{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T10:08:10Z","timestamp":1780481290489,"version":"3.54.1"},"reference-count":26,"publisher":"The Endocrine Society","issue":"7","license":[{"start":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T00:00:00Z","timestamp":1780444800000},"content-version":"vor","delay-in-days":14,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,5,20]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Smith\u2013Magenis syndrome (SMS) is a rare neurodevelopmental disorder characterized by intellectual disability, behavioral dysregulation, and hyperphagia-driven obesity. While dual glucose-dependent insulinotropic polypeptide (GIP) and glucagon-like peptide-1 (GLP-1) receptor agonists have demonstrated efficacy in the general population, their use in patients with SMS has not been described. We report the case of a 31-year-old woman with SMS (17p11.2 deletion) treated with tirzepatide, a dual GIP\/GLP-1 receptor agonist. The patient presented with lifelong obesity (body mass index [BMI] 32.0-32.9\u2005kg\/m2 in adulthood) and aggressive behaviors refractory to standard management. Following initiation of tirzepatide, titrated to 5\u2005mg weekly, she achieved 9.4% weight loss (7.3\u2005kg) over 10 months, along with improvement in fasting glucose levels. Concurrently, caregivers reported notable behavioral improvements, including reduced food-seeking behavior and impulsivity. Quantitative analysis demonstrated a significant reduction in aggression. The treatment was well tolerated. This case suggests that tirzepatide may represent a promising therapeutic option for SMS, targeting both metabolic and central nervous system pathways involved in its phenotype.<\/jats:p>","DOI":"10.1210\/jcemcr\/luag152","type":"journal-article","created":{"date-parts":[[2026,5,19]],"date-time":"2026-05-19T11:39:37Z","timestamp":1779190777000},"source":"Crossref","is-referenced-by-count":0,"title":["Tirzepatide for weight and behavior management in a patient with Smith-Magenis syndrome"],"prefix":"10.1210","volume":"4","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3446-9192","authenticated-orcid":false,"given":"X Charlene","family":"Liao","sequence":"first","affiliation":[{"name":"Independent Scholar , Palo Alto, CA 94303, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-0077-5554","authenticated-orcid":false,"given":"Tao","family":"Huang","sequence":"additional","affiliation":[{"name":"Independent Scholar , Mountain View, CA 94041, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9515-881X","authenticated-orcid":false,"given":"David S","family":"Hong","sequence":"additional","affiliation":[{"name":"Department of Psychiatry and Behavioral Sciences, Stanford University, Stanford, CA 94305 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5467-9264","authenticated-orcid":false,"given":"Liqun","family":"Luo","sequence":"additional","affiliation":[{"name":"Howard Hughes Medical Institute, Department of Biology, Stanford University, Stanford, CA 94305 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2026,6,3]]},"reference":[{"issue":"3","key":"2026060305173400000_luag152-B1","doi-asserted-by":"crossref","first-page":"393","DOI":"10.1002\/ajmg.1320240303","article-title":"Interstitial deletion of (17)(p11.2p11.2) in nine 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