Testicular invading refractory multiple myeloma during bortezomib treatment successfully treated with lenalidomide: a case report.

Dear Editor, A 64-year-old man was diagnosed with symptomatic multiple myeloma (MM) in June 2009. No extramedullary region including the testes was detected. Serum protein electrophoresis showed IgA-κmonoclonal protein. Durie-Salmon stagewas IIIA and International Staging System stage was II. Treatment was initiated with pulsed dexamethasone and bortezomib (1.3 mg/m) twice weekly with dexamethasone (20 mg on the day of bortezomib administration and the next day).Without any adverse events, he achieved very good partial response (VGPR) after three cycles of bortezomib therapy [1]. Subsequently, he received high-dose melphalan (200 mg/m) followed by autologous stem cell transplantation (ASCT) in November 2009. After ASCT, he was followed up without any therapeutic agents. However, in September 2010, he complained of hip pain, and serum IgA level was elevated to 2,014 mg/dl. As recurrence of MM was diagnosed, we started weekly bortezomib with dexamethasone. The patient soon experienced improvement of symptoms, and serum IgA decreased to normal levels. In July 2011, during weekly bortezomib and dexamethasone, the patient noticed enlargement of his scrotum. Magnetic resonance image revealed enlarged bilateral testes. Serum IgA levels and all other laboratory data were normal. Bilateral orchidectomy was performed in October 2011. Microscopic examination showed positive results for CD56, CD138, and IgA and κ light chain (Fig. 1a, b). The pathological diagnosis wasmetastasis of plasma cell myeloma to the bilateral testes, and the right spermatic cord had positive margins. In November 2011, serum IgA was elevated to 2,304 mg/dl. At relapse of systemicMM including spermatic margins after orchidectomy, a 28-day cycle of lenalidomide (25 mg on days 1–21) and dexamethasone (40 mg on days 1, 8, 15, and 22) was initiated [2]. Serum IgA levels decreased to 1,026 mg/dl and reached a plateau without stump recurrence of the spermatic cord. After 6 cycles of lenalidomide, the patient was treated with cyclophosphamide, vincristine, adriamycin, and dexamethasone (C-VAD) and achieved VGPR after two cycles of C-VAD therapy. In August 2012, he successfully underwent a second ASCT. Two months after ASCT, he started a 28-day cycle of lenalidomide maintenance therapy (10mg on days 1–21) [3]. As of the time of writing, the patient remains alive without recurrence.