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Rheumatoid arthritis and antisynthetase syndrome. A three cases report
G Model
ARTICLE IN PRESS
BONSOI-4328; No. of Pages 2
Joint Bone Spine xxx (2016) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Letter to the Editor Rheumatoid arthritis and antisynthetase syndrome. A three cases report
a r t i c l e
i n f o
Keywords: Rheumatoid arthritis Antisynthetase syndrome Overlap syndrome Anti-Jo1 antibodies
The association of rheumatoid arthritis (RA) with antisynthetase syndrome (ASS) is uncommon and represents a diagnosis problem. This situation may be a new overlap syndrome. We report three such cases (Table 1). They are two women, a man, aged 52–70 years, two cases of non-erosive, ACPA positive polyarthritis (1 and 7 years duration), and one erosive, RF positive nodular RA (23 years duration). ASS was diagnosed at the same time in one case, more than ten years before, and 20 years after in the other cases, in front of moderate muscular signs (2 cases), interstitial lung disease and positive anti-Jo1 serum testing in all cases. Systemic features were noted: pulmonary arterial hypertension (PAH) and monoclonal gammopathy of unknown significance (MGUS) (1 case), Raynaud’s phenomenon and anti-SSa antibodies (1 case), vasculitis and axonal neuropathy (1 case). Treatment was based on corticosteroids associated with methotrexate in all cases, and rituximab in the two most recent cases, with partial improvement of the different symptoms. These
three cases fulfill simultaneously RA and ASS classification criteria. Arthritis is present in more than half of ASS cases [1], and ASS may mimic RA [2]. Associated features such as interstitial lung disease, Raynaud’s phenomenon, presence of ANA may suggest ASS. A recent study collected 40 cases of ACPA negative polyarthritis revealing ASS [3]. The mean delay between the onset of polyarthritis and ASS diagnosis was 27 months, with infrequent and late occurring (40 months for lung involvement) pulmonary and muscular symptoms, skin involvement in 1/4 of the cases, Raynaud’s phenomenon in 1/3 and antisynthetase antibodies in 70% of the cases. In first analysis, ACPA presence seems discriminant between ASS and RA, but ACPA may be present in some cases of myositis [4]. ASS may be associated with subluxing artropathy [5,6], ACPA positivity in ASS is associated with presence of erosions [6,7], and in this situation, the nosologic diagnosis between ASS and RA may be difficult. A recent collaborative study collected 17 such cases of ASS with positive ACPA [7]. Presence of anti-SSa antibodies in case of ASS is associated with a more severe interstitial lung disease [1]. Some cases of an association of RA with ASS were reported [8,9], sometimes under anti TNF treatment [9,10]. The auto-antibody is mainly Jo1, but sometimes it is another type of antisynthetase antibody (PL-7, PL-12) [10]. These cases may correspond to a real overlap syndrome between RA and ASS, fulfilling both classification criteria, and with potential therapeutic implications. This situation highlights the usefulness of lung evaluation in case of a recent onset arthritis, and the potential benefit of anti CD 20 targeted therapy upon the several systemic features presented by these patients.
Table 1 Summary of the 3 cases. Age Sex
Joint involvement
Muscle involvement
Lung involvement
ACPA/RF /Jo1
Other
Treatment
Evolution
70 F
Polyarthritis Non-erosive 2007 Polyarthritis non-erosive Duration: 1 year
Myositis 1995
ILD Fibrosis 1995 ILD Fibrosis Duration: 1 year
+/−/+
PAH MGUS
Stabilization
+/−/+
Raynaud Normal capillaroscopy Cheiroarthropathy Anti-SSa+, BGSA− Vasculitis Axonal neuropathy
PDN MTX RTX PDN MTX RTX
PDN HCQ MTX CTX
Infectious complications
52 M
60 F
Erosive RA Duration: 23 years
Myalgia PET + muscle
Myositis EMG myogene pattern Duration: 2 years
ILD
ND/+/+
Improvement
F: female; M: male; ILD: interstitial lung disease; PAH: pulmonary arterial hypertension; MGUS: monoclonal gammopathy of unknown significance; PDN: prednisone; MTX: methotrexate; RTX: rituximab; HCQ: hydroxychloroquine; CTX: cyclophosphamide; RA: rheumatoid arthritis; EMG: electromyography; BGSA: accessory salivary gland biopsy.
http://dx.doi.org/10.1016/j.jbspin.2015.10.014 1297-319X/© 2016 Société franc¸aise de rhumatologie. Published by Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Wendling D, et al. Rheumatoid arthritis and antisynthetase syndrome. A three cases report. Joint Bone Spine (2016), http://dx.doi.org/10.1016/j.jbspin.2015.10.014
G Model BONSOI-4328; No. of Pages 2
ARTICLE IN PRESS Letter to the Editor / Joint Bone Spine xxx (2016) xxx–xxx
2
Disclosure of interest The authors declare that they have no competing interest. References [1] Iaccarino L, Gatto M, Bettio S, et al. Overlap connective tissue disease syndromes. Autoimmun Rev 2013;12:363–73. [2] Mumm GE, McKown KM, Bell CL. Antisynthetase syndrome presenting as rheumatoid-like polyarthritis. J Clin Rheumatol 2010;16:307–12. [3] Lefèvre G, Meyer A, Launay D, et al. Seronegative polyarthritis revealing antisynthetase syndrome: a multicentre study of 40 patients. Rheumatology (Oxford) 2015;54:927–32. [4] Labrador-Horrillo M, Martinez MA, Selva-O’Callaghan A, et al. Anti-cyclic citrullinated peptide and anti-keratin antibodies in patients with idiopathic inflammatory myopathy. Rheumatology (Oxford) 2009;48:676–9. [5] Meyer O, Charlanne H, Cherin P, et al. Subluxing arthropathy: an unusual manifestation of the antisynthetase syndrome. Ann Rheum Dis 2009;68:152–3. [6] Cavagna L, Fusetti C, Montecucco C, et al. Anticyclic citrullinated peptide antibodies as markers of erosive arthritis in antisynthetase syndrome. J Rheumatol 2010;37:1967. [7] Meyer A, Lefevre G, Bierry G, et al. In antisynthetase syndrome, ACPA are associated with severe and erosive arthritis: an overlapping rheumatoid arthritis and antisynthetase syndrome. Medicine 2015;94:e523. [8] Park CK, Kim TJ, Cho YN, et al. Development of antisynthetase syndrome in a patient with rheumatoid arthritis. Rheumatol Int 2011;31:529–32.
[9] Ishikawa Y, Yukawa N, Ohmura K, et al. Etanercept-induced anti-Jo-1-antibodypositive polymyositis in a patient with rheumatoid arthritis: a case report and review of the literature. Clin Rheumatol 2010;29:563–6. [10] Ishikawa Y, Yukawa N, Kawabata D, et al. A case of antisynthetase syndrome in a rheumatoid arthritis patient with anti-PL-12 antibody following treatment with etanercept. Clin Rheumatol 2011;30:429–32.
Daniel Wendling ∗ Cécile Golden Matthieu Monet Frank Verhoeven Xavier Guillot Clément Prati Service de rhumatologie, CHRU de Besanc¸on, boulevard Fleming, 25030 Besanc¸on, France ∗ Corresponding author. E-mail address: [email protected] (D. Wendling)
Accepted 21 October 2015 Available online xxx
Please cite this article in press as: Wendling D, et al. Rheumatoid arthritis and antisynthetase syndrome. A three cases report. Joint Bone Spine (2016), http://dx.doi.org/10.1016/j.jbspin.2015.10.014
ARTICLE IN PRESS
BONSOI-4328; No. of Pages 2
Joint Bone Spine xxx (2016) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Letter to the Editor Rheumatoid arthritis and antisynthetase syndrome. A three cases report
a r t i c l e
i n f o
Keywords: Rheumatoid arthritis Antisynthetase syndrome Overlap syndrome Anti-Jo1 antibodies
The association of rheumatoid arthritis (RA) with antisynthetase syndrome (ASS) is uncommon and represents a diagnosis problem. This situation may be a new overlap syndrome. We report three such cases (Table 1). They are two women, a man, aged 52–70 years, two cases of non-erosive, ACPA positive polyarthritis (1 and 7 years duration), and one erosive, RF positive nodular RA (23 years duration). ASS was diagnosed at the same time in one case, more than ten years before, and 20 years after in the other cases, in front of moderate muscular signs (2 cases), interstitial lung disease and positive anti-Jo1 serum testing in all cases. Systemic features were noted: pulmonary arterial hypertension (PAH) and monoclonal gammopathy of unknown significance (MGUS) (1 case), Raynaud’s phenomenon and anti-SSa antibodies (1 case), vasculitis and axonal neuropathy (1 case). Treatment was based on corticosteroids associated with methotrexate in all cases, and rituximab in the two most recent cases, with partial improvement of the different symptoms. These
three cases fulfill simultaneously RA and ASS classification criteria. Arthritis is present in more than half of ASS cases [1], and ASS may mimic RA [2]. Associated features such as interstitial lung disease, Raynaud’s phenomenon, presence of ANA may suggest ASS. A recent study collected 40 cases of ACPA negative polyarthritis revealing ASS [3]. The mean delay between the onset of polyarthritis and ASS diagnosis was 27 months, with infrequent and late occurring (40 months for lung involvement) pulmonary and muscular symptoms, skin involvement in 1/4 of the cases, Raynaud’s phenomenon in 1/3 and antisynthetase antibodies in 70% of the cases. In first analysis, ACPA presence seems discriminant between ASS and RA, but ACPA may be present in some cases of myositis [4]. ASS may be associated with subluxing artropathy [5,6], ACPA positivity in ASS is associated with presence of erosions [6,7], and in this situation, the nosologic diagnosis between ASS and RA may be difficult. A recent collaborative study collected 17 such cases of ASS with positive ACPA [7]. Presence of anti-SSa antibodies in case of ASS is associated with a more severe interstitial lung disease [1]. Some cases of an association of RA with ASS were reported [8,9], sometimes under anti TNF treatment [9,10]. The auto-antibody is mainly Jo1, but sometimes it is another type of antisynthetase antibody (PL-7, PL-12) [10]. These cases may correspond to a real overlap syndrome between RA and ASS, fulfilling both classification criteria, and with potential therapeutic implications. This situation highlights the usefulness of lung evaluation in case of a recent onset arthritis, and the potential benefit of anti CD 20 targeted therapy upon the several systemic features presented by these patients.
Table 1 Summary of the 3 cases. Age Sex
Joint involvement
Muscle involvement
Lung involvement
ACPA/RF /Jo1
Other
Treatment
Evolution
70 F
Polyarthritis Non-erosive 2007 Polyarthritis non-erosive Duration: 1 year
Myositis 1995
ILD Fibrosis 1995 ILD Fibrosis Duration: 1 year
+/−/+
PAH MGUS
Stabilization
+/−/+
Raynaud Normal capillaroscopy Cheiroarthropathy Anti-SSa+, BGSA− Vasculitis Axonal neuropathy
PDN MTX RTX PDN MTX RTX
PDN HCQ MTX CTX
Infectious complications
52 M
60 F
Erosive RA Duration: 23 years
Myalgia PET + muscle
Myositis EMG myogene pattern Duration: 2 years
ILD
ND/+/+
Improvement
F: female; M: male; ILD: interstitial lung disease; PAH: pulmonary arterial hypertension; MGUS: monoclonal gammopathy of unknown significance; PDN: prednisone; MTX: methotrexate; RTX: rituximab; HCQ: hydroxychloroquine; CTX: cyclophosphamide; RA: rheumatoid arthritis; EMG: electromyography; BGSA: accessory salivary gland biopsy.
http://dx.doi.org/10.1016/j.jbspin.2015.10.014 1297-319X/© 2016 Société franc¸aise de rhumatologie. Published by Elsevier Masson SAS. All rights reserved.
Please cite this article in press as: Wendling D, et al. Rheumatoid arthritis and antisynthetase syndrome. A three cases report. Joint Bone Spine (2016), http://dx.doi.org/10.1016/j.jbspin.2015.10.014
G Model BONSOI-4328; No. of Pages 2
ARTICLE IN PRESS Letter to the Editor / Joint Bone Spine xxx (2016) xxx–xxx
2
Disclosure of interest The authors declare that they have no competing interest. References [1] Iaccarino L, Gatto M, Bettio S, et al. Overlap connective tissue disease syndromes. Autoimmun Rev 2013;12:363–73. [2] Mumm GE, McKown KM, Bell CL. Antisynthetase syndrome presenting as rheumatoid-like polyarthritis. J Clin Rheumatol 2010;16:307–12. [3] Lefèvre G, Meyer A, Launay D, et al. Seronegative polyarthritis revealing antisynthetase syndrome: a multicentre study of 40 patients. Rheumatology (Oxford) 2015;54:927–32. [4] Labrador-Horrillo M, Martinez MA, Selva-O’Callaghan A, et al. Anti-cyclic citrullinated peptide and anti-keratin antibodies in patients with idiopathic inflammatory myopathy. Rheumatology (Oxford) 2009;48:676–9. [5] Meyer O, Charlanne H, Cherin P, et al. Subluxing arthropathy: an unusual manifestation of the antisynthetase syndrome. Ann Rheum Dis 2009;68:152–3. [6] Cavagna L, Fusetti C, Montecucco C, et al. Anticyclic citrullinated peptide antibodies as markers of erosive arthritis in antisynthetase syndrome. J Rheumatol 2010;37:1967. [7] Meyer A, Lefevre G, Bierry G, et al. In antisynthetase syndrome, ACPA are associated with severe and erosive arthritis: an overlapping rheumatoid arthritis and antisynthetase syndrome. Medicine 2015;94:e523. [8] Park CK, Kim TJ, Cho YN, et al. Development of antisynthetase syndrome in a patient with rheumatoid arthritis. Rheumatol Int 2011;31:529–32.
[9] Ishikawa Y, Yukawa N, Ohmura K, et al. Etanercept-induced anti-Jo-1-antibodypositive polymyositis in a patient with rheumatoid arthritis: a case report and review of the literature. Clin Rheumatol 2010;29:563–6. [10] Ishikawa Y, Yukawa N, Kawabata D, et al. A case of antisynthetase syndrome in a rheumatoid arthritis patient with anti-PL-12 antibody following treatment with etanercept. Clin Rheumatol 2011;30:429–32.
Daniel Wendling ∗ Cécile Golden Matthieu Monet Frank Verhoeven Xavier Guillot Clément Prati Service de rhumatologie, CHRU de Besanc¸on, boulevard Fleming, 25030 Besanc¸on, France ∗ Corresponding author. E-mail address: [email protected] (D. Wendling)
Accepted 21 October 2015 Available online xxx
Please cite this article in press as: Wendling D, et al. Rheumatoid arthritis and antisynthetase syndrome. A three cases report. Joint Bone Spine (2016), http://dx.doi.org/10.1016/j.jbspin.2015.10.014