Vigabatrin for seizures treatment in patients with tuberous sclerosis complex: an efficacy and safety study_Journal of Epilepsy

Authors：

 HUANGLulu ^1,2 ,  PANGLingyu ¹ , WANGYangyang ^1,2 , HULinyan ¹ , GUOShufang ^1,2 , GAOYang ¹ , LIUYujie ¹ , ZHANGMengna ¹ , MAShufang ¹ ,  ZOULiping ^1,3

1. Department of Pediatrics, General Hospital of Chinese PLA, Beijing 100853, China;
2. Medical School of Nankai University, Tianjin 300071, China;
3. Institute of Epilepsy, Beijing Institute for Brain Disorders, Beijing 100069, China;

Corresponding author：

ZOULiping, Email: zouliping21@hotmail.com

Keywords：

Vigabatrin; Tuberous sclerosis complex; Epilepsy; Infantile spasms

DOI：

10.7507/2096-0247.20160040

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Objective To evaluate the efficacy, tolerability and safety of vigabatrin (VGB) for seizure treatment in patients with tuberous sclerosis complex (TSC). Methods Forty-one epilepsy patients with tuberous sclerosis complex, admitted from January 2015 to December 2015, were included in our study; they were treated with VGB with an initial dose of 20 mg/(kg·d), and a maintenance dose of 50~ 100 mg/(kg·d). Baseline seizure frequency were evaluated by the parents or the guardian, and investigated the efficacy, tolerability, adverse reactions and safety in 3 and 6 months after treatment, and compared with the baseline. The treatment outcomes were evaluated by seizure frequency as completely seizure free (100% seizure reduction), markedly effective (75%~99% seizure reduction), effective (50%~74% seizure reduction) and invalid ( < 50% seizure reduction). Adverse reactions were observed during treatment. Results The completely seizure free rates after 3 and 6 months treatment were 51.2% and 57.9%; and the total effective rates (completely seizure free+markedly effective+effective) were 90.2% and 89.5%.During the 6 months, only one patients stopped VGB use because of the poor efficacy and the difficulties to buy this medicine. 14 patients appeared adverse reactions, including drowsiness, agitation, hyperactivity and myoclonus, which were transient and mild. No patients had clinically perceivable visual-field changes on clinical examination. Conclusion VGB is a effective treatment in TSC patients with epilepsy, and have a good security in short term.

Citation： HUANGLulu, PANGLingyu, WANGYangyang, HULinyan, GUOShufang, GAOYang, LIUYujie, ZHANGMengna, MAShufang, ZOULiping. Vigabatrin for seizures treatment in patients with tuberous sclerosis complex: an efficacy and safety study. Journal of Epilepsy, 2016, 2(3): 216-219. doi: 10.7507/2096-0247.20160040 Copy

1.	Osborne JP, Fryer A, Webb D. Epidemiology of tuberous sclerosis. Ann Ny Acad Sci, 1991, 61(21):125-127.
2.	Curatolo P, Bombardieri R, Jozwiak S. Tuberous sclerosis. Lancet, 2008, 372(9639):657-668.
3.	Wang S, Fallah A. Optimal management of seizures associated with tuberous sclerosis complex:current and emerging options. Neuropsychiatr Dis Treat, 2014, 10(8):2021-2030.
4.	Thiele EA. Managing and understanding epilepsy in tuberous sclerosis complex. Epilepsia, 2010, 51(Suppl 1):90-91.
5.	Angehagen M, Ben-Menachem E, Ronnback L, et al. Novel mechanisms of action of three antiepileptic drugs, vigabatrin, tiagabine, and topiramate. Neurochem Res, 2003, 28(2):333-340.
6.	Shields WD, Pellock JM. Vigabatrin 35 years later-from mechanism of action to benefit-risk considerations. Acta Neurol Scand, 2011, 192(Suppl 2):1-4.
7.	Aicardi J, Mumford JP, Dumas C, et al. Vigabatrin as initial therapy for infantile spasms:a European retrospective survey. Sabril IS Investigator and Peer Review Groups. Epilepsia, 1996, 37(7):638-642.
8.	Schwarz MD, Li M, Tsao J, et al. A lack of clinically apparent vision loss among patients treated with vigabatrin with infantile spasms:The UCLA experience. Epilepsy Behav, 2016, 57(Pt A):29-33.
9.	Nabbout RC, Chiron C, Mumford J, et al. Vigabatrin in partial seizures in children. J Child Neurol, 1997, 12(3):172-177.
10.	Willmore LJ, Abelson MB, Ben-Menachem E, et al. Vigabatrin:2008 update. Epilepsia, 2009, 50(2):163-173.
11.	Riikonen RS. Favourable prognostic factors with infantile spasms. Eur J Paediatr Neurol, 2010, 14(1):13-18.
12.	Hancock E, Osborne J, Milner P. Treatment of infantile spasms. Cochrane Database Syst Rev, 2003, (3):Cd001770.
13.	Chiron C, Dumas C, Jambaque I, et al. Randomized trial comparing vigabatrin and hydrocortisone in infantile spasms due to tuberous sclerosis. Epilepsy Res, 1997, 26(2):389-395.
14.	Yum MS, Lee EH, Ko TS. Vigabatrin and mental retardation in tuberous sclerosis:infantile spasms versus focal seizures. J Child Neurol, 2013, 28(3):308-313.
15.	Appleton RE. The role of vigabatrin in the management of infantile epileptic syndromes. Neurology, 1993, 43(11 Suppl 5):21-23.
16.	Elterman RD, Shields WD, Bittman RM, et al. Vigabatrin for the treatment of infantile spasms:final report of a randomized trial. J Child Neurol, 2010, 25(11):1340-1347.
17.	Gaily E, Jonsson H, Lappi M. Visual fields at school-age in children treated with vigabatrin in infancy. Epilepsia, 2009, 50(2):206-216.
18.	Taghdiri MM, Ashrafi MR, Bakhshandeh-Bali MK, et al. Clinical trial of vigabatrin as adjunctive therapy in children with refractory epilepsy. Iran J Pediatr, 2013, 23(6):653-658.
19.	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.

1. Osborne JP, Fryer A, Webb D. Epidemiology of tuberous sclerosis. Ann Ny Acad Sci, 1991, 61(21):125-127.
2. Curatolo P, Bombardieri R, Jozwiak S. Tuberous sclerosis. Lancet, 2008, 372(9639):657-668.
3. Wang S, Fallah A. Optimal management of seizures associated with tuberous sclerosis complex:current and emerging options. Neuropsychiatr Dis Treat, 2014, 10(8):2021-2030.
4. Thiele EA. Managing and understanding epilepsy in tuberous sclerosis complex. Epilepsia, 2010, 51(Suppl 1):90-91.
5. Angehagen M, Ben-Menachem E, Ronnback L, et al. Novel mechanisms of action of three antiepileptic drugs, vigabatrin, tiagabine, and topiramate. Neurochem Res, 2003, 28(2):333-340.
6. Shields WD, Pellock JM. Vigabatrin 35 years later-from mechanism of action to benefit-risk considerations. Acta Neurol Scand, 2011, 192(Suppl 2):1-4.
7. Aicardi J, Mumford JP, Dumas C, et al. Vigabatrin as initial therapy for infantile spasms:a European retrospective survey. Sabril IS Investigator and Peer Review Groups. Epilepsia, 1996, 37(7):638-642.
8. Schwarz MD, Li M, Tsao J, et al. A lack of clinically apparent vision loss among patients treated with vigabatrin with infantile spasms:The UCLA experience. Epilepsy Behav, 2016, 57(Pt A):29-33.
9. Nabbout RC, Chiron C, Mumford J, et al. Vigabatrin in partial seizures in children. J Child Neurol, 1997, 12(3):172-177.
10. Willmore LJ, Abelson MB, Ben-Menachem E, et al. Vigabatrin:2008 update. Epilepsia, 2009, 50(2):163-173.
11. Riikonen RS. Favourable prognostic factors with infantile spasms. Eur J Paediatr Neurol, 2010, 14(1):13-18.
12. Hancock E, Osborne J, Milner P. Treatment of infantile spasms. Cochrane Database Syst Rev, 2003, (3):Cd001770.
13. Chiron C, Dumas C, Jambaque I, et al. Randomized trial comparing vigabatrin and hydrocortisone in infantile spasms due to tuberous sclerosis. Epilepsy Res, 1997, 26(2):389-395.
14. Yum MS, Lee EH, Ko TS. Vigabatrin and mental retardation in tuberous sclerosis:infantile spasms versus focal seizures. J Child Neurol, 2013, 28(3):308-313.
15. Appleton RE. The role of vigabatrin in the management of infantile epileptic syndromes. Neurology, 1993, 43(11 Suppl 5):21-23.
16. Elterman RD, Shields WD, Bittman RM, et al. Vigabatrin for the treatment of infantile spasms:final report of a randomized trial. J Child Neurol, 2010, 25(11):1340-1347.
17. Gaily E, Jonsson H, Lappi M. Visual fields at school-age in children treated with vigabatrin in infancy. Epilepsia, 2009, 50(2):206-216.
18. Taghdiri MM, Ashrafi MR, Bakhshandeh-Bali MK, et al. Clinical trial of vigabatrin as adjunctive therapy in children with refractory epilepsy. Iran J Pediatr, 2013, 23(6):653-658.
19. 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.

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