Pressure injuries are an accepted complication of prone positioning during the neurosurgical procedures. Horseshoe headrest are intended to reduce the incidence and severity of such injuries by allowing limited areas of contact between the skin of dependent areas of contact and the supporting surfaces. We report a case where a patient positioned prone over a horseshoe headrest developed inadvertent unilateral facial pressure injuries following a 6-h long craniovertebral junction (CVJ) surgery. We attempt to highlight this complication, analyze its causation, and briefly review the existing literature related to similar reported injuries.