TY - JOUR
T1 - Clinical follow-up predictors of disease pattern change in anti-Jo1 positive anti-synthetase syndrome
T2 - Results from a multicenter, international and retrospective study
AU - Bartoloni, Elena
AU - Gonzalez-Gay, Miguel A.
AU - Scirè, Carlo
AU - Castaneda, Santos
AU - Gerli, Roberto
AU - Lopez-Longo, Francisco Javier
AU - Martinez-Barrio, Julia
AU - Govoni, Marcello
AU - Furini, Federica
AU - Pina Murcia, Trinitario
AU - Iannone, Florenzo
AU - Giannini, Margherita
AU - Nuño, Laura
AU - Quartuccio, Luca
AU - Ortego-Centeno, Norberto
AU - Alunno, Alessia
AU - Specker, Christopher
AU - Montecucco, Carlomaurizio
AU - Triantafyllias, Konstantinos
AU - Balduzzi, Silvia
AU - Sifuentes Giraldo, Walter Alberto
AU - Paolazzi, Giuseppe
AU - Bravi, Elena
AU - Schwarting, Andreas
AU - Pellerito, Raffaele
AU - Russo, Alessandra
AU - Selmi, Carlo
AU - Saketkoo, Lesley Ann
AU - Fusaro, Enrico
AU - Parisi, Simone
AU - Pipitone, Nicolò
AU - Franceschini, Franco
AU - Cavazzana, Ilaria
AU - Neri, Rossella
AU - Barsotti, Simone
AU - Codullo, Veronica
AU - Cavagna, Lorenzo
PY - 2017/3/1
Y1 - 2017/3/1
N2 - Objective Arthritis, myositis and interstitial lung disease (ILD) constitute the classic clinical triad of anti-synthetase syndrome (ASSD). These patients experience other accompanying features, such as Raynaud's phenomenon, fever or mechanic's hands. Most ASSD patients develop the complete triad during the follow-up. In the present study we aimed to determine whether the subsequent appearance of accompanying features may suggest the development of triad findings lacking at the onset in anti-Jo1 positive ASSD patients. Methods Anti-Jo1 positive patients presenting with incomplete ASSD (no > 2 classic triad features) were assessed. Clinical characteristics and clusters of disease manifestations were retrospectively collected and analyzed in a large international multicenter cohort of ASSD patients. Results 165 patients (123 women) with incomplete ASSD were identified. Ninety-five patients (57.5%) developed new classic triad manifestations after 15 months median (IQR 9–51) and 40 (24%) developed new accompanying features after 19 months median (IQR 6–56) from disease onset. During the follow-up, the ex-novo occurrence of triad features was observed in 32 out of 40 patients (80%) with new accompanying findings and in 63 out of 125 patients (50.5%) without new accompanying findings (p = 0.002). In patients with at least one new accompanying feature the odds ratio for the occurrence of new triad manifestations was 3.94 with respect to patients not developing ex-novo accompanying findings (95% CI 1.68–9.21, p = 0.002). Conclusion Anti-Jo1 ASSD patients with incomplete forms at disease onset are at high risk for the subsequent occurrence of lacking classic triad findings. Although all ASSD patients should be carefully assessed for the occurrence of new triad features, a closer follow-up should be considered in the subgroup of patients developing ex novo accompanying findings. These patients, indeed, have near four-fold increased risk for new classic triad manifestation occurrence with respect to patients not presenting ex novo accompanying findings.
AB - Objective Arthritis, myositis and interstitial lung disease (ILD) constitute the classic clinical triad of anti-synthetase syndrome (ASSD). These patients experience other accompanying features, such as Raynaud's phenomenon, fever or mechanic's hands. Most ASSD patients develop the complete triad during the follow-up. In the present study we aimed to determine whether the subsequent appearance of accompanying features may suggest the development of triad findings lacking at the onset in anti-Jo1 positive ASSD patients. Methods Anti-Jo1 positive patients presenting with incomplete ASSD (no > 2 classic triad features) were assessed. Clinical characteristics and clusters of disease manifestations were retrospectively collected and analyzed in a large international multicenter cohort of ASSD patients. Results 165 patients (123 women) with incomplete ASSD were identified. Ninety-five patients (57.5%) developed new classic triad manifestations after 15 months median (IQR 9–51) and 40 (24%) developed new accompanying features after 19 months median (IQR 6–56) from disease onset. During the follow-up, the ex-novo occurrence of triad features was observed in 32 out of 40 patients (80%) with new accompanying findings and in 63 out of 125 patients (50.5%) without new accompanying findings (p = 0.002). In patients with at least one new accompanying feature the odds ratio for the occurrence of new triad manifestations was 3.94 with respect to patients not developing ex-novo accompanying findings (95% CI 1.68–9.21, p = 0.002). Conclusion Anti-Jo1 ASSD patients with incomplete forms at disease onset are at high risk for the subsequent occurrence of lacking classic triad findings. Although all ASSD patients should be carefully assessed for the occurrence of new triad features, a closer follow-up should be considered in the subgroup of patients developing ex novo accompanying findings. These patients, indeed, have near four-fold increased risk for new classic triad manifestation occurrence with respect to patients not presenting ex novo accompanying findings.
KW - Anti-synthetase syndrome
KW - Interstitial lung disease
KW - mechanic's hand
KW - Myositis
KW - Prognosis
KW - Raynaud's phenomenon
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U2 - 10.1016/j.autrev.2017.01.008
DO - 10.1016/j.autrev.2017.01.008
M3 - Review article
AN - SCOPUS:85011295962
VL - 16
SP - 253
EP - 257
JO - Autoimmunity Reviews
JF - Autoimmunity Reviews
SN - 1568-9972
IS - 3
ER -