Forskning
Udskriv Udskriv
Switch language
Rigshospitalet - en del af Københavns Universitetshospital
Udgivet

Imprinting disorders in children born after ART: a Nordic study from the CoNARTaS group

Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

DOI

  1. Proteome of fluid from human ovarian small antral follicles reveals insights in folliculogenesis and oocyte maturation

    Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

  2. N-acetylcysteine protects ovarian follicles from ischemia-reperfusion injury in xenotransplanted human ovarian tissue

    Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

  3. SARS-CoV-2 in first trimester pregnancy: a cohort study

    Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

  4. Familial resemblance in markers of testicular function in fathers and their young sons: a cross-sectional study

    Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

  1. Pregnancy outcomes following oocyte donation

    Publikation: Bidrag til tidsskriftReviewForskningpeer review

  2. Are the Danish stillbirth rates still record low? A nationwide ecological study

    Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

  3. Natarbejde og komplikationer i graviditeten

    Publikation: Bidrag til tidsskriftReviewForskningpeer review

  4. I denne sælsomme juletid.....

    Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

Vis graf over relationer

STUDY QUESTION: Is the risk of imprinting disorders increased in children conceived after ART?

SUMMARY ANSWER: We found an adjusted odds ratio (AOR) of 2.84 [95% CI: 1.34-6.01] for Beckwith-Wiedemann syndrome in ART children, while the risk of Prader-Willi syndrome, Silver-Russell syndrome or Angelman syndrome was not increased in children conceived after ART.

WHAT IS KNOWN ALREADY: Earlier studies, most of them small, have suggested an association between ART and imprinting disorders.

STUDY DESIGN, SIZE, DURATION: This was a binational register-based cohort study. All children conceived by ART in Denmark (n = 45 393, born between 1994 and 2014) and in Finland (n = 29 244, born between 1990 and 2014) were identified. The full background populations born during the same time periods in the two countries were included as controls. Odds ratios of imprinting disorders in ART children compared with naturally conceived (NC) children were calculated. The median follow-up time was 8 years and 9 months for ART children and 11 years and 9 months for NC children.

PARTICIPANTS/MATERIALS, SETTING, METHODS: From the national health registries in Denmark and Finland, we identified all children diagnosed with Prader-Willi syndrome (n = 143), Silver-Russell syndrome (n = 69), Beckwith-Wiedemann syndrome (n = 105) and Angelman syndrome (n = 72) born between 1994/1990 and 2014, respectively.

MAIN RESULTS AND THE ROLE OF CHANCE: We identified a total of 388 children diagnosed with imprinting disorders; 16 of these were conceived after ART. The overall AOR for the four imprinting disorders in ART children compared with NC children was 1.35 [95% CI: 0.80-2.29], but since eight ART children were diagnosed with Beckwith-Wiedemann syndrome, the AOR for this specific imprinting disorder was 2.84 [95% CI: 1.34-6.01]. The absolute risk of Beckwith-Wiedemann syndrome in children conceived after ART was still low: 10.7 out of 100 000 newborns. The risks of Prader-Willi syndrome, Silver-Russell syndrome and Angelman syndrome were not increased in children conceived after ART.

LIMITATIONS, REASONS FOR CAUTION: Imprinting disorders are rare events and our results are based on few ART children with imprinting disorders. The aetiology is complex and only partly clarified, and the clinical diagnoses are challenged by a broad phenotypic spectrum.

WIDER IMPLICATIONS OF THE FINDINGS: In the existing studies, results on the risk of imprinting disorders in children conceived after ART are ambiguous. This study adds that the risk of imprinting disorders in ART children is very small and perhaps restricted to Beckwith-Wiedemann syndrome.

STUDY FUNDING/COMPETING INTEREST(S): This work was supported by the Nordic Trial Alliance: a pilot project jointly funded by the Nordic Council of Ministers and NordForsk (grant number: 71450), the Nordic Federation of Obstetrics and Gynecology (grant numbers: NF13041, NF15058, NF16026 and NF17043) and the Interreg Öresund-Kattegat-Skagerak European Regional Development Fund (ReproUnion project). The authors have no conflicts of interest related to this work.

TRIAL REGISTRATION NUMBER: N/A.

OriginalsprogEngelsk
TidsskriftHuman reproduction (Oxford, England)
Vol/bind35
Udgave nummer5
Sider (fra-til)1178-1184
Antal sider7
ISSN0268-1161
DOI
StatusUdgivet - 1 maj 2020

Bibliografisk note

© The Author(s) 2020. Published by Oxford University Press on behalf of the European Society of Human Reproduction and Embryology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.

ID: 61423435