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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 14  |  Issue : 2  |  Page : 145-149

Ravaged mandibular ramus: Two rare case presentations of unicystic ameloblastoma with a view on management


1 Department of Oral and Maxiilofacial Surgery, Appolo Adlux Hospital, Cable junction, Angamaly, Kerala, India
2 Department of Oral Pathology, Malabar Dental College and Research Centre, Edappal, Kerala, India

Date of Submission05-Jun-2021
Date of Decision10-Oct-2021
Date of Acceptance28-Jan-2022
Date of Web Publication01-Jul-2022

Correspondence Address:
S Akhil
Department of Oral Pathology, Malabar Dental College and Research Centre, Manoor Chekanoor Road, Mudur P O, Malappuram, Edappal - 679 578, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jorr.jorr_43_21

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  Abstract 


Unicystic ameloblastoma (UA) is a rare, slow-growing, persistent, locally invasive, and cystic variant of ameloblastoma. This article presents two different variants of UA. The first case was a rare presentation of UA in an 11-year-old patient involving the impacted second molar, which is otherwise usually associated with an impacted mandibular third molar. It was diagnosed with UA-luminal type and treated by a piezoelectric ultrasonic device with minimal trauma to soft tissue. The second one was associated with impacted mandibular third molar extending the entire ramus of mandible till the coronoid which was diagnosed with UA-mural type and treated by cystic enucleation with platelet-rich fibrin graft. Recurrence was not observed during a 6-month follow-up. The aim of this case presentation was to give an insight into the destructive nature of UA and its diagnosis and management to maxillofacial surgeons as well as dental practitioners.

Keywords: Ameloblastoma, mandible, unicystic ameloblastoma


How to cite this article:
Mathew J, Akhil S, Thomas J, Ali F. Ravaged mandibular ramus: Two rare case presentations of unicystic ameloblastoma with a view on management. J Oral Res Rev 2022;14:145-9

How to cite this URL:
Mathew J, Akhil S, Thomas J, Ali F. Ravaged mandibular ramus: Two rare case presentations of unicystic ameloblastoma with a view on management. J Oral Res Rev [serial online] 2022 [cited 2022 Aug 16];14:145-9. Available from: https://www.jorr.org/text.asp?2022/14/2/145/349709




  Introduction Top


Ameloblastoma is the most common odontogenic tumor after odontoma.[1] It is a locally invasive true neoplasm that arises from the remnants of the dental lamina and odontogenic epithelium that represent about 1% of all oral tumors.[2] In 2017, the World Health Organization classified conventional ameloblastoma into:

  • Ameloblastoma, unicystic type
  • Ameloblastoma, extraosseous/peripheral type
  • Metastasizing (malignant) ameloblastoma.[3]


The unicystic ameloblastoma (UA) is the second and sporadic growth pattern seen among the intraosseous ameloblastomas occur as a single cystic cavity, with or without luminal proliferation. It has an incidence rate of around 6% of total ameloblastomas and is commonly seen during the second and third decades of life.[4]

In this article, we report two cases of different variants of UA, one involving the impacted second molar and the other one affecting the impacted mandibular third molar.


  Case Reports Top


Case report-1

An 11-year-old female patient reported to a private dental clinic with the chief complaint of pain and swelling in relation to the lower right back tooth region for 1 month [Figure 1]. The swelling was ovoid with a size measuring approximately 5 cm × 4 cm. On intraoral examination, dental caries was noticed in relation to 46. Root canal treatment was started and canals were accessed for the same. While taking intraoral periapical radiographs for working length, a huge radiolucency involving the distal root of 46 and 47 was observed. The patient was referred to the oral surgery department of our institute. Intraoral examination revealed a slight diffuse swelling in the posterior region of the right side of the mandible. The swelling was firm in consistency with a smooth surface. On palpation, the buccal and lingual cortical plate appeared to be expanded. The patient was advised to take cone-beam computed tomography (CBCT), but due to financial constraints, they declined to do so. A panoramic radiograph was taken which revealed a large well-defined unilocular radiolucency of size nearly 6 cm × 5 cm. It was extending from the mesial aspect of 46 cm–2 cm below the sigmoid notch and superoinferiorly extending from the alveolar crest to the inferior border of the mandible in its most significant dimension. There was thinning of the inferior border of the mandible without any break in its continuity [Figure 2]. Based on the clinical and radiographic characteristics, a provisional diagnosis of osteolytic lesion was made. Routine blood examinations were carried out, and it was found to be normal. Aspiration from the cystic cavity showed a straw-colored fluid which when sent for protein estimation, showed a total protein content of more than 5 g/100 ml. After obtaining the necessary consent from the patient's parents for treatment, the lesion was approached intraorally and an excision biopsy with a quadrilateral flap design was done under general anesthesia. A piezoelectric ultrasonic device was used to remove the lesion by generating mechanical micrometric ultrasonic vibrations, which allowed the preservation of the bone surface without damaging the normal cell and minimal trauma to the soft tissue. Neurovascular bundle and vital structures were preserved with complete enucleation of lesion along with the extraction of 46, 47. The opening of enucleated area was maintained with a removable obturator. No drain was placed since wound margins were free of bone and wound dehiscence was expected. The histopathological examination revealed multiple cystic spaces with fibrous wall and lumen. It showed the presence of numerous odontogenic islands [Figure 3]. A cyst was lined by odontogenic epithelium, which exhibited basal columnar cells with palisading nuclei that showed a reversal of polarity and basilar cytoplasmic vacuolization. Superficial cells resembled loose stellate reticulum and the lumen contain amphiphilic material. The connective tissue wall showed numerous small islands and strands of odontogenic epithelium. A final diagnosis of UA-luminal type was made based on clinical and histopathological findings. The patient is kept under observation and postoperative radiographs were taken with 3-month and 6-month [Figure 4] duration, which showed excellent bone regeneration. Further postoperative follow-up was not possible since the patient had refused to visit our college citing the COVID-19 pandemic situation.
Figure 1: Extra oral view of the patient

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Figure 2: Orthopantamograph (OPG) showing a well-defined radiolucency extending along ramus to lower border of mandible

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Figure 3: Photomicrograph showing multiple ameloblastomatous follicles in the cystic wall (H and E, ×4)

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Figure 4: OPG showing bone deposition at the enucleated area after 6 months

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Case report–2

A 24-year-old male patient presented to the department of oral and maxillofacial surgery with a chief complaint of pain and swelling in the right lower back tooth region for 2 weeks [Figure 5]. The swelling was slow-growing for the past 6 months with mild pain, which was gradual in onset, continuous, and localized. He used to take painkillers frequently without medical consultation. On extraoral examination, an obvious facial asymmetry was detected with diffuse swelling of size 4 cm × 3 cm extending anteroposteriorly from the corner of the mouth to the angle of the mandible and superioinferiorly from the ala-tragal line to the inferior border of the mandible. Intraoral examination revealed an ill-defined swelling of 2 cm diameter in the right vestibular sulcus in relation to 46, 47, which was seen extending from the 46 region to the pterygomandibular raphe anteroposteriorly. A panoramic radiograph was taken which showed a large unilocular radiolucency of approximately 8 cm × 4 cm size, extending from the root apex of 46 to the coronoid process involving the ramus of the mandible [Figure 6]. Tooth 48 appeared to be impacted, abutting the inferior border of the mandible. CBCT revealed lingual expansion [Figure 7] and a provisional diagnosis of a cystic lesion was made. Routine blood investigations were found to be normal.
Figure 5: Extraoral view of the patient

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Figure 6: Orthopantamograph (OPG) showing a well-defined radiolucency extending along ramus with third molar seen at pushed to lower border of mandible

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Figure 7: CBCT showing the expansion of buccal and lingual plates

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After obtaining consent, cystic enucleation of the entire lesion with the removal of 46, 47, and impacted 48 was carried out. Platelet-rich fibrin (PRF) graft was placed with sutures at enucleated site since the author had encountered many cases of extensive mandibular lesions which showed good healing after placement of PRF.

Histopathological examination revealed a highly inflamed connective tissue capsule which was lined by 3–4 layers of short columnar epithelium. Some sections showed ameloblastomatous epithelium proliferating into the connective tissue capsule in a plexiform pattern with few peripheral columnar ameloblastic-like cells [Figure 8]. The features were suggestive of a UA-mural variant arising from a dentigerous cyst. The patient was recalled after 3 months and the postoperative radiograph showed satisfactory healing [Figure 9]. We were not able to carry out further follow-up examinations as the patient did not turn up due to the COVID-19 pandemic.
Figure 8: Photomicrograph showing proliferation of ameloblastomatous epithelium into the connective tissue capsule in a plexiform pattern (H and E, 4×)

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Figure 9: OPG showing bone deposition at the enucleated area after 3 months

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  Discussion Top


In 1977, Robinson and Martinez first described the existence of UA and advocated for recognition as a separate entity. The term UA refers to those cystic lesions which showed clinical, radiographical, or gross features of a jaw cyst, but on histologic examination showed a typical ameloblastomatous epithelial lining, with the presence or absence of luminal and or mural tumor proliferation.[5] The lesions are usually asymptomatic, more than 90% of UA reported are found in the posterior region of the mandible involving the third molar.[6] However, in our initial case report which is mentioned in this article, the lesion was associated with a second molar, which is a rare occurrence.

Gupta et al. also reported similar UA cases in an 18-year-old female patient with an impacted mandibular second molar. In this case, the lower border of the mandible was partially damaged, and thinning was evident. Hemimandibulectomy was their treatment of choice.[7] As compared to their presentation, in our case, the lower border of the mandible was thinner without breaking in its continuity, thus preserving the bone surface intact. Complete enucleation of the lesion was carried out without damaging the cell along with minimal trauma to the soft tissue and vital structures.

Unilocular and multilocular radiographical patterns of UA are seen. Vickers and Gorlin described the histological criteria for the diagnosis of UA. Ackermann classified three histopathological variants of UA as, luminal (type 1), intraluminal or plexiform (type 2), and mural (type 3) [Table 1].[8]
Table 1: Histopathologiocal variants of unicystic ameloblastoma

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Tandon A in 2014 reported a case of an aggressive counterpart of UA with dysplastic features. Surgery (jaw resection) is a mode of treatment in ameloblastoma, which can be conservative or radical resection. Long-term follow-up of the lesion is necessary after meticulous treatment.[9] The primary objective of the treatment of ameloblastoma is tumor elimination, restoration, esthetics, and the prevention of recurrence of the lesion.[10]

Three factors which complicate the treatment of ameloblastoma in children and adolescents include:

  1. The continuing facial growth and different bone physiology (more cancellous bone, increased bone turnover, and reactive periosteum)
  2. The presence of unerupted teeth and the difficulty in the initial diagnosis
  3. The predominance of the UA.


All of the above factors can create obstacles in resection and reconstruction effects on maxillofacial development to some extent.[11] Treatment planning or prognosis of a UA may depend on patient age, tumor size, location, radiographic appearance, final histopathology, and whether it is a recurrent lesion or initial presentation. Several treatment approaches including enucleation and marsupialization with an adequate area of resection are put forward, but primarily conservative treatment has been the treatment of choice suggested for younger patients. Carnoy's solution, when applied over the enucleated area showed less recurrence of UA due to its deep penetration and fixation properties.[12]

When patients came with complaints of pain and swelling in the maxilla or mandible, it is advisable to take suitable radiographs so that lesions in those areas can be detected at the earliest.


  Conclusion Top


The primary treatment option for ameloblastoma in young patients should be timely intervention and conservative surgery. Thorough knowledge of various surgical procedures added with excellent diagnosing skills and its meticulous application should prima facie be the motto of any maxillofacial surgeon as it will be of immense help in the psychological well-being and functional rehabilitation of the patient. Long-term follow-up is deemed necessary, due to its risk of recurrence.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patients have given their consent for 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.
Masthan K, Anitha N, Krupaa J, Manikkam S. Ameloblastoma. J Pharm Bioall Sci 2015;7:S167-70.  Back to cited text no. 1
    
2.
Agani Z, Hamiti-Krasniqi V, Recica J, Loxha MP, Kurshumliu F, Rexhepi A. Maxillary unicystic ameloblastoma: A case report. BMC Res Notes 2016;9:469.  Back to cited text no. 2
    
3.
Soluk-Tekkeşin M, Wright JM. The world health organization classification of odontogenic lesions: A summary of the changes of the 2017 (4th) edition. Turk Patoloji Derg 2018;34:1-18.  Back to cited text no. 3
    
4.
Dave PM, Galinde JS, Poonja KS. Unicystic ameloblastoma. J Contemp Dent 2015:5:40-2.  Back to cited text no. 4
    
5.
Al Sineedi F, Aruveetil YA, Kavarodi AM, Harbi SO. Bilocular unicystic ameloblastoma of the mandible in a 9 yr old child – A diagnostic and management dilemma. Saudi Dent J 2018;30:250-5.  Back to cited text no. 5
    
6.
Kubbi JR, Tipirisety S, Dubbudu R, Oruganti RV. Unicystic ameloblastoma of mandible: Nondentigerous variant – A rare case report. J Indian Acad Oral Med Radiol 2016;28:420-3.  Back to cited text no. 6
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7.
Gupta N, Saxena S, Rathod VC, Aggarwal P. Unicystic ameloblastoma of the mandible. J Oral Maxillofac Pathol 2011;15:228-31.  Back to cited text no. 7
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8.
Saxena N, Choudhary SH, Aldhuwayhi SD, Thakare A. Infected multilocular unicystic ameloblastoma involving ramus and coronoid process: A rare case report. Contemp Clin Dent 2020;11:179-83.  Back to cited text no. 8
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9.
Tandon A. Malignant squamous changes in unicystic ameloblastoma: A case report and review of literature. J Interdiscip Med Dent Sci 2014;2:132.  Back to cited text no. 9
    
10.
Agbaje JO, Olumuyiwa Adisa A, Ivanova Petrova M, Adenike Olusanya A, Osayomi T, Ajibola Effiom O, et al. Biological profile of ameloblastoma and its location in the jaw in 1246 Nigerians. Oral Surg Oral Med Oral Pathol Oral Radiol 2018;126:424-31.  Back to cited text no. 10
    
11.
Kim J, Nam E, Yoon S. Conservative management (marsupialization) of unicystic ameloblastoma: Literature review and a case report. Maxillofac Plast Reconstr Surg 2017;39:38.  Back to cited text no. 11
    
12.
Bhutia O, Roychoudhury A, Arora A, Mallick S. Management of unicystic ameloblastoma of the mandible in a 5-year old child. Natl J Maxillofac Surg 2013;4:232-4.  Back to cited text no. 12
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