Introduction: Although pneumocephalus is very common after intracranial or spinal surgeries, pneumoventricle is uncommon. Tension pneumoventricle (tPV) occurs when air in the ventricles expands to cause neurological deficits or mass effect. It is usually seen with a combination of a ball-valve defect postoperatively that sucks in air and a ventriculoperitoneal shunt that drains cerebrospinal fluid and allows the ingress of air. Case Report: A young man developed delayed tPV after surgery for craniopharyngioma. He required multiple surgeries and occlusion of the ventriculoperitoneal shunt before the tPV resolved. Conclusions: The probable mechanisms of tPV and the importance of early recognition and treatment are discussed. A review of the literature of this uncommon entity has also been performed.