Patients with autism spectrum disorders (ASDs) may have difficulty tolerating conventional dental treatment due to aberrant sensory responsiveness. The purpose of this report was to describe the successful treatment of obstructive sleep apnea (OSA) in a nonverbal 20-year-old male patient with ASD using a dental appliance. A series of appointments prepared the patient for the required treatment procedures and desensitized him for use of the final appliance. The final appliance improved outcomes of a post-treatment sleep study, indicating successful treatment of OSA. Understanding the specific challenges of patients with ASD and the patience and foresight of providers in approaching these challenges, in collaboration with caregivers, can contribute to improved health outcomes for these patients.