Reduced Cortical Complexity in Children with Prader-Willi Syndrome and Its Association with Cognitive Impairment and Developmental Delay

Lukoshe, Akvile; Hokken-Koelega, Anita; Lugt, Aad van der; White, Tonya

doi:10.1371/journal.pone.0107320

Cited by 36 publications

(21 citation statements)

References 58 publications

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“…Specifically, the age × cognitive interaction effects provide evidence that the relationships between local cortical gyrification and sustained attention, inhibitory control, motor response variability, perceptual sensitivity, and both verbal and nonverbal conceptualization abilities differ at different ages during adolescence. These changing brain-behavior relationships were mostly but not exclusively seen in frontoparietal and temporoparietal brain regions where local gyrification values linearly decreased with age and where prior studies show links to individual differences in intellectual ability for healthy adults (Docherty et al, 2015; Gregory et al, 2016) and individuals with neurodevelopmental disorders (Lukoshe et al, 2014; Treble et al, 2013). Even though these interaction effects were found for all tests examined and in widespread frontoparietal cortices, statistical mediation analyses showed age-related changes in cortical folding causally influenced only motor response inhibition ability.…”

Section: Discussionmentioning

confidence: 86%

Adolescent maturation of the relationship between cortical gyrification and cognitive ability

2017

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There are changes to the degree of cortical folding from gestation through adolescence into young adulthood. Recent evidence suggests that degree of cortical folding is linked to individual differences in general cognitive ability in healthy adults. However, it is not yet known whether age-related cortical folding changes are related to maturation of specific cognitive abilities in adolescence. To address this, we examined the relationship between frontoparietal cortical folding as measured by a Freesurfer-derived local gyrification index (lGI) and performance on subtests from the Wechsler Abbreviated Scale of Intelligence and scores from Conner’s Continuous Performance Test-II in 241 healthy adolescents (ages 12–25 years). We hypothesized that age-related lGI changes in the frontoparietal cortex would contribute to cognitive development. A secondary goal was to explore if any gyrification-cognition relationships were either test-specific or sex-specific. Consistent with previous studies, our results showed a reduction of frontoparietal local gyrification with age. Also, as predicted, all cognitive test scores (i.e., Vocabulary, Matrix Reasoning, the CPT-II Commission, Omission, Variabiltiy, d’) showed age × cognitive ability interaction effects in frontoparietal and temporoparietal brain regions. Mediation analyses confirmed a causal role of age-related cortical folding changes only for CPT-II Commission errors. Taken together, the results support the functional significance of cortical folding, as well as provide the first evidence that cortical folding maturational changes play a role in cognitive development.

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Section: Discussionmentioning

confidence: 86%

Adolescent maturation of the relationship between cortical gyrification and cognitive ability

2017

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“…Non-food-stimulated resting-state studies have also implicated a deregulation of energy intake and impairment in inhibitory control in PWS [Zhang et al, 2013[Zhang et al, , 2015. Only a few studies have focused on anatomical alterations in PWS [Lukoshe et al, 2014;Ogura et al, 2011;Yamada et al, 2006]. Compared with controls, PWS subjects exhibited smaller gray matter volumes [Ogura et al, 2011], reduced cortical complexity [Lukoshe et al, 2014], and decreased white matter integrity [Yamada et al, 2006].…”

Section: Introductionmentioning

confidence: 99%

Brain structural alterations in obese children with and withoutPrader‐Willi Syndrome

Zhang

Deneen

et al. 2017

Human Brain Mapping

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Prader-Willi syndrome (PWS) is a genetic imprinting disorder that is mainly characterized by hyperphagia and childhood obesity. Previous neuroimaging studies revealed that there is a significant difference in brain activation patterns between obese children with and without PWS. However, whether there are differences in the brain structure of obese children with and without PWS remains elusive. In the current study, we used T1-weighted and diffusion tensor magnetic resonance imaging to investigate alterations in the brain structure, such as the cortical volume and white matter integrity, in relation to this eating disorder in 12 children with PWS, 18 obese children without PWS (OB) and 18 healthy controls. Compared with the controls, both the PWS and OB groups exhibited alterations in cortical volume, with similar deficit patterns in 10 co-varying brain regions in the bilateral dorsolateral and medial prefrontal cortices, right anterior cingulate cortex, and bilateral temporal lobe. The white matter integrities of the above regions were then examined with an analysis method based on probabilistic tractography. The PWS group exhibited distinct changes in the reduced fractional anisotropy of white matter fibers connected to the co-varying regions, whereas the OB group did not. Our findings indicated that PWS and OB share similar gray matter alterations that are responsible for the development of eating disorders. Additionally, the distinct white matter alterations might explain the symptoms associated with food intake in PWS, including excessive hyperphagia and constant hunger. Hum Brain Mapp 38:4228-4238, 2017. © 2017 Wiley Periodicals, Inc.

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“…MRI studies in PWS suggested that lower cortical complexity partially underlies cognitive impairment and developmental delay [4], with structural brain abnormalities and different neurodevelopmental patterns between children with a deletion and an mUPD [5]. Certain cognitive skills improved significantly during GH treatment, while GH-untreated children with PWS showed a deterioration of cognitive functioning [6, 7].…”

Section: Introductionmentioning

confidence: 99%

Effect of cessation of GH treatment on cognition during transition phase in Prader-Willi syndrome: results of a 2-year crossover GH trial

Kuppens

Mahabier

Bakker

et al. 2016

Orphanet J Rare Dis

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BackgroundPatients with Prader-Willi syndrome (PWS) have a cognitive impairment. Growth hormone (GH) treatment during childhood improves cognitive functioning, while cognition deteriorates in GH-untreated children with PWS. Cessation of GH treatment at attainment of adult height (AH) might deteriorate their GH-induced improved cognition, while continuation might benefit them. We, therefore, investigated the effects of placebo versus GH administration on cognition in young adults with PWS who were GH-treated for many years during childhood and had attained AH.MethodTwo-year, randomized, double-blind, placebo-controlled cross-over study in 25 young adults with PWS. Cross-over intervention with placebo and GH (0.67 mg/m2/day), both during 1 year.ResultsTotal (TIQ), verbal (VIQ) and performance IQ (PIQ) did not deteriorate during 1 year of placebo, compared to GH treatment (p > 0.322). Young adults with a lower TIQ had significantly more loss of TIQ points during placebo versus GH, in particular VIQ decreased more in those with a lower VIQ. The effect of placebo versus GH on TIQ, VIQ and PIQ was not different for gender or genotype.ConclusionsCompared to GH treatment, 1 year of placebo did not deteriorate cognitive functioning of GH-treated young adults with PWS who have attained AH. However, patients with a lower cognitive functioning had more loss in IQ points during placebo versus GH treatment. The reassuring finding that 1 year of placebo does not deteriorate cognitive functioning does, however, not exclude a gradual deterioration of cognitive functioning on the long term.Trial registration ISRCTN24648386, NTR1038, Dutch Trial Register, www.trialregister.nl. Registered 16 August 2007.

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Reduced Cortical Complexity in Children with Prader-Willi Syndrome and Its Association with Cognitive Impairment and Developmental Delay

Cited by 36 publications

References 58 publications

Adolescent maturation of the relationship between cortical gyrification and cognitive ability

Adolescent maturation of the relationship between cortical gyrification and cognitive ability

Brain structural alterations in obese children with and withoutPrader‐Willi Syndrome

Effect of cessation of GH treatment on cognition during transition phase in Prader-Willi syndrome: results of a 2-year crossover GH trial

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