Elsevier

Autoimmunity Reviews

Volume 11, Issue 10, August 2012, Pages 739-745
Autoimmunity Reviews

Review
Comparison of long-term outcome between anti-Jo1- and anti-PL7/PL12 positive patients with antisynthetase syndrome

https://doi.org/10.1016/j.autrev.2012.01.006Get rights and content

Abstract

The aims of the present study were to: compare the characteristics between antisynthetase syndrome (ASS) patients with anti-Jo1 antibody and those with anti-PL7/PL12 antibody. The medical records of 95 consecutive patients with ASS were reviewed. Seventy-five of these patients had anti-Jo1 antibody; the other patients had anti-PL7 (n = 15) or anti-PL12 (n = 5) antibody. At ASS diagnosis, the prevalence of myalgia (p = 0.007) and muscle weakness (p = 0.02) was significantly lower in the group of anti-PL7/PL12-positive patients than in those with anti-Jo1 antibody; median value of CK (p = 0.00003) was also lower in anti-PL7/PL12 patients. Anti-Jo1 positive patients developed more rarely myositis resolution (21.3% vs. 46.2%); in addition, the overall recurrence rate of myositis was higher in anti-Jo1 positive patients than in patients with anti-PL7/PL12 antibody (65.9% vs. 19.4%). Anti-Jo1-positive patients, compared with those with anti-PL7/PL12 antibody, more often experienced: joint involvement (63.3%vs. 40%) and cancer (13.3% vs. 5%). By contrast, anti-PL7/PL12 positive patients, compared with those with anti-Jo1 antibody, more commonly exhibited: ILD (90% vs. 68%); in anti-PL7/PL12 positive patients, ILD was more often symptomatic at diagnosis, and led more rarely to resolution of lung manifestations (5.6% vs. 29.4%). Finally, the group of anti-PL7/PL12 positive patients more commonly experienced gastrointestinal manifestations related to ASS (p = 0.02). Taken together, although anti-Jo1 positive patients with ASS share some features with those with anti-PL7/PL12 antibody, they exhibit many differences regarding clinical phenotype and long-term outcome. Our study underscores that the presence of anti-Jo1 antibody results in more severe myositis, joint impairment and increased risk of cancer. On the other hand, the presence of anti-PL7/PL12 antibody is markedly associated with: early and severe ILD, and gastrointestinal complications. Thus, our study interestingly indicates that the finding for anti-Jo1 and anti-PL7/PL12 antibodies impacts both the long-term outcome and prognosis of patients with ASS.

Introduction

Antisynthetase syndrome (ASS) is characterized by polymyositis/dermatomyositis (PM/DM) associated with antisynthetase antibodies, fever, arthritis, Raynaud's phenomenon, mechanic's hands and interstitial lung disease (ILD) [1], [2], [3], [4], [5], [6], [7], [8], [9], [10], [11]. To date, 8 antisynthetase antibodies have been identified in patients with ASS, i.e.: anti-histidyl (anti-Jo1), anti-threonyl (anti-PL7), anti-alanyl (anti-PL12), anti-isoleucyl (anti-OJ), anti-glycyl (anti-EJ), anti-asparaginyl (anti-KS), anti-phenylalanyl (anti-Zo) and anti-tyrosyl-tRNA (anti-YRS) synthetase [12]. Anti-Jo1 antibody is the more common of antisynthetase antibodies, being encountered in 60 to 80% of patients with ASS; on the other hand, anti-PL7 and anti-PL12 antibodies are less frequent, being reported in 10–15% and 5–10%, respectively, of patients with ASS [12], [13]. Among the clinical manifestations, ILD is the most frequent complication of ASS, occurring in 70 to 89% of patients; in patients with ASS, ILD has been found to result in increased morbidity and mortality [12], [13], [14], [15], [16], [17], [18], [19], [20].

However, to date, no series has previously compared the long-term outcome of ASS between anti-Jo1- and anti-PL7/PL12-positive patients. Therefore, the aims of the current study were to compare both the characteristics and long-term outcome between ASS patients with anti-Jo1 antibody and those with anti-PL7/PL12 antibody.

Section snippets

Patients and methods

This retrospective study began with a search of the institutional centers' medical record index, which provides access to the diagnoses of the centers' patients. The first electronic search involved use of the codes PM and DM to identify patients with a diagnosis of PM/DM seen from January 1995 to January 2011 in 6 academic centers (Lille, Paris, Poitiers, Rennes, Rouen, Tours). The diagnosis of PM/DM was based on Bohan and Peter criteria [21], [22]: 1) symmetric muscle weakness, 2) increased

General characteristics of anti-PL7 and anti-PL12 positive patients with ASS

The 20 anti-PL7- (n = 15) and anti-PL12- (n = 5) positive patients comprised 8 men and 12 women with a median age of 59 years [range: 31–79] at ASS diagnosis; only 2 of these 20 patients had DM (10% of cases).

Ten patients had myalgia (50%), and 8 exhibited muscle weakness (40%). The patients experienced ASS-related complications: fever (n = 6), Raynaud's phenomenon (n = 8), mechanic's hands (n = 6), joint impairment (n = 8), esophageal dysfunction (n = 4), gastrointestinal manifestations (n = 3), ILD (n = 18),

Discussion

Patients with ASS auto-antibodies exhibit characteristic features. However, in our review of the Medline database, we found no series comparing both the features and long-term outcome between ASS patients with anti-Jo1 antibody and those with anti-PL7/PL12 antibody. Thus, the current study is, to the best of our knowledge, the first to compare the characteristics between anti-Jo1 and anti-PL7/PL12-positive patients. Our series considered 95 consecutive anti-Jo1 (n = 75) and anti-PL7/PL12- (n = 20)

Take-home messages

  • Our study underscores that anti-Jo1 positive patients with ASS share some features with anti-PL7/PL12 antibody with some differences regarding clinical phenotype and long-term outcome.

  • Anti-Jo1 positive patients more often developed severe myositis and joint manifestations, including erosive arthritis.

  • Anti-Jo1 positive patients more often exhibited cancer. Our findings suggest that these patients require evaluation for cancer, especially digestive and gynecologic cancers which were responsible

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