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1. The Association Between Age at Diagnosis and Disease Characteristics and Damage in Patients With ANCA-Associated Vasculitis.

Author

Corbridge, Thomas, Khalidi, Nader, Koening, Curry, Langford, Carol, McAlear, Carol, Monach, Paul, Moreland, Larry, Pagnoux, Christian, Rhee, Rennie, Seo, Philip, Silver, Jared, Specks, Ulrich, Warrington, Kenneth, Wechsler, Michael, Merkel, Peter, Bloom, Jessica, Pickett-Nairn, Kaci, Silveira, Lori, Fuhlbrigge, Robert, Cuthbertson, David, and Akuthota, Praveen

Subjects
Child, Middle Aged, Young Adult, Humans, Female, Aged, Male, Antibodies, Antineutrophil Cytoplasmic, Prospective Studies, Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis, Granulomatosis with Polyangiitis, Microscopic Polyangiitis, Hemorrhage, Churg-Strauss Syndrome
Abstract

OBJECTIVE: This study examined the relationship between age at diagnosis and disease characteristics and damage in patients with antineutrophil cytoplasmic antibody (ANCA)-associated vasculitis (AAV). METHODS: Analysis of a prospective longitudinal cohort of patients with granulomatosis with polyangiitis (GPA), microscopic polyangiitis (MPA), and eosinophilic GPA (EGPA) in the Vasculitis Clinical Research Consortium (2013-2021). Disease cohorts were divided by age at diagnosis (years): children (65). Data included demographics, ANCA type, clinical characteristics, Vasculitis Damage Index (VDI) scores, ANCA Vasculitis Index of Damage (AVID) scores, and novel disease-specific and non-disease-specific damage scores built from VDI and AVID items. RESULTS: Analysis included data from 1020 patients with GPA/MPA and 357 with EGPA. Female predominance in GPA/MPA decreased with age at diagnosis. AAV in childhood was more often GPA and proteinase 3-ANCA positive. Children with GPA/MPA experienced more subglottic stenosis and alveolar hemorrhage; children and young adults with EGPA experienced more alveolar hemorrhage, need for intubation, and gastrointestinal involvement. Older adults (GPA/MPA) had more neurologic manifestations. After adjusting for disease duration, medications, tobacco, and ANCA, all damage scores increased with age at diagnosis for GPA/MPA (P

Published
2023

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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7. The impact of treatment with avacopan on health-related quality of life in antineutrophil cytoplasmic antibody-associated vasculitis: a post-hoc analysis of data from the ADVOCATE trial

Author

Au Peh, Chen, Chakera, Aron, Cooper, Bruce, Kurtkoti, Jagadeesh, Langguth, Daman, Levidiotis, Vicki, Luxton, Grant, Mount, Peter, Mudge, David, Noble, Euan, Phoon, Richard, Ranganathan, Dwarakanathan, Ritchie, Angus, Ryan, Jessica, Suranyi, Michael, Rosenkranz, Alexander, Lhotta, Karl, Kronbichler, Andreas, Demoulin, Nathalie, Bovy, Christophe, Hellemans, Rachel, Hougardy, Jean-Michel, Sprangers, Ben, Wissing, Karl Martin, Pagnoux, Christian, Barbour, Sean, Brachemi, Soumeya, Cournoyer, Serge, Girard, Louis-Philippe, Laurin, Louis-Philippe, Liang, Patrick, Philibert, David, Walsh, Michael, Tesar, Vladimir, Becvar, Radim, Horak, Pavel, Rychlik, Ivan, Szpirt, Wladimir, Dieperink, Hans, Gregersen, Jon Waarst, Ivarsen, Per, Krarup, Elizabeth, Lyngsoe, Cecilie, Rigothier, Claire, Augusto, Jean-Francois, Belot, Alexandre, Chauveau, Dominique, Cornec, Divi, Jourde-Chiche, Noemie, Ficheux, Maxence, Karras, Alexandre, Klein, Alexandre, Maurier, Francois, Mesbah, Rafik, Moranne, Olivier, Neel, Antoine, Quemeneur, Thomas, Saadoun, David, Terrier, Benjamin, Zaoui, Philippe, Schaier, Matthias, Benck, Urs Tobias, Bergner, Raoul, Busch, Martin, Floege, Juergen, Grundmann, Franziska, Haller, Hermann, Haubitz, Marion, Hellmich, Bernhard, Henes, Joerg Christoph, Hohenstein, Bernd, Hugo, Christian, Iking-Konert, Christof, Arndt, Fabian, Kubacki, T, Kotter, Ina, Lamprecht, Peter, Lindner, Tom, Halbritter, Jan, Mehling, Heidrun, Schönermarck, Ulf, Venhoff, Nils, Vielhauer, Volker, Witzke, Oliver, Szombati, Istvan, Szucs, Gabriella, Garibotto, Giacomo, Alberici, Federico, Brunetta, Enrico, Dagna, Lorenzo, De Vita, Salvatore, Emmi, Giacomo, Gabrielli, Armando, Manenti, Lucio, Pieruzzi, Federico, Roccatello, Dario, Salvarani, Carlo, Harigai, Masayoshi, Dobashi, Hiroaki, Atsumi, Tatsuya, Fujimoto, Shoichi, Hagino, Noboru, Ihata, Atsushi, Kaname, Shinya, Kaneko, Yuko, Katagiri, Akira, Katayama, Masao, Kirino, Yohei, Kitagawa, Kiyoki, Komatsuda, Atsushi, Kono, Hajime, Kurasawa, Takahiko, Matsumura, Ryutaro, Mimura, Toshihide, Morinobu, Akio, Murakawa, Yohko, Naniwa, Taio, Nanki, Toshihiro, Ogawa, Noriyoshi, Oshima, Hisaji, Sada, Kenei, Sugiyama, Eiji, Takeuchi, Tohru, Taki, Hirofumi, Tamura, Naoto, Tsukamoto, Tatsuo, Yamagata, Kunihiro, Yamamura, Masahiro, van Daele, Paulus Leon Arthur, Rutgers, Abraham, Teng, Y.K. Onno, Walker, Robert, Chua, Ignatius, Collins, Michael, Rabindranath, Kannaiyan, de Zoysa, Janak, Svensson, My Hanna Sofia, Grevbo, Bard-Waldum, Kalstad, Synove, Little, Mark, Clarkson, Michael, Molloy, Eamonn, Agraz Pamplona, Irene, Anton, Jordi, Barrio Lucia, Vicente, Ciggaran, Secundino, Cinta Cid, Maria, Diaz Encarnacion, Montserrat, Fulladosa Oliveras, Xavier, Jose Soler, Maria, Marco Rusinol, Helena, Praga, Manuel, Quintana Porras, Luis, Segarra, Alfons, Bruchfeld, Annette, Segelmark, Marten, Soveri, Inga, Thomaidi, Eleni, Westman, Kerstin, Neumann, Thomas, Burnier, Michel, Daikeler, Thomas, Dudler, Jean, Hauser, Thomas, Seeger, Harald, Vogt, Bruno, Burton, James, Al Jayyousi, Reem, Amin, Tania, Andrews, Jacqueline, Baines, Laura Anne, Brogan, Paul, Dasgupta, Bhaskar, Doulton, Timothy William Ronald, Flossmann, Oliver, Griffin, Sian V., Harper, Janice Marian, Harper, Lorraine, Kidder, Dana, Klocke, Rainer, Lanyon, Peter Charles, Luqmani, Raashid, McLaren, John Stuart, Makanjuola, David Osagie, McCann, Liza, Nandagudi, Anupama C., Selvan, Shilpa, O'Riordan, Edmond, Patel, Mumtaz, Patel, Rajan Kantilal, Pusey, Charles Dickson, Rajakariar, Ravindra, Robson, Joanna C., Robson, Michael, Salama, Alan David, Smyth, Lucy, Sznajd, Jan, Taylor, Joanne, Sreih, Antonie G., Belilos, Elise, Bomback, Andrew S., Carlin, Jeffrey, Chang Chen Lin, Yih, Derebail, Vimal K., Dragoi, Serban, Dua, Anisha, Forbess, Lindsy, Geetha, Duvuru, Gipson, Patrick, Gohh, Reginald, Greenwood, Gregory Todd, Hugenberg, Steven T., Jimenez, Richard A.H., Kaskas, Marwan Omar, Kermani, Tanaz, Kivitz, Alan J., Koening, Curry, Langford, Carol A., Marder, Galina, Mohamed, Amr Ahmed El-Huesseini, Monach, Paul, Neyra, Nilda Roxana, Niemer, Gregory W., Niles, John, Obi, Reginald, Owens, Charles, Parks, Deborah L., Podoll, Amber S., Rovin, Brad, Sam, R, Shergy, William Julius, Silva, Arnold Lawrence, Specks, Ulrich, Spiera, Robert, Springer, Jason M., Striebich, Christopher Charles, Swarup, Areena, Thakar, Surabhi, Tiliakos, Athan N., Tsai, Yong, Waguespack, Dia R., Chester Wasko, Mary, Strand, Vibeke, Jayne, David R W, Horomanski, Audra, Yue, Huibin, Bekker, Pirow, and Merkel, Peter A

Published
2023
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8. 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

9. 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. Signal Regulatory Protein α Expression in Systemic Vasculitis.

Author

Banerjee, Shubhasree, Rose, Eileen, Panicker, Sandip, Dugan, John, Khalidi, Nader, Koening, Curry L., Langford, Carol A., Monach, Paul A., Pagnoux, Christian, McAlear, Carol A., and Merkel, Peter A.

Subjects
VASCULITIS, CELL migration, BIOPSY, MACROPHAGES, MONOCYTES, CROHN'S disease, RESEARCH funding, NEUTROPHILS, CELL proliferation, MICROSCOPIC polyangiitis, KRUSKAL-Wallis Test, MYELOID cells, GIANT cell arteritis, CELLULAR signal transduction, GENE expression, GRANULOMATOSIS with polyangiitis, IMMUNOHISTOCHEMISTRY, MICROBIOLOGICAL assay, STAINS & staining (Microscopy), MEMBRANE proteins, PHAGOCYTOSIS, TEMPORAL arteries, KIDNEYS
Abstract

Objective: Signal regulatory protein α (SIRPα) is found primarily on myeloid cells, including macrophages and neutrophils; binds to CD47; and regulates phagocytosis, antigen presentation, cellular fusion, cell proliferation, and migration. Therefore, SIRPα may be involved in the pathogenesis of autoimmune diseases, including systemic vasculitis. This study aimed to assess SIRPα expression in tissue samples from patients with vasculitis. Methods: Immunohistochemical staining for SIRPα was performed on temporal artery (TA), kidney, and lung biopsy samples from patients with giant cell arteritis (GCA), patients with microscopic polyangiitis (MPA), patients with granulomatosis with polyangiitis (GPA), and patients without vasculitis. A score of SIRPα+ expression was calculated, derived from the percentages of monocytes, macrophages, and dendritic cells and neutrophils with different staining intensities in affected tissues. Results: A total of 46 samples from patients with different vasculitides (GCA, MPA, and GPA) were included in the study. Tissue samples included TA samples from 15 patients with GCA; kidney samples from 11 and 9 patients with GPA and MPA, respectively; and lung samples from 11 patients with GPA. Most tissue samples from patients with active vasculitis (15 of 15 TA samples, 17 of 20 kidney samples, and 9 of 11 lung samples) showed SIRPα staining. SIRPα staining intensity was less in kidney samples compared to TA and lung samples. Conclusion: This study demonstrates high‐level expression of SIRPα in macrophages and monocytes in affected tissue in systemic vasculitis. These findings provide a foundation for further studies exploring the role of the SIRPα–CD47 pathway in the pathogenesis of systemic vasculitis and the potential for the blockade of SIRPα and/or the depletion of SIRPα+ cells as treatment of systemic vasculitis. [ABSTRACT FROM AUTHOR]

Published
2024
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11. 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

12. 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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13. Clinical Characteristics and Outcomes of Polyarteritis Nodosa: An International Study.

Author

Karadag, Omer, Bolek, Ertugrul Cagri, Ayan, Gizem, Mohammad, Aladdin J., Grayson, Peter C., Pagnoux, Christian, Martín‐Nares, Eduardo, Monti, Sara, Abe, Yoshiyuki, Alberici, Federico, Alibaz‐Oner, Fatma, Cuthbertson, David, Dagna, Lorenzo, Direskeneli, Haner, Khalidi, Nader A., Koening, Curry, Langford, Carol A., McAlear, Carol A., Monach, Paul A., and Moroni, Luca

Subjects
PROTEINURIA, SURVIVAL rate, INTERPROFESSIONAL relations, CREATININE, RESEARCH funding, SCIENTIFIC observation, SYMPTOMS, DESCRIPTIVE statistics, AGE distribution, CENTRAL nervous system, RETROSPECTIVE studies, WORLD health, LONGITUDINAL method, MEDICAL records, ACQUISITION of data, SOCIODEMOGRAPHIC factors, POLYARTERITIS nodosa, GASTROINTESTINAL diseases
Abstract

Objective: We describe the demographics, clinical features, disease course, and survival of polyarteritis nodosa (PAN) through an international collaboration (GLOBAL‐PAN). Methods: Patients with PAN were recruited between 1990 and 2020 from observational cohorts of nine countries across Europe, Japan, and North America. Eligibility was retrospectively defined using the European Medicines Agency classification algorithm. Patients with PAN related to hepatitis B virus (n = 12) and two monogenic diseases mimicking PAN, deficiency of adenosine deaminase 2 enzyme (n = 16) or familial Mediterranean fever (n = 11), were excluded. Data regarding organ involvement, relapse, disease‐related damage, and survival were analyzed. Results: Three hundred fifty‐eight patients (female:male ratio 174:184), including those with systemic PAN (sPAN, n = 282) and cutaneous PAN (n = 76), were included. Twenty‐five were pediatric onset. Mean ± SD age at diagnosis was 44.3 ± 18.1 years. Constitutional symptoms (71.5%), cutaneous involvement (70.5%), musculoskeletal findings (69.1%), and neurologic features (48.0%) were common manifestations. Among patients with sPAN, gastrointestinal involvement and proteinuria over 400 mg/day were reported in 52.2% and 11.2%, respectively. During a median (interquartile range) 59.6 (99.5) months of follow‐up, relapse occurred in 48.5% of patients. One, 5‐ and 10‐year survival rates for sPAN were 97.1%, 94.0%, and 89.0%, respectively. Predictors of death for sPAN included age ≥65 years at diagnosis, serum creatinine at diagnosis >140 μmol/L, gastrointestinal manifestations, and central nervous system (CNS) involvement. Conclusion: The spectrum of PAN remains a complex, multifaceted disease. Relapse is common. Age ≥65 years and serum creatinine >140 μmol/L at diagnosis, as well as gastrointestinal and CNS involvement, are independent predictors of death in sPAN. [ABSTRACT FROM AUTHOR]

Published
2024
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14. Pulmonary Eosinophilic Granulomatosis with Polyangiitis Has IgG4 Plasma Cells and Immunoregulatory Features

Author

Dong, Zachary M., Lin, Edwin, Wechsler, Michael E., Weller, Peter F., Klion, Amy D., Bochner, Bruce S., Delker, Don A., Hazel, Mark W., Fairfax, Keke, Khoury, Paneez, Akuthota, Praveen, Merkel, Peter A., Dyer, Anne-Marie, Langford, Carol, Specks, Ulrich, Gleich, Gerald J., Chinchilli, Vernon M., Raby, Benjamin, Yandell, Mark, and Clayton, Frederic

Published
2020
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15. The Birmingham Vasculitis Activity Score as a Measure of Disease Activity in Patients with Giant Cell Arteritis.

Author

Kermani, Tanaz, Cuthbertson, David, Carette, Simon, Hoffman, Gary, Khalidi, Nader, Koening, Curry, Langford, Carol, McKinnon-Maksimowicz, Kathleen, McAlear, Carol, Monach, Paul, Seo, Philip, Warrington, Kenneth, Ytterberg, Steven, Merkel, Peter, and Matteson, Eric

Subjects
BIRMINGHAM VASCULITIS ACTIVITY SCORE, COHORT STUDY, DISEASE ACTIVITY, GIANT CELL ARTERITIS, Aged, Aged, 80 and over, Female, Giant Cell Arteritis, Humans, Longitudinal Studies, Male, Middle Aged, Prospective Studies, Severity of Illness Index, Symptom Assessment
Abstract

OBJECTIVE: To evaluate the performance of the Birmingham Vasculitis Activity Score (BVAS) in the assessment of disease activity in giant cell arteritis (GCA). METHODS: Patients with GCA enrolled in a prospective, multicenter, longitudinal study with symptoms of active vasculitis during any visit were included. Spearmans rank correlation was used to explore the association of the BVAS with other measures of disease activity. RESULTS: During a mean (SD) followup of 2.3 (1.6) years, symptoms of active GCA were present in 236 visits in 136 subjects (100 female, 74%). Median (range) BVAS1 (new/worse symptoms) was 1 (0-10) and median (range) BVAS2 (persistent symptoms) was 0 (0-5). Median (range) physicians global assessment (PGA) was 4 (0-9) for disease activity in the past 28 days and 2 (0-9) for activity on the day of the visit. Important ischemic manifestations of active vasculitis not recorded by the BVAS included tongue/jaw claudication (27%), upper extremity claudication (15%), lower extremity claudication (5%), carotidynia (7%), and ischemic retinopathy (5%). During 25 visits (11%) with active disease, all symptoms of active vasculitis were placed in the Other category yet still resulted in a BVAS1 and BVAS2 of 0. BVAS1 moderately correlated with PGA for the past 28 days (Spearmans correlation 0.50) and physician-rated disease activity for the past 28 days (Spearmans correlation 0.46). CONCLUSION: The BVAS has limited utility in GCA. Patients with active GCA can have a BVAS of 0. Many important ischemic symptoms attributable to active vasculitis are not included in the composite score.

Published
2016

17. The impact of treatment with avacopan on health-related quality of life in antineutrophil cytoplasmic antibody-associated vasculitis: a post-hoc analysis of data from the ADVOCATE trial

Author

Strand, Vibeke, primary, Jayne, David R W, additional, Horomanski, Audra, additional, Yue, Huibin, additional, Bekker, Pirow, additional, Merkel, Peter A, additional, Au Peh, Chen, additional, Chakera, Aron, additional, Cooper, Bruce, additional, Kurtkoti, Jagadeesh, additional, Langguth, Daman, additional, Levidiotis, Vicki, additional, Luxton, Grant, additional, Mount, Peter, additional, Mudge, David, additional, Noble, Euan, additional, Phoon, Richard, additional, Ranganathan, Dwarakanathan, additional, Ritchie, Angus, additional, Ryan, Jessica, additional, Suranyi, Michael, additional, Rosenkranz, Alexander, additional, Lhotta, Karl, additional, Kronbichler, Andreas, additional, Demoulin, Nathalie, additional, Bovy, Christophe, additional, Hellemans, Rachel, additional, Hougardy, Jean-Michel, additional, Sprangers, Ben, additional, Wissing, Karl Martin, additional, Pagnoux, Christian, additional, Barbour, Sean, additional, Brachemi, Soumeya, additional, Cournoyer, Serge, additional, Girard, Louis-Philippe, additional, Laurin, Louis-Philippe, additional, Liang, Patrick, additional, Philibert, David, additional, Walsh, Michael, additional, Tesar, Vladimir, additional, Becvar, Radim, additional, Horak, Pavel, additional, Rychlik, Ivan, additional, Szpirt, Wladimir, additional, Dieperink, Hans, additional, Gregersen, Jon Waarst, additional, Ivarsen, Per, additional, Krarup, Elizabeth, additional, Lyngsoe, Cecilie, additional, Rigothier, Claire, additional, Augusto, Jean-Francois, additional, Belot, Alexandre, additional, Chauveau, Dominique, additional, Cornec, Divi, additional, Jourde-Chiche, Noemie, additional, Ficheux, Maxence, additional, Karras, Alexandre, additional, Klein, Alexandre, additional, Maurier, Francois, additional, Mesbah, Rafik, additional, Moranne, Olivier, additional, Neel, Antoine, additional, Quemeneur, Thomas, additional, Saadoun, David, additional, Terrier, Benjamin, additional, Zaoui, Philippe, additional, Schaier, Matthias, additional, Benck, Urs Tobias, additional, Bergner, Raoul, additional, Busch, Martin, additional, Floege, Juergen, additional, Grundmann, Franziska, additional, Haller, Hermann, additional, Haubitz, Marion, additional, Hellmich, Bernhard, additional, Henes, Joerg Christoph, additional, Hohenstein, Bernd, additional, Hugo, Christian, additional, Iking-Konert, Christof, additional, Arndt, Fabian, additional, Kubacki, T, additional, Kotter, Ina, additional, Lamprecht, Peter, additional, Lindner, Tom, additional, Halbritter, Jan, additional, Mehling, Heidrun, additional, Schönermarck, Ulf, additional, Venhoff, Nils, additional, Vielhauer, Volker, additional, Witzke, Oliver, additional, Szombati, Istvan, additional, Szucs, Gabriella, additional, Garibotto, Giacomo, additional, Alberici, Federico, additional, Brunetta, Enrico, additional, Dagna, Lorenzo, additional, De Vita, Salvatore, additional, Emmi, Giacomo, additional, Gabrielli, Armando, additional, Manenti, Lucio, additional, Pieruzzi, Federico, additional, Roccatello, Dario, additional, Salvarani, Carlo, additional, Harigai, Masayoshi, additional, Dobashi, Hiroaki, additional, Atsumi, Tatsuya, additional, Fujimoto, Shoichi, additional, Hagino, Noboru, additional, Ihata, Atsushi, additional, Kaname, Shinya, additional, Kaneko, Yuko, additional, Katagiri, Akira, additional, Katayama, Masao, additional, Kirino, Yohei, additional, Kitagawa, Kiyoki, additional, Komatsuda, Atsushi, additional, Kono, Hajime, additional, Kurasawa, Takahiko, additional, Matsumura, Ryutaro, additional, Mimura, Toshihide, additional, Morinobu, Akio, additional, Murakawa, Yohko, additional, Naniwa, Taio, additional, Nanki, Toshihiro, additional, Ogawa, Noriyoshi, additional, Oshima, Hisaji, additional, Sada, Kenei, additional, Sugiyama, Eiji, additional, Takeuchi, Tohru, additional, Taki, Hirofumi, additional, Tamura, Naoto, additional, Tsukamoto, Tatsuo, additional, Yamagata, Kunihiro, additional, Yamamura, Masahiro, additional, van Daele, Paulus Leon Arthur, additional, Rutgers, Abraham, additional, Teng, Y.K. Onno, additional, Walker, Robert, additional, Chua, Ignatius, additional, Collins, Michael, additional, Rabindranath, Kannaiyan, additional, de Zoysa, Janak, additional, Svensson, My Hanna Sofia, additional, Grevbo, Bard-Waldum, additional, Kalstad, Synove, additional, Little, Mark, additional, Clarkson, Michael, additional, Molloy, Eamonn, additional, Agraz Pamplona, Irene, additional, Anton, Jordi, additional, Barrio Lucia, Vicente, additional, Ciggaran, Secundino, additional, Cinta Cid, Maria, additional, Diaz Encarnacion, Montserrat, additional, Fulladosa Oliveras, Xavier, additional, Jose Soler, Maria, additional, Marco Rusinol, Helena, additional, Praga, Manuel, additional, Quintana Porras, Luis, additional, Segarra, Alfons, additional, Bruchfeld, Annette, additional, Segelmark, Marten, additional, Soveri, Inga, additional, Thomaidi, Eleni, additional, Westman, Kerstin, additional, Neumann, Thomas, additional, Burnier, Michel, additional, Daikeler, Thomas, additional, Dudler, Jean, additional, Hauser, Thomas, additional, Seeger, Harald, additional, Vogt, Bruno, additional, Burton, James, additional, Al Jayyousi, Reem, additional, Amin, Tania, additional, Andrews, Jacqueline, additional, Baines, Laura Anne, additional, Brogan, Paul, additional, Dasgupta, Bhaskar, additional, Doulton, Timothy William Ronald, additional, Flossmann, Oliver, additional, Griffin, Sian V., additional, Harper, Janice Marian, additional, Harper, Lorraine, additional, Kidder, Dana, additional, Klocke, Rainer, additional, Lanyon, Peter Charles, additional, Luqmani, Raashid, additional, McLaren, John Stuart, additional, Makanjuola, David Osagie, additional, McCann, Liza, additional, Nandagudi, Anupama C., additional, Selvan, Shilpa, additional, O'Riordan, Edmond, additional, Patel, Mumtaz, additional, Patel, Rajan Kantilal, additional, Pusey, Charles Dickson, additional, Rajakariar, Ravindra, additional, Robson, Joanna C., additional, Robson, Michael, additional, Salama, Alan David, additional, Smyth, Lucy, additional, Sznajd, Jan, additional, Taylor, Joanne, additional, Sreih, Antonie G., additional, Belilos, Elise, additional, Bomback, Andrew S., additional, Carlin, Jeffrey, additional, Chang Chen Lin, Yih, additional, Derebail, Vimal K., additional, Dragoi, Serban, additional, Dua, Anisha, additional, Forbess, Lindsy, additional, Geetha, Duvuru, additional, Gipson, Patrick, additional, Gohh, Reginald, additional, Greenwood, Gregory Todd, additional, Hugenberg, Steven T., additional, Jimenez, Richard A.H., additional, Kaskas, Marwan Omar, additional, Kermani, Tanaz, additional, Kivitz, Alan J., additional, Koening, Curry, additional, Langford, Carol A., additional, Marder, Galina, additional, Mohamed, Amr Ahmed El-Huesseini, additional, Monach, Paul, additional, Neyra, Nilda Roxana, additional, Niemer, Gregory W., additional, Niles, John, additional, Obi, Reginald, additional, Owens, Charles, additional, Parks, Deborah L., additional, Podoll, Amber S., additional, Rovin, Brad, additional, Sam, R, additional, Shergy, William Julius, additional, Silva, Arnold Lawrence, additional, Specks, Ulrich, additional, Spiera, Robert, additional, Springer, Jason M., additional, Striebich, Christopher Charles, additional, Swarup, Areena, additional, Thakar, Surabhi, additional, Tiliakos, Athan N., additional, Tsai, Yong, additional, Waguespack, Dia R., additional, and Chester Wasko, Mary, additional

Published
2023
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18. 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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19. Measuring treatment outcomes and change in disease activity in giant cell arteritis: a systematic literature review informing the development of the EULAR-ACR response criteria on behalf of the EULAR-ACR response criteria in giant cell arteritis task force

Author

Sanchez-Alvarez, Catalina, primary, Bond, Milena, additional, Soowamber, Medha, additional, Camellino, Dario, additional, Anderson, Melanie, additional, Langford, Carol A, additional, Dejaco, Christian, additional, Touma, Zahi, additional, and Ramiro, Sofia, additional

Published
2023
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20. The Association Between Age at Diagnosis and Disease Characteristics and Damage in Patients With ANCA‐Associated Vasculitis.

Author

Bloom, Jessica L., Pickett‐Nairn, Kaci, Silveira, Lori, Fuhlbrigge, Robert C., Cuthbertson, David, Akuthota, Praveen, Corbridge, Thomas C., Khalidi, Nader A., Koening, Curry L., Langford, Carol A., McAlear, Carol A., Monach, Paul A., Moreland, Larry W., Pagnoux, Christian, Rhee, Rennie L., Seo, Philip, Silver, Jared, Specks, Ulrich, Warrington, Kenneth J., and Wechsler, Michael E.

Subjects
AGE distribution, INTUBATION, ANTINEUTROPHIL cytoplasmic antibodies, ALVEOLAR process, GRANULOMATOSIS with polyangiitis, NEUROLOGIC manifestations of general diseases, AGE factors in disease, SYMPTOMS, LARYNGEAL diseases, DISEASE duration, VASCULITIS, LONGITUDINAL method, MICROSCOPIC polyangiitis, HEMORRHAGE, TOBACCO
Abstract

Objective: This study examined the relationship between age at diagnosis and disease characteristics and damage in patients with antineutrophil cytoplasmic antibody (ANCA)–associated vasculitis (AAV). Methods: Analysis of a prospective longitudinal cohort of patients with granulomatosis with polyangiitis (GPA), microscopic polyangiitis (MPA), and eosinophilic GPA (EGPA) in the Vasculitis Clinical Research Consortium (2013–2021). Disease cohorts were divided by age at diagnosis (years): children (<18), young adults (18–40), middle‐aged adults (41–65), and older adults (>65). Data included demographics, ANCA type, clinical characteristics, Vasculitis Damage Index (VDI) scores, ANCA Vasculitis Index of Damage (AVID) scores, and novel disease‐specific and non‐disease‐specific damage scores built from VDI and AVID items. Results: Analysis included data from 1020 patients with GPA/MPA and 357 with EGPA. Female predominance in GPA/MPA decreased with age at diagnosis. AAV in childhood was more often GPA and proteinase 3‐ANCA positive. Children with GPA/MPA experienced more subglottic stenosis and alveolar hemorrhage; children and young adults with EGPA experienced more alveolar hemorrhage, need for intubation, and gastrointestinal involvement. Older adults (GPA/MPA) had more neurologic manifestations. After adjusting for disease duration, medications, tobacco, and ANCA, all damage scores increased with age at diagnosis for GPA/MPA (P < 0.001) except the disease‐specific damage score, which did not differ (P = 0.44). For EGPA, VDI scores increased with age at diagnosis (P < 0.009), whereas all other scores were not significantly different. Conclusion: Age at diagnosis is associated with clinical characteristics in AAV. Although VDI and AVID scores increase with age at diagnosis, this is driven by non‐disease‐specific damage items. [ABSTRACT FROM AUTHOR]

Published
2023
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21. 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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22. 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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23. Risk of relapse of ANCA-associated vasculitis among patients homozygous for the proteinase 3 gene Val119Ile polymorphism

Author

Casal Moura, Marta, primary, Deng, Zuoming, additional, Brooks, Stephen R, additional, Tew, Wei, additional, Fervenza, Fernando C, additional, Kallenberg, Cees G M, additional, Langford, Carol A, additional, Merkel, Peter A, additional, Monach, Paul A, additional, Seo, Philip, additional, Spiera, Robert F, additional, St Clair, E William, additional, Stone, John H, additional, Prunotto, Marco, additional, Grayson, Peter C, additional, and Specks, Ulrich, additional

Published
2023
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24. Identification of new risk loci and pathways involved in GCA pathogenesis by a genome-wide.

Author

Borrego Yaniz, Gonzalo, Ortiz-Fernández, Lourdes, Kerick, Martin, Madrid Paredes, Adela, Vaglio, Augusto, Hernández-Rodríguez, José, Mackie, Sarah, Castañeda, Santos, Solans-Laqué, Roser, Mestre, Jaume, Dasgupta, Bhaskar, Watts, Richard, Khalidi, Nader, Langford, Carol, Ytterberg, Steven R, Beretta, Lorenzo, Govoni, Marcelo, Emmi, Giacomo, Cimmino, Marco A., Witte, Torsten, Neumann, Thomas, Holle, Julia, Schönau, Verena, Pugnet, Gregory, Papo, Thomas, Haroche, Julien, Mahr, Alfred, Mouthon, Luc, Molberg, Oyvind, Diamantopoulos, Andreas P., Voskuyl, Alexandre E., Daikeler, Thomas, Berger, Christoph, Molloy, Eamonn, Blockmans, Daniel, Ortego-Centeno, Norberto, Brouwer, Elisabeth, Lamprecht, Peter, Klapa, Sebastian, Salvarani, Carlo, Merkel, Peter A., Cid, María C., González-Gay, M. A., Morgan, Ann, Martin Ibáñez, Javier, Márquez, Ana, Borrego Yaniz, Gonzalo, Ortiz-Fernández, Lourdes, Kerick, Martin, Madrid Paredes, Adela, Vaglio, Augusto, Hernández-Rodríguez, José, Mackie, Sarah, Castañeda, Santos, Solans-Laqué, Roser, Mestre, Jaume, Dasgupta, Bhaskar, Watts, Richard, Khalidi, Nader, Langford, Carol, Ytterberg, Steven R, Beretta, Lorenzo, Govoni, Marcelo, Emmi, Giacomo, Cimmino, Marco A., Witte, Torsten, Neumann, Thomas, Holle, Julia, Schönau, Verena, Pugnet, Gregory, Papo, Thomas, Haroche, Julien, Mahr, Alfred, Mouthon, Luc, Molberg, Oyvind, Diamantopoulos, Andreas P., Voskuyl, Alexandre E., Daikeler, Thomas, Berger, Christoph, Molloy, Eamonn, Blockmans, Daniel, Ortego-Centeno, Norberto, Brouwer, Elisabeth, Lamprecht, Peter, Klapa, Sebastian, Salvarani, Carlo, Merkel, Peter A., Cid, María C., González-Gay, M. A., Morgan, Ann, Martin Ibáñez, Javier, and Márquez, Ana

Published
2023

25. 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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26. Vasculitis

Author

Langford, Carol A., Shah, Samir K., editor, and Clair, Daniel G., editor

Published
2014
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31. Vitamin D status in ANCA-associated vasculitis

Author

Doubelt, Irena, primary, 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, additional, Seo, Philip, additional, Specks, Ulrich, additional, Warrington, Kenneth J, additional, Merkel, Peter A, additional, and Pagnoux, Christian, additional

Published
2022
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32. 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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33. Autoreactive plasmablasts after B cell depletion with rituximab and relapses in ANCA ‐associated vasculitis

Author

Berti, Alvise, primary, Hillion, Sophie, additional, Konig, Maximilian F., additional, Casal Moura, Marta, additional, Hummel, Amber M., additional, Carmona, Eva, additional, Peikert, Tobias, additional, Fervenza, Fernando C., additional, Kallenberg, Cees G.M., additional, Langford, Carol A., additional, Merkel, Peter A., additional, Monach, Paul A., additional, Seo, Philip, additional, Spiera, Robert F., additional, Brunetta, Paul, additional, St. Clair, E. William, additional, Harris, Kristina M., additional, Stone, John H., additional, Grandi, Guido, additional, Pers, Jacques‐Olivier, additional, Specks, Ulrich, additional, and Cornec, Divi, additional

Published
2022
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34. Urine eosinophil‐derived neurotoxin: A potential marker of activity in select eosinophilic disorders

Author

Makiya, Michelle A., primary, Khoury, Paneez, additional, Kuang, Fei Li, additional, Mata, Alexis Dominique, additional, Mahmood, Sana, additional, Bowman, Abbie, additional, Espinoza, David, additional, Kovacs, Nicholas, additional, Brown, Thomas, additional, Holland, Nicole, additional, Wetzler, Lauren, additional, Ware, JeanAnne M., additional, Dyer, Anne‐Marie, additional, Akuthota, Praveen, additional, Bochner, Bruce S., additional, Chinchilli, Vernon M., additional, Gleich, Gerald J., additional, Langford, Carol, additional, Merkel, Peter A., additional, Specks, Ulrich, additional, Weller, Peter F., additional, Wechsler, Michael E., additional, Prussin, Calman, additional, Fay, Michael P., additional, and Klion, Amy D., additional

Published
2022
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37. ANCA-Associated Vasculitis

Author

Stone, John H., Ozaki, Shoichi, Keogh, Karina, Specks, Ulrich, Langford, Carol A., Rasmussen, Niels, Kallenberg, Cees G. M., Bajema, Ingeborg M., and Stone, John H., editor

Published
2009
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38. Serum S100 Proteins as a Marker of Disease Activity in Large Vessel Vasculitis

Author

Springer, Jason Michael, Monach, Paul, Cuthbertson, David, Carette, Simon, Khalidi, Nader A., McAlear, Carol A., Pagnoux, Christian, Seo, Philip, Warrington, Kenneth J., Ytterberg, Steven R., Hoffman, Gary, Langford, Carol, Hamilton, Thomas, Foell, Dirk, Vogl, Thomas, Holzinger, Dirk, Merkel, Peter A., Roth, Johannes, and Hajj-Ali, Rula A.

Published
2018
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39. Contributors

Author

Abramson, Steven, primary, An, KaiNan, additional, Andrade, Felipe, additional, Ardoin, Stacy P., additional, Barton, Anne, additional, Baughman, Robert P., additional, Beaton, Dorcas E., additional, Beere, Helen M., additional, Beltran, Javier, additional, Bending, David, additional, Bennett, Robert M., additional, Bermas, Bonnie L., additional, Bertsias, George, additional, Bhardwaj, Nina, additional, Bijlsma, Johannes W.J., additional, Bockenstedt, Linda K., additional, Boers, Maarten, additional, Boilard, Eric, additional, Boin, Francesco, additional, Boumpas, Dimitrios T., additional, Boyle, David L., additional, Bradley, Sean, additional, Brown, Matthew, additional, Buch, Maya, additional, Buckley, Christopher D., additional, Budd, Ralph C., additional, Burg, Nathalie, additional, Burns, Christopher M., additional, Cannella, Amy C., additional, Carter, John D., additional, Chakravarty, Eliza F., additional, Chakravarty, Soumya D., additional, Chang, Christopher, additional, Cheng, Joseph S., additional, Chiodo, Christopher P., additional, Chung, Sharon, additional, Cleland, Leslie G., additional, Cohen, Stanley, additional, Colbert, Robert A., additional, Cook, Paul P., additional, Craft, Joseph E., additional, Crofford, Leslie J., additional, Cronstein, Bruce N., additional, Crow, Mary K., additional, Crowson, Cynthia S., additional, Culley, Kirsty L., additional, Cunnane, Gaye, additional, Dall'Era, Maria, additional, Darrah, Erika, additional, Davis, John M., additional, Bari, Cosimo De, additional, Dell'Accio, Francesco, additional, Diamond, Betty, additional, Di Cesare, Paul E., additional, Dixit, Rajiv, additional, Drenth, Joost P.H., additional, Dustin, Michael L., additional, El-Gabalawy, Hani S., additional, Elmamoun, Musaab, additional, Erickson, Alan R., additional, Erkan, Doruk, additional, Eyre, Stephen, additional, Fanouriakis, Antonis, additional, Felson, David T., additional, Field, Max, additional, Filer, Andrew, additional, Firestein, Gary S., additional, Fishman, Felicity G., additional, FitzGerald, Oliver, additional, Flaherty, John P., additional, Fors, César E., additional, Fortner, Karen A., additional, Gabriel, Sherine E., additional, Gasque, Philippe, additional, Gershwin, M. Eric, additional, Gladue, Heather S., additional, Goldring, Mary B., additional, Goldring, Steven R., additional, Golightly, Yvonne M., additional, Goodman, Stuart, additional, Gordon, Siamon, additional, Grassi, Walter, additional, Green, Douglas R., additional, Greenspan, Adam, additional, Gregersen, Peter, additional, Grimaldi, Christine, additional, Guilherme, Luiza, additional, Hajj-Ali, Rula A., additional, Haudenschild, Dominik R., additional, Hellmann, David B., additional, Holmdahl, Rikard, additional, Hsu, Joyce J., additional, Huddleston, James I., additional, Hudson, Alan P., additional, Huizinga, Thomas W.J., additional, Hunder, Gene G., additional, Iversen, Maura D., additional, Jacobs, Johannes W.G., additional, Jen, Ho, additional, Jordan, Joanne M., additional, Jorizzo, Joseph L., additional, Kalil, Jorge, additional, Kaufman, Kenton, additional, Kaufman, William S., additional, Kavanaugh, Arthur, additional, Keenan, Robert T., additional, Kenna, Tony, additional, Kerr, Darcy A., additional, Koch, Alisa E., additional, Kono, Dwight H., additional, Korsten, Peter, additional, Krakow, Deborah, additional, Krasnokutsky, Svetlana, additional, Lafeber, Floris P.J.G., additional, Lambert, Robert G.W., additional, Lane, Nancy E., additional, Langford, Carol A., additional, Laskin, Daniel M., additional, Layh-Schmitt, Gerlinde, additional, Lee, Lela A., additional, Lee, Tzielan C., additional, Lockshin, Michael D., additional, Lozada, Carlos J., additional, Lundberg, Ingrid E., additional, Luqmani, Raashid, additional, Luyten, Frank P., additional, Mader, Reuven, additional, Maksymowych, Walter, additional, Markenson, Joseph A., additional, Martin, Scott David, additional, Matteson, Eric L., additional, McGregor, Laura, additional, McInnes, Iain B., additional, McNamara, Elizabeth K., additional, Mikuls, Ted R., additional, Miller, Mark S., additional, Ming, Pedro Azevedo, additional, Moder, Kevin G., additional, Monach, Paul A., additional, Moulton, Vaishali R., additional, Nagaraju, Kanneboyina, additional, Nelson, Amanda E., additional, Nigrovic, Peter A., additional, Nistala, Kiran, additional, O'Dell, James R., additional, Okada, Yasunori, additional, Østergaard, Mikkel, additional, Otero, Miguel, additional, Palmer, Bradley M., additional, Panush, Richard S., additional, Peng, Stanford L., additional, Pillai, Shiv, additional, Pillinger, Michael H., additional, Plüddemann, Annette, additional, Polston, Gregory R., additional, Porcelli, Steven A., additional, Price, Mark D., additional, Reed, Ann M., additional, Reveille, John D., additional, Robinson, Angela B., additional, Robinson, Philip, additional, Robinson, William H., additional, Roosendaal, Goris, additional, Rosen, Antony, additional, Rosenbaum, James T., additional, Rosenberg, Andrew E., additional, Ruderman, Eric M., additional, Saag, Kenneth G., additional, Salmon, Jane E., additional, Sammaritano, Lisa R., additional, Samuels, Jonathan, additional, Sandborg, Christy I., additional, Sawalha, Amr H., additional, Saxena, Amit, additional, Schett, Georg, additional, Schutgens, Roger E.G., additional, Seldin, David C., additional, Shah, Binita, additional, Sikora, Keith A., additional, Simon, Anna, additional, Siraj, Dawd S., additional, Sorkin, Linda S., additional, St. Clair, E. William, additional, Stamp, Lisa K., additional, Stone, John H., additional, Suarez-Fueyo, Abel, additional, Svensson, Camilla I., additional, Sweiss, Nadera J., additional, Swigart, Carrie R., additional, Szekanecz, Zoltán, additional, Tait, Stephen, additional, Tanne, Antoine, additional, Taylor, Peter C., additional, Terkeltaub, Robert, additional, Theofilopoulos, Argyrios N., additional, Thornhill, Thomas S., additional, Torok, Kathryn S., additional, Toth, Michael J., additional, Tozman, Elaine C., additional, Trouw, Leendert A., additional, Tsokos, George C., additional, Tugwell, Peter, additional, Tutuncu, Zuhre, additional, Upadhyaya, Shivam, additional, Van, Annette H.M., additional, van der Linden, Sjef, additional, Van, Jos W.M., additional, Van, Jacob M., additional, Meter, Heather Van, additional, van Vollenhoven, Ronald F., additional, van Vulpen, Lize F.D., additional, Varga, John, additional, Vaseer, Samera, additional, Vasquez-Castellanos, Raul, additional, Veale, Douglas J., additional, Wakefield, Richard J., additional, Wallace, Mark S., additional, Wang, Ruoning, additional, Wang, Tingting, additional, Warshaw, David M., additional, Wedderburn, Lucy R., additional, Werth, Victoria P., additional, Wigley, Fredrick M., additional, Wofsy, David, additional, Wollheim, Frank A., additional, Wondimu, Elisabeth, additional, Wong, Cyrus, additional, Wortmann, Robert L., additional, Yelin, Edward, additional, Zayat, Ahmed, additional, Zou, Yong-Rui, additional, and Zurier, Robert B., additional

Published
2017
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41. 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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42. Association of baseline soluble immune checkpoints with the risk of relapse in PR3-ANCA vasculitis following induction of remission

Author

Gamerith, Gabriele, primary, Mildner, Finn, additional, Merkel, Peter A, additional, Harris, Kristina, additional, Cooney, Laura, additional, Lim, Noha, additional, Spiera, Robert, additional, Seo, Philip, additional, Langford, Carol A, additional, Hoffman, Gary S, additional, St Clair, E William, additional, Fervenza, Fernando C, additional, Monach, Paul, additional, Ytterberg, Steven R, additional, Geetha, Duvuru, additional, Amann, Arno, additional, Wolf, Dominik, additional, Specks, Ulrich, additional, Stone, John H, additional, and Kronbichler, Andreas, additional

Published
2022
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45. The Pharmacogenomic Association of Fcγ Receptors and Cytochrome P450 Enzymes With Response to Rituximab or Cyclophosphamide Treatment in Antineutrophil Cytoplasmic Antibody–Associated Vasculitis

Author

Cartin‐Ceba, Rodrigo, Indrakanti, Divya, Specks, Ulrich, Stone, John H., Hoffman, Gary S., Kallenberg, Cees G. M., Langford, Carol A., Merkel, Peter A., Spiera, Robert F., Monach, Paul A., St.Clair, E. William, Seo, Philip, Tchao, Nadia K., Ytterberg, Steven R., Brunetta, Paul G., Song, Huijuan, Birmingham, Dan, and Rovin, Brad H.

Published
2017
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47. Activation of a Latent Epitope Causing Differential Binding of Antineutrophil Cytoplasmic Antibodies to Proteinase 3.

Author

Casal Moura, Marta, Thompson, Gwen E., Nelson, Darlene R., Fussner, Lynn A., Hummel, Amber M., Jenne, Dieter E., Emerling, Daniel, Fervenza, Fernando C., Kallenberg, Cees G. M., Langford, Carol A., McCune, W. Joseph, Merkel, Peter A., Monach, Paul A., Seo, Philip, Spiera, Robert F., St. Clair, E. William, Ytterberg, Steven R., Stone, John H., Robinson, William H., and Specks, Ulrich

Subjects
GENETIC mutation, ANTINEUTROPHIL cytoplasmic antibodies, PROTEOLYTIC enzymes, MONOCLONAL antibodies, GRANULOMATOSIS with polyangiitis, IMMUNOASSAY, COMPARATIVE studies, ANTIGENS, VASCULITIS, LONGITUDINAL method
Abstract

Objective: Proteinase 3 (PR3) is the major antigen for antineutrophil cytoplasmic antibodies (ANCAs) in the systemic autoimmune vasculitis, granulomatosis with polyangiitis (GPA). PR3‐targeting ANCAs (PR3‐ANCAs) recognize different epitopes on PR3. This study was undertaken to study the effect of mutations on PR3 antigenicity. Methods: The recombinant PR3 variants, iPR3 (clinically used to detect PR3‐ANCAs) and iHm5 (containing 3 point mutations in epitopes 1 and 5 generated for epitope mapping studies) immunoassays and serum samples from patients enrolled in ANCA‐associated vasculitis (AAV) trials were used to screen for differential PR3‐ANCA binding. A patient‐derived monoclonal ANCA 518 (moANCA518) that selectively binds to iHm5 within the mutation‐free epitope 3 and is distant from the point mutations of iHm5 was used as a gauge for remote epitope activation. Selective binding was determined using inhibition experiments. Results: Rather than reduced binding of PR3‐ANCAs to iHm5, we found substantially increased binding of the majority of PR3‐ANCAs to iHm5 compared to iPR3. This differential binding of PR3‐ANCA to iHm5 is similar to the selective moANCA518 binding to iHm5. Binding of iPR3 to monoclonal antibody MCPR3‐2 also induced recognition by moANCA518. Conclusion: The preferential binding of PR3‐ANCAs from patients, such as the selective binding of moANCA518 to iHm5, is conferred by increased antigenicity of epitope 3 on iHm5. This can also be induced on iPR3 when captured by monoclonal antibody MCPR2. This previously unrecognized characteristic of PR3‐ANCA interactions with its target antigen has implications for studying antibody‐mediated autoimmune diseases, understanding variable performance characteristics of immunoassays, and design of potential novel treatment approaches. [ABSTRACT FROM AUTHOR]

Published
2023
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48. Autoreactive Plasmablasts After B Cell Depletion With Rituximab and Relapses in Antineutrophil Cytoplasmic Antibody–Associated Vasculitis.

Author

Berti, Alvise, Hillion, Sophie, Konig, Maximilian F., Moura, Marta Casal, Hummel, Amber M., Carmona, Eva, Peikert, Tobias, Fervenza, Fernando C., Kallenberg, Cees G.M., Langford, Carol A., Merkel, Peter A., Monach, Paul A., Seo, Philip, Spiera, Robert F., Brunetta, Paul, St. Clair, E. William, Harris, Kristina M., Stone, John H., Grandi, Guido, and Pers, Jacques‐Olivier

Subjects
RITUXIMAB, FLOW cytometry, B cells, ANTINEUTROPHIL cytoplasmic antibodies, DISEASE relapse, RESEARCH funding, VASCULITIS, DISEASE remission
Abstract

Objective: Autoreactive B cells are responsible for antineutrophil cytoplasmic antibody (ANCA) production in ANCA‐associated vasculitis (AAV). Rituximab (RTX) depletes circulating B cells, including autoreactive B cells. We aimed to evaluate changes and associations with relapse of the circulating autoreactive B cell pool following therapeutic B cell depletion in AAV. Methods: Sequential flow cytometry was performed on 148 samples of peripheral blood mononuclear cells from 23 patients with proteinase 3 (PR3)–ANCA–positive AAV who were treated with RTX for remission induction and monitored after stopping therapy during long‐term follow‐up in a prospective clinical trial. PR3 was used as a ligand to target autoreactive PR3‐specific (PR3+) B cells. B cell recurrence was considered as the first blood sample with ≥10 B cells/μl after RTX treatment. Results: At B cell recurrence, PR3+ B cell frequency among B cells was higher than baseline (P < 0.01). Within both PR3+ and total B cells, frequencies of transitional and naive subsets were higher at B cell recurrence than at baseline, while memory subsets were lower (P < 0.001 for all comparisons). At B cell recurrence, frequencies of B cells and subsets did not differ between patients who experienced relapse and patients who remained in remission. In contrast, the plasmablast frequency within the PR3+ B cell pool was higher in patients who experienced relapse and associated with a shorter time to relapse. Frequencies of PR3+ plasmablasts higher than baseline were more likely to be found in patients who experienced relapse within the following 12 months compared to those in sustained remission (P < 0.05). Conclusion: The composition of the autoreactive B cell pool varies significantly following RTX treatment in AAV, and early plasmablast enrichment within the autoreactive pool is associated with future relapses. [ABSTRACT FROM AUTHOR]

Published
2023
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49. 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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50. An International Delphi Exercise to Identify Items of Importance for Measuring Response to Treatment in ANCA-Associated Vasculitis

Author

Quinn, Kaitlin A., primary, Monti, Sara, additional, Christensen, Robin, additional, Jayne, David, additional, Langford, Carol A., additional, Lanier, Georgia E., additional, Mahr, Alfred, additional, Pagnoux, Christian, additional, Shea, Beverley, additional, Viðarsdóttir, Maria Bjork, additional, Tomasson, Gunnar, additional, and Merkel, Peter A., additional

Published
2022
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