[Cervical lymphangioma in the adult: a report of a significant case]

Lymphangioma in the adult is quite rare: the most common localization in the head and neck region is at the posterior triangle of the cervical lateral region (75%). Moreover, it favors the right latero-cervical area (72%) thus supporting a disembryogenetic pathogenesis. Indeed, according to Dowd and Goetsch, the lymphatic tissues are rearranged in this area, creating the principle head and neck drainage. The presence of disembryogenetic alterations in the lymphatic architecture could go on unnoticed until the lymphatic circulation equilibrium is altered by some anatomo-functional event compromising the involved area.

G.P., a 41-year-old male, presented a swelling in the right submandibular region since approximately 4 months and which had increased in size over the last 30 days. The case history revealed that, at the age of 18 he had been admitted to the hospital for surgical removal of an angiomatose phlegmonous neoformation while, at the age of 18 38, he had undergone surgery for a lipoma in the right temporo-zigomatic area. Head and neck CT using a contrast medium revealed a formation of mixed density, predominantly liquid, apparently multiloculated. The patient underwent surgery, via a right submandibular cervicotomy approach, to remove the cystic neoformation and attached submandibular gland. In addition, the plurilobate mass showed submentonier, parapharyngeal, subdigastric and subhyoid extensions. Histology led to a diagnosis of "cystic lymphangioma". The clinical case reported is quite unusual in that:--the submandibular (anterior triangle) location is rare, accounting for only 6.25% of the cervical manifestations;--the previous case history of this patient underlines the importance of an embryogenic alteration in the regional lymph network;--this condition, clinically manifest in infancy as a submandibular (angiomatose) phlegmon became latent in adulthood after surgery for right temporo-zigomatic lipoma. In light of the above data, it can be hypothesized that a disembryogenetic origin of the adult lymphangioma, associated with a predisposition (scarred areas further compromising the lymph vessel architecture) and some precipitating event (surgical trauma in adulthood), have lead to delayed development of the amartoma.