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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 24  |  Issue : 1  |  Page : 60-63

“Intrabony” palatal pleomorphic adenoma: A case report and review of the literature


1 Department of Surgery, Benjamin Carson Snr. School of Medicine, Babcock University; Department of Surgery, Dental Unit, Babcock University Teaching Hospital, Ilisan-Remo, Ogun State, Nigeria
2 Department of Surgery, Dental Unit, Babcock University Teaching Hospital, Ilisan-Remo, Ogun State, Nigeria
3 Department of Biochemistry, Benjamin Carson Snr. School of Medicine, Babcock University High School, Ilisan-Remo, Ogun State, Nigeria

Date of Submission02-Nov-2019
Date of Decision29-Jan-2020
Date of Acceptance04-Mar-2020
Date of Web Publication31-Mar-2021

Correspondence Address:
Dr. Adekunle Moses Adetayo
Department of Surgery, Benjamin Carson Snr. School of Medicine, Babcock University, Ilisan-Remo, Ogun State, Nigeria, Dental Unit, Babcock University Teaching Hospital, Ilisan-Remo, Ogun State
Nigeria
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DOI: 10.4103/smj.smj_59_19

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  Abstract 


Pleomorphic adenoma (PA) is the most common neoplasm of the large salivary glands. It derives its name from the architectural pleomorphism seen by light microscopy. It is also known as the mixed tumor salivary type, which describes its pleomorphic appearance as opposed to its dual origin from epithelial and myoepithelial elements. We present a case of an adult who presented with a palatal swelling, which computed tomography (CT) scan showed to be “intrabony” but after considering other planes of the CT scan It was finally considered to be an intrabony extension of the palatal PA. The mass was successfully removed and the residual palatal defect left to granulate. Following 2 years of patient follow-up, there was no recurrence but the presence of a oronasal defect about 1 cm in diameter in the palate.

Keywords: Defect, intrabony, palate, pleomorphic adenoma


How to cite this article:
Adetayo AM, Oladapo OA, Adetayo MO. “Intrabony” palatal pleomorphic adenoma: A case report and review of the literature. Sahel Med J 2021;24:60-3

How to cite this URL:
Adetayo AM, Oladapo OA, Adetayo MO. “Intrabony” palatal pleomorphic adenoma: A case report and review of the literature. Sahel Med J [serial online] 2021 [cited 2021 Apr 22];24:60-3. Available from: https://www.smjonline.org/text.asp?2021/24/1/60/312741




  Introduction Top


Pleomorphic adenoma (PA) is the most common neoplasm of the large salivary glands. It derives its name from the architectural pleomorphism seen by light microscopy.[1] It is also known as the mixed tumor salivary type, which describes its pleomorphic appearance as opposed to its dual origin from epithelial and myoepithelial elements. PA affects mostly the parotid gland, less frequently the minor salivary glands, and accounts for 73% of all salivary tumors.[2] In terms of location, the palate is the most common site for mixed tumor of minor salivary glands owing to the preponderance of minor salivary glands in this region.[3] Other sites commonly affected are the lips, buccal mucosa, tongue, floor of the mouth, tonsil, pharynx, neck, and nasal cavity.[1] The majority of minor salivary gland PAs occurs in the second decade of life and there is a slight predilection for the female gender.[2]

The clinical presentation of PA of the hard palate varies from a firm or rubbery submucosal mass to a soft lipoma-like mass without ulceration or surrounding inflammation.[2] A combination of bony hard and firm or only bony hard consistencies is rare.[4] Furthermore, pain and tenderness are unusual except when infected. Radiographically, little is seen in plain radiographs with panoramic view only showing if the palatal bone is intact or not.[2] The use of plain views is deficient due to limitations which includes variable magnification, distortion and superimposition of structures[3] Computed tomography (CT) scan, especially two-phase helical CT, is an indispensable tool in the evaluation of palatal PA as it gives both bone and soft tissue information.[3] Differential diagnosis of the palatal PA includes other minor salivary gland tumors, particularly mucoepidermoid carcinoma.[1]

Histologically, PA has a highly variable appearance.[1] It appears as an encapsulated lesion when it develops in major salivary glands, while the capsule is incomplete when it forms in minor salivary glands.[1] Classically, it is biphasic and is characterized by admixture of polygonal epithelial and spindle-shaped myoepithelial elements in a variable background stroma that may be mucoid, myxoid, cartilaginous, or hyaline.[5]

The treatment of choice for PA in minor salivary gland is wide local excision with the removal of periosteum or bone if they are involved.[6]


  Case Report Top


A 31-year-old male secondary school teacher presented to the oral and maxillofacial surgery clinic with a palatal mass of 16-year duration. The mass was slowly growing and relatively asymptomatic until a year before presentation that the patient noted a faster growth rate from the mass and that his upper teeth seemed to have shifted. He also noticed that the way he talked has changed. On examination, the mass extended posteriorly from the first premolar on the right side, and bulging over the midline, to the soft palate, sparing the uvula, about 5 cm × 5 cm. It had soft to rubbery consistency posteriorly and firm consistency more anteriorly, with notable palatal expansion toward the palatal rugae. The first to third molars were mobile (Grade II). A differential diagnosis of palatal PA was made and the patient was asked to do fine-needle aspiration and cytology (FNAC) and a CT scan of the face. He, however, could not continue with the treatment plan due to financial constraint. He presented again 2 years later. The swelling has increased in dimension, now 7 cm × 5 cm [Figure 1]. FNAC showed admixture of clusters of epithelial and mesenchymal cells on a background of amorphous material. CT scan shows intrabony growth of the mass with superior and inferior bony boundaries [Figure 2].
Figure 1: Preoperative picture showing a palatal mass

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Figure 2: Computed tomography scan showing the mass completely intrabony

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Posterior low-level maxillectomy was successfully done on the right side under general anesthesia. There was a residual oroantral and oronasal communication of about 3 cm × 2 cm, which was treated by suturing a sufratulle pack in place intraoperatively, and changed postoperatively on an alternate day basis for 2 weeks. It is important to say that the patient was given the options of intermediate and definitive obturator, but he could not afford the cost of any form. Nevertheless, he seems to have healed well as the communication reduced to about 1 cm × 1 cm in dimension after almost 2 years of follow-up [Figure 3].
Figure 3: Postoperative oronasal fistula

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  Discussion Top


PA exhibits diverse variation in sizes. Because the tumor appears as an asymptomatic mass, and due to its slow growth, it can reach a very large proportions before patient presentation. [1],[2] The late presentation and of course, the large size is evident in our patient presenting, 16 years after initially seeing a swelling in his mouth. Compromise of vital functions such as breathing, feeding, and phonetics has also been documented.[4] Our patient, however, complained of only defect in phonetics.

Radiographic investigation of maxillofacial swellings is indispensable due to the presence of so many vital structures and the diverse nature of diseases that can occur[6] as it provides information on location and extent and/or proximity to vital structures.[5] Plain X-ray plays an insignificant part in the diagnosis of salivary gland tumor of the palate, due to image distortion as pointed out by Patel et al.[2] Ultrasonography and magnetic resonance imaging (MRI) have also been used with different limitations.[3] CT is the goal standard and has an advantage over MRI when evaluating bone, especially in diagnosing erosion and perforation of the bony part of the palate.[7] We found multiple slices in different planes, coronal, axial, and para-sagittal, very useful as the coronal view initially gave the mass as entirely intrabony. This was confusing because of other differential diagnoses of a palatal mass, such as giant-cell fibroma; osteochondroma of the palate; and malignant ones such as primary intraosseous carcinoma and osteosarcoma that could grow de novo from the bone. While all these usually present with bony hard consistency, at least at their early stages, PA as a rule is never bony hard, because of its soft tissue origin. However, when the lesion was viewed in different planes [Figure 4], it was confirmed to have secondarily become “intrabony.”
Figure 4: Axial computed tomography showing the mass without bony floor

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Treatment of palatal PA requires a wide local excision of the tumor including its surrounding capsule,[4] together with clear margins involving the periosteum and associated mucosa, because surgical exposure of the tumor or its capsule could cause spillage and this increases the risk of recurrence.[3] In our patient, a posterior low-level maxillectomy seemed like the most practicable because of the well-outlined bony borders on the CT scans. This was essentially to ensure a total removal of the lesion. Patigaroo et al.[4] and Bordoy-Soto et al.[6] also pointed out the need to do a maxillectomy in very large cases, and in Patigaroo et al.'s[4] review, a case required a total maxillectomy with orbital floor reconstruction.[4]

Reconstruction of the palate after surgical intervention is a challenging endeavor.[3] Consideration of the goals of reconstruction from both functional and esthetic view will help in deciding which reconstruction options will benefit the patient. Various reconstruction methods for the palatal defect have been reported.[8],[9] These methods include leave alone to granulate when the defect is limited to just soft tissue without involving bone. .[8] Bony defects involving the hard palate can be treated conservatively with an obturator.[8] However, bony defects that involve the upper alveolar ridge and/or parts of the maxilla will cause a significant cosmetic and functional deformity and therefore may require a tissue transfer.[9] Each reconstructive option has its merits and demerits.

In the leave alone modality, the time of surgery is short as there is no need for tissue transfer and it is cost-friendly because maxillary obturator might seem expensive to some patients as in our case. The major challenge here is the healing, which is not predictable as there are many factors, local and systemic, that affect it. The obturator placement is a good option too. The duration of surgery is also short, but the cost and availability of a maxillary prosthodontics could really limit its use, excluding the need to change and refabricate it. Tissue transfer is arguably the best choice in that, there is no foreign body introduction i.e. maxillary obturator, there is good improvement in aesthetics and function and replacement of lost tissue in like for like manner, but the technical expertise, cost and duration of surgery are a great draw back. Notwithstanding, we used the option of leave alone to granulate in our patient, without placing an obturator. This was because the patient could not afford the cost of this prosthesis, but the patient seemed to have done very well. Plan is in process to close the remaining oronasal fistula primarily, but the patient is yet to consent almost 2 years after the first surgery.


  Conclusion Top


PA of the palate is a slow and progressively growing lesion that exhibits firm consistency and variable dimensions which can vary from small dimension without any symptom to a giant mass with phonetic, masticatory, and respiratory disturbances. CT scan with multiplanar images is indispensable, especially to rule out other differential. Leave alone option in reconstruction is also feasible, especially in carefully selected cases as it reduces or prevents the complications seen with complex tissue transfer and artificial prosthesis use.

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.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Neville B, Damm D, Allen C, Bouquot J. Salivary glan pathology. In: Oral and Maxillofacial Pathology. Ch. 11. 2nd ed. New York: W.B Saunders Company; 2002. p. 410.  Back to cited text no. 1
    
2.
Vellios F, Shafer WG. Tumors of minor salivary glands. Surg Gynecol Obstet 1959;108:450-6.  Back to cited text no. 2
    
3.
Patel MF, Ord R, Brennan PA. Radiological investigations and imaging techniques. In: Langdon JD, Mohan F, editors. Operative Oral and Maxillofacial Surgery. Ch. 1. 2nd ed. London: CRC Press. 2017. p. 3.  Back to cited text no. 3
    
4.
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. 4
    
5.
Kawahara A, Harada H, Kage M, Yokoyama T, Kojiro M. Characterization of the epithelial components in pleomorphic adenoma of the salivary gland. Acta Cytol 2002;46:1095-100.  Back to cited text no. 5
    
6.
Bordoy-Soto MA, Vélez-Gimón HJ, Hernández MF, Xavier-Briceño W. Giant pleomorphic adenoma of the palate. Case report and literature review. Revista Odontológica Mexicana 2016;20:e252-7.  Back to cited text no. 6
    
7.
Noghreyan A, Gatot A, Maor E, Fliss DM. Palatal pleomorphic adenoma in a child. J Laryngol Otol 1995;109:343-5.  Back to cited text no. 7
    
8.
Genden EM, Buchbinder D, Urken ML. The submental island flap for palatal reconstruction: A novel technique. J Oral Maxillofac Surg 2004;62:387-90.  Back to cited text no. 8
    
9.
Germain MA, Hartl DM, Marandas P, Juliéron M, Demers G. Free flap reconstruction in the treatment of tumors involving the hard palate. Eur J Surg Oncol 2006;32:335-9.  Back to cited text no. 9
    


    Figures

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



 

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