Evidence map›Paper›PMID 29719672›Full record

ArticleMolecular autism2018

Abnormal coherence and sleep composition in children with Angelman syndrome: a retrospective EEG study.

Hanna den Bakker, Michael S Sidorov, Zheng Fan, David J Lee, Lynne M Bird, Catherine J Chu, Benjamin D Philpot

Open access · goldAbstract read
In one paragraph

Article in Molecular autism, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 32 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
32citing papers in PubMed, 1 pooled it
29.8field-weighted citation impact, top 1% 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

32 citing papers in PubMed, 1 synthesis or guideline pooled it, 70 citations in OpenAlex.

  1. Use of Wearable Sensors in Angelman Syndrome: A Systematic Review.Journal of intellectual disability research : JIDR · 2026
    Pooled it
  2. Article
  3. Article
  4. Article
  5. Review
  6. Remote EEG acquisition in Angelman syndrome using PANDABox-EEG.Journal of neurodevelopmental disorders · 2025
    Article
  7. Article
  8. Review
  9. Sleep disturbance in Angelman syndrome patients.Orphanet journal of rare diseases · 2024
    Review
  10. Article
  11. Article
  12. Review
  13. Review
  14. Article
  15. Evaluation of electroencephalography biomarkers for Angelman syndrome during overnight sleep.Autism research : official journal of the International Society for Autism Research · 2022
    Article
  16. Article
  17. Article
  18. Article
  19. 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

7 authors at 3 institutions in 1 country.

Hanna den Bakker *1Department of Cell Biology and Physiology, University of North Carolina, Chapel Hill, NC 27599 USA.
Michael S Sidorov *1Department of Cell Biology and Physiology, University of North Carolina, Chapel Hill, NC 27599 USA.
Zheng Fan4Department of Neurology, University of North Carolina, Chapel Hill, NC 27599 USA.
David J Lee5Department of Neurosciences, University of California, San Diego, CA USA.
Lynne M Bird6Department of Pediatrics, University of California, San Diego, CA USA.
Catherine J Chu8Department of Neurology, Massachusetts General Hospital, Boston, MA 02114 USA.
Benjamin D Philpot1Department of Cell Biology and Physiology, University of North Carolina, Chapel Hill, NC 27599 USA.ORCID 0000-0003-2746-9143
University of North Carolina at Chapel Hill · USUniversity of California San Diego · USBoston Medical Center · US

Funding

Postdoctoral Research in Neurodevelopmental DisordersT32HD040127 · NICHD · UNIV OF NORTH CAROLINA CHAPEL HILL · PI BENJAMIN D PHILPOT, Mark D Shen · 2001 to 2026
$8.5M
Identification of Cortical Biomarkers for Seizure Risk in Childhood EpilepsyK23NS092923 · NINDS · MASSACHUSETTS GENERAL HOSPITAL · PI CHU, CATHERINE J · 2015 to 2020
$1.0M
White Matter Fiber Tract Pathology in a Genetically-Defined Neurodevelopmental DisorderR56NS097831 · NINDS · UNIV OF NORTH CAROLINA CHAPEL HILL · PI HAZLETT, HEATHER CODY, PHILPOT, BENJAMIN D · 2017 to 2017
$544k
NICHD NIH HHS T32 HD040127NINDS NIH HHS K23 NS092923NINDS NIH HHS R56 NS097831
6 · The paper itself

Abstract

Background: Angelman syndrome (AS) is a neurodevelopmental disorder characterized by intellectual disability, speech and motor impairments, epilepsy, abnormal sleep, and phenotypic overlap with autism. Individuals with AS display characteristic EEG patterns including high-amplitude rhythmic delta waves. Here, we sought to quantitatively explore EEG architecture in AS beyond known spectral power phenotypes. We were motivated by studies of functional connectivity and sleep spindles in autism to study these EEG readouts in children with AS. Methods: We analyzed retrospective wake and sleep EEGs from children with AS (age 4-11) and age-matched neurotypical controls. We assessed long-range and short-range functional connectivity by measuring coherence across multiple frequencies during wake and sleep. We quantified sleep spindles using automated and manual approaches. Results: During wakefulness, children with AS showed enhanced long-range EEG coherence across a wide range of frequencies. During sleep, children with AS showed increased long-range EEG coherence specifically in the gamma band. EEGs from children with AS contained fewer sleep spindles, and these spindles were shorter in duration than their neurotypical counterparts. Conclusions: We demonstrate two quantitative readouts of dysregulated sleep composition in children with AS-gamma coherence and spindles-and describe how functional connectivity patterns may be disrupted during wakefulness. Quantitative EEG phenotypes have potential as biomarkers and readouts of target engagement for future clinical trials and provide clues into how neural circuits are dysregulated in children with AS.

Indexed as

Gamma RhythmSleep StagesAngelman SyndromeCase-Control StudiesChildDelta RhythmFemaleHumansMaleAngelman syndromeBiomarkerCoherenceEEGSpindlesUBE3A

Identifiers

PMID29719672
PMCPMC5924514
OpenAlexW2807260013

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.