Compartment syndrome is a limb-threatening and life-threatening emergency resulting from elevated intracompartmental pressure. Prompt surgical intervention and treatment are necessary to prevent irreparable damage to muscle and nerve tissues. Leukemic infiltration of the muscle is an unusual cause of compartment syndrome and has been documented to occur secondary to hyperleukocytic leukemias, most commonly in acute myeloid leukemia. We present a rare case of multiple compartment syndrome in the buttock and thigh of an 11-year-old male patient with chronic myelomonocytic leukemia. The diagnosis of acute compartment syndrome was delayed, causing irreversible tissue damage. Physicians are generally unfamiliar with leukemia-induced complications and may not initially suspect leukemic compartment syndrome because of its rarity. Awareness of its clinical features is critical, because early diagnosis and prompt surgical debridement can prevent significant morbidity and even death.