研究目的
To assess cross-sectional and longitudinal microperimetry and full-field static perimetry-derived retinal sensitivity with conventional and volumetric indices of retinal function in childhood-onset Stargardt disease (STGD1).
研究成果
The study demonstrated that children with childhood-onset STGD1 can reliably undergo functional testing using microperimetry and full-field static perimetry. Volumetric indices derived from topographic analysis provide reliable measures of retinal sensitivity, with good test-retest reliability and interocular symmetry. A greater rate of progression was observed in children compared to adults, highlighting the potential for these methods in monitoring disease progression and designing treatment trials.
研究不足
The study had a relatively small adult subgroup (n=13), which may limit generalizability. Test-retest variability could vary by factors like age and disease severity, and individual variability was not quantified. Follow-up duration varied, and increased follow-ups would provide more accurate progression rates. The influence of fixation stability and location was not explored. The custom grids may introduce biases in mean sensitivity calculations.
1：Experimental Design and Method Selection:
The study used a prospective design with molecularly confirmed childhood-onset STGD1 subjects. Methods included full-field static perimetry and microperimetry using custom grids, with computation of mean sensitivity (MS) and volumetric indices (VTOT, V30). Statistical analysis involved intraclass correlation coefficient (ICC), paired t-tests, independent t-tests, and Pearson correlation coefficients to assess reliability, symmetry, and progression rates.
2：0). Statistical analysis involved intraclass correlation coefficient (ICC), paired t-tests, independent t-tests, and Pearson correlation coefficients to assess reliability, symmetry, and progression rates.
Sample Selection and Data Sources:
2. Sample Selection and Data Sources: Subjects were recruited from an ongoing natural history study at Moorfields Eye Hospital and UCL Institute of Ophthalmology. Inclusion criteria: molecularly confirmed STGD1 with childhood onset. Subjects under 18 at baseline were children, 18 or older were adults. Data included baseline and follow-up perimetry tests with a minimum 12-month follow-up.
3：List of Experimental Equipment and Materials:
Equipment: Octopus900 (Haag-Streit AG) for full-field static perimetry, Nidek MP-1 (Nidek Technologies) for microperimetry, Spectralis OCT (Heidelberg Engineering) for OCT scans. Materials: Pupil dilation agents (
4：5% phenylephrine hydrochloride, 1% tropicamide ophthalmic solution). Software:
EyeSuite for Octopus900, manufacturer's software for Nidek MP-1, Visual Field Modelling and Analysis (VFMA) for topographic analysis.
5：Experimental Procedures and Operational Workflow:
For full-field static perimetry: Monocular testing with Octopus900, custom grid of 185 locations, background
6：4 apostilbs, Goldmann size V stimulus, 200 ms duration, GATE strategy. Fixation monitored by technician. Tests with reliability factor >30% excluded. For microperimetry:
Monocular testing with Nidek MP-1, pupils dilated, custom grid of 44 locations, background 4 apostilbs, Goldmann size III stimulus, 200 ms duration, 4-2 dB full-threshold strategy. Fixation monitored, OCT scan used for grid placement. Volumetric indices computed using VFMA software.
7：Data Analysis Methods:
Statistical analysis performed with SPSS. Normality tested with Shapiro-Wilk test. ICC for test-retest reliability. Paired t-tests for interocular symmetry. Independent t-tests for progression rate differences. Pearson correlation for correlations between metrics.
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