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Abstract Background Epidemiological research on fractures in children under the age of two is of great importance to help understand differences between accidental and abusive trauma. Objective This systematic review aimed to evaluate studies reporting on the incidence of fractures in children under two years of age, excluding birth injuries. Secondary outcome measures included fracture location, mechanisms of injury and fracture characteristics. Methods A systematic literature review (1946 to February 7th 2024), including prospective and retrospective cohort studies and cross-sectional cohort studies, was performed. Studies including children from other age groups were included if the actual measures for those aged 0–2 years could be extracted. We also included studies restricted to infants. Annual incidence rates of fractures were extracted and reported as the main result. Critical appraisal of was performed using the Appraisal tool for Cross-Sectional Studies. Results Twelve moderate to good quality studies met eligibility criteria, of which seven were based on data from medical records and five were registry studies. Studies investigated different aspects of fractures, making comprehensive synthesis challenging. There was an overall annual fracture incidence rate of 5.3 to 9.5 per 1,000 children from 0–2 years of age; with commonest sites being the radius/ulna (25.2–40.0%), followed by tibia/fibula (17.3–27.6%) and the clavicle (14.6–14.8%) (location based on 3 studies with a total of 407 patients). In infants, the reported incidence ranged between 0.7 to 4.6 per 1,000 (based on 3 studies), with involvement of the clavicle in 22.2% and the distal humerus in 22.2% of cases (based on 1 study). Only a single metaphyseal lesion was reported (proximal humerus of an 11-month-old infant). Fracture mechanisms were detailed in four studies, with fall from chair, bed, table, own height or fall following indoor activities causing 50–60% of fractures. Conclusions There is a paucity of good quality data on fracture incidence in children under the age of two. Larger, prospective and unbiased studies would be helpful in determining normal pattern of injuries, so that differences from abusive trauma may be better understood.