Clinical spectrum time course in anti jo-1 positive antisynthetase syndrome: Results from an international retrospective multicenter study

Lorenzo Cavagna, Laura Nuño, Carlo Alberto Scirè, Marcello Govoni, Francisco Javier Lopez Longo, Franco Franceschini, Rossella Neri, Santos Castañeda, Walter Alberto Sifuentes Giraldo, Roberto Caporali, Florenzo Iannone, Enrico Fusaro, Giuseppe Paolazzi, Raffaele Pellerito, Andreas Schwarting, Lesley Ann Saketkoo, Norberto Ortego-Centeno, Luca Quartuccio, Elena Bartoloni, Christof SpeckerTrinitario Pina Murcia, Renato La Corte, Federica Furini, Valentina Foschi, Javier Bachiller Corral, Paolo Airò, Ilaria Cavazzana, Julia Martínez-Barrio, Michelle Hinojosa, Margherita Giannini, Simone Barsotti, Julia Menke, Kostantinos Triantafyllias, Rosetta Vitetta, Alessandra Russo, Gianluigi Bajocchi, Elena Bravi, Giovanni Barausse, Roberto Bortolotti, Carlo Selmi, Simone Parisi, Carlomaurizio Montecucco, Miguel Angel González-Gay, Ken Rosenthal

Research output: Contribution to journalArticle

60 Citations (Scopus)

Abstract

Anti Jo-1 antibodies are the main markers of the anti-synthetase syndrome (ASSD), an autoimmune disease clinically characterized by the occurrence of arthritis, myositis, and interstitial lung disease (ILD). These manifestations usually co-occur (for practical purpose complete forms) in the same patient, but cases with only 1 or 2 of these findings (for practical purpose incomplete forms) have been described. In incomplete forms, the ex novo occurrence of further manifestations is possible, although with frequencies and timing not still defined. The aim of this international, multicenter, retrospective study was to characterize the clinical time course of anti Jo-1 positive ASSD in a large cohort of patients. Included patients should be anti Jo-1 positive and with at least 1 feature between arthritis, myositis, and ILD. We evaluated the differences between complete and incomplete forms, timing of clinical picture appearance and analyzed factors predicting the appearance of further manifestations in incomplete ASSD. Finally, we collected 225 patients (58 males and 167 females) with a median follow-up of 80 months. At the onset, complete ASSD were 44 and incomplete 181. Patients with incomplete ASSD had frequently only 1 of the classic triad findings (110 cases), in particular, isolated arthritis in 54 cases, isolated myositis in 28 cases, and isolated ILD in 28 cases. At the end of follow-up, complete ASSD were 113, incomplete 112. Only 5 patients had an isolated arthritis, only 5 an isolated myositis, and 15 an isolated ILD. During the follow-up, 108 patients with incomplete forms developed further manifestations. Single main feature onset was the main risk factor for the ex novo appearance of further manifestation. ILD was the prevalent ex novo manifestation (74 cases). In conclusion, ASSD is a condition that should be carefully considered in all patients presenting with arthritis, myositis, and ILD, even when isolated. The ex novo appearance of further manifestations in patients with incomplete forms is common, thus indicating the need for an adequate clinical and instrumental follow-up. Furthermore, the study clearly suggested that in ASSD multidisciplinary approach involving Rheumatology, Neurology, Pneumology, and Internal Medicine specialists is mandatory.

Original languageEnglish (US)
Article numbere1144
JournalMedicine
Volume94
Issue number32
DOIs
StatePublished - Aug 1 2015
Externally publishedYes

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Ligases
Multicenter Studies
Retrospective Studies
Interstitial Lung Diseases
Myositis
Arthritis
Antisynthetase syndrome
Pulmonary Medicine
Rheumatology
Neurology
Internal Medicine
Autoimmune Diseases

ASJC Scopus subject areas

  • Medicine(all)

Cite this

Cavagna, L., Nuño, L., Scirè, C. A., Govoni, M., Longo, F. J. L., Franceschini, F., ... Rosenthal, K. (2015). Clinical spectrum time course in anti jo-1 positive antisynthetase syndrome: Results from an international retrospective multicenter study. Medicine, 94(32), [e1144]. https://doi.org/10.1097/MD.0000000000001144

Clinical spectrum time course in anti jo-1 positive antisynthetase syndrome : Results from an international retrospective multicenter study. / Cavagna, Lorenzo; Nuño, Laura; Scirè, Carlo Alberto; Govoni, Marcello; Longo, Francisco Javier Lopez; Franceschini, Franco; Neri, Rossella; Castañeda, Santos; Giraldo, Walter Alberto Sifuentes; Caporali, Roberto; Iannone, Florenzo; Fusaro, Enrico; Paolazzi, Giuseppe; Pellerito, Raffaele; Schwarting, Andreas; Saketkoo, Lesley Ann; Ortego-Centeno, Norberto; Quartuccio, Luca; Bartoloni, Elena; Specker, Christof; Murcia, Trinitario Pina; La Corte, Renato; Furini, Federica; Foschi, Valentina; Corral, Javier Bachiller; Airò, Paolo; Cavazzana, Ilaria; Martínez-Barrio, Julia; Hinojosa, Michelle; Giannini, Margherita; Barsotti, Simone; Menke, Julia; Triantafyllias, Kostantinos; Vitetta, Rosetta; Russo, Alessandra; Bajocchi, Gianluigi; Bravi, Elena; Barausse, Giovanni; Bortolotti, Roberto; Selmi, Carlo; Parisi, Simone; Montecucco, Carlomaurizio; González-Gay, Miguel Angel; Rosenthal, Ken.

In: Medicine, Vol. 94, No. 32, e1144, 01.08.2015.

Research output: Contribution to journalArticle

Cavagna, L, Nuño, L, Scirè, CA, Govoni, M, Longo, FJL, Franceschini, F, Neri, R, Castañeda, S, Giraldo, WAS, Caporali, R, Iannone, F, Fusaro, E, Paolazzi, G, Pellerito, R, Schwarting, A, Saketkoo, LA, Ortego-Centeno, N, Quartuccio, L, Bartoloni, E, Specker, C, Murcia, TP, La Corte, R, Furini, F, Foschi, V, Corral, JB, Airò, P, Cavazzana, I, Martínez-Barrio, J, Hinojosa, M, Giannini, M, Barsotti, S, Menke, J, Triantafyllias, K, Vitetta, R, Russo, A, Bajocchi, G, Bravi, E, Barausse, G, Bortolotti, R, Selmi, C, Parisi, S, Montecucco, C, González-Gay, MA & Rosenthal, K 2015, 'Clinical spectrum time course in anti jo-1 positive antisynthetase syndrome: Results from an international retrospective multicenter study', Medicine, vol. 94, no. 32, e1144. https://doi.org/10.1097/MD.0000000000001144
Cavagna, Lorenzo ; Nuño, Laura ; Scirè, Carlo Alberto ; Govoni, Marcello ; Longo, Francisco Javier Lopez ; Franceschini, Franco ; Neri, Rossella ; Castañeda, Santos ; Giraldo, Walter Alberto Sifuentes ; Caporali, Roberto ; Iannone, Florenzo ; Fusaro, Enrico ; Paolazzi, Giuseppe ; Pellerito, Raffaele ; Schwarting, Andreas ; Saketkoo, Lesley Ann ; Ortego-Centeno, Norberto ; Quartuccio, Luca ; Bartoloni, Elena ; Specker, Christof ; Murcia, Trinitario Pina ; La Corte, Renato ; Furini, Federica ; Foschi, Valentina ; Corral, Javier Bachiller ; Airò, Paolo ; Cavazzana, Ilaria ; Martínez-Barrio, Julia ; Hinojosa, Michelle ; Giannini, Margherita ; Barsotti, Simone ; Menke, Julia ; Triantafyllias, Kostantinos ; Vitetta, Rosetta ; Russo, Alessandra ; Bajocchi, Gianluigi ; Bravi, Elena ; Barausse, Giovanni ; Bortolotti, Roberto ; Selmi, Carlo ; Parisi, Simone ; Montecucco, Carlomaurizio ; González-Gay, Miguel Angel ; Rosenthal, Ken. / Clinical spectrum time course in anti jo-1 positive antisynthetase syndrome : Results from an international retrospective multicenter study. In: Medicine. 2015 ; Vol. 94, No. 32.
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abstract = "Anti Jo-1 antibodies are the main markers of the anti-synthetase syndrome (ASSD), an autoimmune disease clinically characterized by the occurrence of arthritis, myositis, and interstitial lung disease (ILD). These manifestations usually co-occur (for practical purpose complete forms) in the same patient, but cases with only 1 or 2 of these findings (for practical purpose incomplete forms) have been described. In incomplete forms, the ex novo occurrence of further manifestations is possible, although with frequencies and timing not still defined. The aim of this international, multicenter, retrospective study was to characterize the clinical time course of anti Jo-1 positive ASSD in a large cohort of patients. Included patients should be anti Jo-1 positive and with at least 1 feature between arthritis, myositis, and ILD. We evaluated the differences between complete and incomplete forms, timing of clinical picture appearance and analyzed factors predicting the appearance of further manifestations in incomplete ASSD. Finally, we collected 225 patients (58 males and 167 females) with a median follow-up of 80 months. At the onset, complete ASSD were 44 and incomplete 181. Patients with incomplete ASSD had frequently only 1 of the classic triad findings (110 cases), in particular, isolated arthritis in 54 cases, isolated myositis in 28 cases, and isolated ILD in 28 cases. At the end of follow-up, complete ASSD were 113, incomplete 112. Only 5 patients had an isolated arthritis, only 5 an isolated myositis, and 15 an isolated ILD. During the follow-up, 108 patients with incomplete forms developed further manifestations. Single main feature onset was the main risk factor for the ex novo appearance of further manifestation. ILD was the prevalent ex novo manifestation (74 cases). In conclusion, ASSD is a condition that should be carefully considered in all patients presenting with arthritis, myositis, and ILD, even when isolated. The ex novo appearance of further manifestations in patients with incomplete forms is common, thus indicating the need for an adequate clinical and instrumental follow-up. Furthermore, the study clearly suggested that in ASSD multidisciplinary approach involving Rheumatology, Neurology, Pneumology, and Internal Medicine specialists is mandatory.",
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T1 - Clinical spectrum time course in anti jo-1 positive antisynthetase syndrome

T2 - Results from an international retrospective multicenter study

AU - Cavagna, Lorenzo

AU - Nuño, Laura

AU - Scirè, Carlo Alberto

AU - Govoni, Marcello

AU - Longo, Francisco Javier Lopez

AU - Franceschini, Franco

AU - Neri, Rossella

AU - Castañeda, Santos

AU - Giraldo, Walter Alberto Sifuentes

AU - Caporali, Roberto

AU - Iannone, Florenzo

AU - Fusaro, Enrico

AU - Paolazzi, Giuseppe

AU - Pellerito, Raffaele

AU - Schwarting, Andreas

AU - Saketkoo, Lesley Ann

AU - Ortego-Centeno, Norberto

AU - Quartuccio, Luca

AU - Bartoloni, Elena

AU - Specker, Christof

AU - Murcia, Trinitario Pina

AU - La Corte, Renato

AU - Furini, Federica

AU - Foschi, Valentina

AU - Corral, Javier Bachiller

AU - Airò, Paolo

AU - Cavazzana, Ilaria

AU - Martínez-Barrio, Julia

AU - Hinojosa, Michelle

AU - Giannini, Margherita

AU - Barsotti, Simone

AU - Menke, Julia

AU - Triantafyllias, Kostantinos

AU - Vitetta, Rosetta

AU - Russo, Alessandra

AU - Bajocchi, Gianluigi

AU - Bravi, Elena

AU - Barausse, Giovanni

AU - Bortolotti, Roberto

AU - Selmi, Carlo

AU - Parisi, Simone

AU - Montecucco, Carlomaurizio

AU - González-Gay, Miguel Angel

AU - Rosenthal, Ken

PY - 2015/8/1

Y1 - 2015/8/1

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