Many patients with mental retardation have a limited awareness of abnormalities in the oral cavity, making early detection of oral cancer difficult. Moreover, during examination and treatment, these patients may not understand well-intentioned verbal explanations and may express their anxiety through nonverbal communication. Herein we discuss a case in which favorable results were obtained when surgical treatment was performed on a maxillary gingival carcinoma patient with mental retardation. The patient was a 61-year-old man who was admitted to our hospital with maxillary gingival pain. A biopsy revealed well-differentiated oral squamous cell carcinoma(cT4aN0M0, Stage ⅣA). Hemi-maxillectomy was performed and postoperative recovery was uneventful. A maxillary prosthesis was provided for oral rehabilitation. At the 5-year follow-up, there were no signs of recurrence or metastasis. This case illustrates the importance of collaboration between oral maxillofacial surgeons and specialists from related departments for the comprehensive management of patients with mental retardation.