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Int J Clin Exp Pathol 2013;6(9):1929-1934

Case Report
Primary ductal adenocarcinoma of the lacrimal sac: the first reported case

Mitsuaki Ishida, Muneo Iwai, Keiko Yoshida, Akiko Kagotani, Hideaki Kohzaki, Masahiko Arikata, Takeshi Shimizu, Hidetoshi Okabe

Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, Department of Otorhinolaryngology, Shiga University of
Medical Science, Shiga, Japan

Received July 4, 2013; Accepted July 21, 2013; Epub August 15, 2013; Published September 1, 2013

Abstract: Primary lacrimal sac tumor is extremely rare, and moreover, glandular tumor is exceptional. Herein, we described the first
documented case of primary ductal adenocarcinoma of the lacrimal sac. A 79-year-old Japanese female presented with persistent swelling of
her left lower eyelid. Computed tomography demonstrated an irregular-shaped tumor involving the left lacrimal sac, lower eyelid, sinonasal
tract, and internal side of the left orbit. Biopsy from the eyelid revealed a poorly differentiated adenocarcinoma. Histopathological study of the
resected lacrimal sac tumor revealed an infiltrative neoplastic growth that was composed of cribriform structures with comedonecrosis. The
neoplastic cells had relatively rich granular eosinophilic cytoplasm and large round to oval nuclei containing conspicuous nucleoli. The left
cervical lymph nodes had metastatic carcinoma. Immunohistochemically, the neoplastic cells were diffusely positive for gross cystic disease
fluid protein-15 and androgen receptor. Moreover, mammalian target of rapamycin (mTOR), 4E-BP1, and p4E-BP1 were expressed. According
to these results, an ultimate diagnosis of primary ductal adenocarcinoma of the lacrimal sac was made. Only 9 cases of primary lacrimal sac
adenocarcinoma have been reported, and this is the first reported case of ductal adenocarcinoma of the lacrimal sac. Ductal adenocarcinoma
of the salivary gland shows an aggressive clinical course, and the present case had multiple cervical lymph node metastases. This report is
the first to demonstrate that mTOR pathway proteins, which are central proteins involved in carcinogenesis, are activated in ductal
adenocarcinoma. Therefore, mTOR inhibitor may be a potential candidate for treatment of this highly aggressive carcinoma. (IJCEP1307009).

Keywords: Ductal adenocarcinoma, lacrimal sac, mTOR pathway

Address correspondence to: Dr. Mitsuaki Ishida, Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, Shiga
University of Medical Science, Tsukinowa-cho, Seta, Otsu, Shiga, 520-2192, Japan. Tel: +81-77-548-2603; Fax: +81-77-548-2407; E-mail:
mitsuaki@belle.shiga-med.ac.jp