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1. Faster Sensitivity Loss around Dense Scotomas than for Overall Macular Sensitivity in Stargardt Disease: ProgStar Report No. 14

Author

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

Published
2020
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2. Longitudinal Changes of Fixation Location and Stability Within 12 Months in Stargardt Disease: ProgStar Report No. 12

Author

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

Published
2018
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3. Human Cone Visual Pigment Deletions Spare Sufficient Photoreceptors to Warrant Gene Therapy

Author

Cideciyan, Artur V, Hufnagel, Robert B, Carroll, Joseph, Sumaroka, Alexander, Luo, Xunda, Schwartz, Sharon B, Dubra, Alfredo, Land, Megan, Michaelides, Michel, Gardner, Jessica C, Hardcastle, Alison J, Moore, Anthony T, Sisk, Robert A, Ahmed, Zubair M, Kohl, Susanne, Wissinger, Bernd, and Jacobson, Samuel G

Subjects
Rare Diseases, Neurosciences, Orphan Drug, Eye Disease and Disorders of Vision, Gene Therapy, Genetics, Eye, Adolescent, Adult, Animals, Child, Child, Preschool, Color Vision Defects, Female, Gene Deletion, Genetic Therapy, Humans, Mice, Middle Aged, Mutation, Retinal Cone Photoreceptor Cells, Rod Opsins
Abstract

Human X-linked blue-cone monochromacy (BCM), a disabling congenital visual disorder of cone photoreceptors, is a candidate disease for gene augmentation therapy. BCM is caused by either mutations in the red (OPN1LW) and green (OPN1MW) cone photoreceptor opsin gene array or large deletions encompassing portions of the gene array and upstream regulatory sequences that would predict a lack of red or green opsin expression. The fate of opsin-deficient cone cells is unknown. We know that rod opsin null mutant mice show rapid postnatal death of rod photoreceptors. Using in vivo histology with high-resolution retinal imaging, we studied a cohort of 20 BCM patients (age range 5-58) with large deletions in the red/green opsin gene array. Already in the first years of life, retinal structure was not normal: there was partial loss of photoreceptors across the central retina. Remaining cone cells had detectable outer segments that were abnormally shortened. Adaptive optics imaging confirmed the existence of inner segments at a spatial density greater than that expected for the residual blue cones. The evidence indicates that human cones in patients with deletions in the red/green opsin gene array can survive in reduced numbers with limited outer segment material, suggesting potential value of gene therapy for BCM.

Published
2013

4. Mutations in RPGR and RP2 Account for 15% of Males with Simplex Retinal Degenerative DiseaseX-Linked Mutations in Simplex Males

Author

Branham, Kari, Othman, Mohammad, Brumm, Matthew, Karoukis, Athanasios J, Atmaca-Sonmez, Pelin, Yashar, Beverly M, Schwartz, Sharon B, Stover, Niamh B, Trzupek, Karmen, Wheaton, Dianna, Jennings, Barbara, Ciccarelli, Maria Laura, Jayasundera, K Thiran, Lewis, Richard A, Birch, David, Bennett, Jean, Sieving, Paul A, Andreasson, Sten, Duncan, Jacque L, Fishman, Gerald A, Iannaccone, Alessandro, Weleber, Richard G, Jacobson, Samuel G, Heckenlively, John R, and Swaroop, Anand

Subjects
Neurosciences, Rare Diseases, Clinical Research, Genetics, Neurodegenerative, Eye Disease and Disorders of Vision, Aetiology, 2.1 Biological and endogenous factors, Eye, DNA Mutational Analysis, Eye Proteins, Female, GTP-Binding Proteins, Genetic Diseases, X-Linked, Genetic Testing, Humans, Intracellular Signaling Peptides and Proteins, Male, Membrane Proteins, Mutation, Pedigree, Random Amplified Polymorphic DNA Technique, Registries, Retinitis Pigmentosa, Sex Distribution, Biological Sciences, Medical and Health Sciences, Ophthalmology & Optometry
Abstract

PurposeTo determine the proportion of male patients presenting simplex retinal degenerative disease (RD: retinitis pigmentosa [RP] or cone/cone-rod dystrophy [COD/CORD]) with mutations in the X-linked retinal degeneration genes RPGR and RP2.MethodsSimplex males were defined as patients with no known affected family members. Patients were excluded if they had a family history of parental consanguinity. Blood samples from a total of 214 simplex males with a diagnosis of retinal degeneration were collected for genetic analysis. The patients were screened for mutations in RPGR and RP2 by direct sequencing of PCR-amplified genomic DNA.ResultsWe identified pathogenic mutations in 32 of the 214 patients screened (15%). Of the 29 patients with a diagnosis of COD/CORD, four mutations were identified in the ORF15 mutational hotspot of the RPGR gene. Of the 185 RP patients, three patients had mutations in RP2 and 25 had RPGR mutations (including 12 in the ORF15 region).ConclusionsThis study represents mutation screening of RPGR and RP2 in the largest cohort, to date, of simplex males affected with RP or COD/CORD. Our results demonstrate a substantial contribution of RPGR mutations to retinal degenerations, and in particular, to simplex RP. Based on our findings, we suggest that RPGR should be considered as a first tier gene for screening isolated males with retinal degeneration.

Published
2012

8. Visual Acuity Loss and Associated Risk Factors in the Retrospective Progression of Stargardt Disease Study (ProgStar Report No. 2)

Author

Scholl, Hendrik P.N., Strauss, Rupert W., Wolfson, Yulia, Bittencourt, Millena, Shah, Syed Mahmood, Ahmed, Mohammed, Schonbach, Etienne, Fujinami, Kaoru, Traboulsi, Elias, Ehlers, Justis, Marino, Meghan, Crowe, Susan, Briggs, Rachael, Borer, Angela, Pinter, Anne, Fecko, Tami, Brugnoni, Nikki, Sunness, Janet S., Applegate, Carol, Russell, Leslie, Michaelides, Michel, Moore, Anthony, Webster, Andrew, Connor, Sophie, McCudden, Victoria, Pefkianaki, Maria, Aboshiha, Jonathan, Liew, Gerald, Holder, Graham, Robson, Anthony, King, Alexa, Cajas Narvaez, Daniela Ivanova, Barnard, Katy, Grigg, Catherine, Dunbar, Hannah, Obadeyi, Yetunde, Girard-Claudon, Karine, Swann, Hilary, Rughani, Avani, Amoah, Charles, Carrington, Dominic, Bibi, Kanom, Co, Emerson Ting, Carter, Andrew, Georgiou, Anne, Lewis, Selma, Shaheen, Saddaf, Shinmar, Harpreet, Burton, Linda, Bernstein, Paul, Wegner, Kimberley, Sawyer, Briana Lauren, Carlstrom, Bonnie, Farnsworth, Kellian, Fry, Cyrie, Chandler, Melissa, Jenkins, Glen, Creel, Donnel, Birch, David, Wang, Yi-Zhong, Rodriguez, Luis, Locke, Kirsten, Klein, Martin, Mejia, Paulina, Cideciyan, Artur V., Jacobson, Samuel G., Schwartz, Sharon B., Matsui, Rodrigo, Gruzensky, Michaela, Roman, Alejandro J., Zrenner, Eberhart, Nasser, Fadi, Hahn, Gesa Astrid, Wilhelm, Barbara, Peters, Tobias, Beier, Benjamin, Koenig, Tilman, Kramer, Susanne, Sahel, José-Alain, Mohand-Said, Saddek, Audo, Isabelle, Laurent-Coriat, Caroline, Sliesoraityte, Ieva, Zeitz, Christina, Boyard, Fiona, Tran, Minh Ha, Chapon, Mathias, Chaumette, Céline, Amaudruz, Juliette, Ganem, Victoria, Sancho, Serge, Girmens, Aurore, Wojciechowski, Robert, Khan, Shazia, Emmert, David G., Cain, Dennis, Herring, Mark, Bassinger, Jennifer, Liberto, Lisa, West, Sheila, Ervin, Ann-Margret, Munoz, Beatriz, Kong, Xiangrong, Dreger, Kurt, Jones, Jennifer, Sadda, Srinivas, Jha, Anamika, Ho, Alex, Kramer, Brendan, Hariri, Amirhossein, Blanquel, Gloria Rebecca, Lam, Ngoc, Pitetta, Sean, Shi, Yue, Tawdros, Rita, Petrossian, Christine, Jenkins, Dennis, Gupta, Muneeswar, Zhou, Yong Dong, Aguilar, Katherine, Chan, Cynthia, Santos, Lisa, Seo, Brian, Sison, Christopher, Perez, Silvia, Chao, Stephanie, Miyasato, Kelly, Higgins, Julia, Luna, Zoila, Menchaca, Anita, Gonzalez, Norma, Robledo, Vicky, Carig, Karen, Baker, Kirstie, Ellenbogen, David, Russell, James, Bluemel, Daniel, Moreno, Alex, Pham, Royal, Sanford, Theo, Linares, Daisy, Tran, Mei, and Muñoz, Beatriz

Published
2016
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11. Human Gene Therapy for RPE65 Isomerase Deficiency Activates the Retinoid Cycle of Vision but with Slow Rod Kinetics

Author

Cideciyan, Artur V., Aleman, Tomas S., Boye, Sanford L., Schwartz, Sharon B., Kaushal, Shalesh, Roman, Alejandro J., Pang, Ji-jing, Sumaroka, Alexander, Windsor, Elizabeth A. M., Wilson, James M., Flotte, Terence R., Fishman, Gerald A., Heon, Elise, Stone, Edwin M., Byrne, Barry J., Jacobson, Samuel G., and Hauswirth, William W.

Published
2008
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12. Human Cone Photoreceptor Dependence on RPE65 Isomerase

Author

Jacobson, Samuel G., Aleman, Tomas S., Cideciyan, Artur V., Heon, Elise, Golczak, Marcin, Beltran, William A., Sumaroka, Alexander, Schwartz, Sharon B., Roman, Alejandro J., Windsor, Elizabeth A. M., Wilson, James M., Aguirre, Gustavo D., Stone, Edwin M., and Palczewski, Krzysztof

Published
2007
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16. Safety of Recombinant Adeno-Associated Virus Type 2–RPE65 Vector Delivered by Ocular Subretinal Injection

Author

Jacobson, Samuel G., Acland, Gregory M., Aguirre, Gustavo D., Aleman, Tomas S., Schwartz, Sharon B., Cideciyan, Artur V., Zeiss, Caroline J., Komaromy, Andras M., Kaushal, Shalesh, Roman, Alejandro J., Windsor, Elizabeth A.M., Sumaroka, Alexander, Pearce-Kelling, Susan E., Conlon, Thomas J., Chiodo, Vincent A., Boye, Sanford L., Flotte, Terence R., Maguire, Albert M., Bennett, Jean, and Hauswirth, William W.

Published
2006
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17. Determining consequences of retinal membrane guanylyl cyclase (RetGC1) deficiency in human Leber congenital amaurosis en route to therapy: residual cone-photoreceptor vision correlates with biochemical properties of the mutants

Author

Jacobson, Samuel G., Cideciyan, Artur V., Peshenko, Igor V., Sumaroka, Alexander, Olshevskaya, Elena V., Cao, Lihui, Schwartz, Sharon B., Roman, Alejandro J., Olivares, Melani B., Sadigh, Sam, Yau, King-Wai, Heon, Elise, Stone, Edwin M., and Dizhoor, Alexander M.

Published
2013
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18. Gene Therapy for Leber Congenital Amaurosis Caused by RPE65 Mutations: Safety and Efficacy in 15 Children and Adults Followed Up to 3 Years

Author

Jacobson, Samuel G., Cideciyan, Artur V., Ratnakaram, Ramakrishna, Heon, Elise, Schwartz, Sharon B., Roman, Alejandro J., Peden, Marc C., Aleman, Tomas S., Boye, Sanford L., Sumaroka, Alexander, Conlon, Thomas J., Calcedo, Roberto, Pang, Ji-Jing, Erger, Kirsten E., Olivares, Melani B., Mullins, Cristina L., Swider, Malgorzata, Kaushal, Shalesh, Feuer, William J., Iannaccone, Alessandro, Fishman, Gerald A., Stone, Edwin M., Byrne, Barry J., and Hauswirth, William W.

Published
2012
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21. Faster Sensitivity Loss around Dense Scotomas than for Overall Macular Sensitivity in Stargardt Disease: ProgStar Report No. 14

Author

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

Published
2020
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23. ABCA4 disease progression and a proposed strategy for gene therapy

Author

Cideciyan, Artur V., Swider, Malgorzata, Aleman, Tomas S., Tsybovsky, Yaroslav, Schwartz, Sharon B., Windsor, Elizabeth A.M., Roman, Alejandro J., Sumaroka, Alexander, Steinberg, Janet D., Jacobson, Samuel G., Stone, Edwin M., and Palczewski, Krzysztof

Published
2009

30. Variegated yet non-random rod and cone photoreceptor disease patterns in RPGR-ORF15-associated retinal degeneration

Author

Charng, Jason, primary, Cideciyan, Artur V, additional, Jacobson, Samuel G, additional, Sumaroka, Alexander, additional, Schwartz, Sharon B, additional, Swider, Malgorzata, additional, Roman, Alejandro J, additional, Sheplock, Rebecca, additional, Anand, Manisha, additional, Peden, Marc C, additional, Khanna, Hemant, additional, Heon, Elise, additional, Wright, Alan F, additional, and Swaroop, Anand, additional

Published
2018
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31. Longitudinal Changes of Fixation Location and Stability Within 12 Months in Stargardt Disease: ProgStar Report No. 12

Author

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

Published
2018
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32. Leber congenital amaurosis caused by Lebercilin (LCA5) mutation: Retained photoreceptors adjacent to retinal disorganization

Author

Jacobson, Samuel G., Aleman, Tomas S., Cideciyan, Artur V., Sumaroka, Alexander, Schwartz, Sharon B., Windsor, Elizabeth A.M., Swider, Malgorzata, Herrera, Waldo, and Stone, Edwin M.

Subjects
Adult, Spectroscopy, Near-Infrared, genetic structures, Adolescent, Infant, Pupil, Retinal Pigment Epithelium, Middle Aged, Blindness, eye diseases, Retina, Child, Preschool, Mutation, Humans, sense organs, Child, Eye Proteins, Microtubule-Associated Proteins, Tomography, Optical Coherence, Research Article, Photoreceptor Cells, Vertebrate
Abstract

Purpose To determine the retinal disease expression in the rare form of Leber congenital amaurosis (LCA) caused by Lebercilin (LCA5) mutation. Methods Two young unrelated LCA patients, ages six years (P1) and 25 years (P2) at last visit, both with the same homozygous mutation in the LCA5 gene, were evaluated clinically and with noninvasive studies. En face imaging was performed with near-infrared (NIR) reflectance and autofluorescence (AF); cross-sectional retinal images were obtained with optical coherence tomography (OCT). Dark-adapted thresholds were measured in the older patient; and the transient pupillary light reflex was recorded and quantified in both patients. Results Both LCA5 patients had light perception vision only, hyperopia, and nystagmus. P1 showed a prominent central island of retinal pigment epithelium (RPE) surrounded by alternating elliptical-appearing areas of decreased and increased pigmentation. Retinal laminar architecture at and near the fovea was abnormal in both patients. Foveal outer nuclear layer (ONL) was present in P1 and P2 but to different degrees. With increasing eccentricity, there was retinal laminar disorganization. Regions of pericentral and midperipheral retina in P1, but not P2, could retain measurable ONL and less laminopathy. P2 had a small central island of perception with >5 log units of sensitivity loss. Pupillary responsiveness was present in both LCA5 patients; the thresholds were abnormally elevated by ≥5.5 log units. Conclusions LCA5 patients had evidence of retained photoreceptors mainly in the central retina. Retinal remodeling was present in pericentral regions in both patients. The NIR reflectance and NIR-AF imaging in the younger patient suggested preserved RPE in retinal regions with retained photoreceptors. Detailed phenotype studies in other LCA5 patients with longitudinal follow-up will help determine the feasibility of future intervention in this rare disease.

Published
2009

33. Outcome Measures for Clinical Trials of Leber Congenital Amaurosis Caused by the Intronic Mutation in the CEP290 Gene

Author

Jacobson, Samuel G., primary, Cideciyan, Artur V., additional, Sumaroka, Alexander, additional, Roman, Alejandro J., additional, Charng, Jason, additional, Lu, Monica, additional, Choi, Windy, additional, Sheplock, Rebecca, additional, Swider, Malgorzata, additional, Kosyk, Mychajlo S., additional, Schwartz, Sharon B., additional, Stone, Edwin M., additional, and Fishman, Gerald A., additional

Published
2017
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35. Variegated yet non-random rod and cone photoreceptor disease patterns inRPGR-ORF15-associated retinal degeneration

Author

Charng, Jason, primary, Cideciyan, Artur V, additional, Jacobson, Samuel G, additional, Sumaroka, Alexander, additional, Schwartz, Sharon B, additional, Swider, Malgorzata, additional, Roman, Alejandro J, additional, Sheplock, Rebecca, additional, Anand, Manisha, additional, Peden, Marc C, additional, Khanna, Hemant, additional, Heon, Elise, additional, Wright, Alan F, additional, and Swaroop, Anand, additional

Published
2016
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37. Visual Acuity Loss and Associated Risk Factors in the Retrospective Progression of Stargardt Disease Study (ProgStar Report No. 2)

Author

Kong, Xiangrong, primary, Strauss, Rupert W., additional, Michaelides, Michel, additional, Cideciyan, Artur V., additional, Sahel, José-Alain, additional, Muñoz, Beatriz, additional, West, Sheila, additional, Scholl, Hendrik P.N., additional, Wolfson, Yulia, additional, Bittencourt, Millena, additional, Shah, Syed Mahmood, additional, Ahmed, Mohammed, additional, Schonbach, Etienne, additional, Fujinami, Kaoru, additional, Traboulsi, Elias, additional, Ehlers, Justis, additional, Marino, Meghan, additional, Crowe, Susan, additional, Briggs, Rachael, additional, Borer, Angela, additional, Pinter, Anne, additional, Fecko, Tami, additional, Brugnoni, Nikki, additional, Sunness, Janet S., additional, Applegate, Carol, additional, Russell, Leslie, additional, Moore, Anthony, additional, Webster, Andrew, additional, Connor, Sophie, additional, McCudden, Victoria, additional, Pefkianaki, Maria, additional, Aboshiha, Jonathan, additional, Liew, Gerald, additional, Holder, Graham, additional, Robson, Anthony, additional, King, Alexa, additional, Cajas Narvaez, Daniela Ivanova, additional, Barnard, Katy, additional, Grigg, Catherine, additional, Dunbar, Hannah, additional, Obadeyi, Yetunde, additional, Girard-Claudon, Karine, additional, Swann, Hilary, additional, Rughani, Avani, additional, Amoah, Charles, additional, Carrington, Dominic, additional, Bibi, Kanom, additional, Co, Emerson Ting, additional, Carter, Andrew, additional, Georgiou, Anne, additional, Lewis, Selma, additional, Shaheen, Saddaf, additional, Shinmar, Harpreet, additional, Burton, Linda, additional, Bernstein, Paul, additional, Wegner, Kimberley, additional, Sawyer, Briana Lauren, additional, Carlstrom, Bonnie, additional, Farnsworth, Kellian, additional, Fry, Cyrie, additional, Chandler, Melissa, additional, Jenkins, Glen, additional, Creel, Donnel, additional, Birch, David, additional, Wang, Yi-Zhong, additional, Rodriguez, Luis, additional, Locke, Kirsten, additional, Klein, Martin, additional, Mejia, Paulina, additional, Jacobson, Samuel G., additional, Schwartz, Sharon B., additional, Matsui, Rodrigo, additional, Gruzensky, Michaela, additional, Roman, Alejandro J., additional, Zrenner, Eberhart, additional, Nasser, Fadi, additional, Hahn, Gesa Astrid, additional, Wilhelm, Barbara, additional, Peters, Tobias, additional, Beier, Benjamin, additional, Koenig, Tilman, additional, Kramer, Susanne, additional, Mohand-Said, Saddek, additional, Audo, Isabelle, additional, Laurent-Coriat, Caroline, additional, Sliesoraityte, Ieva, additional, Zeitz, Christina, additional, Boyard, Fiona, additional, Tran, Minh Ha, additional, Chapon, Mathias, additional, Chaumette, Céline, additional, Amaudruz, Juliette, additional, Ganem, Victoria, additional, Sancho, Serge, additional, Girmens, Aurore, additional, Wojciechowski, Robert, additional, Khan, Shazia, additional, Emmert, David G., additional, Cain, Dennis, additional, Herring, Mark, additional, Bassinger, Jennifer, additional, Liberto, Lisa, additional, Ervin, Ann-Margret, additional, Munoz, Beatriz, additional, Kong, Xiangrong, additional, Dreger, Kurt, additional, Jones, Jennifer, additional, Sadda, Srinivas, additional, Jha, Anamika, additional, Ho, Alex, additional, Kramer, Brendan, additional, Hariri, Amirhossein, additional, Blanquel, Gloria Rebecca, additional, Lam, Ngoc, additional, Pitetta, Sean, additional, Shi, Yue, additional, Tawdros, Rita, additional, Petrossian, Christine, additional, Jenkins, Dennis, additional, Gupta, Muneeswar, additional, Zhou, Yong Dong, additional, Aguilar, Katherine, additional, Chan, Cynthia, additional, Santos, Lisa, additional, Seo, Brian, additional, Sison, Christopher, additional, Perez, Silvia, additional, Chao, Stephanie, additional, Miyasato, Kelly, additional, Higgins, Julia, additional, Luna, Zoila, additional, Menchaca, Anita, additional, Gonzalez, Norma, additional, Robledo, Vicky, additional, Carig, Karen, additional, Baker, Kirstie, additional, Ellenbogen, David, additional, Russell, James, additional, Bluemel, Daniel, additional, Moreno, Alex, additional, Pham, Royal, additional, Sanford, Theo, additional, Linares, Daisy, additional, and Tran, Mei, additional

Published
2016
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40. Whole-Exome Sequencing Identifies LRIT3 Mutations as a Cause of Autosomal-Recessive Complete Congenital Stationary Night Blindness

Author

Zeitz, Christina, Jacobson, Samuel G., Hamel, Christian P., Bujakowska, Kinga, Neuillé, Marion, Orhan, Elise, Zanlonghi, Xavier, Lancelot, Marie-Elise, Michiels, Christelle, Schwartz, Sharon B., Bocquet, Béatrice, Antonio, Aline, Audier, Claire, Letexier, Mélanie, Saraiva, Jean-Paul, Luu, Tien D., Sennlaub, Florian, Nguyen, Hoan, Poch, Olivier, Dollfus, Hélène, Lecompte, Odile, Kohl, Susanne, Sahel, José-Alain, Bhattacharya, Shomi S., and Audo, Isabelle

Published
2013
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41. Extending the sufficient component cause model to describe the Stable Unit Treatment Value Assumption (SUTVA)

Author

Campbell, Ulka Bawle, Gatto, Nicolle M., and Schwartz, Sharon B.

Subjects
Epidemiology
Abstract

Causal inference requires an understanding of the conditions under which association equals causation. The exchangeability or no confounding assumption is well known and well understood as central to this task. More recently the epidemiologic literature has described additional assumptions related to the stability of causal effects. In this paper we extend the Sufficient Component Cause Model to represent one expression of this stability assumption--the Stable Unit Treatment Value Assumption. Approaching SUTVA from an SCC model helps clarify what SUTVA is and reinforces the connections between interaction and SUTVA.

Published
2012
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42. Gene Therapy for Leber Congenital Amaurosis caused by RPE65 mutations: Safety and Efficacy in Fifteen Children and Adults Followed up to Three Years

Author

Jacobson, Samuel G., Cideciyan, Artur V., Ratnakaram, Ramakrishna, Heon, Elise, Schwartz, Sharon B., Roman, Alejandro J., Peden, Marc C., Aleman, Tomas S., Boye, Sanford L., Sumaroka, Alexander, Conlon, Thomas J., Calcedo, Roberto, Pang, Ji-jing, Erger, Kirsten E., Olivares, Melani B., Mullins, Cristina L., Swider, Malgorzata, Kaushal, Shalesh, Feuer, Willam J., Iannaccone, Alessandro, Fishman, Gerald A., Stone, Edwin M., Byrne, Barry J., and Hauswirth, William W.

Subjects
Adult, Male, cis-trans-Isomerases, Adolescent, Genetic Vectors, Leber Congenital Amaurosis, Visual Acuity, Article, Young Adult, Humans, Child, Eye Proteins, Pupil, Genetic Therapy, Dependovirus, Treatment Outcome, Mutation, Female, Injections, Intraocular, Visual Fields, Carrier Proteins, Photic Stimulation, Psychomotor Performance, Tomography, Optical Coherence, Follow-Up Studies, Photoreceptor Cells, Vertebrate
Abstract

To determine the safety and efficacy of subretinal gene therapy in the RPE65 form of Leber congenital amaurosis using recombinant adeno-associated virus 2 (rAAV2) carrying the RPE65 gene.Open-label, dose-escalation phase I study of 15 patients (range, 11-30 years of age) evaluated after subretinal injection of the rAAV2- RPE65 vector into the worse-functioning eye. Five cohorts represented 4 dose levels and 2 different injection strategies.Primary outcomes were systemic and ocular safety. Secondary outcomes assayed visual function with dark-adapted full-field sensitivity testing and visual acuity with Early Treatment Diabetic Retinopathy Study charts. Further assays included immune responses to the vector, static visual fields, pupillometry, mobility performance, and optical coherence tomography.No systemic toxicity was detected; ocular adverse events were related to surgery. Visual function improved in all patients to different degrees; improvements were localized to treated areas. Cone and rod sensitivities increased significantly in the study eyes but not in the control eyes. Minor acuity improvements were recorded in many study and control eyes. Major acuity improvements occurred in study eyes with the lowest entry acuities and parafoveal fixation loci treated with subretinal injections. Other patients with better foveal structure lost retinal thickness and acuity after subfoveal injections.Gene therapy for Leber congenital amaurosis caused by RPE65 mutations is sufficiently safe and substantially efficacious in the extrafoveal retina. There is no benefit and some risk in treating the fovea. No evidence of age-dependent effects was found. Our results point to specific treatment strategies for subsequent phases.Gene therapy for inherited retinal disease has the potential to become a future part of clinical practice.clinicaltrials.gov Identifier: NCT00481546.

Published
2011

46. Blue Cone Monochromacy: Visual Function and Efficacy Outcome Measures for Clinical Trials

Author

Luo, Xunda, primary, Cideciyan, Artur V., additional, Iannaccone, Alessandro, additional, Roman, Alejandro J., additional, Ditta, Lauren C., additional, Jennings, Barbara J., additional, Yatsenko, Svetlana A., additional, Sheplock, Rebecca, additional, Sumaroka, Alexander, additional, Swider, Malgorzata, additional, Schwartz, Sharon B., additional, Wissinger, Bernd, additional, Kohl, Susanne, additional, and Jacobson, Samuel G., additional

Published
2015
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48. Are social causes so different from all other causes? A comment on Sander Greenland

Author

Susser, Ezra S. and Schwartz, Sharon B.

Subjects
Public health, Epidemiology, Medical policy
Abstract

Sander Greenland's elegant paper raises deep questions about the way in which we choose and evaluate public health actions. We agree with the main thrust of his argument with respect to public health policy. We have some concerns, however, about his treatment of social causes, and on this point we focus our critique.

Published
2005
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