ArticleProceedings of the National Academy of Sciences of the United States of America2013
Leptin resistance is a secondary consequence of the obesity in ciliopathy mutant mice.
Article in Proceedings of the National Academy of Sciences of the United States of America, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 51 papers, 1 of them a synthesis that pooled it.
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Who cites it
51 citing papers in PubMed, 1 synthesis or guideline pooled it, 96 citations in OpenAlex.
- The sequenced rat brain transcriptome--its use in identifying networks predisposing alcohol consumption.The FEBS journal · 2015Pooled it
- Primary cilia: master conductors of cellular communication in development and disease.Nature reviews. Nephrology · 2026Review
- Resolution of chronic idiopathic urticaria with setmelanotide in a patient with Bardet-Biedl Syndrome: A case report.Obesity pillars · 2025Article
- Cilia in the brain display region-dependent oscillations of length and orientation.PLoS biology · 2025Article
- Should GLP-1 receptor agonist therapy be used to treat obesity in Bardet-Biedl syndrome?The Journal of clinical investigation · 2025Article
- Tonic ubiquitination of the central body weight regulator melanocortin receptor 4 (MC4R) promotes its constitutive exit from cilia.PLoS biology · 2025Article
- Treatment of Hypothalamic Obesity With GLP-1 Analogs.Journal of the Endocrine Society · 2024Review
- Circadian cilia transcriptome in mouse brain across physiological and pathological states.Molecular brain · 2024Article
- A Comprehensive Review of Syndromic Forms of Obesity: Genetic Etiology, Clinical Features and Molecular Diagnosis.Current obesity reports · 2024Review
- POMC Neuron BBSome Regulation of Body Weight is Independent of its Ciliary Function.Function (Oxford, England) · 2024Article
- Elderly rats fed with a high-fat high-sucrose diet developed sex-dependent metabolic syndrome regardless of long-term metformin and liraglutide treatment.Frontiers in endocrinology · 2023Article
- Mechanisms of Weight Control by Primary Cilia.Molecules and cells · 2022Review
- Pyrogallol-Phloroglucinol-6, 6-Bieckol Restored Primary Cilia Length, Which Was Decreased by High-Fat Diet in Visceral Adipose Tissue, and Decreased Adipogenesis.International journal of endocrinology · 2022Article
- Large-scale analysis reveals spatiotemporal circadian patterns of cilia transcriptomes in the primate brain.Journal of neuroscience research · 2021Article
- Hypothalamic primary cilium: A hub for metabolic homeostasis.Experimental & molecular medicine · 2021Review
- Melanocortin 4 receptor signals at the neuronal primary cilium to control food intake and body weight.The Journal of clinical investigation · 2021Article
- Artificial Intelligence Approaches to Assessing Primary Cilia.Journal of visualized experiments : JoVE · 2021Article
- A mouse model of BBS identifies developmental and homeostatic effects of BBS5 mutation and identifies novel pituitary abnormalities.Human molecular genetics · 2021Article
- Cilia signaling and obesity.Seminars in cell & developmental biology · 2021Review
- Ventromedial hypothalamic primary cilia control energy and skeletal homeostasis.The Journal of clinical investigation · 2021Article
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Authors and funding
9 authors at 2 institutions in 2 countries.
Funding
Abstract
Although primary cilia are well established as important sensory and signaling structures, their function in most tissues remains unknown. Obesity is a feature associated with some syndromes of cilia dysfunction, such as Bardet-Biedl syndrome (BBS) and Alström syndrome, as well as in several cilia mutant mouse models. Recent data indicate that obesity in BBS mutant mice is due to defects in leptin receptor trafficking and leptin resistance. Furthermore, induction of cilia loss in leptin-responsive proopiomelanocortin neurons results in obesity, implicating cilia on hypothalamic neurons in regulating feeding behavior. Here, we directly test the importance of the cilium as a mediator of the leptin response. In contrast to the current dogma, a longitudinal study of conditional Ift88 cilia mutant mice under different states of adiposity indicates that leptin resistance is present only when mutants are obese. Our studies show that caloric restriction leads to an altered anticipatory feeding behavior that temporarily abrogates the anorectic actions of leptin despite normalized circulating leptin levels. Interestingly, preobese Bbs4 mutant mice responded to the anorectic effects of leptin and did not display other phenotypes associated with defective leptin signaling. Furthermore, thermoregulation and activity measurements in cilia mutant mice are inconsistent with phenotypes previously observed in leptin deficient ob/ob mice. Collectively, these data indicate that cilia are not directly involved in leptin responses and that a defect in the leptin signaling axis is not the initiating event leading to hyperphagia and obesity associated with cilia dysfunction.
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