Primary sternal osteomyelitis (PSO) caused by Salmonella is a rare condition and most commonly associated with sickle cell disease. Only one such case has been previously reported in an infant (age, <1 year) worldwide. The present study reported on two infantile cases of PSO caused by Salmonella in the absence of any hematological diseases. A total of two male infants (age, ≤1 year) were referred to our hospital for fever and rapid breathing accompanied by a chest wall mass involving the lower end of the sternum. Imaging findings on CT and ultrasound, which included sternal segment dislocation, lytic destruction and periosteal elevation, confirmed the diagnosis of PSO. Blood and purulent material cultures confirmed that the causative pathogen was Salmonella. The infants were completely cured by sequential intravenous and oral antibiotics followed by surgical debridement. The infants remained symptom-free and local recurrence of PSO was not detected at follow-up. PSO caused by Salmonella in the absence of any hematological diseases is a rare condition. Unfamiliarity with this disease may lead to a delay in diagnosis and serious complications. The current case report presents two cases of PSO along with a brief overview of the characteristics and management modalities for this condition, and it provides a comprehensive reference for pediatricians regarding this rare disease, particularly in infants. Copyright: © Qian et al.