{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,4]],"date-time":"2026-03-04T05:16:25Z","timestamp":1772601385742,"version":"3.50.1"},"reference-count":53,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2019,10,31]],"date-time":"2019-10-31T00:00:00Z","timestamp":1572480000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Pediatric Obesity"],"published-print":{"date-parts":[[2020,1]]},"abstract":"<jats:title>Summary<\/jats:title><jats:sec><jats:title>Background<\/jats:title><jats:p>The clinical phenotype of patients with monogenic obesity due to mutations in the leptin receptor (<jats:italic>LEPR)<\/jats:italic> or melanocortin 4 receptor (<jats:italic>MC4R<\/jats:italic>) gene is characterized by impaired satiety and hyperphagia, leading to extreme, sometimes life\u2010threatening weight gain.<\/jats:p><\/jats:sec><jats:sec><jats:title>Subjects\/methods<\/jats:title><jats:p>In a case series, we analysed the effect of an off\u2010label methylphenidate (MPH) use for 1 year as an individual treatment approach on eating behaviour (Child Eating Behaviour Questionnaire [CEBQ]), appetite (visual analogue scales) and body mass index (BMI) trajectories in five patients with severe obesity due to mutations in the <jats:italic>LEPR<\/jats:italic> (n = 3) or <jats:italic>MC4R<\/jats:italic> (n <jats:italic>= 2<\/jats:italic>) gene.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>After 1 year use of MPH (20 mg\/day divided in two to three doses), BMI (\u0394 BMI<jats:sub>T0\u2212T1<\/jats:sub> \n<jats:inline-graphic xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"graphic\/ijpo12577-math-0001.png\" xlink:title=\"urn:x-wiley:20476302:media:ijpo12577:ijpo12577-math-0001\"\/>: \u22120.7 \u00b1 0.9 kg\/m<jats:sup>2<\/jats:sup>), BMI standard deviation score (SDS) (\u0394 BMI\u2010SDS<jats:sub>T0\u2212T1<\/jats:sub> \n<jats:inline-graphic xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"graphic\/ijpo12577-math-0002.png\" xlink:title=\"urn:x-wiley:20476302:media:ijpo12577:ijpo12577-math-0002\"\/>: \u22120.32 \u00b1 0.20), and %BMIP95 (\u0394 %BMIP95<jats:sub>T0\u2212T1<\/jats:sub> \n<jats:inline-graphic xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" xlink:href=\"graphic\/ijpo12577-math-0003.png\" xlink:title=\"urn:x-wiley:20476302:media:ijpo12577:ijpo12577-math-0003\"\/>: \u22126.6 \u00b1 7.8%) decreased. BMI\u2010SDS velocity decreased from +0.17 \u00b1 0.22 to \u22120.30 \u00b1 0.20. Appetite and CEBQ subscale scores for \u201cfood responsiveness\u201d and \u201cenjoyment of food\u201d decreased. We observed adverse effects with increase in self\u2010reported frequency of disordered sleep, nervousness, hyperactivity, and tics.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>The observed decrease in BMI trajectories with MPH use for one year is clinically meaningful in this group of patients, since the natural course would have been associated with a pronounced increase in BMI, leading to comorbidities and complications over time.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ijpo.12577","type":"journal-article","created":{"date-parts":[[2019,10,31]],"date-time":"2019-10-31T11:04:00Z","timestamp":1572519840000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":26,"title":["Methylphenidate in children with monogenic obesity due to <i>LEPR<\/i> or <i>MC4R<\/i> deficiency improves feeling of satiety and reduces BMI\u2010SDS\u2014A case series"],"prefix":"10.1111","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8693-2647","authenticated-orcid":false,"given":"Stephanie","family":"Brandt","sequence":"first","affiliation":[{"name":"Division of Pediatric Endocrinology and Diabetes, Department of Pediatrics and Adolescent Medicine University Medical Center Ulm  Ulm Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Julia","family":"von Schnurbein","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Diabetes, Department of Pediatrics and Adolescent Medicine University Medical Center Ulm  Ulm Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Belinda","family":"Lennerz","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Diabetes, Department of Pediatrics and Adolescent Medicine University Medical Center Ulm  Ulm Germany"},{"name":"Division of Endocrinology and New Balance Foundation Obesity Prevention Center Boston Children's Hospital and Harvard Medical School, Harvard University, Boston  Boston Massachusetts"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Katja","family":"Kohlsdorf","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Diabetes, Department of Pediatrics and Adolescent Medicine University Medical Center Ulm  Ulm Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Heike","family":"Vollbach","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Diabetes, Department of Pediatrics and Adolescent Medicine University Medical Center Ulm  Ulm Germany"},{"name":"Pediatric Endocrinology and Diabetology Division, Children's Hospital University of Bonn  Bonn Germany"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Christian","family":"Denzer","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Diabetes, 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