TY - JOUR
T1 - Regional cerebral blood flow and abnormal eating behavior in Prader-Willi syndrome
AU - Ogura, Kaeko
AU - Fujii, Toshikatsu
AU - Abe, Nobuhito
AU - Hosokai, Yoshiyuki
AU - Shinohara, Mayumi
AU - Fukuda, Hiroshi
AU - Mori, Etsuro
N1 - Funding Information:
We would like to express our gratitude to the individuals with PWS who participated in our study and their families and the members of the Japan PWS Society (Takenoko No Kai). We also thank Yoichi Sawada for helping us to recruit the control subjects and Makoto Saito for his assistance with data collection. This work was partly supported by a Grant-in-Aid for Scientific Research on Priority Areas, System Study on Higher-Order Brain Functions, from the Ministry of Education, Culture, Sports, Science and Technology (MEXT) of Japan ( 20020004 to E.M.). This work was also partially supported by the Global COE Program of the Basic & Translational Research Center for Global Brain Science, MEXT, Japan.
PY - 2013/5
Y1 - 2013/5
N2 - Background: Prader-Willi syndrome (PWS) is a genetically determined neurodevelopmental disorder and is generally regarded as a genetic model of obesity. Individuals with PWS exhibit behavioral symptoms including temper tantrums, rigid thinking, and compulsive behavior. The most striking feature of PWS is abnormal eating behavior, including hyperphagia, intense preoccupation with food, and incessant food seeking. To explore brain regions associated with the behavioral symptoms of PWS, we investigated differences in resting-state regional cerebral blood flow (rCBF) between individuals with PWS and healthy controls. Correlation analyses were also performed to examine the relationship between rCBF and altered eating behavior in PWS individuals. Methods: Twelve adults with PWS and 13 age- and gender-matched controls underwent resting-state single photon emission computerized tomography (SPECT) with N-isopropyl-p-[123I] iodoamphetamine (IMP). The rCBF data were analyzed on a voxel-by-voxel basis using SPM5 software. Results: The results demonstrated that compared with controls, individuals with PWS had significantly lower rCBF in the right thalamus, left insular cortex, bilateral lingual gyrus, and bilateral cerebellum. They had significantly higher rCBF in the right inferior frontal gyrus, left middle/inferior frontal gyrus (anterior and posterior clusters), and bilateral angular gyrus. Additionally, rCBF in the left insula, which was significantly lower in PWS individuals, was negatively correlated with the eating behavior severity score. Conclusions: These results suggest that specific brain regions, particularly the left insula, may be partly responsible for the behavioral symptoms in PWS.
AB - Background: Prader-Willi syndrome (PWS) is a genetically determined neurodevelopmental disorder and is generally regarded as a genetic model of obesity. Individuals with PWS exhibit behavioral symptoms including temper tantrums, rigid thinking, and compulsive behavior. The most striking feature of PWS is abnormal eating behavior, including hyperphagia, intense preoccupation with food, and incessant food seeking. To explore brain regions associated with the behavioral symptoms of PWS, we investigated differences in resting-state regional cerebral blood flow (rCBF) between individuals with PWS and healthy controls. Correlation analyses were also performed to examine the relationship between rCBF and altered eating behavior in PWS individuals. Methods: Twelve adults with PWS and 13 age- and gender-matched controls underwent resting-state single photon emission computerized tomography (SPECT) with N-isopropyl-p-[123I] iodoamphetamine (IMP). The rCBF data were analyzed on a voxel-by-voxel basis using SPM5 software. Results: The results demonstrated that compared with controls, individuals with PWS had significantly lower rCBF in the right thalamus, left insular cortex, bilateral lingual gyrus, and bilateral cerebellum. They had significantly higher rCBF in the right inferior frontal gyrus, left middle/inferior frontal gyrus (anterior and posterior clusters), and bilateral angular gyrus. Additionally, rCBF in the left insula, which was significantly lower in PWS individuals, was negatively correlated with the eating behavior severity score. Conclusions: These results suggest that specific brain regions, particularly the left insula, may be partly responsible for the behavioral symptoms in PWS.
KW - Chromosome 15q
KW - Eating behavior
KW - Insular cortex
KW - Neurodevelopmental disorder
KW - Obesity
KW - Regional cerebral blood flow
KW - SPECT
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U2 - 10.1016/j.braindev.2012.07.013
DO - 10.1016/j.braindev.2012.07.013
M3 - Article
C2 - 22921862
AN - SCOPUS:84875906604
SN - 0387-7604
VL - 35
SP - 427
EP - 434
JO - Brain and Development
JF - Brain and Development
IS - 5
ER -