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Erin P. Dooley, Craig Boote, Natalie Dora, John D. West, Christina S. Kamma-Lorger, Andrew J. Quantock, Keith M. Meek; The Structural Role Of Pax6 Over-expression In The Formation Of Microcorneas. Invest. Ophthalmol. Vis. Sci. 2011;52(14):1989.
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To better understand the structural role of Pax6 over-expression in hemizygous PAX77+/- transgenic mice (carrying 5-6 copies of the human PAX6 gene) in the formation of microcorneas.
Corneas of CBA-PAX77+/- mice (hemizygous PAX77+/- mice on a CBA/Ca genetic background) and wild-type (CBA/Ca) mice were dissected, preserved in buffered paraformaldehyde and examined by wide angle X-ray scattering at station I02 at the Diamond Light Source (Didcot, UK). The corneas were scanned using a 200 µm x 200 µm beam at a scanning interval of 0.25 mm with a 10 second exposure time. X-ray scatter patterns were recorded at each position, and subsequent data analysis quantified the preferential orientation and distribution of stromal collagen fibrils across the cornea from limbus to limbus.
PAX77+/- corneas were smaller than the wild-type strain. Differences in the preferential alignment and distribution of stromal collagen fibrils were evident between the PAX77+/- and wild-type strains. Preliminary data showed that in PAX77+/-corneas collagen lamellae were oriented in a unilateral inferior/superior direction throughout, unlike wild-type corneas which showed a more annular arrangement of lamellae.
Pax6 over-expression in PAX77+/- corneas is associated with changes in collagen lamella arrangement, which may be related to the microcornea phenotype.
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