title: Primary ductal adenocarcinoma of the lacrimal sac: the first reported case.
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.
authors.affiliation: Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, Shiga University of Medical Science Shiga, Japan.
authors.forename: Mitsuaki
authors.initials: M
authors.lastname: Ishida
authors.forename: Muneo
authors.initials: M
authors.lastname: Iwai
authors.forename: Keiko
authors.initials: K
authors.lastname: Yoshida
authors.forename: Akiko
authors.initials: A
authors.lastname: Kagotani
authors.forename: Hideaki
authors.initials: H
authors.lastname: Kohzaki
authors.forename: Masahiko
authors.initials: M
authors.lastname: Arikata
authors.forename: Takeshi
authors.initials: T
authors.lastname: Shimizu
authors.forename: Hidetoshi
authors.initials: H
authors.lastname: Okabe
mesh_terms: D048868:Adaptor Proteins, Signal Transducing / Q000032:analysis; D000230:Adenocarcinoma / Q000737:chemistry / Q000188:drug therapy / Q000556:secondary* / Q000601:surgery; D000368:Aged; D000970:Antineoplastic Agents / Q000627:therapeutic use; D014408:Biomarkers, Tumor / Q000032:analysis; D001706:Biopsy; D018797:Cell Cycle Proteins; D005134:Eye Neoplasms / Q000737:chemistry / Q000188:drug therapy / Q000473:pathology* / Q000601:surgery; D005260:Female; D006801:Humans; D007150:Immunohistochemistry; D007765:Lacrimal Apparatus / Q000473:pathology* / Q000601:surgery; D007766:Lacrimal Apparatus Diseases / Q000188:drug therapy / Q000378:metabolism / Q000473:pathology* / Q000601:surgery; D008207:Lymphatic Metastasis; D058990:Molecular Targeted Therapy; D018579:Patient Selection; D010750:Phosphoproteins / Q000032:analysis; D010766:Phosphorylation; D057285:Precision Medicine; D047428:Protein Kinase Inhibitors / Q000627:therapeutic use; D058570:TOR Serine-Threonine Kinases / Q000032:analysis / Q000037:antagonists & inhibitors; D014057:Tomography, X-Ray Computed
chemical_list: D048868:Adaptor Proteins, Signal Transducing; D000970:Antineoplastic Agents; D014408:Biomarkers, Tumor; D018797:Cell Cycle Proteins; C087000:EIF4EBP1 protein, human; D010750:Phosphoproteins; D047428:Protein Kinase Inhibitors; C546842:MTOR protein, human; D058570:TOR Serine-Threonine Kinases
keywords: Ductal adenocarcinoma; lacrimal sac; mTOR pathway
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