An unusual case of remitting seronegative symmetrical synovitis with pitting edema: Case report and literature review

Joseph, Anne D. D.; Kumanan, T.; Aravinthan, N.; Suganthan, Navaneethakrishnan

doi:10.1177/2050313x20910920

Cited by 8 publications

(23 citation statements)

References 18 publications

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“…[ 1 ], but so far the pathological mechanisms remain poorly understood [ 2 – 5 ]. The main diagnostic elements for RS3PE are: (1) pitting edema of the hands and feet, (2) acute onset of polyarthritis, (3) age over 50 years, (4) negative serology for rheumatoid factor [ 3 , 5 , 8 ]. An association with positive human leukocyte antigen (HLA) B7 [ 1 , 3 – 5 ] or a high level of vascular endothelial growth factor (VEGF) was observed in several cases, but their role and diagnostic value are debated [ 2 – 4 , 7 , 9 ].…”

Section: Discussionmentioning

confidence: 99%

“…The infection-induced form seems to be related to parvovirus or Streptobacillus moniliformis infection [ 2 , 5 , 6 , 9 ]. RS3PE can also coexist with other autoimmune diseases, such as Sjögren’s syndrome, systemic lupus erythematosus (SLE), polyarteritis nodosa, or ankylosing spondylitis [ 3 , 4 ]. Concerning PMR, some authors suggested that RS3PE is a part of the same spectrum because their clinical presentation can overlap.…”

Section: Discussionmentioning

confidence: 99%

“…In most cases of LORA (unlike in the young population), the clinical presentation involves the large proximal joints and not small joints, as in the case of our patient [ 13 ]. The absence of RF is not pathognomonic, but the negative antibodies anti-CCP make this diagnosis less likely [ 3 , 5 ].…”

Section: Discussionmentioning

confidence: 99%

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Idiopathic remitting seronegative symmetrical synovitis with pitting edema syndrome mimicking symptoms of polymyalgia rheumatica: a case report

et al. 2022

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Background Remitting seronegative symmetrical synovitis with pitting edema is a rare rheumatic condition of the elderly population that is well described but whose mechanisms remain little studied. This syndrome is characterized by symmetrical swelling located mainly on the dorsal part of the hands and the feet. Because of possible heterogeneous clinical presentation, it can easily mimic the onset of other rheumatic diseases or appear associated with them. Here we report a case of a patient who developed remitting seronegative symmetrical synovitis with pitting edema with preexisting shoulder and hip girdle pain associated with progressive fatigue, indicating a possible differential diagnosis of polymyalgia rheumatica. We reviewed and compared classification for remitting seronegative symmetrical synovitis with pitting edema and polymyalgia rheumatica and discussed other differential diagnoses. Case presentation An 84-year-old Caucasian woman presented to our General Medicine Unit with acute onset of symmetrical hands and feet edema, leading to functional limitation due to pain and stiffness. Additionally, she was complaining about neck, shoulder, and pelvic girdle pain present for about 2 months associated with worsening asthenia. Blood tests showed an elevated level of C-reactive protein and erythrocyte sedimentation rate, as well as absence of anti-cyclic citrullinated peptide antibodies and rheumatoid factor. As all criteria of remitting seronegative symmetrical synovitis with pitting edema syndrome were present, the patient was treated with low-dose prednisone, with a rapid and complete resolution of symptoms. She remains asymptomatic without treatment 2 years after the onset of symptoms, without any evident oncologic etiology. Conclusions This case is an example of a classic representation of remitting seronegative symmetrical synovitis with pitting edema syndrome with clinical elements suggesting a concomitant existing early stage of polymyalgia rheumatica. These two entities, classified in the group of seronegative arthritis, can coexist (up to 10% of cases), with remitting seronegative symmetrical synovitis with pitting edema appearing as an initial or late manifestation of polymyalgia rheumatica. It is essential to remind that remitting seronegative symmetrical synovitis with pitting edema is associated with a higher risk of cancer (30%). A proper diagnosis allows the clinician to precisely define the appropriate therapy duration to limit its side effects in the elderly and remain aware of the potential risk of underlying malignancy.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Idiopathic remitting seronegative symmetrical synovitis with pitting edema syndrome mimicking symptoms of polymyalgia rheumatica: a case report

et al. 2022

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“…Radiologically, there are no erosions. [10] Tenosynovitis has been demonstrated with the imaging of extremities using ultrasonography and /magnetic resonance imaging. In our cases, laboratory and radiological findings were consistent with those reported in the literature.…”

Section: Discussionmentioning

confidence: 99%

Remitting Seronegative Symmetrical Synovitis with Pitting Edema (RS3PE) Syndrome: A Single-Center Experience

Tezcan

Gülcemal

Limon

et al. 2021

Journal of Contemporary Medicine

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Objective: Remitting seronegative symmetrical synovitis with pitting edema (RS3PE) is a rare disorder characterized by pitting edema, the onset of acute polyarthritis, and negative rheumatoid factor. The absence of joint erosion and good long-term response to low-dose steroids are its other characteristics. The present study aimed to evaluate the characteristics and accompanying conditions of RS3PE for contributing to the literature on the issue. Material and method: This is cross-sectional descriptive study was carried out in a rheumatology clinic between January 2019- October 2020. Fifteen patients over 18 diagnosed with RS3PE were included in the study. Results: Of 15 patients diagnosed with RS3PE, seven were female, and eight male. The following comorbid disorders were present: relapsing polychondritis, scleroderma, seropositive rheumatoid arthritis, gout, monoclonal gammopathy of unknown significance, and primary myelofibrosis. Most patients other than the ones who have hematological malignities and those diagnosed with relapsing polychondritis responded rapidly to steroids, and no recurrence occurred. Conclusion: RS3PE may be associated with neoplasia, drugs, and various rheumatismal conditions, suggesting that it may be heterogeneous and considered a paraneoplastic rheumatic disease. The rare occurrence of this syndrome leads clinicians to miss it commonly. It is essential to increase awareness of this entity among clinicians.

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“…6 Other studies reported the presence of edema with delayed lymphatic drainage. 7 Classification criteria for RS3PE have not yet been established. Olive et al in 1997 proposed that all the following diagnostic criteria had to be satisfied in order to make a diagnosis of RS3PE: i) pitting edema in all 4 limbs; ii) acute onset; iii) age >50 years; and iv) negative RF.…”

Section: Introductionmentioning

confidence: 99%

RS3PE and the role of ultrasound. A case report and brief review of the literature

et al. 2021

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Remitting seronegative symmetric synovitis with pitting edema (RS3PE) is a rare syndrome characterised by acute onset of symmetrical distal synovitis and tenosynovitis, associated with severe pitting edema of the dorsum of the hands and feet, seronegativity for autoimmunity and dramatic response to glucocorticoids. First described by McCarty et al. in 1985, RS3PE is reported mostly in elderly males, but it can also rarely occur in young people. Although it was initially regarded as a form of rheumatoid arthritis (RA), it is now considered a clinical distinct entity that can be associated with other rheumatologic conditions or be secondary to underlying diseases, such as cancer. As revealed by magnetic resonance imaging (MRI), the typical landmark of RS3PE is extensor tenosynovitis. Due to extensive subcutaneous edema of the extremities, the clinical assessment of synovitis and tenosynovitis is difficult. On the other hand, the use of MRI is expensive, time-consuming and requires experienced staffs. Musculoskeletal ultrasound (MSUS) may be a viable, reliable and cost-effective tool for evaluation of RS3PE patients. We hereby report the case of an 84 year-old woman presenting with bilateral hand and wrist swelling and morning stiffness. Clinical examination, laboratory tests and imaging led to a diagnosis of RS3PE. The aim of this report is to discuss the role of MSUS in RS3PE for the purpose of diagnosis and differential diagnosis compared with other rheumatologic conditions.

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An unusual case of remitting seronegative symmetrical synovitis with pitting edema: Case report and literature review

Cited by 8 publications

References 18 publications

Idiopathic remitting seronegative symmetrical synovitis with pitting edema syndrome mimicking symptoms of polymyalgia rheumatica: a case report

Idiopathic remitting seronegative symmetrical synovitis with pitting edema syndrome mimicking symptoms of polymyalgia rheumatica: a case report

Remitting Seronegative Symmetrical Synovitis with Pitting Edema (RS3PE) Syndrome: A Single-Center Experience

RS3PE and the role of ultrasound. A case report and brief review of the literature

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