September 2016
Volume 57, Issue 12
Open Access
ARVO Annual Meeting Abstract  |   September 2016
Decreased corneal biomechanical properties in children with osteogenesis imperfecta
Author Affiliations & Notes
  • Brenda L Bohnsack
    University of Michigan, Ann Arbor, Michigan, United States
  • Lisa Lagrou
    University of Michigan, Ann Arbor, Michigan, United States
  • Sayoko Eileen Moroi
    University of Michigan, Ann Arbor, Michigan, United States
  • Footnotes
    Commercial Relationships   Brenda Bohnsack, None; Lisa Lagrou, None; Sayoko Moroi, None
  • Footnotes
    Support  NIH Grant K08EY022912-01; Research to Prevent Blindness Career Development Award; Edward Mallinckrodt Jr Foundation Grant; Paul Lichter, M.D. Research Discovery Fund; Alcon Research Institute Young Investigators Grant
Investigative Ophthalmology & Visual Science September 2016, Vol.57, 2375. doi:
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    • Get Citation

      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)

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Abstract

Purpose : 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.

Methods : 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

Results : 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.

Conclusions : 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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