Neurocognitive outcomes of individuals with a sex chromosome trisomy: XXX, XYY, or XXY: a systematic review
Aim: To review systematically the neurodevelopmental characteristics of individuals with sex chromosome trisomies (SCTs). Method: A bibliographic search identified English-language articles on SCTs. The focus was on studies unbiased by clinical referral, with power of at least 0.69 to detect an effe...
| Main Authors: | , , , , |
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| Format: | Journal article |
| Language: | English |
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Wiley
2010
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| _version_ | 1797088992208879616 |
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| author | Leggett, V Jacobs, P Nation, K Scerif, G Bishop, D |
| author2 | American Academy for Cerebral Palsy and Developmental Medicine (AACPDM) |
| author_facet | American Academy for Cerebral Palsy and Developmental Medicine (AACPDM) Leggett, V Jacobs, P Nation, K Scerif, G Bishop, D |
| author_sort | Leggett, V |
| collection | OXFORD |
| description | Aim: To review systematically the neurodevelopmental characteristics of individuals with sex chromosome trisomies (SCTs). Method: A bibliographic search identified English-language articles on SCTs. The focus was on studies unbiased by clinical referral, with power of at least 0.69 to detect an effect size of 1.0. Results: We identified 35 articles on five neonatally identified samples that had adequate power for our review. An additional 11 studies were included where cases had been identified for reasons other than neurodevelopmental concerns. Individuals with an additional X chromosome had mean IQs that were within broadly normal limits but lower than the respective comparison groups, with verbal IQ most affeted. Cognitive outcomes were poorest for females with XXX. Males with XYY had normal-range IQs, but all three SCT groups (XXX, XXY, and XYY) had marked difficulties in speech and language, motor skills, and educational achievement. Nevertheless, most adults with SCTs lived independently. Less evidence was available for the brain structure and for attention, social, and psychiatric outcomes. Within each group there was much variation. Interpretation: Individuals with SCTs are at risk of cognitive and behavioural difficulties. However, the evidence base is slender, and further research is needed to ascertain the nature, severity, and causes of these difficulties in unselected samples. |
| first_indexed | 2024-03-07T02:58:04Z |
| format | Journal article |
| id | oxford-uuid:affeaf7e-0fdc-46e2-b3da-61465735f4b3 |
| institution | University of Oxford |
| language | English |
| last_indexed | 2024-03-07T02:58:04Z |
| publishDate | 2010 |
| publisher | Wiley |
| record_format | dspace |
| spelling | oxford-uuid:affeaf7e-0fdc-46e2-b3da-61465735f4b32022-03-27T03:53:21ZNeurocognitive outcomes of individuals with a sex chromosome trisomy: XXX, XYY, or XXY: a systematic reviewJournal articlehttp://purl.org/coar/resource_type/c_dcae04bcuuid:affeaf7e-0fdc-46e2-b3da-61465735f4b3Genetics (medical sciences)Experimental psychologyEnglishOxford University Research Archive - ValetWiley2010Leggett, VJacobs, PNation, KScerif, GBishop, DAmerican Academy for Cerebral Palsy and Developmental Medicine (AACPDM)British Paediatric Neurology Association (BPNA)Aim: To review systematically the neurodevelopmental characteristics of individuals with sex chromosome trisomies (SCTs). Method: A bibliographic search identified English-language articles on SCTs. The focus was on studies unbiased by clinical referral, with power of at least 0.69 to detect an effect size of 1.0. Results: We identified 35 articles on five neonatally identified samples that had adequate power for our review. An additional 11 studies were included where cases had been identified for reasons other than neurodevelopmental concerns. Individuals with an additional X chromosome had mean IQs that were within broadly normal limits but lower than the respective comparison groups, with verbal IQ most affeted. Cognitive outcomes were poorest for females with XXX. Males with XYY had normal-range IQs, but all three SCT groups (XXX, XXY, and XYY) had marked difficulties in speech and language, motor skills, and educational achievement. Nevertheless, most adults with SCTs lived independently. Less evidence was available for the brain structure and for attention, social, and psychiatric outcomes. Within each group there was much variation. Interpretation: Individuals with SCTs are at risk of cognitive and behavioural difficulties. However, the evidence base is slender, and further research is needed to ascertain the nature, severity, and causes of these difficulties in unselected samples. |
| spellingShingle | Genetics (medical sciences) Experimental psychology Leggett, V Jacobs, P Nation, K Scerif, G Bishop, D Neurocognitive outcomes of individuals with a sex chromosome trisomy: XXX, XYY, or XXY: a systematic review |
| title | Neurocognitive outcomes of individuals with a sex chromosome trisomy: XXX, XYY, or XXY: a systematic review |
| title_full | Neurocognitive outcomes of individuals with a sex chromosome trisomy: XXX, XYY, or XXY: a systematic review |
| title_fullStr | Neurocognitive outcomes of individuals with a sex chromosome trisomy: XXX, XYY, or XXY: a systematic review |
| title_full_unstemmed | Neurocognitive outcomes of individuals with a sex chromosome trisomy: XXX, XYY, or XXY: a systematic review |
| title_short | Neurocognitive outcomes of individuals with a sex chromosome trisomy: XXX, XYY, or XXY: a systematic review |
| title_sort | neurocognitive outcomes of individuals with a sex chromosome trisomy xxx xyy or xxy a systematic review |
| topic | Genetics (medical sciences) Experimental psychology |
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