High-Resolution Retinal Imaging Reveals Preserved Cone Photoreceptor Density and Choroidal Thickness in Female Carriers of Choroideremia

Ophthalmic Surg Lasers Imaging Retina. 2019 Feb 1;50(2):76-85. doi: 10.3928/23258160-20190129-03.

Abstract

Background and objective: To characterize the photoreceptors and choroidal morphology of heterozygous female carriers of choroideremia who typically do not have any visual defects but can have severe funduscopic changes.

Patients and methods: This was a clinical case series study. Detailed ophthalmic examinations were performed on six female carriers from four families with choroideremia. The subfoveal choroidal thickness (SFCT) was determined by spectral-domain optical coherence tomography (SD-OCT) and the cone photoreceptor density by adaptive optics (AO) retinal imaging. SFCT and cone densities of the carriers were compared to that of normal eyes of healthy subjects.

Results: The mean age of the carriers was 42.5 years. Fundus photographs showed diffuse, patchy depigmentation; however, the SFCT was within the normal limits. AO retinal imaging revealed preserved cone densities at temporal eccentricities from 2 to 8 angular degrees.

Conclusions: The findings indicate that despite the presence of distinctive depigmentation of the retinal pigment epithelium in female carriers of choroideremia, their cone photoreceptor densities and SFCT are well-preserved. These observations may account for the good visual acuity and lack of an awareness of visual disturbances. [Ophthalmic Surg Lasers Imaging Retina. 2019;50:76-85.].

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Aged
  • Case-Control Studies
  • Child
  • Choroideremia / diagnostic imaging*
  • Choroideremia / genetics
  • Diagnostic Techniques, Ophthalmological
  • Female
  • Heterozygote
  • Humans
  • Middle Aged
  • Ophthalmoscopy / methods*
  • Pedigree
  • Retina / diagnostic imaging*
  • Retinal Cone Photoreceptor Cells*
  • Visual Acuity
  • Young Adult