Objective:  This systematic review aimed to inform researchers and policymakers about what validated outcome measures are available to evaluate clinical genetics services (CGS) and the need for new measures.Methods:  Validated outcome measures used to evaluate CGS were identified from a systematic literature review. Subjective outcome measures were assumed to have been validated only if some form of psychometric assessment was reported.Results:  A total of 1688 titles and abstracts were identified, and 61 articles met the inclusion criteria for the final review, which covered 67 validated outcome measures. There were 37 nongenetics-specific and 30 genetics-specific measures identified. No single validated outcome measure encompassed all potential patient benefits from using a CGS. A variety of different domains were identified, including anxiety and depression, coping, decision-making, distress, family environment, health status, knowledge, mood, perception of risk, perceived personal control, psychological impact, quality of life, satisfaction and expectations, self-esteem, spiritual well-being, and worry. Some important aspects of patient benefit from CGS are not covered by existing outcome measures.Conclusions:  New research is necessary to develop the array of outcome measures required to quantify the benefits CGS offer patients living with the effects of genetic conditions. These need to be suitable for use in prospective evaluation studies to provide robust evidence for decision-makers to inform service development and commissioning. This includes prioritization of the existing validated outcome measures in terms of their usefulness and relevance to the measurement and valuation of patient benefits from a CGS.