A characteristic case of melanotic neuroectodermal tumor of infancy is reported and the literature regarding this lesion is reviewed. The case reported shows the salient features of previously described melanotic neuroectodermal tumors of infancy; it occurs in infants of less than 1 year of age; it most commonly arises from bone (jaws and skull), has distinctive gross and histologic features, is benign, and is cured by total local excision. Since the teeth in the mandible and maxilla are undergoing development about the time that this tumor occurs, it is not surprising to find tooth buds in close proximity to the expanding neoplastic mass. When odontogenic rests are found in association with the lesion, it must be assumed that it is a collision of the two structures and not that the odontogenic epithelium is the origin for the lesion as previously thought. This is especially true since odontogenic epithelium is not found in the tumor when it occurs outside the jaws. This is the type of lesion that will most probably first be noticed by a dentist and should be managed by the oral surgeon. It is important to note that proper advice regarding future dental problems should be given to the parents of the infant. Such possibilities as crowding or absence of the deciduous or permanent teeth, malocclusion, and possible malformation of the remaining maxillary ridge should be explained. Close dental follow-up is mandatory since recurrence is possible.

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