While primarily a hematologic disease, sickle cell anemia is notorious for its multisystemic manifestations, particularly in episodes of vaso-occlusive crisis. Multifocal acute calvarial infarction with associated epidural hemorrhage has rarely been reported in sickle cell vaso-occlusive crisis. In this article, we reported a unique case of a 15-year-old male presenting with sickle cell vaso-occlusive crisis and neuroimaging findings of multifocal calvarial bone infarction and epidural hemorrhage. Radiologists and clinicians should be cognizant of this rare complication of sickle cell anemia to ensure appropriate diagnosis and timely treatment.
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