In the April issue of this journal, we noted with interest a case report by Nel et al. [1] of multiple adenomatoid odontogenic tumors (AOTs) in a 9-year-old male patient with linear epidermal nevi and unilateral oral mucosal papillomatosis, highly compatible with Schimmelpenning syndrome. In their Discussion section, the authors referred to two previously published English-language reports of syndromic multiple AOTs.
Their report was timely, as at about the same time (March 2021) we published a brief literature review that included an overlooked meeting abstract describing multiple AOTs associated with epidermal nevus syndrome in 1980 [2]. It is worth noting that this, in fact, was the first published description of this condition by a Japanese group of oral surgeons and pathologists [3]. The patient was a 3-year-old boy who presented with linear epidermal nevi on the left upper lip and cheek, multifocal papillomatous growths on the left buccal mucosa and soft palate, and two follicular AOTs in the left maxilla containing a deciduous canine and first deciduous molar, respectively. The former tooth exhibited enamel hypoplasia [3]. Unfortunately, this Japanese-language abstract appeared in the local domestic scientific literature [3] and our English-language report was also not indexed in PubMed [2]. We would like to direct any interested readers to our historical review on syndromic multiple AOTs [2], and hopefully this notification will be of assistance to radiologists, surgeons and pathologists intending to submit reports on this topic in the future.
