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Epigenetic Mechanisms of ART-Related Imprinting Disorders: Lessons From iPSC and Mouse Models.

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cam.issuedOnline2021-10-26
dc.contributor.authorHoránszky, Alex
dc.contributor.authorBecker, Jessica
dc.contributor.authorZana, Melinda
dc.contributor.authorFerguson-Smith, Anne C
dc.contributor.authorDinnyés, András
dc.contributor.orcidBecker, Jessica [0000-0002-8217-1385]
dc.contributor.orcidZana, Melinda [0000-0001-5725-1300]
dc.contributor.orcidDinnyés, András [0000-0003-3791-2583]
dc.date.accessioned2021-10-29T02:13:22Z
dc.date.available2021-10-29T02:13:22Z
dc.date.issued2021-10-26
dc.date.updated2021-10-29T02:13:21Z
dc.description.abstractThe rising frequency of ART-conceived births is accompanied by the need for an improved understanding of the implications of ART on gametes and embryos. Increasing evidence from mouse models and human epidemiological data suggests that ART procedures may play a role in the pathophysiology of certain imprinting disorders (IDs), including Beckwith-Wiedemann syndrome, Silver-Russell syndrome, Prader-Willi syndrome, and Angelman syndrome. The underlying molecular basis of this association, however, requires further elucidation. In this review, we discuss the epigenetic and imprinting alterations of in vivo mouse models and human iPSC models of ART. Mouse models have demonstrated aberrant regulation of imprinted genes involved with ART-related IDs. In the past decade, iPSC technology has provided a platform for patient-specific cellular models of culture-associated perturbed imprinting. However, despite ongoing efforts, a deeper understanding of the susceptibility of iPSCs to epigenetic perturbation is required if they are to be reliably used for modelling ART-associated IDs. Comparing the patterns of susceptibility of imprinted genes in mouse models and IPSCs in culture improves the current understanding of the underlying mechanisms of ART-linked IDs with implications for our understanding of the influence of environmental factors such as culture and hormone treatments on epigenetically important regions of the genome such as imprints.
dc.identifier.doi10.17863/CAM.77471
dc.identifier.eissn2073-4425
dc.identifier.issn2073-4425
dc.identifier.urihttps://www.repository.cam.ac.uk/handle/1810/330027
dc.languageen
dc.publisherMDPI AG
dc.subjectgenomic imprinting
dc.subjectimprinting disorders
dc.subjectassisted reproductive technology
dc.subjectDNA methylation
dc.subjectmouse models
dc.subjectiPSCs
dc.titleEpigenetic Mechanisms of ART-Related Imprinting Disorders: Lessons From iPSC and Mouse Models.
dc.typeArticle
dcterms.dateAccepted2021-10-25
prism.issueIdentifier11
prism.publicationNameGenes (Basel)
prism.volume12
pubs.funder-project-idEuropean Commission Horizon 2020 (H2020) Marie Sk?odowska-Curie actions (812660 - DohART-NET)
rioxxterms.licenseref.urihttps://creativecommons.org/licenses/by/4.0/
rioxxterms.versionVoR
rioxxterms.versionofrecord10.3390/genes12111704

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