Longitudinal Changes in Scotopic and Mesopic Macular Function as Assessed with Microperimetry in Patients With Stargardt Disease: SMART Study Report No. 2 - 23/03/22

Doi : 10.1016/j.ajo.2021.10.014

Xiangrong Kong ¹^2,^3,^4,^{^{1
Drs. Kong and Ibrahim-Ahmed contributed equally to this work.}} , Mohamed Ibrahim-Ahmed ^5,^{^{1
Drs. Kong and Ibrahim-Ahmed contributed equally to this work.}}, Millena G. Bittencourt ¹, Rupert W. Strauss ^6,^7,^8,^{^{4
The Kepler University clinic and Medical University of Graz are affiliated centers in the European Reference Network for Orphan diseases (ERN-EYE).}}, David G. Birch ⁹, Artur V. Cideciyan ¹⁰, Ann-Margaret Ervin ^1,³, Alexander Ho ^11,^{^{2
Dr. Ho is currently at Labcorp Drug Development, Los Angles, CA.}}, Janet S. Sunness ¹², Isabelle S. Audo ¹³, Michel Michaelides ⁶, Eberhart Zrenner ¹⁴, SriniVas Sadda ¹¹, Michael S. Ip ¹¹, Sheila West ¹, Hendrik P.N. Scholl ^1,^15,¹⁶
for the
SMART Study Group^³
The SMART Study Group members are listed in the Appendix.
¹ Wilmer Eye Institute at the Johns Hopkins University (X.K., M.G.B., A.-M.E., S.W., H.P.N.S.), Baltimore, Maryland, USA
² Department of Biostatistics (X.K.), Bloomberg School of Public Health, Johns Hopkins University, Baltimore, Maryland, USA
³ Department of Epidemiology (X.K., A.-M.E.), Bloomberg School of Public Health, Johns Hopkins University, Baltimore, Maryland, USA
⁴ Department of Health Behavior and Society (X.K.), Bloomberg School of Public Health, Johns Hopkins University, Baltimore, Maryland, USA
⁵ Ocular Imaging Research and Reading Center (M.I.-A.), Sunnyvale, California, USA
⁶ Moorfields Eye Hospital and UCL Institute of Ophthalmology (R.W.S., M.M.), London, UK
⁷ Department of Ophthalmology (R.W.S.), Kepler University Clinic, Linz, Linz, Austria
⁸ Department of Ophthalmology (R.W.S.), Medical University of Graz, Graz, Austria
⁹ Retina Foundation of the Southwest (D.G.B.), Dallas, Texas, USA
¹⁰ Scheie Eye Institute (A.V.C.), University of Pennsylvania, Philadelphia, Pennsylvania, USA
¹¹ Doheny Image Reading Center (A.H., S.V.S., M.S.I.), Los Angeles, California, USA
¹² Greater Baltimore Medical Center (J.S.S.), Baltimore, Maryland, USA
¹³ CHNO des Quinze-Vingts (I.S.A.), DHU Sight Restore, INSERM-DHOS CIC 1423, Sorbonne Universités, UPMC Univ Paris 06, INSERM, CNRS, Institut de la Vision, Paris, France
¹⁴ Universitäts-Augenklinik (E.Z.), University of Tübingen, Tübingen, Germany
¹⁵ Institute of Molecular and Clinical Ophthalmology Basel (IOB) (H.P.N.S.), Basel, Switzerland
¹⁶ Department of Ophthalmology (H.P.N.S.), University of Basel, Basel, Switzerland

Riassunto

Purpose

To estimate and compare cross-sectional scotopic versus mesopic macular sensitivity losses measured by microperimetry, and to report and compare the longitudinal rates of scotopic and mesopic macular sensitivity losses in ABCA4 gene−associated Stargardt disease (STGD1).

Design

This was a multicenter prospective cohort study.

Methods

Participants comprised 127 molecularly confirmed STGD1 patients enrolled from 6 centers in the United States and Europe and followed up every 6 months for up to 2 years.

The Nidek MP-1S device was used to measure macular sensitivities of the central 20° under mesopic and scotopic conditions. The mean deviations (MD) from normal for mesopic macular sensitivity for the fovea (within 2° eccentricity) and extrafovea (4°-10° eccentricity), and the MD for scotopic sensitivity for the extrafovea, were calculated. Linear mixed effects models were used to estimate mesopic and scotopic changes. Main outcome measures were baseline mesopic mean deviation (mMD) and scotopic MD (sMD) and rates of longitudinal changes in the mMDs and sMD.

Results

At baseline, all eyes had larger sMD, and the difference between extrafoveal sMD and mMD was 10.7 dB (P < .001). Longitudinally, all eyes showed a statistically significant worsening trend: the rates of foveal mMD and extrafoveal mMD and sMD changes were 0.72 (95% CI = 0.37-1.07), 0.86 (95% CI = 0.58-1.14), and 1.12 (95% CI = 0.66-1.57) dB per year, respectively.

Conclusions

In STGD1, in extrafovea, loss of scotopic macular function preceded and was faster than the loss of mesopic macular function. Scotopic and mesopic macular sensitivities using microperimetry provide alternative visual function outcomes for STGD1 treatment trials.

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PARTICIPANT DISPOSITION AND DEMOGRAPHICS

MEAN DEVIATIONS AT THE BASELINE VISIT

LONGITUDINAL CHANGES IN MEAN DEVIATIONS

DISCUSSION

Supplemental Material available at AJO.com. Inquiries to Xiangrong Kong Woods, 155B The Wilmer Eye Institute, 600 N. Wolfe Street, Baltimore, MD 21287, USA.

Esportazione

Vol 236

P. 32-44 - aprile 2022 Ritorno al numero

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Longitudinal Changes in Scotopic and Mesopic Macular Function as Assessed with Microperimetry in Patients With Stargardt Disease: SMART Study Report No. 2 - 23/03/22

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Results

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