• Katsuyoshi Idei, Mitsuru Kitamura, Marie N Shimanuki, Makoto Hosoya, Nobuyoshi Tsuzuki, Natsuki Hasebe, Takanori Nishiyama, Hiroyuki Ozawa, and Naoki Oishi.
• Department of Otorhinolaryngology, Head and Neck Surgery, Keio University Hospital, Tokyo, Japan.
• Medicine (Baltimore). 2023 Nov 24; 102 (47): e36124e36124.

RationaleHypertrophic pachymeningitis (HP) is a local or diffuse fibrous thickness of the dura mater of the brain or spinal cord, caused by infection or connective tissue disease. Headache is the most common clinical symptom, followed by various cranial nerve disorders such as visual impairment, diplopia, and hearing loss. HP can be classified into secondary and idiopathic. Here, we report a case of bilateral progressive profound sensorineural hearing loss diagnosed in a patient with idiopathic HP, where a cochlear implant was effectively used.Patient ConcernsThe patient was a 77-year-old woman. Hearing loss gradually progressed bilaterally, and magnetic resonance imaging showed a space-occupying lesion with a continuous contrast enhancement in the bilateral internal auditory canals, and diffused dural thickening from the middle to the posterior cranial fossa.DiagnosesA trans-labyrinthine biopsy was conducted, and a definite diagnosis of idiopathic HP was made. Thickening of the dura mater in the bilateral internal auditory canals was thought to cause profound hearing loss.Interventions And OutcomesA cochlear implant was implemented 4 months after biopsy, and a favorable hearing response was obtained postoperatively.LessonsThis is the first report of a cochlear implant in a patient with idiopathic HP. Cochlear implantation was considered a good treatment for profound hearing loss due to idiopathic HP, which provides a reference for patients to receive timely and correct treatment.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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