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. |