|Ahead of print publication
Bilateral parotid gland metastasis from a breast invasive ductal carcinoma
Syrine Ben Dhia1, Imtinene Belaid1, Wided Stita2, Makrem Hochlaf1, Faten Ezzairi1, Slim Ben Ahmed1
1 Department of Medical Oncology, University of Sousse, Medical School of Sousse, Farhat Hached Hospital, Sousse, Tunisie
2 Department of Pathology, University of Sousse, Medical School of Sousse, Ibn El Jazzar Hospital, Kairouan, Tunisie
Syrine Ben Dhia,
Rue Ibn El Jazzar, Immeuble Heyla 4eme Etage Aprt Numero 4, 4000 Sousse
Source of Support: None, Conflict of Interest: None
Metastases to the parotid gland are very rare. We report the second case of bilateral metastases to the parotid gland from a breast invasive ductal carcinoma. A 50-year-old female was treated for an early left breast cancer in 2007. A pulmonary metastatic relapse was diagnosed in 2013. A metastatic skin extension required several lines of treatment from June 2014 to July 2016. Bilateral parotid gland metastases from a breast invasive ductal carcinoma were confirmed in December 2016. The patient died on May 2017 from cerebral metastases. Only 16 cases of metastasis to the parotid gland from breast cancer have been reported in the literature. Only one case had a bilateral involvement. Prognosis is poor, and there are no specific guidelines for the treatment.
Keywords: Breast cancer, distant metastasis, parotid gland
| > Introduction|| |
Metastatic salivary gland tumors are rare clinical events. Metastasis to the parotid gland from breast cancer is very rare. We report a case of carcinoma of mammary invasive duct carcinoma with bilateral parotid metastases. To the best of our knowledge, it is the second case published in the literature.
| > Case Report|| |
A 50-year-old woman was referred to our institution with a left-sided breast lump in July 2007. A core biopsy was performed. Histology showed a Grade 2 Bloom–Richardson invasive ductal carcinoma with positive hormone receptors and HER2 negative. There was no evidence of metastatic disease. Clinical stage was T4bN0M0. The patient received four cycles of neoadjuvant chemotherapy fluorouracil epirubicin cyclophosphamide 100 (fluorouracil 500 mg/m2, epirubicin 100 mg/m2, and cyclophosphamide 500 mg/m2) and underwent a radical left mastectomy with lymph node dissection. Histological stage was pT4bN1. She received four cycles of adjuvant chemotherapy paclitaxel followed by a locoregional radiotherapy and a selective estrogen receptor modulator (Tamoxifen®) for 2 years.
The patient was lost to follow-up. She came back in 2013. A pulmonary metastatic relapse was diagnosed. An anthracycline-based chemotherapy was indicated. She received five cycles of anthracycline chemotherapy leading to a partial response, followed by an aromatase inhibitor therapy. A metastatic skin extension required several lines of treatment (paclitaxel, estrogen receptor modulator, and nonsteroidal aromatase inhibitor) from June 2014 to July 2016 with an initial response followed by a clinical progression.
In December 2016, after 11 months of clinical stability, the patient presented with a progressive painless swelling in the right parotid gland. There was a 1.5 cm × 2.0 cm preauricular tumor palpable over the area of the right parotid. The mass was slightly tender, elastic, and adherent to the surrounding tissues. The overlying skin was normal, and regional lymph nodes were not palpable. [Figure 1] Ultrasonography study revealed a nodular heterogeneous ill-defined lesion of the right parotid gland not reveled by physical examination [Figure 2] fine needle aspiration biopsy reveled the presence of malignant celles of breast origin [Figure 3] and [Figure 4] fine needle aspiratio.
|Figure 1: Patient presenting at physical examination with a mass of the right parotid of 5×2 cm, irregular and firm, not painful at palpation with skin involvement|
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|Figure 2: Ultrasonographic study revealed a nodular heterogeneous ill-defined lesion of the right parotid gland of 21 X 22 X 14 mm. A similar lesion was found in the left parotid gland measuring 10 X 9 X 6 mm. It was an unexpected discovery on the ultrasonography, physical examination did not reveal any abnormality on the left parotid gland. We did not proceed to a biopsy. The lesion was considered secondary radiologically considering the clinical context.|
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|Figure 3: (a) Parotid: invasive ductal carcinoma arranged in sheets and small nests (H and E, ×100), (b) Parotid: middle-sized cells with abundant basophilic cytoplasm, vacuolar in places with irregular hyperchromatic nuclei. Abundant mitotic figures are present (H and E, ×400|
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|Figure 4: Parotid: large and intense immunostain with anti-estrogen receptors antibody (a) and antiprogesterone antibody (b) the nuclei of the malignant cell are highlighted in brown|
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A radical parotidectomy was not possible. The patient received three cycles of palliative chemotherapy with capecitabine and vinorelbine with extension of the metastatic skin nodules and a bone scan showing multiple bone metastases.
A hemostatic radiotherapy was necessary to control a local bleeding of the right parotid invasion, and a chemotherapy with gemcitabine was indicated. Few weeks later, the patient presented tonic–clonic seizures. Cerebral metastases were diagnosed, but the patient died in May 2017.
| > Discussion|| |
Most salivary gland tumors are benign (80%) and occur in the parotid glands. Salivary gland cancer is rare and represents 6% of all head-and-neck tumors. The most commonly involved salivary gland is the parotid gland. Parotid gland metastases are rare and are reported to arise from head-and-neck tumors. They are usually secondary to a melanoma or a squamous cell carcinoma. The most common sites where breast cancer spreads are bone, lung, liver, and brain.
Parotid gland metastases from breast carcinoma are very rare. The anatomical location of the parotid gland can be the reason for this rarity. The metastatic process is not yet elucidated. There are three routes of spread for the parotid metastasis metastasis: locoregional extension from a primitive tumor, through the lymph ducts for head-and-neck cancers or hematogenous dissemination from distant solid tumors: lung cancer, renal cancer, and rarely breast cancer which is the most likely route of spread in this case., Only 16 cases have been reported in the literature from 1982 to 2017 among which 14 were reported in 2011 in a review of the literature.
Mean age of the patients was 55 years (41–71 years). Thirteen patients presented with a mass or a swelling of the parotid gland, which is the typical presentation of parotid gland tumors. Only four patients presented with a palsy of the facial nerve, suggesting a malignant lesion. Our patient is the second case of bilateral parotid metastasis from breast cancer. Patients had exclusive parotid metastases except for three cases.
Ultrasonographic study is the most common imaging procedure. Findings are usually not specific showing a poorly defined heterogeneous mass with a hypervascular area. Diagnosis is usually made from a fine-needle aspiration biopsy. The distinction between a primary salivary duct carcinoma and a metastatic ductal breast carcinoma is a challenge for the cytopathologist because of morphological and immunocytochemical similarities.,
The absence of hormone receptors expression suggests a primary tumor of the parotid gland. Computed tomography and MRI allow distinction between malignant and benign tumors and are helpful in determining the locoregional extent and the involved lymph nodes.6 Fluorodeoxyglucose-positron emission tomography may improve staging and is recommended for the locoregional evaluation in case of exclusive parotid gland metastasis. However, radiological workup cannot distinguish a primary malignant gland tumor from a metastasis.
There are no reliable treatment modalities for parotid metastases. Total or partial parotidectomy for exclusive parotid metastases is still controversial, and adjuvant radiotherapy or chemotherapy is a matter of debate. Some authors, recommend a parotidectomy followed by radiotherapy for tumors restricted to the parotid gland. For Shuang Shi and QiGen Fang, partial parotidectomy with preservation of facial nerve followed by chemotherapy could be indicated for metastatic tumors to the parotid gland. However, Jecker and Hartwein stated that parotid surgery would not improve patient survival. Among the reported cases, 12 patients had a partial or a total parotidectomy. Two of them did not receive any complementary treatment. Four patients did not have surgery and were treated with exclusive radiotherapy or a combination of radiation and chemotherapy. Our patient had a non resectable tumor because of the local invasion. She received a hemostatic radiotherapy and a palliative chemotherapy. A personalized treatment is needed adapted to the individual situation and the disease course of the patient.
This site of metastases reflects a disseminated disease like in this case where the patient had a pulmonary metastatic relapse 3 years before and developed skin and bone metastases few months after the diagnosis of metastases to the parotid gland. This might explain the poor prognosis of those patients with a 5-year survival rate of only 10%.
| > Conclusion|| |
Despite the fact that parotid metastases are a rare clinical event, any parotid mass in a patient with a prior history of a malignancy must be considered as metastatic and investigated until proven otherwise. Given the rarity of this entity, there are no specific guidelines for their management. Prognosis is poor with a 5-year survival of 10%.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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Conflicts of interest
There are no conflicts of interest.
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[Figure 1], [Figure 2], [Figure 3], [Figure 4]