Infantile fibromatosis (IF) is a benign, nonmetastasizing but locally aggressive tumor. Pterygomandibular space is deep and obscure, and IF in this space is seldom reported. We describe a typical case of IF in the pterygomandibular space that occurred in a 3-year-old girl. Intraoral incisional biopsy revealed the diagnosis of IF, and surgical resection of the tumor was performed. We intend to heighten the awareness of this tumor and emphasize the pathologic features and identifications of IF and discuss the diagnosis and treatment of this disease. For the high recurrence risk, follow-up with clinical examination and magnetic resonance imaging in pediatric patients is recommended.
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