Hyperghrelinemia in Prader-Willi syndrome begins in early infancy long before the onset of hyperphagia

Circulating total ghrelin levels are elevated in older children and adults with Prader‐Willi syndrome (PWS). However, the presence or absence of hyperghrelinemia in young children with PWS remains controversial. We hypothesized that a more robust way to analyze appetite‐regulating hormones in PWS wo...

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Bibliographic Details
Published in:American journal of medical genetics. Part A 2015-01, Vol.167A (1), p.69-79
Main Authors: Kweh, Frederick A., Miller, Jennifer L., Sulsona, Carlos R., Wasserfall, Clive, Atkinson, Mark, Shuster, Jonathan J., Goldstone, Anthony P., Driscoll, Daniel J.
Format: Article
Language:English
Subjects:
Age Distribution
Child
Child, Preschool
Fasting - blood
Female
ghrelin
Ghrelin - blood
Humans
hyperghrelinemia
hyperphagia
Hyperphagia - blood
Infant
Insulin Resistance
Male
nutritional phase
obesity
Obesity, Morbid - blood
Prader-Willi
Prader-Willi Syndrome - blood
Prader-Willi Syndrome - classification
Siblings
Young Adult
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Summary:Circulating total ghrelin levels are elevated in older children and adults with Prader‐Willi syndrome (PWS). However, the presence or absence of hyperghrelinemia in young children with PWS remains controversial. We hypothesized that a more robust way to analyze appetite‐regulating hormones in PWS would be by nutritional phases rather than age alone. Our objectives were to compare total serum ghrelin levels in children with PWS by nutritional phase as well as to compare total ghrelin levels in PWS (5 weeks to 21 years of age) to normal weight controls and individuals with early‐onset morbid obesity (EMO) without PWS. Fasting serum total ghrelin levels were measured in 60 subjects with PWS, 39 subjects with EMO of unknown etiology, and in 95 normal non‐obese sibling controls of PWS or EMO subjects (SibC) in this 12 year longitudinal study. Within PWS, total ghrelin levels were significantly (P 
ISSN:1552-4825
1552-4833
DOI:10.1002/ajmg.a.36810