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Allogeneic stem cell transplantation for hepatosplenic gammadelta T-cell lymphoma

Hepatosplenic gammadelta T-cell lymphoma (HSTCL) was first described by Farcet et al. in 1990 [1]. Most cases of HSCTL occur in young men. Patients typically present with hepatosplenomegaly and bone marrow infiltration with resultant cytopenias but comparatively little lymphadenopathy. The prognosis of HSCTL is poor with reported median survivals of 8 – 16 months and few instances of longterm disease free survival with conventional chemotherapy [2,3]. Recently, a limited number of cases treated with allogeneic SCT for HSTCL have been reported [2 – 15]. We previously reported a patient of HSTCL successfully treated with allogeneic bone marrow transplantation (BMT) from an HLAidentical sibling [6]. In this report, we provide an update of this patient with 7-years follow-up and review the literature for allogeneic SCT for HSTCL. A 23-year-old Japanese male was admitted to our hospital in June 1999 with abdominal distension, malaise, and night sweats. As previously described, the patient was diagnosed with hepatosplenic gamma/delta T-cell lymphoma and treated with intensive B-NHL86 protocol chemotherapy. After the two courses of chemotherapy, he achieved complete remission (CR). Thereafter, he received allogeneic BMT in August 1999 from an HLAidentically matched younger sister. The conditioning regimen consisted of four fractionated 12-Gy total body irradiation (TBI) on Day 79 and Day 78 and high-dose etoposide (60 mg/kg), which was administered as 24-h continuous intravenous infusion on Day 74. Graft-versus-host disease (GVHD) prophylaxis consisted of intravenous cyclosporine and short-term methotrexate. He had evidence of grade II acute GVHD of the skin and gut, which required no steroid treatment. Chimerism evaluation assessed by fluorescent in situ hybridization (FISH) using a mixture of X and Y chromosome-specific probes revealed complete donor chimerism of bone marrow cells on Day þ28. Because of high risk of disease relapse after transplantation, cyclosporine was tapered rapidly and finished on Day þ70. Thereafter, mild chronic GVHD of the liver and skin developed at 4 months after BMT, requiring treatment with oral cyclosporine for 8 months. At 5 years post-BMT, bone marrow examination revealed complete donor chimerism by sex mismatched FISH. Seven years after BMT, the patient is alive and free of disease. Treatment modalities for HSTCL have considerable heterogeneity [3], including splenectomy, corticosteroids, purine analogue, anthracycline containing regimens such as CHOP (cyclophosphamide, hydroxydaunomycin, vincristine and prednisone) or CHOP-like regimen, second or third generation aggressive lymphoma regimen such as IEV (ifosphamide, epirubicin and etoposide) or modified MACOP-B (methotrexate, etoposide instead of adriamycin, cyclophosphamide, vincristine, predonisone and bleomycin), alemtuzumab and autologous and allogeneic stem cell transplantation. However, such treatments have limited efficacy and the vast majority of patients will die from progressive disease. Although allogeneic SCT, which is the only potentially curative therapy, has been attempted to treat