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Journal of Case Reports and Images in Dentistry, Vol. 5, 2019.
J Case Rep Images Dent 2019;5:100029Z07CC2019. www.edoriumjournals.com/case-reports/jcrd
Carracha et al. 1
CASE REPORT OPEN ACCESS
Pleomorphic adenoma of the submandibular gland
Clara Carracha, Andreia Silva, Margarida Santos, Luís Nunes da Silva, Paulo Valejo Coelho
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.
Clara Carracha1, Andreia Silva1, Margarida Santos2, Luís Nunes da Silva3, Paulo Valejo Coelho4
Affiliations: 1Resident Physicians of the 6th year of Max-illofacial Surgery at Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal; 2Resident Physician of the 1st year of Anatomical Pathology at Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal; 3Gradu-ate Assistant in the Department of Maxillofacial Surgery at Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal; 4Head of Maxillofacial Surgery Department at Centro Hospitalar Universitário de Lisboa Central, Lisbon, Portugal.Corresponding Author: Clara Carracha, Avenida Dr. José Grilo Evangelista, 207, 5º frente, 2890-007 Alcochete, Por-tugal; Email address: [email protected]
Received: 29 December 2018Accepted: 08 May 2019Published: 24 July 2019
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
CLINICAL IMAGE PEER REVIEWED | OPEN ACCESS
Figure 1: Preoperative CT scan—axial (A), coronal (B), and sagittal (C) view showing a hypodense nodule in the right submaxillary gland (orange arrow).
Figure 2: (A) After surgical excision in block, revealing the submaxillary gland and the nodule (blue arrow), and the macroscopic visualization of the interior of the nodule (B).
Journal of Case Reports and Images in Dentistry, Vol. 5, 2019.
J Case Rep Images Dent 2019;5:100029Z07CC2019. www.edoriumjournals.com/case-reports/jcrd
Carracha et al. 2
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 fine-needle 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.
Keywords: Pediatric age, Pleomorphic adenoma, Salivar tumor
How to cite this article
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.
Article ID: 100029Z07CC2019
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doi: 10.5348/100029Z07CC2019CL
REFERENCES
1. Molina EJ, Mayer K, Khurana J, Grewal H. Pleomorphic adenoma of the submandibular gland. J Pediatr Surg 2008;43(6):1224–6.
2. Lingam RK, Daghir AA, Nigar E, Abbas SA, Kumar M. Pleomorphic adenoma (benign mixed tumour) of the salivary glands: Its diverse clinical, radiological, and histopathological presentation. Br J Oral Maxillofac Surg 2011;49(1):14–20.
3. Rai S, Sodhi SP, Sandhu SV. Pleomorphic adenoma of sbmandibular gland: An uncommon occurrence. Natl J Maxillofac Surg 2011;2(1):66–8.
4. Fu H, Wang J, Wang L, Zhang Z, He Y. Pleomorphic adenoma of the salivary glands in children and adolescents. J Pediatr Surg 2012;47(4):715–9.
5. Alves CA, Ribeiro Júnior O, Borba AM, et al. Pleomorphic multicentric adenoma in the aubmandibular gland. Head Neck Pathol w2007;1(2):178–80.
6. Witt RL. Salivary Gland Diseases: Surgical and Medical Management. 1ed. New York: Thieme Medical Publishers; 2005.
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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.
Author ContributionsClara Carracha – Conception of the work, Design of the work, Acquisition of data, Analysis of data, Interpretation 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
Figure 3: Pleomorphic adenoma (H&E, 40x)—Mixed benign tumor with epithelial component with ducts and myoepithelial cells dispersed in a myxoid stroma.
Journal of Case Reports and Images in Dentistry, Vol. 5, 2019.
J Case Rep Images Dent 2019;5:100029Z07CC2019. www.edoriumjournals.com/case-reports/jcrd
Carracha et al. 3
aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolvedAndreia Silva – Analysis of data, Interpretation 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 resolvedMargarida Santos – Acquisition of data, Analysis of data, Interpretation 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 resolvedLuís Nunes da Silva – Conception of the work, Design of the work, Interpretation of data, 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 resolvedPaulo Valejo Coelho – Conception 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
Guarantor of SubmissionThe corresponding author is the guarantor of submission.
Source of SupportNone.
Consent StatementWritten informed consent was obtained from the patient for publication of this article.
Conflict of InterestAuthors declare no conflict of interest.
Data AvailabilityAll relevant data are within the paper and its Supporting Information files.
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.
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