# Brain MRI to pre-symptomatically predict seizure onset for Sturge-Weber Syndrome

> **NIH NIH R21** · BOSTON CHILDREN'S HOSPITAL · 2022 · $271,133

## Abstract

Abstract
Sturge-Weber syndrome (SWS) is a rare neurological disease, and its biggest concern is neurocognitive
impairments by school age (6-10 years). To improve neurocognitive outcomes, Kenndy Krieger Institute (Dr.
Comi, co-PI of this R21) and Boston Children’s Hospital (Dr. Pinto, co-PI of this R21) have been the leading or
key sites in various clinical trials, to test new treatment (NCT02332655 (2014-19); NCT0304980 (2017-19);
NCT04447846 (2019-21)), or to develop neuroimaging biomarkers that can select at-risk patients
(NCT01345305 (2010-2012); NCT01425944 (2010-2020); NCT04717427 (2021-2024)). However, all these trials
focus on the post-symptomatic phase – after seizure symptoms have occurred. Our recent evidence suggested
that pre-symptomatic treatment – treating patients before seizure symptoms occur, ideally before 2 years of
age – may delay or avoid seizure symptoms. This is important, because those without seizure symptoms by 2
years of age (10-25% of SWS patients) often enjoy good neurocognitive outcomes by school age. Motivated by
this, multidisciplinary experts gathered and reached a consensus in 2018, 2019, and 2021, calling for immediate
investigations of pre-symptomatic treatments. In a timely response to this call, KKI and BCH, the two largest
national centers that treat SWS pre-symptomatically, are planning for a trial to comprehensively evaluate the
effect of anti-epilepsy drugs Levetiracetam, in two treatment arms (Levetiracetam with versus without low-dose
aspirin) for pre-symptomatic treatment. The bottleneck issue for this planned trial, though, is the lack of a
biomarker to accurately and pre-symptomatically identify SWS patients who are at risk to develop seizure
symptoms by 2 years of age. Those at-risk patients should be ideal candidates to be included in our planned
trial. This R21 aims to address this bottleneck biomarker problem. We plan to retrospectively build the largest
multi-site presymptomatic database from clinical data in KKI and BCH (Aim 1). We will thoroughly evaluate two
clinical and brain MRI biomarkers to identify at-risk patients pre-symptomatically (Aim 2). The central
hypothesis is that sophisticated features that artificial intelligence (AI) algorithms extract from clinical and brain
MRI could serve as a biomarker to pre-symptomatically identify SWS patients at risk of developing seizure
symptoms by 2 years of age. This is the first AI-powered, large-dataset-driven rigorous study for presymptomatic
clinical and MRI biomarkers for this rare disease. Such a biomarker will be immediately used in our planned
clinical trials to evaluate Levetiracetam with or without low-dose aspirin for pre-symptomatic treatment.

## Key facts

- **NIH application ID:** 10423733
- **Project number:** 1R21TR004265-01
- **Recipient organization:** BOSTON CHILDREN'S HOSPITAL
- **Principal Investigator:** ANNE M COMI
- **Activity code:** R21 (R01, R21, SBIR, etc.)
- **Funding institute:** NIH
- **Fiscal year:** 2022
- **Award amount:** $271,133
- **Award type:** 1
- **Project period:** 2022-08-15 → 2024-07-31

## Primary source

NIH RePORTER: https://reporter.nih.gov/project-details/10423733

## Citation

> US National Institutes of Health, RePORTER application 10423733, Brain MRI to pre-symptomatically predict seizure onset for Sturge-Weber Syndrome (1R21TR004265-01). Retrieved via AI Analytics 2026-05-23 from https://api.ai-analytics.org/grant/nih/10423733. Licensed CC0.

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