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 language | English (US) |
|---|---|
| Article number | e1144 |
| Journal | Medicine |
| Volume | 94 |
| Issue number | 32 |
| DOIs | |
| State | Published - Aug 1 2015 |
| Externally published | Yes |
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ASJC Scopus subject areas
- Medicine(all)
Cite this
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 journal › Article
}
TY - JOUR
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
N2 - 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.
AB - 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.
UR - http://www.scopus.com/inward/record.url?scp=84942436488&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=84942436488&partnerID=8YFLogxK
U2 - 10.1097/MD.0000000000001144
DO - 10.1097/MD.0000000000001144
M3 - Article
C2 - 26266346
AN - SCOPUS:84942436488
VL - 94
JO - Medicine; analytical reviews of general medicine, neurology, psychiatry, dermatology, and pediatries
JF - Medicine; analytical reviews of general medicine, neurology, psychiatry, dermatology, and pediatries
SN - 0025-7974
IS - 32
M1 - e1144
ER -