We report a novel fusion of the MYST4 and CBP genes in an acute myeloid leukaemia (AML)-M4 patient exhibiting t(10;16)(q22;p13) and t(11;17)(q23;q21). The t(10;16)(q22;p13) resulted in a rearrangement, where MYST4-CBP and CBP-MYST4 chimaeric transcripts were products of in-frame fusions of MYST4 exon 17 to CBP exon 6 and CBP exon 4 to MYST4 exon 18 respectively. The potential resulting chimaeric proteins showed similarities with MYST3-CBP, MYST3-P300 and MYST3-NCOA2 putative fusion proteins found in other cases of AML.