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BackgroundRemitting seronegative symmetrical synovitis with pitting edema (RS3PE) syndrome is characterized by acute symmetrical synovitis, and pitting edema involving, the dorsal part of hands and feet. It affects more men aged more than 50 years. The inflammatory markers are usually elevated, with negative rheumatoid factor. The underlying etiology is not clear but has been associated with drugs exposure, infections, malignancy in 30% of cases, and coexist with other autoimmune diseases. The clinical course is benign with significant improvement to low-dose prednisolone therapy for 2 to 3 months. It has been rarely reported in dialysis patients.Methods and case descriptionA 54 years old male, known to have long standing uncontrolled diabetes mellitus, hypertension, chronic kidney disease and diabetic retinopathy. Recently, he was treated for left foot cellulitis and advanced renal failure (813 micromol/L) initiated on hemodialysis. He presented one week later with progressive painful bilateral upper limb and lower limb pitting edema, with limited range of movement in hands and reduce urine output. He was resumed on hemodialysis for management of volume overload. Despite removal of almost 12 liters of fluids during consecutive dialysis sessions and improvement of lower limb edema, his bilateral hand edema was persistence with pain and limited movement. Further investigations were done to evaluate for rheumatological condition.ResultsHand X ray was normal and ultrasound of the hand revealed features of synovitis, diffuse subcutaneous oedema and possible cellulitis. He was started on IV antibiotics without major improvement and blood culture was negative. CT chest showed left pleural effusion. The ESR was elevated 120, while serology for HBV. HCV, HIV, and parvovirus were negative. In addition, the autoimmune workup were negative (CCP, RF, ANA, ANCA, C3, C4, ACEI). He was diagnosed with remitting seronegative symmetric synovitis with pitting edema (RS3PE). He was treated with IV hydrocortisone once 100 mg, followed by oral prednisolone 10 mg for 5 days with dramatic improvement in bilateral hand edema and movement. He had hyperglycemia steroid related managed with adjustment of insulin therapy. At follow up in 1 week, patient regain full function of both hands, and he was on regular hemodialysis trice per week.ConclusionRS3PE is rare in hemodialysis patients, with favorable response to steroid therapy. We used shorter duration of 5 days steroid therapy with complete recovery and no recurrence at 1 year follow up. Screening for underlying autoimmune, infection or malignancy in RS3PE is essential. |
