OBJECTIVE: The comparatively poor music appreciation in patients with cochlear implants might be ascribed to an inadequate exposure to music; however, the effect of training on music perception in prelingually deafened children with cochlear implants remains unknown. This study aimed to investigate whether previous musical education improves pitch perception ability in these children. METHODS: Twenty-seven children with congenital/prelingual deafness of profound degree were studied. Test stimuli consisted of 2 sequential piano tones, ranging from C (256 Hz) to B (495 Hz). Children were asked to identify the pitch relationship between the 2 tones (same, higher, or lower). Effects of musical training duration, pitch-interval size, current age, age of implantation, gender, and type of cochlear implant on accuracy of pitch perception were evaluated. RESULTS: The duration of musical training positively correlated with the correct rate of pitch perception. Pitch perception performance was better in children who had a cochlear implant and were older than 6 years than in those who were aged ≤6 years (ie, preschool). Effect of pitch-interval size was insignificant on pitch perception, and there was no correlation between pitch perception and the age of implantation, gender, or type of cochlear implant. CONCLUSIONS: Musical training seems to improve pitch perception ability in prelingually deafened children with a cochlear implant. Auditory plasticity might play an important role in such enhancement. This suggests that incorporation of a structured training program on music perception early in life and as part of the postoperative rehabilitation program for prelingually deafened children with cochlear implants would be beneficial. A longitudinal study is needed to show whether improvement of music performance in these children is measurable by use of auditory evoked potentials.