|Year : 2012 | Volume
| Issue : 3 | Page : 172-175
Plexiform ameloblastoma of the maxilla with extension to sinonasal cavity: Presenting with loosening of teeth
Sankappa P Sinhasan
Department of Pathology, Indira Gandhi Medical College and Research Institute, Pondicherry, India
|Date of Submission||26-Oct-2012|
|Date of Acceptance||23-Jan-2013|
|Date of Web Publication||29-Apr-2013|
Sankappa P Sinhasan
Department of Pathology, Indira Gandhi Medical College and Research Institute, Pondicherry
Source of Support: None, Conflict of Interest: None
Ameloblastomas are locally aggressive jaw tumors with a high propensity for recurrence that are believed to arise from remnants of odontogenic epithelium, lining of odontogenic cysts, and the basal layer of the overlying oral mucosa. The unusual presenting symptoms, as well as the highly destructive nature of these lesions when arising in the maxilla, make them worthy of consideration in the differential diagnosis of nasal and maxillary masses. We present a rare case of right maxillary ameloblastoma in a 48 year old male patient who presented with the main complaint of loosening of molar teeth. History also revealed unilateral nasal obstruction of long duration about which the patient was least concerned. The computed tomography scan revealed a soft tissue polypoidal mass in the right maxillary sinus involving alveolar arch of the maxilla, with expansion of sinus and erosion of posterolateral wall. Initial biopsy confirmed ameloblastoma, which was followed by excision of the tumor by radical surgery. The case represents an unusual presentation of a rare odontogenic tumor.
Keywords: Ameloblastoma, maxilla, sinonasal
|How to cite this article:|
Sinhasan SP. Plexiform ameloblastoma of the maxilla with extension to sinonasal cavity: Presenting with loosening of teeth. Indian J Oral Sci 2012;3:172-5
|How to cite this URL:|
Sinhasan SP. Plexiform ameloblastoma of the maxilla with extension to sinonasal cavity: Presenting with loosening of teeth. Indian J Oral Sci [serial online] 2012 [cited 2019 Nov 20];3:172-5. Available from: http://www.indjos.com/text.asp?2012/3/3/172/111185
| Introduction|| |
Ameloblastomas are epithelial-derived odontogenic tumors that typically originate in jaw bones, primarily involving the mandible (80% of the time) and less often the maxilla (20% of the time) in the region of the third molar.  In case of upper maxillary lesions, radical resection is advised due to the spongy osteoarchitecture of the maxilla, which facilitates the diffusion of the tumor to the maxillary antrum, sinus ethmoidalis, pterygomaxillary fossa, temporal fossa, and base of skull. Complete excision in this area is difficult.  This article aims to report a case of a plexiform ameloblastoma of rare location, emphasizing the peculiar clinical presentation and histopathological aspects that are relevant for the diagnosis and treatment.
| Case Report|| |
A 48-year-old male patient was presented to the dental clinic at our District Hospital, Hassan, Karnataka, India, with a primary complaint of loosening of the right-sided upper molar teeth. There was no history of trauma and his past medical history was unremarkable. History also revealed right-sided nasal obstruction of long duration about which the patient was least concerned. Intraoral examination revealed a symptomatic soft tissue mass hanging in the retromolar trigone area with ulceration, reddish coloration, and irregular surface of imprecise outlines [Figure 1]. Loosening of upper molar teeth was confirmed due to mass effect of the tumoral growth. Incisional biopsy of the mass was taken under local anesthesia and sent for histopathological examination. The computed tomography (CT) scan revealed a soft tissue polypoidal mass in the right maxillary sinus involving alveolar arch of maxilla with the expansion of sinus and erosion of posterolateral wall [Figure 2]. The soft tissue mass also showed extension to frontal and ethmoid sinuses on the right side. The left side nasal cavity was normal. The CT diagnosis of "dental cyst with possibility of right maxillary sinus malignancy" was issued. The biopsy revealed fragments of a benign neoplasia of odontogenic origin, characterized by the proliferation of small odontogenic epithelium islands and cords interlarded by a myxoid to fibrous stroma. In the center of the islands, loosely organized cells were found, resembling the stellate reticulum of the enamel origin. From these findings, a histopathological diagnosis was issued as Ameloblastoma of Maxilla. The patient was posted for wide resection of tumor by radical maxillectomy [Figure 3] under general anesthesia after the grave risk consent.
|Figure 1: Intraoral examination showing tumoral lesion with areas of surface ulceration in the right maxilla corresponding to retromolar trigone location|
Click here to view
|Figure 2: The CT image showing a soft tissue polypoidal mass in the right maxillary sinus involving alveolar arch of maxilla, with expansion of sinus and erosion of posterolateral wall|
Click here to view
Gross examination of the surgical specimen consisted of a portion of the maxilla, extending from distal of the canine to distal of the second molar. The whole specimen measured 10 × 6 × 5 cm [Figure 4]. The tumoral area was a solid mass, rubbery to spongy in consistency. Cut surface showed grey white to myxoid appearance. The specimen was subjected to histopathological examination.
|Figure 4: Gross specimen: Portion of the maxilla with the tumor having rubbery to spongy nature showing myxoid appearance|
Click here to view
Microscopic examination of hematoxylin and eosin stained sections showed predominantly plexiform pattern, comprising network of long anastomosing cords of odontogenic epithelium [Figure 5] showing focal areas of squamous metaplasia with acanthomatous appearance [Figure 5]. The epithelial strands are bounded at the periphery by a layer of columnar cells having hyperchromatic nucleus, with subnuclear vacuolation, peripheral palisading with reversed polarized nuclei [Figure 6]. The epithelium at focal areas shows squamous metaplasia with acanthomatous appearance [Figure 7]. In the center of the islands, loosely organized cells were found, resembling the stellate reticulum of the enamel origin. The stroma was myxoid to desmoplastic, with sparse lymphoplasma cell infiltration. Areas of cystic change with accumulation of foamy macrophages were also seen. A small focus of calcification was seen within the plexiform strands of tumor cells. Tumor extension into sinonasal respiratory epithelium was noted at focal areas. Microscopic impression of "Plexiform Ameloblastoma of Maxilla with extension to sinonasal cavity" was issued.
|Figure 5: Plexiform pattern of ameloblastoma showing back-to-back anastomosing cords of islands of odontogenic epithelium. In the center of the islands, loosely organized cells were found, resembling the stellate reticulum of the enamel origin. (H and E, ×10 magnification)|
Click here to view
|Figure 6: Ameloblastoma showing epithelium with a peripheral palisading of columnar cells having hyperchromatic nucleus, with subnuclear vacuolation and reversed polarized nuclei. The stroma is desmoplastic. (H and E, ×40 magnification)|
Click here to view
|Figure 7: Squamous metaplasia within the center of the ameloblastoma islands is characteristic of acanthomatous growth pattern. (H and E, ×10 magnification)|
Click here to view
Post-operatively, the patient is asymptomatic without recurrence after 6 months of follow-up. However, he has been advised for long-term follow-up.
| Discussion|| |
The ameloblastoma is a relatively rare dental tumor, described for the first time in 1827 by Cusack, and so named by Ivey and Churchill in 1934.  According to Larsonn et al., its incidence is 0.6 cases per million, while Shear et al. found an incidence of 0.31 cases per million in a white population in South Africa.  General pathologists will see odontogenic tumors in 0.003% of their specimens.  Ameloblastoma of the maxilla can cause an increase in volume, pain, poor occlusion, and paresthesia of the affected area.  In the case reported here, the patient had a loosening of upper molar teeth with painful symptomatology and areas of ulceration, probably because of the size of the lesion and the trauma during mastication. Moreover, the tumor had extended into the sinonasal region from a primary gnathic origin. Involvement of the sinonasal region by maxillary ameloblastoma usually causes symptoms such as obstruction and epistaxis. They may require a longer period of time before attaining sufficient size to present with symptoms.
Ameloblastoma may manifest with a number of histological patterns, including the follicular, plexiform, acanthomatous, keratinizing, desmoplastic, granular cell, basal cell, and clear cell types. , In our case, it was predominantly plexiform pattern, although foci of follicular, acanthomatous, and desmoplastic areas were also seen. Hence, it is not uncommon to see more than one histological patterns in the same case. These have been designated as 'hybrid lesions'. 
In spite of the apparent and gross circumscription of the ameloblastomas, they have the potential for recurrence because they fail to produce a capsule; it is therefore recommended that they should always be treated by complete surgical resection.  Considering the fact that conservative treatment of the maxillary bones offers local recurrence in 100% of the cases and 60% of mortality rate, it is important that the patients with this pathology have a life-long follow-up with the help of CT and magnetic resonance tomography (MRT).  The therapy of the ameloblastoma is surgical, but the most suitable operation has not been established yet. The decision to use a radical or conservative approach depends on various factors: (1) the dimensions and location of the lesion, (2) the growth rate and relationship with the surrounding structures, (3) the histological type, (4) the clinical characteristics, in the recurrences, (5) general conditions of health and age of the patient.  While planning the treatment of ameloblastoma, it is important to understand the growth characteristics and removing the full extension of tumor, including the surrounding tissue. Other factors to be considered during operation of ameloblastoma are solid nature of tumor and destruction of the inferior border of mandible. Reconstruction procedures are good for functioning and aesthetic mandibular problems.  Reconstruction within the head and neck is challenging and various developments include the use of custom-made implants, robotic surgery, tissue engineering, distraction osteogenesis, and facial allotransplantation. 
| Conclusion|| |
We conclude that ameloblastoma can also present as loosening of teeth with its displacement as an unusual clinical presentation, which should not be neglected as old age-related root resorption and disruption. The initial intralesional biopsies are efficient because they are easy to perform and yield near accurate diagnosis. The ameloblastomas may show different patterns of histology in different areas, which needs thorough sampling of the tissue with maximum study of various areas of tumor. Therefore, for intraoral tumoral lesion, we think that it is advisable, considering the following: the site and extension of the lesion, age and general conditions of the patient, to remove the lesion in a conservative manner in the first surgical step, and according to the histological aspect, evaluate a possible radical resection.
| Acknowledgments|| |
The author expresses his gratitude to Dr. Bhagawan, Professor and Head of Department, Department of Dentistry, and Dr. Raghavendra Prasad, Assistant Professor, Department of ENT, Hassan Institute of Medical Sciences (HIMS), Hassan, Karnataka, India, for the help rendered.
| References|| |
|1.||Kenneth D, McClatchey, Zarbo RJ. The Jaws and Oral Cavity. In: Sternberg SS, editor. Diagnostic surgical pathology. 3 rd ed. Philadelphia: Lippincott Williams and Wilkins; 1999. p. 842-6. |
|2.||Di Cosola M, Turco M, Bizzoca G, Tavoulari K, Capodiferro S, Escudero CN, et al. Ameloblastoma of the jaw and maxillary bone: Clinical study and report of our experience. AvOdontoestomatol 2007;23:367-73. |
|3.||Alves PM, Pereira KM, Vasconcelos MG, Souza LB, Queiroz LM, Medeiros AM. Desmoplastic ameloblastoma in maxilla: Report of case and review of the literature. Int J Morphol 2008;26:263-8. |
|4.||Shashikanth MC, Neetha MC, Ali IM, Shambulingappa P. Desmoplastic ameloblastoma in the maxilla: A case report and review of literature. Indian J Dent Res 2007;18:214-7. |
|5.||Schafer DR, Thompson LD, Smith BC, Wenig BM. Primary ameloblastoma of the sinonasal tract: A clinicopathologic study of 24 cases. Cancer 1998;82:667-74. |
|6.||Memon GA, Khatoon S, Junejo A, Jamal A. Clinical Pattern and Management of Ameloblastoma at Tertiary Care Hospital of Sindh. Med Channel 2012;16:189-91. |
|7.||Balasundaram I, Al-Hadad I, Parmar S. Recent advances in reconstructive oral and maxillofacial surgery. Br J Oral Maxillofac Surg 2012;50:695-705. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7]