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We analyzed chromatin-associated RNA in human induced pluripotent cells (iPSCs) upon depletion of hybrid small nucleolar long non-coding RNAs (sno-lncRNAs) and 5\u2019 snoRNA capped and polyadenylated long non-coding RNAs (SPA-lncRNAs) transcribed from the locus deleted in PWS. We found that rapid ablation of these lncRNAs affects transcription of specific gene classes. Downregulated genes contribute to neurodevelopment and neuronal maintenance, while upregulated genes are predominantly involved in the negative regulation of cellular metabolism and apoptotic processes. Our data reveal the importance of SPA-lncRNAs and sno-lncRNAs in controlling gene expression in iPSCs and provide a platform for synthetic experimental approaches in PWS studies. We conclude that ncRNAs transcribed from the PWS locus are critical regulators of a transcriptional signature, which is important for neuronal differentiation and development.<\/jats:p>","DOI":"10.1093\/hmg\/ddac228","type":"journal-article","created":{"date-parts":[[2022,9,8]],"date-time":"2022-09-08T09:06:32Z","timestamp":1662627992000},"page":"608-620","source":"Crossref","is-referenced-by-count":21,"title":["Non-coding RNAs associated with Prader\u2013Willi syndrome regulate transcription of neurodevelopmental genes in human induced pluripotent stem cells"],"prefix":"10.1093","volume":"32","author":[{"given":"Monika","family":"Sledziowska","sequence":"first","affiliation":[{"name":"School of Biosciences, University of Birmingham , Edgbaston, Birmingham B15 2TT , UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kinga","family":"Winczura","sequence":"additional","affiliation":[{"name":"School of Biological Sciences, University of Manchester, Michael Smith Building , Oxford Road, Manchester M13 9PT , UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Matt","family":"Jones","sequence":"additional","affiliation":[{"name":"School of Life Sciences, Gibbet Hill Campus, University of Warwick , Coventry CV4 7AL , UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ruba","family":"Almaghrabi","sequence":"additional","affiliation":[{"name":"Institute for Cancer and Genomic Sciences, University of Birmingham , Edgbaston, Birmingham B15 2TT , UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hannah","family":"Mischo","sequence":"additional","affiliation":[{"name":"School of Immunology & Microbial Sciences, King\u2019s College London , London SE1 9RT , UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daniel","family":"Hebenstreit","sequence":"additional","affiliation":[{"name":"School of Life Sciences, Gibbet Hill Campus, University of Warwick , Coventry CV4 7AL , UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Paloma","family":"Garcia","sequence":"additional","affiliation":[{"name":"Institute for Cancer and Genomic Sciences, University of Birmingham , Edgbaston, Birmingham B15 2TT , UK"},{"name":"Birmingham Centre for Genome Biology, University of Birmingham , Edgbaston, Birmingham B15 2TT , UK"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3355-1741","authenticated-orcid":false,"given":"Pawel","family":"Grzechnik","sequence":"additional","affiliation":[{"name":"School of Biological Sciences, University of Manchester, Michael Smith Building , Oxford Road, Manchester M13 9PT , UK"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2022,9,9]]},"reference":[{"key":"2023020311102731200_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. 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