{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,19]],"date-time":"2026-02-19T00:19:26Z","timestamp":1771460366889,"version":"3.50.1"},"reference-count":32,"publisher":"The Endocrine Society","issue":"8","license":[{"start":{"date-parts":[[2021,4,2]],"date-time":"2021-04-02T00:00:00Z","timestamp":1617321600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000062","name":"National Institute of Diabetes and Digestive and Kidney Diseases","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000062","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100007270","name":"University of Michigan","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007270","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,7,13]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>Data quantifying the impact of metreleptin therapy on survival in non-human immunodeficiency virus (HIV)-related generalized lipodystrophy (GL) and partial lipodystrophy (PL) are unavailable.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>This study aimed to estimate the treatment effect of metreleptin on survival in patients with GL and PL.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Design\/Setting\/Patients<\/jats:title>\n                  <jats:p>Demographic and clinical characteristics were used to match metreleptin-treated and metreleptin-na\u00efve patients with GL and PL. Differences in mortality risk were estimated between matched cohorts of metreleptin-treated and metreleptin-na\u00efve patient cohorts using Cox proportional hazard models. Sensitivity analyses assessed the impact of study assumptions and the robustness of results.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Outcome Measures<\/jats:title>\n                  <jats:p>This study assessed time-to-mortality and risk of mortality.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>The analysis evaluated 103 metreleptin-na\u00efve patients with characteristics matched to 103 metreleptin-treated patients at treatment initiation. Even after matching, some metabolic and organ abnormalities were more prevalent in the metreleptin-treated cohort due to bias toward treating more severely affected patients. A Cox proportional hazards model associated metreleptin therapy with an estimated 65% decrease in mortality risk (hazard ratio [HR] 0.348, 95% confidence interval (CI): 0.134\u20130.900; P = 0.029) even though the actual number of events were relatively small. Results were robust across a broad range of alternate methodological assumptions. Kaplan\u2013Meier estimates of time-to-mortality for the metreleptin-treated and the matched metreleptin-na\u00efve cohorts were comparable.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>Metreleptin therapy was associated with a reduction in mortality risk in patients with lipodystrophy syndromes despite greater disease severity in treated patients, supporting the view that metreleptin can have a positive disease-modifying impact. Confirmatory studies in additional real-world and clinical datasets are warranted.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgab216","type":"journal-article","created":{"date-parts":[[2021,4,6]],"date-time":"2021-04-06T14:39:26Z","timestamp":1617719966000},"page":"e2953-e2967","source":"Crossref","is-referenced-by-count":24,"title":["Effect of Leptin Therapy on Survival in Generalized and Partial Lipodystrophy: A Matched Cohort Analysis"],"prefix":"10.1210","volume":"106","author":[{"given":"Keziah","family":"Cook","sequence":"first","affiliation":[{"name":"Analysis Group Inc., Menlo Park, CA 94025, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Omer","family":"Ali","sequence":"additional","affiliation":[{"name":"Analysis Group Inc., Menlo Park, CA 94025, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Baris","family":"Akinci","sequence":"additional","affiliation":[{"name":"Dokuz Eylul University, Izmir, 35220, Turkey"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maria Cristina","family":"Foss de Freitas","sequence":"additional","affiliation":[{"name":"University of S\u00e3o Paulo \u2013 Ribeirao Preto Medical School, S\u00e3o Paulo, 14049, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"suffix":"Jr","given":"Renan Magalh\u00e3es","family":"Montenegro","sequence":"additional","affiliation":[{"name":"Federal University of Cear\u00e1, Cear\u00e1, 60020, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Virginia Oliveira","family":"Fernandes","sequence":"additional","affiliation":[{"name":"Federal University of Cear\u00e1, Cear\u00e1, 60020, Brazil"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Deepshekhar","family":"Gupta","sequence":"additional","affiliation":[{"name":"Analysis Group Inc., Menlo Park, CA 94025, 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