Case Report - (2023) Volume 14, Issue 3
Received: 15-Apr-2023, Manuscript No. jch-22-82233;
Editor assigned: 17-Apr-2023, Pre QC No. P-82233;
Reviewed: 28-Apr-2023, QC No. Q-82233;
Revised: 03-May-2023, Manuscript No. R-82233;
Published:
10-May-2023
, DOI: 10.37421/2157-7099.2022.13.688
Citation: Chen, Hsiao-Ching. “Metastatic Carcinoma ex Pleomorphic Adenoma in Pleural Effusion.” J Cytol Histol 13 (2023): 688.
Copyright: © 2023 Chen HC. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
Carcinoma ex pleomorphic adenoma (CXPA) arising from primary or recurrent pleomorphic adenoma is characterized by the presence of longstanding palpable mass with growth spurt mainly in the parotid region. Overall, patients with CXPA have a poor prognosis. Pleural effusion involved by CXPA is precedingly rare, compared to that common dissemination from pulmonary or mammary origin. Here we reported a metastatic CXPA in pleural effusion from an 82-year-old male. The clinical presentation, cytological morphology, and immunohistochemistry confirmed the diagnosis. When dealing patients with prolonged history of pleomorphic adenoma, underwent recent rapid growth of malignant transformation and malignant cells presented in the pleural effusion, metastatic CXPA should be seriously considered.
Carcinoma ex pleomorphic adenoma (CXPA) • Pleural effusion • Immunohistochemistry
Carcinoma ex pleomorphic adenoma (CXPA) defined as: a pleomorphic adenoma in which an epithelial malignancy is derived, is a rare, aggressive, poorly understood malignant neoplasm of salivary gland [1]. CXPA constitutes approximately 3.6% of all salivary gland tumors, 5% of pleomorphic adenomas undergo malignant transformation, and 11.6% of all malignant salivary gland neoplasms. It is a distinctive but relatively common high-grade adenocarcinoma arising from the excretory ductal epithelium of the major salivary gland, especially the parotid gland [1-3]. The clinical findings typical of this tumor include history of a slow growing, ulcerated, painless mass that enlarges rapidly; aggressive and infiltrative behavior with local recurrence, distant metastasis, or significant mortality [1-5].
Metastasis of salivary gland neoplasms to pleural effusion is an unusual finding. Cases of mucoepidermoid carcinoma involving peritoneal and pericardial fluid and adenoid cystic carcinoma metastasizing to the pleural fluid had been reported before [6-8]. In this article, metastatic CXPA in pleural effusion presented here and hignlighted the diagnostic challenges on a cytology specimen.
An 82-year-old male presented with receiving excision of pleomorphic adenoma at right parotid 35 years ago and an enlarged lymph node was found at right supraclavicular area recently. CT revealed a 2 cm heterogeneously enhanced tumor at right parotid tail. Fine needle aspiration was then performed. The Pap-stained smears showed a mixed population of cells composed of benign and malignant elements. Some clusters showed a metachromatic stroma in a background. The benign epithelial cells component appeared as small round to oval cells with bland oval nuclei. The malignant population consisted of large cells with large pleomorphic nuclei having coarse chromatin and high nucleo-cytoplasmic ratio. Malignant change occurring in a background of pleomorphic adenoma was suggested. Based on the abovementioned cytological features,malignant transformation from previous right parotid tumor was diagnosed (Figure 1).
Figure 1. FNA smear showing malignant epithelial cells with pleomorphic nuclei, coarse chromatin and increased N/C ratio (Papanicolaou’s stain, original magnification x20).
Sequential study revealed multiple distant metastases to rib, T-spine, mediastinal lymph node and subsequently pleural effusion was sent for cytological study and cell block was made at the same time for further investigation.
The cytology report showed some clusters of plump hyperchromatic oval neoplastic cells with vesicular nuclei and pinkish cytoplasm (Figures 2 and 3). The tumor cells showed positive staining for CK7, lysozyme and occasionally positive for CEA (Figure 4) while negative for TTF-1, S100 and actin. The features suggested metastatic CXPA, compatible with salivary duct origin.
Figure 2. Pleural effusion cytology showing ball-like clusters of neoplastic cells (Papanicolaou’s stain, original magnification x10 in A, x40 in B).
Figure 3. Cell block preparation illustrating clusters of plump hyperchromatic neoplastic cells occasionally with lumens within the clusters (H&E, original magnification x20).
Figure 4. Immunohistochemical stain showing positive staining for CK7 (A) Lysozyme, (B) and (C) CEA (original magnification x20).
He received salvage radiotherapy with partially shrinkage of the tumor. General condition became worse after radiotherapy and he passed away after completion of radiotherapy due to pneumonia.
We thought that our case was a carcinomatous transformation in pleomorphic adenoma involved by pleural fluid. Our case had all the features of malignant transformation along with local and distant metastasis. Multiple distant metastases have been reported, including lung, breast, gingiva, vagina and inguinal lymph node [9-14]. Immunoprofiles were believed to be of potential assistance in the diagnosis of salivary gland tumors and in the prediction of histogenesis. The typical clinical presentation in a patient with CXPA is a longstanding history of pleomorphic adenoma and a sudden period of rapid growth. CXPA usually is a more poorly circumscribed mass than benign pleomorphic adenoma; is most likely to occur within the major salivary gland, especially the parotid gland; and typically occurs in patients in the sixth to eight decades of life [4,5].
CXPA has been named as carcinoma ex mixed tumor, carcinoma ex adenoma and carcinoma ex benign pleomorphic adenoma [4,5].
The differential diagnosis of a slow growing parotid mass that has recently exhibited a growth spurt should raise the suspicion of a CXPA. A CXPA can be mistaken for a benign PA. It can also be misdiagnosed as other benign and malignant salivary gland tumors. Cytologic differential diagnosis included mucoepidermoid carcinoma, a high- grade salivary gland adenocarcinoma and metastatic adenocarcinoma which are difficult to classify should include CXPA in its differential [15,16].
The diagnosis of metastatic CXPA in pleural effusion is possible on cytology if presence of unequivocal malignancy in association with features of PA. Correlation with clinical history and careful scrutiny interpretation of the cytomorphologic features help arriving at a conclusive diagnosis. This critical information is necessary for the clinician to plan out the therapeutic modalities for a better outcome.
None.
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