Myelofibrosis is a clonal myeloproliferative disorder characterized by splenomegaly, abnormal deposition of reticulin and collagen in the bone marrow, extramedullary hematopoiesis, dacryocytosis and leukoerythroblastic blood smear. Development and sustainment of fibrosis are mediated by complex network of several cytokines. Osteosclerosis is the most frequently observed bone change in myelofibrosis. We present an atypical case of leukemic transformation in myelofibrosis associated with diffuse osteolytic lesions and extremely elevated lactate dehydrogenase in serum, which indicates high bone turnover during leukemic infiltration and bone destruction