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Although craniopharyngiomas account for a large percentage of pediatric intracranial tumors, there is a bimodal age distribution. Most of these neoplasms are suprasellar or sellar in location. In this report we describe an unusual case of an infrasellar craniopharyngioma in a child. Only four previous cases of infrasellar craniopharyngiomas with no sellar involvement have been described. Infrasellar craniopharyngiomas are part of the continuum representing intracranial craniopharyngiomas and ameloblastomas of the jaw. A transnasal endoscopic biopsy was performed with a preliminary diagnosis of craniopharyngioma. The patient then underwent a radical resection of the infrasellar tumor via a subfrontal transbasal approach. This case illustrates a rare and unusual location for a craniopharyngioma. Craniopharyngiomas should be considered in the differential diagnosis of infrasellar neoplasms. Infrasellar craniopharyngiomas compromise part of the spectrum of tumors originating from enamel-forming neural crest cells.