Chronic recurrent multifocal osteomyelitis has recently been classified as an autoinflammatory disorder characterized by episodes of systemic inflammation including serological signs of inflammation occurring in the absence of autoantibodies or pathogen agents. The insidious onset of pain with swelling and tenderness localized over the affected bones are the main manifestations. The metaphysis and epiphyses of the long bones, clavicle and vertebrae are affected. We report a male patient aged 2 years and 2 months with osteomyelitis with lytic destruction in two different single sites with a year difference between the episodes. Histological examination of the bone showed inflammation and chronic osteomyelitis, excluding the existence of infectious osteomyelitis, neoplasm and myeloproliferative disease. Clinical symptoms improved under treatment with nonsteroidal anti-inflammatory drugs. Considering chronic recurrent multifocal osteomyelitis may shorten time to diagnosis in order to avoid potentially unnecessary prolonged courses of intravenous antibiotics, complementary studies and multiple biopsies.