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1. Flortaucipir tau PET findings from former professional and college American football players in the DIAGNOSE CTE research project.

Author

Su, Yi, Protas, Hillary, Luo, Ji, Chen, Kewei, Alosco, Michael, Adler, Charles, Balcer, Laura, Bernick, Charles, Au, Rhoda, Banks, Sarah, Barr, William, Coleman, Michael, Dodick, David, Katz, Douglas, Marek, Kenneth, McClean, Michael, McKee, Ann, Mez, Jesse, Daneshvar, Daniel, Palmisano, Joseph, Peskind, Elaine, Turner, Robert, Wethe, Jennifer, Johnson, Keith, Tripodis, Yorghos, Cummings, Jeffrey, Shenton, Martha, Stern, Robert, Reiman, Eric, and Rabinovici, Gil

Subjects
CTE, PET, Tau, flortaucipir, football, Male, Humans, Middle Aged, Chronic Traumatic Encephalopathy, Football, tau Proteins, Positron-Emission Tomography, Brain Injuries, Traumatic, Carbolines
Abstract

INTRODUCTION: Tau is a key pathology in chronic traumatic encephalopathy (CTE). Here, we report our findings in tau positron emission tomography (PET) measurements from the DIAGNOSE CTE Research Project. METHOD: We compare flortaucipir PET measures from 104 former professional players (PRO), 58 former college football players (COL), and 56 same-age men without exposure to repetitive head impacts (RHI) or traumatic brain injury (unexposed [UE]); characterize their associations with RHI exposure; and compare players who did or did not meet diagnostic criteria for traumatic encephalopathy syndrome (TES). RESULTS: Significantly elevated flortaucipir uptake was observed in former football players (PRO+COL) in prespecified regions (p

Published
2024

2. Association of Loneliness with Functional Connectivity MRI, Amyloid-β PET, and Tau PET Neuroimaging Markers of Vulnerability for Alzheimers Disease.

Author

Zhao, Amanda, Balcer, Laura, Himali, Jayandra, ODonnell, Adrienne, Rahimpour, Yashar, Decarli, Charles, Gonzales, Mitzi, Aparicio, Hugo, Ramos-Cejudo, Jaime, Kenney, Rachel, Beiser, Alexa, Seshadri, Sudha, and Salinas, Joel

Subjects
Alzheimer’s disease, amyloid, dementia, functional neuroimaging, loneliness, longitudinal studies, tau protein, Humans, Alzheimer Disease, Male, Positron-Emission Tomography, Female, Magnetic Resonance Imaging, Amyloid beta-Peptides, Cross-Sectional Studies, tau Proteins, Loneliness, Middle Aged, Aged, Brain, Biomarkers, Neuroimaging
Abstract

BACKGROUND: Loneliness has been declared an epidemic associated with negative physical, mental, and cognitive health outcomes such as increased dementia risk. Less is known about the relationship between loneliness and advanced neuroimaging correlates of Alzheimers disease (AD). OBJECTIVE: To assess whether loneliness was associated with advanced neuroimaging markers of AD using neuroimaging data from Framingham Heart Study (FHS) participants without dementia. METHODS: In this cross-sectional observational analysis, we used functional connectivity MRI (fcMRI), amyloid-β (Aβ) PET, and tau PET imaging data collected between 2016 and 2019 on eligible FHS cohort participants. Loneliness was defined as feeling lonely at least one day in the past week. The primary fcMRI marker was Default Mode Network intra-network connectivity. The primary PET imaging markers were Aβ deposition in precuneal and FLR (frontal, lateral parietal and lateral temporal, retrosplenial) regions, and tau deposition in the amygdala, entorhinal, and rhinal regions. RESULTS: Of 381 participants (mean age 58 [SD 10]) who met inclusion criteria for fcMRI analysis, 5% were classified as lonely (17/381). No association was observed between loneliness status and network changes. Of 424 participants (mean age 58 [SD = 10]) meeting inclusion criteria for PET analyses, 5% (21/424) were lonely; no associations were observed between loneliness and either Aβ or tau deposition in primary regions of interest. CONCLUSIONS: In this cross-sectional study, there were no observable associations between loneliness and select fcMRI, Aβ PET, and tau PET neuroimaging markers of AD risk. These findings merit further investigation in prospective studies of community-based cohorts.

Published
2024

5. Inflammatory biomarkers for neurobehavioral dysregulation in former American football players: findings from the DIAGNOSE CTE Research Project

Author

van Amerongen, Suzan, Pulukuri, Surya V., Tuz-Zahra, Fatima, Tripodis, Yorghos, Cherry, Jonathan D., Bernick, Charles, Geda, Yonas E., Wethe, Jennifer V., Katz, Douglas I., Alosco, Michael L., Adler, Charles H., Balcer, Laura J., Ashton, Nicholas J., Blennow, Kaj, Zetterberg, Henrik, Daneshvar, Daniel H., Colasurdo, Elizabeth A., Iliff, Jeffrey J., Li, Gail, Peskind, Elaine R., Shenton, Martha E., Reiman, Eric M., Cummings, Jeffrey L., and Stern, Robert A.

Published
2024
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6. MOG Antibody–Associated Disease and Thymic Hyperplasia

Author

Hurtubise, Brigitte, Frohman, Elliot M, Galetta, Steven, Balcer, Laura J, Frohman, Teresa C, Lisak, Robert P, Newsome, Scott D, Graves, Jennifer S, Zamvil, Scott S, and Amezcua, Lilyana

Subjects
Biomedical and Clinical Sciences, Immunology, Autoimmune Disease, Neurosciences, Multiple Sclerosis, Neurodegenerative, Rare Diseases, Brain Disorders, 2.1 Biological and endogenous factors, Aetiology, Inflammatory and immune system, Humans, Myelin-Oligodendrocyte Glycoprotein, Thymus Hyperplasia, Autoantibodies, Optic Neuritis, Immune System Diseases
Abstract

Myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is a recently described CNS inflammatory disorder that may manifest with optic neuritis, myelitis, seizures, and/or acute disseminated encephalomyelitis. While MOG-specific antibodies in patients with MOGAD are IgG1, a T-cell-dependent antibody isotype, immunologic mechanisms of this disease are not fully understood. Thymic hyperplasia can be associated with certain autoimmune diseases. In this report we describe a case of MOGAD associated with thymic hyperplasia in a young adult.

Published
2023

7. Associations between near end-of-life flortaucipir PET and postmortem CTE-related tau neuropathology in six former American football players

Author

Alosco, Michael L, Su, Yi, Stein, Thor D, Protas, Hillary, Cherry, Jonathan D, Adler, Charles H, Balcer, Laura J, Bernick, Charles, Pulukuri, Surya Vamsi, Abdolmohammadi, Bobak, Coleman, Michael J, Palmisano, Joseph N, Tripodis, Yorghos, Mez, Jesse, Rabinovici, Gil D, Marek, Kenneth L, Beach, Thomas G, Johnson, Keith A, Huber, Bertrand Russell, Koerte, Inga, Lin, Alexander P, Bouix, Sylvain, Cummings, Jeffrey L, Shenton, Martha E, Reiman, Eric M, McKee, Ann C, and Stern, Robert A

Subjects
Biomedical and Clinical Sciences, Clinical Sciences, Aging, Physical Injury - Accidents and Adverse Effects, Acquired Cognitive Impairment, Brain Disorders, Alzheimer's Disease, Dementia, Neurosciences, Neurodegenerative, Alzheimer's Disease including Alzheimer's Disease Related Dementias (AD/ADRD), Neurological, Humans, Alzheimer Disease, Autopsy, Brain, Brain Injuries, Traumatic, Chronic Traumatic Encephalopathy, Death, Football, Positron-Emission Tomography, tau Proteins, Biomarkers, Chronic traumatic encephalopathy, Neurodegenerative disease, Positron emission tomography imaging, Repetitive head impacts, Tau, Flortaucipir, DIAGNOSE C. T. E. Research Project, Other Physical Sciences, Nuclear Medicine & Medical Imaging, Clinical sciences
Abstract

PurposeFlourine-18-flortaucipir tau positron emission tomography (PET) was developed for the detection for Alzheimer's disease. Human imaging studies have begun to investigate its use in chronic traumatic encephalopathy (CTE). Flortaucipir-PET to autopsy correlation studies in CTE are needed for diagnostic validation. We examined the association between end-of-life flortaucipir PET and postmortem neuropathological measurements of CTE-related tau in six former American football players.MethodsThree former National Football League players and three former college football players who were part of the DIAGNOSE CTE Research Project died and agreed to have their brains donated. The six players had flortaucipir (tau) and florbetapir (amyloid) PET prior to death. All brains from the deceased participants were neuropathologically evaluated for the presence of CTE. On average, the participants were 59.0 (SD = 9.32) years of age at time of PET. PET scans were acquired 20.33 (SD = 13.08) months before their death. Using Spearman correlation analyses, we compared flortaucipir standard uptake value ratios (SUVRs) to digital slide-based AT8 phosphorylated tau (p-tau) density in a priori selected composite cortical, composite limbic, and thalamic regions-of-interest (ROIs).ResultsFour brain donors had autopsy-confirmed CTE, all with high stage disease (n = 3 stage III, n = 1 stage IV). Three of these four met criteria for the clinical syndrome of CTE, known as traumatic encephalopathy syndrome (TES). Two did not have CTE at autopsy and one of these met criteria for TES. Concomitant pathology was only present in one of the non-CTE cases (Lewy body) and one of the CTE cases (motor neuron disease). There was a strong association between flortaucipir SUVRs and p-tau density in the composite cortical (ρ = 0.71) and limbic (ρ = 0.77) ROIs. Although there was a strong association in the thalamic ROI (ρ = 0.83), this is a region with known off-target binding. SUVRs were modest and CTE and non-CTE cases had overlapping SUVRs and discordant p-tau density for some regions.ConclusionsFlortaucipir-PET could be useful for detecting high stage CTE neuropathology, but specificity to CTE p-tau is uncertain. Off-target flortaucipir binding in the hippocampus and thalamus complicates interpretation of these associations. In vivo biomarkers that can detect the specific p-tau of CTE across the disease continuum are needed.

Published
2023

9. Amyloid PET across the cognitive spectrum in former professional and college American football players: findings from the DIAGNOSE CTE Research Project

Author

Stern, Robert A., Trujillo-Rodriguez, Diana, Tripodis, Yorghos, Pulukuri, Surya V., Alosco, Michael L., Adler, Charles H., Balcer, Laura J., Bernick, Charles, Baucom, Zachary, Marek, Kenneth L., McClean, Michael D., Johnson, Keith A., McKee, Ann C., Stein, Thor D., Mez, Jesse, Palmisano, Joseph N., Cummings, Jeffrey L., Shenton, Martha E., and Reiman, Eric M.

Published
2023
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10. Neuropsychological test performance of former American football players

Author

Alosco, Michael L., Barr, William B., Banks, Sarah J., Wethe, Jennifer V., Miller, Justin B., Pulukuri, Surya Vamsi, Culhane, Julia, Tripodis, Yorghos, Adler, Charles H., Balcer, Laura J., Bernick, Charles, Mariani, Megan L., Cantu, Robert C., Dodick, David W., McClean, Michael D., Au, Rhoda, Mez, Jesse, Turner, II, Robert W., Palmisano, Joseph N., Martin, Brett, Hartlage, Kaitlin, Cummings, Jeffrey L., Reiman, Eric M., Shenton, Martha E., and Stern, Robert A.

Published
2023
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12. Examination of parkinsonism in former elite American football players

Author

Alosco, Michael L., Adler, Charles H., Dodick, David W., Tripodis, Yorghos, Balcer, Laura J., Bernick, Charles, Banks, Sarah J., Barr, William B., Wethe, Jennifer V., Palmisano, Joseph N., Martin, Brett, Hartlage, Kaitlin, Cantu, Robert C., Geda, Yonas E., Katz, Douglas I., Mez, Jesse, Cummings, Jeffery L., Shenton, Martha E., Reiman, Eric M., and Stern, Robert A.

Published
2024
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13. Artificial intelligence extension of the OSCAR-IB criteria.

Author

Petzold, Axel, Albrecht, Philipp, Balcer, Laura, Bekkers, Erik, Brandt, Alexander U, Calabresi, Peter A, Deborah, Orla Galvin, Graves, Jennifer S, Green, Ari, Keane, Pearse A, Nij Bijvank, Jenny A, Sander, Josemir W, Paul, Friedemann, Saidha, Shiv, Villoslada, Pablo, Wagner, Siegfried K, Yeh, E Ann, and IMSVISUAL, ERN-EYE Consortium

Subjects
IMSVISUAL, ERN-EYE Consortium, Clinical Sciences, Neurosciences
Abstract

Artificial intelligence (AI)-based diagnostic algorithms have achieved ambitious aims through automated image pattern recognition. For neurological disorders, this includes neurodegeneration and inflammation. Scalable imaging technology for big data in neurology is optical coherence tomography (OCT). We highlight that OCT changes observed in the retina, as a window to the brain, are small, requiring rigorous quality control pipelines. There are existing tools for this purpose. Firstly, there are human-led validated consensus quality control criteria (OSCAR-IB) for OCT. Secondly, these criteria are embedded into OCT reporting guidelines (APOSTEL). The use of the described annotation of failed OCT scans advances machine learning. This is illustrated through the present review of the advantages and disadvantages of AI-based applications to OCT data. The neurological conditions reviewed here for the use of big data include Alzheimer disease, stroke, multiple sclerosis (MS), Parkinson disease, and epilepsy. It is noted that while big data is relevant for AI, ownership is complex. For this reason, we also reached out to involve representatives from patient organizations and the public domain in addition to clinical and research centers. The evidence reviewed can be grouped in a five-point expansion of the OSCAR-IB criteria to embrace AI (OSCAR-AI). The review concludes by specific recommendations on how this can be achieved practically and in compliance with existing guidelines.

Published
2021

26. Reliability of the Neuro-ophthalmic Aspects of a Telemedicine Exam in Concussion (Tele-BCPE) (P11-10.004)

Author

Grossman, Scott, primary, Jack, Alani, additional, Digney, Helena, additional, Bell, Carter, additional, McPherson, Jacob, additional, Saleem, Ghazala, additional, Haider, Mohammad, additional, Leddy, John, additional, Willer, Barry, additional, Galetta, Steven, additional, Busis, Neil, additional, Torres, Daniel, additional, and Balcer, Laura, additional

Published
2024
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27. Life stressors significantly impact long-term outcomes and post-acute symptoms 12-months after COVID-19 hospitalization

Author

Agarwal, Shashank, Andino, Andres, Arena, Vito, Baskharoun, Samuel, Bauman, Kristie, Bell, Lena, Berger, Stephen, Bhagat, Dhristie, Bokhari, Matthew, Bondi, Steven, Canizares, Melanie, Chervinsky, Alexander, Czeisler, Barry M., Dygert, Levi, Fang, Taolin, Flusty, Brent, Friedman, Daniel, Friedman, David, Fuchs, Benjamin, Granger, Andre, Gratch, Daniel, Gurin, Lindsey, Gutman, Josef, Hasanaj, Lisena, Holmes, Manisha, Horng, Jennifer, Huang, Joshua, Ishii, Haruki, Jauregui, Ruben, Ji, Yuan, Kahn, D. Ethan, Koch, Ethan, Krieger, Penina, Kvernland, Alexandra, Lalchan, Rebecca, Lillemoe, Kaitlyn, Lin, Jessica, Liu, Susan B., Madhavan, Maya, Medicherla, Chaitanya, Millar-Vernetti, Patricio, Morgan, Nicole, Olivera, Anlys, Omari, Mirza, Park, George, Patel, Palak, Ristic, Milan, Rosenthal, Jonathan, Sonson, Michael, Snyder, Thomas, Stainman, Rebecca S., Sunwoo, Brian, Talmasov, Daniel, Tamimi, Michael, Thomas, Betsy, Valdes, Eduard, Zhou, Ting, Zhu, Yingrong, Frontera, Jennifer A., Sabadia, Sakinah, Yang, Dixon, de Havenon, Adam, Yaghi, Shadi, Lewis, Ariane, Lord, Aaron S., Melmed, Kara, Thawani, Sujata, Balcer, Laura J., Wisniewski, Thomas, and Galetta, Steven L.

Published
2022
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28. Diagnosis and classification of optic neuritis

Author

Petzold, Axel, Fraser, Clare L, Abegg, Mathias, Alroughani, Raed, Alshowaeir, Daniah, Alvarenga, Regina, Andris, Cécile, Asgari, Nasrin, Barnett, Yael, Battistella, Roberto, Behbehani, Raed, Berger, Thomas, Bikbov, Mukharram M, Biotti, Damien, Biousse, Valerie, Boschi, Antonella, Brazdil, Milan, Brezhnev, Andrei, Calabresi, Peter A, Cordonnier, Monique, Costello, Fiona, Cruz, Franz M, Cunha, Leonardo Provetti, Daoudi, Smail, Deschamps, Romain, de Seze, Jerome, Diem, Ricarda, Etemadifar, Masoud, Flores-Rivera, Jose, Fonseca, Pedro, Frederiksen, Jette, Frohman, Elliot, Frohman, Teresa, Tilikete, Caroline Froment, Fujihara, Kazuo, Gálvez, Alberto, Gouider, Riadh, Gracia, Fernando, Grigoriadis, Nikolaos, Guajardo, José M, Habek, Mario, Hawlina, Marko, Martínez-Lapiscina, Elena H, Hooker, Juzar, Hor, Jyh Yung, Howlett, William, Huang-Link, Yumin, Idrissova, Zhannat, Illes, Zsolt, Jancic, Jasna, Jindahra, Panitha, Karussis, Dimitrios, Kerty, Emilia, Kim, Ho Jin, Lagrèze, Wolf, Leocani, Letizia, Levin, Netta, Liskova, Petra, Liu, Yaou, Maiga, Youssoufa, Marignier, Romain, McGuigan, Chris, Meira, Dália, Merle, Harold, Monteiro, Mário L R, Moodley, Anand, Moura, Frederico, Muñoz, Silvia, Mustafa, Sharik, Nakashima, Ichiro, Noval, Susana, Oehninger, Carlos, Ogun, Olufunmilola, Omoti, Afekhide, Pandit, Lekha, Paul, Friedemann, Rebolleda, Gema, Reddel, Stephen, Rejdak, Konrad, Rejdak, Robert, Rodriguez-Morales, Alfonso J, Rougier, Marie-Bénédicte, Sa, Maria Jose, Sanchez-Dalmau, Bernardo, Saylor, Deanna, Shatriah, Ismail, Siva, Aksel, Stiebel-Kalish, Hadas, Szatmary, Gabriella, Ta, Linh, Tenembaum, Silvia, Tran, Huy, Trufanov, Yevgen, van Pesch, Vincent, Wang, An-Guor, Wattjes, Mike P, Willoughby, Ernest, Zakaria, Magd, Zvornicanin, Jasmin, Balcer, Laura, and Plant, Gordon T

Published
2022
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29. MICK (Mobile Integrated Cognitive Kit) app: Feasibility of an accessible tablet-based rapid picture and number naming task for concussion assessment in a division 1 college football cohort

Author

Bell, Carter A., Rice, Lionel, Balcer, Marc J., Pearson, Randolph, Penning, Brett, Alexander, Aubrey, Roskelly, Jensyn, Nogle, Sally, Tomczyk, Chris P., Tracey, Allie J., Loftin, Megan C., Pollard-McGrandy, Alyssa M., Zynda, Aaron J., Covassin, Tracey, Park, George, Rizzo, John-Ross, Hudson, Todd, Rucker, Janet C., Galetta, Steven L., Balcer, Laura, Kaufman, David I., and Grossman, Scott N.

Published
2022
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31. A young man in “double-trouble”: Hallucinations and cranial nerve palsies: From the National Multiple Sclerosis Society Case Conference Proceedings

Author

Bradshaw, Michael J, Lisak, Robert P, Meltzer, Ethan, Melamed, Esther, Lucas, Ashlea, Freeman, Leorah, Frohman, Teresa C, Costello, Kathleen, Balcer, Laura, Galetta, Steven, Chitnis, Tanuja, Zamvil, Scott S, and Frohman, Elliot M

Subjects
Biomedical and Clinical Sciences, Ophthalmology and Optometry, Adolescent, Anti-N-Methyl-D-Aspartate Receptor Encephalitis, Brain, Congresses as Topic, Cranial Nerve Diseases, Diagnosis, Differential, Encephalitis, Hallucinations, Humans, Male, Neurosciences
Published
2019

36. Toxic Metabolic Encephalopathy in Hospitalized Patients with COVID-19

Author

Frontera, Jennifer A., Melmed, Kara, Fang, Taolin, Granger, Andre, Lin, Jessica, Yaghi, Shadi, Zhou, Ting, Lewis, Ariane, Kurz, Sebastian, Kahn, D. Ethan, de Havenon, Adam, Huang, Joshua, Czeisler, Barry M., Lord, Aaron, Meropol, Sharon B., Troxel, Andrea B., Wisniewski, Thomas, Balcer, Laura, and Galetta, Steven

Published
2021
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37. Normative Data and Conversion Equation for Spectral-Domain Optical Coherence Tomography in an International Healthy Control Cohort

Author

Kenney, Rachel, Liu, Mengling, Hasanaj, Lisena, Joseph, Binu, Al-Hassan, Abdullah A., Balk, Lisanne, Behbehani, Raed, Brandt, Alexander U., Calabresi, Peter A., Frohman, Elliot M., Frohman, Teresa, Havla, Joachim, Hemmer, Bernhard, Jiang, Hong, Knier, Benjamin, Korn, Thomas, Leocani, Letizia, Martínez-Lapiscina, Elena H., Papadopoulou, Athina, Paul, Friedemann, Petzold, Axel, Pisa, Marco, Villoslada, Pablo, Zimmermann, Hanna, Ishikawa, Hiroshi, Schuman, Joel S., Wollstein, Gadi, Chen, Yu, Saidha, Shiv, Thorpe, Lorna E., Galetta, Steven L., and Balcer, Laura J.

Published
2022
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38. Brain morphometry in former American football players: Findings from the DIAGNOSE CTE research project

Author

Arciniega, Hector, primary, Baucom, Zachary H, additional, Tuz-Zahra, Fatima, additional, Tripodis, Yorghos, additional, John, Omar, additional, Carrington, Holly, additional, Kim, Nicholas, additional, Knyazhanskaya, Evdokiya E, additional, Jung, Leonard B, additional, Breedlove, Katherine, additional, Wiegand, Tim L T, additional, Daneshvar, Daniel H, additional, Rushmore, R Jarrett, additional, Billah, Tashrif, additional, Pasternak, Ofer, additional, Coleman, Michael J, additional, Adler, Charles H, additional, Bernick, Charles, additional, Balcer, Laura J, additional, Alosco, Michael L, additional, Koerte, Inga K, additional, Lin, Alexander P, additional, Cummings, Jeffrey L, additional, Reiman, Eric M, additional, Stern, Robert A, additional, Shenton, Martha E, additional, and Bouix, Sylvain, additional

Published
2024
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39. Relapsing White Matter Disease and Subclinical Optic Neuropathy

Author

O'Neill, Kimberly A., primary, Dugue, Andrew, additional, Abreu, Nicolas J., additional, Balcer, Laura J., additional, Branche, Marc, additional, Galetta, Steven, additional, Graves, Jennifer, additional, Kister, Ilya, additional, Magro, Cynthia, additional, Miller, Claire, additional, Newsome, Scott D., additional, Pappas, John, additional, Rucker, Janet, additional, Steigerwald, Connolly, additional, William, Christopher M., additional, Zamvil, Scott S., additional, Grossman, Scott N., additional, and Krupp, Lauren B., additional

Published
2024
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40. Long-term outcomes in patients presenting with optic neuritis: Analyses of the MSBase registry

Author

Kenney, Rachel, Liu, Mengling, Patil, Sachi, Alroughani, Raed, Ampapa, Radek, Bergamaschi, Roberto, Boz, Cavit, Butzkueven, Helmut, Gomez, Jose Cabrera, Cartechini, Elisabetta, Madueño, Sara Eichau, Ferraro, Diana, Grand-Maison, Francois, Granella, Franco, Horakova, Dana, Izquierdo Ayuso, Guillermo, Kalincik, Tomas, Lizrova Preiningerova, Jana, Lugaresi, Alessandra, Onofrj, Marco, Ozakbas, Serkan, Patti, Francesco, Sola, Patrizia, Soysal, Aysun, Spitaleri, Daniele Litterio A., Terzi, Murat, Turkoglu, Recai, van Pesch, Vincent, Saidha, Shiv, Thorpe, Lorna E., Galetta, Steven L., Balcer, Laura J., Kister, Ilya, and Spelman, Tim

Published
2021
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42. A prospective study of long-term outcomes among hospitalized COVID-19 patients with and without neurological complications

Author

Frontera, Jennifer A., Yang, Dixon, Lewis, Ariane, Patel, Palak, Medicherla, Chaitanya, Arena, Vito, Fang, Taolin, Andino, Andres, Snyder, Thomas, Madhavan, Maya, Gratch, Daniel, Fuchs, Benjamin, Dessy, Alexa, Canizares, Melanie, Jauregui, Ruben, Thomas, Betsy, Bauman, Kristie, Olivera, Anlys, Bhagat, Dhristie, Sonson, Michael, Park, George, Stainman, Rebecca, Sunwoo, Brian, Talmasov, Daniel, Tamimi, Michael, Zhu, Yingrong, Rosenthal, Jonathan, Dygert, Levi, Ristic, Milan, Ishii, Haruki, Valdes, Eduard, Omari, Mirza, Gurin, Lindsey, Huang, Joshua, Czeisler, Barry M., Kahn, D. Ethan, Zhou, Ting, Lin, Jessica, Lord, Aaron S., Melmed, Kara, Meropol, Sharon, Troxel, Andrea B., Petkova, Eva, Wisniewski, Thomas, Balcer, Laura, Morrison, Chris, Yaghi, Shadi, and Galetta, Steven

Published
2021
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48. Myelin Oligodendrocyte Glycoprotein Antibody Disease Optic Neuritis: A Structure–Function Paradox?

Author

Ross, Ruby, Kenney, Rachel, Balcer, Laura J., Galetta, Steven L., Krupp, Lauren, O'Neill, Kimberly A., Grossman, Scott N., Fraser, Clare, and Mollan, Susan

Abstract

Supplemental Digital Content is Available in the Text. Background: Myelin oligodendrocyte glycoprotein antibody disease (MOGAD) is a demyelinating disorder that most commonly presents with optic neuritis (ON) and affects children more often than adults. We report 8 pediatric patients with MOG-associated ON and characterize focal optical coherence tomography (OCT) abnormalities over time that help distinguish this condition from the trajectories of other demyelinating disorders. These OCT findings are examined in the context of longitudinal visual function testing. Methods: This is a retrospective case series of 8 pediatric patients with MOG-associated ON who were referred for neuro-ophthalmic evaluation. Longitudinal data for demographics, clinical history, physical examination, and OCT obtained in the course of clinical evaluations were collected through retrospective medical record review. Results: Patients demonstrated acute peripapillary retinal nerve fiber layer (RNFL) thickening in one or both eyes, consistent with optic disc swelling. This was followed by steady patterns of average RNFL thinning, with 9 of 16 eyes reaching significantly low RNFL thickness using OCT platform reference databases (P < 0.01), accompanied by paradoxical recovery of high-contrast visual acuity (HCVA) in every patient. There was no correlation between HCVA and any OCT measures, although contrast sensitivity (CS) was associated with global thickness, PMB thickness, and nasal/temporal (N/T) ratio, and color vision was associated with PMB thickness. There was a lower global and papillomacular bundle (PMB) thickness (P < 0.01) in clinically affected eyes compared with unaffected eyes. There was also a significantly higher N:T ratio in clinically affected eyes compared with unaffected eyes in the acute MOG-ON setting (P = 0.03), but not in the long-term setting. Conclusions: MOG shows a pattern of prominent retinal atrophy, as demonstrated by global RNFL thinning, with remarkable preservation of HCVA but remaining deficits in CS and color vision. These tests may be better clinical markers of vision changes secondary to MOG-ON. Of the OCT parameters measured, PMB thickness demonstrated the most consistent correlation between structural and functional measures. Thus, it may be a more sensitive marker of clinically significant retinal atrophy in MOG-ON. The N:T ratio in acute clinically affected MOG-ON eyes in our study was higher than the N:T ratio of neuromyelitis optica (NMO)-ON eyes and similar to the N:T ratio in multiple sclerosis (MS)-ON eyes as presented in the prior literature. Therefore, MOG may share a more similar pathophysiology to MS compared with NMO. [ABSTRACT FROM AUTHOR]

Published
2024
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