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1. Impact of Risk Factors on COVID-19 Outcomes in Unvaccinated People With Rheumatic Diseases: A Comparative Analysis of Pandemic Epochs Using the COVID-19 Global Rheumatology Alliance Registry

Author

Yazdany, J, Ware, A, Wallace, Z, Bhana, S, Grainger, R, Hachulla, E, Richez, C, Cacoub, P, Hausmann, J, Liew, J, Sirotich, E, Jacobsohn, L, Strangfeld, A, Mateus, E, Hyrich, K, Gossec, L, Carmona, L, Lawson-Tovey, S, Kearsley-Fleet, L, Schaefer, M, Ribeiro, S, Al-Emadi, S, Hasseli, R, Müller-Ladner, U, Specker, C, Schulze-Koops, H, Bernardes, M, Fraga, V, Rodrigues, A, Sparks, J, Ljung, L, Di Giuseppe, D, Tidblad, L, Wise, L, Duarte-García, A, Ugarte-Gil, M, Colunga-Pedraza, I, Martínez-Martínez, M, Alpizar-Rodriguez, D, Xavier, R, Isnardi, C, Pera, M, Pons-Estel, G, Izadi, Z, Gianfrancesco, M, Carrara, G, Scirè, C, Zanetti, A, Machado, P, Wallace, ZS, Hausmann, JS, Liew, JW, Mateus, EF, Hyrich, KL, Ribeiro, SLE, Fraga, VM, Rodrigues, AM, Sparks, JA, Ugarte-Gil, MF, Colunga-Pedraza, IJ, Martínez-Martínez, MU, Xavier, RM, Isnardi, CA, Gianfrancesco, MA, Scirè, CA, Machado, PM, Yazdany, J, Ware, A, Wallace, Z, Bhana, S, Grainger, R, Hachulla, E, Richez, C, Cacoub, P, Hausmann, J, Liew, J, Sirotich, E, Jacobsohn, L, Strangfeld, A, Mateus, E, Hyrich, K, Gossec, L, Carmona, L, Lawson-Tovey, S, Kearsley-Fleet, L, Schaefer, M, Ribeiro, S, Al-Emadi, S, Hasseli, R, Müller-Ladner, U, Specker, C, Schulze-Koops, H, Bernardes, M, Fraga, V, Rodrigues, A, Sparks, J, Ljung, L, Di Giuseppe, D, Tidblad, L, Wise, L, Duarte-García, A, Ugarte-Gil, M, Colunga-Pedraza, I, Martínez-Martínez, M, Alpizar-Rodriguez, D, Xavier, R, Isnardi, C, Pera, M, Pons-Estel, G, Izadi, Z, Gianfrancesco, M, Carrara, G, Scirè, C, Zanetti, A, Machado, P, Wallace, ZS, Hausmann, JS, Liew, JW, Mateus, EF, Hyrich, KL, Ribeiro, SLE, Fraga, VM, Rodrigues, AM, Sparks, JA, Ugarte-Gil, MF, Colunga-Pedraza, IJ, Martínez-Martínez, MU, Xavier, RM, Isnardi, CA, Gianfrancesco, MA, Scirè, CA, and Machado, PM

Abstract

Objective: Approximately one third of individuals worldwide have not received a COVID-19 vaccine. Although studies have investigated risk factors linked to severe COVID-19 among unvaccinated people with rheumatic diseases (RDs), we know less about whether these factors changed as the pandemic progressed. We aimed to identify risk factors associated with severe COVID-19 in unvaccinated individuals in different pandemic epochs corresponding to major variants of concern. Methods: Patients with RDs and COVID-19 were entered into the COVID-19 Global Rheumatology Alliance Registry between March 2020 and June 2022. An ordinal logistic regression model (not hospitalized, hospitalized, and death) was used with date of COVID-19 diagnosis, age, sex, race and/or ethnicity, comorbidities, RD activity, medications, and the human development index (HDI) as covariates. The main analysis included all unvaccinated patients across COVID-19 pandemic epochs; subanalyses stratified patients according to RD types. Results: Among 19,256 unvaccinated people with RDs and COVID-19, those who were older, male, had more comorbidities, used glucocorticoids, had higher disease activity, or lived in lower HDI regions had worse outcomes across epochs. For those with rheumatoid arthritis, sulfasalazine and B-cell–depleting therapy were associated with worse outcomes, and tumor necrosis factor inhibitors were associated with improved outcomes. In those with connective tissue disease or vasculitis, B-cell–depleting therapy was associated with worse outcomes. Conclusion: Risk factors for severe COVID-19 outcomes were similar throughout pandemic epochs in unvaccinated people with RDs. Ongoing efforts, including vaccination, are needed to reduce COVID-19 severity in this population, particularly in those with medical and social vulnerabilities identified in this study.

Published
2024

2. Prolonged COVID-19 symptom duration in people with systemic autoimmune rheumatic diseases: results from the COVID-19 Global Rheumatology Alliance Vaccine Survey

Author

DiIorio, M, Kennedy, K, Liew, JW, Putman, MS, Sirotich, E, Sattui, SE, Foster, G, Harrison, C, Larche, MJ, Levine, M, Moni, TT, Thabane, L, Bhana, S, Costello, W, Grainger, R, Machado, PM, Robinson, PC, Sufka, P, Wallace, ZS, Yazdany, J, Gore-Massy, M, Howard, RA, Kodhek, MA, Lalonde, N, Tomasella, L-A, Wallace, J, Akpabio, A, Alpizar-Rodriguez, D, Beesley, RP, Berenbaum, F, Bulina, I, Chock, EY, Conway, R, Duarte-Garcia, A, Duff, E, Gheita, TA, Graef, ER, Hsieh, E, El Kibbi, L, Liew, DFL, Lo, C, Nudel, M, Singh, AD, Singh, JA, Singh, N, Ugarte-Gil, MF, Hausmann, JS, Simard, JF, Sparks, JA, DiIorio, M, Kennedy, K, Liew, JW, Putman, MS, Sirotich, E, Sattui, SE, Foster, G, Harrison, C, Larche, MJ, Levine, M, Moni, TT, Thabane, L, Bhana, S, Costello, W, Grainger, R, Machado, PM, Robinson, PC, Sufka, P, Wallace, ZS, Yazdany, J, Gore-Massy, M, Howard, RA, Kodhek, MA, Lalonde, N, Tomasella, L-A, Wallace, J, Akpabio, A, Alpizar-Rodriguez, D, Beesley, RP, Berenbaum, F, Bulina, I, Chock, EY, Conway, R, Duarte-Garcia, A, Duff, E, Gheita, TA, Graef, ER, Hsieh, E, El Kibbi, L, Liew, DFL, Lo, C, Nudel, M, Singh, AD, Singh, JA, Singh, N, Ugarte-Gil, MF, Hausmann, JS, Simard, JF, and Sparks, JA

Abstract

OBJECTIVE: We investigated prolonged COVID-19 symptom duration, defined as lasting 28 days or longer, among people with systemic autoimmune rheumatic diseases (SARDs). METHODS: We analysed data from the COVID-19 Global Rheumatology Alliance Vaccine Survey (2 April 2021-15 October 2021) to identify people with SARDs reporting test-confirmed COVID-19. Participants reported COVID-19 severity and symptom duration, sociodemographics and clinical characteristics. We reported the proportion experiencing prolonged symptom duration and investigated associations with baseline characteristics using logistic regression. RESULTS: We identified 441 respondents with SARDs and COVID-19 (mean age 48.2 years, 83.7% female, 39.5% rheumatoid arthritis). The median COVID-19 symptom duration was 15 days (IQR 7, 25). Overall, 107 (24.2%) respondents had prolonged symptom duration (≥28 days); 42/429 (9.8%) reported symptoms lasting ≥90 days. Factors associated with higher odds of prolonged symptom duration included: hospitalisation for COVID-19 vs not hospitalised and mild acute symptoms (age-adjusted OR (aOR) 6.49, 95% CI 3.03 to 14.1), comorbidity count (aOR 1.11 per comorbidity, 95% CI 1.02 to 1.21) and osteoarthritis (aOR 2.11, 95% CI 1.01 to 4.27). COVID-19 onset in 2021 vs June 2020 or earlier was associated with lower odds of prolonged symptom duration (aOR 0.42, 95% CI 0.21 to 0.81). CONCLUSION: Most people with SARDs had complete symptom resolution by day 15 after COVID-19 onset. However, about 1 in 4 experienced COVID-19 symptom duration 28 days or longer; 1 in 10 experienced symptoms 90 days or longer. Future studies are needed to investigate the possible relationships between immunomodulating medications, SARD type/flare, vaccine doses and novel viral variants with prolonged COVID-19 symptoms and other postacute sequelae of COVID-19 among people with SARDs.

Published
2022

3. Risk of severe COVID-19 associated with immune-modifying drugs: Data from PsoProtect and Global Rheumatology Alliance registries.

Author

Mahil, S, Schaefer, M, Dand, N, Yiu, Z, Hyrich, K, Strangfeld, A, Yates, M, Carmona, L, Gossec, L, Mateus, E, Lawson-Tovey, S, Wiek, D, Jacobsohn, L, Isnardi, C, Quintana, R, Soriano, E, Wallace, Z, Bhana, S, Gore-Massey, M, Grainger, R, Hausmann, J, Liew, J, Sirotich, E, Mason, K, Tsakok, T, Meynell, F, Coker, B, Vincent, A, Urmston, D, Mcateer, H, Vesty, A, Kelly, J, Lancelot, C, Moorhead, L, Mackenzie, T, Rossi, M, Rivera, R, Mahe, E, Carugno, A, Magnano, M, Rech, G, Balogh, E, Feldman, S, De La Cruz, C, Di Meglio, P, Contreras, C, Choon, S, Capon, F, Torres, T, Naldi, L, Weinman, J, Brown, M, Galloway, J, Norton, S, Lambert, J, Spuls, P, Van Huizen, A, Jullien, D, Bachelez, H, Mcmahon, D, Freeman, E, Gisondi, P, Puig, L, Warren, R, Langan, S, Sufka, P, Robinson, P, Yazdany, J, Griffiths, C, Barker, J, Machado, P, Smith, C, Mahil SK, Schaefer M, Dand N, Yiu ZZN, Hyrich KL, Strangfeld A, Yates M, Carmona L, Gossec L, Mateus EF, Lawson-Tovey S, Wiek D, Jacobsohn L, Isnardi CA, Quintana R, Soriano ER, Wallace ZS, Bhana S, Gore-Massey M, Grainger R, Hausmann JS, Liew JW, Sirotich E, Mason KJ, Tsakok T, Meynell F, Coker B, Vincent A, Urmston D, McAteer H, Vesty A, Kelly J, Lancelot C, Moorhead L, Mackenzie T, Rossi MT, Rivera R, Mahe E, Carugno A, Magnano M, Rech G, Balogh EA, Feldman SR, De La Cruz C, Di Meglio P, Contreras CR, Choon SE, Capon F, Torres T, Naldi L, Weinman J, Brown MA, Galloway JB, Norton S, Lambert J, Spuls P, Van Huizen AM, Jullien D, Bachelez H, McMahon DE, Freeman EE, Gisondi P, Puig L, Warren RB, Langan SM, Sufka P, Robinson PC, Yazdany J, Griffiths CEM, Barker JN, Machado PM, Smith CH., Mahil, S, Schaefer, M, Dand, N, Yiu, Z, Hyrich, K, Strangfeld, A, Yates, M, Carmona, L, Gossec, L, Mateus, E, Lawson-Tovey, S, Wiek, D, Jacobsohn, L, Isnardi, C, Quintana, R, Soriano, E, Wallace, Z, Bhana, S, Gore-Massey, M, Grainger, R, Hausmann, J, Liew, J, Sirotich, E, Mason, K, Tsakok, T, Meynell, F, Coker, B, Vincent, A, Urmston, D, Mcateer, H, Vesty, A, Kelly, J, Lancelot, C, Moorhead, L, Mackenzie, T, Rossi, M, Rivera, R, Mahe, E, Carugno, A, Magnano, M, Rech, G, Balogh, E, Feldman, S, De La Cruz, C, Di Meglio, P, Contreras, C, Choon, S, Capon, F, Torres, T, Naldi, L, Weinman, J, Brown, M, Galloway, J, Norton, S, Lambert, J, Spuls, P, Van Huizen, A, Jullien, D, Bachelez, H, Mcmahon, D, Freeman, E, Gisondi, P, Puig, L, Warren, R, Langan, S, Sufka, P, Robinson, P, Yazdany, J, Griffiths, C, Barker, J, Machado, P, Smith, C, Mahil SK, Schaefer M, Dand N, Yiu ZZN, Hyrich KL, Strangfeld A, Yates M, Carmona L, Gossec L, Mateus EF, Lawson-Tovey S, Wiek D, Jacobsohn L, Isnardi CA, Quintana R, Soriano ER, Wallace ZS, Bhana S, Gore-Massey M, Grainger R, Hausmann JS, Liew JW, Sirotich E, Mason KJ, Tsakok T, Meynell F, Coker B, Vincent A, Urmston D, McAteer H, Vesty A, Kelly J, Lancelot C, Moorhead L, Mackenzie T, Rossi MT, Rivera R, Mahe E, Carugno A, Magnano M, Rech G, Balogh EA, Feldman SR, De La Cruz C, Di Meglio P, Contreras CR, Choon SE, Capon F, Torres T, Naldi L, Weinman J, Brown MA, Galloway JB, Norton S, Lambert J, Spuls P, Van Huizen AM, Jullien D, Bachelez H, McMahon DE, Freeman EE, Gisondi P, Puig L, Warren RB, Langan SM, Sufka P, Robinson PC, Yazdany J, Griffiths CEM, Barker JN, Machado PM, and Smith CH.

Published
2022

4. Lifetime Allergy Symptoms in IgG4-Related Disease: A Case-Control Study

Author

Sanders S, Fu X, Zhang Y, Perugino CA, Wallwork R, DELLA TORRE E, Harvey L, Harkness T, Long A, Choi HK, Stone JH, Wallace ZS., Sanders, S, Fu, X, Zhang, Y, Perugino, Ca, Wallwork, R, DELLA TORRE, E, Harvey, L, Harkness, T, Long, A, Choi, Hk, Stone, Jh, and Wallace, Zs.

Published
2020

5. Immediate effect of the COVID-19 pandemic on patient health, health-care use, and behaviours: results from an international survey of people with rheumatic diseases

Author

Hausmann, JS, Kennedy, K, Simard, JF, Liew, JW, Sparks, JA, Moni, T, Harrison, C, Larche, MJ, Levine, M, Sattui, SE, Semalulu, T, Foster, G, Surangiwala, S, Thabane, L, Beesley, RP, Durrant, KL, Mateus, EF, Mingolla, S, Nudel, M, Palmerlee, CA, Richards, DP, Liew, DFL, Hill, CL, Bhana, S, Costello, W, Grainger, R, Machado, PM, Robinson, PC, Sufka, P, Wallace, ZS, Yazdany, J, Sirotich, E, Hausmann, JS, Kennedy, K, Simard, JF, Liew, JW, Sparks, JA, Moni, T, Harrison, C, Larche, MJ, Levine, M, Sattui, SE, Semalulu, T, Foster, G, Surangiwala, S, Thabane, L, Beesley, RP, Durrant, KL, Mateus, EF, Mingolla, S, Nudel, M, Palmerlee, CA, Richards, DP, Liew, DFL, Hill, CL, Bhana, S, Costello, W, Grainger, R, Machado, PM, Robinson, PC, Sufka, P, Wallace, ZS, Yazdany, J, and Sirotich, E

Abstract

BACKGROUND: The impact and consequences of the COVID-19 pandemic on people with rheumatic disease are unclear. We developed the COVID-19 Global Rheumatology Alliance Patient Experience Survey to assess the effects of the COVID-19 pandemic on people with rheumatic disease worldwide. METHODS: Survey questions were developed by key stakeholder groups and disseminated worldwide through social media, websites, and patient support organisations. Questions included demographics, rheumatic disease diagnosis, COVID-19 diagnosis, adoption of protective behaviours to mitigate COVID-19 exposure, medication access and changes, health-care access and communication with rheumatologists, and changes in employment or schooling. Adults age 18 years and older with inflammatory or autoimmune rheumatic diseases were eligible for inclusion. We included participants with and without a COVID-19 diagnosis. We excluded participants reporting only non-inflammatory rheumatic diseases such as fibromyalgia or osteoarthritis. FINDINGS: 12 117 responses to the survey were received between April 3 and May 8, 2020, and of these, 10 407 respondents had included appropriate age data. We included complete responses from 9300 adults with rheumatic disease (mean age 46·1 years; 8375 [90·1%] women, 893 [9·6%] men, and 32 [0·3%] participants who identified as non-binary). 6273 (67·5%) of respondents identified as White, 1565 (16·8%) as Latin American, 198 (2·1%) as Black, 190 (2·0%) as Asian, and 42 (0·5%) as Native American or Aboriginal or First Nation. The most common rheumatic disease diagnoses included rheumatoid arthritis (3636 [39·1%] of 9300), systemic lupus erythematosus (2882 [31·0%]), and Sjögren's syndrome (1290 [13·9%]). Most respondents (6921 [82·0%] of 8441) continued their antirheumatic medications as prescribed. Almost all (9266 [99·7%] of 9297) respondents adopted protective behaviours to limit SARS-CoV-2 exposure. A change in employment status occurred in 2524 (27·1%) of 9300) of respond

Published
2021

6. Early experience of COVID-19 vaccination in adults with systemic rheumatic diseases: results from the COVID-19 Global Rheumatology Alliance Vaccine Survey

Author

Sattui, SE, Liew, JW, Kennedy, K, Sirotich, E, Putman, M, Moni, TT, Akpabio, A, Alpizar-Rodriguez, D, Berenbaum, F, Bulina, I, Conway, R, Singh, AD, Duff, E, Durrant, KL, Gheita, TA, Hill, CL, Howard, RA, Hoyer, BF, Hsieh, E, El Kibbi, L, Kilian, A, Kim, AH, Liew, DFL, Lo, C, Miller, B, Mingolla, S, Nudel, M, Palmerlee, CA, Singh, JA, Singh, N, Ugarte-Gil, MF, Wallace, J, Young, KJ, Bhana, S, Costello, W, Grainger, R, Machado, PM, Robinson, PC, Sufka, P, Wallace, ZS, Yazdany, J, Harrison, C, Larche, M, Levine, M, Foster, G, Thabane, L, Rider, LG, Hausmann, JS, Simard, JF, Sparks, JA, Sattui, SE, Liew, JW, Kennedy, K, Sirotich, E, Putman, M, Moni, TT, Akpabio, A, Alpizar-Rodriguez, D, Berenbaum, F, Bulina, I, Conway, R, Singh, AD, Duff, E, Durrant, KL, Gheita, TA, Hill, CL, Howard, RA, Hoyer, BF, Hsieh, E, El Kibbi, L, Kilian, A, Kim, AH, Liew, DFL, Lo, C, Miller, B, Mingolla, S, Nudel, M, Palmerlee, CA, Singh, JA, Singh, N, Ugarte-Gil, MF, Wallace, J, Young, KJ, Bhana, S, Costello, W, Grainger, R, Machado, PM, Robinson, PC, Sufka, P, Wallace, ZS, Yazdany, J, Harrison, C, Larche, M, Levine, M, Foster, G, Thabane, L, Rider, LG, Hausmann, JS, Simard, JF, and Sparks, JA

Abstract

BACKGROUND: We describe the early experiences of adults with systemic rheumatic disease who received the COVID-19 vaccine. METHODS: From 2 April to 30 April 2021, we conducted an online, international survey of adults with systemic rheumatic disease who received COVID-19 vaccination. We collected patient-reported data on clinician communication, beliefs and intent about discontinuing disease-modifying antirheumatic drugs (DMARDs) around the time of vaccination, and patient-reported adverse events after vaccination. RESULTS: We analysed 2860 adults with systemic rheumatic diseases who received COVID-19 vaccination (mean age 55.3 years, 86.7% female, 86.3% white). Types of COVID-19 vaccines were Pfizer-BioNTech (53.2%), Oxford/AstraZeneca (22.6%), Moderna (21.3%), Janssen/Johnson & Johnson (1.7%) and others (1.2%). The most common rheumatic disease was rheumatoid arthritis (42.3%), and 81.2% of respondents were on a DMARD. The majority (81.9%) reported communicating with clinicians about vaccination. Most (66.9%) were willing to temporarily discontinue DMARDs to improve vaccine efficacy, although many (44.3%) were concerned about rheumatic disease flares. After vaccination, the most reported patient-reported adverse events were fatigue/somnolence (33.4%), headache (27.7%), muscle/joint pains (22.8%) and fever/chills (19.9%). Rheumatic disease flares that required medication changes occurred in 4.6%. CONCLUSION: Among adults with systemic rheumatic disease who received COVID-19 vaccination, patient-reported adverse events were typical of those reported in the general population. Most patients were willing to temporarily discontinue DMARDs to improve vaccine efficacy. The relatively low frequency of rheumatic disease flare requiring medications was reassuring.

Published
2021

7. Clinical phenotypes of IgG4-related disease: an analysis of two international cross-sectional cohorts

Author

Wallace, Zs, Zhang, Y, Perugino, Ca, Naden, R, Choi, Hk, Stone, Jh, ACR/EULAR IgG4-RD Classification Criteria Committee, Takashi, Akamizu, Mitsuhiro, Akiyama, Adrian, Bateman, Daniel, Blockmans, Pilar, Brito-Zeron, Corrado, Campochiaro, Mollie, Carruthers, Suresh, Chari, Tsutomu, Chiba, Hyon, Choi, Andreu Fernandez Codina, Lynn, Cornell, Emma, Culver, Emanuel, Della-Torre, Vikram, Deshpande, Jean-Francois, Dicaire, Lingli, Dong, Mikael, Ebbo, Judith, A Ferry, George, Fragkoulis, Fabian, Frost, Luca, Frulloni, Phil, A Hart, Gabriela, Hernandez-Molina, Dai, Inoue, Karuna, Keat, Terumi, Kamisawa, Shigeyuki, Kawa, Mitsuhiro, Kawano, Arezou, Khosroshahi, Hiroshi, Kobayashi, Yuzo, Kodama, Satoshi, Kubo, Kensuke, Kubota, Marco, Lanzillotta, Markus, M Lerch, Yanying, Liu, Matthias, Löhr, Chiara, Marvisi, Ferran, Martinez-Valle, Eduardo, Martin-Nares, Yasufumi, Masaki, Shoko, Matsui, Ichiro, Mizushima, Ray, P Naden, Seiji, Nakamura, Jan, Nordeide, Kenji, Notohara, Kazuichi, Okazaki, Sergio, Paira, Cory, A Perugino, Jovan, Popovic, Manel, Ramos-Casals, James, Rosenbaum, Jay, Ryu, Yasuharu, Sato, Amita, Sharma, Takako, Saeki, Hiroshi, Sekiguchi, Nicolas, Schleinitz, Evgeniya, V Sokol, John, H Stone, James, R Stone, Hiroki, Takahashi, Naoki, Takahashi, Masayuki, Takahira, Yoshiya, Tanaka, Hisanori, Umehara, Vaglio, Augusto, Alejandra, Villamil, Yoko, Wada, Zachary, S Wallace, George, Webster, Kazunori, Yamada, Motohisa, Yamamoto, Joanne, Yi, Giuseppe, Zamboni, Yoh, Zen, Wen, Zhang, Wallace, Z, Zhang, Y, Perugino, Ca, Naden, R, Choi, Hk, Stone, Jh, for the ACR/EULAR IgG4-RD Classification Criteria, Committee, and DELLA TORRE, E

Subjects
Male, IgG4-related disease, cluster analysis, epidemiology, Adult, Americas, Aortitis, Asia, Asian Continental Ancestry Group, Continental Population Groups, Cross-Sectional Studies, Digestive System Diseases, Europe, Female, Humans, Immunoglobulin G, Immunoglobulin G4-Related Disease, Middle Aged, Mikulicz' Disease, Otorhinolaryngologic Diseases, Phenotype, Retroperitoneal Fibrosis, Disease, Retroperitoneal fibrosis, 0302 clinical medicine, Epidemiology, Immunology and Allergy, 030212 general & internal medicine, Latent class model, Cohort, medicine.symptom, medicine.medical_specialty, Immunology, General Biochemistry, Genetics and Molecular Biology, Article, 03 medical and health sciences, Rheumatology, Asian People, Internal medicine, medicine, 030203 arthritis & rheumatology, business.industry, Racial Groups, medicine.disease, Etiology, business
Abstract

ObjectiveIgG4-related disease (IgG4-RD) is a heterogeneous, multiorgan condition of unclear aetiology that can cause organ failure. Difficulty recognising IgG4-RD contributes to diagnostic delays. We sought to identify key IgG4-RD phenotypes.MethodsWe used two cross-sectional studies assembled by an international, multispecialty network of IgG4-RD specialists who submitted 765 cases to derive and replicate phenotypic groups. Phenotype groups of disease manifestations and key covariate distributions across the identified groups were measured using latent class analysis.ResultsIn the derivation cohort (n=493), we identified four groups with distinct manifestations: Group 1 (31%), Pancreato-Hepato-Biliary disease; Group 2 (24%), Retroperitoneal Fibrosis and/or Aortitis; Group 3 (24%), Head and Neck-Limited disease and Group 4 (22%), classic Mikulicz syndrome with systemic involvement. We replicated the identification of four phenotype groups in the replication cohort. Compared with cases in Groups 1, 2 and 4, respectively, cases in Group 3 were more likely to be female (OR 11.60 (95% CI 5.39 to 24.98), 10.35 (95% CI 4.63 to 23.15) and 9.24 (95% CI 3.53 to 24.20)) and Asian (OR 6.68 (95% CI 2.82 to 15.79), 7.43 (95% CI 2.97 to 18.56) and 6.27 (95% CI 2.27 to 17.29)). Cases in Group 4 had a higher median serum IgG4 concentration (1170 mg/dL) compared with groups 1–3 (316, 178 and 445 mg/dL, respectively, pConclusionWe identified four distinctive IgG4-RD phenotypes according to organ involvement. Being Asian or female may predispose individuals to head and neck-limited disease. These phenotypes serve as a framework for identifying IgG4-RD and studying its aetiology and optimal treatment.

Published
2019

8. The 2019 American College of Rheumatology/European League Against Rheumatism Classification Criteria for IgG4-Related Disease

Author

Wallace, Zachary S, Naden, Ray P, Chari, Suresh, Choi, Hyon, Della-Torre, Emanuel, Dicaire, Jean-Francois, Hart, Phil A, Inoue, Dai, Kawano, Mitsuhiro, Khosroshahi, Arezou, Kubota, Kensuke, Lanzillotta, Marco, Okazaki, Kazuichi, Perugino, Cory A, Sharma, Amita, Saeki, Takako, Sekiguchi, Hiroshi, Schleinitz, Nicolas, Stone, James R, Takahashi, Naoki, Umehara, Hisanori, Webster, George, Zen, Yoh, Stone, John H, Akamizu, T, Akiyama, M, Barra, L, Bateman, A, Blockmans, D, Brito-Zeron, P, Campochiaro, C, Carruthers, M, Chari, S, Chiba, T, Choi, H, Cornell, L, Culver, E, Darabian, S, Torre, Ed, Deshpande, V, Dong, L, Ebbo, M, Fernández-Codina, A, Ferry, Ja, Fragkoulis, G, Frost, F, Frulloni, L, Hart, Pa, Hernandez-Molina, G, Inoue, D, Ji, H, Keat, K, Kamisawa, T, Kawa, S, Kawano, M, Khosroshahi, A, Kobayashi, H, Kodama, Y, Kubo, S, Kubota, K, Lanzillotta, M, Leng, H, Lerch, M, Liu, Y, Liu, Z, Löhr, M, Martin-Nares, E, Martinez-Valle, F, Marvisi, C, Masaki, Y, Matsui, S, Mizushima, I, Naden, Rp, Nakamura, S, Nordeide, J, Notohara, K, Okazaki, K, Paira, S, Perugino, Ca, Popovic, J, Ramos-Casals, M, Rosenbaum, J, Ryu, J, Saeki, T, Sato, Y, Schleinitz, N, Sekiguchi, H, Sharma, A, Sokol, Ev, Stone, Jr, Stone, Jh, Sun, W, Takahashi, H, Takahashi, N, Takahira, M, Tanaka, Y, Umehara, H, Vaglio, A, Villamil, A, Wada, Y, Wallace, Zs, Webster, G, Yamada, K, Yamamoto, M, Yi, J, Yi, Y, Zamboni, G, Zen, Y, Zhang, W., Wallace, Z, Naden, Rp, Chari, S, Choi, H, DELLA TORRE, E, Dicaire, Jf, Hart, Pa, Inoue, D, Kawano, M, Khosroshahi, A, Kubota, K, Lanzillotta, M, Okazaki, K, Perugino, Ca, Sharma, A, Saeki, T, Sekiguchi, H, Schleinitz, N, Stone, Jr, Takahashi, N, Umehara, H, Webster, G, Zen, Y, Stone, Jh, and American College of Rheumatology/European League Against Rheumatism IgG4-Related Disease Classification Criteria Working, Group.

Subjects
Adult, Male, medicine.medical_specialty, Consensus, Aged, Diagnosis, Differential, Europe, Female, Humans, Immunoglobulin G4-Related Disease, Middle Aged, Reproducibility of Results, Rheumatology, Societies, Medical, United States, Immunology, MEDLINE, Disease, Internal medicine, Medical, parasitic diseases, Diagnosis, Immunology and Allergy, Medicine, business.industry, medicine.disease, Confidence interval, Test (assessment), Differential, IgG4-related disease, business, Societies, Rheumatism, Decision analysis
Abstract

Objective IgG4-related disease (IgG4-RD) can cause fibroinflammatory lesions in nearly any organ. Correlation among clinical, serologic, radiologic, and pathologic data is required for diagnosis. This work was undertaken to develop and validate an international set of classification criteria for IgG4-RD. Methods An international multispecialty group of 86 physicians was assembled by the American College of Rheumatology (ACR) and the European League Against Rheumatism (EULAR). Investigators used consensus exercises, existing literature, derivation and validation cohorts of 1,879 subjects (1,086 cases, 793 mimickers), and multicriterion decision analysis to identify, weight, and test potential classification criteria. Two independent validation cohorts were included. Results A 3-step classification process was developed. First, it must be demonstrated that a potential IgG4-RD case has involvement of at least 1 of 11 possible organs in a manner consistent with IgG4-RD. Second, exclusion criteria consisting of a total of 32 clinical, serologic, radiologic, and pathologic items must be applied; the presence of any of these criteria eliminates the patient from IgG4-RD classification. Third, 8 weighted inclusion criteria domains, addressing clinical findings, serologic results, radiology assessments, and pathology interpretations, are applied. In the first validation cohort, a threshold of 20 points had a specificity of 99.2% (95% confidence interval [95% CI] 97.2-99.8%) and a sensitivity of 85.5% (95% CI 81.9-88.5%). In the second, the specificity was 97.8% (95% CI 93.7-99.2%) and the sensitivity was 82.0% (95% CI 77.0-86.1%). The criteria were shown to have robust test characteristics over a wide range of thresholds. Conclusion ACR/EULAR classification criteria for IgG4-RD have been developed and validated in a large cohort of patients. These criteria demonstrate excellent test performance and should contribute substantially to future clinical, epidemiologic, and basic science investigations.

Published
2018

10. International Consensus Guidance Statement on the Management and Treatment of IgG4-Related Disease

Author

Khosroshahi A, Wallace ZS, Crowe JL, Akamizu T, Azumi A, Carruthers MN, Chari ST, DELLA TORRE E, Frulloni L, Goto H, Hart PA, Kamisawa T, Kawa S, Kawano M, Kim MH, Kodama Y, Kubota K, Lerch MM, Löhr M, Masaki Y, Matsui S, Mimori T, Nakamura S, Nakazawa T, Ohara H, Okazaki K, Ryu JH, Saeki T, Schleinitz N, Shimatsu A, Shimosegawa T, Takahashi H, Takahira M, Tanaka A, Topazian M, Umehara H, Webster GJ, Witzig TE, Yamamoto M, Zhang W, Chiba T, Stone JH, Second International Symposium on IgG4-Related Disease., Khosroshahi, A, Wallace, Z, Crowe, Jl, Akamizu, T, Azumi, A, Carruthers, Mn, Chari, St, DELLA TORRE, E, Frulloni, L, Goto, H, Hart, Pa, Kamisawa, T, Kawa, S, Kawano, M, Kim, Mh, Kodama, Y, Kubota, K, Lerch, Mm, Löhr, M, Masaki, Y, Matsui, S, Mimori, T, Nakamura, S, Nakazawa, T, Ohara, H, Okazaki, K, Ryu, Jh, Saeki, T, Schleinitz, N, Shimatsu, A, Shimosegawa, T, Takahashi, H, Takahira, M, Tanaka, A, Topazian, M, Umehara, H, Webster, Gj, Witzig, Te, Yamamoto, M, Zhang, W, Chiba, T, Stone, Jh, and Second International Symposium on IgG4-Related, Disease.

Published
2015

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