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ASSESSMENT OF THE RETINAL STRUCTURE IN CHILDREN WITH INCONTINENTIA PIGMENTI

Mangalesh, Shwetha MBBS*; Chen, Xi MD, PhD*; Tran-Viet, Du BS*; Viehland, Christian BS; Freedman, Sharon F. MD*; Toth, Cynthia A. MD*,†

doi: 10.1097/IAE.0000000000001395
Original Study
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Purpose: This report aims at expanding the current knowledge of retinal microanatomy in children with incontinentia pigmenti using hand-held spectral domain optical coherence tomography (SDOCT).

Methods: We reviewed OCT scans from 7 children (4 weeks–13 years) obtained either in the clinic or during an examination under anesthesia. The scans were analyzed for anatomical changes in the outer and inner retina, by certified graders. Medical records were assessed for systemic findings.

Results: We observed abnormal retinal findings unilaterally in three children. We found inner and outer retinal thinning temporally in two participants. This thinning was present prior to and persisted after treatment. One child showed a distorted foveal contour and significant retinal thickening secondary to dense epiretinal membrane and vitreomacular traction. All other children had normal retinae.

Conclusion: Hand-held SDOCT imaging of the retina has brought to light additional retinal structural defects that were not previously reported or visualized via routine clinical ophthalmic examination including retinal photography. Despite a normal foveal structure and visual acuity, we identified inner and outer retinal thinning on SDOCT which may benefit from future functional assessment such as visual field testing.

Children with incontinentia pigmenti showed abnormal retinal thinning of both the inner and outer retinal layers on optical coherence tomography.

*Department of Ophthalmology, Duke University School of Medicine, Durham, North Carolina; and

Department of Biomedical Engineering, Pratt School of Engineering, Duke University, Durham, North Carolina.

Reprint requests: Cynthia A. Toth, MD, Department of Ophthalmology, Duke University Medical Center, 2351 Erwin Road-Box 3802, Durham, NC 27710; e-mail: cynthia.toth@dm.duke.edu

The Hartwell Foundation; The Andrew Family Charitable Foundation; Grant number 1RO1 EY025009-0141 (NIH); Pilot grant from Duke Translational Research Institute (NIH) and NIH Roadmap for Medical Research. Its contents are solely the resonsibility of the authors and do not necessarily represent the official view of NCRR or NIH. The sponsors or funding organizations had no role in the design or conduct of this research.

C. A. Toth recieves royalties through her university from Alcon and research support from Bioptigen and Genentech. She also has unlicensed patents pending in OCT imaging and analysis. S. F. Freedman is a scientific consultant to Pfizer, Inc and Inotek, Inc. The remaining authors has no financial disclosures. The remaining authors has no proprietary interest in the current study.

© 2017 by Ophthalmic Communications Society, Inc.