Median palatine cyst: an unusual presentation of a rare entity

Median palatine cyst: an unusual presentation of a rare entity

BJOM-102.QXD 6/26/01 9:46 PM Page 278 British Journal of Oral and Maxillofacial Surgery (2001) 39, 278–281 © 2001 The British Association of Oral a...

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British Journal of Oral and Maxillofacial Surgery (2001) 39, 278–281 © 2001 The British Association of Oral and Maxillofacial Surgeons doi: 10.1054/bjom.2001.0620, available online at http://www.idealibrary.com on

BRITISH

Journal of Oral and Maxillofacial Surgery

Median palatine cyst: an unusual presentation of a rare entity U. Hadi,* A. Younes,† S. Ghosseini,‡ A. Tawil§ *Clinical Assistant Professor; †Chief Resident; ‡Resident, 5th Year, Department of Otolaryncology–Head and Neck Surgery, American University of Beirut Medical Center; §Associate Professor, American University of Beirut, New York, NY, USA SUMMARY. Median palatine cyst is rare. Mostly, it is asymptomatic and usually is discovered incidentally during routine dental or radiological examination. The case that we report has the following unusual features: Firstly, it is the largest cyst to be reported, measuring 5 cm in diameter. Secondly, there was no swelling on the oral surface of the hard palate contrary to other reports. Rather, it caused elevation of the nasal floor and nasal obstruction. Thirdly, it pushed the inferior and caudal end of the septum into the left nasal chamber. The median palatine cyst was surgically removed by a sublabial degloving approach. The cyst was removed in toto and the palatal bone curetted to ensure adequate removal of any nesting cells that could lead to recurrence in the future. © 2001 The British Association of Oral and Maxillofacial Surgeons

INTRODUCTION

CASE REPORT

Median palatine cysts are rare, non-odontogenic fissural cysts of the hard palate. We know of only 17 cases that had been reported up to 1985 (Table 1).1 It is a diagnostic challenge and must be differentiated from other maxillary cysts.2–5 We operated on a patient with a median palatine cyst, the size of which is the largest reported so far, to our knowledge.

A 48-year-old white woman presented with a one-year history of bilateral nasal obstruction, more pronounced on the left side, which varied from partial to complete, associated with frontal headache and upper gingival pain. There was no history of allergic symptoms. She had had an operation 20 years earlier, at which a cyst was drained through a labial buccal approach; no pathology report was available to identify the nature of the lesion.

Table 1 Reported cases of median palatine cyst First author Rushton15 1930 Hyde16 1938 Sayer17 1943 Frerichs18 1953 Choukas19 1957 Choukas20 1964 Rhymes21 1964 Hatziotis5 1966 Meyer22 1966 Sutherland23 1968 Thornton24 1972 Courage9 1974 Taintor25 1977 Gordon26 1980 Clark7 1981 Yip27 1981 Gingell1 1985 Present case 1999

Age

Sex

Symptoms

Size (cm)

Site

Operation

– 44 48 22 52 42 25 27 38 40 42 23 47 45 13 34 44 48

F M M M M M M M M M F F M M M M F F

Tenderness Pain Pain Hole in palate Swelling Painful swelling Swelling Swelling – Swelling Swelling Swelling Numbness Swelling Swelling Swelling Swelling Nasal obstruction

– 1.34 – 23 22 34 – 22 – – 22 22 – – 44 23 2.53 55

– Anterior hard palate – – Mid-palate Mid-palate – Mid-palate – Anterior palate Anterior and mid-palate Anterior and mid-palate Anterior palate Most of hard palate Anterior and mid-palate Anterior palate Anterior and mid-palate Nasal floor elevation; septal deviation

Marsupialization Excision Enucleation Curettage Enucleation Enucleation Enucleation Excision Enucleation Enucleation Enucleation Enucleation Enucleation Curettage Enucleation Enucleation Enucleation Enucleation

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On examination, anterior rhinoscopy showed severe septal deviation to the right with bilateral elevation of the floor of the nose causing obstruction of more than 40% of the airway. The columella and upper lip looked normal. No swelling was noted in the area of the naso-alveolar groove. Oral examination showed no swelling in the gingivobuccal sulcus. The hard

palate was normal with no bulge or change in its colour or texture. All teeth were vital. Direct fiberoptic nasopharyngoscopy was unremarkable except for the swelling of the nasal floor that had been noted on anterior rhinoscopy. The rest of the examination and routine laboratory studies were not helpful. Computed tomography (CT) showed the presence of a large well-circumscribed opacity occupying the space between the hard palate and the septum (Fig. 1). There was no evidence of bulging of the oral surface of the hard palate or separation of the teeth (Fig. 2). The mass was exposed through a midfacial degloving translabial incision. The palatal mass was dissected and was found to be covered by a thin ballooned layer of bone that was the floor of the nose. The overlying mucosa and mucoperiosteum were gently dissected and lifted from the thinned bone all the way posteriorly, laterally, and towards the inferior aspect of the septum (Fig. 3). The mass consisted of a cyst measuring 5 cm in diameter. It had a smooth outer surface and a wellformed wall that was 1 mm thick. The cyst was inadvertently ruptured and some of its tan–white cheesy content oozed out. The cyst was enucleated (Fig. 4) and the defect in the floor of the palate and nasal cavity was packed with iodoform gauze, which was removed gradually over a period of three days. Culture of the cyst’s contents showed no growth. Microscopic examination showed a fibrous cyst wall lined by benign keratinizing stratified squamous epithelium without adnexal structures. There were no salivary glands, vascular, or neural elements in the cyst wall (Fig. 5). The postoperative course was smooth and uneventful. The patient was symptom-free and had no evidence of recurrence two years after the excision of the cyst.

Fig. 1 A 3-mm coronal CT scan of paranasal sinuses shows no connection between median palatine cyst and the teeth.

Fig. 2 A 3-mm coronal CT scan of paranasal sinuses shows a large median palatine cyst of the hard palate, presenting in the nasal cavity (large arrow). Note the absence of swelling of the oral surface of the hard palate (small arrows). Also note deviation of the caudal end of the septum to opposite side (arrowhead).

Fig. 3 A midfacial, degloving, translabial approach exposes the large palatine cyst that is lying on the nasal side of the hard palate (arrow).

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Fig. 5 Section of a portion of the cyst showing fibrous wall (large arrow) and benign keratinizing squamous epithelium (small arrows) (hematoxylin and eosin, original magnification100).

Fig. 4 Gross appearance of the cyst after its contents had been evacuated.

DISCUSSION The pathogenesis of median palatine cyst is controversial and Gorlin and Goldman6 have questioned its existence. It may be the result of abnormal palatal development during embryogenesis.7 Fusion of the palatal shelves of the maxilla with its ectodermally derived epithelium occurs during the sixth fetal week. Lack of complete disintegration of epithelium at the fusion line may result in the growth of such cysts along the midline of the hard palate.4,8 In vivo, palatal cultures support the concept that such cysts are an anomaly of this process of disintegration of epithelial remnants. However, these epithelial remnants proliferate later in adult life for unknown reasons to form cysts.5 Courage et al.9 suggested that synostosis

caused by the movement during mastication can trigger this process of proliferation. Others considered these cysts to be derived from odontogenic cell rests.3,10 Another theory proposed that they are primordial cysts of supernumerary tooth buds.11 The differential diagnosis of median palatine cyst includes incisive canal papilla cysts and anterior palatine maxillary cysts. Both can mimic palatine cysts because they may both present as a bulge on the oral surface of the hard palate, but they are located anteriorly. Zachariades and Papanikolaou3 stated that it is purely academic to subdivide the cysts of the palatal suture into median alveolar (anterior) and median palatine (posterior). The existence of a true median palatine cyst has been supported by the occurrence of posterior lesions that are too far back to be derived from incisive canal or nasopalatine duct cysts.12 Some oral and maxillofacial pathologists use the term ‘median palatal cyst’ to describe the infantile and neonatal cysts, which are self-limiting.13 These cysts should not be confused with median palatine cysts, despite having the same name. The clinical presentation is characterized by a welldefined swelling along the midline of the palate. It is radiolucent on radiography and may involve the floor of the nose.14 Most are asymptomatic and they are discovered as incidental findings during routine dental or radiological examination.2,5,9 The usual age at the time of diagnosis, as observed from the limited number of the cases reported, is 20–50 years. Men are affected more commonly than women (ratio 4 : 1). The cysts may

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become painful when the nasopalatine nerve is involved by local extension, or when the cyst is secondarily infected causing painful swelling and a sinus opening into the oral cavity.3 The teeth adjacent to the cyst must be vital with positive vitality pulp test to rule out a dental origin.3 Donnelly et al.2 considered this association with vital teeth to be a necessary diagnostic criterion. The cyst is usually treated by either enucleation or marsupialization (Partsch procedure).7 The former is the preferred mode of treatment for fissural cysts as the latter may delay healing. In most reported cases, cysts were managed intraorally by a posteriorly based mucopriosteal flap, enucleation of the cyst, followed by closure and application of a stent. The history of previous nasal surgery in our patient raises the possibility of incomplete excision of a previous cyst which recurred, as the persistence of lining epithelium of any cyst makes recurrence more likely. Our case was managed by simple enucleation through a translabial approach. The mucoperiosteal cover of the nasal cavity floor was elevated. The cyst was enucleated and the area of adhesion to the palate was curetted, debrided, and irrigated to ensure complete removal of epithelial cells and tissue debris. REFERENCES 1. Gingell JC, Levy BA, Depaola LG. Median palatine cyst. J Oral Maxillofac Surg 1985; 43: 47–51. 2. Donnelly JC, Koudelka BM, Hartwell GR. Median palatal cyst. J Endod 1986; 12: 546–549. 3. Zachariades N, Papanikolaou S. The median palatal cyst: does it exist? Report of three cases with oro-medical implications. J Oral Med 1984; 39: 173–176. 4. Cinberg JZ, Solomon MP. Median palatal cyst. A reminder of palate fusion. Ann Otol Rhinol Laryngol 1979; 88: 377–381. 5. Hatziotis J. Median palatine cyst: report of a case. J Oral Surg 1966; 24: 343–346. 6. Gorlin RJ, Goldman HM. Thoma’s Oral Pathology, 6th edn. St Louis; CV Mosby, 1970: 462. 7. Clark M. Median palatal cyst: report of a case of unusual size. N Y State Dent J 1980; 46: 20–22. 8. Meyer AW. Median Anterior Maxillary Cysts. JADA 1931; 18: 1851–1877. 9. Courage GB, North AF, Hansen LS: Median palatine cyst: review of the literature and report of a case. Oral Surg Oral Med Oral Pathol 1974; 37: 745–753. 10. Killey HC, Kay LW. Benign Cystic Lesions of the Jaws, 2nd edn. Edinburgh: Churchill Livingstone, 1972: 71–73. 11. Archer WH. Oral Surgery, 4th edn. Philadelphia: WB Saunders, 1966: 395.

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12. Thoma KH, Goldman HM. Oral Pathology, 5th edn. St Louis: CV Mosby, 1960: 817. 13. Flinck A, Paludan A, Matsson L, Holm AK, Axelsson. Oral findings in a group of newborn Swedish children. Int J Paediatr Dent 1994; 4: 67–73. 14. Thoma KH. Oral Surgery, 4th edn. St Louis: CV Mosby, 1963: 888. 15. Rushton MS. A cyst in the median palatine suture. Br Dent J 1930; 51: 109–110. 16. Hyde WH. Multiple fissural cysts. Am J Orthod Oral Surg 1938; 24: 893–895. 17. Sayer B, Scully JB. Fissural cysts. Am J Orthod Oral Surg 1943; 39: 320–327. 18. Frerichs DW, Spooner SW. Median palatine cyst. Oral Surg 1953; 6: 1181–1185. 19. Choukas NC. Case report of a median palatine cyst with criteria for a differential diagnosis. Oral Surg 1957; 10: 237. 20. Choukas N, Toto PD. Fissural cysts of the palate. Oral Surg 1964; 17: 497–502. 21. Rhymes R. Median palatine cyst: report of cases. J Oral Surg 1964; 22: 513–515. 22. Meyer I. Comments on report of median palatine cyst. J Oral Surg 1966; 24: 346. 23. Sutherland KG. The pathology and treatment of diseases of the palate. Aust Dent J 1968; 13: 111–124. 24. Thornton WE, Allen JW, Byrd DL. Median palatal cyst: report of case. J Oral Surg 1972; 30: 661–663. 25. Taintor JF, Fahid A. Median palatine cyst. Dent Surg 1977; 53: 33–36. 26. Gordon NC, Swann NP, Hansen LS. Median palatine cyst and maxillary antral osteoma: report of an unusual case. J Oral Surg 1980; 38: 361–365. 27. Yip MCM, Nguyen N, Hansen LS. A biochemical and clinical study of an uncommon lesion, the median palatine cyst, associated with pulpless teeth. J Endo 1981; 7: 407–412.

The Authors Usamah Hadi MD, FACS Clinical Assistant Professor Abbas Younes MD Chief Resident Soha Ghosseini Resident, 5th Year American University of Beirut Medical Center Ayman Tawil MD Associate Professor American University of Beirut New York, NY, USA Correspondence and requests for offprints to: Usamah Hadi MD, FACS, Department of Otolaryngology, Head and Neck Surgery, American University of Beirut, 850 Third Avenue, 18th Floor, New York NY 10022, USA. E-mail: [email protected] Accepted 23 January 2001