Psychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.

<h4>Background and objectives</h4>Several studies have suggested a difference in clinical features of intellectual ability and psychiatric illness in the Prader-Willi syndrome (PWS) with the 15q11-q13 paternal deletion and maternal uniparental disomy (mUPD). Our objective was to appraise...

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Autores principales:	Lin Yang, Guo-dong Zhan, Jun-jie Ding, Hui-jun Wang, Duan Ma, Guo-ying Huang, Wen-hao Zhou
Formato:	article
Lenguaje:	EN
Publicado:	Public Library of Science (PLoS) 2013
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/facb60b2c2474fa389db731722d91ad0
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spelling	oai:doaj.org-article:facb60b2c2474fa389db731722d91ad02021-11-18T08:59:36ZPsychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.1932-620310.1371/journal.pone.0072640https://doaj.org/article/facb60b2c2474fa389db731722d91ad02013-01-01T00:00:00Zhttps://www.ncbi.nlm.nih.gov/pmc/articles/pmid/23967326/?tool=EBIhttps://doaj.org/toc/1932-6203<h4>Background and objectives</h4>Several studies have suggested a difference in clinical features of intellectual ability and psychiatric illness in the Prader-Willi syndrome (PWS) with the 15q11-q13 paternal deletion and maternal uniparental disomy (mUPD). Our objective was to appraise evidence on this association through a meta-analysis.<h4>Methods</h4>The electronic records PubMed and EMBASE from 1956 to 2012 were extracted for meta-analysis. Meta-analyses were performed by using fixed effect model. Mean difference, odds ratio, and 95% confidence interval were calculated.<h4>Results</h4>We retrieved a total of 744 PWS cases from 13 studies. These include 423 cases with paternal 15q11-q13 deletions and 318 cases of mUPD. Compare to the PWS cases with mUPD, PWS patients with the paternal 15q11-q13 deletion associated with significantly lower full scale IQ (FSIQ) [mean difference (MD), -2.69; 95%CI, -4.86 to -0.52; p=0.02] and verbal IQ (VIQ) (MD, -7.5; 95%CI, -9.75 to -5.26; p<0.00001) but higher performance IQ (PIQ) (MD, 4.02; 95%CI, 1.13 to 6.91; p=0.006). In contrast, PWS patients with mUPD are associated with significantly higher risk of psychiatric illness [odds rate (OR), 0.14; 95%CI, 0.08 to 0.23; p<0.00001] and higher risk of bipolar disorder (OR, 0.04; 95%CI, 0.01 to 0.23; p=0.0002).<h4>Conclusions</h4>Significant different clinical features of cognitive development and psychiatric illness are associated with PWS with different molecular defects. These findings provide support for evidence based practice to evaluate and manage the PWS syndrome with different molecular defects.Lin YangGuo-dong ZhanJun-jie DingHui-jun WangDuan MaGuo-ying HuangWen-hao ZhouPublic Library of Science (PLoS)articleMedicineRScienceQENPLoS ONE, Vol 8, Iss 8, p e72640 (2013)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Lin Yang Guo-dong Zhan Jun-jie Ding Hui-jun Wang Duan Ma Guo-ying Huang Wen-hao Zhou Psychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.
description	<h4>Background and objectives</h4>Several studies have suggested a difference in clinical features of intellectual ability and psychiatric illness in the Prader-Willi syndrome (PWS) with the 15q11-q13 paternal deletion and maternal uniparental disomy (mUPD). Our objective was to appraise evidence on this association through a meta-analysis.<h4>Methods</h4>The electronic records PubMed and EMBASE from 1956 to 2012 were extracted for meta-analysis. Meta-analyses were performed by using fixed effect model. Mean difference, odds ratio, and 95% confidence interval were calculated.<h4>Results</h4>We retrieved a total of 744 PWS cases from 13 studies. These include 423 cases with paternal 15q11-q13 deletions and 318 cases of mUPD. Compare to the PWS cases with mUPD, PWS patients with the paternal 15q11-q13 deletion associated with significantly lower full scale IQ (FSIQ) [mean difference (MD), -2.69; 95%CI, -4.86 to -0.52; p=0.02] and verbal IQ (VIQ) (MD, -7.5; 95%CI, -9.75 to -5.26; p<0.00001) but higher performance IQ (PIQ) (MD, 4.02; 95%CI, 1.13 to 6.91; p=0.006). In contrast, PWS patients with mUPD are associated with significantly higher risk of psychiatric illness [odds rate (OR), 0.14; 95%CI, 0.08 to 0.23; p<0.00001] and higher risk of bipolar disorder (OR, 0.04; 95%CI, 0.01 to 0.23; p=0.0002).<h4>Conclusions</h4>Significant different clinical features of cognitive development and psychiatric illness are associated with PWS with different molecular defects. These findings provide support for evidence based practice to evaluate and manage the PWS syndrome with different molecular defects.
format	article
author	Lin Yang Guo-dong Zhan Jun-jie Ding Hui-jun Wang Duan Ma Guo-ying Huang Wen-hao Zhou
author_facet	Lin Yang Guo-dong Zhan Jun-jie Ding Hui-jun Wang Duan Ma Guo-ying Huang Wen-hao Zhou
author_sort	Lin Yang
title	Psychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.
title_short	Psychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.
title_full	Psychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.
title_fullStr	Psychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.
title_full_unstemmed	Psychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.
title_sort	psychiatric illness and intellectual disability in the prader-willi syndrome with different molecular defects--a meta analysis.
publisher	Public Library of Science (PLoS)
publishDate	2013
url	https://doaj.org/article/facb60b2c2474fa389db731722d91ad0
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_version_	1718421120723451904

Psychiatric illness and intellectual disability in the Prader-Willi syndrome with different molecular defects--a meta analysis.

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