Evidence map›Paper›PMID 34047077›Full record

Trial reportAnnals of clinical and translational neurology2021

Delta power robustly predicts cognitive function in Angelman syndrome.

Lauren M Ostrowski, Elizabeth R Spencer, Lynne M Bird, Ronald Thibert, Robert W Komorowski, Mark A Kramer, Catherine J Chu

Open access · goldAbstract readClinical TrialMulticenter Study
In one paragraph

Trial report in Annals of clinical and translational neurology, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.

0numbers the graph read from it
0cells of the map it votes in
23citing papers in PubMed
5.2field-weighted citation impact, top 4% of its field
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

23 citing papers in PubMed, 44 citations in OpenAlex.

  1. Review
  2. Cannabidiol corrects sleep deficits and reduces spontaneous seizures in Angelman syndrome model mice.Neuropsychopharmacology : official publication of the American College of Neuropsychopharmacology · 2026
    Article
  3. Article
  4. Article
  5. Article
  6. Article
  7. Article
  8. Remote EEG acquisition in Angelman syndrome using PANDABox-EEG.Journal of neurodevelopmental disorders · 2025
    Article
  9. International journal of molecular sciences · 2025
    Article
  10. Article
  11. Nacc1 Mutation in Mice Models Rare Neurodevelopmental Disorder with Underlying Synaptic Dysfunction.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2024
    Article
  12. Review
  13. Article
  14. Article
  15. Article
  16. Review
  17. Article
  18. Evaluation of electroencephalography biomarkers for Angelman syndrome during overnight sleep.Autism research : official journal of the International Society for Autism Research · 2022
    Article
  19. Article
  20. 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

7 authors at 4 institutions in 1 country.

Lauren M OstrowskiDepartment of Neurology, Massachusetts General Hospital, Boston, Massachusetts, USA.
Elizabeth R SpencerDepartment of Neurology, Massachusetts General Hospital, Boston, Massachusetts, USA.
Lynne M BirdDepartment of Pediatrics, University of California, San Diego, California, USA.
Ronald ThibertDepartment of Neurology, Massachusetts General Hospital, Boston, Massachusetts, USA.
Robert W KomorowskiDivision of Clinical Development, Biogen Inc, Cambridge, Massachusetts, USA.
Mark A KramerDepartment of Mathematics and Statistics, Boston University, Boston, Massachusetts, USA.
Catherine J ChuDepartment of Neurology, Massachusetts General Hospital, Boston, Massachusetts, USA.ORCID 0000-0001-7670-9313
Boston University · USHarvard University · USUniversity of California San Diego · USBiogen (United States) · US

Funding

Identification of Cortical Biomarkers for Seizure Risk in Childhood EpilepsyK23NS092923 · NINDS · MASSACHUSETTS GENERAL HOSPITAL · PI CHU, CATHERINE J · 2015 to 2020
$1.0M
NINDS NIH HHS K23 NS092923
6 · The paper itself

Abstract

objectiveAngelman syndrome (AS) is a severe neurodevelopmental disorder caused by loss of function of the maternally inherited UBE3A gene in neurons. Promising disease-modifying treatments to reinstate UBE3A expression are under development and an early measure of treatment response is critical to their deployment in clinical trials. Increased delta power in EEG recordings, reflecting abnormal neuronal synchrony, occurs in AS across species and correlates with genotype. Whether delta power provides a reliable biomarker for clinical symptoms remains unknown.

methodsWe analyzed combined EEG recordings and developmental assessments in a large cohort of individuals with AS (N = 82 subjects, 133 combined EEG and cognitive assessments, 1.08-28.16 years; 32F) and evaluated delta power as a biomarker for cognitive function, as measured by the Bayley Cognitive Score. We examined the robustness of this biomarker to varying states of consciousness, recording techniques and analysis procedures.

resultsDelta power predicted the Bayley Scale cognitive score (P < 10

interpretationDelta power is a simple, direct measure of neuronal activity that reliably correlates with cognitive function in AS. This electrophysiological biomarker offers an objective, clinically relevant endpoint for treatment response in emerging clinical trials.

Indexed as

AdolescentAdultAngelman SyndromeChildChild, PreschoolCognitionDelta RhythmElectroencephalographyFemaleHumansInfantMalePredictive Value of TestsUbiquitin-Protein LigasesYoung AdultUBE3A protein, humanUbiquitin-Protein Ligases

Identifiers

PMID34047077
PMCPMC8283185
OpenAlexW3165105444

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

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.