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

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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.

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Observational studies: cohort and case-control studies.

Jae Song, Kevin C Chung (2010)

Observational studies constitute an important category of study designs. To address some investigative questions in plastic surgery, randomized controlled trials are not always indicated or ethical to conduct. Instead, observational studies may be the next best method of addressing these types of questions. Well-designed observational studies have been shown to provide results similar to those of randomized controlled trials, challenging the belief that observational studies are second rate. Cohort studies and case-control studies are two primary types of observational studies that aid in evaluating associations between diseases and exposures. In this review article, the authors describe these study designs and methodologic issues, and provide examples from the plastic surgery literature.

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Common and Distinct Clinical Features in Adult Patients with Anti-Aminoacyl-tRNA Synthetase Antibodies: Heterogeneity within the Syndrome

Yasuhito Hamaguchi, Manabu Fujimoto, Takashi Matsushita … (2013)

Objective To identify similarities and differences in the clinical features of adult Japanese patients with individual anti-aminoacyl-tRNA synthetase antibodies (anti-ARS Abs). Methods This was a retrospective analysis of 166 adult Japanese patients with anti-ARS Abs detected by immunoprecipitation assays. These patients had visited Kanazawa University Hospital or collaborating medical centers from 2003 to 2009. Results Anti-ARS Ab specificity included anti-Jo-1 (36%), anti-EJ (23%), anti-PL-7 (18%), anti-PL-12 (11%), anti-KS (8%), and anti-OJ (5%). These anti-ARS Abs were mutually exclusive, except for one serum Ab that had both anti-PL-7 and PL-12 reactivity. Myositis was closely associated with anti-Jo-1, anti-EJ, and anti-PL-7, while interstitial lung disease (ILD) was correlated with all 6 anti-ARS Abs. Dermatomyositis (DM)-specific skin manifestations (heliotrope rash and Gottron’s sign) were frequently observed in patients with anti-Jo-1, anti-EJ, anti-PL-7, and anti-PL-12. Therefore, most clinical diagnoses were polymyositis or DM for anti-Jo-1, anti-EJ, and anti-PL-7; clinically amyopathic DM or ILD for anti-PL-12; and ILD for anti-KS and anti-OJ. Patients with anti-Jo-1, anti-EJ, and anti-PL-7 developed myositis later if they had ILD alone at the time of disease onset, and most patients with anti-ARS Abs eventually developed ILD if they did not have ILD at disease onset. Conclusion Patients with anti-ARS Abs are relatively homogeneous. However, the distribution and timing of myositis, ILD, and rashes differ among patients with individual anti-ARS Abs. Thus, identification of individual anti-ARS Abs is beneficial to define this rather homogeneous subset and to predict clinical outcomes within the “anti-synthetase syndrome.”

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Hierarchical cluster and survival analyses of antisynthetase syndrome: phenotype and outcome are correlated with anti-tRNA synthetase antibody specificity.

Baptiste Hervier, Hervé Devilliers, Raluca Stanciu … (2012)

The clinical phenotype and evolution of antisynthetase syndrome (ASS) are heterogeneous. This study was therefore undertaken to identify subgroups of ASS patients with similar clinico-biological features and outcomes. This retrospective multicentric study included 233 consecutive patients with three different anti-aminoacyl-tRNA-synthetase antibodies (anti-ARS): anti-Jo1 (n=160), anti-PL7 (n=25) and anti-PL12 (n=48). To characterise ASS patients, bivariate, multiple correspondence (MCA), cluster and survival analyses were performed. Interstitial lung disease (ILD) and myositis were the most common ASS manifestations. However, their respective frequencies were correlated to anti-ARS specificity: ILD was more frequent (80% and 88% vs 67%, p=0.014) whereas myositis was less common (44% and 47% vs 74%, p<0.001) in patients with anti-PL7 and anti-PL12 compared to anti-Jo1. The MCA suggested that anti-PL7 and anti-PL12 phenotypes were close to one another and distinct from anti-Jo1. The clustering analysis confirmed these data, identifying subgroups strongly defined by the anti-ARS specificity and other clinical features. Cluster 1 (n=175, 86% of anti-Jo1) defined patients with the most diffuse phenotype, whereas patients from cluster 2 (n=48, 96% of anti-PL12 and anti-PL7) exhibited a disease more restricted to the lung. Patient survival was also conditioned by the anti-ARS specificity, and was significantly lower in patients with anti-PL7/12 rather than anti-Jo1 (p=0.012). Other factors associated with poor survival were mostly related to pulmonary involvement, including severe dyspnea (p=0.003) and isolated ILD (p=0.009) at diagnosis. In patients with ASS, the phenotype and the survival were correlated with the anti-ARS specificity. Copyright © 2012. Published by Elsevier B.V.

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Journal

Journal ID (nlm-ta): J Clin Med

Journal ID (iso-abbrev): J Clin Med

Journal ID (publisher-id): jcm

Title: Journal of Clinical Medicine

Publisher: MDPI

ISSN (Electronic): 2077-0383

Publication date (Electronic): 18 November 2019

Publication date Collection: November 2019

Volume: 8

Issue: 11

Electronic Location Identifier: 2013

Affiliations

[1 ]Department of Rheumatology, University and IRCCS Policlinico S. Matteo Foundation of Pavia and ERN ReCONNET, 27100 Pavia, Italy; gio.zanframundo@ 123456gmail.com (G.Z.); valentina.morandi02@ 123456universitadipavia.it (V.M.); francesco_locatelli@ 123456hotmail.it (F.L.); montecucco@ 123456smatteo.pv.it (C.M.)

[2 ]Department of Internal Medicine, Vall d’Hebron General Hospital, Universitat Autonoma de Barcelona, GEAS group, 08035 Barcelona, Spain; etraller@ 123456vhebron.net (E.T.-A.); aselva@ 123456vhebron.net (A.S.-O.)

[3 ]Department of Pneumology, University and IRCCS Policlinico S. Matteo Foundation of Pavia and ERN Lung, 27100 Pavia, Italy; f.meloni@ 123456smatteo.pv.it

[4 ]Department of Rheumatology, University and ASST Spedali Civili—Brescia and ERN ReCONNET, 25123 Brescia, Italy; ilariacava@ 123456virgilio.it (I.C.); franco.franceschini1@ 123456gmail.com (F.F.)

[5 ]Interstitial Lung Disease and Rheumatology Unit, Instituto Nacional de Enfermedades Respiratorias, Ismael Cosio Villegas, 14080 Mexico City, Mexico; jorroser@ 123456gmail.com (J.R.-S.); ixchelglez19@ 123456gmail.com (M.I.G.P.); medithmejia1965@ 123456gmail.com (M.M.)

[6 ]Department of Rheumatology, Charité—Universitätsmedizin Berlin, 10117 Berlin, Germany; Eugen.Feist@ 123456helios-gesundheit.de (E.F.); Udo.Schneider@ 123456charite.de (U.S.)

[7 ]Department of Rheumatology, Hôpitaux Universitaires de Strasbourg and ERN ReCONNET, 67000 Strasbourg, France; alain.meyer1@ 123456chru-strasbourg.fr

[8 ]Service de Physiologie des Explorations Fonctionnelles, NHC Strasbourg, Université de Strasbourg, 67000 Strasbourg, France; margherita.giannini85@ 123456gmail.com (M.G.); bernard.geny@ 123456chru-strasbourg.fr (B.G.)

[9 ]Department of Rheumatology, Centro Hospitalar e Universitário de Coimbra, 3000-075 Coimbra, Portugal; jdasilva@ 123456ci.uc.pt (J.A.P.d.S.); cjmc85@ 123456gmail.com (C.J.M.C.)

[10 ]Department of Rheumatology, Oslo University Hospital, 0372 Oslo, Norway; oyvind.molberg@ 123456medisin.uio.no (O.M.); helena.andersson@ 123456medisin.uio.no (H.A.)

[11 ]Department of Rheumatology, Cochin Hospital, 75014 Paris, France; veronicacodullo@ 123456yahoo.it

[12 ]Department of Rheumatology, Azienda Ospedaliera Universitaria Pisana, Pisa and ERN ReCONNET, 56126 Pisa, Italy; marta.mosca@ 123456med.unipi.it (M.M.); simone.barsotti.pisa@ 123456gmail.com (S.B.); rneri@ 123456int.med.unipi.it (R.N.)

[13 ]Department of Rheumatology, Azienda Ospedaliero Universitaria S. Anna, 44124 Ferrara, Italy; carloalberto.scire@ 123456unife.it (C.S.); marcello.govoni@ 123456unife.it (M.G.); fefefurini@ 123456gmail.com (F.F.)

[14 ]Department of Rheumatology, Hospital General Universitario Gregorio Marañón, 28007 Madrid, Spain; fjlopezlongo@ 123456hotmail.com (F.J.L.-L.); juliamartinezbarrio@ 123456gmail.com (J.M.-B.)

[15 ]Department of Rheumatology, University of Heidelberg, 69117 Heidelberg, Germany; Hannes.Lorenz@ 123456med.uni-heidelberg.de

[16 ]Department of Rheumatology, University of Padua and ERN ReCONNET, 35122 Padova, Italy; adoria@ 123456unipd.it (A.D.); anna.ghirardello@ 123456unipd.it (A.G.)

[17 ]Department of Rheumatology, Hospital Universitario San Cecilio, 18016 Granada, Spain; nortego@ 123456gmail.com

[18 ]Department of Pneumology, University Hospital of Cattinara, 34149 Trieste, Italy; marco.confalonieri@ 123456asuits.sanita.fvg.it

[19 ]Department of Rheumatology, University Hospital of Cattinara, 34149 Trieste, Italy; paolatomietto@ 123456gmail.com

[20 ]Department of Rheumatology, S. Maria Hospital—IRCCS, 42123 Reggio Emilia, Italy; Nicolo.Pipitone@ 123456asmn.re.it

[21 ]Department of Rheumatology, Severo Ochoa Hospital, 28911 Madrid, Spain; anabelen_r_c@ 123456hotmail.com (A.B.R.C.); mariblazquez@ 123456gmail.com (M.Á.B.C.)

[22 ]Department of Rheumatology and Clinical Immunology, Medical Center—University of Freiburg, Faculty of Medicine, University of Freiburg, 79110 Freiburg, Germany; reinhard.voll@ 123456uniklinik-freiburg.de (R.E.V.); sarah.wendel@ 123456uniklinik-freiburg.de (S.W.)

[23 ]Department of Rheumatology, Ospedale “ Scarlato” Scafati, 84018 Scafati, Italy; scarpasa@ 123456tin.it

[24 ]Department of Rheumatology, HP Metz, Hopital Belle-Ile, 57000 Metz, France; francois.maurier@ 123456hp-metz.fr

[25 ]Department of Rheumatology, ASST Papa Giovanni XXIII, 24127 Bergamo, Italy; mlimonta@ 123456asst-pg23.it

[26 ]Department of Rheumatology, Ospedale di Treviglio, 24047 Treviglio, Italy; paolo_colombelli@ 123456asst-bgovest.it

[27 ]Department of Rheumatology, Ospedale Guglielmo da Saliceto, 29121 Piacenza, Italy; e.arrigoni@ 123456ausl.pc.it (E.A.); bravielena@ 123456yahoo.it (E.B.)

[28 ]Department of Immunology, Azienda Ospedaliera Universitaria Pisana, Pisa and ERN ReCONNET, 56126 Pisa, Italy; paola.migliorini@ 123456med.unipi.it

[29 ]Department of Rheumatology, University Clinic and AOU of Cagliari, 09100 Cagliari, Italy; mathieu@ 123456medicina.unica.it (A.M.); matteopiga@ 123456unica.it (M.P.)

[30 ]Department of Rheumatology, Johann Wolfgang Goethe-Universität, 60590 Frankfurt, Germany; ulrich.drott@ 123456kgu.de (U.D.); christiane.delbrueck@ 123456kgu.de (C.D.)

[31 ]Department of Rheumatology, ACURA Centre for Rheumatic Diseases, 76530 Baden-Baden, Germany; jutta.bauhammer@ 123456gmx.de

[32 ]Department of Rheumatology, Skane University Hospital, 22242 Lund, Sweden; giovanni.cagnotto@ 123456med.lu.se

[33 ]Department of Pneumology, AOU Catania, 95100 Catania, Italy; vancheri@ 123456unict.it (C.V.); dottorsambataro@ 123456gmail.com (G.S.)

[34 ]Department of Rheumatology, University Hospitals, 3000 Leuven, Belgium; ellen.delanghe@ 123456uzleuven.be

[35 ]Department of Rheumatology at CAAD, DiMet, University of Eastern Piedmont (UPO) and AOU “Maggiore della Carità”, 28100 Novara, Italy; pierpaolo.sainaghi@ 123456med.uniupo.it

[36 ]Department of Public Health, Unit of Biostatistics and Clinical Epidemiology, University of Pavia, 27100 Pavia, Italy; cristina.monti@ 123456unipv.it (C.M.); francesca.gigli@ 123456unipv.it (F.G.B.)

[37 ]Department of Rheumatology, Niguarda Hospital, 20162 Milan, Italy; mariaeva.romano@ 123456hotmail.it

[38 ]Department of Pneumology, Ruhrlandklinik, University of Duisburg-Essen and ERN Lung, 45239 Essen, Germany; Francesco.Bonella@ 123456ruhrlandklinik.uk-essen.de

[39 ]Department of Rheumatology, Ruhrlandklinik, University of Duisburg-Essen, 45239 Essen, Germany; ch.specker@ 123456gmail.com

[40 ]Department of Rheumatology, Johannes Gutenberg-University, 55122 Mainz, Germany; schwarting@ 123456uni-mainz.de

[41 ]Department of Rheumatology, Sierrallana Hospital, 39300 Torrelavega, Spain; villa.nacho@ 123456gmail.com

[42 ]Department of Rheumatology, Humanitas Research Hospital, Rozzano, 20089 Milan, Italy; carlo.selmi@ 123456unimi.it (C.S.); dott.ceribelli@ 123456gmail.com (A.C.)

[43 ]Department of Rheumatology, Hospital Universitario La Paz, 28046 Madrid, Spain; lauranuno2@ 123456gmail.com

[44 ]Department of Rheumatology, Hospital Clínico Universitario de Santiago de Compostela, 15702 Santiago de Compostela, Spain; antonio.mera.varela@ 123456sergas.es (A.M.-V.); nair.pg.89@ 123456gmail.com (N.P.G.)

[45 ]Department of Rheumatology, Città della Salute e della Scienza, 10126 Turin, Italy; fusaro.reumatorino@ 123456gmail.com (E.F.); simone.parisi@ 123456hotmail.it (S.P.)

[46 ]Department of Rheumatology, Hospital G. Pini—CTO, 20122 Milan, Italy; Luigi.Sinigaglia@ 123456asst-pini-cto.it (L.S.); Nicoletta.DelPapa@ 123456asst-pini-cto.it (N.D.P.)

[47 ]Department of Rheumatology, Azienda Ospedaliera San Giovanni di Dio, 50143 Firenze, Italy; maubenucci@ 123456tiscali.it

[48 ]Department of Rheumatology, University of Genova 16126 Genova, Italy; cimmino@ 123456unige.it

[49 ]Department of Rheumatology, University La Sapienza and Policlinico Umberto I, 00161 Rome, Italy; Valeria.Riccieri@ 123456uniroma1.it (V.R.); fabrizio.conti@ 123456uniroma1.it (F.C.)

[50 ]Department of Rheumatology, Ospedale San Camillo, 00152 Rome, Italy; giandoreum@ 123456libero.it (G.D.S.); annamariaiuliano@ 123456hotmail.it (A.I.)

[51 ]Department of Internal Medicine, AOU Careggi, 50134 Firenze, Italy; giacomaci@ 123456yahoo.it

[52 ]Department of Immunology, AOU Careggi, 50134 Firenze, Italy; daniele.cammelli@ 123456unifi.it

[53 ]Department of Rheumatology, Azienda Ospedaliera Universitaria di Modena, 41125 Modena, Italy; marco.sebastiani@ 123456unimore.it (M.S.); andreina.manfredi@ 123456gmail.com (A.M.)

[54 ]Department of Rheumatology, Hospital Universitario Ramon y Cajal, 28034 Madrid, Spain; javierbachiller@ 123456gmail.com (J.B.-C.); albertosifuentesg@ 123456gmail.com (W.A.S.G.)

[55 ]Department of Rheumatology, Ospedale Santa Chiara, 38122 Trento, Italy; Giuseppe.Paolazzi@ 123456apss.tn.it

[56 ]University Medical Center- Comprehensive Pulmonary Hypertension Center & Interstitial Lung Disease Clinic Programs, Louisiana State University and Tulane University Schools of Medicine, Pulmonary Division New Orleans, New Orleans, LA 1542, USA

[57 ]Department of Rheumatology, ASL Cuneo 2, 12051 Alba, Italy; rgiorgi@ 123456aslcn2.it

[58 ]Department of Rheumatology, Polytechnic University of Marche, C. Urbani Hospital, 60035 Jesi, Italy; fausto.salaffi@ 123456gmail.com

[59 ]Department of Pneumology, Hospital Universitario Marques de Valdecilla, IDIVAL, University of Cantabria Santander, 39008 Santander, Spain; jmcifrian@ 123456humv.es

[60 ]Department of Clinical Sciences and Community Health, University of Milan and Gaetano Pini Hospital, 20122 Milan, Italy; roberto.caporali@ 123456unimi.it

[61 ]Department of Neurology, IRCCS Mondino Foundation, 27100 Pavia, Italy; enrico.marchioni@ 123456mondino.it

[62 ]Department of Pneumology, Univerity of Milano Bicocca, San Gerardo Hospital, 20900 Monza, Italy; alberto.pesci@ 123456unimib.it (A.P.); giulia.dei@ 123456hotmail.it (G.D.); m.pozzi@ 123456asst-monza.it (M.R.P.)

[63 ]Department of Rheumatology, Mauriziano Hospital, 10126 Turin, Italy; clomater@ 123456mauriziano.it

[64 ]Department of Internal Medicine, Friedrich-Alexander-Universität Erlangen-Nürnberg, 91054 Erlangen, Germany; Joerg.Distler@ 123456uk-erlangen.de (J.D.); Johannes.Knitza@ 123456uk-erlangen.de (J.K.); georg.schett@ 123456uk-erlangen.de (G.S.)

[65 ]Rheumatology Unit—DETO, University of Bari, 70121 Bari, Italy; florenzo.iannone@ 123456uniba.it (F.I.); marco3987@ 123456hotmail.it (M.F.)

[66 ]Clinic of Rheumatology, Department of Medicine, Santa Maria della Misericordia Hospital and University of Udine, 33100 Udine, Italy

[67 ]Rheumatology Unit, Department of Medicine, University of Perugia, 06129 Perugia, Italy; roberto.gerli@ 123456unipg.it (R.G.); elena.bartolonibocci@ 123456unipg.it (E.B.)

[68 ]Department of Rheumatology, AOU Careggi, 50134 Firenze, Italy; s.bellandorandone@ 123456gmail.com (S.B.R.); marco.matuccicerinic@ 123456unifi.it (M.M.-C.)

[69 ]Department of Rheumatology, AO Brunico, 39031 Bruneck, Italy; giuseppe.zampogna@ 123456sabes.it

[70 ]Department of Rheumatology, Hospital Universitario de la Princesa, IIS-Princesa, 28006 Madrid, Spain; efvicenter@ 123456gmail.com (E.V.);

[71 ]Department of Rheumatology, ACURA Center for Rheumatic Diseases, 55543 Bad Kreuznach, Germany; ktriantafyllias@ 123456gmail.com

[72 ]Department of Rheumatology, Hospital Universitario Marques de Valdecilla, IDIVAL, University of Cantabria Santander, 39008 Santander, Spain; rlopezmejias78@ 123456gmail.com (R.L.-M.); miguelaggay@ 123456hotmail.com (M.A.G.-G.)

[73 ]Catedra UAM-Roche, EPID Future, Universitad Autonoma de Madrid, 28006 Madrid, Spain

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[* ]Correspondence: lorenzo.cavagna@ 123456unipv.it ; Tel.: +39-0382-501878

Author information

Giovanni Zanframundo https://orcid.org/0000-0001-5042-1282

Jose Antonio Pereira da Silva https://orcid.org/0000-0002-2782-6780

Simone Barsotti https://orcid.org/0000-0002-4864-4505

Paola Migliorini https://orcid.org/0000-0001-6433-4964

Matteo Piga https://orcid.org/0000-0002-1126-8315

Giovanni Cagnotto https://orcid.org/0000-0002-1682-8158

Gianluca Sambataro https://orcid.org/0000-0001-9933-1202

Ellen De Langhe https://orcid.org/0000-0002-4045-2146

Pier Paolo Sainaghi https://orcid.org/0000-0001-8322-9158

Maurizio Benucci https://orcid.org/0000-0001-8180-6660

Gian Domenico Sebastiani https://orcid.org/0000-0003-2969-6649

Walter Alberto Sifuentes Giraldo https://orcid.org/0000-0003-2548-3988

Johannes Knitza https://orcid.org/0000-0001-9695-0657

Marco Fornaro https://orcid.org/0000-0003-1716-7432

Elena Bartoloni https://orcid.org/0000-0003-4776-2136

Miguel Angel Gonzalez-Gay https://orcid.org/0000-0002-7924-7406

Article

Publisher ID: jcm-08-02013

DOI: 10.3390/jcm8112013

PMC ID: 6912490

PubMed ID: 31752231

SO-VID: 7328f9f1-61ef-4ecc-bcda-5a46614120f2

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Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license ( http://creativecommons.org/licenses/by/4.0/).

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

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Most cited references 18

Observational studies: cohort and case-control studies.

Common and Distinct Clinical Features in Adult Patients with Anti-Aminoacyl-tRNA Synthetase Antibodies: Heterogeneity within the Syndrome

Hierarchical cluster and survival analyses of antisynthetase syndrome: phenotype and outcome are correlated with anti-tRNA synthetase antibody specificity.

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