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Longitudinal Changes of Fixation Location and Stability Within 12 Months in Stargardt Disease: ProgStar Report No. 12

2018; Elsevier BV; Volume: 193; Linguagem: Inglês

10.1016/j.ajo.2018.06.003

1879-1891

Etienne M. Schönbach, Rupert W. Strauß, Xiangrong Kong, Beatriz Muñoz, Mohamed A. Ibrahim, Janet S. Sunness, David G. Birch, Gesa-Astrid Hahn, Fadi Nasser, Eberhart Zrenner, Srinivas R. Sadda, Sheila K. West, Hendrik P. N. Scholl, Hendrik P. N. Scholl, Rupert W. Strauß, Yulia Wolfson, Millena Bittencourt, Syed Mahmood Shah, Mohamed Ahmed, Etienne M. Schönbach, Kaoru Fujinami, Elias I. Traboulsi, Justis P. Ehlers, Meghan J. Marino, Susan Crowe, Rachael Briggs, Angela Borer, Anne Pinter, Tami Fecko, Nikki Burgnoni, Janet S. Sunness, Carol A. Applegate, Leslie Russell, Michel Michaelides, Simona Degli Esposti, Anthony T. Moore, Andrew R. Webster, Sophie Connor, Jade Barnfield, Zaid Salchi, Clara Alfageme, Victoria McCudden, Maria Pefkianaki, Jonathan Aboshiha, Gerald Liew, Graham E. Holder, Anthony G. Robson, Alexa King, Daniela Ivanova Cajas Narvaez, Katy Barnard, Catherine Grigg, Hannah Dunbar, Yetunde Obadeyi, Karine Girard-Claudon, Hilary Swann, Avani Rughani, Charles Amoah, Dominic Carrington, Kanom Bibi, Emerson Ting Co, Mohamed Nafaz Illiyas, Hamida Begum, Andrew Carter, Anne Georgiou, Selma Lewism, Saddaf Shaheen, Harpreet Shinmar, Linda M. Burton, Paul S. Bernstein, Kimberley Wegner, Briana Lauren Sawyer, Bonnie Carlstrom, Kellian Farnsworth, Cyrie Fry, Melissa Chandler, Glen Jenkins, Donnel Creel, David G. Birch, Yi‐Zhong Wang, Luis Rodriguez, Kirsten Locke, Martin Klein, Paulina Mejia, Artur V. Cideciyan, Samuel G. Jacobson, Sharon Schwartz, Rodrigo Matsui, Michaela Gruzensky, Jason Charng, Alejandro J. Román, Eberhart Zrenner, Fadi Nasser, Gesa Astrid Hahn, Barbara Wilhelm, Tobias Peters, Benjamin Beier, Tilman Koenig, Susanne Krämer, José‐Alain Sahel, Saddek Mohand‐Saïd, Isabelle Audo, Caroline Laurent‐Coriat, Ieva Sliesoraitytė, Christina Zeitz, Fiona Boyard, Minh Ha Tran, Mathias Chapon, Céline Chaumette, Juliette Amaudruz, Victoria J. Ganem, Serge Sancho, Aurore Girmens, Hendrik P. N. Scholl, Rupert W. Strauß, Yulia Wolfson, Syed Mahmood Shah, Mohamed Ahmed, Etienne M. Schönbach, Robert Wojciechowski, Shazia Khan, David Emmert, Dennis Cain, Mark Herring, Jennifer Bassinger, Lisa Liberto, Sheila K. West, Ann‐Margret Ervin, Beatriz Muñoz, Xiangrong Kong, Kurt Dreger, Jennifer M. Jones, Srinivas R. Sadda, Michael S. Ip, Anamika Jha, Alexander Ho, Brendan Kramer, Ngoc Lam, Rita Tawdros, Yong Dong Zhou, Johana Carmona, Akihito Uji, Amirhossein Hariri, Amy Lock, Anthony Elshafei, Anushika Ganegoda, Christine Petrossian, Dennis Jenkins, Edward Strnad, Elmira Baghdasaryan, Eric Ito, Feliz Samson, Gloria Blanquel, Handan Akıl, Jhanisus Melendez, Jianqin Lei, Jianyan Huang, Jonathan Chau, Khalil Ghasemi Falavarjani, Kristina Espino, Manfred Li, Maria A. Mendoza, Muneeswar Gupta Nittala, Netali Roded, Nizar Saleh, Ping Huang, Sean Pitetta, Siva Balasubramanian, Sophie Leahy, Sowmya J. Srinivas, Swetha Bindu Velaga, Teresa Margaryan, Tudor Tepelus, Tyler Brown, Wenying Fan, Yamileth Murillo, Yue Shi, Katherine Elizabeth Gonzaga Aguilar, Cynthia Chan, Lisa Santos, Brian Seo, Christopher Sison, Silvia Pérez, Stephanie Chao, Kelly Miyasato, Julia Higgins, Zoila Luna, Anita Menchaca, Norma Gonzalez, Vicky Robledo, Karen Carig, Kirstie Baker, David Ellenbogen, Daniel Bluemel, Theo Sanford, Daisy Linares, Mei Tran, Lorane Nava, Michelle K. Oberoi, Mark Romero, Vivian Chiguil, Grantley Bynum-Bain, Monica Kim, Carolina Mendiguren, Xiwen Huang, Monika Smith, Teresa Margaryan, Natalie Sarreal,

Retinal Imaging and Analysis

Purpose To investigate the natural history of Stargardt disease (STGD1) using fixation location and fixation stability. Design Multicenter, international, prospective cohort study. Methods Fixation testing was performed using the Nidek MP-1 microperimeter as part of the prospective, multicenter, natural history study on the Progression of Stargardt disease (ProgStar). A total of 238 patients with ABCA4 -related STGD1 were enrolled at baseline (bilateral enrollment in 86.6%) and underwent repeat testing at months 6 and 12. Results Outcome measures included the distance of the preferred retinal locus from the fovea (PRL) and the bivariate contour ellipse area (BCEA). After 12 months of follow-up, the change in the eccentricity of the PRL from the anatomic fovea was −0.0014 degrees (95% confidence interval [CI], −0.27 degrees, 0.27 degrees; P = .99). The deterioration in the stability of fixation as expressed by a larger BCEA encompassing 1 standard deviation of all fixation points was 1.21 degrees squared (deg 2 ) (95% CI, −1.23 deg 2 , 3.65 deg 2 ; P = .33). Eyes with increases and decreases in PRL eccentricity and/or BCEA values were observed. Conclusions Our observations point to the complexity of fixation parameters. The association of increasingly eccentric and unstable fixation with longer disease duration that is typically found in cross-sectional studies may be countered within individual patients by poorly understood processes like neuronal adaptation. Nevertheless, fixation parameters may serve as useful secondary outcome parameters in selected cases and for counseling patients to explain changes to their visual functionality.