Long-term Natural History of Atrophy in Eyes with Choroideremia-A Systematic Review and Meta-analysis of Individual-Level Data.

ABSTRACT:

Purpose

To conduct a systematic review and meta-analysis of the natural history of atrophy secondary to choroideremia (CHM).

Clinical relevance

A sensitive and reliable anatomic measure to monitor disease progression is needed in treatment trials for CHM. However, the long-term natural history of the residual retinal pigment epithelium (RPE) is unclear, with reported RPE area decline rates varying widely among patients.

Methods

We searched in 7 literature databases up through July 17, 2019, to identify studies that assessed the residual RPE area in untreated eyes with CHM using fundus autofluorescence (FAF). We sought individual-eye data and investigated the RPE decline pattern using 3 models: the area linear model (ALM), radius linear model (RLM), and area exponentia

SUBMITTER: Shen LL 

PROVIDER: S-EPMC7415675 | biostudies-literature | 2020 Aug

REPOSITORIES: biostudies-literature