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The Developmental Trajectory of Self-Injurious Behaviours in Individuals with Prader Willi Syndrome, Autism Spectrum Disorder and Intellectual Disability

Zebrafish Models of Prader-Willi Syndrome: Fast Track to Pharmacotherapeutics

Department of Oncology, Georgetown University Medical Center, 4000 Reservoir Road, N.W., Washington, DC 20057, USA
Author to whom correspondence should be addressed.
Academic Editor: Danny Camfferman
Received: 31 December 2015 / Revised: 27 February 2016 / Accepted: 1 March 2016 / Published: 8 March 2016
(This article belongs to the Special Issue Prader-Willi Syndrome)
Prader-Willi syndrome (PWS) is a rare genetic neurodevelopmental disorder characterized by an insatiable appetite, leading to chronic overeating and obesity. Additional features include short stature, intellectual disability, behavioral problems and incomplete sexual development. Although significant progress has been made in understanding the genetic basis of PWS, the mechanisms underlying the pathogenesis of the disorder remain poorly understood. Treatment for PWS consists mainly of palliative therapies; curative therapies are sorely needed. Zebrafish, Danio rerio, represent a promising way forward for elucidating physiological problems such as obesity and identifying new pharmacotherapeutic options for PWS. Over the last decade, an increased appreciation for the highly conserved biology among vertebrates and the ability to perform high-throughput drug screening has seen an explosion in the use of zebrafish for disease modeling and drug discovery. Here, we review recent advances in developing zebrafish models of human disease. Aspects of zebrafish genetics and physiology that are relevant to PWS will be discussed, and the advantages and disadvantages of zebrafish models will be contrasted with current animal models for this syndrome. Finally, we will present a paradigm for drug screening in zebrafish that is potentially the fastest route for identifying and delivering curative pharmacotherapies to PWS patients. View Full-Text
Keywords: neuroendocrine; oxytocin; zebrafish neuroendocrine; oxytocin; zebrafish
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MDPI and ACS Style

Spikol, E.D.; Laverriere, C.E.; Robnett, M.; Carter, G.; Wolfe, E.; Glasgow, E. Zebrafish Models of Prader-Willi Syndrome: Fast Track to Pharmacotherapeutics. Diseases 2016, 4, 13.

AMA Style

Spikol ED, Laverriere CE, Robnett M, Carter G, Wolfe E, Glasgow E. Zebrafish Models of Prader-Willi Syndrome: Fast Track to Pharmacotherapeutics. Diseases. 2016; 4(1):13.

Chicago/Turabian Style

Spikol, Emma D., Caroline E. Laverriere, Maya Robnett, Gabriela Carter, Erin Wolfe, and Eric Glasgow. 2016. "Zebrafish Models of Prader-Willi Syndrome: Fast Track to Pharmacotherapeutics" Diseases 4, no. 1: 13.

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