OBJECTIVES: There is growing recognition among researchers, regulatory agencies, policy makers and payers of the value in taking a more patient-centred approach to health outcomes research. Patient-reported outcome (PRO) measures assess the experience of health conditions from the patient perspective, capturing data that may not be accurately reported by clinicians or observers. Methods in which patients can be actively engaged in PRO development and validation beyond their traditional role as passive participants are presented and discussed. METHODS: The extent of patient engagement in PRO research can range from passive (participant) to active (researcher). ‘Active’ engagement requires patient direction in the planning/conduct of research. Specifically, input into the development of study documents (e.g. interview guides), selection of PRO measures or generation of new items, involvement in cognitive testing (e.g. reviewing results, informing instrument modifications) and in evaluation of measurement properties (e.g. scoring or meaningful changes in scores) are methods of active engagement. The convening of patient advisory groups represents a practical means of facilitating patient engagement in the research process. IMPLICATIONS: The optimal level of engagement depends upon the objectives of the research. In particular there is a need to balance methodological rigour with feasibility, cost and timeline considerations. Most importantly, implementation requires careful planning to ensure patients receive adequate support (e.g. PRO and regulatory training) to ensure that they are comfortable, empowered and able to contribute meaningfully to the research process as equal research partners. CONCLUSIONS: There is scope for patients to be more actively engaged in PRO measure development/validation, as research partners rather than just participants. Patient advisory groups present a practical way of engaging patients at key stages and help to ensure that PRO measures are truly patient-centred, valid and credible; assessing what is important to patients, in a way that reflects patients’ experiences.