Rare Metastatic Lesion Affecting the Mouth: Case Report and Review

Abstract

The presence of metastases in the oral cavity is considered quietly rare in the literature. Considering metastasis of hepatocellular carcinoma, the occurrence occurs in approximately 1% of cases. The objective of this study was to report a case of metastasis of hepatocellular carcinoma in gingiva, in a patient with a previous diagnosis of cancer presenting a poor prognosis. Furthermore, a review of reported cases has already been performed and found only 20 reports. This case report emphasizes the importance of including metastases in soft tissue of the mouth, in the differential diagnosis of lesions with benign aspect.

Share and Cite:

Curi, M. , Curra, C. , Condezo, A. , Rodrigues, M. , Pexe, M. , Moraes, B. , DeAntoni, C. , Moreira, R. and Cardoso, C. (2017) Rare Metastatic Lesion Affecting the Mouth: Case Report and Review. Journal of Cancer Therapy, 8, 399-404. doi: 10.4236/jct.2017.84034.

1. Introduction

Hepatocellular carcinoma (HCC) is considered the sixth most common malignant neoplasm in the world, with 748,000 cases per year [1] . Its etiology is associated with hepatitis A, hepatitis B, hepatitis C and alcohol consumption [2] . Extra-hepatic metastases occur in 50% of cases, predominantly affecting the lung, diaphragm, abdominal lymph nodes and bones [1] . In the oral cavity, metastases are extremely rare [2] and are usually found at an advanced stage of cancer [3] . The first report of oral HCC metastasis was described by Dick et al., in 1957 [4] and to date only 20 cases of gingival metastasis have been found in the literature (Table 1) [1] - [21] . This article has reported an additional case report of oral HCC metastasis in gingiva and a literature review.

Table 1. Case reports of Hepatocellular Carcinoma affecting the gingival tissue.

2. Case Report

A 58-year-old male patient reported pain in the gingiva, which impaired chewing. The patient had a history of hepatitis C infection and an advanced Hepatocellular Carcinoma with pulmonary metastasis 4 months ago. In the intraoral physical examination, there was a vegetative nodular lesion of at least 2 cm, pedunculated, in the gingiva between the teeth 37 and 38, which presented severe mobility. One part of the lesion presented an ulcerated surface, covered by necrotic tissue and, another part, was being traumatized during occlusion (Figure 1). Computed tomography, in a soft tissue window, revealed a well-circum- scribed lesion in the premolar and molar region on the left side. It was also possible to see the juxtaposition of the lesion to the teeth and mandible (Figure 2). The patient presented an advanced stage of Hepatocellular Carcinoma and a pet-scan examination showed hypercaptation in the primary lesion of the liver and areas of lung and jaw metastases (Figure 3). In an MRI, it was possible to identify lesions in the liver and, through a bronchoscopy, pulmonary metastases (Figure 4).

Figure 1. Nodular lesion vegetative with ulcerated surface, pedunculated in the gingiva of the region of the teeth 37 and 38 with severe mobility.

(a) (b)

Figure 2.Computed tomography axial sections. (a) There is a well-defined lesion in the posterior region of the mandible on the left side; (b) In the window for bone tissue, there is absence of bone involvement.

Figure 3. Pet-Scan examination, representing hypercaptation of the primary lesion in the liver, and metastases in the lung and mandible.

(a) (b)

Figure 4. (a) Magnetic resonance imaging reveals hepatic impairment; (b) Bronchoscopy revealing many metastases in the pleura and lungs.

Facing the clinical, imaging findings and the systemic conditions of the patient, the diagnostic hypotheses were: Hepatocellular Carcinoma Metastasis or non-neoplastic proliferative lesion. The proper conduct for the oral lesion would be an incisional biopsy, but since the patient had a prognosis of 2 months of life, the medical and dental team chose to completely remove the lesion and extract the involved teeth, so that the patient could have a better quality of life. The microscopic findings were compatible with malignant neoplasm, demonstrating neoplastic hematogenous infiltration, and epithelial tissue with severe epithelial dysplasia (Figure 5(a)). In order to determine the tissue origin of the lesion, an immunohistochemical examination was performed, in which the result was positive for Hepatocellular Carcinoma metastasis (Figure 5(b) and Figure 5(c)).

3. Discussion

Hepatocellular carcinoma is the sixth most common malignant neoplasm, being more frequent in men than in women [3] - [22] . Chronic infection with hepatitis B virus (HBV) and/or hepatitis C virus (HCV) infection are the main risk factors for the development of this cancer [23] . Extra-hepatic metastasis is a fairly com- mon occurrence, affecting more than 50% of patients frequently in the lungs, abdominal lymph nodes, diaphragm and bone [4] . Metastases in the oral cavity are rare and, when they occur the most common primary site is lung carcinoma, followed by breast carcinoma and renal cell carcinoma. Considering hepatocellular carcinoma, its metastasis to the mouth region is even rarer, accounting for approximately 1% of the cases, the most affected mandibular and gingival tissues [2] [3] [4] [5] .

The clinical presentation of soft tissue metastases is generally similar to the characteristics of benign or reactive proliferative lesions, such as pyogenic granuloma, and peripheral giant cell lesion [3] . However, the microscopic examination is fundamental to define the final diagnosis. In the present case, although the history of cancer was known and the main suspicion was of an oral metastasis, the microscopic examination was fundamental to confirm the diagnosis. In addition, even the microscopic appearance of a malignant neoplasm, the immunohistochemical examination was performed to confirm the origin of the lesion.

Figure 5. Microscopic examination compatible with malignant neoplastic infiltration. (a) The epithelial tissue is observed to be smaller, reacting with a severe dysplasia and below the infiltration of neoplastic cells; (b) A detail of altered cellularity, with nuclear hyperchromatism, mitoses and cellular bizarreism; (c) Immunohistochemical examination revealing the positivity for CEA.

Due to the presence of multiple metastases in vital organs, such as the lungs, the conduct of an excisional biopsy, even if the main diagnosis was a malignant lesion, was taken to offer better quality of life of the patient. We consider maintenance of oral functions fundamental in the palliative phase.

4. Conclusion

As conclusion, it is worth emphasizing in this work that although the present case already presented the recognized primary disease, some cases of metastatic oral lesions may manifest firstly than the primary site of the neoplasia, and it is important never to neglect any lesion removed. During the clinical examination process, anamnesis is fundamental in the differential diagnosis and the conduct of each case.

Conflicts of Interest

The authors declare no conflicts of interest.

References

[1] Alrumaih, R.A., Arian, A.A., Alhedyani, A.A., Al-Zaher, N. and Dababo, M.A. (2015) Hepatocellular Carcinoma First Presenting as a Tumor of the Oral Cavity. Hematology/Oncology and Stem Cell Therapy, 8, 130-135.
https://doi.org/10.1016/j.hemonc.2015.03.001
[2] Terada, T. (2011) Hepatocellular Carcinoma Metastatic to the Gingiva as a First Manifestation of Hepatocellular Carcinoma. Journal of Maxillofacial and Oral Surgery, 10, 271-274.
https://doi.org/10.1007/s12663-011-0187-z
[3] Friedrich, R.E. and Zustin, J. (2010) Multiple Distant Metastases of Hepatocellular Carcinoma to the Oral Cavity. In Vivo, 24, 211-214.
[4] Dick, A., Mead, S.G., Mensh, M. and Schatten, W.E. (1957) Primary Hepatoma with Metastasis to the Mandible. American Journal of Surgery, 94, 846-850.
https://doi.org/10.1016/0002-9610(57)90068-5
[5] Pires, F.R., Sagarra, R., Corrêa, M.E., Pereira, C.M., Vargas, P.A. and Lopes, M.A. (2004) Oral Metastasis of a Hepatocellular Carcinoma. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, 97, 359-368.
https://doi.org/10.1016/j.tripleo.2003.09.018
[6] Greenstein, A., Witherspoon, R., Iqbal, F. and Coleman, H. (2013) Hepatocellular Carcinoma Metastasis to the Maxilla: A Rare Case. Australian Dental Journal, 58, 373-375.
https://doi.org/10.1111/adj.12083
[7] Lapeyrolerie, F.M. and Manhold Jr., J.H. (1964) Hepatoma Metastatic to the Gingiva: Report of a Case. Oral Surgery, Oral Medicine, and Oral Pathology, 18, 365-367.
[8] Radden, B.F. and Reade, P.C. (1966) Gingival Metastasis from a Hepatoma. Oral Surgery, Oral Medicine, and Oral Pathology, 21, 621-625.
[9] Lund, B.A., Soule, E.H. and Moertel, C.G. (1970) Hepatocellular Carcinoma with Metastasis to Gingival Mucosa: Report of Case. Journal of Oral Surgery, 28, 604-607.
[10] Kuga, Y., Kitamura, A., Kusaba, I., Aketa, J. and Yamada, N. (1976) Primary Liver Cancer with Metastasis to the Gingiva: Report of a Case. Japanese Journal of Oral and Maxillofacial Surgery, 22, 541-545.
https://doi.org/10.5794/jjoms.22.541
[11] Yoshida, Y., Tsukuda, T., Yoshinari, M. and Sasaki, H. (1976) Two Cases of Metastatic Tumors to the Mouth. Japanese Journal of Oral and Maxillofacial Surgery, 22, 534-540.
https://doi.org/10.5794/jjoms.22.534
[12] Wedgwood, D., Rusen, D. and Balk, S. (1979) Gingival Metastasis from Primary Hepatocellular Carcinoma: Report of a Case. Oral Surgery, Oral Medicine, and Oral Pathology, 47, 263-266.
[13] Morishita, M. and Fukuda, J. (1984) Hepatocellular Carcinoma Metastatic to the Maxillary Incisal Gingiva. Journal of Oral and Maxillofacial Surgery, 42, 812-815.
https://doi.org/10.1016/0278-2391(84)90351-3
[14] Tokuyama, K., Koike, S., Takashima, S., Moriwaki, S., Uyama, K. and Jinno, K. (1984) A Case Report of Pedunculated Hepatoma with Very Rare Remote Metastases after the Resection. Gan No Rinsho, 30, 174-180.
[15] Kanazawa, H. and Sato, K. (1989) Gingival Metastasis from Primary Hepatocellular Carcinoma: Report of a Case and Review of Literature. Journal of Oral and Maxillofacial Surgery, 47, 987-990.
https://doi.org/10.1016/0278-2391(89)90385-6
[16] Llanes, F., Sanz-Ortega, J., Suarez, B. and Sanz-Esponera, J. (1996) Hepatocellular Carcinomas Diagnosed Following Metastasis to the Oral Cavity. Report of 2 Cases. Journal of Periodontology, 67, 717-719.
https://doi.org/10.1902/jop.1996.67.7.717
[17] English III, J.C., Meyer, C., Lewey, S.M. and Zinn, C.J. (2000) Gingival Lesions and Nasal Obstruction in an Immunosuppressed Patient Post-Liver Transplantation. Cutis, 65, 107-109.
[18] Maiorano, E., Piattelli, A. and Favia, G. (2000) Hepatocellular Carcinoma Metastatic to the Oral Mucosa: Report of a Case with Multiple Gingival Localizations. Journal of Periodontology, 71, 641-645.
https://doi.org/10.1902/jop.2000.71.4.641
[19] Papa, F., Ferrara, S., Felicetta, L., Lavorgna, G., Matarazzo, M., Staibano, S., De Rosa, G., Troisi, S. and Claudio, P.P. (2001) Mandibular Metastatic Hepatocellular Carcinoma: Report of a Case Involving Severe and Uncontrollable Hemorrhage. Anticancer Research, 21, 2121-2130.
[20] Ramón Ramirez, J., Seoane, J., Montero, J., Esparza Gómez, G.C. and Cerero, R. (2003) Isolated Gingival Metastasis from Hepatocellular Carcinoma Mimicking a Pyogenic Granuloma. Journal of Clinical Periodontology, 30, 926-929.
https://doi.org/10.1034/j.1600-051X.2003.00391.x
[21] Arai, R., Otsuka, T., Mori, K., Kobayashi, R., Tomizawa, Y., Sohara, N., Kakizaki, S., Hirokawa, T., Kanda, D., Nakayama, H., Nakajima, T., Takagi, H. and Mori, M. (2004) Metastasis of Hepatocellular Carcinoma to the Supramaxillary Gingiva and Right Ventricle. Hepatogastroenterology, 51, 1159-1161.
[22] Altekruse, S.F., Mcglynn, K.A. and Reichman, M.E. (2009) Hepatocellular Carcinoma Incidence, Mortality, and Survival Trends in the United States from 1975 to 2005. Journal of Clinical Oncology, 27, 1485-1491.
https://doi.org/10.1200/JCO.2008.20.7753
[23] Lasiter, J.C., Liess, B.D., Zitsch III, R.P. and Wieberg, J. (2011) An Expansile Mandibular Mass as the Initial Manifestation of Hepatocellular Carcinoma. Ear, Nose & Throat Journal, 90, E19.

Copyright © 2024 by authors and Scientific Research Publishing Inc.

Creative Commons License

This work and the related PDF file are licensed under a Creative Commons Attribution 4.0 International License.