{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T13:23:32Z","timestamp":1791638612823,"version":"4.3.4"},"reference-count":58,"publisher":"Oxford University Press (OUP)","issue":"19","license":[{"start":{"date-parts":[[2020,9,25]],"date-time":"2020-09-25T00:00:00Z","timestamp":1600992000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"DOI":"10.13039\/100002889","name":"Foundation for Prader-Willi Research","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002889","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Connecticut Regenerative Medicine Research Fund"},{"name":"Cascade Fellowship"},{"name":"Levo Therapeutics"},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["HD099975"],"award-info":[{"award-number":["HD099975"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2020,11,25]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Prader-Willi syndrome (PWS) is characterized by neonatal hypotonia, developmental delay and hyperphagia\/obesity. This disorder is caused by the absence of paternally expressed gene products from chromosome 15q11\u2013q13. We previously demonstrated that knocking out ZNF274, a Kruppel-associated box-A-domain zinc finger protein capable of recruiting epigenetic machinery to deposit the H3K9me3 repressive histone modification, can activate expression from the normally silent maternal allele of SNORD116 in neurons derived from PWS induced pluripotent stem cells (iPSCs). However, ZNF274 has many other targets in the genome in addition to SNORD116. Depleting ZNF274 will surely affect the expression of other important genes and disrupt other pathways. Here, we used CRISPR\/Cas9 to delete ZNF274 binding sites at the SNORD116 locus to determine whether activation of the maternal copy of SNORD116 could be achieved without altering ZNF274 protein levels. We obtained similar activation of gene expression from the normally silenced maternal allele in neurons derived from PWS iPSCs, compared with ZNF274 knockout, demonstrating that ZNF274 is directly involved in the repression of SNORD116. These results suggest that interfering with ZNF274 binding at the maternal SNORD116 locus is a potential therapeutic strategy for PWS.<\/jats:p>","DOI":"10.1093\/hmg\/ddaa210","type":"journal-article","created":{"date-parts":[[2020,9,18]],"date-time":"2020-09-18T15:19:15Z","timestamp":1600442355000},"page":"3285-3295","source":"Crossref","is-referenced-by-count":23,"title":["Specific ZNF274 binding interference at\n                    <i>SNORD116<\/i>\n                    activates the maternal transcripts in Prader-Willi syndrome neurons"],"prefix":"10.1093","volume":"29","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7176-0427","authenticated-orcid":false,"given":"Ma\u00e9va","family":"Langou\u00ebt","sequence":"first","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dea","family":"Gorka","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Clarisse","family":"Orniacki","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Cl\u00e9mence M","family":"Dupont-Thibert","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael S","family":"Chung","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heather R","family":"Glatt-Deeley","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Noelle","family":"Germain","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leann J","family":"Crandall","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Justin L","family":"Cotney","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"},{"name":"Institute for Systems Genomics, University of Connecticut, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christopher E","family":"Stoddard","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marc","family":"Lalande","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"},{"name":"Institute for Systems Genomics, University of Connecticut, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stormy J","family":"Chamberlain","sequence":"additional","affiliation":[{"name":"Department of Genetics and Genome Sciences, University of Connecticut School of Medicine, Farmington, CT 06030, USA"},{"name":"Institute for Systems Genomics, University of Connecticut, Farmington, CT 06030, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2020,9,25]]},"reference":[{"key":"2020112602364159900_ref1","doi-asserted-by":"crossref","first-page":"1249","DOI":"10.1007\/s40618-015-0312-9","article-title":"Prader-Willi syndrome: a review of clinical, genetic, and endocrine findings","volume":"38","author":"Angulo","year":"2015","journal-title":"J. Endocrinol. Investig."},{"key":"2020112602364159900_ref2","doi-asserted-by":"crossref","first-page":"10","DOI":"10.1038\/gim.0b013e31822bead0","article-title":"Prader-Willi syndrome","volume":"14","author":"Cassidy","year":"2012","journal-title":"Genet. Med."},{"key":"2020112602364159900_ref3","volume-title":"Human Growth Hormone Treatment for Children with Prader-Willi Syndrome: A Review of Clinical Effectiveness, Cost-Effectiveness, and Guidelines","author":"Edge","year":"2018"},{"key":"2020112602364159900_ref4","doi-asserted-by":"crossref","first-page":"229","DOI":"10.1016\/j.endinu.2018.01.006","article-title":"Treatment with growth hormone in the prader-willi syndrome","volume":"65","author":"Moix Gil","year":"2018","journal-title":"Endocrinol. Diabetes Nutr."},{"key":"2020112602364159900_ref5","first-page":"166","article-title":"Cognitive improvements in children with Prader-Willi syndrome following Pitolisant treatment-patient reports","volume":"24","author":"Pullen","year":"2019","journal-title":"J. Pediatr. Pharmacol. Ther."},{"key":"2020112602364159900_ref6","doi-asserted-by":"crossref","first-page":"344","DOI":"10.1016\/j.omtm.2019.03.001","article-title":"Preclinical testing in translational animal models of Prader-Willi syndrome: overview and gap analysis","volume":"13","author":"Carias","year":"2019","journal-title":"Mol. Ther. Methods Clin. Dev."},{"key":"2020112602364159900_ref7","doi-asserted-by":"crossref","first-page":"529","DOI":"10.1007\/7854_2017_28","article-title":"Oxytocin and Prader-Willi syndrome","volume":"35","author":"Kabasakalian","year":"2018","journal-title":"Curr. Top. Behav. 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