{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T15:00:57Z","timestamp":1788447657846,"version":"build-2803163510"},"reference-count":16,"publisher":"The Endocrine Society","issue":"12","license":[{"start":{"date-parts":[[2025,4,30]],"date-time":"2025-04-30T00:00:00Z","timestamp":1745971200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,11,18]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Context<\/jats:title>\n                    <jats:p>Tirzepatide, a dual glucose-dependent insulinotropic peptide\/glucagon-like peptide 1 (GIP\/GLP1) receptor agonist, was recently approved for type 2 diabetes and weight management. Alstr\u00f6m syndrome (AS) is a rare, genetic, multisystemic disorder, characterized by cone-rod dystrophy, progressive hearing loss, obesity, and diabetes with profound insulin resistance due to marked hyperphagia.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>Here we highlight the potential of tirzepatide as a novel therapeutic option for improving glycemic outcomes, metabolic dysfunction\u2013associated steatotic liver disease (MASLD), and effectively reducing body weight in individuals with AS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We present the first 2 reported cases of people living with AS treated with tirzepatide.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Two individuals with AS, previously treated with semaglutide, received tirzepatide at our clinic. The first, a 23-year-old man with 18 months of treatment, experienced a weight loss of \u221228 kg (113.6 to 83\u2005kg, \u221226.9%); glycated hemoglobin A1c decreased by \u22120.4% (6.7% to 6.3%), with considerable reductions in daily insulin doses of \u221296\u2005IU\/day (\u221283%; 58 to 20\u2005IU insulin glargine and 58 to 0\u2005IU prandial insulin), while maintaining oral antidiabetics. Hepatic steatosis, with a previous fat fraction of 20%, resolved as confirmed by magnetic resonance imaging (MRI). The second, a 20-year-old man with previously well-controlled diabetes, was followed up for 9 months and showed a weight reduction of \u22129.5\u2005kg (132 kg to 122.5\u2005kg; \u22127.2%) with a reduction of hepatic lipid content from 21% at the latest MRI to 11% after approximately 3 months of therapy.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Tirzepatide shows great effectiveness with regard to body weight, MASLD, and insulin resistance in AS. Follow-up studies with larger cohorts have to be performed to confirm these findings.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1210\/clinem\/dgaf258","type":"journal-article","created":{"date-parts":[[2025,4,29]],"date-time":"2025-04-29T08:10:48Z","timestamp":1745914248000},"page":"3364-3369","source":"Crossref","is-referenced-by-count":9,"title":["Effectiveness of the Dual GIP\/GLP1-Agonist Tirzepatide in 2 Cases of Alstr\u00f6m Syndrome, a Rare Obesity Syndrome"],"prefix":"10.1210","volume":"110","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7786-8156","authenticated-orcid":false,"given":"Moritz","family":"Ferch","sequence":"first","affiliation":[{"name":"Division of Endocrinology and Metabolism, Department of Medicine III, Medical University of Vienna , Vienna 1090 ,","place":["Austria"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabel","family":"Peitsch","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism, 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