Evidence map›Paper›PMID 39641935›Full record

ArticleEpilepsia2025

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

Carmen Stevering, Maarten Lequin, Kinga Szczepaniak, Krzysztof Sadowski, Saba Ishrat, Alberto De Luca, Alexander Leemans, Willem Otte, David J Kwiatkowski, Paolo Curatolo and 24 more

Abstract read
In one paragraph

Article in Epilepsia, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Article
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

34 authors.

Carmen SteveringDepartment of Pediatric Neurology, University Medical Center Utrecht Brain Center, Utrecht, The Netherlands.ORCID 0000-0002-2294-2344
Maarten LequinDepartment of Radiology, University Medical Center, Utrecht, The Netherlands.ORCID 0000-0001-7488-1734
Kinga SzczepaniakResearch Department, Department of Neurology and Epileptology, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0002-0468-1777
Krzysztof SadowskiResearch Department, Department of Neurology and Epileptology, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0002-0730-2226
Saba IshratImage Sciences Institute, Division Imaging and Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.ORCID 0009-0009-6599-9226
Alberto De LucaImage Sciences Institute, Division Imaging and Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.ORCID 0000-0002-2553-7299
Alexander LeemansImage Sciences Institute, Division Imaging and Oncology, University Medical Center Utrecht, Utrecht, The Netherlands.ORCID 0000-0002-9306-6126
Willem OtteDepartment of Pediatric Neurology, University Medical Center Utrecht Brain Center, Utrecht, The Netherlands.ORCID 0000-0003-1511-6834
David J KwiatkowskiBrigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts, USA.ORCID 0000-0002-5668-5219
Paolo CuratoloChild Neurology and Psychiatry Unit, Systems Medicine Department, Tor Vergata University, Rome, Italy.ORCID 0000-0003-4253-752X
Bernhard WeschkeDepartment of Child Neurology, Charité University Medicine Berlin, Berlin, Germany.ORCID 0000-0003-4533-0743
Kate RineyNeurosciences Unit, Queensland Children's Hospital, South Brisbane, Queensland, Australia.ORCID 0000-0002-1122-3555
Martha FeuchtEpilepsy Center, Department of Pediatrics, Medical University of Vienna, Vienna, Austria.ORCID 0000-0001-7691-8158
Pavel KrsekDepartment of Pediatric Neurology, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic.ORCID 0000-0002-3071-1589
Rima NabboutDepartment of Pediatric Neurology, Reference Center for Rare Epilepsies, Necker-Enfants Malades Hospital, Paris Descartes University, Imagine Institute, Paris, France.ORCID 0000-0001-5877-4074
Anna JansenGenetics Reproduction and Development Research Group, Vrije Universiteit Brussel, Brussels, Belgium.ORCID 0000-0002-3835-2824
Konrad WojdanTransition Technologies Advanced Solutions, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0002-5627-4858
Kamil SijkoTransition Technologies Science, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0002-2203-1065
Jagoda Glowacka-WalasTransition Technologies Science, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0002-7628-8691
Julita BorkowskaResearch Department, Department of Neurology and Epileptology, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0002-9956-9250
Dorota Domanska-PakielaResearch Department, Department of Neurology and Epileptology, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0003-4810-4434
Romina MoaveroChild Neurology and Psychiatry Unit, Systems Medicine Department, Tor Vergata University, Rome, Italy.ORCID 0000-0002-6200-060X
Christoph HertzbergDepartment of Child Neurology, Charité University Medicine Berlin, Berlin, Germany.ORCID 0000-0002-5550-8675
Hanna HulshofDepartment of Pediatric Neurology, University Medical Center Utrecht Brain Center, Utrecht, The Netherlands.ORCID 0000-0003-0122-1174
Theresa SchollEpilepsy Center, Department of Pediatrics, Medical University of Vienna, Vienna, Austria.ORCID 0000-0002-9835-7328
Bořivoj PetrákDepartment of Pediatric Neurology, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic.ORCID 0009-0007-2985-9838
Miroslav MaminakDepartment of Pediatric Neurology, Second Faculty of Medicine, Charles University and Motol University Hospital, Prague, Czech Republic.ORCID 0000-0001-9407-9255
Eleonora AronicaDepartment of (Neuro)Pathology, Amsterdam Neuroscience, Amsterdam University Medical Center, University of Amsterdam, Amsterdam, The Netherlands.ORCID 0000-0002-3542-3770
Jessie De RidderDepartment of Pediatric Neurology, Katholieke Universiteit, Leuven, Belgium.ORCID 0000-0002-8555-7822
Lieven LagaeDepartment of Pediatric Neurology, Katholieke Universiteit, Leuven, Belgium.ORCID 0000-0002-7118-0139
Sergiusz JozwiakResearch Department, Department of Neurology and Epileptology, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0003-3350-6326
Katarzyna KotulskaResearch Department, Department of Neurology and Epileptology, Children's Memorial Health Institute, Warsaw, Poland.ORCID 0000-0002-5015-250X
Kees BraunDepartment of Pediatric Neurology, University Medical Center Utrecht Brain Center, Utrecht, The Netherlands.ORCID 0000-0003-3631-0003
Floor JansenDepartment of Pediatric Neurology, University Medical Center Utrecht Brain Center, Utrecht, The Netherlands.ORCID 0000-0003-0745-5668

Funding

7th Framework Program of the European Commission within the Large-Scale Integrating Project EPISTOP 602391-2Medical Research Agency, Poland, and a statutory S196/2022Polish Ministerial funds for scienceVIRAP 2019/ABM/01/00034
6 · The paper itself

Abstract

objectivePrevious 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.

methodsThe 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.

resultsPrevalence 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.

Indexed as

AnticonvulsantsBrainTuberous SclerosisVigabatrinChild, PreschoolCohort StudiesEpilepsyFemaleHumansInfantMagnetic Resonance ImagingMaleProspective StudiesRetrospective StudiesAnticonvulsantsVigabatrinbrain MRIepilepsytuberous sclerosis complexvigabatrin

Identifiers

PMID39641935
PMCPMC11827727

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