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Task D-008 · Type D — Disease → mechanism

Why does Angelman syndrome result specifically from loss of the MATERNAL UBE3A allele rather than either parental copy?

Difficulty
medium
Rarity stratum
medium
Evidence type
primary_research + review
MONDO ID
MONDO:0007113
Curator notes

Requires the specific genomic-imprinting fact that the paternal UBE3A allele is epigenetically silenced by the UBE3A-ATS antisense transcript specifically in neurons — a generic "one copy is lost" answer would miss the actual mechanism this task is testing for.

Generation outcome

Abstained
No
Abstention correct
Yes
Gold in retrieved
1.00
Generation cost
$0.000377
Claims
3
Supported
1
Contradicted
2
Unverifiable
0

Gold documents (2)

  • Ube3a unsilencer for the potential treatment of Angelman syndrome.
    pubmed:38977672retrieved ✓source
  • Angelman syndrome: a journey through the brain.
    pubmed:32087041not retrievedsource

Retrieved documents (6)

  • Precision Medicine in Angelman Syndrome.
    pubmed:39168152
  • Genotype-Phenotype Correlations in Angelman Syndrome.
    pubmed:34203304
  • Angelman Syndrome: Identification and Management.
    pubmed:28494826
  • Angelman Syndrome: Multidisciplinary Management.
    pubmed:40643417
  • Ube3a unsilencer for the potential treatment of Angelman syndrome.
    pubmed:38977672gold
  • Prenatal delivery of a therapeutic antisense oligonucleotide achieves broad biodistribution in the brain and ameliorates Angelman syndrome phenotype in mice.
    pubmed:38327047

Generated claims (3)

supportedconfidence 0.95

Angelman syndrome results from the loss of the maternal UBE3A allele.

pubmed:34203304pubmed:38977672pubmed:38327047pubmed:28494826
contradictedconfidence 0.90

The paternal UBE3A allele is epigenetically silenced in neurons by the UBE3A-ATS transcript.

pubmed:38977672pubmed:38327047
contradictedconfidence 0.90

This epigenetic silencing of the paternal UBE3A allele means that only the maternal copy is expressed in neurons, leading to Angelman syndrome when it is lost.

pubmed:38977672pubmed:38327047