Rinsho Shinkeigaku (Clinical Neurology)

Case Report

Two cases of hypertrophic cranial pachymeningitis associated with infection in the external auditory canal and paranasal sinus

Yasutaka Tajima, M.D., Ri-ichiro Kishimoto, M.D., Kazumasa Sudoh, M.D. and Akihisa Matsumoto

Department of Neurology, Sapporo City General Hospital

We here present two cases of hypertrophic cranial pachymeningitis exhibiting unique multiple cranial neuropathies, both of which were associated with otic and paranasal infections.
Case 1: A 76-year-old woman developed headache after undergoing surgical dilatation of the external auditory canal, with subsequent development of a bacterial infection. Neurological examination reveled only bilateral hearing disturbance. MRI and CT scans demonstrated thickening of the dura mater and inflammatory granulation around the left cerebellar tentorium. Based on a diagnosis of hypertrophic pachymeningitis associated with previous infection, antibiotics were administered, followed by oral prednisolone therapy. This treatment relieved the headache and improved the MRI findings. However, 2 months later, the headache became worse and impaired movement of the soft palate, atrophy of the left side of the tongue, and atrophy of the sternocleidomastoideus muscle were noted. MRI revealed aggravated inflammatory changes around the left cerebellar tentorium and their expansion into the jugular foramen. Occulusive changes in the transverse and sigmoid sinuses were also seen.
Case 2: A 78-year-old man developed bilateral visual loss, right frontal headache, and bilateral restriction of eye movement. He had been treated for phemphigus with prednisolone and azathioprine. MRI showed hypertrophic dura mater spreading continuously from the frontal base and ethmoid and frontal sinuses to the falx and right frontal lobe. Since Pseudomonas aeruginosa was cultivated in biopsy specimens from the dura mater, antibiotic agents were administered. The clinical symptoms resolved and MRI findings gradually improved.

(CLINICA NEUROL, 43: 258|264, 2003)
key words: hypertrophic cranial pachymeningitis, sinusitis, external auditory infection, sinus thrombosis, multiple cranial neuropathy

(Received: 12-Dec-02)