Successful treatment of elderly advanced lymphomatoid granulomatosis with rituximab‐CVP combination therapy

To the Editor: We read the conflicting results regarding single rituximab on lymphomatoid granulomatosis (LYG) by Grothey et al. (1) and Polizzotto et al. (2). Here, we report the one elderly patient, with advanced pulmonary LYG, who had involvement of bone marrow at diagnosis, and was successfully and safely treated by rituximab-CVP (cyclophosphamide–vincristine–prednisolone) combination. The 76-year-old female was admitted in August 2005 when the symptoms of dyspnea on exertion and intermittent low grade fever were noted for 1 month and chest X-ray film disclosed lobulated pleural thickening over bilateral chest. The films of chest computerized tomography (CT) found several solid masses at the subpleural region of right lungs and, in addition, there were several enlarged lymph nodes over bilateral lower neck, axilla, and mediastinum. The diagnosis was not made till CT-guided biopsy done twice and pathological findings revealed alveolar parenchyma with extensive mixed inflammatory cell infiltration and fibrosis, among where some large atypical lymphoid cells were immunoreactive for LCA, L26, CD79a, and Epstein–Barr virus latent membrane protein 1. LYG of grade I was diagnosed and involvement of bone marrow was further found. The patient has past history of hypertension and coronary artery disease, and has underwent coronary angioplasty 1 yr earlier. In addition, severe respiratory impairment was noted at diagnosis (Table 1). In term of CD20 expression in tumor tissues, combination therapy with rituximab (375 mg/m) and CVP regimen (cyclophosphamide, 750 mg/m once; vincristine, 1.4 mg/m once; and prednisolone, 60 mg/m/day for 5 d) was given every 3– 4 wk. Total 6 cycles of combination therapy were given and an unconfirmed complete response was achieved. The patient tolerated it well through the whole course and, furthermore, there was a significant improvement of respiratory function (Table 1), with the ECOG grade of performance status from 3 to 0. The patient received additional single rituximab every 3 months as maintenance therapy and, at last follow in November 2006, the disease were still remitted. Because of its rarity, no standard treatment of LGY was established yet. CVP regimen has been used to treat lower grade or indolent lymphoma (3) and Lipford et al. (4) also reported a 50% complete response rate of LGY by using initial cyclophosphamide and prednisolone combination therapy. However, progression to malignant lymphoma following initial therapy was usually refractory to subsequent aggressive chemotherapy (4). Although there were conflicting results on the efficacy of single rituximab (1, 2), Robak et al. (5) recently reported a high activity of the similar combination regimen, rituximab, cladribine, and cyclophosphamide in a patient with advanced LYG. In terms of its safety and efficacy, our case provided additional evidence supporting the use of rituximab and chemotherapy combination in LYG treatment.