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We prospectively examined rates of outpatient oral medication adherence in children after hematopoietic stem cell transplant (post-HSCT). For 6 months after first discharge post-HSCT, 50 patients (aged 0 to 16 years) and their primary caregivers agreed to store 1 oral medication in an electronic pill bottle that date and time stamps each bottle opening. Demographics, disease, donor type, and prescribed post-HSCT medication regimen were collected via chart review. For each patient percent adherence was calculated by dividing the number of doses taken as indicated by the electronic pill bottle by the number of doses prescribed for the same time period. Average percent adherence ranged from 63% at 1 month after discharge to 57% at 6 months after discharge. For patients who received an allogeneic transplant, lower adherence was associated (P .005) with higher infection rates, after controlling for age and time since transplant. No such relationship was observed for patients who received an autologous transplant. This study demonstrates that poor oral medication adherence is prevalent, persistent, and, for patients receiving an allogeneic transplant, associated with increased incidence of infections during the outpatient treatment period. This study highlights the need for further research examining factors that hinder medication adherence as well as monitoring, promoting, and intervening to maximize medication adherence throughout the HSCT course.