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Vigabatrin-associated brain magnetic resonance imaging abnormalities and clinical symptoms in infants with tuberous
Carmen Stevering1, Maarten Lequin2, Kinga Szczepaniak3
1Department of Pediatric Neurology, University Medical Center Utrecht Brain Center, Utrecht, The Netherlands.
Insights
Vigabatrin-associated brain MRI abnormalities (VABAM) are common in infants with tuberous sclerosis complex (TSC). However, these MRI changes in children treated with vigabatrin (VGB) did not correlate with clinical symptoms and resolved after VGB discontinuation.
Area of Science:
- Neurology
- Pediatric Epilepsy
- Neuroimaging
Background:
- Retrospective studies suggest vigabatrin-associated brain abnormalities on MRI (VABAM).
- The clinical significance of VABAM remains unclear.
- Tuberous sclerosis complex (TSC) is a genetic disorder associated with epilepsy and neurological complications.
Purpose of the Study:
- To evaluate the association between vigabatrin (VGB) and specific MRI changes (VABAM) in a large TSC cohort.
- To assess the clinical impact of VABAM in TSC infants.
- To investigate the occurrence of VABAM-like abnormalities in TSC patients not treated with VGB and in non-TSC epilepsy controls.
Main Methods:
- Retrospective and prospective data from 80 TSC patients treated with VGB before age 2 and 23 TSC patients without VGB.
- 29 age-matched non-TSC epilepsy patients not on VGB served as controls.
- VABAM defined as T2/FLAIR hyperintensity or diffusion restriction in specific brain areas; MRI scans analyzed before, during, and after VGB treatment, and once in controls. Symptom evaluation performed concurrently.
Main Results:
- VABAM prevalence was 35.5% in VGB-treated TSC patients.
- VABAM-like abnormalities occurred in 13.5% of non-VGB TSC patients.
- VGB was significantly associated with VABAM (RR=3.57), but TSC and refractory epilepsy were not.
- VABAM resolved entirely after VGB discontinuation in all available cases.
- No significant association was found between VABAM/VABAM-like abnormalities and clinical symptoms (RR=2.76).
Conclusions:
- VABAM are frequent in VGB-treated TSC infants, but similar MRI changes also appear in children without VGB or TSC.
- The etiology of these MRI changes is uncertain, potentially involving myelination, underlying conditions, seizures, or other antiseizure medications.
- VABAM or VABAM-like abnormalities were not associated with clinical symptoms in this cohort.
- The established antiseizure benefits of VGB outweigh the observed risks of VABAM and associated symptoms.
Objective:
Previous retrospective studies have reported vigabatrin-associated brain abnormalities on magnetic resonance imaging (VABAM), although clinical impact is unknown. We evaluated the association between vigabatrin and predefined brain magnetic resonance imaging (MRI) changes in a large homogenous tuberous sclerosis complex (TSC) cohort and assessed to what extent VABAM-related symptoms were reported in TSC infants.
Methods:
The Dutch TSC Registry and the EPISTOP cohort provided retrospective and prospective data from 80 TSC patients treated with vigabatrin (VGB) before the age of 2 years and 23 TSC patients without VGB. Twenty-nine age-matched non-TSC epilepsy patients not receiving VGB were included as controls. VABAM, specified as T2/fluid-attenuated inversion recovery hyperintensity or diffusion restriction in predefined brain areas, were examined on brain MRI before, during, and after VGB, and once in the controls (at approximately age 2 years). Additionally, the presence of VABAM accompanying symptoms was evaluated.
Results:
Prevalence of VABAM in VGB-treated TSC patients was 35.5%. VABAM-like abnormalities were observed in 13.5% of all patients without VGB. VGB was significantly associated with VABAM (risk ratio [RR] = 3.57, 95% confidence interval [CI] = 1.43-6.39), whereas TSC and refractory epilepsy were not. In all 13 VGB-treated patients with VABAM for whom posttreatment MRIs were available, VABAM entirely resolved after VGB discontinuation. The prevalence of symptoms was 11.7% in patients with VABAM or VABAM-like MRI abnormalities and 4.3% in those without, implicating no significant association (RR = 2.76, 95% CI = .68-8.77).
Significance:
VABAM are common in VGB-treated TSC infants; however, VABAM-like abnormalities also occurred in children without either VGB or TSC. The cause of these MRI changes is unknown. Possible contributing factors are abnormal myelination, underlying etiology, recurrent seizures, and other antiseizure medication. Furthermore, the presence of VABAM (or VABAM-like abnormalities) did not appear to be associated with clinical symptoms. This study confirms that the well-known antiseizure effects of VGB outweigh the risk of VABAM and related symptoms.
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