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A bstract Leiomyoma is a benign soft tissue tumor originating from smooth muscle, rarely seen in the oral cavity, due to the scarcity of this tissue in the mouth. The tumor may occur at any age, without sex predilection. Rare reports of this lesion have been in pediatric patients. The lesion is typically asymptomatic and has slow growth, the final diagnosis is defined by histopathological and immunohistochemical examination. Treatment involves resection of the lesion. The lesion is classified histologically as solid leiomyomas, angioleiomyomas (AL), or epithelioid leiomyomas. We report the case of a 1-year-old female child with a painless papule on the hard palate. Histopathological examination revealed biphasic LA showing proliferation of spindle-shaped nuclear cells in straw-shaped bundles or a concentric design. This is the fourth documented case of AL in early childhood. How to cite this article Bezerra TMM, Chaves FN, Carvalho FSR, et al. Oral Angioleiomyoma in Early Childhood Patient: A Case Report of an Uncommon Lesion and Literature Review. Int J Clin Pediatr Dent 2021;14(6):828–832.