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Pleomorphic adenoma of the submandibular gland
1 Resident Physician of the 6th year of Maxillofacial Surgery at Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal
2 Resident Physician of the 1st year of Anatomical Pathology at Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal
3 Graduate Assistant in the Department of Maxillofacial Surgery at Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal
4 Head of Maxillofacial Surgery Department at Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal
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Clara Carracha
Avenida Dr. José Grilo Evangelista, 207, 5º frente, 2890-007 Alcochete,
Portugal
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Article ID: 100029Z07CC2019
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Carracha C, Silva A, Santos M, da Silva LN, Coelho PV. Pleomorphic adenoma of the submandibular gland. J Case Rep Images Dent 2019;5:100029Z07CC2019.ABSTRACT
No Abstract
Keywords: Pediatric age, Pleomorphic adenoma, Salivar tumor
Case Report
A 16-year-old boy was observed in our hospital, by the presence of a right submandibular mass, with progressive and painless growth, with about one year of evolution. The patient denied any other type of symptomatology. Clinical observation revealed the presence of a right submandibular mass, with approximately 2 cm in diameter, a hard/elastic consistency, painless, without adherence to the deep or superficial planes. There were no functional deficits in relation to the marginal branch of the facial nerve, the lingual branch of the trigeminal nerve, or the hypoglossal nerve.
Imaging of the cervical computed tomography (CT) scan revealed a right, hypodense, and hypocapant submandibular nodule, with approximately 2.5 cm (Figure 1A–C), with homogeneous and regular contours. The fine needle aspiration cytology was inconclusive, however, excluding the presence of dysplasia or neoplasia.
The patient was operated under general anesthesia, submitted to the right submandibular gland surgical excision (complete sialoadenectomy). The presence of a nodule was considered an integral part of the right submandibular gland (Figure 2 A, B), regular, capsulated, without invasion of neighboring structures. There were no intercurrences in the postoperative period.
The histological analysis revealed the presence of the submandibular gland about 45×25×20 mm, associated with a well-circumscribed elastic nodule, 22×20×20 mm, whose microscopic analysis was compatible with a pleomorphic adenoma, showing an epithelial component with ducts and myoepithelial cells, dispersed in a myxoid stroma (Figure 3).
After three years of follow-up, there are no signs or symptoms suggestive of relapse.
Discussion
The pleomorphic adenoma is the most frequent benign tumor of the salivary glands, however, it is rarely present in children or adolescents [1]. Known as a benign mixed tumor, pleomorphic adenoma accounts for about 60% of all benign tumors affecting the salivary glands [2], being more frequent in the parotid gland [3]. It occurs in the submandibular and sublingual glands in about 8–10% of cases [3]. Usually, it is a tumor that affects adults between 30 and 60 years of age, being more frequent in females [2], [4]. Clinically, it is characterized by the presence of a regular mass, with painless and progressive growth, as described in the case report. It is able to reach large dimensions, compromising neighboring anatomical structures [5].
The cytologic diagnosis is obtained through fineneedle aspiration [2]. Incisional biopsy is not indicated, since it may lead to the extension of tumor cells externally to the capsule [5]. Imaging diagnosis can be performed by ultrasound or CT scan, but magnetic resonance imaging (MRI) is the first choice exam.
Histologically, it presents as a capsulated tumor, being constituted by a mixture of ductal and myoepithelial cells, whose differentiation is, respectively, epithelial and mesenchymal. It can be classified in cellular type (rich in epithelial cell) or myxoid type (rich in stromal) [6]. Besides this, there are several histological presentations and so, it is called “pleomorphic” [2].
The treatment is the same in adults and children [4], with surgical excision in block, with negative margins, being the first choice option. Recurrence is rare, but the tumor enucleation increases its risk [1]. Surgical complications are not frequent [1]. It is estimated that the risk of malignancy, originating a “carcinoma ex-pleomorphic adenoma,” is around 25% [3]. The acceleration of tumor growth and the appearance of adenopathies, associated with facial paralysis, are signs of probable malignization [4]. Tumors of the salivary glands represent an important group of masses that reach the head and neck [5].
Conclusion
Pleomorphic adenoma is the most frequent tumor, however, in the pediatric age, the inflammatory and congenital cervical masses are more frequent than the tumor origin.
REFERENCE
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SUPPORTING INFORMATION
Author Contributions
Clara Carracha - Conception of the work, Design of the work, Acquisition of data, Analysis of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Andreia Silva - Conception of the work, Design of the work, Drafting the work, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Margarida Santos - Acquisition of data, Analysis of data, Drafting the work, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Luís Nunes da Silva - Conception of the work, Design of the work, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
Paulo Valejo Coelho - Conception of the work, Design of the work, Drafting the work, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.
AcknowledgmentsThe authors are grateful to the other health professionals of the Centro Hospitalar Universitário de Lisboa Central, who also contributed to the therapeutic success of this type of patients.The lead author also thanks her sister for her support.
Source of SupportNone
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
Conflict of InterestAuthors declare no conflict of interest.
Copyright© 2019 Clara Carracha et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information.
