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Intravenous cyclophosphamide pulse therapy in juvenile dermatomyositis A review of efficacy and safety Riley P; Maillard SM; Wedderburn LR; Woo P; Murray KJ; Pilkington CARheumatology (Oxford) 2004[Apr]; 43 (4): 491-6OBJECTIVES: To assess the efficacy and safety of intravenous cyclophosphamide (CYP) used in severe and refractory juvenile dermatomyositis (JDM). METHODS: Retrospective case note review of the outcome of 12 patients. RESULTS: Assessment at 6 months of therapy in 10 of the 12 patients showed a significant improvement in muscle function as assessed by the Childhood Myositis Assessment Scale (CMAS) (P = 0.012), muscle strength (P = 0.008), global extramuscular disease score (P = 0.008), skin disease severity (P = 0.015) and lactate dehydrogenase (P = 0.028). There were reductions in creatine kinase, alanine aminotransferase, prednisolone dose and ESR, but these did not reach statistical significance. Clinical improvement was maintained after CYP until the most recent follow-up (between 6 months and 7 yr) and no severe side-effects were seen. Reversible complications included lymphopenia, herpes zoster infections and alopecia. The median cumulative dose was 4.6 g/m(2) (range 3-9 g/m(2)). The available evidence suggests that, at the doses required, risks of malignancy, infertility and gonadal failure are low. Two patients with severe treatment-resistant disease died after one dose of CYP, both of whom were ventilated prior to commencement of CYP and were thought to have died as a result of their severe disease process, and too early for clinical benefit to be obtained from the drug. CONCLUSIONS: In this cohort of children with severe and refractory JDM, CYP appeared to have provided major clinical benefit with no evidence of serious toxicity in the short term.|Antirheumatic Agents/adverse effects/*therapeutic use[MESH]|Child[MESH]|Child, Preschool[MESH]|Cyclophosphamide/adverse effects/*therapeutic use[MESH]|Dermatomyositis/*drug therapy[MESH]|Drug Administration Schedule[MESH]|Female[MESH]|Humans[MESH]|Immunosuppressive Agents/adverse effects/*therapeutic use[MESH]|Injections, Intravenous[MESH]|Male[MESH]|Retrospective Studies[MESH]|Treatment Outcome[MESH] |
