Echinococcal disease is an endemic disease for eastern Mediterranean countries. Various types of kidney involvement have been reported. Here, we report the first case of echinococcal disease on a transplanted kidney in a patient who was successfully treated with albendazole alone. The patient (a 38-year-old female) was evaluated for elevated creatinine levels 7 months after receiving a living-donor allograft. Standard immunosuppression therapy protocols were applied. Tacrolimus level was normal, and the patient was compliant with treatment. Creatinine level was 1.91 mg/dL (baseline: 1.2 mg/dL); proteinuria level was 1300 mg/day. The graft was found to be normal, as evaluated with standard sonographic methods. A kidney biopsy was performed, which showed that part of the cortical parenchyme was infiltrated by echinococcal protoscolices with hooklets. Because there were no cysts present on the graft, we concluded that disease was at an early stage. The patient was given albendazole for 3 months. After therapy, all echinococcal structures disappeared. Her creatinine level dropped to baseline, and proteinuria resolved. Echinococcal disease can affect transplanted kidneys. Albendazole is a valuable treatment option for patients who are not candidates for surgical resection.