Recurrent copy number variations as risk factors for neurodevelopmental disorders: critical overview and analysis of clinical implications

dc.contributor.authorTorres, Fátimapor
dc.contributor.authorBarbosa, Mafalda Fernanda Cabral Santospor
dc.contributor.authorMaciel, P.por
dc.date.accessioned2017-03-14T17:20:46Z
dc.date.available2017-03-14T17:20:46Z
dc.date.issued2016
dc.date.submitted2015-08
dc.date.updated2017-02-14T11:34:50Z
dc.description.abstractNeurodevelopmental disorders (NDs) encompass a spectrum of neuropsychiatric manifestations. Chromosomal regions 1q21.1, 3q29, 15q11.2, 15q13.3, 16p11.2, 16p13.1 and 22q11 harbour rare but recurrent CNVs that have been uncovered as being important risk factors for several of these disorders. These rearrangements may underlie a broad phenotypical spectrum, ranging from normal development, to learning problems, intellectual disability (ID), epilepsy and psychiatric diseases, such as autism spectrum disorders (ASDs) and schizophrenia (SZ). The highly increased risk of developing neurodevelopmental phenotypes associated with some of these CNVs makes them an unavoidable element in the clinical context in paediatrics, neurology and psychiatry. However, and although finding these risk loci has been the goal of neuropsychiatric genetics for many years, the translation of this recent knowledge into clinical practice has not been trivial. In this article, we will: (1) review the state of the art on recurrent CNVs associated with NDs, namely ASD, ID, epilepsy and SZ; (2) discuss the models used to dissect the underlying neurobiology of disease, (3) discuss how this knowledge can be used in clinical practice.por
dc.description.sponsorshipFEDER through the Programa Operacional Factores de Competitividade — COMPETE and by Portuguese national funds through Fundação para a Ciência e Tecnologiapor
dc.distributioninternationalpor
dc.identifier.citationTorres, F., Barbosa, M., & Maciel, P. (2016). Recurrent copy number variations as risk factors for neurodevelopmental disorders: critical overview and analysis of clinical implications. Journal of Medical Genetics, 53(2), 73-90. doi: 10.1136/jmedgenet-2015-103366por
dc.identifier.doi10.1136/jmedgenet-2015-103366por
dc.identifier.eissn1468-6244por
dc.identifier.issn0022-2593por
dc.identifier.pmid26502893por
dc.identifier.urihttps://hdl.handle.net/1822/45011
dc.language.isoengpor
dc.peerreviewedyespor
dc.publisherBMJ Publishing Grouppor
dc.relationinfo:eu-repo/grantAgreement/FCT/5876-PPCDTI/83026/PTpor
dc.relationinfo:eu-repo/grantAgreement/FCT/5876-PPCDTI/83013/PTpor
dc.relationinfo:eu-repo/grantAgreement/FCT/SFRH/SFRH%2FBDINT%2F51549%2F2011/PTpor
dc.relation.publisherversionhttp://jmg.bmj.com/content/early/2015/10/26/jmedgenet-2015-103366.shortpor
dc.rightsopenAccesspor
dc.subject.fosCiências Médicas::Medicina Básicapor
dc.subject.wosScience & Technologypor
dc.titleRecurrent copy number variations as risk factors for neurodevelopmental disorders: critical overview and analysis of clinical implicationspor
dc.typearticle
dspace.entity.typePublicationen
oaire.citationEndPage90por
oaire.citationIssue2por
oaire.citationStartPage73por
oaire.citationTitleJournal of Medical Geneticspor
oaire.citationVolume53por
sdum.journalJournal of Medical Geneticspor
sdum.publicationstatusinfo:eu-repo/semantics/publishedVersionpor

Ficheiros

Pacote original

A mostrar 1 - 1 de 1
A carregar...
Nome:
j med genet-2015-torres-jmedgenet-2015-103366.pdf
Tamanho:
898.91 KB
Formato:
Adobe Portable Document Format