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Conventional onsite wastewater treatment systems (OWTSs) could potentially contribute to the transmission of infectious diseases caused by waterborne pathogenic microorganisms and become an important human health concern, especially in the areas where OWTSs are used as the major wastewater treatment units. Although previous studies suggested the OWTSs could reduce chemical pollutants as well as effectively reducing microbial contaminants from onsite wastewater, the microbiological quality of effluents and the factors potentially affecting the removal are still understudied. Therefore, the design and optimization of pathogen removal performance necessitate a better mechanistic understanding of the hydrological, geochemical, and biological processes controlling the water quality in OWTSs. To fill the knowledge gaps, the sources of pathogens and common pathogenic indicators, along with their major removal mechanisms in OWTSs were discussed. This review evaluated the effectiveness of pathogen removal in state-of-art OWTSs and investigated the contributing factors for efficient pathogen removal (e.g., system configurations, filter materials, environmental and operational conditions), with the aim to guide the future design for optimized treatment performance.