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Assessment of emotions and behaviour by the Developmental Behaviour Checklist in young people with neurodevelopmental CNVs

BACKGROUND: A number of genomic conditions caused by copy number variants (CNVs) are associated with a high risk of neurodevelopmental and psychiatric disorders (ND-CNVs). Although these patients also tend to have cognitive impairments, few studies have investigated the range of emotion and behaviou...

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Autores principales: Cunningham, Adam C., Hall, Jeremy, Einfeld, Stewart, Owen, Michael J., van den Bree, Marianne B. M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Cambridge University Press 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7794095/
https://www.ncbi.nlm.nih.gov/pubmed/32643597
http://dx.doi.org/10.1017/S0033291720002330
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author Cunningham, Adam C.
Hall, Jeremy
Einfeld, Stewart
Owen, Michael J.
van den Bree, Marianne B. M.
author_facet Cunningham, Adam C.
Hall, Jeremy
Einfeld, Stewart
Owen, Michael J.
van den Bree, Marianne B. M.
author_sort Cunningham, Adam C.
collection PubMed
description BACKGROUND: A number of genomic conditions caused by copy number variants (CNVs) are associated with a high risk of neurodevelopmental and psychiatric disorders (ND-CNVs). Although these patients also tend to have cognitive impairments, few studies have investigated the range of emotion and behaviour problems in young people with ND-CNVs using measures that are suitable for those with learning difficulties. METHODS: A total of 322 young people with 13 ND-CNVs across eight loci (mean age: 9.79 years, range: 6.02–17.91, 66.5% male) took part in the study. Primary carers completed the Developmental Behaviour Checklist (DBC). RESULTS: Of the total, 69% of individuals with an ND-CNV screened positive for clinically significant difficulties. Young people from families with higher incomes (OR = 0.71, CI = 0.55–0.91, p = .008) were less likely to screen positive. The rate of difficulties differed depending on ND-CNV genotype (χ(2) = 39.99, p < 0.001), with the lowest rate in young people with 22q11.2 deletion (45.7%) and the highest in those with 1q21.1 deletion (93.8%). Specific patterns of strengths and weaknesses were found for different ND-CNV genotypes. However, ND-CNV genotype explained no more than 9–16% of the variance, depending on DBC subdomain. CONCLUSIONS: Emotion and behaviour problems are common in young people with ND-CNVs. The ND-CNV specific patterns we find can provide a basis for more tailored support. More research is needed to better understand the variation in emotion and behaviour problems not accounted for by genotype.
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spelling pubmed-77940952022-03-11 Assessment of emotions and behaviour by the Developmental Behaviour Checklist in young people with neurodevelopmental CNVs Cunningham, Adam C. Hall, Jeremy Einfeld, Stewart Owen, Michael J. van den Bree, Marianne B. M. Psychol Med Original Article BACKGROUND: A number of genomic conditions caused by copy number variants (CNVs) are associated with a high risk of neurodevelopmental and psychiatric disorders (ND-CNVs). Although these patients also tend to have cognitive impairments, few studies have investigated the range of emotion and behaviour problems in young people with ND-CNVs using measures that are suitable for those with learning difficulties. METHODS: A total of 322 young people with 13 ND-CNVs across eight loci (mean age: 9.79 years, range: 6.02–17.91, 66.5% male) took part in the study. Primary carers completed the Developmental Behaviour Checklist (DBC). RESULTS: Of the total, 69% of individuals with an ND-CNV screened positive for clinically significant difficulties. Young people from families with higher incomes (OR = 0.71, CI = 0.55–0.91, p = .008) were less likely to screen positive. The rate of difficulties differed depending on ND-CNV genotype (χ(2) = 39.99, p < 0.001), with the lowest rate in young people with 22q11.2 deletion (45.7%) and the highest in those with 1q21.1 deletion (93.8%). Specific patterns of strengths and weaknesses were found for different ND-CNV genotypes. However, ND-CNV genotype explained no more than 9–16% of the variance, depending on DBC subdomain. CONCLUSIONS: Emotion and behaviour problems are common in young people with ND-CNVs. The ND-CNV specific patterns we find can provide a basis for more tailored support. More research is needed to better understand the variation in emotion and behaviour problems not accounted for by genotype. Cambridge University Press 2022-02 2020-07-09 /pmc/articles/PMC7794095/ /pubmed/32643597 http://dx.doi.org/10.1017/S0033291720002330 Text en © The Author(s) 2020 https://creativecommons.org/licenses/by/4.0/This is an Open Access article, distributed under the terms of the Creative Commons Attribution licence (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted re-use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Original Article
Cunningham, Adam C.
Hall, Jeremy
Einfeld, Stewart
Owen, Michael J.
van den Bree, Marianne B. M.
Assessment of emotions and behaviour by the Developmental Behaviour Checklist in young people with neurodevelopmental CNVs
title Assessment of emotions and behaviour by the Developmental Behaviour Checklist in young people with neurodevelopmental CNVs
title_full Assessment of emotions and behaviour by the Developmental Behaviour Checklist in young people with neurodevelopmental CNVs
title_fullStr Assessment of emotions and behaviour by the Developmental Behaviour Checklist in young people with neurodevelopmental CNVs
title_full_unstemmed Assessment of emotions and behaviour by the Developmental Behaviour Checklist in young people with neurodevelopmental CNVs
title_short Assessment of emotions and behaviour by the Developmental Behaviour Checklist in young people with neurodevelopmental CNVs
title_sort assessment of emotions and behaviour by the developmental behaviour checklist in young people with neurodevelopmental cnvs
topic Original Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7794095/
https://www.ncbi.nlm.nih.gov/pubmed/32643597
http://dx.doi.org/10.1017/S0033291720002330
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