsies. Pediatr Neurol, 2012, 46(2): 83-88.
4.	Friedman D, Bogner M, Parker-Menzer K, et al. Vigabatrin for partial-onset seizure treatment in patients with tuberous sclerosis complex. Epilepsy Behav, 2013, 27(1): 118-120.
5.	Northrup H, Krueger DA. Tuberous sclerosis complex diagnostic criteria update: recommendations of the 2012 Iinternational Tuberous Sclerosis Complex Consensus Conference. Pediatr Neurol, 2013, 49(4): 243-254.
6.	Curatolo P, Verdecchia M, Bombardieri R. Vigabatrin for tuberous sclerosis complex. Brain Dev, 2001, 23(7): 649-653.
7.	DaboraSL, Jozwiak S, Franz DN, et al. Mutational analysis inacohort of 224 tuberous sclerosis patients indicates increased severity of TSC2, compared with TSC1, disease in multiple organs. AmJHum Genet, 2001, 68(1): 64-80.
8.	Overwater IE, Bindels-De HK, Rietman AB, et al. Epilepsy in children with tuberous sclerosis complex: Chance of remission and response to antiepileptic drugs. Epilepsia, 2015, 56(8): 1239-1245.
9.	Jeong A, Wong M. Tuberous sclerosis complex asamodel disease for developing new therapeutics for epilepsy. Expert Rev Neurother, 2016(5): 1-11.
10.	Marcotte L, Crino PB. The neurobiology of the tuberous sclerosis complex. Neuromolecular Med, 2006, 8(4): 531-546.
11.	Dimario FJ, Sahin M, Ebrahimi-Fakhari D. Tuberous sclerosis complex. Pediatr Clin North Am, 2015, 62(3): 633-648.
12.	Walker SD, Kalviainen R. Non-vision adverse events with vigabatrin therapy. Acta Neurol Scand Suppl, 2011(192): 72-82.
13.	Plant GT, Sergott RC. Understanding and interpreting vision safety issues with vigabatrin therapy. Acta Neurol Scand Suppl, 2011(192): 57-71.
14.	Sergott RC, Bittman RM, Christen EM, et al. Vigabatrin-induced peripheral visual field defects in patients with refractory partial epilepsy. Epilepsy Res, 2010, 92(2-3): 170-176.
15.	Luchetti A, Amadi A, Gobbi G. Visual field defects associated with vigabratin monotherapy in children.J Neurol Neurosurg Psychiatry, 2000, 69(4): 566.
16.	Pellock JM, Faught E, Sergott RC, et al. Registry initiated to characterize vision loss associated with vigabatrin therapy. Epilepsy Behav, 2011, 22(4): 710-717.
17.	Wild JM, Chiron C, Ahn H, et al. Visual field loss in patients with refractory partial epilepsy treated with vigabatrin: final results from an open-label, observational, multicentre study. CNS Drugs, 2009, 23(11): 965-982.
18.	Kinirons P, Cavalleri GL, Singh R, et al.Apharmacogenetic exploration of vigabatrin-induced visual field constriction. Epilepsy Res, 2006, 70(2-3): 144-152.
19.	Saxena A, Sampson JR. Epilepsy in Tuberous Sclerosis: phenotypes, mechanisms, and treatments. Semin Neurol, 2015, 35(3): 269-276.

1. Chu-Shore CJ, Major P, Camposano S, et al. The natural history of epilepsy in tuberous sclerosis complex. Epilepsia, 2010, 51(7): 1236-1241.
2. Willmore LJ, Abelson MB, Ben-Menachem E, et al. Vigabatrin: 2008 update. Epilepsia, 2009, 50(2): 163-173.
3. Greiner HM, Lynch ER, Fordyce S, et al. Vigabatrin for childhood partial-onset epilepsies. Pediatr Neurol, 2012, 46(2): 83-88.
4. Friedman D, Bogner M, Parker-Menzer K, et al. Vigabatrin for partial-onset seizure treatment in patients with tuberous sclerosis complex. Epilepsy Behav, 2013, 27(1): 118-120.
5. Northrup H, Krueger DA. Tuberous sclerosis complex diagnostic criteria update: recommendations of the 2012 Iinternational Tuberous Sclerosis Complex Consensus Conference. Pediatr Neurol, 2013, 49(4): 243-254.
6. Curatolo P, Verdecchia M, Bombardieri R. Vigabatrin for tuberous sclerosis complex. Brain Dev, 2001, 23(7): 649-653.
7. DaboraSL, Jozwiak S, Franz DN, et al. Mutational analysis inacohort of 224 tuberous sclerosis patients indicates increased severity of TSC2, compared with TSC1, disease in multiple organs. AmJHum Genet, 2001, 68(1): 64-80.
8. Overwater IE, Bindels-De HK, Rietman AB, et al. Epilepsy in children with tuberous sclerosis complex: Chance of remission and response to antiepileptic drugs. Epilepsia, 2015, 56(8): 1239-1245.
9. Jeong A, Wong M. Tuberous sclerosis complex asamodel disease for developing new therapeutics for epilepsy. Expert Rev Neurother, 2016(5): 1-11.
10. Marcotte L, Crino PB. The neurobiology of the tuberous sclerosis complex. Neuromolecular Med, 2006, 8(4): 531-546.
11. Dimario FJ, Sahin M, Ebrahimi-Fakhari D. Tuberous sclerosis complex. Pediatr Clin North Am, 2015, 62(3): 633-648.
12. Walker SD, Kalviainen R. Non-vision adverse events with vigabatrin therapy. Acta Neurol Scand Suppl, 2011(192): 72-82.
13. Plant GT, Sergott RC. Understanding and interpreting vision safety issues with vigabatrin therapy. Acta Neurol Scand Suppl, 2011(192): 57-71.
14. Sergott RC, Bittman RM, Christen EM, et al. Vigabatrin-induced peripheral visual field defects in patients with refractory partial epilepsy. Epilepsy Res, 2010, 92(2-3): 170-176.
15. Luchetti A, Amadi A, Gobbi G. Visual field defects associated with vigabratin monotherapy in children.J Neurol Neurosurg Psychiatry, 2000, 69(4): 566.
16. Pellock JM, Faught E, Sergott RC, et al. Registry initiated to characterize vision loss associated with vigabatrin therapy. Epilepsy Behav, 2011, 22(4): 710-717.
17. Wild JM, Chiron C, Ahn H, et al. Visual field loss in patients with refractory partial epilepsy treated with vigabatrin: final results from an open-label, observational, multicentre study. CNS Drugs, 2009, 23(11): 965-982.
18. Kinirons P, Cavalleri GL, Singh R, et al.Apharmacogenetic exploration of vigabatrin-induced visual field constriction. Epilepsy Res, 2006, 70(2-3): 144-152.
19. Saxena A, Sampson JR. Epilepsy in Tuberous Sclerosis: phenotypes, mechanisms, and treatments. Semin Neurol, 2015, 35(3): 269-276.

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