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Treatment with High-Dose Prednisolone in Vigabatrin-Refractory Infantile Spasms
Wafaa Al-Shehhi1, Vann Chau1, Jennifer Boyd1
1Epilepsy Program, Division of Neurology, Department of Pediatrics, Hospital for Sick Children, Toronto, ON, Canada.
Insights
High-dose prednisolone effectively treated infantile spasms (IS) in children resistant to vigabatrin. This treatment achieved sustained electroclinical remission in two-thirds of patients and was well-tolerated.
Area of Science:
- Pediatric Neurology
- Clinical Pharmacology
Background:
- Infantile spasms (IS) are a severe epilepsy syndrome in infants.
- Vigabatrin is a first-line anti-seizure medication (ASM) for IS, but a significant proportion of patients are refractory.
- Alternative treatments are needed for IS refractory to vigabatrin.
Purpose of the Study:
- To evaluate the efficacy and safety of high-dose oral prednisolone in children with IS refractory to vigabatrin.
- To compare the electroclinical remission rates of prednisolone with published data for adrenocorticotropic hormone (ACTH) therapy.
Main Methods:
- A consecutive series of children with IS and hypsarrhythmia (or variant) on EEG, refractory to vigabatrin, were treated with oral prednisolone (8 mg/kg/day).
- Electroclinical response (ECR) was assessed at 2 and 6 weeks post-treatment initiation.
- Adverse effects were monitored.
Main Results:
- Of 65 children included, 30.8% achieved complete ECR after vigabatrin.
- In 45 children refractory to vigabatrin, prednisolone led to complete ECR in 77.8% at 2 weeks, sustained in 66.7% at 6 weeks.
- Prednisolone was generally well tolerated.
Conclusions:
- High-dose oral prednisolone is an effective and safe treatment for infantile spasms refractory to vigabatrin.
- Sustained electroclinical remission was achieved in two-thirds of patients treated with prednisolone.
- Prednisolone offers a viable alternative for managing refractory IS.
Objectives:
This research aimed to study the short-term seizure outcomes following treatment with 8 mg/kg/day prednisolone in children with infantile spasms (IS) refractory to vigabatrin. We hypothesized that high-dose prednisolone may result in similar rates of electroclinical remission when compared to published ACTH rates.
Methods:
All consecutive children with hypsarrhythmia or hypsarrhythmia variant on EEG with/without IS, who had been treated with vigabatrin as first-line anti-seizure medication (ASM) followed by high-dose oral prednisolone (8 mg/kg/day; maximum 60 mg/day) in cases who did not respond to vigabatrin, were included. Clinical and electroclinical response (ECR) at 2 weeks following initiation of treatment and adverse effects were assessed.
Results:
Sixty-five children were included. A genetic etiology was seen in 38.5% cases. Complete ECR was seen in 30.8% (20/65) of the patients 2 weeks after vigabatrin. Complete ECR was noted in 77.8% (35/45) of the patients, 2 weeks after prednisolone initiation in children who failed vigabatrin, and this was sustained at 6 weeks in 66.7% (30/45) patients. Prednisolone was generally well tolerated.
Conclusions:
High-dose (8 mg/kg/day) oral prednisolone resulted in sustained complete ECR (at 6 weeks) in two-thirds of the children with hypsarrhythmia or hypsarrhythmia variant on EEG with/without parentally reported IS. It was generally well tolerated and found to be safe.
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