Background: Off-road running is a growing sport with little research investigating injury profiles of female participants. Bone stress injuries (BSIs) are a particularly detrimental injury with little known about their incidence and risk factors in female off-road runners. Objective: Collate and review the available evidence reporting epidemiological data and risk factors associated with BSI in female off-road runners. Design: Systematic literature review, without meta-analyses. Data Sources: MEDLINE OVID, PubMed, SPORTDiscus, and MEDLINE EBSCO. Searches were finalized in July 2024. Eligibility Criteria: Studies that reported injury surveillance statistics and/or risk factors associated with BSIs in female off-road runners. Results: Seventeen eligible studies were included, of which all reported surveillance statistics and 2 reported risk factors associated with BSI among 897 female runners. Owing to the scarcity of data, cross-country runners were included in the population of off-road runners. Most BSIs were high severity and in the lower leg, with an overall incidence ranging from 0 to 34.39 BSIs per 100,000 athlete-exposures and prevalence ranging from 0% to 40.9%. Study characteristics were reported alongside risk of bias, quality, and level of evidence assessment outcomes from varying tools. Eight significant intrinsic risk factors were associated with BSIs in female off-road runners: increasing age, disrupted menstruation, previous BSI, increased female triad risk, and lower calcium, vitamin D, and calorie intake. Discussion: Limited by the number of studies available reporting data on the specific target population, which highlights the need for performing high-quality prospective studies in the future, this review summarizes the current epidemiological data and risk factors associated with BSIs in female off-road runners.