Increased patient advocacy has resulted in a shift toward more active patient engagement in the research. A scoping review was conducted to explore the literature on healthcare research priority settings wherein children, youths, or their families were involved in the priority-setting process. Six databases including MEDLINE, CINAHL, PsycINFO, Embase, Web of Science, and Global Health and the James Lind Alliance website were searched up until September 2019. All primary studies involving children (<18 years of age) or families in developing research priorities in health care were included. All retrieved references were uploaded into Covidence, and two independent reviewers screened the search results. Descriptive thematic analysis was used to identify common themes. A total of 30 studies with 4247 participants were included. Less than half of the participants (n = 1237, (33%) were pediatric patients and their families. A total of 455 research priorities were identified. Three common themes emerged: (i) quality of care delivery, (ii) self-efficacy in health behaviors, and (iii) community engagement in care. This scoping review revealed priority research health topics from the perspectives of children, youths, or their families. The findings may be used as a foundation for future research to improve the health outcomes of children, youths, or their families according to their identified priorities.