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1. Early disease and low baseline damage predict response to belimumab in patients with systemic lupus erythematosus

Author

Gatto, Mariele, Saccon, Francesca, Zen, Margherita, Regola, Francesca, Fredi, Micaela, Andreoli, Laura, Tincani, Angela, Urban, Maria Letizia, Emmi, Giacomo, Ceccarelli, Fulvia, Conti, Fabrizio, Bortoluzzi, Alessandra, Govoni, Marcello, Tani, Chiara, Mosca, Marta, Ubiali, Tania, Gerosa, Maria, Bozzolo, Enrica, Canti, Valentina, Cardinaletti, Paolo, Gabrielli, Armando, Tanti, Giacomo, Gremese, Elisa, De Marchi, Ginevra, De Vita, Salvatore, Fasano, Serena, Ciccia, Francesco, Pazzola, Giulia, Salvarani, Carlo, Negrini, Simone, Puppo, Francesco, Di Matteo, Andrea, De Angelis, Rossella, Orsolini, Giovanni, Rossini, Maurizio, Faggioli, Paola, Laria, Antonella, Piga, Matteo, Mathieu, Alessandro, Scarpato, Salvatore, Rossi, Francesca W, de Paulis, Amato, Brunetta, Enrico, Ceribelli, Angela, Selmi, Carlo, Prete, Marcella, Racanelli, Vito, Vacca, Angelo, Bartoloni, Elena, Gerli, Roberto, Larosa, Maddalena, Iaccarino, Luca, Doria, Andrea, Gatto, Mariele, Saccon, Francesca, Zen, Margherita, Regola, Francesca, Fredi, Micaela, Andreoli, Laura, Tincani, Angela, Urban, Maria Letizia, Emmi, Giacomo, Ceccarelli, Fulvia, Conti, Fabrizio, Bortoluzzi, Alessandra, Govoni, Marcello, Tani, Chiara, Mosca, Marta, Ubiali, Tania, Gerosa, Maria, Bozzolo, Enrica, Canti, Valentina, Cardinaletti, Paolo, Gabrielli, Armando, Tanti, Giacomo, Gremese, Elisa, De Marchi, Ginevra, De Vita, Salvatore, Fasano, Serena, Ciccia, Francesco, Pazzola, Giulia, Salvarani, Carlo, Negrini, Simone, Puppo, Francesco, Di Matteo, Andrea, De Angelis, Rossella, Orsolini, Giovanni, Rossini, Maurizio, Faggioli, Paola, Laria, Antonella, Piga, Matteo, Mathieu, Alessandro, Scarpato, Salvatore, Rossi, Francesca W, de Paulis, Amato, Brunetta, Enrico, Ceribelli, Angela, Selmi, Carlo, Prete, Marcella, Racanelli, Vito, Vacca, Angelo, Bartoloni, Elena, Gerli, Roberto, Larosa, Maddalena, Iaccarino, Luca, and Doria, Andrea

Subjects
remission, immune system diseases, LDA, SLE, skin and connective tissue diseases, belimumab, damage
Abstract

To investigate predictors of response, remission, low disease activity (LDA), damage and drug discontinuation in patients with systemic lupus erythematosus (SLE) treated with belimumab.

Published
2020

2. Influence of Antisynthetase Antibodies Specificities on Antisynthetase Syndrome Clinical Spectrum Time Course

Author

Cavagna, Lorenzo, Trallero-Araguás, Ernesto, Meloni, Federica, Cavazzana, Ilaria, Rojas-Serrano, Jorge, Feist, Eugen, Zanframundo, Giovanni, Morandi, Valentina, Meyer, Alain, Pereira da Silva, Jose Antonio, Matos Costa, Carlo Jorge, Molberg, Oyvind, Andersson, Helena, Codullo, Veronica, Mosca, Marta, Barsotti, Simone, Neri, Rossella, Scirè, Carlo, Govoni, Marcello, Furini, Federica, Lopez-Longo, Francisco Javier, Martinez-Barrio, Julia, Schneider, Udo, Lorenz, Hanns-Martin, Doria, Andrea, Ghirardello, Anna, Ortego-Centeno, Norberto, Confalonieri, Marco, Tomietto, Paola, Pipitone, Nicolò, Rodriguez Cambron, Ana Belen, Blázquez Cañamero, María Ángeles, Voll, Reinhard Edmund, Wendel, Sarah, Scarpato, Salvatore, Maurier, Francois, Limonta, Massimiliano, Colombelli, Paolo, Giannini, Margherita, Geny, Bernard, Arrigoni, Eugenio, Bravi, Elena, Migliorini, Paola, Mathieu, Alessandro, Piga, Matteo, Drott, Ulrich, Delbrueck, Christiane, Bauhammer, Jutta, Cagnotto, Giovanni, Vancheri, Carlo, Sambataro, Gianluca, De Langhe, Ellen, Sainaghi, Pier Paolo, Monti, Cristina, Gigli Berzolari, Francesca, Romano, Mariaeva, Bonella, Francesco, Specker, Christof, Schwarting, Andreas, Villa Blanco, Ignacio, Selmi, Carlo, Ceribelli, Angela, Nuno, Laura, Mera-Varela, Antonio, Perez Gomez, Nair, Fusaro, Enrico, Parisi, Simone, Sinigaglia, Luigi, Del Papa, Nicoletta, Benucci, Maurizio, Cimmino, Marco Amedeo, Riccieri, Valeria, Conti, Fabrizio, Sebastiani, Gian Domenico, Iuliano, Annamaria, Emmi, Giacomo, Cammelli, Daniele, Sebastiani, Marco, Manfredi, Andreina, Bachiller-Corral, Javier, Sifuentes Giraldo, Walter Alberto, Paolazzi, Giuseppe, Saketkoo, Lesley Ann, Giorgi, Roberto, Salaffi, Fausto, Cifrián, José Manuel, Caporali, Roberto, Locatelli, Francesco, Marchioni, Enrico, Pesci, Alberto, Dei, Giulia, Pozzi, Maria Rosa, Claudia, Lomater, Distler, Jorg, Knitza, Johannes, Schett, George, Iannone, Florenzo, Fornaro, Marco, Franceschini, Franco, Quartuccio, Luca, Gerli, Roberto, Bartoloni, Elena, Bellando Randone, Silvia, Zampogna, Giuseppe, Gonzalez Perez, Montserrat I., Mejia, Mayra, Vicente, Esther, Triantafyllias, Konstantinos, Lopez-Mejias, Raquel, Matucci-Cerinic, Marco, Selva-O'Callaghan, Albert, Castañeda, Santos, Montecucco, Carlomaurizio, González-Gay, Miguel A., Universitat Autònoma de Barcelona, Universidad de Cantabria, Cavagna, L, Trallero-Araguás, E, Meloni, F, Cavazzana, I, Rojas-Serrano, J, Feist, E, Zanframundo, G, Morandi, V, Meyer, A, Pereira da Silva, J, Matos Costa, C, Molberg, O, Andersson, H, Codullo, V, Mosca, M, Barsotti, S, Neri, R, Scirè, C, Govoni, M, Furini, F, Lopez-Longo, F, Martinez-Barrio, J, Schneider, U, Lorenz, H, Doria, A, Ghirardello, A, Ortego-Centeno, N, Confalonieri, M, Tomietto, P, Pipitone, N, Rodriguez Cambron, A, Blázquez Cañamero, M, Voll, R, Wendel, S, Scarpato, S, Maurier, F, Limonta, M, Colombelli, P, Giannini, M, Geny, B, Arrigoni, E, Bravi, E, Migliorini, P, Mathieu, A, Piga, M, Drott, U, Delbrueck, C, Bauhammer, J, Cagnotto, G, Vancheri, C, Sambataro, G, De Langhe, E, Sainaghi, P, Monti, C, Gigli Berzolari, F, Romano, M, Bonella, F, Specker, C, Schwarting, A, Villa Blanco, I, Selmi, C, Ceribelli, A, Nuno, L, Mera-Varela, A, Perez Gomez, N, Fusaro, E, Parisi, S, Sinigaglia, L, Del Papa, N, Benucci, M, Cimmino, M, Riccieri, V, Conti, F, Sebastiani, G, Iuliano, A, Emmi, G, Cammelli, D, Sebastiani, M, Manfredi, A, Bachiller-Corral, J, Sifuentes Giraldo, W, Paolazzi, G, Saketkoo, L, Giorgi, R, Salaffi, F, Cifrian, J, Caporali, R, Locatelli, F, Marchioni, E, Pesci, A, Dei, G, Pozzi, M, Claudia, L, Distler, J, Knitza, J, Schett, G, Iannone, F, Fornaro, M, Franceschini, F, Quartuccio, L, Gerli, R, Bartoloni, E, Bellando Randone, S, Zampogna, G, Gonzalez Perez, M, Mejia, M, Vicente, E, Triantafyllias, K, Lopez-Mejias, R, Matucci-Cerinic, M, Selva-O'Callaghan, A, Castañeda, S, Montecucco, C, and Gonzalez-Gay, M

Subjects
medicine.medical_specialty, antisynthetase antibodies, antisynthetase syndrome, arthritis, interstitial lung disease, myositis, Medizin, Arthritis, lcsh:Medicine, Antisynthetase syndrome, Interstitial lung disease, Article, NO, 03 medical and health sciences, 0302 clinical medicine, Internal medicine, medicine, antisynthetase antibodies, antisynthetase syndrome, arthritis, interstitial lung disease, myositis, ddc:610, Myositis, 030203 arthritis & rheumatology, Antisynthetase antibodies, biology, business.industry, lcsh:R, Autoantibody, General Medicine, medicine.disease, arthriti, 030228 respiratory system, Time course, Cohort, biology.protein, Antibody, business, antisynthetase antibodie
Abstract

Antisynthetase syndrome (ASSD) is a rare clinical condition that is characterized by the occurrence of a classic clinical triad, encompassing myositis, arthritis, and interstitial lung disease (ILD), along with specific autoantibodies that are addressed to different aminoacyl tRNA synthetases (ARS). Until now, it has been unknown whether the presence of a different ARS might affect the clinical presentation, evolution, and outcome of ASSD. In this study, we retrospectively recorded the time of onset, characteristics, clustering of triad findings, and survival of 828 ASSD patients (593 anti-Jo1, 95 anti-PL7, 84 anti-PL12, 38 anti-EJ, and 18 anti-OJ), referring to AENEAS (American and European NEtwork of Antisynthetase Syndrome) collaborative group's cohort. Comparisons were performed first between all ARS cases and then, in the case of significance, while using anti-Jo1 positive patients as the reference group. The characteristics of triad findings were similar and the onset mainly began with a single triad finding in all groups despite some differences in overall prevalence. The "ex-novo" occurrence of triad findings was only reduced in the anti-PL12-positive cohort, however, it occurred in a clinically relevant percentage of patients (30%). Moreover, survival was not influenced by the underlying anti-aminoacyl tRNA synthetase antibodies' positivity, which confirmed that antisynthetase syndrome is a heterogeneous condition and that antibody specificity only partially influences the clinical presentation and evolution of this condition. ispartof: JOURNAL OF CLINICAL MEDICINE vol:8 issue:11 ispartof: location:Switzerland status: published

Published
2019

3. EEcacy, Safety, and Predictor of Response to Belimumab in a Large Nationwide Cohort Study of Patients with Active Systemic Lupus Erythematosus

Author

Iaccarino, Luca, Saccon, Francesca, Mathieu, Alessandro, Piga, Matteo, Ceribelli, Angela, Selmi, Carlo, Cardinaletti, Paolo, Gabrielli, Armando, Matteo, Andrea Di, Angelis, Rossella De, Faggioli, Paola, Laria, Antonella, Fredi, Micaela, Regola, Francesca, Tincani, Angela, Andreoli, Laura, Pazzola, Giulia, Salvarani, Carlo, Puppo, Francesco, Magnani, Ottavia, Prete, Marcella, Gremese, Elisa, Gerosa, Maria, Ubiali, Tania, Bozzolo, Enrica, Canti, Valentina, Racanelli, Vito, Conti, Fabrizio, Ceccarelli, Fulvia, Bartoloni, Elena, Gerli, Roberto, Lobasso, Antonio, Paulis, Amato De, Marchi, Ginevra De, Vita, Salvatore De, Bortoluzzi, Alessandra, Govoni, Marcello, Benvenuti, Francesco, Zen, Margherita, Mosca, Marta, Tani, Chiara, Rossini, Maurizio, Orsolini, Giovanni, Scarpato, Salvatore, Ernico Brunetta, Zumbo, Aurora, Tanti, Giacomo, Doria, Andrea, Istituto Ortopedico, and Pini, Gaetano

Published
2018
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4. Anti-cyclic citrullinated peptide antibodies in systemic lupus erythematosus patients with articular involvement: a predictive marker for erosive disease?

Author

Taraborelli, M, Inverardi, F, FREDI - NON UTILIZZARE, Micaela, Ceribelli, Angela, Cavazzana, Ilaria, Tincani, Angela, Franceschini, F., and Fredi, Micaela

Subjects
Adult, Male, musculoskeletal diseases, lcsh:Internal medicine, Arthritis, lcsh:Medicine, Systemic Lupus Erythematosus, anti-cyclic citrullinated peptide antibodies, arthtritis, Autoantigens, Peptides, Cyclic, Rheumatology, Antibody Specificity, Predictive Value of Tests, immune system diseases, Hand Deformities, Acquired, Medicine, Rheumatoid factor, Humans, Lupus Erythematosus, Systemic, skin and connective tissue diseases, lcsh:RC31-1245, Autoantibodies, Retrospective Studies, Lupus erythematosus, Predictive marker, Systemic lupus erythematosus, biology, business.industry, Foot Deformities, Acquired, C-reactive protein, lcsh:R, medicine.disease, Prognosis, Predictive value of tests, Immunology, biology.protein, Female, Joints, Antibody, business
Abstract

A small number of systemic lupus erythematosus (SLE) patients develop an erosive disease. Some studies have suggested an association between anti-cyclic citrullinated (anti-CCP) antibodies and this pattern of arthritis, but their exact significance in SLE patients remains unclear. The aim of this study was to evaluate the prevalence of anti-CCP antibodies in SLE patients with different subsets of articular disease. Among 521 SLE patients followed in this center from 1976 to 2011, those with articular involvement (n=298) were selected to take part in the study. We searched for anti-CCP2 IgG antibodies in 198 patients using a commercial enzyme linked immunosorbent assay (Immunoscan RA, Eurodiagnostica). In 174 patients the results for rheumatoid factor (RF) by nephelometry were retrospectively collected. C reactive protein (CRP) was obtained from clinical records. Patients were classified into 3 groups: erosive, non-erosive deforming, non-erosive non-deforming arthritis. Results of the different tests were compared among the groups. Plt;0.05 was considered statistically significant. Anti-CCP antibodies were significantly associated with erosive disease. We also found that RF positivity and increased CRP were more frequent in erosive arthritis and erosive or non-deforming arthritis, respectively, than in non-erosive non-deforming arthritis. This study supports the evidence that anti-CCP antibodies could be a useful marker of erosive disease in SLE patients. Increase in RF and CRP could be an additional means of identifying lupus patients with arthritis at risk of a worse prognosis.

Published
2012

5. PROTEIN AND RNA IMMUNOPRECIPITATION FOR THE IDENTIFICATION OF SPECIFIC SERUM AUTOANTIBODIES IN SYSTEMIC AUTOIMMUNE RHEUMATIC DISEASES

Author

CERIBELLI, ANGELA

Abstract

Serum autoantibodies play a key role in systemic autoimmune rheumatic diseases for diagnostic, classification and prognostic purposes. Research of new autoantibodies has been very active in the last decade in rare connective tissue diseases such as systemic sclerosis and poly/dermatomyositis, with new biomarkers entering the clinical practice. Immunoprecipitation of protein and/or RNA components of the target autoantigens constitutes the gold standard method for the discovery of new autoantibodies in a screening setting but is considered time- and labor-intensive and, accordingly, is performed only in a few laboratories worldwide. As a result, alternative techniques such as ELISA and immunoblotting are often preferred for large-scale testing, despite the lack of standardization. The aims of the present project are (1) to set up protein- and RNA- immunoprecipitation in our laboratory and (2) to describe serum autoantibodies identified in our series of patients affected by systemic autoimmune rheumatic diseases. During the PhD program we were able to perform correctly protein-and RNA-immunoprecipitation in our laboratory as demonstrated by positive reference sera, and then by the identification of known but also new and rare autoantibodies, as represented by two new patterns immunoprecipitated in systemic sclerosis, corresponding to serum anti-hnRNP-L and anti-mitochondrial antibodies. In psoriatic arthritis we also analyzed the concentration of circulating levels of LL37, a recently established target of autoimmune response at the skin level, and we identified an increased production in a subset of patients. In conclusion, performing protein- and RNA-immunoprecipitation as a screening method in our laboratory allows a more complete and specific autoantibody analysis that cannot be performed by the commercial techniques available nowadays, and further analysis of the role of LL37 in psoriatic arthritis patients may help in the identification of a new biomarker in this condition.

Published
2016
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6. Testing for myositis specific autoantibodies: Comparison between line blot and immunoprecipitation assays in 57 myositis sera

Author

Cavazzana, Ilaria, Fredi, Micaela, Ceribelli, Angela, Mordenti, Cristina, Ferrari, Fabio, Carabellese, Nice, Tincani, Angela, Satoh, Minoru, and Franceschini, Franco

Subjects
0301 basic medicine, Adult, Male, Anti-Jo1, Anti-synthetase syndrome, Autoantibodies, Immunoprecipitation, Line blot, Myositis, Immunology, Immunology and Allergy, Pathology, medicine.medical_specialty, Interferon-Induced Helicase, IFIH1, Polymyositis, Sensitivity and Specificity, 03 medical and health sciences, 0302 clinical medicine, medicine, Humans, Retrospective Studies, 030203 arthritis & rheumatology, Adenosine Triphosphatases, Immunoassay, medicine.diagnostic_test, biology, business.industry, Autoantibody, Gold standard (test), Dermatomyositis, Middle Aged, medicine.disease, Molecular biology, Blot, DNA-Binding Proteins, 030104 developmental biology, Antibodies, Antinuclear, biology.protein, Female, Antibody, business, K562 Cells, Transcription Factors
Abstract

Objective To analyze the performance of a line blot assay for the identification of autoantibodies in sera of patients affected by myositis, compared with immunoprecipitation (IP) as gold standard. Methods 66 sera of patients with myositis (23 polymyositis, 8 anti-synthetase syndromes, 29 dermatomyositis and 6 overlap syndromes) were tested by commercial LB (Euroimmun, Lubeck, Germany); 57 sera were analyzed also by IP of K562 cell extract radiolabeled with 35 S-methionine. Inter-rater agreement was calculated with Cohen's k coefficient. Results Myositis-specific antibodies (MSA) were detected in 36/57 sera (63%) by IP and in 39/66 sera (59%) by LB. The most frequent MSA found by LB were anti-Jo1 and anti-Mi2 found in 15% (10/66) of sera, followed by anti-NXP2 and anti-SRP detected in 106% (7/66) of sera. Anti-TIF1gamma and anti-MDA5 were found in 6 (9%) and 5 sera (7.6%), respectively. A good agreement between methods was found only for anti-TIF1γ, anti-MDA5 and anti-NXP-2 antibodies, while a moderate agreement was estimated for anti-Mi2 and anti-EJ. By contrast, a high discordance rate for the detection of anti-Jo1 antibodies was evident (k: 0.3). Multiple positivity for MSA were found in 11/66 (17%) by LB and 0/57 by IP (p: 0001). Comparing the clinical features of these 11 sera, we found total discrepancies between assays in 3 sera (27.3%), a relative discrepancy due to the occurrence of one discordant autoantibody (not confirmed by IP) in 5 cases (45.5%) and a total discrepancy between LB and IP results, but with a relative concordance with clinical features were found in other 3 sera (27.3%). The semiquantitative results do not support the interpretation of the data. Conclusions The use of LB assay allowed the detection of new MSA, such as anti-MDA5, anti-MJ and anti-TIF1gamma antibodies, previously not found with routine methods. However, the high prevalence of multiple positivities and the high discondant rate of anti-Jo1 antibodies could create some misinterpretation of the results from the clinical point of view. These data should be confirmed by enlarging the number of myositis cases.

Published
2016

7. Anti-RNA polymerase III antibodies: A marker of systemic sclerosis with rapid onset and skin thickening progression

Author

Ilaria, Cavazzana, Angela, Ceribelli, Ceribelli, Angela, Paolo, Airo', Airo', Paolo, Stefania, Zingarelli, Zingarelli, Stefania, Angela, Tincani, Tincani, Angela, Franco, Franceschini, and Franceschini, Franco

Subjects
Male, Pathology, medicine.medical_specialty, Anti-nuclear antibody, Immunology, Rapid systemic sclerosis onset, Systemic Sclerosis, Disease, Scleroderma, RNA polymerase III, Antibody Specificity, medicine, Humans, Immunology and Allergy, skin and connective tissue diseases, Autoantibodies, Skin, Scleroderma, Systemic, integumentary system, biology, Anti-RNA polymerase III, Skin thickening, business.industry, Systemic, Autoantibody, RNA Polymerase III, Middle Aged, Anti-nuclear antibodies, medicine.disease, Raynaud's phenomenon, Skin thickening progression, Rapid onset, Disease Progression, biology.protein, Female, Biomarkers, Antibody, business
Abstract

Anti-RNA polymerase III antibodies (ARA) are a specific marker for Systemic Sclerosis (SSc), associated to severe disease with major organ and diffuse cutaneous involvement. In our series, ARA were found in 19 of 216 sera, in 15 cases as isolated antibodies' specificity, with a statistically negative association with other SSc-specific autoantibodies (p: 0.00003). The prevalence of ARA among 73 anticentromere and anti-topoisomerase I (topo I) negative sera, was 20.5%. Patients with isolated ARA had more rapid disease onset, defined as the interval from the appearance of Raynaud's phenomenon to the first symptom other than Raynaud's, than patients with isolated anti-topo I antibodies (median: 2 months vs 13 months; p: 0.0013). A rapid onset of SSc (within 6 months from Raynaud's phenomenon onset) was found in all patients with isolated ARA and only in 34% of those with anti-topo I (p

Published
2009

9. International cohort study of 73 anti-Ku-positive patients: association of p70/p80 anti-Ku antibodies with joint/bone features and differentiation of disease populations by using principal-components analysis

Author

Lakota, Katja, Thallinger, Gerhard, Sodin-Semrl, Snezna, Rozman, Blaz, Ambrozic, Ales, Tomsic, Matija, Praprotnik, Sonja, Cucnik, Sasa, Mrak-Poljsak, Katjusa, Ceribelli, Angela, Cavazzana, Ilaria, Franceschini, Franco, Vencovsky, Jiri, Czirjak, Laszlo, Varju, Cecilia, Steiner, Gunther, Aringer, Martin, Stamenkovic, Bojana, Distler, Oliver, and Matucci-Cerinic, Marco

Published
2012

25. Early and Late Response and Glucocorticoid-Sparing Effect of Belimumab in Patients with Systemic Lupus Erythematosus with Joint and Skin Manifestations: Results from the Belimumab in Real Life Setting Study—Joint and Skin (BeRLiSS-JS)

Author

Margherita Zen, Mariele Gatto, Roberto Depascale, Francesca Regola, Micaela Fredi, Laura Andreoli, Franco Franceschini, Maria Letizia Urban, Giacomo Emmi, Fulvia Ceccarelli, Fabrizio Conti, Alessandra Bortoluzzi, Marcello Govoni, Chiara Tani, Marta Mosca, Tania Ubiali, Maria Gerosa, Enrica P. Bozzolo, Valentina Canti, Paolo Cardinaletti, Armando Gabrielli, Giacomo Tanti, Elisa Gremese, Ginevra De Marchi, Salvatore De Vita, Serena Fasano, Francesco Ciccia, Giulia Pazzola, Carlo Salvarani, Simone Negrini, Andrea Di Matteo, Rossella De Angelis, Giovanni Orsolini, Maurizio Rossini, Paola Faggioli, Antonella Laria, Matteo Piga, Alberto Cauli, Salvatore Scarpato, Francesca Wanda Rossi, Amato De Paulis, Enrico Brunetta, Angela Ceribelli, Carlo Selmi, Marcella Prete, Vito Racanelli, Angelo Vacca, Elena Bartoloni, Roberto Gerli, Elisabetta Zanatta, Maddalena Larosa, Francesca Saccon, Andrea Doria, Luca Iaccarino, Zen, Margherita, Gatto, Mariele, Depascale, Roberto, Regola, Francesca, Fredi, Micaela, Andreoli, Laura, Franceschini, Franco, Letizia Urban, Maria, Emmi, Giacomo, Ceccarelli, Fulvia, Conti, Fabrizio, Bortoluzzi, Alessandra, Govoni, Marcello, Tani, Chiara, Mosca, Marta, Ubiali, Tania, Gerosa, Maria, Bozzolo, Enrica P., Canti, Valentina, Cardinaletti, Paolo, Gabrielli, Armando, Tanti, Giacomo, Gremese, Elisa, De Marchi, Ginevra, De Vita, Salvatore, Fasano, Serena, Ciccia, Francesco, Pazzola, Giulia, Salvarani, Carlo, Negrini, Simone, Di Matteo, Andrea, DE ANGELIS, Rossella, Orsolini, Giovanni, Rossini, Maurizio, Faggioli, Paola, Laria, Antonella, Piga, Matteo, Cauli, Alberto, Scarpato, Salvatore, Wanda Rossi, Francesca, De Paulis, Amato, Brunetta, Enrico, Ceribelli, Angela, Selmi, Carlo, Prete, Marcella, Racanelli, Vito, Vacca, Angelo, Bartoloni, Elena, Gerli, Roberto, Zanatta, Elisabetta, Larosa, Maddalena, Saccon, Francesca, Doria, Andrea, and Iaccarino, Luca

Subjects
disease activity score (DAS)-28, low disease activity, remission, systemic lupus erythematosus, belimumab, Cutaneous LE Area and Severity Index (CLASI), Medicine (miscellaneous)
Abstract

Aim. To assess the efficacy of belimumab in joint and skin manifestations in a nationwide cohort of patients with SLE. Methods. All patients with skin and joint involvement enrolled in the BeRLiSS cohort were considered. Belimumab (intravenous, 10 mg/kg) effectiveness in joint and skin manifestations was assessed by DAS28 and CLASI, respectively. Attainment and predictors of DAS28 remission (

Published
2023

26. Dose-Dependent Impairment of the Immune Response to the Moderna-1273 mRNA Vaccine by Mycophenolate Mofetil in Patients with Rheumatic and Autoimmune Liver Diseases

Author

Maria De Santis, Francesca Motta, Natasa Isailovic, Massimo Clementi, Elena Criscuolo, Nicola Clementi, Antonio Tonutti, Stefano Rodolfi, Elisa Barone, Francesca Colapietro, Angela Ceribelli, Matteo Vecellio, Nicoletta Luciano, Giacomo Guidelli, Marta Caprioli, Clara Rezk, Lorenzo Canziani, Elena Azzolini, Luca Germagnoli, Nicasio Mancini, Ana Lleo, Carlo Selmi, De Santis, Maria, Motta, Francesca, Isailovic, Natasa, Clementi, Massimo, Criscuolo, Elena, Clementi, Nicola, Tonutti, Antonio, Rodolfi, Stefano, Barone, Elisa, Colapietro, Francesca, Ceribelli, Angela, Vecellio, Matteo, Luciano, Nicoletta, Guidelli, Giacomo, Caprioli, Marta, Rezk, Clara, Canziani, Lorenzo, Azzolini, Elena, Germagnoli, Luca, Mancini, Nicasio, Lleo, Ana, and Selmi, Carlo

Subjects
Pharmacology, Infectious Diseases, antirheumatic agents, Drug Discovery, Immunology, mycophenolate mofetil, COVID-19, Pharmacology (medical), autoimmune diseases, vaccination
Abstract

The purpose of this study was to evaluate the efficacy and safety of the Moderna-1273 mRNA vaccine for SARS-CoV-2 in patients with immune-mediated diseases under different treatments. Anti-trimeric spike protein antibodies were tested in 287 patients with rheumatic or autoimmune diseases (10% receiving mycophenolate mofetil, 15% low-dose glucocorticoids, 21% methotrexate, and 58% biologic/targeted synthetic drugs) at baseline and in 219 (76%) 4 weeks after the second Moderna-1273 mRNA vaccine dose. Family members or caretakers were enrolled as the controls. The neutralizing serum activity against SARS-CoV-2-G614, alpha, and beta variants in vitro and the cytotoxic T cell response to SARS-CoV-2 peptides were determined in a subgroup of patients and controls. Anti-SARS-CoV-2 antibody development, i.e., seroconversion, was observed in 69% of the mycophenolate-treated patients compared to 100% of both the patients taking other treatments and the controls (p < 0.0001). A dose-dependent impairment of the humoral response was observed in the mycophenolate-treated patients. A daily dose of >1 g at vaccination was a significant risk factor for non-seroconversion (ROC AUC 0.89, 95% CI 0.80–98, p < 0.0001). Moreover, in the seroconverted patients, a daily dose of >1 g of mycophenolate was associated with significantly lower anti-SARS-CoV-2 antibody titers, showing slightly reduced neutralizing serum activity but a comparable cytotoxic response compared to other immunosuppressants. In non-seroconverted patients treated with mycophenolate at a daily dose of >1 g, the cytotoxic activity elicited by viral peptides was also impaired. Mycophenolate treatment affects the Moderna-1273 mRNA vaccine immunogenicity in a dose-dependent manner, independent of rheumatological disease.

Published
2022

27. A case of psoriatic arthritis triggered by SARS-CoV-2 infection

Author

Maria De Santis, Carlo Selmi, Francesca Motta, M. Caprioli, Nicasio Mancini, Giacomo Maria Guidelli, Matteo Vecellio, Nicoletta Luciano, L. Novelli, Natasa Isailovic, Angela Ceribelli, Massimo Clementi, Nicola Clementi, Novelli, Lucia, Motta, Francesca, Ceribelli, Angela, Guidelli, Giacomo M, Luciano, Nicoletta, Isailovic, Natasa, Vecellio, Matteo, Caprioli, Marta, Clementi, Nicola, Clementi, Massimo, Mancini, Nicasio, Selmi, Carlo, and De Santis, Maria

Subjects
2019-20 coronavirus outbreak, Coronavirus disease 2019 (COVID-19), SARS-CoV-2, business.industry, Severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), Arthritis, Psoriatic, COVID-19, Arthritis, medicine.disease, Virology, Letter to the Editor (Case report), Psoriatic arthritis, Rheumatology, medicine, Humans, Pharmacology (medical), business, AcademicSubjects/MED00360
Published
2020

28. Racial differences in systemic sclerosis disease presentation: A European Scleroderma Trials and Research group study

Author

Jaeger, Veronika K, Tikly, Mohammed, Dong, Xu, Siegert, Elise, Hachulla, Eric, Airò, Paolo, Valentini, Gabriele, Matucci Cerinic, Marco, Distler, Oliver, Cozzi, Franco, Carreira, Patricia, Allanore, Yannick, Müller-Ladner, Ulf, Ananieva, Lidia P, Balbir-Gurman, Alexandra, Distler, Jörg H W, Czirják, Laszlo, Mengtao, Li, Henes, Jörg, Jimenez, Sergio A, Smith, Vanessa, Damjanov, Nemanja, Denton, Christopher P, Delgaldo, Francesco, Saketkoo, Lesley Ann, Walker, Ulrich, A, Randone, Sb, Bannert, B, Iannone, F, Maurer, B, Jordan, S, Dobrota, R, Becker, M, Mihai, C, Becvarare, R, Tomčík, M, Bielecka, Ok, Gindzienska-Sieskiewicz, E, Karaszewska, K, Cutolo, M, Pizzorni, C, Paolino, S, Sulli, A, Ruaro, B, Alessandri, E, Riccardi, A, Giacco, V, Messitini, V, Irace, R, Kedor, C, Casteleyn, V, Hilger, J, Hoeppner, J, Rednic, S, Szabo, I, Petcu, A, Avouac, J, Camelia, F, Desbas, C, Vlachoyiannopoulos, P, Montecucco, C, Caporali, R, Cavagna, L, Stork, J, Inanc, M, Joven, Be, Novak, S, Anic, F, Varju, C, Minier, T, Chizzolini, C, Allai, D, Kucharz, Ej, Kotulska, A, Kopec-Medrek, M, Widuchowska, M, Dolnicar, As, Coleiro, B, Gabrielli, A, Manfredi, L, Benfaremo, D, Ferrarini, A, Bancel, Df, Hij, A, Lansiaux, P, Lazzaroni, Mg, Hesselstrand, R, Wuttge, D, Andréasson, R, Martinovic, D, Bozic, I, Radic, M, Braun-Moscovici, Y, Monaco, Al, Furini, F, Hunzelmann, N, Moinzadeh, P, Pellerito, R, Caimmi, C, Bertoldo, E, Morovic-Vergles, J, Culo, Im, Pecher, Ac, Santamaria, Vo, Heitmann, S, Codagnone, M, Pflugfelder, J, Krasowska, D, Michalska-Jakubus, M, Seidel, M, Hasler, P, Kretschmar, S, Kohm, M, Bajocchi, G, Salvador, Mj, Silva, Japd, Stamenkovic, B, Stankovic, A, Selmi, Cf, Santis, M, Ceribelli, A, Garzanova, L, Koneva, O, Starovoytova, M, Herrick, A, Puppo, F, Negrini, S, Murdaca, G, Engelhart, M, Szücs, G, Szamosi, S, de la Puente, C, Grande, Cs, Villanueva, Mjg, Midtvedt, Sø, Hoffmann-Vold, Am, Launay, D, Sobanski, V, Riccieri, V, Vasile, M, Ionescu, Rm, Opris, D, Sha, A, Woods, A, Gheorghiu, Am, Bojinca, M, Sunderkötter, C, Ehrchen, J, Ingegnoli, F, Mouthon, L, Dunogue, B, Chaigne, B, Legendre, P, Cantatore, Fp, Corrado, A, Ullman, S, Iversen, L, von Mühlen CA, Pozzi, Mr, Eyerich, K, Lauffer, F, Wiland, P, Szmyrka-Kaczmarek, M, Sokolik, R, Morgiel, E, Madej, M, Vanthuyne, M, Frédéric, H, Alegre-Sancho, Jj, Aringer, M, Herrmann, K, Günther, C, Westhovens, R, Langhe, E, Lenaerts, J, Anic, B, Baresic, M, Mayer, M, Üprus, M, Otsa, K, Yavuz, S, Granel, B, Radominski, Sc, De, C, Müller, S, Azevedo, Vf, Mendoza, F, Busquets, J, Popa, S, Agachi, S, Zenone, T, Pileckyte, M, Stebbings, S, Mathieu, A, Vacca, A, Sampaio-Barros, Pd, Stamp, L, Solanki, K, Silva, C, Schollum, J, Barns-Graham, H, Veale, D, O'Rourke, M, Loyo, E, Tineo, C, Paulino, G, Mohamed, Waaa, Rosato, E, Gigante, A, Oksel, F, Yargucu, F, Tanaseanu, Cm, Popescu, M, Dumitrascu, A, Tiglea, I, Foti, R, Visalli, E, Benenati, A, Amato, G, Ancuta, C, Villiger, P, Adler, S, Fröhlich, J, Kayser, C, Eduardo, Al, Fathi, N, Alii, S, Ahmed, M, Hasaneen, S, Hakeem, Ee, de la PG, Lefebvre, P, Martin, Jjg, Sibilia, J, Chatelus, E, Gottenberg, Je, Chifflot, H, Litinsky, I, Galdo, Fd, Abignano, G, Eng, S, Seskute, G, Butrimiene, I, Rugiene, R, Karpec, D, Pascal, M, Kerzberg, E, Bianchi, W, Bianchi, Bv, Bianchi, Dv, Barcellos, Y, Castellví, I, Millan, M, Limonta, M, Rimar, D, Rosner, I, Slobodin, G, Couto, M, Spertini, F, Ribi, C, Buss, G, Marcoccia, A, Bondanini, F, Ciani, A, Kahl, S, Hsu, Vm, Martin, T, Poindron, V, Meghit, K, Moiseev, S, Novikov, P, Chung, L, Kolstad, K, Stark, M, Schmeiser, T, Thiele, A, Majewski, D, Zdrojewski, Z, Zaneta, S, Wierzba, K, Martínez-Barrio, J, López-Longo, Fj, Bernardino, V, Moraes-Fontes, Mf, Rodrigues, Ac, Riemekasten, G, Sommerlatte, S, Jendreck, S, Arnold, S, Levy, Y, Rezus, E, Cardoneanu, A, Burlui, Am, Pamuk, On, Puttini, Ps, Talotta, R, Bongiovanni, S, Poormoghim, H, Andalib, E, Almasi, S, Kötter, I, Krusche, M, Cuomo, G, Danzo, F, Masini, F, Gaches, F, Michaud, M, Cartos, F, Belloli, L, Casu, C, Sfikakis, P, Tektonidou, M, Furst, D, Feldman, Gr, Ramazan, Am, Nurmambet, E, Miroto, A, Suta, C, Andronache, I, Huizinga, Twj, de Vries-Bouwstra, J., Chizzolini, Carlo, Jaeger, Veronika K, Tikly, Mohammed, Xu, Dong, Siegert, Elise, Hachulla, Eric, Airò, Paolo, Valentini, Gabriele, Matucci Cerinic, Marco, Distler, Oliver, Cozzi, Franco, Carreira, Patricia, Allanore, Yannick, Müller-Ladner, Ulf, Ananieva, Lidia P, Balbir-Gurman, Alexandra, Distler, Jörg H W, Czirják, Laszlo, Li, Mengtao, Henes, Jörg, Jimenez, Sergio A, Smith, Vanessa, Damjanov, Nemanja, Denton, Christopher P, Delgaldo, Francesco, Saketkoo, Lesley Ann, Walker, Ulrich A, University of Zurich, Cerinic, Marco Matucci, Walker Ulrich, A, Randone, Silvia Bellando, Bannert, Bettina, Iannone, Florenzoaa, Maurer, Brittaab, Jordan, Suzanaab, Dobrota, Rucsandraab, Becker, Mikeab, Mihai, Carinaa, Becvarare, Radima, Tomcik, Michala, Bielecka, Otylia Kowala, Gindzienska-Sieskiewicz, Ewaa, Karaszewska, Katarzynaa, Cutolo, Maurizioa, Pizzorni, Carmena, Paolino, Sabrinaae, Sulli, Albertoa, Ruaro, Barbara, Alessandri, Elisa, Riccardi, Antonella, Giacco, Veronica, Messitini, Valentina, Irace, Rosaria, Kedor, Claudia, Casteleyn, Vincent, Hilger, Julia, Hoeppner, Jakob, Rednic, Simona, Szabo, Iulia, Petcu, Ana, Avouac, Jérome, Camelia, Frantz, Desbas, Carole, Vlachoyiannopoulos, Panayioti, Montecucco, Carlo Maurizio, Caporali, Roberto, Cavagna, Lorenzo, Stork, Jiri, Inanc, Murat, Joven, Beatriz E., Novak, Srdan, Anic, Felina, Varju, Cecilia, Minier, Tunde, Allai, Daniela, Kucharz, Eugene J., Kotulska, Anna, Kopec-Medrek, Magdalena, Widuchowska, Malgorzata, Dolnicar, Alenka Sipek, Coleiro, Bernard, Gabrielli, Armando, Manfredi, Lucia, Benfaremo, Devi, Ferrarini, Alessia, Bancel, Dominique Farge, Hij, Adrian, Lazzaroni, Maria Grazia, Hesselstrand, Roger, Wuttge, Dirk, Andréasson, Kristofer, Martinovic, Duska, Bozic, Ivona, Radic, Mislav, Braun-Moscovici, Yolanda, Monaco, Andrea Lo, Furini, Federica, Hunzelmann, Nicola, Moinzadeh, Pia, Pellerito, Raffaele, Caimmi, Cristian, Bertoldo, Eugenia, Morovic-Vergles, Jadranka, Culo, Ivana Melanie, Pecher, Ann-Christian, Santamaria, Vera Ortiz, Heitmann, Stefan, Codagnone, Medeleine, Pflugfelder, Johanne, Krasowska, Dorota, Michalska-Jakubus, Malgorzata, Seidel, Matthia, Hasler, Paul, Kretschmar, Samuel, Kohm, Michaela, Bajocchi, Gianluigi, Salvador, Maria João, Da Silva, JoséAntonio Pereira, Stamenkovic, Bojana, Stankovic, Aleksandra, Selmi, Carlo Francesco, De Santis, Maria, Ceribelli, Angela, Garzanova, Ludmila, Koneva, Olga, Starovoytova, Maya, Herrick, Ariane, Puppo, Francesco, Negrini, Simone, Murdaca, Giuseppe, Engelhart, Merete, Szücs, Gabriela, Szamosi, Szilvia, De La Puente, Carlo, Grande, Cristina Sobrino, Villanueva, Maria Jesus Garcia, Midtve, Øyvindbw, Hoffmann-Vold, Anna-Mariabw, Launay, Davidbx, Sobanski, Vincentbx, Riccieri, Valeriaby, Vasile, Massimilianoby, Stefantoni, Katia, Ionescu, Ruxandra Maria, Opris, Daniela, Sha, Ami, Woods, Adrianne, Gheorghiu, Ana Maria, Bojinca, Mihai, Sunderkötter, Cord, Ehrchen, Jan, Ingegnoli, Francesca, Mouthon, Luc, Dunogue, Bertrand, Chaigne, Benjamin, Legendre, Paul, Cantatore, Francesco Paolo, Corrado, Ada, Ullman, Susanne, Iversen, Line, Von Mühlen, Carlos Alberto, Pozzi, Maria Rosa, Eyerich, Kilian, Lauffer, Felix, Wiland, Piotr, Szmyrka-Kaczmarek, Magdalena, Sokolik, Renata, Morgiel, Ewa, Madej, Marta, Vanthuyne, Marie, Frédéric, Houssiau, Alegre-Sancho, Juan Jose, Aringer, Martin, Herrmann, Kristine, Günther, Claudia, Westhovens, Rene, De Langhe, Ellen, Lenaerts, Jan, Anic, Branimir, Baresic, Marko, Mayer, Miroslav, Üprus, Maria, Otsa, Kati, Yavuz, Sule, Granel, Brigitte, Radominski, Sebastião Cezar, De Souza Müller, Carolina, Feijóazevedo, Valderílio, Mendoza, Fabian, Busquets, Joanna, Popa, Sergei, Agachi, Svetlana, Zenone, Thierry, Pileckyte, Margarita, Stebbings, Simon, Jordan, Sarah, Mathieu, Alessandro, Vacca, Alessandra, Sampaio-Barros, Percival D., Stamp, Lisa, Solanki, Kamal, Silva, Cherumi, Schollum, Joanne, Barns-Graham, Helen, Veale, Dougla, O'Rourke, Marie, Loyo, Esthela, Tineo, Carmen, Paulino, Glenny, Mohamed, Walid Ahmed Abdel Atty, Rosato, Edoardo, Gigante, Antonietta, Oksel, Fahrettin, Yargucu, Figen, Tanaseanu, Cristina-Mihaela, Popescu, Monica, Dumitrascu, Alina, Tiglea, Isabela, Foti, Rosario, Visalli, Elisa, Benenati, Alessia, Amato, Giorgio, Ancuta, Codrina, Villiger, Peter, Adler, Sabine, Fröhlich, Johanne, Kayser, Cristiane, Eduardo, Andrade Lui, Fathi, Nihal, Alii, Safa, Ahmed, Marrow, Hasaneen, Samar, El Hakeem, Eman, De La Peña Lefebvre, Paloma García, Martin, Jorge Juan Gonzalez, Sibilia, Jean, Chatelus, Emmanuel, Gottenberg, Jacques Eric, Chifflot, Hélène, Litinsky, Ira, Del Galdo, Francesco, Abignano, Giuseppina, Eng, Sookho, Seskute, Goda, Butrimiene, Irena, Rugiene, Rita, Karpec, Diana, Pascal, Melanie, Kerzberg, Eduardo, Bianchi, Washington, Bianchi, Breno Valdetaro, Bianchi, Dante Valdetaro, Barcellos, Yeda, Castellví, Ivan, Millan, Milena, Limonta, Massimiliano, Rimar, Doron, Rosner, Itzhak, Slobodin, Gleb, Couto, Maura, Spertini, Françoi, Ribi, Camillo, Buss, Guillaume, Marcoccia, Antonella, Bondanini, Francesco, Ciani, Aldo, Kahl, Sarah, Hsu, Vivien M., Martin, Thierry, Poindron, Vincent, Meghit, Kilifa, Moiseev, Sergey, Novikov, Pavel, Chung, Lori, Kolstad, Kathleen, Stark, Marianna, Schmeiser, Tim, Thiele, Astrid, Majewski, Dominik, Zdrojewski, Zbigniew, Zaneta, Smolenska, Wierzba, Karol, Martínez-Barrio, Julia, López-Longo, Francisco Javier, Bernardino, Vera, Moraes-Fontes, Maria Francisca, Rodrigues, Ana Catarina, Riemekasten, Gabriela, Sommerlatte, Sabine, Jendreck, Sebastian, Arnold, Sabrina, Levy, Yair, Rezus, Elena, Cardoneanu, Anca, Burlui, Alexandra Maria, Pamuk, Omer Nuri, Puttini, Piercarlo Sarzi, Talotta, Rossella, Bongiovanni, Sara, Poormoghim, Hadi, Andalib, Elham, Almasi, Simin, Kötter, Ina, Krusche, Matrin, Cuomo, Giovanna, Danzo, Fiammetta, Masini, Francesco, Gaches, Franci, Michaud, Martin, Cartos, Florian, Belloli, Laura, Casu, Cinzia, Sfikakis, Petro, Tektonidou, Maria, Furst, Daniel, Feldman, Gary R., Ramazan, Ana-Maria, Nurmambet, Emel, Miroto, Amalia, Suta, Cristina, Andronache, Iulia, Huizinga, Tom W. J., De Vries-Bouwstra, Jeska, and Walker, Ulrich A.

Subjects
Male, Vital capacity, Organ manifestations, systemic sclerosis, Type I, race difference, Systemic scleroderma, Gastroenterology, Scleroderma, immunology, 0302 clinical medicine, Diffusing capacity, middle aged, pulmonary hypertension, Medicine, Pharmacology (medical), 030212 general & internal medicine, organ manifestations, races, skin and connective tissue diseases, Lung, race, pathophysiology, African Continental Ancestry Group, ddc:616, integumentary system, disease course, Hazard ratio, Races, 10051 Rheumatology Clinic and Institute of Physical Medicine, Pulmonary, Middle Aged, Blacks, cohort analysis, Autoantibodie, 3. Good health, Asians, female, priority journal, DNA Topoisomerases, Type I, Black, centromere, Cohort, Hypertension, organ manifestation, Systemic sclerosis, Female, systemic sclerosi, Human, Adult, Asian Continental Ancestry Group, medicine.medical_specialty, Hypertension, Pulmonary, European Continental Ancestry Group, Black People, 610 Medicine & health, complication, Caucasian, White People, Article, lung, 03 medical and health sciences, Black person, Rheumatology, Asian People, forced vital capacity, Internal medicine, geographic distribution, Humans, controlled study, human, DNA topoisomerase, Aged, Autoantibodies, 030203 arthritis & rheumatology, Scleroderma, Systemic, Asian, business.industry, Whites, Systemic, Odds ratio, medicine.disease, Pulmonary hypertension, major clinical study, mortality, clinical feature, business, DNA Topoisomerases, autoantibody
Abstract

Objectives Racial factors play a significant role in SSc. We evaluated differences in SSc presentations between white patients (WP), Asian patients (AP) and black patients (BP) and analysed the effects of geographical locations. Methods SSc characteristics of patients from the EUSTAR cohort were cross-sectionally compared across racial groups using survival and multiple logistic regression analyses. Results The study included 9162 WP, 341 AP and 181 BP. AP developed the first non-RP feature faster than WP but slower than BP. AP were less frequently anti-centromere (ACA; odds ratio (OR) = 0.4, P < 0.001) and more frequently anti-topoisomerase-I autoantibodies (ATA) positive (OR = 1.2, P = 0.068), while BP were less likely to be ACA and ATA positive than were WP [OR(ACA) = 0.3, P < 0.001; OR(ATA) = 0.5, P = 0.020]. AP had less often (OR = 0.7, P = 0.06) and BP more often (OR = 2.7, P < 0.001) diffuse skin involvement than had WP. AP and BP were more likely to have pulmonary hypertension [OR(AP) = 2.6, P < 0.001; OR(BP) = 2.7, P = 0.03 vs WP] and a reduced forced vital capacity [OR(AP) = 2.5, P < 0.001; OR(BP) = 2.4, P < 0.004] than were WP. AP more often had an impaired diffusing capacity of the lung than had BP and WP [OR(AP vs BP) = 1.9, P = 0.038; OR(AP vs WP) = 2.4, P < 0.001]. After RP onset, AP and BP had a higher hazard to die than had WP [hazard ratio (HR) (AP) = 1.6, P = 0.011; HR(BP) = 2.1, P < 0.001]. Conclusion Compared with WP, and mostly independent of geographical location, AP have a faster and earlier disease onset with high prevalences of ATA, pulmonary hypertension and forced vital capacity impairment and higher mortality. BP had the fastest disease onset, a high prevalence of diffuse skin involvement and nominally the highest mortality.

Published
2020

29. PFAPA syndrome and Behçet's disease: a comparison of two medical entities based on the clinical interviews performed by three different specialists

Author

Francesco Caso, Donato Rigante, Carlo Selmi, Antonio Vitale, Marco Cattalini, Giovanni Lapadula, Luisa Costa, Luca Cantarini, Giuseppe Lopalco, Angela Ceribelli, Mauro Galeazzi, Enrico Brunetta, Florenzo Iannone, Giulia Bersani, Laura Martin Nieves, Cantarini, Luca, Vitale, Antonio, Bersani, Giulia, Nieves, Laura Martin, Cattalini, Marco, Lopalco, Giuseppe, Caso, Francesco, Costa, Luisa, Iannone, Florenzo, Lapadula, Giovanni, Galeazzi, Mauro, Ceribelli, Angela, Brunetta, Enrico, Selmi, Carlo, and Rigante, Donato

Subjects
0301 basic medicine, Pediatrics, Behcet's disease, Disease, Behçet’s disease, 0302 clinical medicine, Stomatitis, Pediatric, Behçet's disease, Behcet Syndrome, Pharyngitis, General Medicine, Syndrome, Middle Aged, Settore MED/38 - PEDIATRIA GENERALE E SPECIALISTICA, Italy, Recurrent fever, Autoinflammation, medicine.symptom, Human, Lymphadeniti, Adult, medicine.medical_specialty, PFAPA syndrome, Adolescent, Fever, Interviews as Topic, 03 medical and health sciences, Young Adult, Rheumatology, Lymphadenitis, medicine, Internal Medicine, Humans, Medical history, Symptom onset, 030203 arthritis & rheumatology, business.industry, medicine.disease, Stomatiti, Interleukin-1, stomatognathic diseases, 030104 developmental biology, Pharyngiti, Physical therapy, business
Abstract

The pediatric syndrome characterized by periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis (PFAPA) and adult Behçet's disease share some clinical manifestations and are both polygenic autoinflammatory disorders with interleukin-1β showing to play a pivotal role. However, the diagnosis is mostly clinical and we hypothesize that specific criteria may be addressed differently by different physicians. To determine the diagnostic variability, we compared the answers of 80 patients with a definite diagnosis of Behçet's disease (age 42.1 ± 13.7 years) obtained by separate telephone interviews conducted by a rheumatologist, a pediatrician, and an internist working largely in the field of autoinflammatory disorders. Questions were related to the age of symptom onset, the occurrence of recurrent fevers during childhood, and the association with oral aphthosis, cervical adenitis and/or pharyngitis, previous treatments, possible growth impairment, the time lapse between PFAPA-like symptoms and the onset of Behçet's disease, and the occurrence of Behçet-related manifestation during childhood. The rheumatologist identified 30 % of patients with Behçet's disease fulfilling PFAPA syndrome diagnostic criteria, compared to the pediatrician and the internist identifying 10 and 7.5 %, respectively. Most of the patients suffered from recurrent oral aphthosis in childhood also without fever (50, 39, and 48 % with each interviewer), yet no patient fulfilled the Behçet's disease diagnostic criteria. Our data suggest that physician awareness and expertise are central to the diagnosis of autoinflammatory disorders through an accurate collection of the medical history.

Published
2014

30. The clinical phenotype of Systemic Sclerosis patients with anti-PM/Scl antibodies: results from the EUSTAR cohort

Author

Vanessa Smith, Ellen De Langhe, Elisabetta Zanatta, Jorge Rojas-Serrano, Christopher P. Denton, Roger Hesselstrand, Lorenzo Cavagna, Franco Franceschini, Montserrat-Ixchel Gonzalez-Perez, Angela Ceribelli, Konstantinos Triantafyllias, Marie-Elise Truchetet, Federica Furini, Giacomo De Luca, Alessio Furloni, Francesco Del Galdo, Yannick Allanore, Ilaria Cavazzana, Hadi Poormoghim, Corrado Campochiaro, G. Abignano, M. G. Lazzaroni, Francesca Ingegnoli, Paolo Airò, Silvia Bellando-Randone, Emiliano Marasco, Eugenia Bertoldo, Enrico Colombo, Eric Hachulla, S. Zingarelli, Oliver Distler, Jeska DeVries-Bouwstra, Simone Barsotti, Lazzaroni, Maria-Grazia, Marasco, Emiliano, Campochiaro, Corrado, DeVries-Bouwstra, Jeska, Gonzalez-Perez, Montserrat-Ixchel, Rojas-Serrano, Jorge, Hachulla, Eric, Zanatta, Elisabetta, Barsotti, Simone, Furini, Federica, Triantafyllias, Konstantino, Abignano, Giuseppina, Truchetet, Marie-Elise, De Luca, Giacomo, De Langhe, Ellen, Hesselstrand, Roger, Ingegnoli, Francesca, Bertoldo, Eugenia, Smith, Vanessa, Bellando-Randone, Silvia, Poormoghim, Hadi, Colombo, Enrico, Ceribelli, Angela, Furloni, Alessio, Zingarelli, Stefania, Cavazzana, Ilaria, Franceschini, Franco, Del Galdo, Francesco, Denton, Christopher P, Cavagna, Lorenzo, Distler, Oliver, Allanore, Yannick, and Airò, Paolo

Subjects
Adult, Male, Myositis and muscle disease, medicine.medical_specialty, Scleroderma Renal Crisis, Laboratory Diagnosis, Systemic scleroderma, behavioral disciplines and activities, Gastroenterology, Scleroderma, Rheumatology, Calcinosis, Internal medicine, Scleroderma and related disorders, Myositis and muscle disease, Autoantigens and Autoantibodies, Biomarkers, Laboratory Diagnosis, medicine, Humans, Pharmacology (medical), autoantigens and autoantibodies, biomarkers, laboratory diagnosis, myositis and muscle disease, scleroderma and related disorders, Autoantibodies, Europe, Exoribonucleases, Exosome Multienzyme Ribonuclease Complex, Female, Middle Aged, Phenotype, Retrospective Studies, Scleroderma, Systemic, Registries, skin and connective tissue diseases, integumentary system, business.industry, Systemic, Autoantigens and Autoantibodies, Autoantibody, Interstitial lung disease, Cancer, Dermatomyositis, medicine.disease, Scleroderma and related disorders, Cohort, business, Biomarkers
Abstract

Objective To evaluate clinical associations of anti-PM/Scl antibodies in patients with SSc in a multicentre international cohort, with particular focus on unresolved issues, including scleroderma renal crisis (RC), malignancies, and functional outcome of interstitial lung disease (ILD). Methods (1) Analysis of SSc patients from the EUSTAR database: 144 anti-PM/Scl+ without SSc-specific autoantibodies were compared with 7202 anti-PM/Scl−, and then to 155 anti-Pm/Scl+ with SSc-specific antibodies. (2) Case–control study: additional data were collected for 165 anti-PM/Scl+ SSc patients (85 from the EUSTAR registry) and compared with 257 anti-PM/Scl− SSc controls, matched for sex, cutaneous subset, disease duration and age at SSc onset. Results Patients with isolated anti-PM/Scl+, as compared with anti-Pm/Scl−, had higher frequency of muscle involvement, ILD, calcinosis and cutaneous signs of DM, but similar frequency of SRC and malignancies (either synchronous with SSc onset or not). The presence of muscle involvement was associated with a more severe disease phenotype. Although very frequent, ILD had a better functional outcome in cases than in controls. In patients with both anti-PM/Scl and SSc-specific antibodies, a higher frequency of typical SSc features than in those with isolated anti-PM/Scl was observed. Conclusion The analysis of the largest series of anti-PM/Scl+ SSc patients so far reported helps to delineate a specific clinical subset with muscle involvement, cutaneous DM, calcinosis and ILD characterized by a good functional outcome. SRC and malignancies do not seem to be part of this syndrome.

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