Case of ductal adenocarcinoma ex pleomorphic adenoma of the lacrimal gland.

Ductal adenocarcinoma of the lacrimal gland is extremely rare; to our knowledge, only seven de novo cases and one case of ductal adenocarcinoma ex pleomorphic adenoma have been reported in the literature. Here, we report a case of ductal adenocarcinoma ex pleomorphic adenoma of the lacrimal gland. A 70-year-old Japanese female received the resection of the recurrent lacrimal gland tumor (second surgery), under the clinical diagnosis of recurrent pleomorphic adenoma, fifteen years after the initial surgical resection. The resected tumor was composed of recurrent pleomorphic adenoma and adenocarcinoma. Adenocarcinoma component showed infiltrative papillo-tubular or microcystic growth of carcinoma cells, which had pleomorphic large nuclei with prominent nucleoli and rich eosinophilic cytoplasm. Large dilated ducts with central comedonecrosis were observed. Accordingly, the diagnosis of ductal adenocarcinoma ex pleomorphic adenoma was made. The tumor recurred locally eight months after the second surgery, and tumor exenteration was performed(third surgery). The histopathological features of the third surgical specimen were as same as those of the second surgical specimen. In the previously reported eight cases and the present case of lacrimal ductal adenocarcinomas, recurrence took place in 5 cases and two patients died from multiple metastases. These data suggests that lacrimal ductal adenocarcinoma appears to have a poor prognosis, similar to salivary duct carcinoma, which is one of the most aggressive salivary carcinoma. And further study is needed to clarify the clinicopathological features of the lacrimal ductal adenocarcinoma.