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Grey matter volume and cortical structure in Prader-Willi syndrome compared to typically developing young adults.
Neuroimage Clin 2018; 17:899-909NC

Abstract

Prader-Willi syndrome (PWS) is a neurodevelopmental disorder of genomic imprinting, presenting with a characteristic overeating disorder, mild to moderate intellectual disability, and a variable range of social and behavioral difficulties. Consequently, widespread alterations in neural structure and developmental and maturational trajectory would be expected. To date, there have been few quantitative and systematic studies of brain morphology in PWS, although alterations of volume and of cortical organisation have been reported. This study aimed to investigate, in detail, the structure of grey matter and cortex in the brain in a sample of young adults with PWS in a well-matched case-controlled analysis. 20 young adults with PWS, aged 19-27 years, underwent multiparameter mapping magnetic resonance imaging sequences, from which measures of grey matter volume, cortical thickness and magnetisation transfer saturation, as a proxy measure of myelination, were examined. These variables were investigated in comparison to a control group of 40 typically developing young adults, matched for age and sex. A voxel-based morphometry analysis identified large and widespread bilateral clusters of both increased and decreased grey matter volume in the brain in PWS. In particular, widespread areas of increased volume encompassed parts of the prefrontal cortex, especially medially, the majority of the cingulate cortices, from anterior to posterior aspects, insula cortices, and areas of the parietal and temporal cortices. Increased volume was also reported in the caudate, putamen and thalamus. The most ventromedial prefrontal areas, in contrast, showed reduced volume, as did the parts of the medial temporal lobe, bilateral temporal poles, and a small cluster in the right lateral prefrontal cortex. Analysis of cortical structure revealed that areas of increased volume in the PWS group were largely driven by greater cortical thickness. Conversely, analysis of myelin content using magnetisation transfer saturation indicated that myelination of the cortex was broadly similar in the PWS and control groups, with the exception of highly localised areas, including the insula. The bilateral nature of these abnormalities suggests a systemic biological cause, with possible developmental and maturational mechanisms discussed, and may offer insight into the contribution of imprinted genes to neural development.

Authors+Show Affiliations

Department of Psychiatry, University of Cambridge, Cambridge, UK.Department of Psychiatry, University of Cambridge, Cambridge, UK.Department of Psychiatry, University of Cambridge, Cambridge, UK.Department of Psychiatry, University of Cambridge, Cambridge, UK. Cambridgeshire and Peterborough NHS Foundation Trust, UK. National Institute for Health Research (NIHR) Collaborations for Leadership in Applied Health Care Research and Care (CLAHRC), East of England, UK.

Pub Type(s)

Journal Article
Research Support, Non-U.S. Gov't

Language

eng

PubMed ID

29527494

Citation

Manning, Katherine E., et al. "Grey Matter Volume and Cortical Structure in Prader-Willi Syndrome Compared to Typically Developing Young Adults." NeuroImage. Clinical, vol. 17, 2018, pp. 899-909.
Manning KE, Tait R, Suckling J, et al. Grey matter volume and cortical structure in Prader-Willi syndrome compared to typically developing young adults. Neuroimage Clin. 2018;17:899-909.
Manning, K. E., Tait, R., Suckling, J., & Holland, A. J. (2018). Grey matter volume and cortical structure in Prader-Willi syndrome compared to typically developing young adults. NeuroImage. Clinical, 17, pp. 899-909. doi:10.1016/j.nicl.2017.12.027.
Manning KE, et al. Grey Matter Volume and Cortical Structure in Prader-Willi Syndrome Compared to Typically Developing Young Adults. Neuroimage Clin. 2018;17:899-909. PubMed PMID: 29527494.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Grey matter volume and cortical structure in Prader-Willi syndrome compared to typically developing young adults. AU - Manning,Katherine E, AU - Tait,Roger, AU - Suckling,John, AU - Holland,Anthony J, Y1 - 2017/12/20/ PY - 2017/10/30/received PY - 2017/12/14/revised PY - 2017/12/19/accepted PY - 2018/3/13/entrez PY - 2018/3/13/pubmed PY - 2019/1/31/medline KW - ACC, anterior cingulate cortex KW - ANTS, Advanced Normalisation Tools Software KW - BMI, body mass index KW - CamBA, Cambridge Brain Analysis software KW - Cortical thickness KW - FA, flip angle KW - GLM, general linear model KW - GM, grey matter KW - Genomic imprinting KW - Grey matter KW - IQ, intelligence quotient KW - MPM, multiparameter mapping KW - MRI, magnetic resonance imaging KW - MT, magnetisation transfer KW - Multiparameter mapping KW - Myelination KW - NHS, National Health Service KW - NSPN, NeuroScience in Psychiatry Network KW - OFC, orbitofrontal cortex KW - PD, proton density KW - PFC, prefrontal cortex KW - PWS, Prader-Willi syndrome KW - PWSA UK, Prader-Willi Syndrome Association UK KW - Prader-Willi syndrome KW - TE, echo time KW - TIV, total intracranial volume KW - TR, repetition time KW - UPD, uniparental disomy KW - WM, white matter SP - 899 EP - 909 JF - NeuroImage. Clinical JO - Neuroimage Clin VL - 17 N2 - Prader-Willi syndrome (PWS) is a neurodevelopmental disorder of genomic imprinting, presenting with a characteristic overeating disorder, mild to moderate intellectual disability, and a variable range of social and behavioral difficulties. Consequently, widespread alterations in neural structure and developmental and maturational trajectory would be expected. To date, there have been few quantitative and systematic studies of brain morphology in PWS, although alterations of volume and of cortical organisation have been reported. This study aimed to investigate, in detail, the structure of grey matter and cortex in the brain in a sample of young adults with PWS in a well-matched case-controlled analysis. 20 young adults with PWS, aged 19-27 years, underwent multiparameter mapping magnetic resonance imaging sequences, from which measures of grey matter volume, cortical thickness and magnetisation transfer saturation, as a proxy measure of myelination, were examined. These variables were investigated in comparison to a control group of 40 typically developing young adults, matched for age and sex. A voxel-based morphometry analysis identified large and widespread bilateral clusters of both increased and decreased grey matter volume in the brain in PWS. In particular, widespread areas of increased volume encompassed parts of the prefrontal cortex, especially medially, the majority of the cingulate cortices, from anterior to posterior aspects, insula cortices, and areas of the parietal and temporal cortices. Increased volume was also reported in the caudate, putamen and thalamus. The most ventromedial prefrontal areas, in contrast, showed reduced volume, as did the parts of the medial temporal lobe, bilateral temporal poles, and a small cluster in the right lateral prefrontal cortex. Analysis of cortical structure revealed that areas of increased volume in the PWS group were largely driven by greater cortical thickness. Conversely, analysis of myelin content using magnetisation transfer saturation indicated that myelination of the cortex was broadly similar in the PWS and control groups, with the exception of highly localised areas, including the insula. The bilateral nature of these abnormalities suggests a systemic biological cause, with possible developmental and maturational mechanisms discussed, and may offer insight into the contribution of imprinted genes to neural development. SN - 2213-1582 UR - https://www.unboundmedicine.com/medline/citation/29527494/Grey_matter_volume_and_cortical_structure_in_Prader_Willi_syndrome_compared_to_typically_developing_young_adults_ L2 - https://linkinghub.elsevier.com/retrieve/pii/S2213-1582(17)30332-7 DB - PRIME DP - Unbound Medicine ER -