Assessment of Hyperphagia in Prader‐Willi Syndrome

Dykens, Elisabeth M.; Maxwell, Melissa A.; Pantino, Elizabeth; Kossler, Rebecca; Roof, Elizabeth

doi:10.1038/oby.2007.216

Cited by 162 publications

(159 citation statements)

References 37 publications

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“…Validated questionnaires were used in the study to evaluate the effects of IN-OT on behavior, socialization, and anxiety, including: Aberrant Behavior Checklist (ABC); Repetitive Behavior Scale-Revised (RBS-R) (Lam and Aman, 2007); and Social Responsiveness Scale (SRS) (Constantino et al, 2003). Additionally, the Hyperphagia Questionnaire (HQ) (Dykens, Maxwell, Pantino, Kossler, & Roof, 2007) was administered to determine the effects on appetite in PWS. All questionnaires were modified to reflect that the respondent was answering the questions for the past 24 hr only, as they were used repeatedly during the course of the study.…”

Section: | Materials and Methodsmentioning

confidence: 99%

Oxytocin treatment in children with Prader–Willi syndrome: A double‐blind, placebo‐controlled, crossover study

Miller

Tamura

Butler

et al. 2017

American J of Med Genetics Pt A

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Prader–Willi syndrome (PWS) is a rare, complex multisystem genetic disorder which includes hypothalamic dysfunction, hyperphagia, cognitive and behavioral problems, increased anxiety, and compulsive behaviors. Individuals with PWS have a deficit of oxytocin producing neurons in the paraventricular nucleus of the hypothalamus. Oxytocin plays a role in regulation of feeding behaviors, social interactions, and emotional reactivity, which are all issues that significantly affect the quality of life for individuals with this syndrome. We performed a double-blind, placebo-controlled, crossover study in 24 children with PWS at three academic institutions using 5 days of intranasal oxytocin (IN-OT) or 5 days of intranasal placebo spray, followed by a 4 week washout period, and then patients returned for 5 days of treatment with the alternate source. Questionnaires, including the Aberrant Behavior Checklist, Social Responsiveness Scale, Repetitive Behavior Scale − Revised, and the Hyperphagia Questionnaire, as well as Clinical Global Impression scales were administered. Blood testing for sodium, potassium, and glucose levels on days 2, 4, and 6, and a 24 hr diet recall. All scales factor improvement from Day 3 to Day 6 favored oxytocin over placebo. No single factor showed a statistically significant difference (P < 0.05) between groups at Day 6. The drug effect appeared to be diminished at Day 14. There was no evidence of a difference between oxytocin and placebo in safety lab parameters, 60 min post dose vital signs, weight, or diet parameters. The results from this study suggest that low dose intranasal oxytocin is safe for individuals with PWS and may result in reduction in appetite drive, and improvements in socialization, anxiety, and repetitive behaviors. Further, long-term studies with a larger population of participants are necessary to confirm these findings. The results of this study are encouraging that oxytocin may be a safe and effective treatment for many of the issues that negatively impact individuals with PWS.

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Section: | Materials and Methodsmentioning

confidence: 99%

Oxytocin treatment in children with Prader–Willi syndrome: A double‐blind, placebo‐controlled, crossover study

Miller

Tamura

Butler

et al. 2017

American J of Med Genetics Pt A

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“…Therefore, it is common for children with PWS to be obese. In fact, it is estimated that the rate of obesity among individuals with PWS is around 79% (Dykens, Maxwell, Pantino, Kossler, & Roof, 2007). Although a rare disorder in children, it is the most common genetically identified cause of obesity and has been observed in all ethnic and racial groups with the highest prevalence among Whites (Butler, Hanchett, & Thompson, 2006).…”

Section: Prader-willi Syndromementioning

confidence: 99%

Obesity rates in special populations of children and potential interventions

Holcomb

Pufpaff

McIntosh

2009

Psychology in the Schools

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Two human immunodeficiency virus‐infected patients who needed etoposide therapy to control exacerbations of Castleman disease received four infusions of rituximab. Clinical and virologic relapses with increased blood human herpesvirus‐8 DNA levels occurred in both patients 4 and 24 weeks later and were associated with a worsening of Kaposi sarcoma. Am. J. Hematol. 79:337–339, 2005. © 2005 Wiley‐Liss, Inc.

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“…Russell and Oliver (2003) Roof et al, 2000), very few studies have used such reports due to issues with reliability and validity (Dykens et al, 2007).…”

Section: Assessment Of Internal States Within Individuals With Intellmentioning

confidence: 99%

The expression and assessment of emotions and internal states in individuals with severe or profound intellectual disabilities

Adams¹,

Oliver²

2011

Clinical Psychology Review

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The expression of emotions and internal states by individuals with severe or profound intellectual disabilities is a comparatively under-researched area. Comprehensive or standardised methods of assessing or understanding the emotions and internal states within this population, whose ability to communicate is significantly compromised, do not exist.The literature base will be discussed and compared to that within the general population.Methods of assessing broader internal states, notably depression, anxiety, and pain within severe or profound intellectual disabilities are also addressed. Finally, this review will examine methods of assessing internal states within genetic syndromes, including hunger, social anxiety and happiness within Prader-Willi, Fragile-X and Angelman syndrome. This will then allow for the identification of robust methodologies used in assessing the expression of these internal states, some of which may be useful when considering how to assess emotions within individuals with intellectual disabilities.

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Assessment of Hyperphagia in Prader‐Willi Syndrome

Cited by 162 publications

References 37 publications

Oxytocin treatment in children with Prader–Willi syndrome: A double‐blind, placebo‐controlled, crossover study

Oxytocin treatment in children with Prader–Willi syndrome: A double‐blind, placebo‐controlled, crossover study

Obesity rates in special populations of children and potential interventions

The expression and assessment of emotions and internal states in individuals with severe or profound intellectual disabilities

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