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RS3PE syndrome presenting as vascular endothelial growth factor associated disorder
  1. K Arima1,
  2. T Origuchi2,
  3. M Tamai1,
  4. N Iwanaga1,
  5. Y Izumi1,
  6. M Huang1,
  7. F Tanaka1,
  8. M Kamachi1,
  9. K Aratake1,
  10. H Nakamura1,
  11. H Ida1,
  12. M Uetani3,
  13. A Kawakami1,
  14. K Eguchi1
  1. 1First Department of Internal Medicine, Graduate School of Biomedical Science, Nagasaki University, 1-7-1 Sakamoto, Nagasaki 852-8501, Japan
  2. 2Nagasaki University School of Health Sciences, Nagasaki University, 1-7-1 Sakamoto, Nagasaki 852-8520, Japan
  3. 3Department of Radiology and Radiation Research, Graduate School of Biomedical Science, Nagasaki University, 1-7-1 Sakamoto, Nagasaki 852-8501, Japan
  1. Correspondence to:
    Professor K Eguchi
    kzarima-ngsumin.ac.jp

Abstract

Objectives: To characterise serum concentrations of various cytokines and detection by magnetic resonance imaging (MRI) of synovial hypervascularity in patients with remitting seronegative symmetrical synovitis with pitting oedema (RS3PE) syndrome before and after corticosteroid treatment.

Methods: Vascular endothelial growth factor165 (VEGF165), tumour necrosis factor α (TNFα), and interleukin 1β (IL1β) were measured by enzyme linked immunosorbent assay (ELISA) in serum samples from three patients with RS3PE syndrome. As controls, serum samples from 26 healthy volunteers, 12 patients with rheumatoid arthritis, 10 patients with systemic lupus erythematosus, 13 patients with polymyositis/dermatomyositis, 13 patients with vasculitis syndrome, and 6 patients with mixed connective tissue disease were also analysed. Synovial hypervascularity of patients with RS3PE syndrome was estimated by rate of enhancement (E-rate) in a dynamic MRI study.

Results: Serum concentrations of VEGF165 (mean (SD) 2223.3 (156.3) pg/ml) were significantly higher in patients with active RS3PE syndrome than in controls before corticosteroid treatment. TNFα and IL1β levels were similar in patients and controls. Synovial hypervascularity in affected joints and subcutaneous oedema decreased during corticosteroid treatment, in parallel with the fall in serum VEGF165.

Conclusions: VEGF promotes synovial inflammation and vascular permeability in patients with RS3PE syndrome, suggesting that RS3PE can be classified as a VEGF associated disorder.

  • E-rate, rate of enhancement
  • IL1β, interleukin 1β
  • MRI, magnetic resonance imaging
  • RS3PE syndrome, remitting seronegative symmetrical synovitis with pitting oedema syndrome
  • STIR, short time inversion recovery
  • TE, echo delay time
  • TNFα, tumour necrosis factor α
  • TR, repetition time
  • VEGF, vascular endothelial growth factor
  • RS3PE syndrome
  • vascular endothelial growth factor
  • hypervascularity
  • cytokines

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