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Clin Exp Ophthalmol. 2018 Apr;46(3):247-259. doi: 10.1111/ceo.13022. Epub 2017 Aug 31.

Novel mobility test to assess functional vision in patients with inherited retinal dystrophies.

Author information

1
Spark Therapeutics, Inc., Philadelphia, Pennsylvania, USA.
2
Center for Cellular and Molecular Therapeutics, Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.
3
Statistics Collaborative, Inc., Washington, District of Columbia, USA.
4
Westat, Inc., Philadelphia, Pennsylvania, USA.
5
Perelman School of Medicine, University of Pennsylvania, Philadelphia, Pennsylvania, USA.

Abstract

IMPORTANCE:

This novel endpoint tracks functional vision changes in patients with inherited retinal dystrophies (IRDs) over time.

BACKGROUND:

The aims of the study were to determine whether a multi-luminance mobility test (MLMT) can detect functional vision changes over time in subjects with IRDs and to assess natural history and potential effects of investigational agents.

DESIGN:

This is a prospective, observational study.

PARTICIPANTS:

Sixty-two subjects were enrolled. Sixty (29 normal sighted and 31 visually impaired) were eligible; 54 (28 visually impaired and 26 normal-sighted) completed all testing visits.

METHODS:

Subjects navigated MLMT courses three times over 1 year. At each visit, subjects completed testing using individual eyes, and both eyes, at up to nine standardized, increasing luminance levels (range 1 to 400 lux). Accuracy and speed were evaluated and compared with visual acuity (VA), visual field (VF) and a visual function questionnaire.

MAIN OUTCOME MEASURES:

Accuracy and speed of normal and visually impaired subjects on MLMT, and reliability and content validity of MLMT were the main outcome measures.

RESULTS:

MLMT distinguished normal-sighted from visually impaired subjects. All control subjects passed all MLMT attempts at all tested light levels. Visually impaired subjects' performance varied widely; some declined over 1 year. Performance declined markedly below certain VA and VF thresholds. Concordance on performance on two baseline visits was high: correlations for accuracy were 94% and 98% for lowest common and highest common lux levels.

CONCLUSIONS AND RELEVANCE:

MLMT differentiated visually impaired from control populations and, in visually impaired subjects, identified a range of performances; and tracked performance declines over time, consistent with these progressive conditions.

KEYWORDS:

functional vision; inherited retinal dystrophy; luminance; mobility test

PMID:
28697537
PMCID:
PMC5764825
DOI:
10.1111/ceo.13022
[Indexed for MEDLINE]
Free PMC Article

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