Key result
Top schizophrenia-associated SNPs in individuals with 22q11.2 deletion syndrome were significantly enriched in genes predicted to be regulated by miRNAs disrupted by DGCR8 haploinsufficiency (17% vs 9.3% expected, p=0.03).
Absolute Event Rate: 17% vs 9.3%
p-value: p=0.03
This paper proposes a theoretical model linking miRNA processing deficits in 22q11.2 deletion syndrome to the significantly increased risk of schizophrenia.
Additional rare CNVs overlapping miRNA targets were associated with schizophrenia in 22q11.2DS; leaves open multi-hit mechanisms for prospective validation.
In humans, the most common genomic disorder is a hemizygous deletion of a 1.5-3 Mb region of chromosome 22q11.2. The resultant 22q11.2 deletion syndrome (22q11.2DS) can affect multiple organ systems, and most notably includes cardiac, craniofacial, and neurodevelopmental defects. Individuals with 22q11.2DS have a 20-25-fold risk of developing schizophrenia compared to individuals from the general population, making 22q11.2DS the strongest known molecular genetic risk factor for schizophrenia. Although the deleted region includes DGCR8, a gene coding for a miRNA processing protein, the exact mechanism by which this deletion increases risk is unknown. Importantly, several lines of evidence suggest that miRNAs may modulate risk for schizophrenia in other, non-22q11.2DS populations. Here we present a theory which mechanistically explains the link between 22q11.2DS, miRNAs, and schizophrenia risk. We outline the testable predictions generated by this theory and present preliminary data in support of our model. Further experimental validation of this model could provide important insights into the etiology of both 22q11.2DS and more common forms of schizophrenia.
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Brzustowicz et al. (2012) studied Schizophrenia in 22q11.2 deletion syndrome (n=317). miRNA target enrichment (DGCR8 haploinsufficiency) vs. Simulated random SNP distribution was evaluated on Enrichment of GWAS association signals in genes predicted to be regulated by miRNAs decreased by DGCR8 haploinsufficiency (p=0.03). Top schizophrenia-associated SNPs in individuals with 22q11.2 deletion syndrome were significantly enriched in genes predicted to be regulated by miRNAs disrupted by DGCR8 haploinsufficiency (17% vs 9.3% expected, p=0.03).
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