The first case report of bilateral spontaneous otogenic pneumocephalus.

BACKGROUND: Pneumocephalus is commonly associated with cranial trauma including surgical interventions. Spontaneous pneumocephalus on the other hand is a rare diagnosis. Reported cases were predominantly unilateral. We report unique case of a bilateral spontaneous pneumocephalus probably related to bilateral tegmen defect related to repeated Politzer maneuvers.

CASE DESCRIPTION: A 78-year-old man suffered from vertigo, tinnitus, instability and mild expressive aphasia. In the last 12 weeks before clinical deterioration he underwent series of Politzer maneuvers performed for repetitive inflammation of upper respiratory tract with otitis media. Brain computed tomography revealed a large bilateral collection of intracranial air with associated tegmen defects. Primarily, surgical revision of middle fossa and multilayer reconstruction of the dura and skull base defect on the left side was performed. After five days, the other side was reconstructed in similar way. Postoperative course was uneventful with brisk improvement of symptomatology. Both computed tomography scans undertaken after the first and second surgery showed satisfying regression of intracranial air.

CONCLUSION: Spontaneous pneumocephalus represents a very rare condition often associated with increased middle ear pressure that can be caused by autoinflation (e.g. Valsalva or Politzer maneuver), sneezing or coughing. Described case represents unique presentation of the bilateral spontaneous pneumocephalus associated with bilateral tegmen and dura mater defects managed surgically. Despite its rarity spontaneous pneumocephalus originating from the temporal bone should be considered as a possible diagnosis in patients with suggestive otologic presentation (e.g. tinnitus, instability, hearing loss) and associated non-specific neurological symptomatology.