Objective This is the first description of long-term follow-up of a case of Freeman-Sheldon syndrome. Microstomia was treated with a mouth expander for 2 to 3 hours per day before active orthodontic treatment. Separate impressions were necessary in each quadrant of both upper and lower jaws because of limited mouth opening. Conclusion Orthodontic treatment improved the patient's Class II malocclusion, which was accompanied by crowding and a deep bite.