{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,17]],"date-time":"2026-10-17T14:00:48Z","timestamp":1792245648993,"version":"4.4.0"},"reference-count":140,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2021,2,15]],"date-time":"2021-02-15T00:00:00Z","timestamp":1613347200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Genet."],"abstract":"<jats:p>\n                    Prader-Willi Syndrome (PWS) is a rare neurodevelopmental disorder that affects approximately 1 in 20,000 individuals worldwide. Symptom progression in PWS is classically characterized by two nutritional stages. Stage 1 is hypotonia characterized by poor muscle tone that leads to poor feeding behavior causing failure to thrive in early neonatal life. Stage 2 is followed by the development of extreme hyperphagia, also known as insatiable eating and fixation on food that often leads to obesity in early childhood. Other major features of PWS include obsessive-compulsive and hoarding behaviors, intellectual disability, and sleep abnormalities. PWS is genetic disorder mapping to imprinted 15q11.2-q13.3 locus, specifically at the paternally expressed\n                    <jats:italic>SNORD116<\/jats:italic>\n                    locus of small nucleolar RNAs and noncoding host gene transcripts.\n                    <jats:italic>SNORD116<\/jats:italic>\n                    is processed into several noncoding components and is hypothesized to orchestrate diurnal changes in metabolism through epigenetics, according to functional studies. Here, we review the current status of epigenetic mechanisms in PWS, with an emphasis on an emerging role for\n                    <jats:italic>SNORD116<\/jats:italic>\n                    in circadian and sleep phenotypes. We also summarize current ongoing therapeutic strategies, as well as potential implications for more common human metabolic and psychiatric disorders.\n                  <\/jats:p>","DOI":"10.3389\/fgene.2021.624581","type":"journal-article","created":{"date-parts":[[2021,2,15]],"date-time":"2021-02-15T14:33:26Z","timestamp":1613399606000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":34,"title":["Epigenetics in Prader-Willi Syndrome"],"prefix":"10.3389","volume":"12","author":[{"given":"Aron Judd P.","family":"Mendiola","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janine M.","family":"LaSalle","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2021,2,15]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"106874","DOI":"10.1016\/j.nlm.2018.05.011","article-title":"Cognitive deficits in the Snord116 deletion mouse model for Prader-Willi syndrome","volume":"165","author":"Adhikari","year":"2019","journal-title":"Neurobiol. Learn. Mem."},{"key":"ref2","doi-asserted-by":"publisher","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":"ref3","doi-asserted-by":"publisher","first-page":"377","DOI":"10.1038\/nn.3651","article-title":"Circadian behavior is light-reprogrammed by plastic DNA methylation","volume":"17","author":"Azzi","year":"2014","journal-title":"Nat. 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