Objective To develop and preliminarily evaluate a case management-based care program for preschool children with epilepsy. Methods The study consisted of three phases: a cross-sectional survey of 22 hospitals, development of the care program via Delphi method (17 experts), and a non-concurrent controlled trial involving 104 children (52 each in control and intervention groups). Results The developed program comprised 6 domains and 33 items. After one month of intervention, the intervention group showed significantly higher medication adherence (P=0.012) and caregiver knowledge scores (P<0.001) compared to the control group. Conclusion The case management-based care program is feasible and effective in improving medication adherence and caregiver knowledge in preschool children with epilepsy.