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Brenda L Bohnsack, Lisa Lagrou, Sayoko Eileen Moroi; Decreased corneal biomechanical properties in children with osteogenesis imperfecta. Invest. Ophthalmol. Vis. Sci. 2016;57(12):2375.
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© ARVO (1962-2015); The Authors (2016-present)
Osteogenesis imperfecta (OI) is a genetic disease due to type I collagen defects. In adults, OI is associated with degenerative ocular pathologies, however, eye findings in children are not well-described.
A prospective, comparative, cross-sectional study of pediatric patients (2-19 years) diagnosed with OI. Thirteen patients with OI and five control patients underwent complete ophthalmic examinations. Additional tests included ocular response analyzer, pachymetry, axial length measurement, automated perimetry, and, retinal nerve fiber layer (RNFL) thickness. Statistically significant differences between OI and control groups were determined using independent-samples t test.Results: We found significantly (p<0.05) decreased corneal hysteresis
We found significantly (p<0.05) decreased corneal hysteresis (8.2±1.4mmHg vs. 10.5±1.3), and central corneal thickness (CCT; 451.9 ± 29.1mm vs. 550.5±30.7 mm) in OI patients compared to controls. There was no significant difference in corneal resistance factor (9.9±1.3 mmHg vs. 10.5 ±0.8 mmHg). None of these differences correlated with age, race, or gender. IOP corrected for corneal thickness and hysteresis was significantly higher in OI patients than controls (19.1 ± 3.0 mmHg vs. 13.7 ± 0.7 mmHg). There were no differences in keratometry, axial length and RNFL thickness in OI patients compared to controls.
Strength, flexibility and organization of type-I-collagen are critical for corneal structure and function. In our study, we found that children with OI had decreased corneal hysteresis and CCT, which resulted in artificially low IOP readings. In adults, OI has been associated with primary open angle glaucoma and keratoconus. Our findings suggest that in OI, alterations in corneal biomechanical properties are present at a young age and affected individuals should be routinely screened for glaucoma and corneal pathologies.
This is an abstract that was submitted for the 2016 ARVO Annual Meeting, held in Seattle, Wash., May 1-5, 2016.
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