Drug induced toxic myopathy is repeatedly reported in familial Mediterranean fever (FMF) patients receiving cyclosporine (CsA) and colchicine combination. Protracted febrile myalgia syndrome (PFMS) is a severe form of FMF which may need aggressive treatment including high dose of steroids. A 34 years old male who is known to have FMF for more than 15 years. He developed renal failure due to secondary amyloidosis. He was on hemodialysis for 6 months till he received live unrelated renal transplant which is functioning normally. There were no acute attacks of FMF for 3 years before transplantation. He was on 0.5 mg colchicine once daily which was continued after transplantation. His immunosuppressive regime was antithymocyte globulin as induction and prednisolone, mycophenolate mofetil and CsA as maintenance therapy. After 2 months he presented with severe myopathy and muscle biopsy showed evidence of toxic myopathy most likely due to cyclosporine when it was initiated in addition to colchicine. CsA was changed to sirolimus and colchicine was stopped. He was gradually improving apart from residual chronic myalgia requiring analgesics. Prednisolone dose was reduced gradually to 5 mg daily. Eight months posttransplant he was readmitted with severe arthralgia, prolonged fever, pleuritic pain, diffuse abdominal pain, macroscopic hematuria, protienuria and diarrhea. All investigations for infections, internal malignancy, hyperparathyroidism and rheumatic diseases were negative other than high C-reactive protein (192mg/l) and erythrocyte sedimentation rate (120mm/hour). The clinical diagnosis was acute attack of FMF presenting as PFMS as he was not receiving colchicine for 6 months. In spite of the history of toxic myopathy 6 months earlier he was challenged with colchicine as 0.5mg twice daily which was well tolerated. There was significant improvement of most of his symptoms within days and graft function continued to be normal. FMF may reactivate after renal transplantation if colchicine is discontinued and present as PFMS which responded to restarting colchicine in our case in spite of the history of toxic myopathy.