Clinical follow-up predictors of disease pattern change in anti-Jo1 positive anti-synthetase syndrome: Results from a multicenter, international and retrospective study

Elena Bartoloni, Miguel A. Gonzalez-Gay, Carlo Scirè, Santos Castaneda, Roberto Gerli, Francisco Javier Lopez-Longo, Julia Martinez-Barrio, Marcello Govoni, Federica Furini, Trinitario Pina Murcia, Florenzo Iannone, Margherita Giannini, Laura Nuño, Luca Quartuccio, Norberto Ortego-Centeno, Alessia Alunno, Christopher Specker, Carlomaurizio Montecucco, Konstantinos Triantafyllias, Silvia BalduzziWalter Alberto Sifuentes Giraldo, Giuseppe Paolazzi, Elena Bravi, Andreas Schwarting, Raffaele Pellerito, Alessandra Russo, Carlo Selmi, Lesley Ann Saketkoo, Enrico Fusaro, Simone Parisi, Nicolò Pipitone, Franco Franceschini, Ilaria Cavazzana, Rossella Neri, Simone Barsotti, Veronica Codullo, Lorenzo Cavagna

Research output: Contribution to journalReview article

19 Citations (Scopus)

Abstract

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.

Original languageEnglish
Pages (from-to)253-257
Number of pages5
JournalAutoimmunity Reviews
Volume16
Issue number3
DOIs
Publication statusPublished - Mar 1 2017

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Ligases
Multicenter Studies
Retrospective Studies
Raynaud Disease
Myositis
Interstitial Lung Diseases
Arthritis

Keywords

  • Anti-synthetase syndrome
  • Interstitial lung disease
  • mechanic's hand
  • Myositis
  • Prognosis
  • Raynaud's phenomenon

ASJC Scopus subject areas

  • Immunology and Allergy
  • Immunology

Cite this

Clinical follow-up predictors of disease pattern change in anti-Jo1 positive anti-synthetase syndrome : Results from a multicenter, international and retrospective study. / Bartoloni, Elena; Gonzalez-Gay, Miguel A.; Scirè, Carlo; Castaneda, Santos; Gerli, Roberto; Lopez-Longo, Francisco Javier; Martinez-Barrio, Julia; Govoni, Marcello; Furini, Federica; Pina Murcia, Trinitario; Iannone, Florenzo; Giannini, Margherita; Nuño, Laura; Quartuccio, Luca; Ortego-Centeno, Norberto; Alunno, Alessia; Specker, Christopher; Montecucco, Carlomaurizio; Triantafyllias, Konstantinos; Balduzzi, Silvia; Sifuentes Giraldo, Walter Alberto; Paolazzi, Giuseppe; Bravi, Elena; Schwarting, Andreas; Pellerito, Raffaele; Russo, Alessandra; Selmi, Carlo; Saketkoo, Lesley Ann; Fusaro, Enrico; Parisi, Simone; Pipitone, Nicolò; Franceschini, Franco; Cavazzana, Ilaria; Neri, Rossella; Barsotti, Simone; Codullo, Veronica; Cavagna, Lorenzo.

In: Autoimmunity Reviews, Vol. 16, No. 3, 01.03.2017, p. 253-257.

Research output: Contribution to journalReview article

Bartoloni, E, Gonzalez-Gay, MA, Scirè, C, Castaneda, S, Gerli, R, Lopez-Longo, FJ, Martinez-Barrio, J, Govoni, M, Furini, F, Pina Murcia, T, Iannone, F, Giannini, M, Nuño, L, Quartuccio, L, Ortego-Centeno, N, Alunno, A, Specker, C, Montecucco, C, Triantafyllias, K, Balduzzi, S, Sifuentes Giraldo, WA, Paolazzi, G, Bravi, E, Schwarting, A, Pellerito, R, Russo, A, Selmi, C, Saketkoo, LA, Fusaro, E, Parisi, S, Pipitone, N, Franceschini, F, Cavazzana, I, Neri, R, Barsotti, S, Codullo, V & Cavagna, L 2017, 'Clinical follow-up predictors of disease pattern change in anti-Jo1 positive anti-synthetase syndrome: Results from a multicenter, international and retrospective study', Autoimmunity Reviews, vol. 16, no. 3, pp. 253-257. https://doi.org/10.1016/j.autrev.2017.01.008
Bartoloni, Elena ; Gonzalez-Gay, Miguel A. ; Scirè, Carlo ; Castaneda, Santos ; Gerli, Roberto ; Lopez-Longo, Francisco Javier ; Martinez-Barrio, Julia ; Govoni, Marcello ; Furini, Federica ; Pina Murcia, Trinitario ; Iannone, Florenzo ; Giannini, Margherita ; Nuño, Laura ; Quartuccio, Luca ; Ortego-Centeno, Norberto ; Alunno, Alessia ; Specker, Christopher ; Montecucco, Carlomaurizio ; Triantafyllias, Konstantinos ; Balduzzi, Silvia ; Sifuentes Giraldo, Walter Alberto ; Paolazzi, Giuseppe ; Bravi, Elena ; Schwarting, Andreas ; Pellerito, Raffaele ; Russo, Alessandra ; Selmi, Carlo ; Saketkoo, Lesley Ann ; Fusaro, Enrico ; Parisi, Simone ; Pipitone, Nicolò ; Franceschini, Franco ; Cavazzana, Ilaria ; Neri, Rossella ; Barsotti, Simone ; Codullo, Veronica ; Cavagna, Lorenzo. / Clinical follow-up predictors of disease pattern change in anti-Jo1 positive anti-synthetase syndrome : Results from a multicenter, international and retrospective study. In: Autoimmunity Reviews. 2017 ; Vol. 16, No. 3. pp. 253-257.
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abstract = "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.",
keywords = "Anti-synthetase syndrome, Interstitial lung disease, mechanic's hand, Myositis, Prognosis, Raynaud's phenomenon",
author = "Elena Bartoloni and Gonzalez-Gay, {Miguel A.} and Carlo Scir{\`e} and Santos Castaneda and Roberto Gerli and Lopez-Longo, {Francisco Javier} and Julia Martinez-Barrio and Marcello Govoni and Federica Furini and {Pina Murcia}, Trinitario and Florenzo Iannone and Margherita Giannini and Laura Nu{\~n}o and Luca Quartuccio and Norberto Ortego-Centeno and Alessia Alunno and Christopher Specker and Carlomaurizio Montecucco and Konstantinos Triantafyllias and Silvia Balduzzi and {Sifuentes Giraldo}, {Walter Alberto} and Giuseppe Paolazzi and Elena Bravi and Andreas Schwarting and Raffaele Pellerito and Alessandra Russo and Carlo Selmi and Saketkoo, {Lesley Ann} and Enrico Fusaro and Simone Parisi and Nicol{\`o} Pipitone and Franco Franceschini and Ilaria Cavazzana and Rossella Neri and Simone Barsotti and Veronica Codullo and Lorenzo Cavagna",
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month = "3",
day = "1",
doi = "10.1016/j.autrev.2017.01.008",
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volume = "16",
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journal = "Autoimmunity Reviews",
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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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DO - 10.1016/j.autrev.2017.01.008

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JO - Autoimmunity Reviews

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