ABSTRACT
Despite recent advances including new therapeutic options and availability of primary prophylaxis in haemophiliacs, haemophilic synovitis is still the major clinical problem in significant patient population worldwide. We retrospectively reviewed our 10-year experience with Y-90 radiosynovectomy to determine the outcome in the knee joints of patients with haemophilic synovitis. Between 2002 and 2012, 82 knee joints of 67 patients with haemophilic synovitis were treated with Y-90 radiosynovectomy. The mean age was 16.8 ± 7.8 years (range: 5-39 years). The mean follow-up period was 39.6 ± 25.6 months (range: 12-95 months). Failure of therapy represented re-bleeding after a radiosynovectomy was used as an end point in patient time to progression (TTP) analysis. The median TTP was calculated as 72.0 ± 3.6 months (95% CI 64.8-79.1 months) in Kaplan-Meier analysis. The 1, 3 and 5-year survival rates were 89%, 73% and 63% respectively. Longer TTP (hazard ratio for progression, 2.5; P = 0.00) was evident in patients who have greater reduction in bleeding frequency within 6 months after radiosynovectomy. We did not find a relationship between the TTP and the following variables: age, type and severity of haemophilia, the presence or absence of inhibitor, the radiological score, range of motion status of joints and the pretreatment bleeding frequency. We concluded that Y-90 radiosynovectomy in knee joint represents an important resource for the treatment of haemophilic synovitis, markedly reducing joint bleeding and long-term durability, irrespective of the radiographic stage and inhibitor status.
Subject(s)
Hemophilia A/complications , Knee Joint/pathology , Synovitis/etiology , Synovitis/therapy , Yttrium Radioisotopes/therapeutic use , Adolescent , Adult , Child , Child, Preschool , Female , Hemarthrosis/etiology , Hemarthrosis/therapy , Hemophilia A/mortality , Hemophilia A/therapy , Humans , Male , Treatment Outcome , Young Adult , von Willebrand Diseases/complicationsABSTRACT
Radiosynovectomy has been performed successfully for more than 10 years in our hospital. This study investigated the long-term outcome in the context of time to progression (TTP) analysis and the factors influencing TTP following radiosynovectomy with Re-186 in patients with haemophilic synovitis. Radiosynovectomy performed in 165 joints (81 elbows, 74 ankles, 8 shoulder and 2 hip joints) of 106 patients (median age was 18.0 ± 7.5 years; 91 haemophilia A, 13 haemophilia B and 2 von Willebrand's disease between June 2001 and July 2011. The mean follow-up was 48 months (range: 9-120 months). This study revealed that patients' mean TTP after primary radiosynovectomy was satisfactory for both the ankle and elbow joints. There was no TTP differences between the ankle and elbow joint groups (67 vs. 72 months respectively; P = 0.22). We did not find a relationship between the TTP and the following variables: age, type and severity of haemophilia, the presence or absence of inhibitor, the radiological score, range of motion (ROM) status of joints and the pretreatment bleeding frequency. In this study, 18-20% of the treated joints had improved ROM and 82-79% of the treated joints had unchanged ROM after treatment both the ankle and elbow joints respectively. In this report including TTP analysis in the largest series with long-term follow-up, we demonstrated long-term effectiveness of Re-186 radiosynovectomy in haemophilic synovitis. In our experience, the main predictor of outcome following radiosynovectomy is the number of joint bleeding within 6 months after therapy.
Subject(s)
Blood Coagulation Disorders, Inherited/complications , Radioisotopes/therapeutic use , Radiopharmaceuticals/therapeutic use , Rhenium/therapeutic use , Synovitis/radiotherapy , Adolescent , Adult , Child , Child, Preschool , Disease Progression , Disease-Free Survival , Female , Humans , Male , Prospective Studies , Range of Motion, Articular/physiology , Regression Analysis , Synovitis/physiopathology , Young AdultABSTRACT
PURPOSE: The aim of this study was to investigate the genotoxic effect on the peripheral blood lymphocytes potentially induced by Re-186 in paediatric age group undergoing radiosynovectomy for haemophilic synovitis, by using chromosomal aberration analysis (CA) and the micronuclei (MN) assay for detecting chromosomal aberrations, as well as the sister chromatid exchanges (SCE) technique for assessing DNA damage. METHODS: Cytogenetic analyses were evaluated in 20 boys (mean age: 13.8 +/- 2.7 years) before, and 2 and 90 days after radiosynovectomy from the peripheral lymphocytes of the patients. Joint retention and extra-articular spread of the radionuclides were evaluated by using quantitative gamma camera imaging. RESULTS: Imaging after radiosynovectomy revealed local lymph node visualization in 8 (40%) patients and hepatosplenic visualization in 3 (15%) patients due to extra-articular leakage of radioactive material. The mean frequency of chromosome aberrations (0.2 +/- 0.4/1000 cells) determined prior to the onset of therapy was not significantly increased in comparison with control values obtained 2 days (0.4 +/- 0.5/1000 cells) and 90 days (0.2 +/- 0.4/1000 cells) after therapy (P = 0.754 and P = 1.0). In the analysis of MN and SCE, when we compare the baseline levels, the mean MN and SCE frequencies were slightly higher in the control analyses performed 2 and 90 days after radiosynovectomy but there were no significant differences between baseline and control levels (chi(2) = 2.621, P = 0.270 and F = 0.573, P = 0.569, respectively). CONCLUSION: The major finding of this study with relatively small sample is that, radiosynovectomy with Re-186 does not seem to induce early genotoxic effects on the peripheral blood lymphocytes in paediatric age group.
