SYNCHRONOUS BILATERAL MULTIFOCAL

CANALICULAR ADENOMA: A CASE REPORT

OF AN UNUSUAL FINDINGGelsomina Mansueto, Jessica Falleti, Chiara Mignogna, Gaetano De Rosa

Dipartimento di Scienze Biomorfologiche e Funzionali, Sezione di Anatomia

Patologica e Citopatologia, Università Federico II, Napoli

• A 78 years old woman presented with a bilateral synchronous swelling both in the right and left nose labial wrinkle occurring about 6 years ago and slowly growing.

• Clinical examination showed, on the right side, two nodules of 4 and 2 cm of maximum diameter, while on the left one another two nodular lesions, both of 1 cm: lesions were mobile at palpation, hard, not painful, with sharp border and covered with intact skin.

• The ultra sonogram study showed in the both left (Fig.1A) and right (Fig.1B) nose labial wrinkle, multiple non-homogeneous isoechoic nodules, with peri- and intra-lesional vascularization at colour-Doppler.

• The patient has been undergone to a surgical excision of all masses through an intra-oral approach.

• On the left side one of the nodules was adherent to Stenone’s duct, so it needed to be first sectioned and then anastomosed to mucosa.

• Histology showed a neoplastic proliferation constituted of double rows of columnar epithelial cells that form branching and interconnecting cords and narrow channels (“canaliculi”) in a very loose stroma (Fig.2).

• Lining epithelial cells varied from cuboidal to tall columnar with amphophilic or eosinophilic cytoplasm and round nuclei; pleomorphism or pleochromatism and mitosis were absent (Fig.3).

• benign neoplasms are the most frequent tumors of minor salivary glands and, among these, canalicular adenoma is second to pleomorphic adenoma

• several cases with multifocal canalicular adenoma are described in literature: most common site is upper lip and buccal mucosa

• none case described a synchronous and bilateral occurrence of a multifocal canalicular adenoma

• multifocal presence of canalicular adenoma could be explained with small tumor island foci adjacent to the main lesion but also within capsule or in around normal salivary tissue

• differential diagnosis should include some benign lesion like multiple epidermal and sebaceous cysts, adnexal neoplasm’s and minor salivary glands pathologies like mucocele or neoplasm’s.

• Histologically it must be distinct by a basal cell adenoma and a polymorphous low grade adenocarcinoma: an immunohistochemical assay could be performed, using a panel with cytokeratins 13 and 14, vimentin and alpha smooth actin

• Clinical behaviour is generally benign with a low tendency to recurrence and no locally aggressive

• Our case recurred after one year: we retain that this could been explained both with multiple small tumoral foci within surrounding tissues (multifocal tumor ), both with intra-oral surgical approach that presented much more technical difficulties than to extra-oral approach, especially when, on surgery, one lesion appeared adherent to left Stenone’s duct.

• So we conclude that canalicular adenoma is a benign neoplasm, can be find also in nose labial wrinkle, can be both synchronous and multifocal and bilateral, a surgical excision is an appropriate approach, but an accurate histological study is necessary to exclude a malignancy, accompanied by a long follow-up

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