Cargando…

Altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with Prader-Willi syndrome

BACKGROUND: Prader-Willi syndrome (PWS) is a complex neurodevelopmental disorder, characterized by endocrine problems and hyperphagia, indicating hypothalamic-pituitary dysfunction. However, few studies have explored the underlying neurobiology of the hypothalamus and its functional connectivity wit...

Descripción completa

Detalles Bibliográficos
Autores principales: Lukoshe, Akvile, van Dijk, Suzanne E., van den Bosch, Gerbrich E., van der Lugt, Aad, White, Tonya, Hokken-Koelega, Anita C.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5356363/
https://www.ncbi.nlm.nih.gov/pubmed/28331554
http://dx.doi.org/10.1186/s11689-017-9188-7
_version_ 1782515818707288064
author Lukoshe, Akvile
van Dijk, Suzanne E.
van den Bosch, Gerbrich E.
van der Lugt, Aad
White, Tonya
Hokken-Koelega, Anita C.
author_facet Lukoshe, Akvile
van Dijk, Suzanne E.
van den Bosch, Gerbrich E.
van der Lugt, Aad
White, Tonya
Hokken-Koelega, Anita C.
author_sort Lukoshe, Akvile
collection PubMed
description BACKGROUND: Prader-Willi syndrome (PWS) is a complex neurodevelopmental disorder, characterized by endocrine problems and hyperphagia, indicating hypothalamic-pituitary dysfunction. However, few studies have explored the underlying neurobiology of the hypothalamus and its functional connectivity with other brain regions. Thus, the aim of this study was to examine the anatomical differences of the hypothalamus, mammillary bodies, and pituitary gland as well as resting state functional connectivity of the hypothalamus in children with PWS. METHODS: Twenty-seven children with PWS (13 DEL, 14 mUPD) and 28 typically developing children were included. Manual segmentations by a blinded investigator were performed to determine the volumes of the hypothalamus, mammillary bodies, and pituitary gland. In addition, brain-wide functional connectivity analysis was performed using the obtained masks of the hypothalamus. RESULTS: Children with PWS showed altered resting state functional connectivity between hypothalamus and right and left lateral occipital complex, compared to healthy controls. In addition, children with PWS had on average a 50% smaller pituitary volume, an irregular shape of the pituitary, and a longer pituitary stalk. Pituitary volume did not increase in volume during puberty in PWS. No volumetric differences in the hypothalamus and mammillary bodies were found. In all subjects, the posterior pituitary bright spot was observed. CONCLUSIONS: We report altered functional hypothalamic connectivity with lateral occipital complexes in both hemispheres, which are implicated in response to food and reward system, and absence of connectivity might therefore at least partially contribute to the preoccupation with food in PWS.
format Online
Article
Text
id pubmed-5356363
institution National Center for Biotechnology Information
language English
publishDate 2017
publisher BioMed Central
record_format MEDLINE/PubMed
spelling pubmed-53563632017-03-22 Altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with Prader-Willi syndrome Lukoshe, Akvile van Dijk, Suzanne E. van den Bosch, Gerbrich E. van der Lugt, Aad White, Tonya Hokken-Koelega, Anita C. J Neurodev Disord Research BACKGROUND: Prader-Willi syndrome (PWS) is a complex neurodevelopmental disorder, characterized by endocrine problems and hyperphagia, indicating hypothalamic-pituitary dysfunction. However, few studies have explored the underlying neurobiology of the hypothalamus and its functional connectivity with other brain regions. Thus, the aim of this study was to examine the anatomical differences of the hypothalamus, mammillary bodies, and pituitary gland as well as resting state functional connectivity of the hypothalamus in children with PWS. METHODS: Twenty-seven children with PWS (13 DEL, 14 mUPD) and 28 typically developing children were included. Manual segmentations by a blinded investigator were performed to determine the volumes of the hypothalamus, mammillary bodies, and pituitary gland. In addition, brain-wide functional connectivity analysis was performed using the obtained masks of the hypothalamus. RESULTS: Children with PWS showed altered resting state functional connectivity between hypothalamus and right and left lateral occipital complex, compared to healthy controls. In addition, children with PWS had on average a 50% smaller pituitary volume, an irregular shape of the pituitary, and a longer pituitary stalk. Pituitary volume did not increase in volume during puberty in PWS. No volumetric differences in the hypothalamus and mammillary bodies were found. In all subjects, the posterior pituitary bright spot was observed. CONCLUSIONS: We report altered functional hypothalamic connectivity with lateral occipital complexes in both hemispheres, which are implicated in response to food and reward system, and absence of connectivity might therefore at least partially contribute to the preoccupation with food in PWS. BioMed Central 2017-02-21 /pmc/articles/PMC5356363/ /pubmed/28331554 http://dx.doi.org/10.1186/s11689-017-9188-7 Text en © The Author(s). 2017 Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Research
Lukoshe, Akvile
van Dijk, Suzanne E.
van den Bosch, Gerbrich E.
van der Lugt, Aad
White, Tonya
Hokken-Koelega, Anita C.
Altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with Prader-Willi syndrome
title Altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with Prader-Willi syndrome
title_full Altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with Prader-Willi syndrome
title_fullStr Altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with Prader-Willi syndrome
title_full_unstemmed Altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with Prader-Willi syndrome
title_short Altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with Prader-Willi syndrome
title_sort altered functional resting-state hypothalamic connectivity and abnormal pituitary morphology in children with prader-willi syndrome
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5356363/
https://www.ncbi.nlm.nih.gov/pubmed/28331554
http://dx.doi.org/10.1186/s11689-017-9188-7
work_keys_str_mv AT lukosheakvile alteredfunctionalrestingstatehypothalamicconnectivityandabnormalpituitarymorphologyinchildrenwithpraderwillisyndrome
AT vandijksuzannee alteredfunctionalrestingstatehypothalamicconnectivityandabnormalpituitarymorphologyinchildrenwithpraderwillisyndrome
AT vandenboschgerbriche alteredfunctionalrestingstatehypothalamicconnectivityandabnormalpituitarymorphologyinchildrenwithpraderwillisyndrome
AT vanderlugtaad alteredfunctionalrestingstatehypothalamicconnectivityandabnormalpituitarymorphologyinchildrenwithpraderwillisyndrome
AT whitetonya alteredfunctionalrestingstatehypothalamicconnectivityandabnormalpituitarymorphologyinchildrenwithpraderwillisyndrome
AT hokkenkoelegaanitac alteredfunctionalrestingstatehypothalamicconnectivityandabnormalpituitarymorphologyinchildrenwithpraderwillisyndrome