Invasive fungal infection (IFI) is one of the most challenging complications in children with acute lymphoblastic leukemia (ALL) treatment, but acute fungal osteomyelitis (OM) is rarely encountered. Here, we describe a case of Candida tropicalis osteomyelitis in a 10-year-old patient with Philadelphia chromosome (Ph)–positive ALL. He was on remission induction therapy at the time of neutropenia, and abscess developed in the right arm. The blood and bone cultures were positive for Candida tropicalis. Antibiotics and anti-fungal were started. A magnetic resonance of the arm reveal in intraosseous abscess, suggestive of OM. Surgical irrigation and debridement of the bone were performed immediately. The patient was effectively treated with antifungal therapy and ALL treatment. Now he is full recovery, having complete clinical remission but with sequelae visible by MRI. He is taking oral posaconazole for consolidation until disappearance of the lesion shadows on MRI and having subsequent cycles of chemotherapy in parallel. The recommended treatment for candida osteomyelitis in Ph-positive ALL patients is fungicidal agent combined with surgery and modification chemotherapy with dasatinib. Using combined modalities of treatment seem to be crucial in the successful management of Ph-positive ALL.