Posttransplantation lymphoproliferative disease (PTLD) is a well-known complication of solid organ and bone marrow transplantation. It is agreed that the main causes of PTLD are chronic infection with Epstein-Barr virus (EBV); the intensity, rather then the type, of immunosuppression used; and underlying recipient disease. Hepatitis C virus (HCV) and cytomegalovirus, as cofactors of EBV infection, have been suggested to increase the risk of PTLD. Use of calcineurin inhibitors, anti-CD3 monoclonal antibody (OKT3), and antithymocyte globulin may increase the risk of PTLD. On the other hand, mycophenolate mofetil, sirolimus, and the anti-interleukin-2 receptor monoclonal antibodies Daclizumab and basiliximab have not been demonstrated to increase the risk of PTLD. The incidence of PTLD after liver transplantation (LT) is estimated to be 1.5-3%, but a tonsillar location is extremely rare in adults. Thus, little is known about the best diagnostic tools for and treatment by LT recipients with tonsillar PTLD. Here, we report 2 cases of adult LT recipients with tonsillar PTLD. Tonsillectomy was used as a diagnostic tool and treatment option and resulted in complete remission for >2 years. Considering the high mortality and diagnostic difficulties of PTLD, together with the relatively low risks of tonsillectomy, we recommend tonsillectomy for treating tonsil enlargement of unknown cause and suspected PTLD in LT recipients. A larger series of patients and prospective studies comparing different treatment options will be needed to substantiate our recommendation.