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Hypothalamic loss of Snord116 and Prader-Willi syndrome hyperphagia: the buck stops here?
Juan A. Rodriguez, Jeffrey M. Zigman
Juan A. Rodriguez, Jeffrey M. Zigman
Published January 29, 2018
Citation Information: J Clin Invest. 2018;128(3):900-902. https://doi.org/10.1172/JCI99725.
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Commentary

Hypothalamic loss of Snord116 and Prader-Willi syndrome hyperphagia: the buck stops here?

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Abstract

Hyperphagia and obesity are the best-known manifestations of Prader-Willi syndrome (PWS) and are responsible for most of the overall morbidity and mortality associated with the disease. Yet these PWS symptoms remain poorly understood and without effective pharmacologic therapies. Mouse models attempting to recapitulate both the genetic alterations and marked hyperphagia plus obesity of PWS have been enigmatic, leading to skepticism about the use of mouse models to investigate PWS. In this issue of the JCI, Polex-Wolf and colleagues challenge the skeptics by successfully inducing hyperphagia following bilateral mediobasal hypothalamic deletion of the Snord116 gene from adult mice. Obesity also resulted, although only in a subset of mice. While this approach represents an exciting advance, highlighting a pathologic effect of loss of mediobasal hypothalamic Snord116 expression on the development of PWS’s hallmark symptoms, the variability in the body-weight and body composition responses to this site-selective gene deletion raises several questions.

Authors

Juan A. Rodriguez, Jeffrey M. Zigman

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