Abbreviations: WHO, world health organization; FNAB, fine needle aspiration biopsy; CECT, contrast-enhanced chest computed tomography; HU, hounsfield units; PET-CT, positron emission tomography -computed tomography
IntroductionPleomorphic adenoma is the most common benign neoplasm of salivary glands. It is considered a biologically benign tumour, but, occasionally, it acquires the capability of metastasizing, locally or even distantly. With regards to this, World Health Organization (WHO) classified three different pathologies, included in the definition of "Malignant Mixed Tumour": the Carcinoma Ex Pleomorphic Adenoma, the Carcinosarcoma, both characterized by a histologically malignant transformation of the epithelial component by itself or in combination with the mesenchymal-like one, respectively, and the Metastatic Mixed Tumour. It has been postulated that both Carcinoma ex Pleomorphic Adenoma and Carcinosarcoma probably results from the genetic instabilities and accumulation of key genetic alterations in long-standing pleomorphic adenoma.1 The Metastatic Mixed Tumour is defined as a histologically benign Pleomorphic Adenoma, typically recurrent, that manifests local or distant metastasis. We present a case of recurrent pleomorphic adenoma developing lung metastases.
Case presentationA 45years-old woman with history of recurrent oral pleomorphic adenoma was referred to the Radiology Department to perform a routine chest x-ray pre-operative examination before a planned excision of a left submandibular swelling (Figure 1), present since about three years and diagnosed as a pleomorphic adenoma at a recent fine needle aspiration biopsy (FNAB). The patient had already undergone surgical excision of pleomorphic adenoma in the left part of the palate in 1980 followed, ten years later, by another excision of recurrent disease in the same site. She was free from comorbidities. At chest x-ray two well-defined radio-opaque nodules in both the lower lung fields were depicted, the biggest one in the right lung, projectively in the retro-cardiac region, with a maximum diameter of about 3cm (Figure 2 Case report: We report the case of a 45years-old woman affected by recurrent pleomorphic adenoma since about the age of 30years. During pre-surgical x-ray chest, performed as routine before excision of the last recurrence of pleomorphic adenoma, bilateral lung nodules were discovered, which ended up to be lung metastases.Discussion: it had been proved by evidence that pleomorphic adenoma recurrences significantly increase the possibility both of malignant transformation of the disease and malignant behaviour recurrences that could develop distant localization, though histologically benign.Conclusion: in management of primary pleomorphic adenoma radical surgical excision is crucial, to prevent first of all local recurrences and then distant spread.