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Remitting Seronegative Symmetrical Synovitis With Pitting Edema Syndrome in a Rural Tertiary Care Practice: A Retrospective Analysis

      OBJECTIVES

      To review the clinical and laboratory features of remitting seronegative symmetrical synovitis with pitting edema (RS3PE) in a rural tertiary care rheumatology practice, describe treatments and outcomes, and compare our results to previous reports in the literature.

      PATIENTS AND METHODS

      We performed a retrospective chart review of all patients diagnosed as having RS3PE who were seen in the Department of Rheumatology at Geisinger Medical Center, Danville, PA, from January 1, 1992, to December 31, 2005.

      RESULTS

      We identified 12 men and 2 women, all of whom were white. Mean ± SD age was 74.0±6.6 years; mean ± SD erythrocyte sedimentation rate was 35.9±21.1 mm/h at presentation. Onset of illness was sudden in 9 patients and insidious in 5. All patients were initially treated with prednisone (15-20 mg/d). Although the response in all was excellent, 9 patients received disease-modifying antirheumatic drugs, either because of ongoing disease activity or in an effort to decrease the use of corticosteroids. Hydroxychloroquine was used alone in 7 patients. At the mean ± SD time of last follow-up (31.4±23.1 months), 5 patients continued to receive therapy. Complications of treatment included worsening of preexisting hypertension in 3 patients, gastritis in 2, and exacerbation of preexisting diabetes mellitus in 1. Carpal tunnel syndrome occurred in 6 patients. Duration of therapy ranged from 5 to 120 months (mean, 29 months). Three patients developed malignancies, ie, non-Hodgkin lymphoma, transitional cell carcinoma of the bladder, and prostate carcinoma.

      CONCLUSION

      Our population of patients with RS3PE is similar to those documented in previous reports: elderly, predominantly male, and responsive to corticosteroids. However, our series is clinically differentiated by a greater use of adjunctive disease-modifying antirheumatic drugs (primarily hydroxychloroquine). Confirming previous reports, we also observed a possible association between RS3PE and malignancy.
      ANA (antinuclear antibody), DMARD (disease-modifying antirheumatic drug), ESR (erythrocyte sedimentation rate), GC (glucocorticoid), PMR (polymyalgia rheumatica), RS3PE (remitting seronegative symmetrical synovitis with pitting edema)
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      REFERENCES

        • McCarty DJ
        • O'Duffy JD
        • Pearson L
        • Hunter JB
        Remitting seronegative symmetrical synovitis with pitting edema: RS3PE syndrome.
        JAMA. 1985; 254: 2763-2767
        • Cantini F
        • Salvarani C
        • Olivieri I
        Paraneoplastic remitting seronegative symmetrical synovitis with pitting edema.
        Clin Exp Rheumatol. 1999; 17: 741-744
        • Sibilia J
        • Friess S
        • Schaeverbeke T
        • et al.
        Remitting seronegative symmetrical synovitis with pitting edema (RS3PE): a form of paraneoplastic polyarthritis?.
        J Rheumatol. 1999; 26: 115-120
        • Russell EB
        • Hunter JB
        • Pearson L
        • McCarty DJ
        Remitting, seronegative, symmetrical synovitis with pitting edema—13 additional cases.
        J Rheumatol. 1990; 17: 633-639
        • Olive A
        • del Blanco J
        • Pons M
        • Vaquero M
        • Tena X
        • Catalan Group for the Study of RS3PE
        The clinical spectrum of remitting seronegative symmetrical synovitis with pitting edema.
        J Rheumatol. 1997; 24: 333-336
        • Paira S
        • Graf C
        • Roverano S
        • Rossini J
        Remitting seronegative symmetrical synovitis with pitting oedema: a study of 12 cases.
        Clin Rheumatol. 2002; 21: 146-149
        • Olivo D
        • D'Amore M
        • Lacava R
        • et al.
        Benign edematous polysynovitis in the elderly (RS3PE syndrome).
        Clin Exp Rheumatol. 1994; 12: 669-673
        • Schaeverbeke T
        • Fatout E
        • Marce S
        • et al.
        Remitting seronegative symmetrical synovitis with pitting oedema: disease or syndrome?.
        Ann Rheum Dis. 1995; 54: 681-684
        • Fietta P
        • Manganelli P
        Remitting seronegative symmetrical synovitis with pitting edema and primary Sjogren's syndrome. [letter].
        Clin Exp Rheumatol. 2000; 18: 269-270
        • Manganelli P
        • Delsante G
        • Bianchi G
        • Fietta P
        • Quaini F
        Remitting seronegative symmetrical synovitis with pitting oedema in a patient with myelodysplastic syndrome and relapsing polychondritis.
        Clin Rheumatol. 2001; 20: 132-135
        • Billey T
        • Navaux F
        • Lassoued S
        Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) as the first manifestation of periarteritis nodosa: report of a case.
        Rev Rhum Engl Ed. 1995; 62: 53-54
        • Cantini F
        • Salvarani C
        • Olivieri I
        • et al.
        Remitting seronegative symmetrical synovitis with pitting oedema (RS3PE) syndrome: a prospectivefollow up and magnetic resonance imaging study.
        Ann Rheum Dis. 1999; 58: 230-236
        • Salvarani C
        • Gabriel S
        • Hunder GG
        Distal extremity swelling with pitting edema in polymyalgia rheumatica: report on nineteen cases.
        Arthritis Rheum. 1996; 39: 73-80
        • Kato T
        • Ubara Y
        • Sawa N
        • et al.
        An abrupt onset of seropositive polyarthritis with prominent distal tenosynovitis concomitant with bronchiolitis obliterans organizing pneumonia (BOOP): consideration of the relationship with RS3PE syndrome.
        Intern Med. 2004; 43: 143-147
        • Matsuda M
        • Sakurai K
        • Fushimi T
        • et al.
        Sarcoidosis with high serum levels of vascular endothelial growth factor (VEGF), showing RS3PE-like symptoms in extremities.
        Clin Rheumatol. 2004 Jun; 23 (Epub 2004 Feb 11.): 246-248
        • Olivieri I
        • Salvarani C
        • Cantini F
        RS3PE syndrome: an overview.
        Clin Exp Rheumatol. 2000; 18: S53-S55
        • Schaeverbeke T
        • Vernhes JP
        • Bannwarth B
        • Dehais J
        Is remitting seronegative symmetrical synovitis with pitting oedema (RS3PE syndrome) associated with HLA-A2? [letter].
        Br J Rheumatol. 1995; 34: 889-890
        • Toussirot E
        • Berthier S
        • Wendling D
        • Tiberghien P
        Lack of association between HLA DRB 1* alleles and RS3PE syndrome. [letter].
        Ann Rheum Dis. 1998; 57: 442
        • Russell EB
        • McCarty DJ
        • Schwab J
        • et al.
        RS3PE syndrome: no evidence for retroviruses.
        J Rheumatol. 1994; 21: 1105-1106
        • Perandones CE
        • Colmegna I
        • Arana RM
        Parvovirus B19: another agent associated with remitting seronegative symmetrical synovitis with pitting edema.
        J Rheumatol. 2005; 32: 389-390
        • Mouly S
        • Berenbaum F
        • Kaplan G
        Remitting seronegative symmetrical synovitis with pitting edema following intravesical bacillus Calmette-Guerin instillation.
        J Rheumatol. 2001; 28: 1699-1701
        • Torres A
        • Cuende E
        • De Pablos M
        • Lezuan MJ
        • Michaus L
        • Vesga JC
        Remitting seronegative symmetrical synovitis with pitting edema associated with subcutaneous Streptobacillus moniliformis abscess.
        J Rheumatol. 2001; 28: 1696-1698
        • Pariser KM
        • Canoso JJ
        Remitting, seronegative (A) symmetrical synovitis with pitting edema—two cases of RS3PE syndrome.
        J Rheumatol. 1991; 18: 1260-1262
        • Tada Y
        • Sato H
        • Yoshizawa S
        • et al.
        Remitting seronegative symmetrical synovitis with pitting edema associated with gastric carcinoma.
        J Rheumatol. 1997; 24: 974-975
        • Ethiopia A
        • Bell D
        Gastric carcinoma in association with remitting seronegative symmetrical synovitis with pitting edema.
        J Rheumatol. 1999; 26: 1203-1204
        • Nakashima H
        • Tanaka Y
        • Shigematsu H
        • et al.
        Remitting seronegative symmetrical synovitis with pitting edema syndrome associated with cryptogenic hepatocellular carcinoma [letter].
        Clin Exp Rheumatol. 1999; 17: 390-391
        • Cantini F
        • Olivieri I
        • Salvarani C
        More on remitting seronegative symmetrical synovitis with pitting edema as paraneoplastic syndrome [letter].
        J Rheumatol. 1998; 25: 188-189
        • Olivo D
        • Mattace R
        Concurrence of benign edematous polysynovitis in the elderly (RS3PE syndrome) and endometrial adenocarcinoma.
        Scand J Rheumatol. 1997; 26: 67-68
        • Vinci M
        • Malaguarnera L
        • Pistone G
        RS3PE and Ovarian Cancer [letter].
        Ann Rheum Dis. 2001; 60: 429-431
        • Cobeta-Garcia JC
        • Domingo-Morera JA
        • Martinez-Burgui J
        RS3PE syndrome and chronic lymphoid leukaemia [letter].
        Clin Exp Rheumatol. 1999; 17: 266-267
        • Goldenberg K
        • Rozenbaum M
        • Rosner I
        • Naschitz J
        • Kotler C
        Remitting symmetric seronegative synovitis with pitting edema (RS3PE) secondary to non-Hodgkin's lymphoma [letter].
        Clin Exp Rheumatol. 1998; 16: 767-768
        • Hernandez-Beriain JA
        • Cuesta-Callardo I
        • Segura-Garcia C
        Remitting seronegative symmetrical synovitis with pitting edema: a rheumatic manifestation of myelodysplastic syndrome? [letter].
        Rev Rhum Engl Ed. 1996; 63: 629-630