ArticleJournal of neurodevelopmental disorders2013
Divergent structural brain abnormalities between different genetic subtypes of children with Prader-Willi syndrome.
Article in Journal of neurodevelopmental disorders, 2013. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 23 papers.
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Who cites it
23 citing papers in PubMed, 45 citations in OpenAlex.
- Effect of cessation of GH treatment on cognition during transition phase in Prader-Willi syndrome: results of a 2-year crossover GH trial.Orphanet journal of rare diseases · 2016Trial
- Aberrant local and global neural activation patterns in pediatric Prader-Willi syndrome.Frontiers in neuroscience · 2026Article
- 14 and 6 Hz like spike wave activity is a common finding in young patients with Prader-Willi syndrome.Journal of clinical sleep medicine : JCSM : official publication of the American Academy of Sleep Medicine · 2024Article
- Early psychomotor development and growth hormone therapy in children with Prader-Willi syndrome: a review.European journal of pediatrics · 2024Review
- Progress in Brain Magnetic Resonance Imaging of Individuals with Prader-Willi Syndrome.Journal of clinical medicine · 2023Review
- Differential volume reductions in the subcortical, limbic, and brainstem structures associated with behavior in Prader-Willi syndrome.Scientific reports · 2022Article
- Health Problems in Individuals With PWS Are Associated With Lower Quality of Life for Their Parents: A Snapshot in the Brazilian Population.Frontiers in pediatrics · 2022Article
- Alteration of brain nuclei in obese children with and without Prader-Willi syndrome.Frontiers in neuroinformatics · 2022Article
- A rare familial rearrangement of chromosomes 9 and 15 associated with intellectual disability: a clinical and molecular study.Molecular cytogenetics · 2021Article
- Cerebellar Dysfunction in Adults with Prader Willi Syndrome.Journal of clinical medicine · 2021Article
- Behavioral features in Prader-Willi syndrome (PWS): consensus paper from the International PWS Clinical Trial Consortium.Journal of neurodevelopmental disorders · 2021Review
- The Diagnostic Journey of a Patient with Prader-Willi-Like Syndrome and a Unique HomozygousGenes · 2021Review
- Application of 7 tesla magnetic resonance imaging for pediatric neurological disorders: Early clinical experience.Journal of clinical imaging science · 2021Article
- Observational
- Abnormal Cortical Thickness Is Associated With Deficits in Social Cognition in Patients With Myotonic Dystrophy Type 1.Frontiers in neurology · 2020Article
- Sparse multiple factor analysis to integrate genetic data, neuroimaging features, and attention-deficit/hyperactivity disorder domains.International journal of methods in psychiatric research · 2018Article
- Neuroanatomical and molecular correlates of cognitive and behavioural outcomes in hypogonadal males.Metabolic brain disease · 2018Review
- Grey matter volume and cortical structure in Prader-Willi syndrome compared to typically developing young adults.NeuroImage. Clinical · 2018Article
- Autism spectrum disorder: neuropathology and animal models.Acta neuropathologica · 2017Review
- Aberrant White Matter Microstructure in Children and Adolescents With the Subtype of Prader-Willi Syndrome at High Risk for Psychosis.Schizophrenia bulletin · 2017Article
Corrections and comments
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Authors and funding
5 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundPrader-Willi syndrome (PWS) is a complex neurogenetic disorder with symptoms that indicate not only hypothalamic, but also a global, central nervous system (CNS) dysfunction. However, little is known about developmental differences in brain structure in children with PWS. Thus, our aim was to investigate global brain morphology in children with PWS, including the comparison between different genetic subtypes of PWS. In addition, we performed exploratory cortical and subcortical focal analyses.
methodsHigh resolution structural magnetic resonance images were acquired in 20 children with genetically confirmed PWS (11 children carrying a deletion (DEL), 9 children with maternal uniparental disomy (mUPD)), and compared with 11 age- and gender-matched typically developing siblings as controls. Brain morphology measures were obtained using the FreeSurfer software suite.
resultsBoth children with DEL and mUPD showed smaller brainstem volume, and a trend towards smaller cortical surface area and white matter volume. Children with mUPD had enlarged lateral ventricles and larger cortical cerebrospinal fluid (CSF) volume. Further, a trend towards increased cortical thickness was found in children with mUPD. Children with DEL had a smaller cerebellum, and smaller cortical and subcortical grey matter volumes. Focal analyses revealed smaller white matter volumes in left superior and bilateral inferior frontal gyri, right cingulate cortex, and bilateral precuneus areas associated with the default mode network (DMN) in children with mUPD.
conclusionsChildren with PWS show signs of impaired brain growth. Those with mUPD show signs of early brain atrophy. In contrast, children with DEL show signs of fundamentally arrested, although not deviant brain development and presented few signs of cortical atrophy. Our results of global brain measurements suggest divergent neurodevelopmental patterns in children with DEL and mUPD.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.