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ABSTRACT: We report a unique case of laryngeal dystonia in a 43-year-old male with neurosyphilis who underwent successful treatment with botulinum toxin injection. To date there have been no reports of laryngeal dystonia associated with neurosyphilis. The patient initially presented with strained and stuttering voice despite systemic penicillin therapy. After 2 months of speech therapy with limited relief, the patient received botulinum toxin injection to each thyroarytenoid muscle. Postinjection videostroboscopy showed marked improvement of voice quality.