Abstract
Myeloid sarcoma is a rare ocular manifestation of acute myeloid leukemia. Bilateral manifestation preceding any clinical signs of leukemia is even rarer. We here report the case of a 16-month-old patient with rapidly progressive, inflammatory, acute bilateral exophthalmos associated with exposure keratitis. Computed tomography (CT) scan of the orbit and of the brain showed bilateral tissue-like infiltration into the orbital cavity. Blood tests showed biologic inflammatory syndrome, bicytopenia and circulating blasts 83%. Myelogram was performed based on the presence of circulating blasts; it showed acute myeloid leukemia (grade 4). Clinicians should suspect myeloid sarcoma in patients with bilateral exophthalmos and conduct a careful interpretation of blood test results.