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 Table of Contents  
ORIGINAL ARTICLE
Year : 2022  |  Volume : 9  |  Issue : 1  |  Page : 59-65

Ameloblastoma of the jaws: A retrospective observational study of 131 cases at a tertiary level hospital in Bangladesh


1 Department of Oral and Maxillofacial Surgery, Rajshahi Medical College, Rajshahi, Bangladesh
2 Department of Oral and Maxillofacial Surgery, Dhaka Dental College and Hospital, Dhaka, Bangladesh

Date of Submission26-Nov-2021
Date of Acceptance01-Jul-2022
Date of Web Publication06-Apr-2022

Correspondence Address:
A F. M Shakilur Rahman
Department of Oral and Maxillofacial Surgery, Rajshahi Medical College, Rajshahi
Bangladesh
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jdrr.jdrr_181_21

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  Abstract 


Aim: The purpose of this study is to evaluate the incidence, radiological features, histological variant, and different treatment options of patients with ameloblastoma of the jaws treated at a tertiary-level hospital in Bangladesh. Materials and Methods: Age, gender, tumor location, radiologic features, histological pattern, and treatment modalities were all evaluated in this retrospective observational study. The frequency and percentages of these study variables were analyzed using descriptive statistics. Results: A total of 131 patients were included in this study, 66 of them were female, with a male-to-female ratio of 1:1.01. The patients had a wide age range from 4 to 70 years, a mean age (±standard deviation) of 26.61 (±13.34) years, and a significant incidence in the third decade of life. The left side of the jaws was the most frequently involved site. The mandible (n = 125, 95.42%) was the most frequently affected site, with the body–angle–ramus (n = 73, 58.5%) area being the most frequently involved location. Multilocular radiolucency was seen in most cases (n = 73, 55.73%) on radiography. The most common histological pattern (n = 71, 54.2%) was follicular. The most preferred treatment was the dredging (n = 90, 68.70%) method. The recurrence rate of the dredging treatment method was 5.56%. Conclusions: Ameloblastoma was found to be more common in patients in their third decade of life, with a wide age range. The follicular ameloblastoma was the predominant histological type, with most of them occurring in the mandible's body–angle–ramus area. Most of the patients were treated by the dredging method.

Keywords: Ameloblastoma, Bangladesh, dredging, jaws, mandible


How to cite this article:
Shakilur Rahman A F, Haider IA. Ameloblastoma of the jaws: A retrospective observational study of 131 cases at a tertiary level hospital in Bangladesh. J Dent Res Rev 2022;9:59-65

How to cite this URL:
Shakilur Rahman A F, Haider IA. Ameloblastoma of the jaws: A retrospective observational study of 131 cases at a tertiary level hospital in Bangladesh. J Dent Res Rev [serial online] 2022 [cited 2022 May 26];9:59-65. Available from: https://www.jdrr.org/text.asp?2022/9/1/59/342708




  Introduction Top


Ameloblastoma is a rare, locally invasive, benign, slow-growing epithelial odontogenic tumor.[1] It accounts for only 1.3% of all jaw tumors and cysts. It is the odontogenic tumor with the most clinical significance.[2],[3] Early symptoms are frequently absent, and tumors are only rarely detected in their early stages. It usually appears as a slow-growing, painless swelling [Figure 1]. Patients can also experience pain,[4],[5] draining sinuses, and mucosal ulcerations.[4] Mobility of teeth, ill-fitting dentures, malocclusion, and breathing difficulty are some of the less prevalent signs.[5]
Figure 1: Clinical features: Extra-oral facial swelling

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Most of the ameloblastomas are unilateral (95%) and arise in the jaws' posterior area (85%). The mandible is where most of the tumors are located (80%–93%).[6],[7] A multilocular radiolucency (“soap bubble”) image associated with the resorption of tooth roots is the most common radiographic characteristic.[8] Plexiform and follicular histological patterns are the two most common histological variants of ameloblastoma.[9] Acanthomatous, granular cell, desmoplastic, and basal cell types are among the less prevalent histological variants.[10]

The main goal of ameloblastoma treatment is to eradicate the tumor completely and restore oral function and esthetics.[11] In general, there are two approaches to doing this with surgical management: A conservative method and a radical approach.[11],[12] Enucleation and curettage are the conservative treatments of ameloblastoma, whereas resection or excision of a tumor with a quantifiable periphery of involving bone is employed in the radical treatment.[12] Resection of the mandible has been the standard treatment for ameloblastoma since the risk of recurrence is exceedingly significant if the tumor is treated ineffectively.[13] Radical surgery can result in a variety of negative effects, including facial deformities, tooth loss, and paresthesia. In multi-cystic and invasive patterns, conservative treatment such as enucleation (removal of disease together with the lining epithelium and subsequent repair of the cavity) may not be adequate for ameloblastoma eradication. To avoid such issues, conservative surgical treatment such as dredging method might be implemented.[14]

The objective of this retrospective analysis was to assess the incidence, pattern, and treatment options of ameloblastoma patients diagnosed between January 2016 and June 2018 at a tertiary-level hospital in Bangladesh.


  Materials and Methods Top


Study design

This was a retrospective, observational study conducted in the department of oral and maxillofacial surgery of Dhaka Dental College and Hospital, Bangladesh, between January 2016 and June 2018. No ethical committee approval was obtained as this was a retrospective study and the data were taken from departmental medical records. The authors have agreed to comply with all of the methodology's requirements.

Inclusion and exclusion criteria

From January 2016 to June 2018, hospital records were searched for all cases of ameloblastoma. These cases were diagnosed with ameloblastoma after histological confirmation. In the present study, we excluded patients who had less than a 6-month follow-up period in their medical records.

Data collection procedure

The hospital records were searched to extract the data regarding the patient's age, gender, side, location, radiographic pattern, histopathological diagnosis, and surgical treatment procedure. The anterior (incisor–canine region), body (premolar–molar region), BAR (body–angle–ramus area, which may comprise mandible body, angle, coronoid process, ramus, and condyle), and ramus regions were classified as the location of ameloblastoma in the mandible. The location was classified into anterior (incisor–canine region) and posterior (distal to the canine) regions for maxillary ameloblastoma. The side of the tumor location was subdivided into right, left, and bilateral (crossing the midline). Any recurrence that developed as a result of these cases was not treated as different or new cases. All primary tumor specimens were collected and evaluated with hematoxylin and eosin staining in order to classify them as per the guidelines of the most recent WHO classification of odontogenic tumors.[15],[16] The histopathological patterns were classified into seven types: follicular, plexiform, mixed (either follicular with plexiform or follicular with granular), acanthomatous, granular, desmoplastic, and basal.

Statistical analysis

The frequency and percentages of these study variables were calculated using descriptive statistics. The mean, standard deviation (SD), and range for continuous data were also used in a descriptive analysis of patient data. At 10-year intervals, age was classified into distinct groups. SPSS 23.0 was utilized to conduct the statistical analysis (SPSS, Inc., Chicago, IL, USA).


  Results Top


Patient demographic findings

A total of 131 patients were recorded as histologically diagnosed cases of ameloblastoma. The retrospective analyses of these patients are detailed in [Table 1]. There were 65 male patients, an almost equal ratio to female patients (M:F = 1:1.01). The findings of the study revealed a wide age range (4–70 years), with a mean age (±SD) of 26.61 years (±13.34). The predominant age group was 21–30-year-old (n = 46, 35.11%), followed by 11–20-year-old (n = 44, 33.59%).
Table 1: Analysis of the data from 131 ameloblastoma cases

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Radiologic findings

The most common radiographic presentation was multilocular radiolucency (n = 73, 55.73%) [Figure 2], followed by unilocular radiolucency (n = 57, 43.51%) [Figure 3]. A mixed radio-opaque–radiolucent lesion was seen in only one case (0.76%).
Figure 2: Orthopantomogram shows multilocular radiolucency in the right side of mandible body

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Figure 3: Orthopantomogram shows unilocular radiolucency in the left body–angle–ramus area of the mandible

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Tumor location

All of the tumors were intra-osseous, with a significant predilection for the mandible (n = 125, 95.42%), and six cases involved the maxilla (4.58%) [Figure 4]. The body–angle–ramus (n = 73, 58.5%) was the most commonly affected area in the mandible, followed by the body (n = 34, 27.2%) and the anterior mandible (n = 15, 12%). Ameloblastoma that extended to both sides of the jaws (bilateral-crossing the mid-line) accounted for 11.45% (n = 15), the right side 38.17% (n = 50), and the left side 50.38% (n = 66) cases. In the maxilla, all cases were involved in the posterior region [Figure 5].
Figure 4: The distribution of the ameloblastoma in the jaw

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Figure 5: (a) Intra-oral swelling in the right posterior maxilla. (b) Computed tomography scan axial view shows ameloblastoma in the right maxilla

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Histological findings

On histological evaluation, the follicular pattern was seen in 71 (54.2%) of the 131 cases, followed by plexiform (n = 32, 24.43%), and a mixed (n = 27, 20.61%) histological variant. The mixed histological type was comprised mostly follicular with a plexiform pattern. A follicular with a granular histological pattern was found in three cases. The desmoplastic histological type was present in one case. In this study, we did not record any peripheral ameloblastoma.

Treatment options and recurrences

Most of the patients (n = 95, 72.52%) were treated with conservative surgery (either dredging or enucleation and curettage) and 36 patients (27.48%) underwent resection (en-bloc resection, segmental resection/partial mandibulectomy/hemimandibulectomy/subtotal mandibulectomy, and maxillectomy). Dredging was the most preferred surgical treatment option in our institute, comprising 68.70% (n = 90) of the total cases. Resection was the second modality for the treatment of ameloblastoma in our hospital. The resection was done with (n = 13, 9.92%) or without reconstruction (n = 15, 11.45%) of the surgical defect. The recurrence rate of the dredging treatment method was 5.56% (n = 05) [Table 1].


  Discussion Top


Ameloblastoma is a rare benign odontogenic tumor with a slow growth rate. A systematic review showed that the incidence of ameloblastoma per hospital in Asian or African patients was much greater than in European or American hospitals.[17] Our results showed a mean age of 26.61 years old, with a wide age range (4–70-year-old) and an equal sex distribution. The considerable frequency of ameloblastoma in young individuals is indicated by the highest incidence in the third decade (35.11%). The mandible (95%) showed the most prevalent site in the current study. Reichart et al.[18] reviewed the published articles on 3677 patients with ameloblastoma and compiled the most comprehensive data on the demographics and clinical manifestations of these patients. Ameloblastoma usually presents as a swelling over the affected site (mandible in 80% of cases) at a mean age of 36 years old with an equal gender ratio.[18] The mean age of the Chinese patients was 31.4 years, with a 1.5:1 male-to-female ratio, and 90.8% of the tumors were in the mandible.[19] The Japanese patients had a mean age of 34.7 years, a male-to-female ratio of 1.6:1, and 92.6% of the ameloblastoma was located in the mandible.[20] A retrospective study in Malaysian patients demonstrated a mean age of 30.3 years, a male-to-female ratio of 1.4:1, and a wide age range (7–85 years), and 91.5% of the tumors were located in the mandible.[21] The left side of the jaw was the most affected side in this study. A study conducted to assess the pattern of ameloblastoma in children and adolescents reported a slight predilection for the left side of the jaw.[22] Intapa[10] reported the right quadrant of the jaw as the most involved side. The mandible body–angle–ramus area (posterior/molar-ramus area) was the most affected site in the current analysis. Most of the previous published studies reported similar findings regarding ameloblastoma location.[8],[10],[21],[22],[23],[24],[25]

Histological and radiologic findings are employed to diagnose ameloblastoma. Ameloblastoma is most commonly found in the mandible body–angle–ramus area and is easily recognized by orthopantomogram (OPG) and computed tomography (CT). The most prevalent radiographic appearance in this study was a multilocular radiolucency (55.73%) in OPG, which was consistent with several studies.[8],[10],[21],[23],[24],[26],[27],[28],[29] A mixed radiolucent-radio-opaque lesion was found in one case, which was a case of histologically confirmed desmoplastic ameloblastoma in the anterior mandible. In comparison to the findings of the current study, Bansal et al.[22] reported 59% unilocular radiolucencies. The children appear to have a significant prevalence of unilocular radiolucencies and a lower prevalence of multilocular radiolucencies, which corresponds to a higher prevalence of unicystic ameloblastoma and a lower proportion of multicystic ameloblastoma.[29]

In the 2005 WHO classification for head-and-neck tumors, four distinct variants of ameloblastoma are recognized: (1) peripheral, in which tumor is extra-osseous and demonstrates continuity with the oral mucosa, (2) unicystic ameloblastoma is a single cystic intra-osseous growth observed grossly and radio-graphically, (3) solid/multi-cystic tumor is an invasive tumor that permeates bone marrow spaces and may have multi-cystic foci, and (4) desmoplastic tumor is an infiltrative intra-osseous pattern dominated by the stromal component, resembling a fibro-osseous lesion radio-graphically.[30] All patients were classified following the revised WHO classification from 2017. In 2017, the WHO classified ameloblastoma into three categories: unicystic ameloblastoma, extra-osseous/peripheral forms, and metastasizing ameloblastoma.[15],[16] Since it has no biological importance and can be confused with unicystic ameloblastoma, the term “solid/multi-cystic” for typical ameloblastoma has been eliminated. Desmoplastic ameloblastoma is reclassified as a histological variant rather than a clinicopathologic variant.[16] Central ameloblastoma is a group of intra-osseous ameloblastomas.[30] The mandible is accountable for about 80% of central ameloblastoma, although the maxilla is rarely involved.[18] Follicular, plexiform, acanthomatous, desmoplastic, granular cell, and basal cell patterns are the histological variants of ameloblastoma. All of these histological variants can be seen as single lesions, combinations of two or more histological variants, or hybrid lesions with other odontogenic tumors.[10] Metastasis of histological benign appearing ameloblastoma is known as “metastasizing ameloblastoma.”[31] Ameloblastoma that has been persistent for a prolonged time and has recurred has been observed to rarely progress to malignant ameloblastic carcinoma.[32] The follicular type (54.2%) was the most prevalent histological pattern of ameloblastoma found in the current study, followed by the plexiform pattern (24.43%). The mixed histological (20.61%) variant was the third most common type. We found a case of desmoplastic ameloblastoma. We did not find any cases of peripheral or metastasizing ameloblastoma. The histological findings are consistent with previous studies.[8],[28],[29],[33] The findings of the present study contrast with previous published literature, which found that the majority of patients exhibited a plexiform histological pattern followed by a follicular histological pattern.[10],[21],[23]

Oral function and facial appearance are inextricably linked to the anatomy of the face and oral cavity. As a result, if an oral cavity disease causes facial deformity, the management is insufficient. Maxillofacial deformity results in functional inconveniency, esthetic displeasure, and emotional anguish. As a result, the goal should be to cure the pathology while also restoring the jaw's normal structure and activity. In 1973, the “dredging method” was introduced as an alternate conservative approach to alleviate these difficulties and entirely remove the tumor.[34],[35] The “dredging method” is an option of conservative surgical procedure that involves periodic dredging following deflation and enucleation or just enucleation to speed up new bone synthesis by removing scar tissue from the pathological bony cavity. In large cystic lesions, deflation is performed by removing a piece of the cystic wall, overlying bone, and mucoperiosteum to relieve intra-cystic pressure and promote the establishment of a distinct bony outline.[13],[36] Dredging is done every 2–3 months to speed up the development of new bone and eradicate ameloblastoma cell nests. All specimens must undergo histological examinations to confirm the elimination of any remaining tumor cells and the avoidance of recurrence. When tumor cells are not revealed in microscopic examination of scar tissues following two consecutive dredging, then the follow-up is started. Follow-up on the patients on a frequent basis is an important aspect of the dredging treatment protocol.[36] Dredging was the most commonly used (68.70%) surgical treatment option in our institute for the management of ameloblastoma [Figure 6]. Resection was the second most common (21.37%) treatment option. The recurrence rate of the dredging method and resection was 5.56% and 13.33%, respectively. In a retrospective study, Ruslin et al.[29] reported that 62.5% of patients were treated conservatively (enucleation and/or curettage) and 37.5% were treated radically (resection or maxillectomy). Enucleation with curettage and resection had a recurrence rate of 17.14% and 5%, respectively.[29] A retrospective analysis of the Malaysian population reported enucleation (45.7%) as the treatment of choice for ameloblastoma patients.[21] In a retrospective analysis of 31 cases of ameloblastoma, Giraddi et al.[25] concluded that an ample resection with a safe margin could be a treatment preference. A retrospective study reported a 12.5% recurrence rate for the dredging method in treating patients with ameloblastoma.[36] In a prospective study, the dredging technique as a modified surgical method for treatment of unicystic ameloblastoma in young patients showed no radiographic evidence of recurrence.[37] In the present study, we included patients who had at least a 6-month follow-up period in their medical records. Our institutional protocol for follow-up of treated patients with ameloblastoma by the “dredging method” included an OPG every 6 months for the first 5 years and every 12 months for the subsequent 5 years to diagnose any recurrences. Then, follow-up will be conducted every 2–3 years for the next 10 years. Much of the anatomy can be conserved following surgery if the tumors are noticed early, before the lower border is destroyed, the buccal and lingual cortices are perforated, and the condyle or coronoid process of the mandible is involved, reducing the ensuing morbidity. We also need to consider the patient's age, sex, location, and extent of the tumor while determining the treatment modality and try to avoid unnecessary radical resection in all cases, which will involve less morbidity and higher patient compliance. Systematic employment of the “dredging method” for the restoration of normal structure and activity of the jaw, and the avoidance of recurrence of the lesion, could yield considerable outcomes.
Figure 6: (a) Preoperative orthopantomogram shows uni-cystic ameloblastoma in the left body–angle–ramus area of the mandible. (b) Postoperative orthopantomogram after deflation to relieve intra-cystic pressure (c) Two months after enucleation, orthopantomogram demonstrates bone formation in the left body–angle–ramus area of the mandible (d) Following the first dredging, orthopantomogram reveals normal bone contour with cortical outline and no signs of recurrence (4 months after enucleation) (e) Following the second dredging, orthopantomogram reveals normal bone contour with cortical outline and no signs of recurrence (6 months after enucleation)

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The prolonged surgical phase and lengthy follow-up period were two drawbacks of this dredging method as an alternative technique for the surgical management of unicystic ameloblastoma. To make sure that any remaining pathology was removed and that recurrence was prevented, repeated dredging of scar tissue that had filled up the bone cavity and histological investigation of all specimens were required.


  Conclusions Top


The ameloblastoma pattern was characterized by a mean age of 26.1 years, a lack of gender predilection, and a predilection for the mandible. The body–angle–ramus area was the most affected site in the mandible. The left side of the jaw was predominantly affected. Multilocular radiolucency was more common than unilocular radiolucency on radiographs. The follicular pattern was the most common histological type. Dredging was the most preferred surgical treatment option with a lower recurrence rate.

Acknowledgments

The authors acknowledge the previous and present contributions of surgeons in the hospitals where these patients were treated.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
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