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The combination of catatonia and pneumocephalus is very rare and literature is very scarce on this topic. Here, we report a patient with pneumocephalus and catatonia after persistent drain dysfunctions and infections after a hydrocephalus due to an aquaduct stenosis. The combination of a catatonic syndrome and pneumocephalus in neurosurgical and intensive care practice is a rare phenomenon. One should consider it in patients after complicated neurosurgery with pneumocephalus.