Vigabatrin-associated brain magnetic resonance imaging abnormalities and clinical symptoms in infants with tuberous sclerosis complex.

• Tác giả: Eleonora Aronica, Julita Borkowska, Kees Braun, Paolo Curatolo, Alberto De Luca, Jessie De Ridder, Dorota Domanska-Pakiela, Martha Feucht, Jagoda Glowacka-Walas, Christoph Hertzberg, Hanna Hulshof, Saba Ishrat, Anna Jansen, Floor Jansen, Sergiusz Jozwiak, Katarzyna Kotulska, Pavel Krsek, David J Kwiatkowski, Lieven Lagae, Alexander Leemans, Maarten Lequin, Miroslav Maminak, Romina Moavero, Rima Nabbout, Willem Otte, Bořivoj Petrák, Kate Riney, Krzysztof Sadowski, Theresa Scholl, Kamil Sijko, Carmen Stevering, Kinga Szczepaniak, Bernhard Weschke, Konrad Wojdan
• Ngôn ngữ: eng
• Ký hiệu phân loại: 297.1248 Sources of Islam
• Thông tin xuất bản: United States : Epilepsia , 2025
• Bộ sưu tập: NCBI
• ID: 60155

Tóm tắt

 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.