Psychiatric Illness and Intellectual Disability in the Prader–Willi Syndrome with Different Molecular Defects - A Meta AnalysisReport as inadecuate




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Background and Objectives

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.

Methods

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.

Results

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.

Conclusions

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.



Author: Lin Yang , Guo-dong Zhan , Jun-jie Ding, Hui-jun Wang, Duan Ma, Guo-ying Huang, Wen-hao Zhou

Source: http://plos.srce.hr/



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