{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,11,14]],"date-time":"2026-11-14T15:43:59Z","timestamp":1794671039692,"version":"4.9.1"},"reference-count":17,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2023,11,23]],"date-time":"2023-11-23T00:00:00Z","timestamp":1700697600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2023,11,23]],"date-time":"2023-11-23T00:00:00Z","timestamp":1700697600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Sci Rep"],"abstract":"<jats:title>Abstract<\/jats:title><jats:p>The Hyperphagia Questionnaire for Clinical Trials (HQ-CT) is an observer-reported outcome measure that has been widely used in interventional studies to assess changes in hyperphagic behaviors in individuals with Prader-Willi syndrome (PWS). However, HQ-CT scores in the wider PWS population and the general population have not been reported. Here we report HQ-CT scores from more than 400 individuals with PWS and 600 typical individuals, aged 5\u201326. Overall, HQ-CT scores were significantly higher in those with PWS compared to typically developing individuals at all ages evaluated. In addition, while HQ-CT scores in the typically developing population decreased with age, scores increased with age in PWS. To further understand the variability of HQ-CT scores in the PWS population, semi-structured interviews were conducted with caregivers of a small subset of adults with PWS who had unexpectedly low HQ-CT scores. These caregivers reported that strict adherence to a food routine, food security measures and supervised food preparation reduced the frequency and intensity of hyperphagic behaviors measured by HQ-CT. Thus, hyperphagic behaviors are captured by the HQ-CT for most individuals with PWS, but for some individuals residing in settings with highly structured food routines, HQ-CT scores may not fully reflect the extent of PWS-associated hyperphagia.<\/jats:p>","DOI":"10.1038\/s41598-023-48024-5","type":"journal-article","created":{"date-parts":[[2023,11,23]],"date-time":"2023-11-23T12:02:28Z","timestamp":1700740948000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":28,"title":["Analysis of Hyperphagia Questionnaire for Clinical Trials (HQ-CT) scores in typically developing individuals and those with Prader-Willi syndrome"],"prefix":"10.1038","volume":"13","author":[{"given":"Lisa","family":"Matesevac","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Caroline J.","family":"Vrana-Diaz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jessica E.","family":"Bohonowych","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lauren","family":"Schwartz","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Theresa V.","family":"Strong","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2023,11,23]]},"reference":[{"key":"48024_CR1","doi-asserted-by":"publisher","first-page":"1249","DOI":"10.1007\/s40618-015-0312-9","volume":"38","author":"MA Angulo","year":"2015","unstructured":"Angulo, M. A., Butler, M. G. & Cataletto, M. E. Prader-Willi syndrome: A review of clinical, genetic, and endocrine findings. J. Endocrinol. Invest. 38, 1249\u20131263. https:\/\/doi.org\/10.1007\/s40618-015-0312-9 (2015).","journal-title":"J. Endocrinol. Invest."},{"key":"48024_CR2","doi-asserted-by":"publisher","first-page":"10","DOI":"10.1038\/gim.0b013e31822bead0","volume":"14","author":"SB Cassidy","year":"2012","unstructured":"Cassidy, S. B., Schwartz, S., Miller, J. L. & Driscoll, D. J. Prader-Willi syndrome. Genet. Med. 14, 10\u201326. https:\/\/doi.org\/10.1038\/gim.0b013e31822bead0 (2012).","journal-title":"Genet. Med."},{"key":"48024_CR3","unstructured":"Driscoll, D. J., Miller, J. L., Schwartz, S. & Cassidy, S. B. in GeneReviews(R) (eds M. P. Adam et al.) (University of Washington, 1993)."},{"key":"48024_CR4","doi-asserted-by":"publisher","first-page":"207","DOI":"10.2174\/1573396315666190716120925","volume":"15","author":"MG Butler","year":"2019","unstructured":"Butler, M. G., Miller, J. L. & Forster, J. L. Prader-Willi syndrome\u2014Clinical genetics, diagnosis and treatment approaches: An update. Curr. Pediatr. Rev. 15, 207\u2013244. https:\/\/doi.org\/10.2174\/1573396315666190716120925 (2019).","journal-title":"Curr. Pediatr. Rev."},{"key":"48024_CR5","doi-asserted-by":"publisher","first-page":"1040","DOI":"10.1002\/ajmg.a.33951","volume":"155A","author":"JL Miller","year":"2011","unstructured":"Miller, J. L. et al. Nutritional phases in Prader-Willi syndrome. Am. J. Med. Genet. A 155A, 1040\u20131049. https:\/\/doi.org\/10.1002\/ajmg.a.33951 (2011).","journal-title":"Am. J. Med. Genet. A"},{"key":"48024_CR6","doi-asserted-by":"publisher","first-page":"25","DOI":"10.1186\/s11689-021-09373-2","volume":"13","author":"L Schwartz","year":"2021","unstructured":"Schwartz, L. et al. Behavioral features in Prader-Willi syndrome (PWS): consensus paper from the International PWS clinical trial consortium. J. Neurodev. Disord. 13, 25. https:\/\/doi.org\/10.1186\/s11689-021-09373-2 (2021).","journal-title":"J. Neurodev. Disord."},{"key":"48024_CR7","doi-asserted-by":"publisher","first-page":"e0194655","DOI":"10.1371\/journal.pone.0194655","volume":"13","author":"N Kayadjanian","year":"2018","unstructured":"Kayadjanian, N., Schwartz, L., Farrar, E., Comtois, K. A. & Strong, T. V. High levels of caregiver burden in Prader-Willi syndrome. PLoS One 13, e0194655. https:\/\/doi.org\/10.1371\/journal.pone.0194655 (2018).","journal-title":"PLoS One"},{"key":"48024_CR8","doi-asserted-by":"publisher","first-page":"e0248739","DOI":"10.1371\/journal.pone.0248739","volume":"16","author":"N Kayadjanian","year":"2021","unstructured":"Kayadjanian, N. et al. Characteristics and relationship between hyperphagia, anxiety, behavioral challenges and caregiver burden in Prader-Willi syndrome. PLoS One 16, e0248739. https:\/\/doi.org\/10.1371\/journal.pone.0248739 (2021).","journal-title":"PLoS One"},{"key":"48024_CR9","doi-asserted-by":"publisher","first-page":"1","DOI":"10.4303\/jem\/235968","volume":"4","author":"J Kotler","year":"2016","unstructured":"Kotler, J., Balko, K., Berall, G. & Haig, D. Nutritional phases in prader-willi syndrome: Evolutionary and clinical interpretations. J. Evol. Med. 4, 1\u20137. https:\/\/doi.org\/10.4303\/jem\/235968 (2016).","journal-title":"J. Evol. Med."},{"key":"48024_CR10","doi-asserted-by":"publisher","first-page":"A25","DOI":"10.1016\/j.jval.2015.03.154","volume":"18","author":"S Fehnel","year":"2015","unstructured":"Fehnel, S. et al. Development of the hyperphagia questionnaire for use in Prader-Willi syndrome clinical trials. Value Health 18, A25\u2013A25. https:\/\/doi.org\/10.1016\/j.jval.2015.03.154 (2015).","journal-title":"Value Health"},{"key":"48024_CR11","doi-asserted-by":"publisher","first-page":"1816","DOI":"10.1038\/oby.2007.216","volume":"15","author":"EM Dykens","year":"2007","unstructured":"Dykens, E. M., Maxwell, M. A., Pantino, E., Kossler, R. & Roof, E. Assessment of hyperphagia in Prader-Willi syndrome. Obes. Silver Spring 15, 1816\u20131826. https:\/\/doi.org\/10.1038\/oby.2007.216 (2007).","journal-title":"Obes. Silver Spring"},{"key":"48024_CR12","doi-asserted-by":"publisher","first-page":"1009055","DOI":"10.3390\/genes10090713","volume":"10","author":"J Bohonowych","year":"2019","unstructured":"Bohonowych, J., Miller, J., McCandless, S. E. & Strong, T. V. The global prader-willi syndrome registry: development, launch, and early demographics. Genes Basel 10, 1009055. https:\/\/doi.org\/10.3390\/genes10090713 (2019).","journal-title":"Genes Basel"},{"key":"48024_CR13","doi-asserted-by":"publisher","first-page":"460","DOI":"10.1111\/jar.12950","volume":"35","author":"EM Dykens","year":"2022","unstructured":"Dykens, E. M., Roof, E. & Hunt-Hawkins, H. \u201cThe cure for us is a lot of things\u201d: How young people with Prader-Willi syndrome view themselves and future clinical trials. J. Appl. Res. Intellect. Disabil. 35, 460\u2013470. https:\/\/doi.org\/10.1111\/jar.12950 (2022).","journal-title":"J. Appl. Res. Intellect. Disabil."},{"key":"48024_CR14","doi-asserted-by":"publisher","first-page":"1230","DOI":"10.1080\/13696998.2018.1528980","volume":"21","author":"JH Tsai","year":"2018","unstructured":"Tsai, J. H., Scheimann, A. O., McCandless, S. E., Strong, T. V. & Bridges, J. F. P. Caregiver priorities for endpoints to evaluate treatments for Prader-Willi syndrome: A best-worst scaling. J. Med. Econ. 21, 1230\u20131237. https:\/\/doi.org\/10.1080\/13696998.2018.1528980 (2018).","journal-title":"J. Med. Econ."},{"key":"48024_CR15","doi-asserted-by":"publisher","DOI":"10.1111\/dom.13021","author":"SE McCandless","year":"2017","unstructured":"McCandless, S. E. et al. Effects of MetAP2 inhibition on hyperphagia and body weight in Prader-Willi syndrome: a randomized, double-blind, placebo-controlled trial. Diabetes Obes. Metab. https:\/\/doi.org\/10.1111\/dom.13021 (2017).","journal-title":"Diabetes Obes. Metab."},{"key":"48024_CR16","doi-asserted-by":"publisher","first-page":"1696","DOI":"10.1210\/clinem\/dgad015","volume":"108","author":"E Roof","year":"2023","unstructured":"Roof, E. et al. Intranasal carbetocin reduces hyperphagia, anxiousness, and distress in prader-willi syndrome: CARE-PWS phase 3 trial. J. Clin. Endocrinol. Metab. 108, 1696\u20131708. https:\/\/doi.org\/10.1210\/clinem\/dgad015 (2023).","journal-title":"J. Clin. Endocrinol. Metab."},{"key":"48024_CR17","doi-asserted-by":"publisher","first-page":"1676","DOI":"10.1210\/clinem\/dgad014","volume":"108","author":"JL Miller","year":"2023","unstructured":"Miller, J. L. et al. Diazoxide choline extended-release tablet in people with prader-willi syndrome: a double-blind. Placebo Controll. Trial. J. Clin. Endocrinol. Metab. 108, 1676\u20131685. https:\/\/doi.org\/10.1210\/clinem\/dgad014 (2023).","journal-title":"Placebo Controll. Trial. J. Clin. Endocrinol. 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