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1. Clinical Characteristics of an Internet-Based Cohort of Patient-Reported Diagnosis of Granulomatosis With Polyangiitis and Microscopic Polyangiitis: Observational Study

Author

Springer, Jason Michael, Kermani, Tanaz A, Sreih, Antoine, Shaw, Dianne G, Young, Kalen, Burroughs, Cristina M, and Merkel, Peter A

Subjects
Computer applications to medicine. Medical informatics, R858-859.7, Public aspects of medicine, RA1-1270
Abstract

BackgroundUtilizing the traditional centers of excellence approach to conduct clinical trials involving rare diseases remains challenging. Patient-based registries have been shown to be both feasible and valid in several other diseases. ObjectiveThis report outlines the clinical characteristics of a large internet registry cohort of participants with a self-reported diagnosis of granulomatosis with polyangiitis or microscopic polyangiitis. MethodsPatients with a self-reported diagnosis of granulomatosis with polyangiitis or microscopic polyangiitis in an internet-based prospective longitudinal cohort (from the Vasculitis Patient-Powered Research Network) were included. Data on symptoms, diagnostic testing, and treatment were collected using standardized questionnaires. ResultsThe study compared patients with granulomatosis with polyangiitis (n=762) and patients with microscopic polyangiitis (n=164). Of the cohort, 97.7% (904/925) reported the diagnosis had been confirmed by a physician. Compared to microscopic polyangiitis, patients with granulomatosis with polyangiitis reported significantly more ear, nose, and throat manifestations (granulomatosis with polyangiitis: 641/723, 88.7%; microscopic polyangiitis: 89/164, 54.3%; z=10.42, P

Published
2020
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2. A study of implementation factors for a novel approach to clinical trials: constructs for consideration in the coordination of direct-to-patient online-based medical research

Author

Cronholm, Peter F., Applequist, Janelle, Krischer, Jeffrey, Fontenot, Ebony, Davis, Trocon, Burroughs, Cristina, McAlear, Carol A., Borchin, Renée, Kullman, Joyce, Carette, Simon, Khalidi, Nader, Koening, Curry, Langford, Carol A., Monach, Paul, Moreland, Larry, Pagnoux, Christian, Specks, Ulrich, Sreih, Antoine G., Ytterberg, Steven R., and Merkel, Peter A.

Published
2024
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3. Hypothyroidism in vasculitis.

Author

Kermani, Tanaz, Cuthbertson, David, Carette, Simon, Khalidi, Nader, Koening, Curry, Langford, Carol, McAlear, Carol, Monach, Paul, Moreland, Larry, Pagnoux, Christian, Seo, Philip, Specks, Ulrich, Sreih, Antoine, Warrington, Kenneth, and Merkel, Peter

Subjects
GCA, Takayasu’s arteritis, antineutrophil cytoplasmic antibody, eosinophilic granulomatosis with polyangiitis, granulomatosis with polyangiitis, hypothyroidism, microscopic polyangiitis, polyarteritis nodosa, vasculitis, Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis, Antibodies, Antineutrophil Cytoplasmic, Churg-Strauss Syndrome, Female, Granulomatosis with Polyangiitis, Humans, Hypothyroidism, Longitudinal Studies, Male, Microscopic Polyangiitis, Middle Aged, Prospective Studies
Abstract

OBJECTIVE: To study the prevalence, risk and clinical associations of hypothyroidism among several forms of vasculitis. METHODS: Patients with GCA, Takayasus arteritis (TAK), PAN and the three forms of ANCA-associated vasculitis [AAV; granulomatosis with polyangiitis (GPA), microscopic polyangiitis and eosinophilic granulomatosis with polyangiitis (EGPA)] enrolled in a prospective, multicentre, longitudinal study were included. RESULTS: The study included data on 2085 patients [63% female, 90% White] with a mean age of 54.6 years (s.d. 17.2). Diagnoses were GCA (20%), TAK (11%), PAN (5%), GPA (42%), microscopic polyangiitis (8%) and EGPA (14%). Hypothyroidism was present in 217 patients (10%) (83% female), with a mean age 59.8 years (s.d. 14.5). Age- and sex-adjusted risk of hypothyroidism was GCA, odds ratio (OR) 0.61 (95% CI 0.41, 0.90); TAK, OR 0.57 (95% CI 0.31, 1.03); PAN, OR 0.59 (95% CI 0.25, 1.38); GPA, OR 1.51 (95% CI 1.12, 2.05); microscopic polyangiitis, OR 1.81 (95% CI 1.18, 2.80) and EGPA, OR 0.82 (95% CI 0.52, 1.30). Among patients with AAV, age- and sex-adjusted risk of hypothyroidism was higher with positive MPO-ANCA [OR 1.89 (95% CI 1.39, 2.76)]. The clinical manifestations of vasculitis were similar in patients with and without hypothyroidism, except transient ischaemic attacks, which were more frequently observed in patients with GCA and hypothyroidism (12% vs 2%; P = 0.001). CONCLUSIONS: Differences in the risk of hypothyroidism among vasculitides may be due to genetic susceptibilities or immune responses. This study confirms an association of hypothyroidism with MPO-ANCA.

Published
2022

4. Risk loci involved in giant cell arteritis susceptibility: a genome-wide association study

Author

Callejas, José Luis, Caminal-Montero, Luis, Corbera-Bellalta, Marc, de Miguel, Eugenio, Díaz-López, J. Bernardino, García-Villanueva, María Jesús, Gómez-Vaquero, Carmen, Guijarro-Rojas, Mercedes, Hidalgo-Conde, Ana, Marí-Alfonso, Begoña, Martínez-Berriochoa, Agustín, Morado, Inmaculada C., Narváez, Javier, Ramentol-Sintas, Marc, Martínez-Zapico, Aleida, Martínez-Taboada, Víctor Manuel, Miranda-Filloy, José A., Monfort, Jordi, Pérez-Conesa, Mercedes, Prieto-González, Sergio, Raya, Enrique, Ríos-Fenández, Raquel, Sánchez-Martín, Julio, Sopeña, Bernardo, Tío, Laura, Unzurrunzaga, Ainhoa, Wordsworth, Oliver, Whitwell, Isobel, Brock, Jessica, Douglas, Victoria, Hettiarachchi, Chamila, Bartholomew, Jacqui, Jarrett, Stephen, Smithson, Gayle, Green, Michael, Brown, Pearl Clark, Lawson, Cathy, Gordon, Esther, Lane, Suzanne, Francis, Rebecca, Dasgupta, Bhaskar, Masunda, Bridgett, Calver, Jo, Patel, Yusuf, Thompson, Charlotte, Gregory, Louise, Levy, Sarah, Menon, Ajit, Thompson, Amy, Dyche, Lisa, Martin, Michael, Li, Charles, Laxminarayan, Ramasharan, Wilcox, Louise, de Guzman, Ralph, Isaacs, John, Lorenzi, Alice, Farley, Ross, Hinchcliffe-Hume, Helain, Bejarano, Victoria, Hope, Susan, Nandi, Pradip, Stockham, Lynne, Wilde, Catherine, Durrant, Donna, Lloyd, Mark, Ye, Chee-Seng, Stevens, Rob, Jilani, Amjad, Collins, David, Pegler, Suzannah, Rivett, Ali, Price, Liz, McHugh, Neil, Skeoch, Sarah, O'Kane, Diana, Kirkwood, Sue, Vadivelu, Saravanan, Pugmire, Susan, Sultan, Shabina, Dooks, Emma, Armstrong, Lisa, Sadik, Hala, Nandagudi, Anupama, Abioye, Tolu, Ramos, Angelo, Gumus, Steph, Sofat, Nidhi, Harrison, Abiola, Seward, Abi, Mollan, Susan, Rahan, Ray, Hawkins, Helen, Emsley, Hedley, Bhargava, Anna, Fleming, Vicki, Hare, Marianne, Raj, Sonia, George, Emmanuel, Allen, Nicola, Hunter, Karl, O'Sullivan, Eoin, Bird, Georgina, Magliano, Malgorzata, Manzo, Katarina, Sanghera, Bobbie, Hutchinson, David, Hammonds, Fiona, Sharma, Poonam, Cooper, Richard, McLintock, Graeme, Al-Saffar, Zaid S., Green, Mike, Elliott, Kerry, Neale, Tania, Mallinson, Janine, Lanyon, Peter, Pradere, Marie-Josephe, Jordan, Natasha, Htut, Ei Phyu, Mushapaidzi, Thelma, Abercrombie, Donna, Wright, Sam, Rowlands, Jane, Mukhtyar, Chetan, Kennedy, James, Makkuni, Damodar, Wilhelmsen, Elva, Kouroupis, Michael, John, Lily, Hughes, Rod, Walsh, Margaret, Buckley, Marie, Mackay, Kirsten, Camden-Woodley, Tracey, Redome, Joan, Pearce, Kirsty, Marianayagam, Thiraupathy, Cruz, Carina, Warner, Elizabeth, Atchia, Ishmael, Walker, Claire, Black, Karen, Duffy, Stacey, Fothergill, Lynda, Jefferey, Rebecca, Toomey, Jackie, Rhys-Dillon, Ceril, Pothecary, Carla, Green, Lauren, Toms, Tracey, Maher, Linda, Davis, Diana, Sayan, Amrinder, Thankachen, Mini, Abusalameh, Mahdi, Record, Jessica, Khan, Asad, Stafford, Sam, Hussein, Azza, Williams, Clare, Fletcher, Alison, Johson, Laura, Burnett, Richard, Moots, Robert, Frankland, Helen, Dale, James, Moar, Kirsten, Hollas, Carol, Parker, Ben, Ridings, Derek, Eapen, Sandhya, John, Sindhu, Robson, Jo, Guthrie, Lucy Belle, Fyfe, Rose, Tait, Moira, Marks, Jonathan, Gunter, Emma, Hernandez, Rochelle, Bhat, Smita, Johnston, Paul, Khurshid, Muhammad, Barclay, Charlotte, Kapur, Deepti, Jeffrey, Helen, Hughes, Anna, Slack, Lauren, Thomas, Eleri, Royon, Anna, Hall, Angela, King, Jon, Nyathi, Sindi, Morris, Vanessa, Castelino, Madhura, Hawkins, Ellie, Tomson, Linda, Singh, Animesh, Nunag, Annalyn, O'Connor, Stella, Rushby, Nathan, Hewitson, Nicola, O'Sunmboye, Kenny, Lewszuk, Adam, Boyles, Louise, Perry, Martin, Williams, Emma, Graver, Christine, Defever, Emmanuel, Kamanth, Sanjeet, Kay, Dominic, Ogor, Joe, Winter, Louise, Horton, Sarah, Welch, Gillian, Hollinshead, Kath, Peters, James, Labao, Julius, Dmello, Andrea, Dawson, Julie, Graham, Denise, De Lord, Denise, Deery, Jo, Hazelton, Tracy, Carette, Simon, Chung, Sharon, Cuthbertson, David, Forbess, Lindsy J., Gewurz-Singer, Ora, Hoffman, Gary S., Koening, Curry L., Maksimowicz-McKinnon, Kathleen M., McAlear, Carol A., Moreland, Larry W., Pagnoux, Christian, Seo, Philip, Specks, Ulrich, Spiera, Robert F., Sreih, Antoine, Warrington, Kenneth J., Monach, Paul A., Weisman, Michael, Borrego-Yaniz, Gonzalo, Ortiz-Fernández, Lourdes, Madrid-Paredes, Adela, Kerick, Martin, Hernández-Rodríguez, José, Mackie, Sarah L, Vaglio, Augusto, Castañeda, Santos, Solans, Roser, Mestre-Torres, Jaume, Khalidi, Nader, Langford, Carol A, Ytterberg, Steven, Beretta, Lorenzo, Govoni, Marcello, Emmi, Giacomo, Cimmino, Marco A, Witte, Torsten, Neumann, Thomas, Holle, Julia, Schönau, Verena, Pugnet, Gregory, Papo, Thomas, Haroche, Julien, Mahr, Alfred, Mouthon, Luc, Molberg, Øyvind, Diamantopoulos, Andreas P, Voskuyl, Alexandre, Daikeler, Thomas, Berger, Christoph T, Molloy, Eamonn S, Blockmans, Daniel, van Sleen, Yannick, Iles, Mark, Sorensen, Louise, Luqmani, Raashid, Reynolds, Gary, Bukhari, Marwan, Bhagat, Shweta, Ortego-Centeno, Norberto, Brouwer, Elisabeth, Lamprecht, Peter, Klapa, Sebastian, Salvarani, Carlo, Merkel, Peter A, Cid, María C, González-Gay, Miguel A, Morgan, Ann W, Martin, Javier, and Márquez, Ana

Published
2024
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5. Urine and Plasma Complement Ba Levels During Disease Flares in Patients With Antineutrophil Cytoplasmic Autoantibody–Associated Vasculitis

Author

Almaani, Salem, Song, Huijuan, Suthanthira, Meshora, Toy, Christopher, Fussner, Lynn A., Meara, Alexa, Nagaraja, Haikady, Cuthbertson, David, Khalidi, Nader A., Koening, Curry L., Langford, Carol A., McAlear, Carol A., Moreland, Larry W., Pagnoux, Christian, Seo, Philip, Specks, Ulrich, Sreih, Antoine G., Warrington, Kenneth J., Monach, Paul A., Merkel, Peter A., Rovin, Brad, and Birmingham, Daniel

Published
2023
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6. Exploratory proteomic analysis implicates the alternative complement cascade in primary CNS vasculitis.

Author

Mandel-Brehm, Caleigh, Retallack, Hanna, Knudsen, Giselle M, Yamana, Alex, Hajj-Ali, Rula A, Calabrese, Leonard H, Tihan, Tarik, Sample, Hannah A, Zorn, Kelsey C, Gorman, Mark P, Madan Cohen, Jennifer, Sreih, Antoine G, Marcus, Jacqueline F, Josephson, S Andrew, Douglas, Vanja C, Gelfand, Jeffrey M, Wilson, Michael R, and DeRisi, Joseph L

Subjects
Brain, Humans, Vasculitis, Central Nervous System, Properdin, Neural Cell Adhesion Molecules, Biopsy, Case-Control Studies, Cohort Studies, Proteomics, Complement Pathway, Alternative, Adolescent, Adult, Middle Aged, Complement System Proteins, Complement C5, Complement C8, Complement C9, Female, Male, Complement C4b-Binding Protein, Mass Spectrometry, Gene Ontology, CD59 Antigens, CD55 Antigens, Neurosciences, Biotechnology, Clinical Sciences, Cognitive Sciences, Neurology & Neurosurgery
Abstract

OBJECTIVE:To identify molecular correlates of primary angiitis of the CNS (PACNS) through proteomic analysis of CSF from a biopsy-proven patient cohort. METHODS:Using mass spectrometry, we quantitatively compared the CSF proteome of patients with biopsy-proven PACNS (n = 8) to CSF from individuals with noninflammatory conditions (n = 11). Significantly enriched molecular pathways were identified with a gene ontology workflow, and high confidence hits within enriched pathways (fold change >1.5 and concordant Benjamini-Hochberg-adjusted p < 0.05 on DeSeq and t test) were identified as differentially regulated proteins. RESULTS:Compared to noninflammatory controls, 283 proteins were differentially expressed in the CSF of patients with PACNS, with significant enrichment of the complement cascade pathway (C4-binding protein, CD55, CD59, properdin, complement C5, complement C8, and complement C9) and neural cell adhesion molecules. A subset of clinically relevant findings were validated by Western blot and commercial ELISA. CONCLUSIONS:In this exploratory study, we found evidence of deregulation of the alternative complement cascade in CSF from biopsy-proven PACNS compared to noninflammatory controls. More specifically, several regulators of the C3 and C5 convertases and components of the terminal cascade were significantly altered. These preliminary findings shed light on a previously unappreciated similarity between PACNS and systemic vasculitides, especially anti-neutrophil cytoplasmic antibody-associated vasculitis. The therapeutic implications of this common biology and the diagnostic and therapeutic utility of individual proteomic findings warrant validation in larger cohorts.

Published
2019

7. Arterial lesions in giant cell arteritis: A longitudinal study.

Author

Kermani, Tanaz, Diab, Sehriban, Sreih, Antoine, Cuthbertson, David, Borchin, Renée, Carette, Simon, Forbess, Lindsy, Koening, Curry, McAlear, Carol, Monach, Paul, Moreland, Larry, Pagnoux, Christian, Seo, Philip, Spiera, Robert, Warrington, Kenneth, Ytterberg, Steven, Langford, Carol, Merkel, Peter, and Khalidi, Nader

Subjects
Aortic aneurysm, Computed tomography angiography, Disease activity, Giant cell arteritis, Large-artery stenosis, Magnetic resonance angiography, Aged, Aorta, Thoracic, Axillary Artery, Computed Tomography Angiography, Female, Giant Cell Arteritis, Humans, Longitudinal Studies, Magnetic Resonance Angiography, Male, Middle Aged, Subclavian Artery
Abstract

OBJECTIVES: To evaluate large-vessel (LV) abnormalities on serial imaging in patients with giant cell arteritis (GCA) and discern predictors of new lesions. METHODS: Clinical and imaging data from patients with GCA (including subjects diagnosed by LV imaging) enrolled in a prospective, multicenter, longitudinal study and/or a randomized clinical trial were included. New arterial lesions were defined as a lesion in a previously unaffected artery. RESULTS: The study included 187 patients with GCA, 146 (78%) female, mean (±SD) age at diagnosis 68.5 ± 8.5 years; 39% diagnosed by LV imaging. At least one arterial lesion was present in 123 (66%) on the first study. The most frequently affected arteries were subclavian (42%), axillary (32%), and thoracic aorta (20%). In 106 patients (57%) with serial imaging, new arterial lesions were noted in 41 patients (39%), all of whom had a baseline abnormality, over a mean (±SD) follow-up of 4.39 (2.22) years. New abnormalities were observed in 33% patients by year 2; clinical features of active disease were present at only 50% of these cases. There were no differences in age, sex, temporal artery biopsy positivity, or disease activity in patients with or without new lesions. CONCLUSIONS: In this cohort of patients with GCA, LV abnormalities on first imaging were common. Development of new arterial lesions occurred in patients with arterial abnormalities at first imaging, often in the absence of symptoms of active disease. Arterial imaging should be considered in all patients with GCA at diagnosis and serial imaging at least in patients with baseline abnormalities.

Published
2019

8. Neutrophil activation in patients with anti-neutrophil cytoplasmic autoantibody-associated vasculitis and large-vessel vasculitis

Author

Michailidou, Despina, Duvvuri, Bhargavi, Kuley, Runa, Cuthbertson, David, Grayson, Peter C., Khalidi, Nader A., Koening, Curry L., Langford, Carol A., McAlear, Carol A., Moreland, Larry W., Pagnoux, Christian, Seo, Philip, Specks, Ulrich, Sreih, Antoine G., Warrington, Kenneth J., Mustelin, Tomas, Monach, Paul A., Merkel, Peter A., and Lood, Christian

Published
2022
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10. Evaluation of damage in giant cell arteritis.

Author

Kermani, Tanaz, Sreih, Antoine, Cuthbertson, David, Carette, Simon, Hoffman, Gary, Khalidi, Nader, Koening, Curry, Langford, Carol, McAlear, Carol, Monach, Paul, Moreland, Larry, Pagnoux, Christian, Seo, Philip, Warrington, Kenneth, Ytterberg, Steven, and Merkel, Peter

Subjects
damage, giant cell arteritis, large-artery manifestations, large-vessel vasculitis, vasculitis, Aged, Eye Diseases, Female, Follow-Up Studies, Giant Cell Arteritis, Humans, Intermittent Claudication, Longitudinal Studies, Male, Middle Aged, Odds Ratio, Prospective Studies, Recurrence, Risk Factors, Severity of Illness Index, Time Factors
Abstract

OBJECTIVES: To evaluate damage and variables associated with damage in GCA. METHODS: Patients with GCA enrolled in a prospective, multicentre, longitudinal study were included. Per-protocol assessments were made with the Vasculitis Damage Index and the Large-Vessel Vasculitis Index of Damage. RESULTS: The study included 204 patients: 156 women (76%), mean age at diagnosis 71.3 years (s.d. 8.3), mean follow-up of 3.5 years (s.d. 1.9). One or more damage item was present in 54% at baseline and 79% at the last follow-up on the Vasculitis Damage Index, and 60% at baseline and 82% at the last follow-up on the Large-Vessel Vasculitis Index of Damage. The most frequently observed damage items were large-artery complications (29% cohort) and ocular (22%). Among 117 patients with new damage, most new items were ocular (63 patients), cardiac/vascular (48) and musculoskeletal (34). Of these, treatment-associated items were frequently observed, including cataracts (46 patients), osteoporosis (22) and weight gain (22). Disease-associated new damage included ischaemic optic neuropathy (3 patients), limb claudication (13), arterial occlusions (10) and damage requiring vascular intervention (10). In univariate analysis, the risk of damage increased 22% for every additional year of disease duration [odds ratio (OR) 1.22 (95% CI 1.04, 1.45)]. In 94 patients enrolled within ⩽90 days of diagnosis of GCA, the risk of new damage at the last follow-up decreased 30% for each additional relapse [OR 0.70 (95% CI 0.51, 0.97)]. CONCLUSIONS: Large-artery complications and ocular manifestations are the most commonly occurring items of damage in GCA. Most new damage is associated with treatment. These findings emphasize the cumulative burden of disease in GCA.

Published
2018

11. Risk loci involved in giant cell arteritis susceptibility: a genome-wide association study

Author

Borrego-Yaniz, Gonzalo, primary, Ortiz-Fernández, Lourdes, additional, Madrid-Paredes, Adela, additional, Kerick, Martin, additional, Hernández-Rodríguez, José, additional, Mackie, Sarah L, additional, Vaglio, Augusto, additional, Castañeda, Santos, additional, Solans, Roser, additional, Mestre-Torres, Jaume, additional, Khalidi, Nader, additional, Langford, Carol A, additional, Ytterberg, Steven, additional, Beretta, Lorenzo, additional, Govoni, Marcello, additional, Emmi, Giacomo, additional, Cimmino, Marco A, additional, Witte, Torsten, additional, Neumann, Thomas, additional, Holle, Julia, additional, Schönau, Verena, additional, Pugnet, Gregory, additional, Papo, Thomas, additional, Haroche, Julien, additional, Mahr, Alfred, additional, Mouthon, Luc, additional, Molberg, Øyvind, additional, Diamantopoulos, Andreas P, additional, Voskuyl, Alexandre, additional, Daikeler, Thomas, additional, Berger, Christoph T, additional, Molloy, Eamonn S, additional, Blockmans, Daniel, additional, van Sleen, Yannick, additional, Iles, Mark, additional, Sorensen, Louise, additional, Luqmani, Raashid, additional, Reynolds, Gary, additional, Bukhari, Marwan, additional, Bhagat, Shweta, additional, Ortego-Centeno, Norberto, additional, Brouwer, Elisabeth, additional, Lamprecht, Peter, additional, Klapa, Sebastian, additional, Salvarani, Carlo, additional, Merkel, Peter A, additional, Cid, María C, additional, González-Gay, Miguel A, additional, Morgan, Ann W, additional, Martin, Javier, additional, Márquez, Ana, additional, Callejas, José Luis, additional, Caminal-Montero, Luis, additional, Corbera-Bellalta, Marc, additional, de Miguel, Eugenio, additional, Díaz-López, J. Bernardino, additional, García-Villanueva, María Jesús, additional, Gómez-Vaquero, Carmen, additional, Guijarro-Rojas, Mercedes, additional, Hidalgo-Conde, Ana, additional, Marí-Alfonso, Begoña, additional, Martínez-Berriochoa, Agustín, additional, Morado, Inmaculada C., additional, Narváez, Javier, additional, Ramentol-Sintas, Marc, additional, Martínez-Zapico, Aleida, additional, Martínez-Taboada, Víctor Manuel, additional, Miranda-Filloy, José A., additional, Monfort, Jordi, additional, Pérez-Conesa, Mercedes, additional, Prieto-González, Sergio, additional, Raya, Enrique, additional, Ríos-Fenández, Raquel, additional, Sánchez-Martín, Julio, additional, Sopeña, Bernardo, additional, Tío, Laura, additional, Unzurrunzaga, Ainhoa, additional, Wordsworth, Oliver, additional, Whitwell, Isobel, additional, Brock, Jessica, additional, Douglas, Victoria, additional, Hettiarachchi, Chamila, additional, Bartholomew, Jacqui, additional, Jarrett, Stephen, additional, Smithson, Gayle, additional, Green, Michael, additional, Brown, Pearl Clark, additional, Lawson, Cathy, additional, Gordon, Esther, additional, Lane, Suzanne, additional, Francis, Rebecca, additional, Dasgupta, Bhaskar, additional, Masunda, Bridgett, additional, Calver, Jo, additional, Patel, Yusuf, additional, Thompson, Charlotte, additional, Gregory, Louise, additional, Levy, Sarah, additional, Menon, Ajit, additional, Thompson, Amy, additional, Dyche, Lisa, additional, Martin, Michael, additional, Li, Charles, additional, Laxminarayan, Ramasharan, additional, Wilcox, Louise, additional, de Guzman, Ralph, additional, Isaacs, John, additional, Lorenzi, Alice, additional, Farley, Ross, additional, Hinchcliffe-Hume, Helain, additional, Bejarano, Victoria, additional, Hope, Susan, additional, Nandi, Pradip, additional, Stockham, Lynne, additional, Wilde, Catherine, additional, Durrant, Donna, additional, Lloyd, Mark, additional, Ye, Chee-Seng, additional, Stevens, Rob, additional, Jilani, Amjad, additional, Collins, David, additional, Pegler, Suzannah, additional, Rivett, Ali, additional, Price, Liz, additional, McHugh, Neil, additional, Skeoch, Sarah, additional, O'Kane, Diana, additional, Kirkwood, Sue, additional, Vadivelu, Saravanan, additional, Pugmire, Susan, additional, Sultan, Shabina, additional, Dooks, Emma, additional, Armstrong, Lisa, additional, Sadik, Hala, additional, Nandagudi, Anupama, additional, Abioye, Tolu, additional, Ramos, Angelo, additional, Gumus, Steph, additional, Sofat, Nidhi, additional, Harrison, Abiola, additional, Seward, Abi, additional, Mollan, Susan, additional, Rahan, Ray, additional, Hawkins, Helen, additional, Emsley, Hedley, additional, Bhargava, Anna, additional, Fleming, Vicki, additional, Hare, Marianne, additional, Raj, Sonia, additional, George, Emmanuel, additional, Allen, Nicola, additional, Hunter, Karl, additional, O'Sullivan, Eoin, additional, Bird, Georgina, additional, Magliano, Malgorzata, additional, Manzo, Katarina, additional, Sanghera, Bobbie, additional, Hutchinson, David, additional, Hammonds, Fiona, additional, Sharma, Poonam, additional, Cooper, Richard, additional, McLintock, Graeme, additional, Al-Saffar, Zaid S., additional, Green, Mike, additional, Elliott, Kerry, additional, Neale, Tania, additional, Mallinson, Janine, additional, Lanyon, Peter, additional, Pradere, Marie-Josephe, additional, Jordan, Natasha, additional, Htut, Ei Phyu, additional, Mushapaidzi, Thelma, additional, Abercrombie, Donna, additional, Wright, Sam, additional, Rowlands, Jane, additional, Mukhtyar, Chetan, additional, Kennedy, James, additional, Makkuni, Damodar, additional, Wilhelmsen, Elva, additional, Kouroupis, Michael, additional, John, Lily, additional, Hughes, Rod, additional, Walsh, Margaret, additional, Buckley, Marie, additional, Mackay, Kirsten, additional, Camden-Woodley, Tracey, additional, Redome, Joan, additional, Pearce, Kirsty, additional, Marianayagam, Thiraupathy, additional, Cruz, Carina, additional, Warner, Elizabeth, additional, Atchia, Ishmael, additional, Walker, Claire, additional, Black, Karen, additional, Duffy, Stacey, additional, Fothergill, Lynda, additional, Jefferey, Rebecca, additional, Toomey, Jackie, additional, Rhys-Dillon, Ceril, additional, Pothecary, Carla, additional, Green, Lauren, additional, Toms, Tracey, additional, Maher, Linda, additional, Davis, Diana, additional, Sayan, Amrinder, additional, Thankachen, Mini, additional, Abusalameh, Mahdi, additional, Record, Jessica, additional, Khan, Asad, additional, Stafford, Sam, additional, Hussein, Azza, additional, Williams, Clare, additional, Fletcher, Alison, additional, Johson, Laura, additional, Burnett, Richard, additional, Moots, Robert, additional, Frankland, Helen, additional, Dale, James, additional, Moar, Kirsten, additional, Hollas, Carol, additional, Parker, Ben, additional, Ridings, Derek, additional, Eapen, Sandhya, additional, John, Sindhu, additional, Robson, Jo, additional, Guthrie, Lucy Belle, additional, Fyfe, Rose, additional, Tait, Moira, additional, Marks, Jonathan, additional, Gunter, Emma, additional, Hernandez, Rochelle, additional, Bhat, Smita, additional, Johnston, Paul, additional, Khurshid, Muhammad, additional, Barclay, Charlotte, additional, Kapur, Deepti, additional, Jeffrey, Helen, additional, Hughes, Anna, additional, Slack, Lauren, additional, Thomas, Eleri, additional, Royon, Anna, additional, Hall, Angela, additional, King, Jon, additional, Nyathi, Sindi, additional, Morris, Vanessa, additional, Castelino, Madhura, additional, Hawkins, Ellie, additional, Tomson, Linda, additional, Singh, Animesh, additional, Nunag, Annalyn, additional, O'Connor, Stella, additional, Rushby, Nathan, additional, Hewitson, Nicola, additional, O'Sunmboye, Kenny, additional, Lewszuk, Adam, additional, Boyles, Louise, additional, Perry, Martin, additional, Williams, Emma, additional, Graver, Christine, additional, Defever, Emmanuel, additional, Kamanth, Sanjeet, additional, Kay, Dominic, additional, Ogor, Joe, additional, Winter, Louise, additional, Horton, Sarah, additional, Welch, Gillian, additional, Hollinshead, Kath, additional, Peters, James, additional, Labao, Julius, additional, Dmello, Andrea, additional, Dawson, Julie, additional, Graham, Denise, additional, De Lord, Denise, additional, Deery, Jo, additional, Hazelton, Tracy, additional, Carette, Simon, additional, Chung, Sharon, additional, Cuthbertson, David, additional, Forbess, Lindsy J., additional, Gewurz-Singer, Ora, additional, Hoffman, Gary S., additional, Koening, Curry L., additional, Maksimowicz-McKinnon, Kathleen M., additional, McAlear, Carol A., additional, Moreland, Larry W., additional, Pagnoux, Christian, additional, Seo, Philip, additional, Specks, Ulrich, additional, Spiera, Robert F., additional, Sreih, Antoine, additional, Warrington, Kenneth J., additional, Monach, Paul A., additional, and Weisman, Michael, additional

Published
2024
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12. MIF and D-DT are potential disease severity modifiers in male MS subjects

Author

Benedek, Gil, Meza-Romero, Roberto, Jordan, Kelley, Zhang, Ying, Nguyen, Ha, Kent, Gail, Li, Jia, Siu, Edwin, Frazer, Jenny, Piecychna, Marta, Du, Xin, Sreih, Antoine, Leng, Lin, Wiedrick, Jack, Caillier, Stacy J, Offner, Halina, Oksenberg, Jorge R, Yadav, Vijayshree, Bourdette, Dennis, Bucala, Richard, and Vandenbark, Arthur A

Subjects
Pharmacology and Pharmaceutical Sciences, Biomedical and Clinical Sciences, Multiple Sclerosis, Autoimmune Disease, Neurodegenerative, Brain Disorders, Genetics, Neurosciences, Aetiology, 2.1 Biological and endogenous factors, Neurological, Adult, Animals, Antigens, Differentiation, B-Lymphocyte, Female, Histocompatibility Antigens Class II, Humans, Intramolecular Oxidoreductases, Macrophage Migration-Inhibitory Factors, Male, Mice, Mice, Knockout, Middle Aged, Polymorphism, Genetic, Severity of Illness Index, multiple sclerosis, disease modifier, sex differences
Abstract

Little is known about mechanisms that drive the development of progressive multiple sclerosis (MS), although inflammatory factors, such as macrophage migration inhibitory factor (MIF), its homolog D-dopachrome tautomerase (D-DT), and their common receptor CD74 may contribute to disease worsening. Our findings demonstrate elevated MIF and D-DT levels in males with progressive disease compared with relapsing-remitting males (RRMS) and female MS subjects, with increased levels of CD74 in females vs. males with high MS disease severity. Furthermore, increased MIF and D-DT levels in males with progressive disease were significantly correlated with the presence of two high-expression promoter polymorphisms located in the MIF gene, a -794CATT5-8 microsatellite repeat and a -173 G/C SNP. Conversely, mice lacking MIF or D-DT developed less-severe signs of experimental autoimmune encephalomyelitis, a murine model of MS, thus implicating both homologs as copathogenic contributors. These findings indicate that genetically controlled high MIF expression (and D-DT) promotes MS progression in males, suggesting that these two factors are sex-specific disease modifiers and raising the possibility that aggressive anti-MIF treatment of clinically isolated syndrome or RRMS males with a high-expresser genotype might slow or prevent the onset of progressive MS. Additionally, selective targeting of MIF:CD74 signaling might provide an effective, trackable therapeutic approach for MS subjects of both sexes.

Published
2017

13. OMERACT Endorsement of Patient-reported Outcome Instruments in Antineutrophil Cytoplasmic Antibody-associated Vasculitis.

Author

Robson, Joanna, Tomasson, Gunnar, Milman, Nataliya, Ashdown, Sue, Boonen, Annelies, Casey, George, Cronholm, Peter, Cuthbertson, David, Dawson, Jill, Direskeneli, Haner, Easley, Ebony, Kermani, Tanaz, Farrar, John, Gebhart, Don, Lanier, Georgia, Luqmani, Raashid, Mahr, Alfred, McAlear, Carol, Peck, Jacqueline, Shea, Beverley, Shea, Judy, Sreih, Antoine, Tugwell, Peter, and Merkel, Peter

Subjects
ANCA-ASSOCIATED VASCULITIS, ICF, OMERACT, PATIENT-REPORTED OUTCOMES, PROMIS, Algorithms, Anti-Inflammatory Agents, Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis, Health Status, Humans, Patient Reported Outcome Measures, Quality of Life, Treatment Outcome
Abstract

OBJECTIVE: The antineutrophil cytoplasmic antibody-associated vasculitides (AAV) are multiorgan diseases. Patients with AAV report impairment in their health-related quality of life (HRQOL) and have different priorities regarding disease assessment compared with physicians. The Outcome Measures in Rheumatology (OMERACT) Vasculitis Working Group previously received endorsement for a core set of domains in AAV. Two approaches to measure patient-reported outcomes (PRO) were presented at OMERACT 2016. METHODS: A novel 5-step tool was used to facilitate assessment of the instruments by delegates: the OMERACT Filter 2.0 Instrument Selection Algorithm, with a red-amber-green checklist of questions, including (1) good match with domain (face and content validity), (2) feasibility, (3) do numeric scores make sense (construct validity)?, (4) overall ratings of discrimination, and (5) can individual thresholds of meaning be defined? Delegates gave an overall endorsement. Three generic Patient-Reported Outcomes Measurement Information System (PROMIS) instruments (fatigue, physical functioning, and pain interference) and a disease-specific PRO, the AAV-PRO (6 domains related to symptoms and HRQOL), were presented. RESULTS: OMERACT delegates endorsed the use of the PROMIS instruments for fatigue, physical functioning, and pain interference (87.6% overall endorsement) and the disease-specific AAV-PRO instrument (89.4% overall endorsement). CONCLUSION: The OMERACT Vasculitis Working Group gained endorsement by OMERACT for use of the PROMIS and the AAV-PRO in clinical trials of vasculitis. These instruments are complementary to each other. The PROMIS and the AAV-PRO need further work to assess their utility in longitudinal settings, including their ability to discriminate between treatments of varying efficacy in the setting of a randomized controlled trial.

Published
2017

16. Mitochondrial-mediated inflammation and platelet activation in giant cell arteritis

Author

Michailidou, Despina, primary, Grayson, Peter C., additional, Hermanson, Payton, additional, Chapa, Jorge Armando Gonzalez, additional, Cuthbertson, David, additional, Khalidi, Nader A., additional, Koening, Curry L., additional, Langford, Carol A., additional, McAlear, Carol A., additional, Moreland, Larry W., additional, Pagnoux, Christian, additional, Seo, Philip, additional, Sreih, Antoine G., additional, Warrington, Kenneth J., additional, Monach, Paul A., additional, Merkel, Peter A., additional, and Lood, Christian, additional

Published
2023
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20. Considerations for Colorectal Neoplasia Detection in Inflammatory Bowel Disease Clinical Trials.

Author

Yang, Mira M., Usiskin, Keith, Ahmad, Harris A., Ather, Shabana, Sreih, Antoine, Canavan, James B., Farraye, Francis A., and Ma, Christopher

Subjects
INFLAMMATORY bowel diseases, CLINICAL trials, TUMORS, COLORECTAL cancer
Abstract

Background: High-quality colonoscopic surveillance can lead to earlier and increased detection of colorectal neoplasia in patients with inflammatory bowel disease (IBD). In IBD clinical trials, endoscopy is used to assess mucosal disease activity before and after treatment but also provides an opportunity to surveil for colorectal neoplasia during follow-up. Summary: Best practices for colorectal cancer identification in IBD clinical trials require engagement and collaboration between the clinical trial sponsor, site endoscopist and/or principal investigator, and central read team. Each team member has unique responsibilities for maximizing dysplasia detection in IBD trials. Key Messages: Sponsors should work in accordance with scientific guidelines to standardize imaging procedures, design the protocol to ensure the trial population is safeguarded, and oversee trial conduct. The site endoscopist should remain updated on best practices to tailor sponsor protocol-required procedures to patient needs, examine the mucosa for disease activity and potential dysplasia during all procedures, and provide optimal procedure videos for central read analysis. Central readers may detect dysplasia or colorectal cancer and a framework to report these findings to trial sponsors is essential. Synergistic relationships between all team members in IBD clinical trials provide an important opportunity for extended endoscopic evaluation and colorectal neoplasia identification. [ABSTRACT FROM AUTHOR]

Published
2024
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22. Neutrophil extracellular trap formation in anti-neutrophil cytoplasmic antibody-associated and large-vessel vasculitis

Author

Michailidou, Despina, primary, Kuley, Runa, additional, Wang, Ting, additional, Hermanson, Payton, additional, Grayson, Peter C., additional, Cuthbertson, David, additional, Khalidi, Nader A., additional, Koening, Curry L., additional, Langford, Carol A., additional, McAlear, Carol A., additional, Moreland, Larry W., additional, Pagnoux, Christian, additional, Seo, Philip, additional, Specks, Ulrich, additional, Sreih, Antoine G., additional, Warrington, Kenneth J., additional, Monach, Paul A., additional, Merkel, Peter A., additional, and Lood, Christian, additional

Published
2023
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23. Rituximab versus azathioprine for maintenance of remission for patients with ANCA-associated vasculitis and relapsing disease: an international randomised controlled trial

Author

Smith, Rona M, primary, Jones, Rachel B, additional, Specks, Ulrich, additional, Bond, Simon, additional, Nodale, Marianna, additional, Al-jayyousi, Reem, additional, Andrews, Jacqueline, additional, Bruchfeld, Annette, additional, Camilleri, Brian, additional, Carette, Simon, additional, Cheung, Chee Kay, additional, Derebail, Vimal, additional, Doulton, Tim, additional, Ferraro, Alastair, additional, Forbess, Lindsy, additional, Fujimoto, Shouichi, additional, Furuta, Shunsuke, additional, Gewurz-Singer, Ora, additional, Harper, Lorraine, additional, Ito-Ihara, Toshiko, additional, Khalidi, Nader, additional, Klocke, Rainer, additional, Koening, Curry, additional, Komagata, Yoshinori, additional, Langford, Carol, additional, Lanyon, Peter, additional, Luqmani, Raashid, additional, McAlear, Carol, additional, Moreland, Larry W, additional, Mynard, Kim, additional, Nachman, Patrick, additional, Pagnoux, Christian, additional, Peh, Chen Au, additional, Pusey, Charles, additional, Ranganathan, Dwarakanathan, additional, Rhee, Rennie L, additional, Spiera, Robert, additional, Sreih, Antoine G, additional, Tesar, Vladamir, additional, Walters, Giles, additional, Wroe, Caroline, additional, Jayne, David, additional, and Merkel, Peter A, additional

Published
2023
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24. Rituximab versus azathioprine for maintenance of remission for patients with ANCA-associated vasculitis and relapsing disease: an international randomised controlled trial

Author

Smith, Rona M, Jones, Rachel B, Specks, Ulrich, Bond, Simon, Nodale, Marianna, Al-Jayyousi, Reem, Andrews, Jacqueline, Bruchfeld, Annette, Camilleri, Brian, Carette, Simon, Cheung, Chee Kay, Derebail, Vimal, Doulton, Tim, Ferraro, Alastair, Forbess, Lindsy, Fujimoto, Shouichi, Furuta, Shunsuke, Gewurz-Singer, Ora, Harper, Lorraine, Ito-Ihara, Toshiko, Khalidi, Nader, Klocke, Rainer, Koening, Curry, Komagata, Yoshinori, Langford, Carol, Lanyon, Peter, Luqmani, Raashid, McAlear, Carol, Moreland, Larry W, Mynard, Kim, Nachman, Patrick, Pagnoux, Christian, Peh, Chen Au, Pusey, Charles, Ranganathan, Dwarakanathan, Rhee, Rennie L, Spiera, Robert, Sreih, Antoine G, Tesar, Vladamir, Walters, Giles, Wroe, Caroline, Jayne, David, Merkel, Peter A, RITAZAREM Co-Investigators, Smith, Rona M [0000-0002-7438-5156], Nodale, Marianna [0000-0002-0333-8918], Fujimoto, Shouichi [0000-0002-0025-4030], Furuta, Shunsuke [0000-0003-2482-2685], Harper, Lorraine [0000-0003-1343-9234], Khalidi, Nader [0000-0002-8270-2617], Rhee, Rennie L [0000-0002-4907-0304], Spiera, Robert [0000-0003-2911-6800], Walters, Giles [0000-0003-4854-9353], Jayne, David [0000-0002-1712-0637], and Apollo - University of Cambridge Repository

Subjects
granulomatosis with polyangiitis, Remission Induction, Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis, Antibodies, Antineutrophil Cytoplasmic, rituximab, Treatment Outcome, Recurrence, Azathioprine, B-lymphocytes, therapeutics, Humans, systemic vasculitis, Cyclophosphamide, Immunosuppressive Agents
Abstract

Peer reviewed: True, Acknowledgements: The RITAZAREM trial is directed by the European Vasculitis Society and the Vasculitis Clinical Research Consortium (VCRC). The primary sponsor is Cambridge University Hospitals NHS Foundation Trust, and there are collaboration and data-sharing agreements with the University of Pennsylvania and the University of Miyazaki and Okayama University in Japan., Funder: Research Committee on Intractable Vasculitides, the Ministry of Health, Labour and Welfare of Japan, OBJECTIVE: Following induction of remission with rituximab in anti-neutrophil cytoplasmic antibody-associated vasculitis (AAV) relapse rates are high, especially in patients with history of relapse. Relapses are associated with increased exposure to immunosuppressive medications, the accrual of damage and increased morbidity and mortality. The RITAZAREM trial compared the efficacy of repeat-dose rituximab to daily oral azathioprine for prevention of relapse in patients with relapsing AAV in whom remission was reinduced with rituximab. METHODS: RITAZAREM was an international randomised controlled, open-label, superiority trial that recruited 188 patients at the time of an AAV relapse from 29 centres in seven countries between April 2013 and November 2016. All patients received rituximab and glucocorticoids to reinduce remission. Patients achieving remission by 4 months were randomised to receive rituximab intravenously (1000 mg every 4 months, through month 20) (85 patients) or azathioprine (2 mg/kg/day, tapered after month 24) (85 patients) and followed for a minimum of 36 months. The primary outcome was time to disease relapse (either major or minor relapse). RESULTS: Rituximab was superior to azathioprine in preventing relapse: HR 0.41; 95% CI 0.27 to 0.61, p

Published
2023
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25. Developing Methods to Link Patient Records across Data Sets that Preserve Patient Privacy

Author

Haynes, Kevin, primary, Agiro, Abiy, additional, Chen, Xiaoxue, additional, Stephenson, Judith, additional, Eshete, Biruk, additional, Sutphen, Rebecca, additional, Bourquardez Clark, Elizabeth, additional, Burroughs, Cristina, additional, Nowell, W. Benjamin, additional, Curtis, Jeffrey, additional, Loud, Sara, additional, McBurney, Robert, additional, Merkel, Peter, additional, Sreih, Antoine, additional, and Young, Kalen, additional

Published
2020
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26. Ocular manifestations of ANCA-associated vasculitis

Author

Junek, Mats L, primary, Zhao, Lily, additional, Garner, Stephanie, additional, Cuthbertson, David, additional, Pagnoux, Christian, additional, Koening, Curry L, additional, Langford, Carol A, additional, McAlear, Carol A, additional, Monach, Paul A, additional, Moreland, Larry W, additional, Rhee, Rennie L, additional, Seo, Philip, additional, Specks, Ulrich, additional, Sreih, Antoine G, additional, Warrington, Kenneth, additional, Wechsler, Michael E, additional, Merkel, Peter A, additional, and Khalidi, Nader A, additional

Published
2022
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27. Ocular manifestations of ANCA-associated vasculitis.

Author

Junek, Mats L, Zhao, Lily, Garner, Stephanie, Cuthbertson, David, Pagnoux, Christian, Koening, Curry L, Langford, Carol A, McAlear, Carol A, Monach, Paul A, Moreland, Larry W, Rhee, Rennie L, Seo, Philip, Specks, Ulrich, Sreih, Antoine G, Warrington, Kenneth, Wechsler, Michael E, Merkel, Peter A, and Khalidi, Nader A

Subjects
STATISTICS, CATARACT, OCULAR manifestations of general diseases, MULTIPLE regression analysis, ANTINEUTROPHIL cytoplasmic antibodies, RETROSPECTIVE studies, GRANULOMATOSIS with polyangiitis, RESEARCH funding, HEARING disorders, VASCULITIS, LONGITUDINAL method, CONJUNCTIVITIS
Abstract

Objectives ANCA-associated vasculitis (AAV) is a group of multisystem diseases that can have several ocular manifestations. There are published data on ocular manifestations of granulomatosis with polyangiitis (GPA), but few for eosinophilic granulomatosis with polyangiitis (EGPA) or microscopic polyangiitis (MPA). There is little information concerning chronicity, complications, and association with other cranial manifestations of AAV. Methods This study retrospectively analysed longitudinal multicentre cohorts of individuals with AAV followed between 2006 and 2022. Data included diagnosis, demographics, cranial manifestations of disease, presence of manifestations at onset of disease and/or follow-up, and ocular complications of disease. Univariate and multivariable logistic regression analysis assessed associations across disease manifestations. Results Data from 1441 patients were analysed, including 395 with EGPA, 876 with GPA, and 170 with MPA. Ocular manifestations were seen within 23.1% of patients: 39 (9.9%) with EGPA, 287 (32.7%) with GPA, and 12 (7.1%) with MPA at any time in the disease course. There were more ocular manifestations at onset (n  = 224) than during follow-up (n  = 120). The most common disease-related manifestations were conjunctivitis/episcleritis and scleritis. In multivariable analysis, dacryocystitis, lacrimal duct obstruction, and retro-orbital disease were associated with sinonasal manifestations of GPA; ocular manifestations were associated with hearing loss in MPA. The most common ocular complications and/or damage seen were cataracts (n  = 168) and visual impairment (n  = 195). Conclusion Ocular manifestations occur in all forms of AAV, especially in GPA. Clinicians should be mindful of the wide spectrum of ocular disease in AAV, caused by active vasculitis, disease-associated damage, and toxicities of therapy. [ABSTRACT FROM AUTHOR]

Published
2023
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30. Self-Reported Data and Physician-Reported Data in Patients With Eosinophilic Granulomatosis With Polyangiitis: Comparative Analysis

Author

Doubelt, Irena, primary, Springer, Jason M, additional, Kermani, Tanaz A, additional, Sreih, Antoine G, additional, Burroughs, Cristina, additional, Cuthbertson, David, additional, Carette, Simon, additional, Khalidi, Nader A, additional, Koening, Curry L, additional, Langford, Carol, additional, McAlear, Carol A, additional, Moreland, Larry W, additional, Monach, Paul A, additional, Shaw, Dianne G, additional, Seo, Philip, additional, Specks, Ulrich, additional, Warrington, Kenneth J, additional, Young, Kalen, additional, Merkel, Peter A, additional, and Pagnoux, Christian, additional

Published
2022
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31. 965: EFFICACY AND SAFETY OF DEUCRAVACITINIB, AN ORAL, SELECTIVE TYROSINE KINASE 2 INHIBITOR, IN PATIENTS WITH MODERATELY TO SEVERELY ACTIVE ULCERATIVE COLITIS: 12-WEEK RESULTS FROM THE PHASE 2 LATTICE-UC STUDY

Author

Danese, Silvio, primary, Panaccione, Remo, additional, D'Haens, Geert R., additional, Peyrin-Biroulet, Laurent, additional, Schreiber, Stefan, additional, Kobayashi, Taku, additional, Gawdis-Wojnarska, Beata, additional, Korga, Patryk, additional, Aguilar, Humberto, additional, Sharkey, Brian, additional, Sreih, Antoine, additional, Radosti, Courtney, additional, Patel, Aditya, additional, Canavan, James B., additional, and Rubin, David T., additional

Published
2022
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32. Identification of Susceptibility Loci in IL6, RPS9/LILRB3, and an Intergenic Locus on Chromosome 21q22 in Takayasu Arteritis in a Genome-Wide Association Study

Author

Renauer, Paul A., Saruhan-Direskeneli, Guher, Coit, Patrick, Adler, Adam, Aksu, Kenan, Keser, Gokhan, Alibaz-Oner, Fatma, Aydin, Sibel Z., Kamali, Sevil, Inanc, Murat, Carette, Simon, Cuthbertson, David, Hoffman, Gary S., Akar, Servet, Onen, Fatos, Akkoc, Nurullah, Khalidi, Nader A., Koening, Curry, Karadag, Omer, Kiraz, Sedat, Langford, Carol A., Maksimowicz-McKinnon, Kathleen, McAlear, Carol A., Ozbalkan, Zeynep, Ates, Askin, Karaaslan, Yasar, Duzgun, Nursen, Monach, Paul A., Ozer, Huseyin T. E., Erken, Eren, Ozturk, Mehmet A., Yazici, Ayten, Cefle, Ayse, Onat, Ahmet Mesut, Kisacik, Bunyamin, Pagnoux, Christian, Kasifoglu, Timucin, Seyahi, Emire, Fresko, Izzet, Seo, Philip, Sreih, Antoine G., Warrington, Kenneth J., Ytterberg, Steven R., Cobankara, Veli, Cunninghame-Graham, Deborah S., Vyse, Timothy J., Pamuk, Omer N., Tunc, Ercan S., Dalkilic, Ediz, Bicakcigil, Muge, Yentur, Sibel P., Wren, Jonathan D., Merkel, Peter A., Direskeneli, Haner, and Sawalha, Amr H.

Published
2015
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33. Dynamic Changes in the Nasal Microbiome Associated With Disease Activity in Patients With Granulomatosis With Polyangiitis

Author

Rhee, Rennie L., primary, Lu, Jiarui, additional, Bittinger, Kyle, additional, Lee, Jung‐Jin, additional, Mattei, Lisa M., additional, Sreih, Antoine G., additional, Chou, Sherry, additional, Miner, Jonathan J., additional, Cohen, Noam A., additional, Kelly, Brendan J., additional, Lee, Hongzhe, additional, Grayson, Peter C., additional, Collman, Ronald G., additional, and Merkel, Peter A., additional

Published
2021
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34. Identification of susceptibility loci for Takayasu arteritis through a large multi-ancestral genome-wide association study

Author

Ortiz-Fernandez, Lourdes, Saruhan-Direskeneli, Guher, Alibaz-Oner, Fatma, Kaymaz-Tahra, Sema, Coit, Patrick, Kong, Xiufang, Kiprianos, Allan P., Maughan, Robert T., Aydin, Sibel Z., Aksu, Kenan, Keser, Gokhan, Kamali, Sevil, Inanc, Murat, Springer, Jason, Akar, Servet, Onen, Fatos, Akkoc, Nurullah, Khalidi, Nader A., Koening, Curry, Karadag, Omer, Kiraz, Sedat, Forbess, Lindsy, Langford, Carol A., McAlear, Carol A., Ozbalkan, Zeynep, Yavuz, Sule, Cetin, Gozde Yildirim, Alpay-Kanitez, Nilufer, Chung, Sharon, Ates, Askin, Karaaslan, Yasar, McKinnon-Maksimowicz, Kathleen, Monach, Paul A., Ozer, Huseyin T. E., Seyahi, Emire, Fresko, Izzet, Cefle, Ayse, Seo, Philip, Warrington, Kenneth J., Ozturk, Mehmet A., Ytterberg, Steven R., Cobankara, Veli, Onat, Ahmet Mesut, Duzgun, Nursen, Bicakcigil, Muge, Yentur, Sibel P., Lally, Lindsay, Manfredi, Angelo A., Baldissera, Elena, Erken, Eren, Yazici, Ayten, Kisacik, Bunyamin, Kasifoglu, Timucin, Dalkilic, Ediz, Cuthbertson, David, Pagnoux, Christian, Sreih, Antoine, Reales, Guillermo, Wallace, Chris, Wren, Jonathan D., Cunninghame-Graham, Deborah S., Vyse, Timothy J., Sun, Ying, Chen, Huiyong, Grayson, Peter C., Tombetti, Enrico, Jiang, Lindi, Mason, Justin C., Merkel, Peter A., Direskeneli, Haner, Sawalha, Amr H., Ortiz-Fernandez, Lourdes, Saruhan-Direskeneli, Guher, Alibaz-Oner, Fatma, Kaymaz-Tahra, Sema, Coit, Patrick, Kong, Xiufang, Kiprianos, Allan P., Maughan, Robert T., Aydin, Sibel Z., Aksu, Kenan, Keser, Gokhan, Kamali, Sevil, Inanc, Murat, Springer, Jason, Akar, Servet, Onen, Fatos, Akkoc, Nurullah, Khalidi, Nader A., Koening, Curry, Karadag, Omer, Kiraz, Sedat, Forbess, Lindsy, Langford, Carol A., McAlear, Carol A., Ozbalkan, Zeynep, Yavuz, Sule, Cetin, Gozde Yildirim, Alpay-Kanitez, Nilufer, Chung, Sharon, Ates, Askin, Karaaslan, Yasar, McKinnon-Maksimowicz, Kathleen, Monach, Paul A., Ozer, Huseyin T. E., Seyahi, Emire, Fresko, Izzet, Cefle, Ayse, Seo, Philip, Warrington, Kenneth J., Ozturk, Mehmet A., Ytterberg, Steven R., Cobankara, Veli, Onat, Ahmet Mesut, Duzgun, Nursen, Bicakcigil, Muge, Yentur, Sibel P., Lally, Lindsay, Manfredi, Angelo A., Baldissera, Elena, Erken, Eren, Yazici, Ayten, Kisacik, Bunyamin, Kasifoglu, Timucin, Dalkilic, Ediz, Cuthbertson, David, Pagnoux, Christian, Sreih, Antoine, Reales, Guillermo, Wallace, Chris, Wren, Jonathan D., Cunninghame-Graham, Deborah S., Vyse, Timothy J., Sun, Ying, Chen, Huiyong, Grayson, Peter C., Tombetti, Enrico, Jiang, Lindi, Mason, Justin C., Merkel, Peter A., Direskeneli, Haner, and Sawalha, Amr H.

Abstract

Takayasu arteritis is a rare inflammatory disease of large arteries. We performed a genetic study in Takayasu arteritis comprising 6,670 individuals (1,226 affected individuals) from five different populations. We discovered HLA risk factors and four non-HLA susceptibility loci in VPS8, SVEP1, CFL2, and chr13q21 and reinforced IL12B, PTK2B, and chr21q22 as robust susceptibility loci shared across ancestries. Functional analysis proposed plausible underlying disease mechanisms and pinpointed ETS2 as a potential causal gene for chr21q22 association. We also identified >60 candidate loci with suggestive association (p < 5 x 10(-s)) and devised a genetic risk score for Takayasu arteritis. Takayasu arteritis was compared to hundreds of other traits, revealing the closest genetic relatedness to inflammatory bowel disease. Epigenetic patterns within risk loci suggest roles for monocytes and B cells in Takayasu arteritis. This work enhances understanding of the genetic basis and pathophysiology of Takayasu arteritis and provides clues for potential new therapeutic targets.

Published
2021
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37. Efficacy of leflunomide in the treatment of vasculitis

Author

Mustapha, Noura, primary, Barra, Lillian, additional, Carette, Simon, additional, Cuthbertson, David, additional, Khalidi, Nader A., additional, Koening, Curry L., additional, Langford, Carol A., additional, McAlear, Carol A., additional, Milman, Nataliya, additional, Moreland, Larry W., additional, Monach, Paul A., additional, Seo, Philip, additional, Specks, Ulrich, additional, Sreih, Antoine G., additional, Ytterberg, Steve Y., additional, Merkel, Peter A., additional, and Pagnoux, Christian, additional

Published
2021
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38. P027 Factors Associated with Biologic Discontinuation in Patients with Inflammatory Bowel Disease in TARGET-IBD

Author

Nguyen Joehl, Zhuo Joe, Long Millie, Barnes Edward, Sreih Antoine, Hildebrand Janet, Hanson John, Gazis Derek, Canavan James, Ahmad Harris, and Crawford Julie

Subjects
medicine.medical_specialty, Text mining, Hepatology, business.industry, Internal medicine, Gastroenterology, medicine, In patient, medicine.disease, business, Inflammatory bowel disease, Discontinuation
Published
2020

39. Rituximab as therapy to induce remission after relapse in ANCA-associated vasculitis

Author

Smith, Rona M, Jones, Rachel Bronwen, Specks, Ulrich, Bond, Simon, Nodale, Marianna, Aljayyousi, Reem, Andrews, Jacqueline, Bruchfeld, Annette, Camilleri, Brian, Carette, Simon, Cheung, Chee Kay, Derebail, Vimal, Doulton, Tim, Forbess, Lindsy, Fujimoto, Shouichi, Furuta, Shunsuke, Gewurz-Singer, Ora, Harper, Lorraine, Ito-Ihara, Toshiko, Khalidi, Nader, Klocke, Rainer, Koening, Curry, Komagata, Yoshinori, Langford, Carol, Lanyon, Peter, Luqmani, Raashid Ahmed, Makino, Hirofumi, McAlear, Carole, Monach, Paul, Moreland, Larry W, Mynard, Kim, Nachman, Patrick, Pagnoux, Christian, Pearce, Fiona, Peh, Chen Au, Pusey, Charles, Ranganathan, Dwarakanathan, Rhee, Rennie L, Spiera, Robert, Sreih, Antoine G, Tesar, Vladimir, Walters, Giles, Weisman, Michael H, Wroe, Caroline, Merkel, Peter, Jayne, David, RITAZAREM Co-Investigators, Arimura, Y, Clarkson, M, De Zoysa, J, Endo, T, Hamano, Y, Kono, H, Lawman, S, Muso, E, Sada, K, Smith, R, Suzuki, K, Tsukamoto, T, Uchida, S, Vaglio, A, Watts, R, Smith, Rona M [0000-0002-7438-5156], Jones, Rachel Bronwen [0000-0003-4790-283X], Nodale, Marianna [0000-0002-0333-8918], Harper, Lorraine [0000-0003-1343-9234], Rhee, Rennie L [0000-0002-4907-0304], Walters, Giles [0000-0003-4854-9353], and Apollo - University of Cambridge Repository

Subjects
Vasculitis, B cells, granulomatosis with polyangiitis, treatment, systemic vasculitis
Abstract

Funder: Research Committee on Intractable Vasculitides; The Ministry of Health, Labour and Welfare of Japan., Objectives: Evaluation of rituximab and glucocorticoids as therapy to induce remission after relapse in ANCA-associated vasculitis (AAV) in a prospective observational cohort of patients enrolled into the induction phase of the RITAZAREM trial. Methods: Patients relapsing with granulomatosis with polyangiitis or microscopic polyangiitis were prospectively enrolled and received remission-induction therapy with rituximab (4×375 mg/m2) and a higher or lower dose glucocorticoid regimen, depending on physician choice: reducing from either 1 mg/kg/day or 0.5 mg/kg/day to 10 mg/day by 4 months. Patients in this cohort achieving remission were subsequently randomised to receive one of two regimens to prevent relapse. Results: 188 patients were studied: 95/188 (51%) men, median age 59 years (range 19–89), prior disease duration 5.0 years (range 0.4–34.5). 149/188 (79%) had previously received cyclophosphamide and 67/188 (36%) rituximab. 119/188 (63%) of relapses had at least one major disease activity item, and 54/188 (29%) received the higher dose glucocorticoid regimen. 171/188 (90%) patients achieved remission by 4 months. Only six patients (3.2% of the study population) did not achieve disease control at month 4. Four patients died in the induction phase due to pneumonia (2), cerebrovascular accident (1), and active vasculitis (1). 41 severe adverse events occurred in 27 patients, including 13 severe infections. Conclusions: This large prospective cohort of patients with relapsing AAV treated with rituximab in conjunction with glucocorticoids demonstrated a high level of efficacy for the reinduction of remission in patients with AAV who have relapsed, with a similar safety profile to previous studies.

Published
2020
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41. Self-Reported Data and Physician-Reported Data in Patients With Eosinophilic Granulomatosis With Polyangiitis: Comparative Analysis (Preprint)

Author

Doubelt, Irena, primary, Springer, Jason M, additional, Kermani, Tanaz A, additional, Sreih, Antoine G, additional, Burroughs, Cristina, additional, Cuthbertson, David, additional, Carette, Simon, additional, Khalidi, Nader A, additional, Koening, Curry L, additional, Langford, Carol, additional, McAlear, Carol A, additional, Moreland, Larry W, additional, Monach, Paul A, additional, Shaw, Dianne G, additional, Seo, Philip, additional, Specks, Ulrich, additional, Warrington, Kenneth J, additional, Young, Kalen, additional, Merkel, Peter A, additional, and Pagnoux, Christian, additional

Published
2021
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42. Identification of susceptibility loci for Takayasu arteritis through a large multi-ancestral genome-wide association study

Author

Ortiz-Fernández, Lourdes, primary, Saruhan-Direskeneli, Güher, additional, Alibaz-Oner, Fatma, additional, Kaymaz-Tahra, Sema, additional, Coit, Patrick, additional, Kong, Xiufang, additional, Kiprianos, Allan P., additional, Maughan, Robert T., additional, Aydin, Sibel Z., additional, Aksu, Kenan, additional, Keser, Gokhan, additional, Kamali, Sevil, additional, Inanc, Murat, additional, Springer, Jason, additional, Akar, Servet, additional, Onen, Fatos, additional, Akkoc, Nurullah, additional, Khalidi, Nader A., additional, Koening, Curry, additional, Karadag, Omer, additional, Kiraz, Sedat, additional, Forbess, Lindsy, additional, Langford, Carol A., additional, McAlear, Carol A., additional, Ozbalkan, Zeynep, additional, Yavuz, Sule, additional, Çetin, Gozde Yildirim, additional, Alpay-Kanitez, Nilufer, additional, Chung, Sharon, additional, Ates, Askin, additional, Karaaslan, Yasar, additional, McKinnon-Maksimowicz, Kathleen, additional, Monach, Paul A., additional, Ozer, Hüseyin T.E., additional, Seyahi, Emire, additional, Fresko, Izzet, additional, Cefle, Ayse, additional, Seo, Philip, additional, Warrington, Kenneth J., additional, Ozturk, Mehmet A., additional, Ytterberg, Steven R., additional, Cobankara, Veli, additional, Onat, Ahmet Mesut, additional, Duzgun, Nurşen, additional, Bıcakcıgil, Muge, additional, Yentür, Sibel P., additional, Lally, Lindsay, additional, Manfredi, Angelo A., additional, Baldissera, Elena, additional, Erken, Eren, additional, Yazici, Ayten, additional, Kısacık, Bünyamin, additional, Kaşifoğlu, Timuçin, additional, Dalkilic, Ediz, additional, Cuthbertson, David, additional, Pagnoux, Christian, additional, Sreih, Antoine, additional, Reales, Guillermo, additional, Wallace, Chris, additional, Wren, Jonathan D., additional, Cunninghame-Graham, Deborah S., additional, Vyse, Timothy J., additional, Sun, Ying, additional, Chen, Huiyong, additional, Grayson, Peter C., additional, Tombetti, Enrico, additional, Jiang, Lindi, additional, Mason, Justin C., additional, Merkel, Peter A., additional, Direskeneli, Haner, additional, and Sawalha, Amr H., additional

Published
2021
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43. Dual Effect of the Macrophage Migration Inhibitory Factor Gene on the Development and Severity of Human Systemic Lupus Erythematosus

Author

Sreih, Antoine, Ezzeddine, Rana, Leng, Lin, LaChance, Avery, Yu, Geraldine, Mizue, Yuka, Subrahmanyan, Lakshman, Pons-Estel, Bernardo A., Abelson, Anna-Karin, Gunnarsson, Iva, Svenungsson, Elisabet, Cavett, Joshua, Glenn, Stuart, Zhang, Lin, Montgomery, Ruth, Perl, Andras, Salmon, Jane, Alarcón-Riquelme, Marta E., Harley, John B., and Bucala, Richard

Published
2011
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44. Patterns of Arterial Disease in Takayasu Arteritis and Giant Cell Arteritis

Author

Gribbons, K. Bates, primary, Ponte, Cristina, additional, Carette, Simon, additional, Craven, Anthea, additional, Cuthbertson, David, additional, Hoffman, Gary S., additional, Khalidi, Nader A., additional, Koening, Curry L., additional, Langford, Carol A., additional, Maksimowicz-McKinnon, Kathleen, additional, McAlear, Carol A., additional, Monach, Paul A., additional, Moreland, Larry W., additional, Pagnoux, Christian, additional, Quinn, Kaitlin A., additional, Robson, Joanna C., additional, Seo, Philip, additional, Sreih, Antoine G., additional, Suppiah, Ravi, additional, Warrington, Kenneth J., additional, Ytterberg, Steven R., additional, Luqmani, Raashid, additional, Watts, Richard, additional, Merkel, Peter A., additional, and Grayson, Peter C., additional

Published
2020
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45. S0878 Real World Treatment Patterns and Treatment Persistence Among Ulcerative Colitis and Crohn’s Disease Patients in the Corrona Inflammatory Bowel Disease National Registry

Author

Sauk, Jenny S., primary, Horst, Sara, additional, Hudesman, David P, additional, Zhuo, Joe, additional, Ahmad, Harris, additional, Sreih, Antoine, additional, Harrison, Ryan W., additional, Mackey, Rachel H., additional, Crabtree, Margaux, additional, Emeanuru, Kelechi, additional, and Cross, Raymond K., additional

Published
2020
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46. Outcome Measures in Large Vessel Vasculitis: Relationship Between Patient‐, Physician‐, Imaging‐, and Laboratory‐Based Assessments

Author

Rimland, Casey A., primary, Quinn, Kaitlin A., additional, Rosenblum, Joel S., additional, Schwartz, Mollie N., additional, Bates Gribbons, K., additional, Novakovich, Elaine, additional, Sreih, Antoine G., additional, Merkel, Peter A., additional, Ahlman, Mark A., additional, and Grayson, Peter C., additional

Published
2020
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47. Patterns of clinical presentation in Takayasu's arteritis

Author

Quinn, Kaitlin A., primary, Gribbons, K. Bates, additional, Carette, Simon, additional, Cuthbertson, David, additional, Khalidi, Nader A., additional, Koening, Curry L., additional, Langford, Carol A., additional, McAlear, Carol A., additional, Monach, Paul A., additional, Moreland, Larry W., additional, Pagnoux, Christian, additional, Seo, Philip, additional, Sreih, Antoine G., additional, Warrington, Kenneth J., additional, Ytterberg, Steven R., additional, Novakovich, Elaine, additional, Merkel, Peter A., additional, and Grayson, Peter C., additional

Published
2020
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48. Rituximab as therapy to induce remission after relapse in ANCA-associated vasculitis

Author

Smith, Rona M, primary, Jones, Rachel Bronwen, additional, Specks, Ulrich, additional, Bond, Simon, additional, Nodale, Marianna, additional, Aljayyousi, Reem, additional, Andrews, Jacqueline, additional, Bruchfeld, Annette, additional, Camilleri, Brian, additional, Carette, Simon, additional, Cheung, Chee Kay, additional, Derebail, Vimal, additional, Doulton, Tim, additional, Forbess, Lindsy, additional, Fujimoto, Shouichi, additional, Furuta, Shunsuke, additional, Gewurz-Singer, Ora, additional, Harper, Lorraine, additional, Ito-Ihara, Toshiko, additional, Khalidi, Nader, additional, Klocke, Rainer, additional, Koening, Curry, additional, Komagata, Yoshinori, additional, Langford, Carol, additional, Lanyon, Peter, additional, Luqmani, Raashid Ahmed, additional, Makino, Hirofumi, additional, McAlear, Carole, additional, Monach, Paul, additional, Moreland, Larry W, additional, Mynard, Kim, additional, Nachman, Patrick, additional, Pagnoux, Christian, additional, Pearce, Fiona, additional, Peh, Chen Au, additional, Pusey, Charles, additional, Ranganathan, Dwarakanathan, additional, Rhee, Rennie L, additional, Spiera, Robert, additional, Sreih, Antoine G, additional, Tesar, Vladimir, additional, Walters, Giles, additional, Weisman, Michael H, additional, Wroe, Caroline, additional, Merkel, Peter, additional, and Jayne, David, additional

Published
2020
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50. P038 Factors Associated with Impaired Patient-Reported Outcomes and Work Productivity Among Patients with Ulcerative Colitis in Remission

Author

Ahmad Harris, Zhuo Joe, Emeanuru Kelechi, Sreih Antoine, Harrison Ryan, Nguyen Joehl, Sauk Jenny, Cross Raymond, Kerti Samantha, Horst Sara, and Hudesman David

Subjects
medicine.medical_specialty, Work productivity, Text mining, Hepatology, business.industry, Internal medicine, Gastroenterology, medicine, MEDLINE, business, medicine.disease, Ulcerative colitis
Published
2020

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