ArticleThe Journal of clinical endocrinology and metabolism2015
A Report of Three Cases With Acquired Generalized Lipodystrophy With Distinct Autoimmune Conditions Treated With Metreleptin.
Article in The Journal of clinical endocrinology and metabolism, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT03936829 (Cyclophosphamide in the Treatment of Panniculitis Associated Acquired Lipodystrophy Syndrome With Type 1 Diabetes), which is not on this map. Cited by 18 papers.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
Cyclophosphamide in the Treatment of Panniculitis Associated Acquired Lipodystrophy Syndrome With Type 1 Diabetes
Who cites it
18 citing papers in PubMed, 41 citations in OpenAlex.
- Lipodystrophies and female reproduction: pathophysiology, clinical spectrum, and management across the reproductive lifespan.Hormones (Athens, Greece) · 2026Review
- Leptin Therapy Improves Metabolic Dysfunction in Immune Checkpoint Inhibitor-induced Lipodystrophy.JCEM case reports · 2026Article
- Leptin and leptin resistance in obesity: current evidence, mechanisms and future directions.Endocrine connections · 2025Review
- Diagnosis, treatment and management of lipodystrophy: the physician perspective on the patient journey.Orphanet journal of rare diseases · 2024Article
- Commentary: A rapid action plan to improve diagnosis and management of lipodystrophy syndromes.Frontiers in endocrinology · 2024Article
- Effect of Leptin Therapy on Survival in Generalized and Partial Lipodystrophy: A Matched Cohort Analysis.The Journal of clinical endocrinology and metabolism · 2021Article
- The pleiotropic roles of leptin in metabolism, immunity, and cancer.The Journal of experimental medicine · 2021Review
- Brown Adipose Tissue: New Challenges for Prevention of Childhood Obesity. A Narrative Review.Nutrients · 2021Review
- Effects of Metreleptin on Patient Outcomes and Quality of Life in Generalized and Partial Lipodystrophy.Journal of the Endocrine Society · 2021Article
- Apolipoprotein CIII and Angiopoietin-like Protein 8 are Elevated in Lipodystrophy and Decrease after Metreleptin.Journal of the Endocrine Society · 2021Article
- Tissue-Specific Effects of Leptin on Glucose and Lipid Metabolism.Endocrine reviews · 2021Review
- Selection of a Full Agonist Combinatorial Antibody that Rescues Leptin Deficiency In Vivo.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2020Article
- Brown Adipose Tissue Response to Cold Stimulation Is Reduced in Girls With Autoimmune Hypothyroidism.Journal of the Endocrine Society · 2019Article
- Update on Therapeutic Options in Lipodystrophy.Current diabetes reports · 2018Review
- A Novel Generalized Lipodystrophy-Associated Progeroid Syndrome Due to Recurrent Heterozygous LMNA p.T10I Mutation.The Journal of clinical endocrinology and metabolism · 2018Article
- Potential association of LMNA-associated generalized lipodystrophy with juvenile dermatomyositis.Clinical diabetes and endocrinology · 2018Article
- Case of lipoatrophic diabetes induced by juvenile dermatomyositis.Journal of diabetes investigation · 2017Article
- Efficacy and Safety of Metreleptin in Patients with Partial Lipodystrophy: Lessons from an Expanded Access Program.Journal of diabetes & metabolism · 2016Article
Corrections and comments
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Authors and funding
4 authors at 1 institution in 1 country.
Funding
Abstract
contextAcquired generalized lipodystrophy (AGL) is associated with leptin deficiency as a result of adipose tissue loss and hypertriglyceridemia, insulin resistance, and hepatic steatosis. It may coexist with other autoimmune diseases such as Hashimoto's thyroiditis, rheumatoid arthritis, hemolytic anemia, and chronic active hepatitis. Metreleptin therapy has been shown to improve metabolic abnormalities in lipodystrophy, but the effect on AGL patients with active autoimmune disease is unknown. CASE DESCRIPTION: We report 3 cases of pediatric patients with AGL and distinct active autoimmune diseases who were treated with metreleptin over a period of 4-6 years. Case 1 is a 9-year-old girl with active juvenile dermatomyositis, who was successfully treated with leptin with no worsening of her dermatomoysitis. Case 2 is a 16-year-old female with Graves' disease, who could discontinue all her antidiabetic medication completely with improved triglyceride levels. Case 3 is an 11-year-old boy with active autoimmune hepatitis and chronic urticaria, whose hyperphagia has resolved and his liver enzymes and hepatosplenomegaly have improved.
conclusionMetreleptin therapy is of considerable clinical benefit to reduce insulin resistance and hypertriglyceridemia and did not appear to alter the clinical course of autoimmune disease nor clinical efficacy of immunosuppressive treatments. Our observations suggest that risk or presence of autoimmune disease should not lead to withholding of metreleptin treatment from patients with AGL, but should prompt close clinical follow up in light of cautionary preclinical data.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.