Aim: To highlight the potential difficulties in diagnosing neoplastic lesion of the head and neck in children within autism spectrum. Background: Ameloblastic fibromas are a variation of odontogenic tumors that are located in the posterior mandible in 70% of cases. The tumors may be either unilocular or multilocular when observed radiographically. Ameloblastic fibromas tend to have well-defined, scalloped margins radiographically but may also be corticated. In close to 75% of all cases, an impacted tooth is associated with the lesion. Although not confined to patients who are in their first or second decades of life, ameloblastic fibromas most often arise in this population. Case description: A 9-year-old male diagnosed with autism presented with tenderness in the left mandible. The parents were able to elucidate the child's problem as tingling rather than pain. The patient was referred for histopathological diagnosis and treatment. After evaluation and biopsy, the lesion was identified as a pediatric ameloblastic fibroma. He subsequently underwent the conservative approach of marsupialization and curettage without complication. Six-month follow-up revealed no evidence of recurrence and normal eruption patterns of the succedaneous teeth in the affected area. Conclusion:In this specific case, the conservative approach to treatment appears to have been appropriate. This may not be the appropriate course for every case; as such, each case will have an individualized approach. Earlier recognition with careful inspection can reduce potential complications. Clinical significance: We might be missing early diagnosis of ameloblastic fibroma and other significant orofacial neoplasms in patients who are nonverbal or nondescriptive such as those with autism. Moreover, careful inspection of radiographic and clinical signs cannot be overemphasized.