DOI: 10.1097/mao.0000000000004935 ISSN: 1531-7129

Efficacy of Cochlear Implantation in Kelch-like Protein–11 (KLHL11) IgG–Associated Rhombencephalitis

Karl R. Khandalavala, Ty J. Skyles, Kayla R. Goulson, John I. Lane, Divyanshu Dubey, Matthew L. Carlson

Objective:

To describe cochlear implant outcomes in a patient with bilateral rapidly progressive hearing loss secondary to Kelch-like protein–11 (KLHL11) IgG rhombencephalitis.

Patient:

A 26-year-old male presenting to neurotology clinic at a tertiary care center for evaluation of bilateral rapidly progressive sensorineural hearing loss, unresponsive to systemic corticosteroid therapy.

Interventions:

Bilateral simultaneous cochlear implantation.

Main Outcome Measures:

Postimplant speech testing (CNC word, AzBio sentence scoring), and changes to hearing function with fluctuations in systemic disease symptomatology.

Results:

Preoperative testing demonstrated poor speech perception (bilateral AzBio sentence in quiet: 43%, right-sided CNCw score: 38%, and left-sided CNCw score: 0%). Despite systemic disease progression of KLHL11-IgG rhombencephalitis, including worsening cerebellar ataxia and dysarthria, cochlear implant performance did not decrease at the time of last follow-up. At 1-year, binaural and monaural speech testing were both significantly improved compared with the 1-month testing (bilateral AzBio sentence in quiet: 91%, right-sided CNCw score: 70%, and left-sided CNCw score: 32%).

Conclusions:

Cochlear implantation may offer meaningful auditory rehabilitation in patients with KLHL11-IgG associated rhombencephalitis with relative auditory nerve sparing, despite ongoing disease progression. Further studies are required to evaluate the durability and generalizability of cochlear implantation across the spectrum of disease-related hearing loss.

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