Enterobius vermicularis infection, an oro-faecal transmitted parasitosis, is a frequent finding in infant communities. Enterobius vermicularis is located predominantly in the caecum, appendix, and proximal areas of the ileum and colon, even if reports of some rare extra-intestinal cases have appeared in the literature. The case is reported here of a 62-year-old male presenting a mass in the right submandibular triangle. Histological examination, following removal of submandibular gland, revealed a granulomatous sialoadenitis due to Enterobius vermicularis. A review of the international literature confirms that this is a very rare site and it would appear to be the first report concerning enterobiasis in the salivary glands. Authors advance a hypothesis concerning a possible pathogenic mechanism.