Purpose To report a case of acute myeloid leukemia with bilateral proptosis as the sole presenting sign. Design Observational case report. Methods A patient with bilateral proptosis was seen in consultation by pediatric ophthalmology. Results Complete blood count, computerized tomography, and bone marrow biopsy confirmed the diagnosis of acute myeloid leukemia, with the proptosis due to diffuse infiltration of all extraocular muscles. Conclusion In a child with the sudden onset of proptosis without any other systemic findings, the diagnosis of acute leukemia must be considered.
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