While myelodysplastic syndrome (MDS) and acute leukemia (AL) are well-known secondary diseases after administration of chemotherapy, particularly alkylating agents, they have only rarely been reported in the context of purine analog treatment. In all cases there was an interval of several months before onset of the secondary disease and cytogenetic analyses showed typical chromosomal aberrations. We report the case of a 68-year old male Caucasian with low-grade lymphoma who developed a MDS during ongoing first-line treatment with the purine analog 2-CDA (Cladribine). Furthermore, a normal karyotype was present initially and at all consecutive control evaluations. Thus, this case represents another rare report of an evolution of purine analog treatment-associated MDS; the absence of cytogenetic aberrations might suggest a different mechanism in the pathogenesis of this secondary disease.

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