Background context Complications associated with C1 lateral mass screw placement are relatively infrequent. The most commonly feared complications include neural or vascular injury. Although both vertebral artery and internal carotid artery injuries have been discussed in the literature, there have been no reports of posterior inferior cerebellar artery (PICA) injury from C1 lateral mass screw placement. We report a case of patient who had a cerebellar stroke after C1 lateral mass screw placement, secondary to injury of an aberrant PICA. Purpose To describe the normal anatomy of the PICA, the anatomic variations previously reported in the literature, the sequela and symptoms of a patient with PICA injury, and the relevance to C1 lateral mass screw placement. No previous reports of PICA injury with a cerebellar stroke have been reported with C1 lateral mass screw instrumentation. Study design Case report and literature review. Methods The patient underwent an Occiput-C6 posterior instrumentation for a pathologic fracture, secondary to multiple myeloma. In the postoperative period, the patient was found to have dysarthria, imbalance, and dysdiadochokinesia. Urgent computed tomography confirmed well placed C1 lateral mass screws. Magnetic resonance imaging/Magnetic Resonance Angiography showed an infarct in the PICA distribution with an abnormal variant of the PICA coursing extracranially around C1. Neurologic monitoring did not detect the injury intraoperatively. Results The patient was treated with anticoagulation and he made a reasonable recovery from his stroke. Conclusions We report the first case of an aberrant PICA injured during a C1 lateral mass screw placement, resulting in a cerebellar stroke. Consideration should be given to abnormal PICA variation when placing C1 lateral mass screws. © 2014 Elsevier Inc. All rights reserved.