|ORIGINAL RESEARCH REPORT
|Year : 2018 | Volume
| Issue : 2 | Page : 107-111
Metastatic oral and maxillofacial tumors in a tertiary referral hospital: Retrospective analysis of eight cases and review of the literature
Ramat Oyebunmi Braimah1, Abdurrazaq Olanrewaju Taiwo2, Adebayo Aremu Ibikunle1, Olajide Soyele3, Saddiku Malami Sahabi4
1 Department of Dental and Maxillofacial Surgery, Usmanu Danfodiyo University Teaching Hospital, Sokoto, Sokoto State, Nigeria
2 Department of Surgery/Dental and Maxillofacial Surgery, College of Health Sciences, Usmanu Danfodiyo University/Usmanu Danfodiyo University Teaching Hospital, Sokoto, Sokoto State, Nigeria
3 Department of Oral and Maxillofacial Surgery and Oral Pathology, Obafemi Awolowo University Teaching Hospital, Ile-Ife, Osun State, Nigeria
4 Department of Morbid Anatomy, Faculty of Basic Medical Sciences, College of Health Sciences, Usmanu Danfodiyo University; Departments of Histopathology, Usmanu Danfodiyo University Teaching Hospital, Sokoto, Sokoto State, Nigeria
|Date of Web Publication||21-May-2018|
Dr. Ramat Oyebunmi Braimah
Department of Dental and Maxillofacial Surgery, Usmanu Danfodiyo University Teaching Hospital, Sokoto, Sokoto State
Source of Support: None, Conflict of Interest: None
Introduction: Metastatic tumors are lesions that originate from a distant site and manifest in their secondary site remote from the primary. They are said to be rare in the maxillofacial region. Patients and Methods: This was a retrospective study from the Department of Dental and Maxillofacial Surgery and Department of Histopathology, Usmanu Danfodiyo University Teaching Hospital, Sokoto, northwest Nigeria, from 2010 to 2016. Results: A total of 259 cases of malignant tumors were seen, of which 8 cases (3.1%) were metastasis. There were four males and four females (ratio 1:1). The age ranged from 20 to 75 years with mean ± standard deviation (55.6 ± 18.8 years). Mandible was the chosen location for all the metastatic tumors in this series with the right body-ramus region frequently involved in 4 (50%), closely followed by the left body-ramus in 3 (37.5%) and 1 (12.5%) case seen in the parasymphyseal-symphyseal region. The prostate was the most frequent primary organ that metastasized to the maxillofacial region (3, 37.5%). Conclusion: High level of suspicion is required for early identification of these lesions, and prompt referral is paramount as these tumors can be the first evidence of hidden malignancy at distant sites such as prostate, colon, lungs, breast, and kidneys.
Keywords: Metastasis, oral and maxillofacial, primary cancers, prostate, thyroid
|How to cite this article:|
Braimah RO, Taiwo AO, Ibikunle AA, Soyele O, Sahabi SM. Metastatic oral and maxillofacial tumors in a tertiary referral hospital: Retrospective analysis of eight cases and review of the literature. J Clin Sci 2018;15:107-11
|How to cite this URL:|
Braimah RO, Taiwo AO, Ibikunle AA, Soyele O, Sahabi SM. Metastatic oral and maxillofacial tumors in a tertiary referral hospital: Retrospective analysis of eight cases and review of the literature. J Clin Sci [serial online] 2018 [cited 2018 Aug 14];15:107-11. Available from: http://www.jcsjournal.org/text.asp?2018/15/2/107/232818
| Introduction|| |
Tumors that originate from distant sites of the body, not contiguous spread from adjacent sites and/or tumors that recur locally, are generally referred to as metastatic tumors. Metastatic tumors to the oral and maxillofacial region have been reported to be extremely rare (1%–3% of all malignancies).,,, Out of these, about 65% have been reported to originate from the breast, lung, liver, kidney, and pancreas, while 35% originate from the testicles, uterus, thyroid, colon, rectum, stomach, and prostate. The mandibular body-ramus region has been described as the most common location of metastatic lesions in the maxillofacial region. Mechanism of spread of cancer into the maxillofacial region is vague; however, the seed-soil theory proposed by Paget has now gained popularity. Clinically, these metastatic tumors are of great importance because their appearance may be the initial sign of a distant undetected (or occult) malignancy. We present eight cases of histologically diagnosed oral and maxillofacial metastasis.
| Patients and Methods|| |
This was a retrospective study with data collected from the Department of Dental and Maxillofacial Surgery and Department of Histopathology, Usmanu Danfodiyo University Teaching Hospital, Sokoto, northwest Nigeria, from 2010 to 2016. Case notes, histology reports, and operation notes were retrieved, and data extracted include age, sex, diagnosis, site of metastatic lesion (the mandible was divided into symphyseal, parasymphyseal, body, ramus, condyle, and coronoid), site of primary lesion, first patient presentation, time interval between primary and secondary, other clinical symptoms, duration of metastasis, treatment done, follow-up, and outcome.
Ethical approval for the study was obtained from the Ethical and Research Committee, Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria.
Data were stored and analyzed using IBM SPSS Statistics for Windows Version 20 (IBM Corporation, Armonk, NY, USA) and results were presented as simple frequencies and descriptive statistics. P < 0.05 was considered statistically significant.
| Results|| |
A total of 259 cases of malignant tumors of the maxillofacial region were seen during the study period, of which 8 cases (3.1%) were metastatic lesions. There were four males and four females (ratio 1:1). The age ranged from 20 to 75 years with mean ± standard deviation age (55.6 ± 18.8 years) [Table 1]. There is no statistical significant difference when the age was compared with gender of the subjects.
|Table 1: Distribution of age and gender of subjects with metastatic jaw tumors|
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Most of the patients, i.e., 5 (62.5%) presented first to the oral and maxillofacial surgeons, 2 (25.0%) patients were referred from the general surgeons, while only one (12.5%) patient was referred from an obstetrics and gynecologist [Table 2]. Time interval between onset of primary and secondary lesions ranged between 9 months and 4 years in 6 (75%) patients, while in 2 (25%) patients, this period was not known. Duration of secondary lesion before presentation in the oral and maxillofacial surgery clinic was between 6 weeks and 1 year.
|Table 2: Distribution of clinical parameters of subjects with metastatic jaw tumors|
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The mandible was the chosen location for all the metastatic tumors in this series with the right side frequently involved 4 (50%), closely followed by the left side 3 (37.5%). The body-ramus region of the mandible was the most preferred site (7, 75%), while one (25%) gingiva metastasis occurred in the symphyseal region [Table 3].
|Table 3: Distribution of histological diagnosis and primary site of tumor|
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Radiolucent, radiopacity, and mixed radiographic features were seen in the review. Radiopacity and mixed radiodensity lesions were common with the prostatic metastasis [Table 2].
The prostate was the most frequent primary organ with secondaries to the maxillofacial region in 3 (37.5%) cases, while the thyroid, breast, thigh muscle, and colon have one (12.5%) each [Table 3]. Adenocarcinoma was the histological diagnosis seen in the prostate metastasis with high, intermediate, and infiltrating grades as variants. The histological diagnosis for the breast is invasive ductal carcinoma Grade II, while for thyroid, colon, and thigh are follicular adenocarcinoma, adenocarcinoma, and alveolar rhabdomyosarcoma (RMS), respectively [Table 3].
A multidisciplinary approach involving relevant specialties was employed in the management of these patients. Such include surgery and chemoradiation. Follow-up period of 5 (62.5%) patients ranged from 2 to 6 months, while 3 (37.5%) patients had no record of follow-up. As at the last follow-up time, 3 (37.5%) patients have passed away, 2 (25.0%) patients are still alive, while the status of 3 (37.5%) patients that had no follow-up record could not be ascertained [Table 4].
| Discussion|| |
For any malignant tumor to metastasize, it usually undergoes a complex course by detaching from the primary tumor, spread in tissues, and then invade blood or lymphatic channels. It has to survive the rigor of travel in blood circulation by evading tumor monitoring cells and settle in the capillaries of the secondary site, extravasate through the vessel wall, and invade the target organ and thereafter proliferate within the target tissue.,
Some studies previously reported that 1%–3% of malignant tumors metastasize to the oral and maxillofacial region while others have reported a rate of 1%–8%.,, Adebayo and Ajike  have reported a prevalence rate of 1% in Nigeria over a 20-year period; this study, however, observed a prevalence rate of 3.1% over a period of 7 years within the same region (northwest Nigeria). Lack of information, poverty, inaccessibility to quality health care, and inadequate workforce could be contributory to the previous low prevalence., However, better access and awareness may be responsible in the current study, leading to more patients been seen as compared to the old study.
Findings of metastasis in early adulthood [Table 1] may signal an increase in childhood tumors in this region. Equal gender distribution has been reported for metastasis to the jaw bone. This present study showed no gender predilection; however, gender differences have been reported in association with nature of the primary tumor and preferred area of distant metastasis.,, In our study, three prostate metastases were seen in males, while cases of uterus, breast, colon, and thyroid metastasis were seen in females. The only spread from the thigh muscle (alveolar RMS) was seen in a male. All the cases of oral metastasis in the current study occurred in the mandible with 7 (87.5%) occurring in the molar-angle-ramus region, while only one (12.5%) case of gingival involvement occurred in the anterior (symphyseal) region. This finding has been corroborated in the literature by Lim et al. The soft tissue metastasis in the current study occurred on the attached gingivae. Chronically inflamed gingivae can trap malignant tumor cells by the rich capillary network and favorable microenvironment, leading to proliferation of metastatic cells.
Most of our cases are metastasis from the prostate, i.e., 3 (37.5%). This contrasts sharply with the findings of Adebayo and Ajike  from the same region of Nigeria where thyroid metastasis was predominant in 3 (50%) cases. [Figure 1]a shows the clinical photograph of the only patient in our series with thyroid metastasis to the mandible while [Figure 1]b and [Figure 1]c shows photomicrograph of the primary and jaw metastasis, respectively. Prostate cancer has predilection for jaw bones because of its significant hematopoietic red marrow component. RMS has been reported to be a childhood tumor and the third most common solid tumor in children. RMS can occur in any site on the body, but it is primarily found in the head and neck, orbit, genitourinary tract, genitals, and extremities. Its metastasis to the jaw is reported to be rare. At least 15% of children with RMS present with metastatic disease which usually has a poor prognosis. The only patient with this condition in our series [Figure 2] discharged himself against medical advice, and his current status cannot be ascertained.
|Figure 1: (a) Clinical photograph of patient with thyroid swelling and right mandibular metastasis. (b) Photomicrograph of the primary tumor in the thyroid gland showing extensive capsular invasion by proliferating thyroid follicles (H and E, ×200). (c) Photomicrograph of metastatic tumor in the mandible showing proliferating colloid filled thyroid follicles (H and E, ×200)|
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|Figure 2: Clinical photograph of patient with alveolar rhabdomyosarcoma with right jaw metastasis|
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No specific radiographic feature has been associated with metastatic lesions to the oral and maxillofacial region; it ranges from osteolytic to osteoblastic and mixed. From the present study, metastasis from the prostate presented as osteoblastic or mixed in 3 (37.5%) cases. This is in agreement with the literature. Osteolytic lesions have been reported to be associated with breast, thyroid, and lung cancers  which is similar to our findings. Most of our patients are poor; therefore, advanced investigative procedures could not be carried out. With these constraints, managing these conditions in sub-Saharan Africa could be very challenging.
A multidisciplinary approach including surgery and chemoradiation therapy was employed in our cases which are in tandem with the literature. Although three patients were lost to follow up, two patients are being followed up and receiving chemotherapy.
| Conclusion|| |
Oral and maxillofacial metastasis creates a diagnostic challenge to the dental practitioner because clinical findings frequently imitate reactive or benign lesions and even odontogenic infections. High index of suspicion is important.
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.
The authors would like to thank Laboratory technical staff, Department of Histopathology, Usmanu Danfodiyo University Teaching Hospital, Sokoto, Nigeria.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
van der Waal RI, Buter J, van der Waal I. Oral metastases: Report of 24 cases. Br J Oral Maxillofac Surg 2003;41:3-6.
Kumar GS, Manjunatha BS. Metastatic tumors to the jaws and oral cavity, report of 65 cases. J Oral Maxillofac Pathol 2013;17:71-5.
] [Full text]
Tamiolakis D, Tsamis I, Thomaidis V, Lambropoulou M, Alexiadis G, Venizelos I,
Jaw bone metastases: Four cases. Acta Dermatovenerol Alp Pannonica Adriat 2007;16:21-5.
Adebayo E, Ajike S. Report of six cases of metastatic jaw tumors in Nigerians. Niger J Surg Res 2004;6:30-3.
Gandhiraj SK. Oral metastasis as the first manifestation of an extragonadal primary choriocarcinima of pancreas. J Indian Acad Oral Med Radiol 2013;25:59-62.
Lim SY, Kim SA, Ahn SG, Kim HK, Kim SG, Hwang HK,
Metastatic tumours to the jaws and oral soft tissues: A retrospective analysis of 41 Korean patients. Int J Oral Maxillofac Surg 2006;35:412-5.
Fidler IJ, Poste G. The “seed and soil” hypothesis revisited. Lancet Oncol 2008;9:808.
Bircan A, Baykul T, Kapucuoglu N, Öztürk O. Small cell carcinoma of the lung with mandible metastasis. Turk Toraks Dernegi 2008;9:185-7.
Hanahan D, Weinberg RA. The hallmarks of cancer. Cell 2000;100:57-70.
Singh H, Kumar P, Nirwan A, Kaur R. Possible pathogenetic mechanisms and overview of metastatic tumours to the oral cavity. Internet J Oncol 2010;8:1-6.
Bassey GO, Osunde OD, Anyanechi CE. Maxillofacial tumours and tumor-like lesions in a Nigerian teaching hospital: An eleven year retrospective analysis. Afr Health Sci 2014;14:56-63.
Fasunla AJ, Lasisi AO. Sinonasal cancer: A 10-year review in a tertiary health institution. J Natl Med Assoc 2007;99:1407-10.
Rajappa S, Loya A, Rao RD. Metastasis to oral cavity: A report of two cases and review of literature. Indian J Med Pediatr Oncol 2005;26:43-6.
Hirshberg A, Shnaiderman-Shapiro A, Kaplan I, Berger R. Metastatic tumours to the oral cavity – Pathogenesis and analysis of 673 cases. Oral Oncol 2008;44:743-52.
Menezes JD, Cappellari PF, Capelari MM, Gonçalves PZ, Toledo GL, Toledo Filho JL,
Mandibular metastasis of adenocarcinoma from prostate cancer: Case report according to epidemiology and current therapeutical trends of the advanced prostate cancer. J Appl Oral Sci 2013;21:490-5.
Oberlin O, Rey A, Lyden E, Bisogno G, Stevens MC, Meyer WH,
Prognostic factors in metastatic rhabdomyosarcomas: Results of a pooled analysis from United States and European cooperative groups. J Clin Oncol 2008;26:2384-9.
Peñarrocha Diago M, Bagán Sebastián JV, Alfaro Giner A, Escrig Orenga V. Mental nerve neuropathy in systemic cancer. Report of three cases. Oral Surg Oral Med Oral Pathol 1990;69:48-51.
[Figure 1], [Figure 2]
[Table 1], [Table 2], [Table 3], [Table 4]