An 18-month-old boy with sickle cell disease developed progressive left periorbital edema and temporalis fullness. Imaging revealed two inferotemporal subperiosteal fluid collections and adjacent temporalis muscle thickening. After undergoing an anterior orbitotomy with drainage and biopsy, the patient was diagnosed as having a vaso-occlusive sickle cell crisis involving the lateral orbit. [J Pediatr Ophthalmol Strabismus. 2020;57:e78-e81.].

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