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Long-term remission after multiple relapses in an elderly patient with lymphomatoid granulomatosis after rituximab and high-dose cytarabine chemotherapy without stem-cell transplantation.

Introduction Use of the anti-CD20 monoclonal antibody rituximab has markedly improved clinical outcomes for patients with diffuse large B-cell lymphoma (DLBCL) over the last decade. However, there are some histologic subtypes of DLBCL for which prognoses have not fully improved, even after rituximab therapy. Epstein-Barr virus (EBV) –positive B-cell lymphoproliferative disorders (LPDs) have been well documented over the last decade, and one disorder that is representative of this group is lymphomatoid granulomatosis (LYG), as defined in the 2008 WHO classification guidelines. LYG, a rare subtype of DLBCL, is an angiocentric and angiodestructive lymphoproliferative disease involving extranodal sites, especially bilateral lungs. This disease involves development of lesions comprising EBV-positive B cells admixed with reactive T cells, which are usually predominant. Some cases of EBV-positive LPDs have morphologic and histologic overlap with classic Hodgkin’s lymphoma (cHL), including the presence of Hodgkin’s and ReedSternberg (HRS) –like giant cells. Most LYG is aggressive; the median survival is less than 2 years. The proportion of EBV-positive B cells relative to the reactive lymphocyte background is related to the grading of LYG. Patients with low-grade lesions usually achieve a durable response to interferon alfa-2b (IFN-2b) therapy because of its antiviral, antiproliferative, and immunomodulatory properties, whereas those with grade 3 lesions have been treated with chemotherapy in combination with rituximab, in a similar manner to management of patients with DLBCL. However, the optimal treatment strategy for patients with LYG has not yet been established. Although case reports of efficacious rituximab monotherapy, rituximabcontaining chemotherapy, and high-dose chemotherapy with autologous stem-cell transplantation have been published, to our knowledge, there has been no previous report of successful treatment resulting in a long-term complete remission (CR) of more than 10 years. Here we present the case of an elderly patient who experienced long-term CR, after previous multiple recurrences, following the administration of a regimen containing rituximab and high-dose cytarabine.