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Publication : Inactivation of the microRNA-183/96/182 cluster results in syndromic retinal degeneration.

First Author  Lumayag S Year  2013
Journal  Proc Natl Acad Sci U S A Volume  110
Issue  6 Pages  E507-16
PubMed ID  23341629 Mgi Jnum  J:193822
Mgi Id  MGI:5469752 Doi  10.1073/pnas.1212655110
Citation  Lumayag S, et al. (2013) Inactivation of the microRNA-183/96/182 cluster results in syndromic retinal degeneration. Proc Natl Acad Sci U S A 110(6):E507-16
abstractText  The microRNA-183/96/182 cluster is highly expressed in the retina and other sensory organs. To uncover its in vivo functions in the retina, we generated a knockout mouse model, designated "miR-183C(GT/GT)," using a gene-trap embryonic stem cell clone. We provide evidence that inactivation of the cluster results in early-onset and progressive synaptic defects of the photoreceptors, leading to abnormalities of scotopic and photopic electroretinograms with decreased b-wave amplitude as the primary defect and progressive retinal degeneration. In addition, inactivation of the miR-183/96/182 cluster resulted in global changes in retinal gene expression, with enrichment of genes important for synaptogenesis, synaptic transmission, photoreceptor morphogenesis, and phototransduction, suggesting that the miR-183/96/182 cluster plays important roles in postnatal functional differentiation and synaptic connectivity of photoreceptors.
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