{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,24]],"date-time":"2026-10-24T16:37:06Z","timestamp":1792859826555,"version":"4.5.0"},"reference-count":0,"publisher":"S. Karger AG","license":[{"start":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T00:00:00Z","timestamp":1792713600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/karger.com\/pages\/terms-and-conditions"},{"start":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T00:00:00Z","timestamp":1792713600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/karger.com\/pages\/terms-and-conditions"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Horm Res Paediatr"],"abstract":"<jats:p>Background: Monogenic obesity comprises a group of rare genetic disorders characterized by severe early-onset obesity and hyperphagia. Pharmacological treatment options for severe early-onset obesity remain limited, particularly in young children. Case Presentation: We report two children younger than 6 years of age with severe early-onset obesity, including one with genetically confirmed monogenic obesity, who showed reductions in BMI-SDS during semaglutide treatment. Case-1: A 4.5-year-old girl with a homozygous ADCY3 (c.1102G&gt;A) variant presented with severe obesity, hyperphagia, hypertension, hepatosteatosis and Blount\u2019s disease. Baseline BMI was 48.0 kg\/m2 (+5.5 SDS). Semaglutide was initiated at 0.125 mg\/week and titrated to 1.5 mg\/week. After 18 months, BMI-SDS decreased from +5.5 to +4.1 (-25.5%), accompanied by improved in hyperphagia and resolution of left ventricular hypertrophy. No significant adverse effects were observed, and linear growth remained age appropriate. Case-2: A 3.3-year-old boy with hyperphagia, severe obesity and dyslipidaemia was diagnosed with Klinefelter syndrome (47,XXY). Clinical Exome Sequencing identified no causative variant for obesity. Baseline BMI was 32.6 kg\/m2 (+5.7 SDS). Semaglutide was initiated at 0.0625 mg\/week and gradually titrated to 2.0 mg\/week. After 21 months, BMI-SDS decreased from +5.7 to +4.4 (-22%). Treatment was generally well tolerated, with normal linear growth. Conclusion: This two cases describe reductions BMI-SDS during semaglutide treatment in very young children with severe early-onset obesity, including one child with genetically confirmed ADCY3-related monogenic obesity. These findings suggest a potential clinical benefit of semaglutide in this young population, warranting evaluation in larger studies.<\/jats:p>","DOI":"10.1159\/hrp\/adaag048","type":"journal-article","created":{"date-parts":[[2026,10,24]],"date-time":"2026-10-24T16:01:35Z","timestamp":1792857695000},"source":"Crossref","is-referenced-by-count":0,"title":["SEMAGLUTIDE TREATMENT IN TWO VERY YOUNG CHILDREN WITH SEVERE EARLY-ONSET OBESITY"],"prefix":"10.1159","author":[{"given":"Aylin Tugba","family":"Canbaz","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7627-1996","authenticated-orcid":false,"given":"Ahmet","family":"Kahveci","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilknur","family":"Kurt","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Senol","family":"Demir","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bilgen Bilge","family":"Ge\u00e7kinli","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Abdullah","family":"Bereket","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9946-235X","authenticated-orcid":false,"given":"Belma","family":"Haliloglu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"127","published-online":{"date-parts":[[2026,10,23]]},"container-title":["Hormone Research in Paediatrics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/karger.com\/article-pdf\/doi\/10.1159\/hrp\/adaag048","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,10,24]],"date-time":"2026-10-24T16:01:36Z","timestamp":1792857696000},"score":1,"resource":{"primary":{"URL":"https:\/\/karger.com\/article\/doi\/10.1159\/hrp\/adaag048"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,10,23]]},"references-count":0,"URL":"https:\/\/doi.org\/10.1159\/hrp\/adaag048","archive":["Portico"],"relation":{"is-supplemented-by":[{"id-type":"doi","id":"10.6084\/m9.figshare.34744443","asserted-by":"subject"}]},"ISSN":["1663-2818","1663-2826"],"issn-type":[{"value":"1663-2818","type":"print"},{"value":"1663-2826","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,10,23]]}}}