Angioimmunoblastic T-cell lymphoma (AITL) is a peripheral T-cell lymphoma characterised by a very aggressive clinical course. Despite conventional treatment, usually intensive adriamycin-containing chemotherapy, patients affected by AITL have a poor prognosis with a median survival of less than 3 years [1]. High-dose chemotherapy with stem cell transplantation (HDCT/ASCT) may rescue patients who fail to achieve the complete remission (CR) after induction chemotherapy, finally leading to an improved long-term event-free survival [2], but chemosensitivity is the widely accepted requirement at the time of transplantation [3]. In this scenario, efforts to identify novel approaches aimed at overcoming resistance to conventional cytotoxic agents are warranted. Here we present a case of AITL refractory both to first line chemotherapy and HDCT/ASCT that achieved a 6-month lasting CR with thalidomide alone. The patient was a 52-year-old male who presented in November 2005 complaining of a 2-month history of systemic symptoms. Physical examination and positron emission tomography/computed tomography scan (PET/CT) showed generalised lymphadenopathies, marked splenomegaly, mild hepatomegaly and bilateral pleural effusions. Laboratory evaluation documented pancytopenia with evidence of concomitant autoimmune hemolytic anemia (increased unconjugated bilirubin, reduction of plasma haptoglobin levels and positive direct antiglobulin test) and hypergammaglobulinemia. Biopsy of a left axillary lymph node confirmed AITL. Trephine bone marrow biopsy showed a diffuse infiltrate of lymphoma cells.

Because of the hemolytic process, first line therapy was based on prednisone 1 mg/kg/day with a rapid improvement of the hemoglobin level. Starting in December 2005 the patient received MACOP-B regimen (methotrexate 400 mg/m2 weeks 2, 6 and 10 with leucovorin rescue, doxorubicin 50 mg/m2 weeks 1, 3, 5, 7, 9 and 11, cyclophosphamide 350 mg/m2 weeks 1, 3, 5, 7, 9 and 11, vincristine 1.4 mg/m2 weeks 2, 4, 6, 8, 10 and 12, prednisone 40 mg/m2 weeks 1–12, and bleomycin 10 mg/m2 weeks 4, 8 and 12) that induced only a partial reduction of B-symptoms. BFM protocol (cycle A: methotrexate 3000 mg/m2 day 1 with leucovorin rescue, vincristine 2 mg/m2 day 1, cyclophosphamide 200 mg/m2 days 1–4, doxorubicin 25 mg/m2 day 4, dexamethasone 10 mg/m2 days 1–4; cycle B: methotrexate 3000 mg/m2 day 1 with leucovorin rescue, vincristine 2 mg/m2 day 1, ifosfamide 800 mg/m2 days 1–4, cytosine arabinoside 150 mg/m2 day 4, VM-26 100 mg/m2 day 4, dexamethasone 10 mg/m2 days 1–4) was delivered as salvage therapy starting in April 2006. After two cycles of the latter therapy, although the complete resolution of systemic symptoms was observed, only a partial reduction (550%) of lymphadenopathies and of splenomegaly was documented. Bone marrow lymphoma infiltration reduced to less than 10%. We then addressed the