Copy Number Variation Screen Identifies a Rare De Novo Deletion at Chromosome 15q13.1-13.3 in a Child with Language Impairment.

A significant proportion of children (up to 7% in the UK) present with pronounced language difficulties that cannot be explained by obvious causes like other neurological and medical conditions. A substantial genetic component is predicted to underlie such language problems. Copy number variants (CN...

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Main Authors: Kerry A Pettigrew, Emily Reeves, Ruth Leavett, Marianna E Hayiou-Thomas, Anahita Sharma, Nuala H Simpson, Angela Martinelli, Paul Thompson, Charles Hulme, Margaret J Snowling, Dianne F Newbury, Silvia Paracchini
Format: Article
Language:English
Published: Public Library of Science (PLoS) 2015-01-01
Series:PLoS ONE
Online Access:https://doi.org/10.1371/journal.pone.0134997
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author Kerry A Pettigrew
Emily Reeves
Ruth Leavett
Marianna E Hayiou-Thomas
Anahita Sharma
Nuala H Simpson
Angela Martinelli
Paul Thompson
Charles Hulme
Margaret J Snowling
Dianne F Newbury
Silvia Paracchini
author_facet Kerry A Pettigrew
Emily Reeves
Ruth Leavett
Marianna E Hayiou-Thomas
Anahita Sharma
Nuala H Simpson
Angela Martinelli
Paul Thompson
Charles Hulme
Margaret J Snowling
Dianne F Newbury
Silvia Paracchini
author_sort Kerry A Pettigrew
collection DOAJ
description A significant proportion of children (up to 7% in the UK) present with pronounced language difficulties that cannot be explained by obvious causes like other neurological and medical conditions. A substantial genetic component is predicted to underlie such language problems. Copy number variants (CNVs) have been implicated in neurodevelopmental and psychiatric conditions, such as autism and schizophrenia, but it is not fully established to what extent they might contribute to language disorders. We conducted a CNV screen in a longitudinal cohort of young children with language-related difficulties (n = 85), focusing on single events at candidate loci. We detected a de novo deletion on chromosome 15q13.1-13.3. The adjacent 15q11-13.1 locus is disrupted in Prader-Willi and Angelman syndromes, while disruptions across the breakpoints (BP1-BP6) have previously been implicated in different neurodevelopmental phenotypes including autism, intellectual disability (ID), seizures and developmental delay (DD). This is the first report of a deletion at BP3-BP5 being linked to a deficit confined to language impairment, in the absence of ID, expanding the range of phenotypes that implicate the chromosome 15q13 locus.
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spelling doaj-art-1dcc9a14322a463ea26f3495f3ad5eb42025-08-20T02:22:46ZengPublic Library of Science (PLoS)PLoS ONE1932-62032015-01-01108e013499710.1371/journal.pone.0134997Copy Number Variation Screen Identifies a Rare De Novo Deletion at Chromosome 15q13.1-13.3 in a Child with Language Impairment.Kerry A PettigrewEmily ReevesRuth LeavettMarianna E Hayiou-ThomasAnahita SharmaNuala H SimpsonAngela MartinelliPaul ThompsonCharles HulmeMargaret J SnowlingDianne F NewburySilvia ParacchiniA significant proportion of children (up to 7% in the UK) present with pronounced language difficulties that cannot be explained by obvious causes like other neurological and medical conditions. A substantial genetic component is predicted to underlie such language problems. Copy number variants (CNVs) have been implicated in neurodevelopmental and psychiatric conditions, such as autism and schizophrenia, but it is not fully established to what extent they might contribute to language disorders. We conducted a CNV screen in a longitudinal cohort of young children with language-related difficulties (n = 85), focusing on single events at candidate loci. We detected a de novo deletion on chromosome 15q13.1-13.3. The adjacent 15q11-13.1 locus is disrupted in Prader-Willi and Angelman syndromes, while disruptions across the breakpoints (BP1-BP6) have previously been implicated in different neurodevelopmental phenotypes including autism, intellectual disability (ID), seizures and developmental delay (DD). This is the first report of a deletion at BP3-BP5 being linked to a deficit confined to language impairment, in the absence of ID, expanding the range of phenotypes that implicate the chromosome 15q13 locus.https://doi.org/10.1371/journal.pone.0134997
spellingShingle Kerry A Pettigrew
Emily Reeves
Ruth Leavett
Marianna E Hayiou-Thomas
Anahita Sharma
Nuala H Simpson
Angela Martinelli
Paul Thompson
Charles Hulme
Margaret J Snowling
Dianne F Newbury
Silvia Paracchini
Copy Number Variation Screen Identifies a Rare De Novo Deletion at Chromosome 15q13.1-13.3 in a Child with Language Impairment.
PLoS ONE
title Copy Number Variation Screen Identifies a Rare De Novo Deletion at Chromosome 15q13.1-13.3 in a Child with Language Impairment.
title_full Copy Number Variation Screen Identifies a Rare De Novo Deletion at Chromosome 15q13.1-13.3 in a Child with Language Impairment.
title_fullStr Copy Number Variation Screen Identifies a Rare De Novo Deletion at Chromosome 15q13.1-13.3 in a Child with Language Impairment.
title_full_unstemmed Copy Number Variation Screen Identifies a Rare De Novo Deletion at Chromosome 15q13.1-13.3 in a Child with Language Impairment.
title_short Copy Number Variation Screen Identifies a Rare De Novo Deletion at Chromosome 15q13.1-13.3 in a Child with Language Impairment.
title_sort copy number variation screen identifies a rare de novo deletion at chromosome 15q13 1 13 3 in a child with language impairment
url https://doi.org/10.1371/journal.pone.0134997
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