Abstract
Oncocytic carcinomas of the salivary glands are rare. The submandibular gland is a rare location. It is usually associated with a poor prognosis and recurrence after treatment. We report a case of a 52‐year‐old man with a lymph node and brain metastasis recurrence of an oncocytic carcinoma of the submandibular gland.
Keywords: brain metastasis, lymph node metastasis, oncocytic carcinoma, submandibular gland
Oncocytic carcinoma of the submandibular gland is a rare but aggressive tumor which require long‐term follow‐up as recurrence can occur even after years.
1. INTRODUCTION
Oncocytic carcinomas of the salivary glands are uncommon. It accounts for 0.5% of all epithelial salivary gland malignancies. 1 It occurs usually in the parotid glands, but some cases of oncocytic carcinoma of the submandibular glands (OCSG) have been reported. It is an aggressive tumor. Local recurrence and metastasis are frequent. We report a case of a 52‐year‐old male with an OCSG and recurrence with cerebral metastasis after initial treatment.
2. METHOD
We report a case of an oncocytic carcinoma of the submandibular gland with lymph node and brain metastasis recurrence 3 years later.
3. CASE PRESENTATION
A 52‐year‐old man, alcoholic and smoker, with no notable medical history, consulted with a complaint of a swelling under the left jaw evolving since 1 year.
The clinical examination showed a 1.5 cm swelling in the left submandibular region. It was soft, painless without local inflammatory signs. Lymph nodes were free, and the rest of the clinical examination, particularly the bronchopulmonary examination, was normal.
Biological analysis was normal. Cervical ultrasound revealed a poorly limited, hypoechoic nodule on the outer edge of the left submandibular gland. It measures approximately 9.9 × 13.6 mm.
He was operated on. He underwent a left submandibulectomy. The pathological examination of the surgical specimen showed on gross examination, a whitish 1.5 cm tumor. Microscopic examination showed a carcinomatous proliferation in favor of an oncocytic carcinoma of the submandibular gland which was infiltrated by a carcinomatous proliferation of trabecular and cord‐like architecture without visible tubular structures. The tumor cells are polyhedral, with a vesicular, strongly nucleated nucleus, an abundant eosinophilic, and finely granular cytoplasm. It is accompanied by a fibrous reaction and numerous perineural sheaths (Figure 1).
FIGURE 1.
(HE 100×/200×): Infiltration of the salivary parenchyma by a carcinomatous proliferation of trabecular and cordonal architecture without visible tubular structures. The tumor cells are polyhedral, provided with a vesicular and strongly nucleolated nucleus and an abundant eosinophilic and finely granular cytoplasm.
Three years later, brain metastases revealed by headaches were diagnosed. The patient was operated on. Pathological examination of the surgical specimen revealed a brain metastasis of a CK7+ moderately differentiated carcinoma. A CTAP scan was performed in search of the primary tumor, showing suspicious left latero‐cervical adenopathies in sectors II, III, IV, and V.
The patient was reoperated with intraoperative findings of calcified adenopathies adherent to the posterior border of the sternocleidomastoid muscle and to the left jugular vein, creating a true cervical shield. An adenectomy of two sector III adenopathies was performed with morphological aspects and an immunohistochemical profile consistent with a lymph node location of the oncocytic carcinoma previously diagnosed in the submandibular gland: The lymph node parenchyma infiltrated by a malignant tumor proliferation arranged in clusters, cords, and trabeculae with a sparse, richly vascularized stroma of endocrinoid appearance. The tumor cells are large, polyhedral, with abundant, slightly granular eosinophilic cytoplasm. The nuclei are enlarged, vesicular, and frankly atypical, with a prominent eosinophilic nucleolus (Figure 2). There are numerous mitotic figures and several lymphatic emboli. Immunohistochemical staining was positive for CK7 and negative for CK20, P63, TTF1, CDX2, GATA3, and thyroglobulin.
FIGURE 2.
(HE 100×): Lymph node metastasis from oncocytic carcinoma of the submandibular gland.
It was thus an aggressive oncocytic carcinoma of the submandibular gland with lymph node and brain metastasis recurrence.
4. DISCUSSION
Oncocytic carcinoma of salivary glands have been first described by Bauer and Bauer in 1953. 2 It is rare accounting for 0.5% of all epithelial salivary gland malignancies and 0.18% of all epithelial salivary gland tumors. 1 It occurs usually in the parotid glands. The OCSG is a very rare entity. Only few cases have been reported in the literature.
Epidemiological features are difficult to precise because of the relatively few cases in the literature. It seems more frequent between the fifth and sixth decade with a male‐to‐female ratio of 1 as reported by most of series like those of Thompson et al. 3 and Nakada et al. 4
Most patients present with a painless left sub‐mandibular mass.
On gross examination, the tumor can be partially cystic, multicystic with solid foci, or completely solid. 5
Histologically, oncocytic carcinomas are composed of malignant oncocytes with adenocarcinomatous phenotypes, infiltrative qualities and usually invade muscle, lymphatics, and nerves. 6
The World Health Organization histological typing of salivary gland tumors determined the diagnostic indicators of oncocytic carcinoma arising in the salivary gland which are (1) a lack of encapsulation, (2) regional nodal or distant metastasis, (3) intravascular, lymphatic, or perineural invasion, and (4) mitoses or cellular pleomorphism. 3
The oncocytic differentiation can be demonstrated by using immunohistochemical stains that reveal positivity for antibodies against mitochondrial antigen, keratin (Citok AE1, Citok AE3), and alpha‐1‐antichymotrypsin.
The differential diagnosis includes many tumors with oncocytoid cells, especially mucoepidermoid carcinoma and acinic cell carcinoma. The first shows a diffuse positivity of p63 on immunochemistry staining, and the second is positive for SOX10 (SRY‐related HMG‐box 10). 7
Non‐neoplastic cystic swellings of the submandibular region should also be considered: submandibular branchial cyst, dermoid/epidermoid cyst, mucocele, hydatid cyst, cystic hygroma, and salivary duct cyst. 8
Treatment consists of a complete surgical resection of the tumor mass and its metastases. Adjuvant radiotherapy helps sometimes reach a local control. 9 Chemotherapy is usually ineffective and rarely considered. 10
Oncocytic carcinoma of the submandibular glands is characterized by high rates of local recurrence and lymph metastatic nodes. Goode and Corio reported that small OC has a better prognosis with a cut‐off of two centimeters. 11
Nakada et al indicated distant metastasis as the most significant prognostic indicator. 4
In our case, despite a size of 1.5 cm, a recurrence with lymph metastatic nodes and cerebral metastases occurred.
5. CONCLUSION
Oncocytic carcinoma of the submandibular gland is a rare tumor with a poor prognosis. Even after surgical treatment, a close monitoring is mandatory to detect recurrence.
AUTHOR CONTRIBUTIONS
SN and GG: Wrote the paper. GG: reviewed the literature. SN, AD, AC, and MKM: Contributed to the pathology pictures as well as the interpretation of figures. HB: Contributed to the therapeutic decisions. EC: The head of the pathology department at the Mohamed Taher Maamouri Hospital.
FUNDING INFORMATION
None.
CONFLICT OF INTEREST
None.
CONSENT
Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.
ACKNOWLEDGEMENT
Published with consent of the patient.
Nechi S, Gharbi G, Douggaz A, et al. Recurrence with lymph node and brain metastasis of an oncocytic carcinoma of the submandibular gland: A case report. Clin Case Rep. 2022;10:e06728. doi: 10.1002/ccr3.6728
DATA AVAILABILITY STATEMENT
Patient's personal data have been respected. No data are available for this submission.
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Data Availability Statement
Patient's personal data have been respected. No data are available for this submission.