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CASE REPORT - SALIVARY PATHOLOGY
Year : 2020  |  Volume : 10  |  Issue : 2  |  Page : 533-536

Surgical management of palatal pleomorphic adenoma (PPA) recurrence after 10 years, treated at a Brazilian center - A case report


1 Department of Implantodontics, School of Dentistry, Paulist University, Sao Paulo, Brazil
2 Department of Dentistry and Restoration, School of Dentistry, Paulist University, Sao Paulo, Brazil
3 Department of Oral and Maxillofacial Surgery, Hospital Regional Sul, Sao Paulo, Brazil
4 Department of Oral and Maxillofacial Surgery, Hopsital Sirio-Libanes, Sao Paulo, Brazil
5 Department of Radiation Oncology, A.C. Camargo Cancer Center, Sao Paulo, Brazil

Date of Submission07-Apr-2020
Date of Acceptance27-Aug-2020
Date of Web Publication23-Dec-2020

Correspondence Address:
Dr. Wilber Edison Bernaola-Paredes
Department of Radiation Oncology, A.C. Camargo Cancer Center, Sao Paulo
Brazil
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DOI: 10.4103/ams.ams_107_20

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  Abstract 


Pleomorphic adenoma, considered the most frequent benign mixed neoplasm of the minor salivary glands, occurs mainly in the region of the hard palate, with slight predilection in females and peak of incidence between the third and fifth decades of life. An increase in recurrence rates has been associated with the histopathological variants of the tumor, cellular characteristics, stroma, and capsule rupture during surgical removal of the lesion. The present case report aims to describe the surgical approach performed on the patient, a 45-year-old woman with a recurrent Pleomorphic Adenoma (PA) in the region of the hard palate on the right side, 10 years after initial enucleation of the lesion; her main complaint was an increase in volume in the palatal region. After extensive local surgical excision of the tumor and 2 years of follow-up, there were no signs of recurrence. Computed tomography and a correct histopathological diagnosis are essential to enable the establishment of an appropriate surgical treatment, with the purpose of achieving complete removal of the lesion, with wide surgical margins, including the lining mucosa and the underlying periosteum, as described in the present case.

Keywords: Hard palate, local neoplasm, pleomorphic adenoma, recurrence, salivary gland neoplasm, surgical pathology


How to cite this article:
De Lima FF, Bezerra CP, Rocha AC, Martins IS, Bernaola-Paredes WE. Surgical management of palatal pleomorphic adenoma (PPA) recurrence after 10 years, treated at a Brazilian center - A case report. Ann Maxillofac Surg 2020;10:533-6

How to cite this URL:
De Lima FF, Bezerra CP, Rocha AC, Martins IS, Bernaola-Paredes WE. Surgical management of palatal pleomorphic adenoma (PPA) recurrence after 10 years, treated at a Brazilian center - A case report. Ann Maxillofac Surg [serial online] 2020 [cited 2021 Apr 22];10:533-6. Available from: https://www.amsjournal.com/text.asp?2020/10/2/533/304383


  Introduction Top


Pleomorphic adenoma (PA) is the most common benign tumor of salivary glands of the head and neck region, accounting for about 40%–70% of all major and minor salivary gland tumors. It usually presents as a slow-growing submucosal mass in the hard palate when minor glands are affected. The palate is the most common intraoral site of PA, followed by the upper lip, buccal mucosa, floor of the mouth, tongue, tonsil, and retromolar regions.[1],[2],[3],[4],[5]

The majority of minor salivary gland PAs occur in the second decade of life,[1] but other epidemiological data[3],[6] have shown a higher prevalence in the fourth and sixth decades, with slight predilection in the female gender.[1],[3],[6]

Clinically, it presents as a painless, immobile nodular lesion of firm consistency that is slow-growing, covered by healthy mucosa and of normal color.[3],[6],[7],[8] In few cases, mainly related to the parotid gland, it could grow more rapidly.[9] Moreover, while PAs affect the palatal region, their clinical presentation is typically characterized by a firm or rubbery submucosal mass without ulceration or surrounding inflammation.[1]

These lesions can affect the underlying bone and may extend to the maxillary sinus.[10] Thus, to aid the clinical examination, computed tomography (CT) is the gold standard imaginological assessment required, as it delimits the region affected by the lesion and reduces the chances of recurrence or postoperative complications.

Histologically, the lesion presents a well-delimited neoplasm with a fibrous connective tissue capsule, consisting of cells with ductal and myoepithelial elements.[6],[9],[11] In addition, myxoid, ductiform, hyaline, plasmacytoid, osteoid, and chondroid areas are found, which is why the term benign mixed tumor has been applied to it.[4],[9],[12],[13]

Classically, the most reported form of treatment in the literature is wide local excision with the removal of the periosteum or bone if these tissues are involved. Simple enucleation procedures, which may or may not remove covering tissue, are associated with high local recurrence of the tumor and should be avoided.[1],[5],[9]

The aim of this brief report was to describe our experience with surgical management of a recurrent palatal PA in a female patient and to compare our clinical results with those of others found in the contemporary literature review.


  Case Report Top


The patient, a 46-year-old woman, was referred to the outpatient clinic of our Division of the Oral and Maxillofacial Surgery Department at a state hospital in Sao Paulo, Brazil, to have several complaints addressed. Among these was swelling, pain in the hard palate, making it difficult to adapt her dental prosthesis correctly in the region where a surgical procedure for cyst removal had been performed 10 years previously.

An extra-oral examination showed no facial changes. Relative to the intraoral assessment, an exophytic lesion with a nodular aspect was found, which had a firm consistency, was located in the anterolateral portion of the hard palate, fixed on the right side, measuring approximately 2.2 cm × 3 cm. It was slow-growing, and the patient reported no significant pain complaints about it. CT for soft and hard tissues was performed with the purpose of delimiting the local invasiveness of tumor that showed no bone breakdown or erosions in palatal bone, as visualized in [Figure 1].
Figure 1: (a) Intraoral examination showed well-delimited swelling in the anterior region of the hard palate. (b) In computed tomography scan, generating images in axial section, size, and extension of lesions associated with surrounding tissues may be visualized. (c) Relative to hard tissues, intact hard palate bone may be observed in the axial section of computed tomography

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Histopathological assessment was made after an incisional biopsy, which confirmed our initial diagnosis of PA. Thus, the surgical planning consisted of the complete removal of the tumor by wide surgical excision involving the periosteum and surrounding mucosa.

The surgical technique was started with regional antisepsis using chlorhexidine 0, 12%, and 2% for purposes of intra- and extra-oral decontamination, respectively. Local anesthesia was performed in conjunction with conscious sedation for better patient comfort. Moreover, a rhomboidal incision surrounding the lesion, up to the bone level, was made with an electric scalpel and the divulsion of the periosteum was performed using a molt periosteal elevator number 9. The lesion was detached and removed immediately, as shown in [Figure 2].
Figure 2: (a) Wide local excision for removal of the tumor in the hard palate, which included underlying mucosal-periosteal tissue, (b) Macroscopically, well-delineated submucosal mass covered with normal mucous membrane may be visualized, corresponding to the surgical specimen

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Indeed, the surgical specimen was fixed in a 10% formaldehyde solution and sent for definitive histopathological examination by the oral pathology laboratory. Finally, the wound was allowed to granulate and heal by itself, and the patient's obturator prosthesis was repackaged with silicone material to avoid further contamination of the surgical site and diminish painful symptoms. Corticosteroid and analgesic medications were recommended for use in the following 5 days.

Histopathology with hematoxylin and eosin staining [Figure 3] showed the presence of proliferation of cells such as fusiform/plasmacytoid myoepithelial tumor-type cells. The neoplastic parenchyma was made up of epithelial cells arranged in cord-like and duct-like cell patterns. These were distributed in small stroma mixtures that confirmed the characteristics of a benign PA.
Figure 3: Epithelial cells arranged in duct-form or cord-like cell patterns, along with areas of epidermoid metaplasia

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The patient was followed up weekly for the first 4 weeks. During this period, complaints of painful symptoms were recorded in the initial 15 days [Figure 4]. Moreover, the patient has been followed up clinically and radiographically for 1 year and 6 months (October 2016 to April 2018) without signs of recurrence.
Figure 4: Follow-up after (a) 7 days, (b) 4 weeks, and (c) Almost 2 months after surgical procedure

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  Discussion Top


PA is one of the common benign salivary neoplasms, affecting major[13] and minor salivary glands.[9],[12] According to Waldron et al., 1988,[10] relative to their study with 466 cases, 174 cases of these neoplasms were located in minor salivary glands. In the majority of cases, the hard palate was affected in almost 54%, as found in other studies.[8],[9] In recent studies, authors found high injury rates in the hard palate region, ranging from 40% to 70% of the total number of cases.[1],[12]

The diagnosis of PPA is based on the medical history reported by the patient, intra- and extra-oral clinical examination, with the main clinical sign being an increase in volume in the region of the hard palate, as shown in this case report. CT has become an essential tool for detecting the exact location, size, and extent of the lesion, and in the contemporary literature,[1],[14] it is considered an image examination superior to MRI with reference to the tumor characteristics mentioned above. The definitive diagnosis will be determined by histopathological analysis.[14]

A retrospective study[2] evaluated the clinicopathological characteristics of 74 patients with PPA and determined that of this group, 40 (84%) of the adenomas were partially encapsulated and that they had a larger amount of plasmacytoid myoepithelial tumor cells (50 cases). These histopathological characteristics were found in this case report after histological examination and could probably be associated with the recurrence shown in our patient 10 years after the initial surgical removal of the tumor.

PA, the most common benign neoplasm in minor salivary glands, has a predilection for female patients as has been shown by recent studies.[14],[15] Patigaroo et al.,[1] however, reported predominance of palatal PA in males over females, and the most common age group was 16–30 years. This present case report showed a single case of a 45-year-old female patient with recurrence of a PPA that had been submitted to surgical removal 10 years previously. This was consistent with the relevant literature that showed a peak of incidence between the third and fifth decades of life.[14],[15]

The surgical technique is a relevant factor in order to avoid the recurrence of tumors. Indeed, simple enucleation is associated with clinical failure and local recurrence. For this reason, it should be avoided,[1],[14],[16] whereas a conservative wide local surgical excision, instead of a resective surgical approach, would be the treatment of choice that involves complete removal of mucosal-periosteal tissue surrounding the lesion, or the curettage of bone affected, as described in the retrospective study.[14] Bone removal is usually not necessary if the PA has not invaded any bone tissue.[1],[6],[9],[12]

In cases of erosions detected in the hard palate by means of CT that indicated a wide destruction and/or rupture of the floor of the nasal cavity and/or paranasal sinuses, the best alternative would be the removal of tumor in conjunction with the compromised bone, with enlarged margins, as described in the literature.[1],[14],[16] For more advanced cases, partial maxillectomy has been described as the main approach, followed by rehabilitation with obturator prostheses.[17]

Recurrences usually appear due to an inadequate surgical technique, as previously described, which could happen in a long-term period of follow-up, after a period of 2 years onward after the surgical procedure. However, other previous studies[9],[14] have detected recurrences between 7 and 10 years after initial surgery, as shown in this case report, as already mentioned.

Multiple recurrences of PPA, which take a longer time to develop, are associated with a higher probability of these becoming a carcinoma ex-PA (ca ex PA), which is an aggressive malignancy that can lead to death in most cases.[9],[10],[12],[13],[14] Histopathological findings and other stroma alterations have been associated with malignant transformation such as focal necrosis, extensive hyalinization, vascular and capsular invasion, and atypical mitosis; however, these features are rarely seen in these tumors as shown in this case report and by other recent retrospective studies.[15]

Recurrence rates vary from 0% to 14%, and the majority were shown in cases in which the surgeon opted for an extremely conservative excision (simple enucleation) only, without removing a safety margin, and often with rupture of the lesion capsule, so that the tumor achieved greater capacity for spreading into the adjacent tissues.[1],[13]


  Conclusion Top


PPA is the most frequent benign neoplasm in the minor salivary glands, and its recurrence may occur after a prolonged period of follow-up of the lesion, due to its wide spectrum of cellular and histopathological variants or an inappropriate surgical approach. A correct surgical technique that involves the complete removal of the lesion, margins involved, including the lining mucosa and the underlying periosteum has shown a reduction in the rates of local tumor recurrence. Nevertheless, an accurate clinical, tomographic, and histopathological evaluation will be essential for the establishment of an adequate surgical treatment plan. A secondary healing process with obturator prostheses showed positive results in the present case after a considerable period of clinical follow-up.

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.

Acknowledgment

The authors thank the Department of Oral and Maxillofacial Surgery, Hospital Regional Sul, Sao Paulo, Brazil (BR), including the technical team from the Oral Pathology Laboratory who participated in the work at the outpatient clinic, who supported this work.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Patigaroo SA, Patigaroo FA, Ashraf J, Mehfooz N, Shakeel M, Khan NA, et al. Pleomorphic adenoma of hard palate: An experience. J Maxillofac Oral Surg 2014;13:36-41.  Back to cited text no. 1
    
2.
Wu YC, Wang YP, Cheng SJ, Chen HM, Sun A, Chang JY. Clinicopathological study of 74 palatal pleomorphic adenomas. J Formos Med Assoc 2016;115:25-30.  Back to cited text no. 2
    
3.
Chidzonga MM, Lopez Perez VM, Portilla Alvarez AL. Pleomorphic adenoma of the salivary glands. Clinicopathologic study of 206 cases in Zimbabwe. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1995;79:747-9.  Back to cited text no. 3
    
4.
Debnath SC, Saikia AK, Debnath A. Pleomorphic adenoma of the palate. J Maxillofac Oral Surg 2010;9:420-3.  Back to cited text no. 4
    
5.
Jorge J, Pires FR, Alves FA, Perez DE, Kowalski LP, Lopes MA, et al. Juvenile intraoral pleomorphic adenoma: Report of five cases and review of the literature. Int J Oral Maxillofac Surg 2002;31:273-5.  Back to cited text no. 5
    
6.
Porto DE, Cavalcante JR, Cavalcante Júnior JR, da Costa MC, Pereira SM. Pleomorphic adenoma of parotid gland: A case report. Brazilian Journal of Oral and Maxillofacial Surgery 2014;14:15-8.  Back to cited text no. 6
    
7.
de Sousa RI, dos Santos MG, Oliveira JM, Mendonça VB, Pereira JV. Adenoma Pleomorphic adenoma of submandibular gland: A case report and current literature review. Brazilian Journal of Oral and Maxillofacial Surgery 2013;13:9-14.  Back to cited text no. 7
    
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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;49:14-20.  Back to cited text no. 8
    
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Erdem MA, Cankaya AB, Güven G, Olgaç V, Kasapoğlu C. Pleomorphic adenoma of the palate. J Craniofac Surg 2011;22:1131-4.  Back to cited text no. 9
    
10.
Waldron CA, El-Mofty SK, Gnepp DR. Tumors of the intraoral minor salivary glands: A demographic and histologic study of 426 cases. Oral Surg Oral Med Oral Pathol 1988;66:323-33.  Back to cited text no. 10
    
11.
Tian Z, Li L, Wang L, Hu Y, Li J. Salivary gland neoplasms in oral and maxillofacial regions: A 23-year retrospective study of 6982 cases in an eastern Chinese population. Int J Oral Maxillofac Surg 2010;39:235-42.  Back to cited text no. 11
    
12.
Oliveira LJ, Castro HH, Leão PL, Leal RM, Horta MC, Souza PE. managment of palatal pleomorphic adenoma: Two case reports and literature review. Portuguese Journal of Stomatology, Dental Medicine and Maxillofacial Surgery 2016;57:55-61.  Back to cited text no. 12
    
13.
Bonet-Loscertales M, Armengot-Carceller M, Gaona-Morales J, Basterra-Alegría J. Multicentric recurrent parotid pleomorphic adenoma in a child. Med Oral Patol Oral Cir Bucal 2010;15:743-5.  Back to cited text no. 13
    
14.
Moon SY. Surgical management of the palatal pleomorphic adenoma. J Craniofac Surg 2019;30:e580-2.  Back to cited text no. 14
    
15.
Lopes ML, Barroso KM, Henriques ÁC, Dos Santos JN, Martins MD, de Souza LB. Pleomorphic adenomas of the salivary glands: Retrospective multicentric study of 130 cases with emphasis on histopathological features. Eur Arch Otorhinolaryngol 2017;274:543-51.  Back to cited text no. 15
    
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Gupta M, Gupta M. Pleomorphic adenoma of the hard palate. BMJ Case Rep 2013;2013:1-2.  Back to cited text no. 16
    
17.
Brauner E, Jamshir S, Guarino G, Ciolfi A, Valentini V, Pompa G, et al. Pleomorphic adenoma rehabilitative treatment in growing up patient: A 20-years follow-up. Eur Rev Med Pharmacol Sci 2016;11:17.  Back to cited text no. 17
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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