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Hydrocephalus, an extremely rare complication of craniocervical junction injuries, is postulated to result from compression of the fourth ventricular cerebrospinal fluid (CSF) outlets by fractured and displaced bone fragments, a swollen upper spinal cord or adhesions formed after a traumatic subarachnoid haemorrhage. We present the case of a 21-year-old woman for whom an injury to the cervical spine complicated by a type I atlanto-occipital dislocation contributed to the development of non-communicating hydrocephalus. The hydrocephalus was probably a consequence of impaired CSF circulation at the fourth ventricular outlets (the foramina of Luschka and Magendie), caused by post-haemorrhagic adhesions formed after severe injury to the craniocervical junction., (Copyright © 2023. Published by Elsevier España, S.L.U.)