{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,29]],"date-time":"2026-09-29T13:22:20Z","timestamp":1790688140152,"version":"4.1.0"},"reference-count":45,"publisher":"Wiley","issue":"8","license":[{"start":{"date-parts":[[2022,5,12]],"date-time":"2022-05-12T00:00:00Z","timestamp":1652313600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["dom-pubs.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Diabetes Obesity Metabolism"],"published-print":{"date-parts":[[2022,8]]},"abstract":"Abstract<\/jats:title>\n \n Aim<\/jats:title>\n To describe baseline characteristics and follow\u2010up data in patients with lipodystrophy syndromes treated with metreleptin in a national reference network, in a real\u2010life setting.<\/jats:p>\n <\/jats:sec>\n \n Patients and Methods<\/jats:title>\n Clinical and metabolic data from patients receiving metreleptin in France were retrospectively collected, at baseline, at 1\u2009year and at the latest follow\u2010up during treatment.<\/jats:p>\n <\/jats:sec>\n \n Results<\/jats:title>\n \n Forty\u2010seven patients with lipodystrophy including generalized lipodystrophy (GLD; n\u00a0=\u00a028) and partial lipodystrophy (PLD; n\u00a0=\u00a019) received metreleptin over the last decade. At baseline, the median (interquartile range [IQR]) patient age was 29.3 (16.6\u201047.6) years, body mass index was 23.8\u00a0(21.2\u201025.7) kg\/m\n 2<\/jats:sup>\n and serum leptin was 3.2 (1.0\u20104.9) ng\/mL, 94% of patients had diabetes (66% insulin\u2010treated), 53% had hypertension and 87% had dyslipidaemia. Metreleptin therapy, administered for a median (IQR) of 31.7 (14.2\u201076.0) months, was ongoing in 77% of patients at the latest follow\u2010up. In patients with GLD, glycated haemoglobin (HbA1c) and fasting triglyceride levels significantly decreased from baseline to 1\u2009year of metreleptin treatment, from 8.4 (6.5\u20109.9)% [68 (48\u201085) mmol\/mol] to 6.8 (5.6\u20107.4)% [51(38\u201057) mmol\/mol], and 3.6 (1.7\u20108.5) mmol\/L to 2.2 (1.1\u20103.7) mmol\/L, respectively (\n P<\/jats:italic>\n \u00a0<\u20090.001), with sustained efficacy thereafter. In patients with PLD, HbA1c was not significantly modified (7.7 [7.1\u20109.1]% [61 (54\u201076) mmol\/mol] at baseline vs. 7.7 [7.4\u20109.5]% [61(57\u201080) mmol\/mol] at 1 year), and the decrease in fasting triglycerides (from 3.3 [1.9\u20109.9] mmol\/L to 2.5 [1.6\u20105.3] mmol\/L;\n P<\/jats:italic>\n \u00a0<\u20090.01) was not confirmed at the latest assessment (5.2 [2.2\u201011.3] mmol\/L). However, among PLD patients, at 1 year, 61% were responders regarding glucose homeostasis, with lower baseline leptin levels compared to nonresponders, and 61% were responders regarding triglyceridaemia. Liver enzymes significantly decreased only in the GLD group.\n <\/jats:p>\n <\/jats:sec>\n \n Conclusions<\/jats:title>\n In this real\u2010life setting study, metabolic outcomes are improved by metreleptin therapy in patients with GLD. The therapeutic indication for metreleptin needs to be clarified in patients with PLD.<\/jats:p>\n <\/jats:sec>","DOI":"10.1111\/dom.14726","type":"journal-article","created":{"date-parts":[[2022,4,21]],"date-time":"2022-04-21T02:09:15Z","timestamp":1650506955000},"page":"1565-1577","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":39,"title":["Therapeutic indications and metabolic effects of metreleptin in patients with lipodystrophy syndromes: Real\u2010life experience from a national reference network"],"prefix":"10.1111","volume":"24","author":[{"given":"H\u00e9l\u00e9na","family":"Mosbah","sequence":"first","affiliation":[{"name":"Endocrinology Department Assistance Publique\u2013H\u00f4pitaux de Paris (AP\u2010HP), Saint\u2013Antoine University Hospital, National Reference Centre for Rare Diseases of Insulin Secretion and Insulin Sensitivity (PRISIS) Paris France"},{"name":"Sorbonne University, Inserm UMR_S 938, 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