We report the molecular characterization of a complex duplication-triplication rearrangement involving exons 45–60 of the DMD gene which shares common genomic organization with the recently described duplication-inverted triplication-duplication (DUP-TRP/INV-DUP) events. Taking into consideration the presence of a predicted PRDM9 binding site in the near vicinity of the junction involving two inverted L1 elements and the inherent properties of X-Y chromosome recombination during male meiosis, we propose an alternative two-step model for the generation of this X-linked DMD DUP-TRP/INV-DUP event.(Figure is included in full-text article.)