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Unusual neoplasm on the hard palate of a child: a case report
© The Author(s). 2017
Received: 26 February 2017
Accepted: 12 May 2017
Published: 2 June 2017
Myoepitheliomas account for less than 1% of salivary gland tumors. They mostly affect the parotid glands of adults during the third to fifth decades.
A 10-year-old Indian boy reported a small swelling in the roof of his mouth of 10 days’ duration. History revealed that the lesion was painless and not associated with bleeding or pus discharge. On examination, a purplish well-circumscribed growth was noted on his posterior hard palate. Magnetic resonance imaging was suggestive of a well-encapsulated hemangioma. An excisional biopsy was performed and histopathology along with immunohistochemistry analysis showed that the lesion was a spindle cell variant of benign myoepithelioma.
Palatal myoepitheliomas are rare and their occurrence in young individuals is rarer.
KeywordsSalivary gland tumor Pediatric Oral cavity Hemangioma Myoepithelioma
Myoepitheliomas are rare benign salivary gland neoplasms accounting for less than 1% of salivary gland tumors. They commonly affect the parotid glands and rarely the minor salivary glands, of which the palate is the most common location. Myoepithelioma affects adults between the third and fifth decades, with no gender predilection. To date, to the best of our knowledge, only seven cases of myoepithelioma of the palate occurring in children and adolescents have been reported in the literature. Here we report a case of benign myoepithelioma of the hard palate in a 10-year-old boy to attest the rarity of this tumor.
His postoperative recovery was uneventful with complete healing of the palatal area and no clinical evidence of recurrence (7 months postoperation). He was advised to have clinical/ radiological follow-ups on an annual basis.
Reports of palatal myoepithelioma in the literature
Kahn and Schoub (1973) 
Nesland et al. (1981) 
Lins and Gnepp (1986) 
Arkuszewski et al. (2005) 
Nwoku et al. (2005) 
Perez et al. (2007) 
Santos et al. (2011) 
Our case (2017)
Spindle cell type
On histologic examination, myoepitheliomas are of four morphological types: spindle cell (most common), epithelioid, plasmacytoid, and clear cell (least common). The most common histopathological type described in palatal myoepithelioma is of the plasmacytoid type, while our case revealed a spindle cell morphology. The spindle cell variant of myoepithelioma is not known to behave differently when compared to other morphological subtypes of myoepithelioma. On immunohistochemical examination, the neoplastic cells stain positive with cytokeratin (AE1/AE3, CK5/6, CK7) and variably positive for S100, smooth muscle actin (SMA), p63 protein, and glial fibrillary acidic protein (GFAP). Usually, a combination of a keratin in conjunction with the detection of S100, vimentin, and/or a myogenic marker is required for confirmation of the diagnosis of a myoepithelioma. The present case showed positivity with S100 and p63, thus confirming the diagnosis of myoepithelioma.
Although minor salivary gland tumors present clinically as a dome-shaped swelling on the palate, there may be altered surface texture of the lesion, as in the present case, which may be trauma induced because the oral cavity is subjected to varying temperatures, pH, and different textures of food substances and beverages apart from the masticatory forces. The unusualness of the present case lies in the fact that the tumor that presented on the hard palate of a young child resembled a hemangioma but was found to be a spindle cell type of myoepithelioma, which is a very rare entity. Surgical excision of myoepithelial tumors with adequate margins decreases the chances for recurrence. Further, the reported recurrence rate is the same as that of pleomorphic adenoma (40 to 65%) . The prognosis for benign myoepitheliomas is generally favorable, provided patients undergo regular follow-up examinations to eliminate local recurrence .
Benign salivary gland neoplasms like myoepitheliomas should be considered in the differential diagnoses of palatal lesions. Although minor salivary gland tumors present as dome-shaped, smooth-surfaced masses on the palate, it should be borne in mind that there can be alteration in the surface characteristics; thus mimicking other vascular and reactive lesions.
The authors would like to express their heartfelt thanks to Dr Keerthilatha M Pai, Dean and Professor, Oral Medicine & Radiology, MCODS, Manipal and Dr Vineetha R, Professor and Head, Oral Medicine & Radiology, MCODS, Manipal for their constant motivation and encouragement.
Availability of data and materials
Data sharing is not applicable to this article as no datasets were generated or analyzed during the current study.
MK: Initial clinical and radiological assessments, and design and coordination of the manuscript. SA: Initial clinical and radiological assessments, and design and final approval of the manuscript. KP: Performed the histopathological and immunohistochemical studies, and final approval of the manuscript. VK: Performed the surgical procedure and final approval of the manuscript. SPT: Performed the surgical procedure and final approval of the manuscript. All authors read and approved the final manuscript.
The authors declare that they have no competing interests.
Consent for publication
Written informed consent was obtained from the patient’s legal guardian(s) for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
Ethics approval and consent to participate
The manuscript does not contain any direct patient identification details and hence ethical committee approval was waived.
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