Gingival hypertrophy is a well-known and extensively documented undesirable side effect of cyclosporine in posttransplant patients. However, severe lip enlargement associated with cyclosporine is less recognized and has seldom been reported in the literature. Lip enlargement may lead to social, physical, and psychological stress, especially in the older childhood and adolescent age groups. We present a case of marked lip hypertrophy and concomitant gingival hypertrophy secondary to cyclosporine (Neoral) treatment in a pediatric bilateral lung transplant recipient. We also discuss the various side effects and treatment considerations available including more recent substitution therapy. Cyclosporine has most effectively and conclusively enabled transplantation of solid organs by reducing transplant-associated morbidity. We believe clinicians should be knowledgeable and aware of lip hypertrophy associated with cyclosporine use. This rare and less understood adverse effect should be recognized during the clinical evaluation of the posttransplant patient.