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JOURNAL ARTICLE
Alaa Al Nofal, Christian Hanna, Aida N Lteif, Siobhan T Pittock, Jonathan D Schwartz, Jane E Brumbaugh, Ana L Creo
OBJECTIVES: There have been recent advances assessing copeptin levels in adults with suspected disorders of vasopressin release. Very limited data exits on copeptin levels in children and infants, especially in a critically-ill hospitalized population where hyper- and hypo-natremia are very common. Our objective is to describe the institutional experience assessing copeptin levels in hospitalized infants and children with hyper- or hypo-natremia. METHODS: We performed a single-center retrospective case series of all infants, children, and adolescents who had an ultrasensitive plasma copeptin level obtained between 2019-2021...
April 10, 2023: Journal of Pediatric Endocrinology & Metabolism: JPEM
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JOURNAL ARTICLE
Avi Gadoth, Michelle F Devine, Sean J Pittock, Andrew McKeon, W Oliver Tobin, Thomas R Gossard, Elena F D Cattaneo, Stuart J McCarter, Erik K St Louis
STUDY OBJECTIVES: To describe a case series of patients with prominent sleep disturbances and their polysomnography findings in six patients with dipeptidyl-peptidase-like protein-6 (DPPX) autoimmunity syndrome. METHODS: Of 13 patients with DPPX autoimmunity evaluated at Mayo Clinic, 6 were seen by Sleep Medicine with polysomnography and were assessed with blood and cerebrospinal fluid, neuroimaging, neuropsychological testing, and evaluation tailored to clinical presentation...
July 2023: Journal of Neurology
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JOURNAL ARTICLE
Djillali Annane, Sean J Pittock, Hrishikesh S Kulkarni, Brian W Pickering, Matt R Khoshnevis, Jason L Siegel, Charles A Powell, Pedro Castro, Tomoko Fujii, Derek Dunn, Keisha Smith, Sanjay Mitter, Shamsah Kazani, Austin Kulasekararaj
BACKGROUND: The complement pathway is a potential target for the treatment of severe COVID-19. We evaluated the safety and efficacy of ravulizumab, a terminal complement C5 inhibitor, in patients hospitalised with severe COVID-19 requiring invasive or non-invasive mechanical ventilation. METHODS: This phase 3, multicentre, open-label, randomised controlled trial (ALXN1210-COV-305) enrolled adult patients (aged ≥18 years) from 31 hospitals in France, Japan, Spain, the UK, and the USA...
March 20, 2023: Lancet Respiratory Medicine
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JOURNAL ARTICLE
Susanna Asseyer, Nasrin Asgari, Jeffrey Bennett, Omer Bialer, Yolanda Blanco, Francesca Bosello, Anna Camos-Carreras, Edgar Carnero Contentti, Sara Carta, John Chen, Claudia Chien, Mashina Chomba, Russell C Dale, Josep Dalmau, Kristina Feldmann, Eoin P Flanagan, Caroline Froment Tilikete, Carolina Garcia-Alfonso, Joachim Havla, Mark Hellmann, Ho Jin Kim, Philipp Klyscz, Frank Konietschke, Chiara La Morgia, Marco Lana-Peixoto, Maria Isabel Leite, Netta Levin, Michael Levy, Sara Llufriu, Pablo Lopez, Itay Lotan, Alessandra Lugaresi, Romain Marignier, Sara Mariotto, Susan P Mollan, Cassandra Ocampo, Frederike Cosima Oertel, Maja Olszewska, Jacqueline Palace, Lekha Pandit, José Luis Peralta Uribe, Sean Pittock, Sudarshini Ramanathan, Natthapon Rattanathamsakul, Albert Saiz, Sara Samadzadeh, Bernardo Sanchez-Dalmau, Deanna Saylor, Michael Scheel, Tanja Schmitz-Hübsch, Jemal Shifa, Sasitorn Siritho, Pia S Sperber, Prem S Subramanian, Alon Tiosano, Adi Vaknin-Dembinsky, Alvaro Jose Mejia Vergara, Adi Wilf-Yarkoni, Luis Alfonso Zarco, Hanna G Zimmermann, Friedemann Paul, Hadas Stiebel-Kalish
UNLABELLED: Optic neuritis (ON) often occurs at the presentation of multiple sclerosis (MS), neuromyelitis optica spectrum disorders (NMOSD), and myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease (MOGAD). The recommended treatment of high-dose corticosteroids for ON is based on a North American study population, which did not address treatment timing or antibody serostatus. The Acute Optic Neuritis Network (ACON) presents a global, prospective, observational study protocol primarily designed to investigate the effect of time to high-dose corticosteroid treatment on 6-month visual outcomes in ON...
2023: Frontiers in Neurology
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JOURNAL ARTICLE
Jeffrey L Bennett, Kazuo Fujihara, Ho Jin Kim, Romain Marignier, Kevin C O'Connor, Robert C Sergott, Anthony Traboulsee, Heinz Wiendl, Jens Wuerfel, Scott S Zamvil, Veronica G Anania, Regine Buffels, Thomas Künzel, Annemarie N Lekkerkerker, Siân Lennon-Chrimes, Sean J Pittock
BACKGROUND: Neuromyelitis optica spectrum disorder (NMOSD) is a rare, autoimmune disease of the central nervous system that produces acute, unpredictable relapses causing cumulative neurological disability. Satralizumab, a humanized, monoclonal recycling antibody that targets the interleukin-6 receptor, reduced NMOSD relapse risk vs. placebo in two Phase 3 trials: SAkuraSky (satralizumab ± immunosuppressive therapy; NCT02028884) and SAkuraStar (satralizumab monotherapy; NCT02073279)...
2023: Frontiers in Neurology
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JOURNAL ARTICLE
Sean J Pittock, Michael Barnett, Jeffrey L Bennett, Achim Berthele, Jérôme de Sèze, Michael Levy, Ichiro Nakashima, Celia Oreja-Guevara, Jacqueline Palace, Friedemann Paul, Carlo Pozzilli, Marcus Yountz, Kerstin Allen, Yasmin Mashhoon, Ho Jin Kim
OBJECTIVE: CHAMPION-NMOSD (NCT04201262) is a phase 3, open-label, externally controlled interventional study evaluating the efficacy and safety of the terminal complement inhibitor ravulizumab in adult patients with anti-aquaporin-4 antibody-positive (AQP4+) neuromyelitis optica spectrum disorder (NMOSD). Ravulizumab binds the same complement component 5 epitope as the approved therapeutic eculizumab but has a longer half-life, enabling an extended dosing interval (8 vs 2 weeks)...
June 2023: Annals of Neurology
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JOURNAL ARTICLE
John J Chen, Eoin P Flanagan, Sean J Pittock, Nicole Caroline Stern, Nanthaya Tisavipat, M Tariq Bhatti, Kevin D Chodnicki, Deena A Tajfirouz, Sepideh Jamali, Amy Kunchok, Eric R Eggenberger, Marie A Di Nome, Elias S Sotirchos, Eleni S Vasileiou, Amanda D Henderson, Anthony C Arnold, Laura Bonelli, Heather E Moss, Sylvia Elizabeth Villarreal Navarro, Tanyatuth Padungkiatsagul, Hadas Stiebel-Kalish, Itay Lotan, Adi Wilf-Yarkoni, Helen Danesh-Meyer, Stefan Ivanov, Saif Huda, Mirasol Forcadela, David Hodge, Pascale Poullin, Julie Rode, Caroline Papeix, Samir Saheb, Marine Boudot de la Motte, Catherine Vignal, Yael Hacohen, Julie Pique, Elisabeth Maillart, Romain Deschamps, Bertrand Audoin, Romain Marignier
PURPOSE: To evaluate the effectiveness of plasma exchange (PLEX) for optic neuritis (ON). METHODS: We conducted an international multicenter retrospective study evaluating the outcomes of ON following PLEX. Outcomes were compared to raw data from the Optic Neuritis Treatment Trial (ONTT) using a matched subset. RESULTS: A total of 395 ON attack treated with PLEX from 317 patients were evaluated. The median age was 37 years (range 9 to 75) and 71% were female...
February 21, 2023: American Journal of Ophthalmology
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Hadas Stiebel-Kalish, Kerstin Rubarth, Karny Shouchane-Blum, Alon Tiosano, Itay Lotan, Mark A Hellmann, Adi Wilf-Yarkoni, Omer Bialer, Eoin P Flanagan, Sean J Pittock, M Tariq Bhatti, Tanja Schmitz-Hübsch, Friedemann Paul, Susanna Asseyer, John J Chen
No abstract text is available yet for this article.
February 20, 2023: Scientific Reports
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JOURNAL ARTICLE
A Moth, J Benning, J Glover, V Brown, L Pittock, N Woznitza, K Piper
INTRODUCTION: Radiographers can accurately report musculoskeletal and chest radiographs, but there is paucity of research comparing the performance of reporting radiographers (RRs) with consultant radiologists when interpreting and reporting abdominal radiographs. This study assessed interobserver agreement in the clinical setting between reporting radiographers and a consultant radiologist compared to an expert gastrointestinal radiologist in a District General Hospital. Major discordant reports affecting patient management were also examined...
February 13, 2023: Radiography
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REVIEW
Brenda Banwell, Jeffrey L Bennett, Romain Marignier, Ho Jin Kim, Fabienne Brilot, Eoin P Flanagan, Sudarshini Ramanathan, Patrick Waters, Silvia Tenembaum, Jennifer S Graves, Tanuja Chitnis, Alexander U Brandt, Cheryl Hemingway, Rinze Neuteboom, Lekha Pandit, Markus Reindl, Albert Saiz, Douglas Kazutoshi Sato, Kevin Rostasy, Friedemann Paul, Sean J Pittock, Kazuo Fujihara, Jacqueline Palace
Serum antibodies directed against myelin oligodendrocyte glycoprotein (MOG) are found in patients with acquired CNS demyelinating syndromes that are distinct from multiple sclerosis and aquaporin-4-seropositive neuromyelitis optica spectrum disorder. Based on an extensive literature review and a structured consensus process, we propose diagnostic criteria for MOG antibody-associated disease (MOGAD) in which the presence of MOG-IgG is a core criterion. According to our proposed criteria, MOGAD is typically associated with acute disseminated encephalomyelitis, optic neuritis, or transverse myelitis, and is less commonly associated with cerebral cortical encephalitis, brainstem presentations, or cerebellar presentations...
March 2023: Lancet Neurology
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JOURNAL ARTICLE
Nicholas H Chia, Vyanka Redenbaugh, John J Chen, Sean J Pittock, Eoin P Flanagan
BACKGROUND: Data on corpus callosum involvement in myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) are limited. OBJECTIVE: The objective of the study was to compare callosal lesions in MOGAD, multiple sclerosis (MS), and aquaporin-4-IgG positive neuromyelitis optica spectrum disorder (AQP4+NMOSD). RESULTS: Callosal lesion frequency was similar in MOGAD (38/171 (22%)), MS (24/72 (33%)), and AQP4+NMOSD (18/63 (29%)). Clinical phenotypes included encephalopathy (47%) and focal supratentorial (21%) or infratentorial (45%) deficits...
May 2023: Multiple Sclerosis: Clinical and Laboratory Research
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JOURNAL ARTICLE
Laura Cacciaguerra, Pearse Morris, W Oliver Tobin, John J Chen, Samantha A Banks, Paul Elsbernd, Vyanka Redenbaugh, Jan-Mendelt Tillema, Federico Montini, Elia Sechi, A Sebastian Lopez-Chiriboga, Nicholas Zalewski, Yong Guo, Maria A Rocca, Massimo Filippi, Sean J Pittock, Claudia F Lucchinetti, Eoin P Flanagan
BACKGROUND AND OBJECTIVES: Studies on tumefactive brain lesions in myelin oligodendrocyte glycoprotein-immunoglobulin G (IgG)-associated disease (MOGAD) are lacking. We sought to characterize the frequency clinical, laboratory, and MRI features of these lesions in MOGAD and compare them with those in multiple sclerosis (MS) and aquaporin-4-IgG-positive neuromyelitis optica spectrum disorder (AQP4+NMOSD). METHODS: We retrospectively searched 194 patients with MOGAD and 359 patients with AQP4+NMOSD with clinical/MRI details available from the Mayo Clinic databases and included those with ≥1 tumefactive brain lesion (maximum transverse diameter ≥2 cm) on MRI...
March 28, 2023: Neurology
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JOURNAL ARTICLE
T Lidgett, L Pittock, K Piper, N Woznitza
INTRODUCTION: Radiographers are transitioning from using "Red Dot" annotations to flag abnormal emergency X-ray images, to providing written preliminary clinical evaluation (PCE) diagnostic comments. This study explored the impact of local training on radiographers' PCE participation and accuracy performance during a trial period. METHODS: Ten radiographers provided PCE comments for adult appendicular trauma X-ray examinations performed in the Emergency Department of an English public hospital over a 19 week trial period...
January 19, 2023: Radiography
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JOURNAL ARTICLE
Jennifer A McCombe, Cecilia Zivelonghi, Nisa Vorasoot, Masoud Majed, Eoin P Flanagan, Divyanshu Dubey, Sean J Pittock, Andrew McKeon, Anastasia Zekeridou
α -amino-3-hydroxy-5-methyl-4-isoxazole propionic acid receptor (AMPAR) encephalitis is rare but treatable. We reviewed the clinical and autoantibody profiles of 52 AMPAR-IgG-positive patients (median age 48 years [range 12-81]; 38 female) identified at the Mayo Clinic neuroimmunology laboratory. Main presentation was encephalitis; symptoms other than encephalitis associated with co-existing antibodies (p = 0.004). A tumor was found in 33/44; mostly thymoma. Most patients had partial (14/29) or complete (11/29) immunotherapy response...
February 15, 2023: Journal of Neuroimmunology
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MULTICENTER STUDY
Sara Carta, Álvaro Cobo Calvo, Thaís Armangué, Albert Saiz, Christian Lechner, Kevin Rostásy, Markus Breu, Matthias Baumann, Romana Höftberger, Ilya Ayzenberg, Carolin Schwake, Maria Sepulveda, Eugenia Martínez-Hernández, Gemma Olivé-Cirera, Georgina Arrambide, Mar Tintoré, Raphael Bernard-Valnet, Renaud Du Pasquier, Fabienne Brilot, Sudarshini Ramanathan, Kathrin Schanda, Alberto Gajofatto, Sergio Ferrari, Elia Sechi, Eoin P Flanagan, Sean J Pittock, Vyanka Redenbaugh, Markus Reindl, Romain Marignier, Sara Mariotto
BACKGROUND AND OBJECTIVES: Although the diagnosis of myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) is based on serum MOG antibodies (MOG-Abs) positivity, patients with coexisting or restricted MOG-Abs in the CSF have been reported. The aim of this study is to characterize the relevance of CSF MOG-Abs positivity in clinical practice. METHODS: Eleven medical centers retrospectively collected clinical and laboratory data of adult and pediatric patients with suspected inflammatory CNS disease and MOG-Abs positivity in serum and/or CSF using live cell-based assays...
March 14, 2023: Neurology
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MULTICENTER STUDY
Hadas Stiebel-Kalish, Kerstin Rubarth, Karny Shouchane-Blum, Alon Tiosano, Itay Lotan, Mark A Hellmann, Adi Wilf-Yarkoni, Omer Bialer, Eoin P Flanagan, Sean J Pittock, M Tariq Bhatti, Tanja Schmitz-Hübsch, Paul Friedemann, Susanna Asseyer, John J Chen
Optic neuritis (ON) is a frequent presentation at onset of multiple sclerosis (MS), neuromyelitis optica spectrum disorder (NMOSD), and myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD). The pathophysiology underlying these diseases, especially MOGAD, is still being elucidated. While obesity has been reported to potentially be a risk factor for MS, this has not been explored in NMOSD or MOGAD. We aimed to investigate a possible association between obesity (body mass index [BMI] > 30 kg/m2 ) in patients with MOGAD, aquaporin 4-IgG positive NMOSD (AQP4-IgG+ NMOSD) or MS...
December 9, 2022: Scientific Reports
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JOURNAL ARTICLE
Eoin P Flanagan, Michael D Geschwind, A Sebastian Lopez-Chiriboga, Kyle M Blackburn, Sanchit Turaga, Sophie Binks, Jennifer Zitser, Jeffrey M Gelfand, Gregory S Day, S Richard Dunham, Stefanie J Rodenbeck, Stacey L Clardy, Andrew J Solomon, Sean J Pittock, Andrew McKeon, Divyanshu Dubey, Anastasia Zekeridou, Michel Toledano, Lindsey E Turner, Steven Vernino, Sarosh R Irani
IMPORTANCE: Autoimmune encephalitis misdiagnosis can lead to harm. OBJECTIVE: To determine the diseases misdiagnosed as autoimmune encephalitis and potential reasons for misdiagnosis. DESIGN, SETTING, AND PARTICIPANTS: This retrospective multicenter study took place from January 1, 2014, to December 31, 2020, at autoimmune encephalitis subspecialty outpatient clinics including Mayo Clinic (n = 44), University of Oxford (n = 18), University of Texas Southwestern (n = 18), University of California, San Francisco (n = 17), University of Washington in St Louis (n = 6), and University of Utah (n = 4)...
January 1, 2023: JAMA Neurology
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JOURNAL ARTICLE
(no author information available yet)
In this issue of Journal of Neuro-Ophthalmology, Drs. Marc J. Dinkin, Deborah I. Friedman, and Mark L. Moster discuss the following 6 articles: Mollan SP, Mitchell JL, Yiangou A, Ottridge RS, Alimajstorovic Z, Cartwright DM, Hickman SJ, Markey KA, Singhal R, Tahrani AA, Frew E, Brock K, Sinclair AJ. Association of amount of weight lost after bariatric surgery with intracranial pressure in women with idiopathic intracranial hypertension. Neurology. 2022;99. doi:10.1212/WNL.0000000000200839.Nia AM, Srinivasan VM, Lall R, Kan P...
December 1, 2022: Journal of Neuro-ophthalmology: the Official Journal of the North American Neuro-Ophthalmology Society
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JOURNAL ARTICLE
Cristina Valencia-Sanchez, Yong Guo, Karl N Krecke, John J Chen, Vyanka Redenbaugh, Mayra Montalvo, Paul M Elsbernd, Jan-Mendelt Tillema, Sebastian Lopez-Chiriboga, Adrian Budhram, Elia Sechi, Amy Kunchok, Divyanshu Dubey, Sean J Pittock, Claudia F Lucchinetti, Eoin P Flanagan
Cerebral cortical encephalitis (CCE) is a recently described myelin oligodendrocyte glycoprotein antibody-associated disease (MOGAD) phenotype. In this observational retrospective study, we characterized 19 CCE patients (6.7% of our MOGAD cohort). Headache (n = 15, 79%), seizures (n = 13, 68%), and encephalopathy (n = 12, 63%) were frequent. Magnetic resonance imaging revealed unilateral (n = 12, 63%) or bilateral (n = 7, 37%) cortical T2 hyperintensity and leptomeningeal enhancement (n = 17, 89%)...
February 2023: Annals of Neurology
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JOURNAL ARTICLE
Jeffrey L Bennett, Orhan Aktas, William A Rees, Michael A Smith, Michele Gunsior, Li Yan, Dewei She, Daniel Cimbora, Sean J Pittock, Brian G Weinshenker, Friedemann Paul, Romain Marignier, Dean Wingerchuk, Gary Cutter, Ari Green, Hans-Peter Hartung, Ho Jin Kim, Kazuo Fujihara, Michael Levy, Eliezer Katz, Bruce A C Cree
BACKGROUND: Inebilizumab is an anti-CD19 antibody approved for the treatment of neuromyelitis optica spectrum disorder (NMOSD) in adults with aquaporin-4 autoantibodies. The relationship between B-cell, plasma-cell (PC), and immunoglobulin depletion with longitudinal reductions in NMOSD activity after inebilizumab treatment was characterised post hoc in an exploratory analysis from the N-MOmentum study (NCT02200770). METHODS: Peripheral blood CD20+ B cells, PC gene signature, and immunoglobulin levels were assessed throughout N-MOmentum (follow-up ≥2...
November 9, 2022: EBioMedicine
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