Sickle cell anemia (SCA) is a hematologic disease that can cause severe infectious complications like osteomyelitis. Osteomyelitis commonly affects the long bones in children. However, atypical localizations have also been rarely reported. Herein, we aimed to present a pediatric patient with SCA, who was diagnosed as clavicular osteomyelitis. Our patient was admitted with painful and restricted movement of the right arm and shoulder. Physical examination revealed swelling, tenderness, erythema and localized temperature rise on the right clavicle. The patient received intravenous antibiotic treatment with a diagnosis of osteomyelitis. The patient, whose clinical and laboratory findings resolved, was discharged with oral treatment. On follow-up, there was no complication. Osteomyelitis is an infection that may cause serious complications in children with SCA. It is necessary to be kept in mind for differential diagnosis of the clinically suspected cases. It is possible to obtain full recovery with early diagnosis and appropriate duration and dose of antimicrobial treatment without surgery.