Background: Very late relapse of anaplastic large cell lymphoma (ALCL) is an extremely rare event and until now only one such a case has been reported in the literature.Case Report: We report a 43-year-old patient who developed T cell ALK – 1 (anaplastic lymphoma kinase) negative ALCL 141/2 years after diagnosis of Hodgkin’s disease (HD), mixed cellularity type. The patient was successfully treated with MOPP/ABVD regimen and radiotherapy. On review of the first histopathology performed 141/2 years before, the original diagnosis of HD was revised to that of ALCL. The PCR analysis of TCR delta chain gene and heavy chain immunoglobulin gene rearrangements was performed in lymph node samples from the first diagnosis and relapse to detect monoclonality. Both tests have shown no clonal rearrangements of the genes, suggested germ line patterns and possible dissociation between genotype and phenotype of the neoplastic cells. The patient received 6 courses of CHOP (cyclophosphamide, adriamycin, vincristine and prednisone) and the second complete response was achieved. Moreover eosinophilia gradually decreased and peripheral blood count normalized after two courses of CHOP treatment.Conclusions: To our knowledge this is the latest relapse of ALCL presented in the literature so far. Relapse of ALCL was accompanied with significant peripheral blood eosinophilia and eosinophilic infiltration of the bone marrow and lymphoma tumour.

Keywords: ALK negative, anaplastic large cell lymphoma, Hodgkin’s Disease, late relapse