Evidence map›Paper›PMID 42563964›Full record

ArticleAnnals of the Child Neurology Society2024

Longitudinal prospective study of Sturge-Weber syndrome urine angiogenic factors and neurological outcome.

Brooke Kimbrell, Kieran D McKenney, SangEun Yeom, Isabelle Iannotti, Alyssa Day, Kelly Harmon, Alison Sebold, Lindsay Smegal, Katherine Kaplan, Cassie Daisy and 6 more

Abstract read
In one paragraph

Article in Annals of the Child Neurology Society, 2024. 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. Review
  2. Biomarker development in Sturge-Weber syndrome.Journal of neurodevelopmental disorders · 2025
    Review
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

16 authors.

Brooke KimbrellDepartment of Pediatrics Johns Hopkins School of Medicine Baltimore Maryland USA.ORCID 0000-0002-9331-8012
Kieran D McKenneyDepartment of Neurology and Developmental Medicine Hugo Moser Kennedy Krieger Research Institute Baltimore Maryland USA.
SangEun YeomDepartment of Neurology and Developmental Medicine Hugo Moser Kennedy Krieger Research Institute Baltimore Maryland USA.
Isabelle IannottiDepartment of Neurology Boston Children's Hospital and Harvard Medical School Boston Massachusetts USA.
Alyssa DayDepartment of Neurology and Developmental Medicine Hugo Moser Kennedy Krieger Research Institute Baltimore Maryland USA.
Kelly HarmonDepartment of Neurology and Developmental Medicine Hugo Moser Kennedy Krieger Research Institute Baltimore Maryland USA.
Alison SeboldDepartment of Neurology and Developmental Medicine Hugo Moser Kennedy Krieger Research Institute Baltimore Maryland USA.
Lindsay SmegalDepartment of Neurology and Developmental Medicine Hugo Moser Kennedy Krieger Research Institute Baltimore Maryland USA.
Katherine KaplanVascular Biology Program Boston Children's Hospital Boston Massachusetts USA.
Cassie DaisyVascular Biology Program Boston Children's Hospital Boston Massachusetts USA.
Rama AldakhlallahVascular Biology Program Boston Children's Hospital Boston Massachusetts USA.
Michael TaylorDepartment of Pediatrics University of Cincinnati College of Medicine Cincinnati Ohio USA.
Anna PintoDepartment of Neurology Boston Children's Hospital and Harvard Medical School Boston Massachusetts USA.
Adrienne HammillDepartment of Pediatrics University of Cincinnati College of Medicine Cincinnati Ohio USA.
Marsha A MosesVascular Biology Program Boston Children's Hospital Boston Massachusetts USA.
Anne ComiDepartment of Pediatrics Johns Hopkins School of Medicine Baltimore Maryland USA.

Funding

VCRC Administration UnitU54NS065705 · NINDS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI KIM, HELEN · 2009 to 2023
$20.8M
NINDS NIH HHS U54 NS065705
6 · The paper itself

Abstract

Objective: This study identified biomarkers of neurological outcome in Sturge-Weber syndrome (SWS) via urine angiogenic factors and captured longitudinally derived natural history data within an SWS cohort. Methods: This longitudinal, prospective, multicentered study of 61 people with SWS aged 0.4-55 years reports port-wine birthmark score, Neuroscore, Neuro-Quality of Life, and urine angiogenic factors over a two-year period. Results: Cognitive Neuroscore worsened over time for children aged 0-2 years. Male sex was associated with worsening Cognitive Function Neuroscore during the study. Age of seizure onset before 2 years was strongly associated with worse Neuroscore. Children with SWS had low Neuro-Quality of Life related to cognitive function. Seizure severity, male sex, and earlier age of seizure onset were associated with worse Neuro-Quality of Life in school-aged children. Children with SWS have elevated basic fibroblast growth factor in their urine compared with controls, whereas higher vascular endothelial growth factor was associated with better Neuroscore. Interpretation: This study is the first multicenter, prospective, and longitudinal study of people with SWS. It identifies significant clinical prognostic factors such as age of seizure onset and male sex, informs symptom progression over time by age group, and suggests that further study of angiogenic mechanisms and potential biomarkers are needed.

Indexed as

bFGFbiomarkersNeuro‐Quality of LifeNeuroscoreSturge‐Weber syndromeurine angiogenic factors

Identifiers

PMID42563964
PMCPMC13359193

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.