Sex chromosome trisomies are not associated with atypical lateralization for language

Wilson, Alexander C.; Bishop, Dorothy V. M.

doi:10.1111/dmcn.13929

Cited by 12 publications

(9 citation statements)

References 33 publications

(83 reference statements)

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“…In addition, handedness and language laterality were assessed. Results from laterality assessments were unremarkable and are not considered further here ( Wilson & Bishop, 2018a; Wilson & Bishop, 2018b). …”

Section: Methodsmentioning

confidence: 99%

Stage 2 Registered Report: Variation in neurodevelopmental outcomes in children with sex chromosome trisomies: testing the double hit hypothesis

Newbury¹,

Simpson²,

Thompson³

et al. 2018

Wellcome Open Res

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Background: The presence of an extra sex chromosome is associated with an increased rate of neurodevelopmental difficulties involving language. The 'double hit' hypothesis proposes that the adverse impact of the extra sex chromosome is amplified when genes that are expressed from the sex chromosomes interact with autosomal variants that usually have only mild effects. We predicted that the impact of an additional sex chromosome on neurodevelopment would depend on common autosomal variants involved in synaptic functions. Methods: We analysed data from 130 children with sex chromosome trisomies (SCTs: 42 girls with trisomy X, 43 boys with Klinefelter syndrome, and 45 boys with XYY). Two comparison groups were formed from 370 children from a twin study. Three indicators of phenotype were: (i) Standard score on a test of nonword repetition; (ii). A language factor score derived from a test battery; (iii) A general scale of neurodevelopmental challenges based on all available information. Preselected regions of two genes, CNTNAP2 and NRXN1, were tested for association with neurodevelopmental outcomes using Generalised Structural Component Analysis. Results: There was wide phenotypic variation in the SCT group, as well as overall impairment on all three phenotypic measures. There was no association of phenotype with CNTNAP2 or NRXN1 variants in either the SCT group or the comparison groups. Supplementary analyses found no indication of any impact of trisomy type on the results, and exploratory analyses of individual SNPs confirmed the lack of association. Conclusions: We cannot rule out that a double hit may be implicated in the phenotypic variability in children with SCTs, but our analysis does not find any support for the idea that common variants in CNTNAP2 or NRXN1 are associated with the severity of language and neurodevelopmental impairments that often accompany an extra X or Y chromosome. Stage 1 report: http://dx.doi.org/10.12688/wellcomeopenres.13828.2

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Section: Methodsmentioning

confidence: 99%

Stage 2 Registered Report: Variation in neurodevelopmental outcomes in children with sex chromosome trisomies: testing the double hit hypothesis

Newbury¹,

Simpson²,

Thompson³

et al. 2018

Wellcome Open Res

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“…Children were seen for an individual assessment at home or school. The language, reading and nonverbal ability tests ( Table 1) were given in an initial session lasting around 90 minutes, followed by an assessment of laterality, the results of which are described elsewhere ( Wilson & Bishop, 2018).…”

Section: Methodsmentioning

confidence: 99%

“…We aimed to recruit families with twin children aged between 6 years 0 months and 11 years 11 months, with over-representation of those where one or both twins had language or literacy problems. Further details are provided in Wilson and Bishop ( Wilson & Bishop, 2018)…”

Section: Participantsmentioning

confidence: 99%

Language phenotypes in children with sex chromosome trisomies

et al. 2019

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Background Sex chromosome trisomies (47,XXX, 47,XXY and 47,XYY) are known to be a risk factor for language disorder, but it is hard to predict outcomes, because many cases are identified only when problems are found. Methods We recruited children aged 5-16 years with all three types of trisomy, and divided them into a High Bias group, identified in the course of investigations for neurodevelopmental problems, and a Low Bias group, identified via prenatal screening or other medical investigations. Children from a twin sample were used to compare pattern and severity of language problems: they were subdivided according to parental concerns about language/history of speech-language therapy into a No Concerns group (N = 118) and a Language Concerns group (N = 57). Children were assessed on a psychometric battery and a standardized parent checklist. After excluding children with intellectual disability, autism or hearing problems, the sample included 28 XXX, 18 XXY and 14 XYY Low Bias cases and 7 XXX, 13 XXY and 17 XYY High Bias cases. Results Variation within each trisomy group was substantial: within the Low Bias group, overall language scores were depressed relative to normative data, but around one third had no evidence of problems. There was no effect of trisomy type, and the test profile was similar to the Language Concerns comparison group. The rate of problems was much greater in the High Bias children with trisomies. Conclusions When advising parents after discovery of a trisomy, it is important to emphasise that, though there is an increased risk of language problems, there is a very wide range of outcomes. Severe language problems are more common in those identified via genetic testing for neurodevelopmental problems but these are not characteristic of children identified on prenatal screening.

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“…At the same time, the variety of authors note reduction in the severity of the left-sided asymmetry for one reason or another during the certain speech disorders (Key et al, 2007;Kurth et al, 2018;Naoufal et al, 2016;Wilson & Bishop, 2018). One of the most common causes associated with the growth of speech problems is called left-handedness (Nikolaeva et al, 1995).…”

Section: Introductionmentioning

confidence: 99%

The Peculiarity of Lateral Preferences and the Quality of the Task Performance within the Go/Go Paradigm in Children with Complex Language Disabilities

Nikolaeva¹,

Брониславовна²,

Nikiforova³

2020

PSYCH

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The present study has tested the hypothesis of the connection of speech problems in children with left-handedness with the realisation of some high-speed reactions, in particular, a simple sensorimotor reaction in the paradigm of go/go. The handedness is determined on the basis of a set of tests; their results correlate with the results of dichotic testing. The study involves 90 children without any speech disorders and 47 children with complex speech disturbances (CSD). It is shown that the children with CSD are more likely to be not left-handed but mixed-handed in comparison with children with no speech disorders. The probability of speech diagnosis is connected with the mother's age at birth and her level of education. The older the mother at birth is and the higher the level of her education is, the less likelihood of speech diagnosis is. The children without any CSD are more steady and better at performing the task in the go/go paradigm in comparison with the children with CSD. The stability of the task performance is associated with the age of the parents. The older the father and mother are, the more effective the child without any CSD during the second part of the task works.

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Sex chromosome trisomies are not associated with atypical lateralization for language

Cited by 12 publications

References 33 publications

Stage 2 Registered Report: Variation in neurodevelopmental outcomes in children with sex chromosome trisomies: testing the double hit hypothesis

Stage 2 Registered Report: Variation in neurodevelopmental outcomes in children with sex chromosome trisomies: testing the double hit hypothesis

Language phenotypes in children with sex chromosome trisomies

The Peculiarity of Lateral Preferences and the Quality of the Task Performance within the Go/Go Paradigm in Children with Complex Language Disabilities

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