Evidence map›Paper›PMID 39679858›Full record

ArticleAmerican journal of medical genetics. Part A2025

Community-Sourced Reporting of Mortalities in Angelman Syndrome (1979-2022).

Adriana T Gomes, Amanda Moore, Meagan Cross, Cristy Hardesty, Kelly David, Maria Galan Sampedro, Sophie Dube, Sharon Weil-Chalker, Adela Gentil Montepagano, Ursula Christel and 5 more

Abstract read
In one paragraph

Article in American journal of medical genetics. Part A, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing 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

1 citing paper in PubMed.

  1. Article
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

15 authors.

Adriana T GomesUCSD, San Diego, California, USA.ORCID 0009-0000-3598-460X
Amanda MooreAngelman Syndrome Foundation, Aurora, Illinois, USA.ORCID 0009-0007-6499-8593
Meagan CrossFAST Australia, Salisbury, Australia.ORCID 0000-0002-1281-498X
Cristy HardestyPatient Advocate, New Market, Alabama, USA.ORCID 0009-0006-1958-663X
Kelly DavidFoundation for Angelman Syndrome Therapeutics (FAST), Austin, Texas, USA.ORCID 0009-0001-9057-6198
Maria Galan SampedroFAST Spain, Almería, Almería, Spain.
Sophie DubeAngelman Quebec Foundation, Pierrefonds, Canada.ORCID 0009-0006-6435-6673
Sharon Weil-ChalkerPerinatal Cardiology Consultants, Havertown, Pennsylvania, USA.ORCID 0009-0000-2217-5328
Adela Gentil MontepaganoClínica Angelman de Buenos Aires, San Isidro, Buenos Aires, Argentina.
Ursula ChristelThe Angelman Network, Warkworth, New Zealand.ORCID 0009-0009-4429-0189
Rachel MartinAngelmanUK, Nuneaton, UK.ORCID 0009-0003-6747-6118
Anne WheelerRTI International, Raleigh, North Carolina, USA.ORCID 0000-0003-2693-5616
Wen-Hann TanBoston Children's Hospital, Boston, Massachusetts, USA.ORCID 0000-0002-1593-6149
Lynne M BirdUCSD, Rady Children's Hospital San Diego, San Diego, California, USA.ORCID 0000-0003-4833-3747
Terry Jo BichellCOMBINEDBrain, Brentwood, Tennessee, USA.ORCID 0000-0002-1055-4993

Funding

Training and Career Development ComponentU54HD061222 · NICHD · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI PERCY, ALAN KENNETH · 2009 to 2019
$12.4M
RARE DISEASE CRC FOR NEW THERAPIES AND NEW DIAGNOSTICSU54RR019478 · NCRR · BAYLOR COLLEGE OF MEDICINE · PI BEAUDET, ARTHUR L. · 2003 to 2008
$7.3M
NCRR NIH HHS U54 RR019478NICHD NIH HHS U54 HD061222
6 · The paper itself

Abstract

Angelman syndrome (AS) is a severe genetic neurodevelopmental disorder with an estimated prevalence of 1:20,000. Life expectancy appears to be normal, however, data regarding lifespan in AS are scarce. Until 2018, there was no unique diagnosis code for AS, thus true incidence, prevalence, mortality and morbidity rates are unknown. A social media effort was launched by caregivers of people with AS to gather community-sourced data to understand AS mortality risks. Information on 220 deaths was verified with obituaries and public postings. Respiratory illness was the primary cause of death among people with AS overall, followed by accidents and seizures. Surprisingly, sudden unexpected death in sleep (SUDS) was the fourth most common cause, which has not been reported previously. Approximately 91% of people with AS have epilepsy, thus some SUDS cases may represent sudden unexpected deaths in epilepsy (SUDEP). Causes of death vary by age, and differ from the general population. Though there are obvious limitations to data collected through social media, grass roots science is a starting point to amass preliminary data and inform future epidemiological research on AS.

Indexed as

Angelman SyndromeAdolescentAdultCause of DeathChildChild, PreschoolEpilepsyFemaleHumansInfantMaleMiddle AgedPrevalenceSeizuresSocial MediaSudden Unexpected Death in EpilepsyAngelmancause of deathmortalitySUDEPSUDSUBE3A

Identifiers

PMID39679858
PMCPMC12166961

What OpenQuestion holds

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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.