Giant inclusions in the blast cells resembling that of Chediak-Higashi syndrome have been reported only in a handful of cases with acute leukaemia. There is a paucity of data on the ultrastructural appearance of such unique light microscopic findings. As many of such patients were reported in late seventies and early eighties [1-4] the outcome of treatment using present day multiple induction/consolidation regimens in such patients has also been rarely reported due to the rarity of such patients. We report here on a patient with acute myeloid leukaemia showing pseudo-Chediak-Higashi inclusions in leukaemic blasts with unusual ultrastructural morphology.