Vigabatrin as initial therapy for infantile spasms: A European retrospective survey

J. Aicardi; J. P. Mumford; C. Dumas; S. Wood; E. Hauser; H. Steinböck; M. Szyper; V. Holsteen; J. Ostergaard; S. A. Pedersen; K. Taudorf; M. A. Barthez-Carpentier; N. Badinand-Hubert; P. Berquin; J. Boulloche; M. Bourgeois; J. P. Carriere; B. Chabrol; C. Chiron; O. Claris; B. Echenne; D. Gauthier-Morel; M. O. Livet; N. Lopez; J. Mancini; J. C. Netter; D. Quillerou; C. H. Richelme; C. Rousselle; A. De Saint Martin; M. De Swarte; G. Auerswald; U. Brandl; G. Kurlemann; H. Siemes; H. L. Spohr; W. F.M. Aarts; J. H. Begeer; D. J. Heersma; L. A.E.M. Laan; A. C.B. Peters; G. B. Cavazzutti; P. Curatolo; A. Fois; E. Franzoni; G. Gobbi; G. Incorpora; F. Vigevano; J. Campistol; J. Campos; C. Casas; J. L. Herranz; M. Nieto; J. M. Prats; P. Amark; G. Blennow; K. Theorell; B. Tonnby; C. A. Haenggeli; M. Weissert; R. Appleton; R. Robinson; C. De Sousa; N. Thomas; F. Andermann; R. Appleton; S. Boyd; W. Brandi; D. Buti; O. Dulac; K. Farrell; S. Moshe; J. Motte; P. Plouin; W. D. Shields

doi:10.1111/j.1528-1157.1996.tb00627.x

Vigabatrin as initial therapy for infantile spasms: A European retrospective survey

J. Aicardi, J. P. Mumford, C. Dumas, S. Wood, E. Hauser, H. Steinböck, M. Szyper, V. Holsteen, J. Ostergaard, S. A. Pedersen, K. Taudorf, M. A. Barthez-Carpentier, N. Badinand-Hubert, P. Berquin, J. Boulloche, M. Bourgeois, J. P. Carriere, B. Chabrol, C. Chiron, O. ClarisB. Echenne, D. Gauthier-Morel, M. O. Livet, N. Lopez, J. Mancini, J. C. Netter, D. Quillerou, C. H. Richelme, C. Rousselle, A. De Saint Martin, M. De Swarte, G. Auerswald, U. Brandl, G. Kurlemann, H. Siemes, H. L. Spohr, W. F.M. Aarts, J. H. Begeer, D. J. Heersma, L. A.E.M. Laan, A. C.B. Peters, G. B. Cavazzutti, P. Curatolo, A. Fois, E. Franzoni, G. Gobbi, G. Incorpora, F. Vigevano, J. Campistol, J. Campos, C. Casas, J. L. Herranz, M. Nieto, J. M. Prats, P. Amark, G. Blennow, K. Theorell, B. Tonnby, C. A. Haenggeli, M. Weissert, R. Appleton, R. Robinson, C. De Sousa, N. Thomas, F. Andermann, R. Appleton, S. Boyd, W. Brandi, D. Buti, O. Dulac, K. Farrell, S. Moshe, J. Motte, P. Plouin, W. D. Shields

Research output: Contribution to journal › Article › peer-review

180 Scopus citations

Abstract

Purpose: The efficacy and tolerability of vigabatrin (VGB) as an add-on therapy in the treatment of infantile spasm (IS) prompted physicians to explore its use as the first drug in this seizure type. Methods: Our retrospective study included 250 infants diagnosed with IS: the data obtained were subjected to peer-group review. Of this infant population, 192 infants were considered to have classic IS and had received VGB as their first treatment for the spasms. There was a slight preponderance of boys (57%) in this population. Mean age of IS onset was 5.8 months; 60% had typical hypsarrhythmia. Results: Initial suppression of spasms was obtained in 68% of infants with a median time to response of 4 days at an average VGB dose of 99 mg/kg/day. The best response was seen in those infants with tuberous sclerosis(96% response) and in those younger than 3 months at onset of spasms (90% response). Of these infants, 43 (22%) of 192 subsequently had other types of seizures, and a recurrence of infantile spasms occurred in 28 (21%) of 131 responders. At the end of this study, 96 of 192 infants who could be evaluated were seizure free with VGB monotherapy. Treatment appeared to be well tolerated, with only 33 (13%) infants with adverse events, of which the most common were somnolence (15 patients) and hyperkinesia (eight patients). In only two cases did adverse events require VGB withdrawal. Conclusion: This study supports the opinion that VGB may be considered an initial treatment for IS regardless of cause.

Original language	English (US)
Pages (from-to)	638-642
Number of pages	5
Journal	Epilepsia
Volume	37
Issue number	7
DOIs	https://doi.org/10.1111/j.1528-1157.1996.tb00627.x
State	Published - 1996
Externally published	Yes

Keywords

Infantile spasms
Monotherapy
Retrospective study
Vigabatrin

ASJC Scopus subject areas

Neurology
Clinical Neurology

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Aicardi, J., Mumford, J. P., Dumas, C., Wood, S., Hauser, E., Steinböck, H., Szyper, M., Holsteen, V., Ostergaard, J., Pedersen, S. A., Taudorf, K., Barthez-Carpentier, M. A., Badinand-Hubert, N., Berquin, P., Boulloche, J., Bourgeois, M., Carriere, J. P., Chabrol, B., Chiron, C., ... Shields, W. D. (1996). Vigabatrin as initial therapy for infantile spasms: A European retrospective survey. Epilepsia, 37(7), 638-642. https://doi.org/10.1111/j.1528-1157.1996.tb00627.x

Vigabatrin as initial therapy for infantile spasms : A European retrospective survey. / Aicardi, J.; Mumford, J. P.; Dumas, C.; Wood, S.; Hauser, E.; Steinböck, H.; Szyper, M.; Holsteen, V.; Ostergaard, J.; Pedersen, S. A.; Taudorf, K.; Barthez-Carpentier, M. A.; Badinand-Hubert, N.; Berquin, P.; Boulloche, J.; Bourgeois, M.; Carriere, J. P.; Chabrol, B.; Chiron, C.; Claris, O.; Echenne, B.; Gauthier-Morel, D.; Livet, M. O.; Lopez, N.; Mancini, J.; Netter, J. C.; Quillerou, D.; Richelme, C. H.; Rousselle, C.; De Saint Martin, A.; De Swarte, M.; Auerswald, G.; Brandl, U.; Kurlemann, G.; Siemes, H.; Spohr, H. L.; Aarts, W. F.M.; Begeer, J. H.; Heersma, D. J.; Laan, L. A.E.M.; Peters, A. C.B.; Cavazzutti, G. B.; Curatolo, P.; Fois, A.; Franzoni, E.; Gobbi, G.; Incorpora, G.; Vigevano, F.; Campistol, J.; Campos, J.; Casas, C.; Herranz, J. L.; Nieto, M.; Prats, J. M.; Amark, P.; Blennow, G.; Theorell, K.; Tonnby, B.; Haenggeli, C. A.; Weissert, M.; Appleton, R.; Robinson, R.; De Sousa, C.; Thomas, N.; Andermann, F.; Appleton, R.; Boyd, S.; Brandi, W.; Buti, D.; Dulac, O.; Farrell, K.; Moshe, S.; Motte, J.; Plouin, P.; Shields, W. D.

In: Epilepsia, Vol. 37, No. 7, 1996, p. 638-642.

Research output: Contribution to journal › Article › peer-review

Aicardi, J, Mumford, JP, Dumas, C, Wood, S, Hauser, E, Steinböck, H, Szyper, M, Holsteen, V, Ostergaard, J, Pedersen, SA, Taudorf, K, Barthez-Carpentier, MA, Badinand-Hubert, N, Berquin, P, Boulloche, J, Bourgeois, M, Carriere, JP, Chabrol, B, Chiron, C, Claris, O, Echenne, B, Gauthier-Morel, D, Livet, MO, Lopez, N, Mancini, J, Netter, JC, Quillerou, D, Richelme, CH, Rousselle, C, De Saint Martin, A, De Swarte, M, Auerswald, G, Brandl, U, Kurlemann, G, Siemes, H, Spohr, HL, Aarts, WFM, Begeer, JH, Heersma, DJ, Laan, LAEM, Peters, ACB, Cavazzutti, GB, Curatolo, P, Fois, A, Franzoni, E, Gobbi, G, Incorpora, G, Vigevano, F, Campistol, J, Campos, J, Casas, C, Herranz, JL, Nieto, M, Prats, JM, Amark, P, Blennow, G, Theorell, K, Tonnby, B, Haenggeli, CA, Weissert, M, Appleton, R, Robinson, R, De Sousa, C, Thomas, N, Andermann, F, Appleton, R, Boyd, S, Brandi, W, Buti, D, Dulac, O, Farrell, K, Moshe, S, Motte, J, Plouin, P & Shields, WD 1996, 'Vigabatrin as initial therapy for infantile spasms: A European retrospective survey', Epilepsia, vol. 37, no. 7, pp. 638-642. https://doi.org/10.1111/j.1528-1157.1996.tb00627.x

Aicardi, J. ; Mumford, J. P. ; Dumas, C. ; Wood, S. ; Hauser, E. ; Steinböck, H. ; Szyper, M. ; Holsteen, V. ; Ostergaard, J. ; Pedersen, S. A. ; Taudorf, K. ; Barthez-Carpentier, M. A. ; Badinand-Hubert, N. ; Berquin, P. ; Boulloche, J. ; Bourgeois, M. ; Carriere, J. P. ; Chabrol, B. ; Chiron, C. ; Claris, O. ; Echenne, B. ; Gauthier-Morel, D. ; Livet, M. O. ; Lopez, N. ; Mancini, J. ; Netter, J. C. ; Quillerou, D. ; Richelme, C. H. ; Rousselle, C. ; De Saint Martin, A. ; De Swarte, M. ; Auerswald, G. ; Brandl, U. ; Kurlemann, G. ; Siemes, H. ; Spohr, H. L. ; Aarts, W. F.M. ; Begeer, J. H. ; Heersma, D. J. ; Laan, L. A.E.M. ; Peters, A. C.B. ; Cavazzutti, G. B. ; Curatolo, P. ; Fois, A. ; Franzoni, E. ; Gobbi, G. ; Incorpora, G. ; Vigevano, F. ; Campistol, J. ; Campos, J. ; Casas, C. ; Herranz, J. L. ; Nieto, M. ; Prats, J. M. ; Amark, P. ; Blennow, G. ; Theorell, K. ; Tonnby, B. ; Haenggeli, C. A. ; Weissert, M. ; Appleton, R. ; Robinson, R. ; De Sousa, C. ; Thomas, N. ; Andermann, F. ; Appleton, R. ; Boyd, S. ; Brandi, W. ; Buti, D. ; Dulac, O. ; Farrell, K. ; Moshe, S. ; Motte, J. ; Plouin, P. ; Shields, W. D. / Vigabatrin as initial therapy for infantile spasms : A European retrospective survey. In: Epilepsia. 1996 ; Vol. 37, No. 7. pp. 638-642.

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title = "Vigabatrin as initial therapy for infantile spasms: A European retrospective survey",

abstract = "Purpose: The efficacy and tolerability of vigabatrin (VGB) as an add-on therapy in the treatment of infantile spasm (IS) prompted physicians to explore its use as the first drug in this seizure type. Methods: Our retrospective study included 250 infants diagnosed with IS: the data obtained were subjected to peer-group review. Of this infant population, 192 infants were considered to have classic IS and had received VGB as their first treatment for the spasms. There was a slight preponderance of boys (57%) in this population. Mean age of IS onset was 5.8 months; 60% had typical hypsarrhythmia. Results: Initial suppression of spasms was obtained in 68% of infants with a median time to response of 4 days at an average VGB dose of 99 mg/kg/day. The best response was seen in those infants with tuberous sclerosis(96% response) and in those younger than 3 months at onset of spasms (90% response). Of these infants, 43 (22%) of 192 subsequently had other types of seizures, and a recurrence of infantile spasms occurred in 28 (21%) of 131 responders. At the end of this study, 96 of 192 infants who could be evaluated were seizure free with VGB monotherapy. Treatment appeared to be well tolerated, with only 33 (13%) infants with adverse events, of which the most common were somnolence (15 patients) and hyperkinesia (eight patients). In only two cases did adverse events require VGB withdrawal. Conclusion: This study supports the opinion that VGB may be considered an initial treatment for IS regardless of cause.",

keywords = "Infantile spasms, Monotherapy, Retrospective study, Vigabatrin",

author = "J. Aicardi and Mumford, {J. P.} and C. Dumas and S. Wood and E. Hauser and H. Steinb{\"o}ck and M. Szyper and V. Holsteen and J. Ostergaard and Pedersen, {S. A.} and K. Taudorf and Barthez-Carpentier, {M. A.} and N. Badinand-Hubert and P. Berquin and J. Boulloche and M. Bourgeois and Carriere, {J. P.} and B. Chabrol and C. Chiron and O. Claris and B. Echenne and D. Gauthier-Morel and Livet, {M. O.} and N. Lopez and J. Mancini and Netter, {J. C.} and D. Quillerou and Richelme, {C. H.} and C. Rousselle and {De Saint Martin}, A. and {De Swarte}, M. and G. Auerswald and U. Brandl and G. Kurlemann and H. Siemes and Spohr, {H. L.} and Aarts, {W. F.M.} and Begeer, {J. H.} and Heersma, {D. J.} and Laan, {L. A.E.M.} and Peters, {A. C.B.} and Cavazzutti, {G. B.} and P. Curatolo and A. Fois and E. Franzoni and G. Gobbi and G. Incorpora and F. Vigevano and J. Campistol and J. Campos and C. Casas and Herranz, {J. L.} and M. Nieto and Prats, {J. M.} and P. Amark and G. Blennow and K. Theorell and B. Tonnby and Haenggeli, {C. A.} and M. Weissert and R. Appleton and R. Robinson and {De Sousa}, C. and N. Thomas and F. Andermann and R. Appleton and S. Boyd and W. Brandi and D. Buti and O. Dulac and K. Farrell and S. Moshe and J. Motte and P. Plouin and Shields, {W. D.}",

year = "1996",

doi = "10.1111/j.1528-1157.1996.tb00627.x",

language = "English (US)",

volume = "37",

pages = "638--642",

journal = "Epilepsia",

issn = "0013-9580",

publisher = "Wiley-Blackwell",

number = "7",

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TY - JOUR

T1 - Vigabatrin as initial therapy for infantile spasms

T2 - A European retrospective survey

AU - Aicardi, J.

AU - Mumford, J. P.

AU - Dumas, C.

AU - Wood, S.

AU - Hauser, E.

AU - Steinböck, H.

AU - Szyper, M.

AU - Holsteen, V.

AU - Ostergaard, J.

AU - Pedersen, S. A.

AU - Taudorf, K.

AU - Barthez-Carpentier, M. A.

AU - Badinand-Hubert, N.

AU - Berquin, P.

AU - Boulloche, J.

AU - Bourgeois, M.

AU - Carriere, J. P.

AU - Chabrol, B.

AU - Chiron, C.

AU - Claris, O.

AU - Echenne, B.

AU - Gauthier-Morel, D.

AU - Livet, M. O.

AU - Lopez, N.

AU - Mancini, J.

AU - Netter, J. C.

AU - Quillerou, D.

AU - Richelme, C. H.

AU - Rousselle, C.

AU - De Saint Martin, A.

AU - De Swarte, M.

AU - Auerswald, G.

AU - Brandl, U.

AU - Kurlemann, G.

AU - Siemes, H.

AU - Spohr, H. L.

AU - Aarts, W. F.M.

AU - Begeer, J. H.

AU - Heersma, D. J.

AU - Laan, L. A.E.M.

AU - Peters, A. C.B.

AU - Cavazzutti, G. B.

AU - Curatolo, P.

AU - Fois, A.

AU - Franzoni, E.

AU - Gobbi, G.

AU - Incorpora, G.

AU - Vigevano, F.

AU - Campistol, J.

AU - Campos, J.

AU - Casas, C.

AU - Herranz, J. L.

AU - Nieto, M.

AU - Prats, J. M.

AU - Amark, P.

AU - Blennow, G.

AU - Theorell, K.

AU - Tonnby, B.

AU - Haenggeli, C. A.

AU - Weissert, M.

AU - Appleton, R.

AU - Robinson, R.

AU - De Sousa, C.

AU - Thomas, N.

AU - Andermann, F.

AU - Appleton, R.

AU - Boyd, S.

AU - Brandi, W.

AU - Buti, D.

AU - Dulac, O.

AU - Farrell, K.

AU - Moshe, S.

AU - Motte, J.

AU - Plouin, P.

AU - Shields, W. D.

PY - 1996

Y1 - 1996

N2 - Purpose: The efficacy and tolerability of vigabatrin (VGB) as an add-on therapy in the treatment of infantile spasm (IS) prompted physicians to explore its use as the first drug in this seizure type. Methods: Our retrospective study included 250 infants diagnosed with IS: the data obtained were subjected to peer-group review. Of this infant population, 192 infants were considered to have classic IS and had received VGB as their first treatment for the spasms. There was a slight preponderance of boys (57%) in this population. Mean age of IS onset was 5.8 months; 60% had typical hypsarrhythmia. Results: Initial suppression of spasms was obtained in 68% of infants with a median time to response of 4 days at an average VGB dose of 99 mg/kg/day. The best response was seen in those infants with tuberous sclerosis(96% response) and in those younger than 3 months at onset of spasms (90% response). Of these infants, 43 (22%) of 192 subsequently had other types of seizures, and a recurrence of infantile spasms occurred in 28 (21%) of 131 responders. At the end of this study, 96 of 192 infants who could be evaluated were seizure free with VGB monotherapy. Treatment appeared to be well tolerated, with only 33 (13%) infants with adverse events, of which the most common were somnolence (15 patients) and hyperkinesia (eight patients). In only two cases did adverse events require VGB withdrawal. Conclusion: This study supports the opinion that VGB may be considered an initial treatment for IS regardless of cause.

AB - Purpose: The efficacy and tolerability of vigabatrin (VGB) as an add-on therapy in the treatment of infantile spasm (IS) prompted physicians to explore its use as the first drug in this seizure type. Methods: Our retrospective study included 250 infants diagnosed with IS: the data obtained were subjected to peer-group review. Of this infant population, 192 infants were considered to have classic IS and had received VGB as their first treatment for the spasms. There was a slight preponderance of boys (57%) in this population. Mean age of IS onset was 5.8 months; 60% had typical hypsarrhythmia. Results: Initial suppression of spasms was obtained in 68% of infants with a median time to response of 4 days at an average VGB dose of 99 mg/kg/day. The best response was seen in those infants with tuberous sclerosis(96% response) and in those younger than 3 months at onset of spasms (90% response). Of these infants, 43 (22%) of 192 subsequently had other types of seizures, and a recurrence of infantile spasms occurred in 28 (21%) of 131 responders. At the end of this study, 96 of 192 infants who could be evaluated were seizure free with VGB monotherapy. Treatment appeared to be well tolerated, with only 33 (13%) infants with adverse events, of which the most common were somnolence (15 patients) and hyperkinesia (eight patients). In only two cases did adverse events require VGB withdrawal. Conclusion: This study supports the opinion that VGB may be considered an initial treatment for IS regardless of cause.

KW - Infantile spasms

KW - Monotherapy

KW - Retrospective study

KW - Vigabatrin

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