{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T22:39:04Z","timestamp":1791326344034,"version":"4.3.1"},"reference-count":26,"publisher":"Wiley","issue":"9","license":[{"start":{"date-parts":[[2013,7,29]],"date-time":"2013-07-29T00:00:00Z","timestamp":1375056000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"JSPS KAKENHI","award":["24791086"],"award-info":[{"award-number":["24791086"]}]},{"name":"Kawano Masanori Memorial Foundation for the Promotion of Pediatrics"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J of Med Genetics Pt A"],"published-print":{"date-parts":[[2013,9]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:label\/><jats:p>Prader\u2013Willi syndrome (PWS), a complex genetic disorder, arises from suppressed expression of paternally inherited imprinted genes on chromosome 15q11\u2010q13. Characteristics include short stature, intellectual disability, behavioral problems, hypogonadism, obesity, and reduced bone and muscle mass. Testosterone replacement (TR) remains controversial due to concerns regarding behavioral problems. To evaluate the effects of TR on secondary sexual characteristics, body composition, and behavior in adult males with PWS, 22 male PWS patients over the age of 16 with behavioral scores of less than grade 4 on the Modified Overt Aggression Scale (MOAS) underwent monthly intramuscular TR (125\u2009mg). Pubertal change, body composition and behavior were evaluated before and after 24 months of therapy. Serum testosterone, LH, and FSH did not change. Increased pubic hair was observed in 16 of 22 patients (72.7%). Percent body fat decreased from 47.55\u2009\u00b1\u20092.06% to 39.75\u2009\u00b1\u20091.60% (n\u2009=\u200918) (<jats:italic>P<\/jats:italic>\u2009=\u20090.018). Bone mineral density increased from 0.8505\u2009\u00b1\u20090.0426\u2009g\/cm<jats:sup>2<\/jats:sup> to 0.9035\u2009\u00b1\u20090.0465\u2009g\/cm<jats:sup>2<\/jats:sup> (n\u2009=\u200918) (<jats:italic>P<\/jats:italic>\u2009=\u20090.036), and lean body mass increased from 18093.4\u2009\u00b1\u2009863.0\u2009g to 20312.1\u2009\u00b1\u20091027.2\u2009g (n\u2009=\u200918) (<jats:italic>P<\/jats:italic>\u2009=\u20090.009). The MOAS was unchanged, from 4.5\u2009\u00b1\u20092.0 at the beginning of the study to 3.0\u2009\u00b1\u20091.7 at the end of study indicating no increase in aggression. No behavioral problems were observed. Based on this pilot study, TR with 125\u2009mg monthly is a potentially safe and useful intervention for adult males with PWS. \u00a9 2013 Wiley Periodicals, Inc.<\/jats:p><\/jats:sec>","DOI":"10.1002\/ajmg.a.36048","type":"journal-article","created":{"date-parts":[[2013,7,29]],"date-time":"2013-07-29T19:49:30Z","timestamp":1375127370000},"page":"2167-2173","source":"Crossref","is-referenced-by-count":34,"title":["Testosterone replacement therapy to improve secondary sexual characteristics and body composition without adverse behavioral problems in adult male patients with Prader\u2013Willi syndrome: An observational study"],"prefix":"10.1002","volume":"161","author":[{"given":"Yasuhiro","family":"Kido","sequence":"first","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Satoru","family":"Sakazume","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yoshiko","family":"Abe","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuji","family":"Oto","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hisashi","family":"Itabashi","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masahisa","family":"Shiraishi","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Atsunori","family":"Yoshino","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yuriko","family":"Tanaka","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kazuo","family":"Obata","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nobuyuki","family":"Murakami","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Toshiro","family":"Nagai","sequence":"additional","affiliation":[{"name":"Department of Pediatrics Dokkyo Medical University Koshigaya Hospital Saitama Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2013,7,29]]},"reference":[{"key":"e_1_2_7_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.1999.tb00542.x"},{"key":"e_1_2_7_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/j.1939-4640.2006.tb01177.x"},{"key":"e_1_2_7_4_1","doi-asserted-by":"crossref","first-page":"787","DOI":"10.1210\/edrv.22.6.0447","article-title":"Endocrine dysfunction in Prader\u2010Willi syndrome: A review with special reference to GH","volume":"22","author":"Burman P","year":"2001","journal-title":"Endocr Rev"},{"key":"e_1_2_7_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.34.11.917"},{"key":"e_1_2_7_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2008.165"},{"key":"e_1_2_7_7_1","doi-asserted-by":"publisher","DOI":"10.1038\/gim.0b013e31822bead0"},{"key":"e_1_2_7_8_1","doi-asserted-by":"crossref","first-page":"327","DOI":"10.1007\/s00431-002-1132-4","article-title":"Hypogonadism and pubertal development in Prader\u2010Willi syndrome","volume":"162","author":"Crino A","year":"2003","journal-title":"Eur J Pediatr"},{"key":"e_1_2_7_9_1","doi-asserted-by":"publisher","DOI":"10.1159\/000313377"},{"key":"e_1_2_7_10_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2005-0902"},{"key":"e_1_2_7_11_1","doi-asserted-by":"publisher","DOI":"10.1159\/000100925"},{"key":"e_1_2_7_12_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2008-0649"},{"key":"e_1_2_7_13_1","doi-asserted-by":"publisher","DOI":"10.1515\/jpem.2011.201"},{"key":"e_1_2_7_14_1","doi-asserted-by":"publisher","DOI":"10.1038\/sj.ejcn.1601206"},{"key":"e_1_2_7_15_1","doi-asserted-by":"publisher","DOI":"10.3345\/kjp.2011.54.2.55"},{"key":"e_1_2_7_16_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.bpo.0000226282.01202.4f"},{"key":"e_1_2_7_17_1","first-page":"S57","article-title":"The measurement of aggressive behavior: Reflections on the use of the Overt Aggression Scale and the Modified Overt Aggression Scale","volume":"3","author":"Ratey JJ","year":"1991","journal-title":"J Neuropsychiatry Clin Neurosci"},{"key":"e_1_2_7_18_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1651-2227.2001.tb00451.x"},{"key":"e_1_2_7_19_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2011-1954"},{"key":"e_1_2_7_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ridd.2010.12.014"},{"key":"e_1_2_7_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.34171"},{"key":"e_1_2_7_22_1","doi-asserted-by":"publisher","DOI":"10.1210\/jc.2010-0907"},{"key":"e_1_2_7_23_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2788.2011.01418.x"},{"key":"e_1_2_7_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(08)60072-0"},{"key":"e_1_2_7_25_1","doi-asserted-by":"publisher","DOI":"10.1067\/mpd.2001.118399"},{"key":"e_1_2_7_26_1","doi-asserted-by":"publisher","DOI":"10.1159\/000323442"},{"key":"e_1_2_7_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.juro.2008.03.113"}],"container-title":["American Journal of Medical Genetics Part A"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.a.36048","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1002%2Fajmg.a.36048","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ajmg.a.36048","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,24]],"date-time":"2023-09-24T13:54:35Z","timestamp":1695563675000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ajmg.a.36048"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2013,7,29]]},"references-count":26,"journal-issue":{"issue":"9","published-print":{"date-parts":[[2013,9]]}},"alternative-id":["10.1002\/ajmg.a.36048"],"URL":"https:\/\/doi.org\/10.1002\/ajmg.a.36048","archive":["Portico"],"relation":{},"ISSN":["1552-4825","1552-4833"],"issn-type":[{"value":"1552-4825","type":"print"},{"value":"1552-4833","type":"electronic"}],"subject":[],"published":{"date-parts":[[2013,7,29]]}}}