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Indian Journal of Cancer, Vol. 47, No. 4, October-December, 2010, pp. 475-477 Letter to Editor Double primary-Lymphoepithelioma-like carcinoma of the parotid and papillary carcinoma of the thyroid R Bishnoi1, S Kaushal2, MC Varma1, NK Shukla1, R Ray2 1 Department of Surgical Oncology, Dr. BRA Institute Rotary Cancer Hospital, New Delhi - 110 029, India Correspondence Address: Code Number: cn10114 PMID: 21131769 DOI: 10.4103/0019-509X.73557 Sir, Lymphoepithelioma-like carcinoma (LELC) of the parotid gland is a rare tumor. [1] Double primary involving parotid and thyroid are extremely rare. [2] A 60-year-old female presented with enlarged right parotid, enlarged right lobe of thyroid, and right cervical lymphadenopathy. Computed tomography (CT) scan revealed mass lesion involving right parotid [Figure - 1a], enlarged right lobe of thyroid [Figure - 1b], and bilateral multiple pulmonary metastases [Figure - 1c]. Nasopharynx was normal. Fine-needle aspiration cytology (FNAC) from right parotid was suggestive of non-Hodgkin′s lymphoma. Right cervical lymph node biopsy was suggestive of papillary carcinoma of the thyroid (PCT). CT scan showed an enlarged parotid, suggestive of solid lesion in deep lobe of the right parotid gland; however, FNAC, which was positive for non-Hodgkin′s lymphoma and image-guided biopsy done twice were inconclusive. Although FNAC is not conclusive in making a diagnosis of non-Hodgkin′s lymphoma, which requires a biopsy correlation, it was indicating that the patient has got some malignant tumor of the parotid gland, reported as non-Hodgkin′s lymphoma on FNAC. So provisional diagnosis of double primary-lymphoma of the right parotid, and PCT was done. Although the treatment for non-Hodgkin′s lymphoma is chemoradiotherapy, surgery followed by radiotherapy or chemoradiotherapy is also an alternative treatment for localized extranodal non-Hodgkin′s lymphoma particularly in such cases where surgery has both diagnostic and therapeutic role. This patient already has got a biopsy-proved PCT, treatment of choice for which is surgery, and both the procedures for thyroid and parotid can be done in one sitting under the same general anesthesia. Minimal procedure for parotid tumor is superficial parotidectomy, but in this case tumor was involving only the deep lobe and superficial lobe was normal, so the procedure planned for parotid tumor was facial nerve sparing total parotidectomy. Patient underwent nerve preserving right total parotidectomy, total thyroidectomy, right modified radical neck dissection Type I, central compartment clearance, and left anterolateral neck dissection. Postoperative period was uneventful. Microscopy of parotid tumor showed irregularly shaped islands, trabeculae, and syncytial masses of eosinophilic epitheloid cells widely separated by lymphocyte-rich stroma. Dense lymphocytic infiltrate surrounded nodules of carcinomatous cells and was composed of mixed B and T cells. Immunohistochemical analysis showed that the epithelial cells were strongly positive for pancytokeratin [Figure - 2a] Inset and epithelial membrane antigen. Overall features were suggestive of LELC [Figure - 2a], whereas microscopic examination of thyroid tumor revealed follicular arrangement with cytological features of PCT. On immunohistochemical analysis, tumor cells stained positive for CK 19 [Figure - 2b (Inset)]. Overall features were suggestive of follicular variant of PCT [Figure - 2b]. Ten lymph nodes were isolated from the right side neck dissection specimen, 3 showed metastasis from LELC [Figure - 2c], whereas 2 had metastasis from PCT [Figure - 2d]. Central compartment specimen also showed metastasis from PCT. Patient also investigated for Epstein-Barr virus (EBV) infection as LELC is known to be associated with this infection. She was strongly immunoreactive for EBV-IgG antibodies (patient - 823 mg/ml, N < 12 mg/ml). Final diagnosis of double primary-LELC of the right parotid and follicular variant of PCT was made. Whole body radioiodine uptake scan revealed increased uptake in the mediastinal lymph nodes and in the bilateral lung fields [Figure - 1d] confirming the diagnosis of lung metastasis from PCT. Patient underwent radioiodine ablation of lung metastasis and received postoperative radiotherapy to local site for LELC of the parotid. LELC was first described by Hilderman et al, stating that it mainly occurs in the nasopharynx. [1] It is a rare subtype of undifferentiated tumors of the parotid gland (<0.1%), [3] almost exclusively found among Eskimos and Chinese population, [4] show familial clustering [4] and female preponderance. This tumor mainly occurs in adult females-presents as unilateral mass in the parotid (or submandibular gland). Metastasis to regional lymph nodes is common but distant metastasis to lungs, liver, and bone also occurs. Metastasis to distant sites usually occurs during follow-up and more likely in patients already having neck node metastasis. [5] Histology reveals that LELC is composed of islands of atypical, large, epithelial cells accompanied by a benign dense lymphoplasmacytic infiltrate. [4] It is strongly positive for keratin on immunohistochemistry. [4] Often, seropositivity to EBV is present. [4] Standard treatment is surgery (removal of primary tumor with neck dissection) followed by radiation. [6],[7] This is a very radiosensitive tumor. Chemotherapy may be considered for distant metastasis; [7] common site of failure being distant metastasis. [6] There is a wide variation in the mortality rates but overall prognosis is relatively good with 65% 5-year survival. [4] A patient with a double primary, PCT, and LELC of the parotid was successfully treated in our department. Reports of related cases in the previous literature are rare. LELC is an extremely rare tumor (<0.1% of parotid tumors). Multiple malignancies are also rare. Ours is the first report in English literature of LELC in the setting of double primary. References
Copyright 2010 - Indian Journal of Cancer The following images related to this document are available:Photo images[cn10114f1.jpg] [cn10114f2.jpg] |
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