An investigation into the relationship between vigabatrin, movement disorders, and brain magnetic resonance imaging abnormalities in children with infantile spasms

Choon Yi Fong, John P. Osborne, Stuart W. Edwards, Cheryl Hemingway, Eleanor Hancock, Anthony L. Johnson, Colin R. Kennedy, Rachel Kneen, Marcus Likeman, Andrew L. Lux, Santosh R. Mordekar, Velayutham Murugan, Richard W. Newton, Michael Pike, Michael Quinn, Stefan Spinty, Grace Vassallo, Christopher M. Verity, Andrea Whitney, Finbar J. K. O'Callaghan

Research output: Contribution to journalArticle

19 Citations (Scopus)

Abstract

Aim: We aimed to investigate the relationship between movement disorders, changes on brain magnetic resonance imaging (MRI), and vigabatrin therapy in children with infantile spasms.

Method: Retrospective review and brain MRI analysis of children enrolled in the International Collaborative Infantile Spasms Study (ICISS) who developed a movement disorder on vigabatrin therapy. Comparisons were made with controls within ICISS who had no movement disorder.

Results: Ten of 124 infants had a movement disorder and in eight it had developed on vigabatrin therapy. Two had a movement disorder that resolved on dose-reduction of vigabatrin, one had improvement on withdrawing vigabatrin, two had resolution without any dose change, and in three it persisted despite vigabatrin withdrawal. The typical brain MRI changes associated with vigabatrin therapy were noted in two infants. Ten control infants were identified. Typical MRI changes noted with vigabatrin were noted in three controls.

Interpretation: It is possible that in two out of eight cases, vigabatrin was associated with the development of a movement disorder. In six out of eight cases a causal relationship was less plausible. The majority of infants treated with vigabatrin did not develop a movement disorder. MRI changes associated with vigabatrin do not appear to be specifically related to the movement disorder.
Original languageEnglish
Pages (from-to)862-867
Number of pages6
JournalDevelopmental Medicine and Child Neurology
Volume55
Issue number9
Early online date22 Jun 2013
DOIs
Publication statusPublished - Sep 2013

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Vigabatrin
Infantile Spasms
Movement Disorders
Magnetic Resonance Imaging
Brain
Therapeutics

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An investigation into the relationship between vigabatrin, movement disorders, and brain magnetic resonance imaging abnormalities in children with infantile spasms. / Fong, Choon Yi; Osborne, John P.; Edwards, Stuart W.; Hemingway, Cheryl; Hancock, Eleanor; Johnson, Anthony L.; Kennedy, Colin R.; Kneen, Rachel; Likeman, Marcus; Lux, Andrew L.; Mordekar, Santosh R.; Murugan, Velayutham ; Newton, Richard W.; Pike, Michael; Quinn, Michael; Spinty, Stefan; Vassallo, Grace; Verity, Christopher M.; Whitney, Andrea; O'Callaghan, Finbar J. K.

In: Developmental Medicine and Child Neurology, Vol. 55, No. 9, 09.2013, p. 862-867.

Research output: Contribution to journalArticle

Fong, CY, Osborne, JP, Edwards, SW, Hemingway, C, Hancock, E, Johnson, AL, Kennedy, CR, Kneen, R, Likeman, M, Lux, AL, Mordekar, SR, Murugan, V, Newton, RW, Pike, M, Quinn, M, Spinty, S, Vassallo, G, Verity, CM, Whitney, A & O'Callaghan, FJK 2013, 'An investigation into the relationship between vigabatrin, movement disorders, and brain magnetic resonance imaging abnormalities in children with infantile spasms', Developmental Medicine and Child Neurology, vol. 55, no. 9, pp. 862-867. https://doi.org/10.1111/dmcn.12188
Fong, Choon Yi ; Osborne, John P. ; Edwards, Stuart W. ; Hemingway, Cheryl ; Hancock, Eleanor ; Johnson, Anthony L. ; Kennedy, Colin R. ; Kneen, Rachel ; Likeman, Marcus ; Lux, Andrew L. ; Mordekar, Santosh R. ; Murugan, Velayutham ; Newton, Richard W. ; Pike, Michael ; Quinn, Michael ; Spinty, Stefan ; Vassallo, Grace ; Verity, Christopher M. ; Whitney, Andrea ; O'Callaghan, Finbar J. K. / An investigation into the relationship between vigabatrin, movement disorders, and brain magnetic resonance imaging abnormalities in children with infantile spasms. In: Developmental Medicine and Child Neurology. 2013 ; Vol. 55, No. 9. pp. 862-867.
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abstract = "Aim: We aimed to investigate the relationship between movement disorders, changes on brain magnetic resonance imaging (MRI), and vigabatrin therapy in children with infantile spasms. Method: Retrospective review and brain MRI analysis of children enrolled in the International Collaborative Infantile Spasms Study (ICISS) who developed a movement disorder on vigabatrin therapy. Comparisons were made with controls within ICISS who had no movement disorder. Results: Ten of 124 infants had a movement disorder and in eight it had developed on vigabatrin therapy. Two had a movement disorder that resolved on dose-reduction of vigabatrin, one had improvement on withdrawing vigabatrin, two had resolution without any dose change, and in three it persisted despite vigabatrin withdrawal. The typical brain MRI changes associated with vigabatrin therapy were noted in two infants. Ten control infants were identified. Typical MRI changes noted with vigabatrin were noted in three controls. Interpretation: It is possible that in two out of eight cases, vigabatrin was associated with the development of a movement disorder. In six out of eight cases a causal relationship was less plausible. The majority of infants treated with vigabatrin did not develop a movement disorder. MRI changes associated with vigabatrin do not appear to be specifically related to the movement disorder.",
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AU - Fong, Choon Yi

AU - Osborne, John P.

AU - Edwards, Stuart W.

AU - Hemingway, Cheryl

AU - Hancock, Eleanor

AU - Johnson, Anthony L.

AU - Kennedy, Colin R.

AU - Kneen, Rachel

AU - Likeman, Marcus

AU - Lux, Andrew L.

AU - Mordekar, Santosh R.

AU - Murugan, Velayutham

AU - Newton, Richard W.

AU - Pike, Michael

AU - Quinn, Michael

AU - Spinty, Stefan

AU - Vassallo, Grace

AU - Verity, Christopher M.

AU - Whitney, Andrea

AU - O'Callaghan, Finbar J. K.

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N2 - Aim: We aimed to investigate the relationship between movement disorders, changes on brain magnetic resonance imaging (MRI), and vigabatrin therapy in children with infantile spasms. Method: Retrospective review and brain MRI analysis of children enrolled in the International Collaborative Infantile Spasms Study (ICISS) who developed a movement disorder on vigabatrin therapy. Comparisons were made with controls within ICISS who had no movement disorder. Results: Ten of 124 infants had a movement disorder and in eight it had developed on vigabatrin therapy. Two had a movement disorder that resolved on dose-reduction of vigabatrin, one had improvement on withdrawing vigabatrin, two had resolution without any dose change, and in three it persisted despite vigabatrin withdrawal. The typical brain MRI changes associated with vigabatrin therapy were noted in two infants. Ten control infants were identified. Typical MRI changes noted with vigabatrin were noted in three controls. Interpretation: It is possible that in two out of eight cases, vigabatrin was associated with the development of a movement disorder. In six out of eight cases a causal relationship was less plausible. The majority of infants treated with vigabatrin did not develop a movement disorder. MRI changes associated with vigabatrin do not appear to be specifically related to the movement disorder.

AB - Aim: We aimed to investigate the relationship between movement disorders, changes on brain magnetic resonance imaging (MRI), and vigabatrin therapy in children with infantile spasms. Method: Retrospective review and brain MRI analysis of children enrolled in the International Collaborative Infantile Spasms Study (ICISS) who developed a movement disorder on vigabatrin therapy. Comparisons were made with controls within ICISS who had no movement disorder. Results: Ten of 124 infants had a movement disorder and in eight it had developed on vigabatrin therapy. Two had a movement disorder that resolved on dose-reduction of vigabatrin, one had improvement on withdrawing vigabatrin, two had resolution without any dose change, and in three it persisted despite vigabatrin withdrawal. The typical brain MRI changes associated with vigabatrin therapy were noted in two infants. Ten control infants were identified. Typical MRI changes noted with vigabatrin were noted in three controls. Interpretation: It is possible that in two out of eight cases, vigabatrin was associated with the development of a movement disorder. In six out of eight cases a causal relationship was less plausible. The majority of infants treated with vigabatrin did not develop a movement disorder. MRI changes associated with vigabatrin do not appear to be specifically related to the movement disorder.

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