Alteration of brain nuclei in obese children with and without Prader-Willi syndrome

Introduction: Prader-Willi syndrome (PWS) is a multisystem genetic imprinting disorder mainly characterized by hyperphagia and childhood obesity. Extensive structural alterations are expected in PWS patients, and their influence on brain nuclei should be early and profound. To date, few studies have...

Full description

Bibliographic Details
Main Authors: Ning Wu, Huan Yu, Mingze Xu
Format: Article
Language:English
Published: Frontiers Media S.A. 2022-11-01
Series:Frontiers in Neuroinformatics
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fninf.2022.1032636/full
_version_ 1811219870923096064
author Ning Wu
Huan Yu
Mingze Xu
author_facet Ning Wu
Huan Yu
Mingze Xu
author_sort Ning Wu
collection DOAJ
description Introduction: Prader-Willi syndrome (PWS) is a multisystem genetic imprinting disorder mainly characterized by hyperphagia and childhood obesity. Extensive structural alterations are expected in PWS patients, and their influence on brain nuclei should be early and profound. To date, few studies have investigated brain nuclei in children with PWS, although functional and structural alterations of the cortex have been reported widely.Methods: In the current study, we used T1-weighted magnetic resonance imaging to investigate alterations in brain nuclei by three automated analysis methods: shape analysis to evaluate the shape of 14 cerebral nuclei (bilateral thalamus, caudate, putamen, globus pallidus, hippocampus, amygdala, and nucleus accumbens), automated segmentation methods integrated in Freesurfer 7.2.0 to investigate the volume of hypothalamic subregions, and region of interest-based analysis to investigate the volume of deep cerebellar nuclei (DCN). Twelve age- and sex-matched children with PWS, 18 obese children without PWS (OB) and 18 healthy controls participated in this study.Results: Compared with control and OB individuals, the PWS group exhibited significant atrophy in the bilateral thalamus, pallidum, hippocampus, amygdala, nucleus accumbens, right caudate, bilateral hypothalamus (left anterior-inferior, bilateral posterior, and bilateral tubular inferior subunits) and bilateral DCN (dentate, interposed, and fastigial nuclei), whereas no significant difference was found between the OB and control groups.Discussion: Based on our evidence, we suggested that alterations in brain nuclei influenced by imprinted genes were associated with clinical manifestations of PWS, such as eating disorders, cognitive disability and endocrine abnormalities, which were distinct from the neural mechanisms of obese children.
first_indexed 2024-04-12T07:33:11Z
format Article
id doaj.art-09d5d362b3a54f6e9c43c6d56a3e78fd
institution Directory Open Access Journal
issn 1662-5196
language English
last_indexed 2024-04-12T07:33:11Z
publishDate 2022-11-01
publisher Frontiers Media S.A.
record_format Article
series Frontiers in Neuroinformatics
spelling doaj.art-09d5d362b3a54f6e9c43c6d56a3e78fd2022-12-22T03:42:01ZengFrontiers Media S.A.Frontiers in Neuroinformatics1662-51962022-11-011610.3389/fninf.2022.10326361032636Alteration of brain nuclei in obese children with and without Prader-Willi syndromeNing Wu0Huan Yu1Mingze Xu2Department of Medical Imaging, Yanjing Medical College, Capital Medical University, Beijing, ChinaDepartment of Radiology, Liangxiang Hospital, Beijing, ChinaCenter for MRI Research, Academy for Advanced Interdisciplinary Studies, Peking University, Beijing, ChinaIntroduction: Prader-Willi syndrome (PWS) is a multisystem genetic imprinting disorder mainly characterized by hyperphagia and childhood obesity. Extensive structural alterations are expected in PWS patients, and their influence on brain nuclei should be early and profound. To date, few studies have investigated brain nuclei in children with PWS, although functional and structural alterations of the cortex have been reported widely.Methods: In the current study, we used T1-weighted magnetic resonance imaging to investigate alterations in brain nuclei by three automated analysis methods: shape analysis to evaluate the shape of 14 cerebral nuclei (bilateral thalamus, caudate, putamen, globus pallidus, hippocampus, amygdala, and nucleus accumbens), automated segmentation methods integrated in Freesurfer 7.2.0 to investigate the volume of hypothalamic subregions, and region of interest-based analysis to investigate the volume of deep cerebellar nuclei (DCN). Twelve age- and sex-matched children with PWS, 18 obese children without PWS (OB) and 18 healthy controls participated in this study.Results: Compared with control and OB individuals, the PWS group exhibited significant atrophy in the bilateral thalamus, pallidum, hippocampus, amygdala, nucleus accumbens, right caudate, bilateral hypothalamus (left anterior-inferior, bilateral posterior, and bilateral tubular inferior subunits) and bilateral DCN (dentate, interposed, and fastigial nuclei), whereas no significant difference was found between the OB and control groups.Discussion: Based on our evidence, we suggested that alterations in brain nuclei influenced by imprinted genes were associated with clinical manifestations of PWS, such as eating disorders, cognitive disability and endocrine abnormalities, which were distinct from the neural mechanisms of obese children.https://www.frontiersin.org/articles/10.3389/fninf.2022.1032636/fullchildhood obesitygenetic imprintingPrader-Willi syndromebrain nucleishape analysishypothalamus
spellingShingle Ning Wu
Huan Yu
Mingze Xu
Alteration of brain nuclei in obese children with and without Prader-Willi syndrome
Frontiers in Neuroinformatics
childhood obesity
genetic imprinting
Prader-Willi syndrome
brain nuclei
shape analysis
hypothalamus
title Alteration of brain nuclei in obese children with and without Prader-Willi syndrome
title_full Alteration of brain nuclei in obese children with and without Prader-Willi syndrome
title_fullStr Alteration of brain nuclei in obese children with and without Prader-Willi syndrome
title_full_unstemmed Alteration of brain nuclei in obese children with and without Prader-Willi syndrome
title_short Alteration of brain nuclei in obese children with and without Prader-Willi syndrome
title_sort alteration of brain nuclei in obese children with and without prader willi syndrome
topic childhood obesity
genetic imprinting
Prader-Willi syndrome
brain nuclei
shape analysis
hypothalamus
url https://www.frontiersin.org/articles/10.3389/fninf.2022.1032636/full
work_keys_str_mv AT ningwu alterationofbrainnucleiinobesechildrenwithandwithoutpraderwillisyndrome
AT huanyu alterationofbrainnucleiinobesechildrenwithandwithoutpraderwillisyndrome
AT mingzexu alterationofbrainnucleiinobesechildrenwithandwithoutpraderwillisyndrome