Aneurysmal bone cysts (ABCs) are nonneoplastic bone lesions that may originate of any site of skeleton. The spine can be affect up to 30% of the cases, leading to pain, neurological deficits, and pathological fractures in symptomatic patients. The incidence of craniocervical (occipito-C1-2) occurrence is not known. We describe the surgical technique and clinical results of two patients with craniocervical ABCs that underwent primary surgical resection: An 11-year-old girl with a lesion in the posterior aspect of the atlas, and a 28-year-old man with an important hydrocephalus and a posterior expansible lesion on the left side of his posterior fossa. Total resection was achieved on both lesions, with no surgical morbidity. Even though ABCs are nonneoplastic lesions, subtotal resection is associated with early recurrence. The knowledge of the anatomy of the region in order to achieve the occlusion of arterial feeders prior to surgical resection itself is the key point of the surgical strategy.