Aim– To document a case presenting with ocular manifestations in acute myeloid leukaemia(AML).

Materials and methods–A 4 year old male child presented with severe pain, watering, bulging of right eye.On clinical examination of right eye vision was no perception of light, axial proptosis, necrotic cornea due to exposure keratitis. CT Scan and MRI revealed retro–orbital mass encasing right optic nerve and extending upto right cavernous sinus and paranasal sinuses and intra–cranially into anterior temporal region. Peripheral blood smear showed atypical blast cells suggestive of acute leukaemia. Bone marrow smear showing 90% myeloblasts and flow–cytometry confirmed diagnosis as AML–M2.

Result–patient was put on BFM–87 chemotherapy protocol and after 3 months the proptosis has reduced. 

Conclusion– This case emphasizes that childhood proptosis can rarely be initial manifestation of AML. It shows the aggresive nature of the disease and if presented early, with vigorous treatment one can salvage both eye and visual acuity.