Binucleated lymphocytes on blood smear are known in PPBL characterized by stable and polyclonal lymphocytosis, polyclonal increase of serum IgM, HLA DR7 and strong correlation with additional i(3q) and premature chromosome condensation. In this disorder some reports of clonal Ig rearrangement suggest a follow up of these patients with immunological and genetic studies. Binucleated lymphocytes are rarely described in other clonal B-CLPD as B-CLL or marginal zone B-cell lymphoma (MZL). Chromosome 3 abnormality is never described in B-CLL but trisomy 3 represents the most consistent abnormality characterizing the MZL. We report in a man without previous medical history an unusual B-CLPD with monoclonal lymphocytosis CD5-, characteristic cytology (particularly binucleated lymphocytes) and chromosomic abnormality as dicentric chromosome 3 never previously described in B-CLPD. In this case lymphocytosis is persistent and stable over 24 months, cytologic immunologic and chromosomic abnormalities are unchanged. We discuss the nosologic place of this atypical B-CLPD closely related to PPBL and MZL with at the moment, after 24 months, a quiet evolution that imply nevertheless a careful follow up with regular cytologic, immunological and genetic studies to clarify the issue.