Late contralateral recurrence of retinal detachment in incontinentia pigmenti: A case report.

Abstract

BACKGROUND:

Incontinentia pigmenti (IP) is a rare X-linked genetic disease. It mainly manifests as skin lesions and causes problems in the eyes, teeth, bones, and central nervous system. Of the various ocular manifestations, the most severe with difficult recovery is retinal detachment (RD). Here, we report an unusual case of bilateral asymmetrical RD.

CASE SUMMARY:

We present the case of an 11-year-old Chinese girl with IP who complained of sudden blurring of vision in the left eye. At that time, she had been blind in her right eye for 4 years. RD with traction was observed in both eyes. A massive retinal proliferative membrane, exudation, and hemorrhage were seen in the left eye. We performed vitrectomy in her left eye. Her visual acuity recovered to 20/50, and her retina had flattened within 2 d after surgery. During the 3-mo follow-up, we performed retinal laser treatment of the non-perfused retinal area in her left eye. Eventually, her visual acuity returned to 20/32, and no new retinal abnormalities developed.

CONCLUSION:

In patients with IP with fundal abnormalities in one eye, it is important to focus on the rate of fundal change in the other eye. RD in its early stages can be effectively treated with timely vitrectomy and laser photocoagulation.

• Authors: You-Ran Cai, Yong Liang, Xin Zhong
• Journal: World journal of clinical cases (World J Clin Cases) Vol. 10 Issue 13 Pg. 4171-4176 (May 06 2022) ISSN: 2307-8960 [Print] United States
• PMID: 35665106 (Publication Type: Case Reports)
• Copyright: ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.