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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 16  |  Issue : 2  |  Page : 160-164

Management of paediatric ameloblastoma


200 Military Dental Centre, Pathankot, Punjab, India

Date of Submission02-Aug-2021
Date of Decision27-Feb-2022
Date of Acceptance02-Jun-2022
Date of Web Publication21-Dec-2022

Correspondence Address:
Ajay Premanand Desai
200 Military Dental Centre, Pathankot 145 001, Punjab
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jodd.jodd_34_21

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  Abstract 


Ameloblastoma is a benign odontogenic tumour of epithelial origin that is slowly growing, locally aggressive, has a high recurrence rate but is uncommon in children. It has a peak incidence in the third and fourth decades of life. Treatment of ameloblastoma in children is complicated by three factors: (1) physiologic factors such as the continued facial growth, different bone physiology and the presence of unerupted teeth; (2) difficulty in the initial diagnosis and (3) predominance of the unicystic type of ameloblastoma. To present the outcome and advantage of a conservative treatment approach in the management of unicystic ameloblastoma in a paediatric patient. An 11-year-old individual reported with osteolytic lesion right ramus of the mandible involving the body with impacted teeth mimicking dentigerous cyst. Incisional biopsy was suggestive of ameloblastoma without any known subtype. Considering the age and the subsequent reconstruction options, he was operated for peripheral ostectomy with chemical cauterisation. A satisfactory post-operative outcome was achieved. The case was followed up for 2 years with no sign of recurrence. Ameloblastomas in children differ from those in adults, with a higher percentage of unicystic tumours. Enucleation with peripheral ostectomy seems a viable option in paediatric patients considering the amount of post-operative morbidity associated with aggressive management which can always be taken up a later stage if warranted.

Keywords: Ameloblastoma, paediatric, unicystic


How to cite this article:
Desai AP, Sehgal S. Management of paediatric ameloblastoma. J Dent Def Sect. 2022;16:160-4

How to cite this URL:
Desai AP, Sehgal S. Management of paediatric ameloblastoma. J Dent Def Sect. [serial online] 2022 [cited 2023 Jan 31];16:160-4. Available from: http://www.journaldds.org/text.asp?2022/16/2/160/364519




  Introduction Top


Ameloblastomas account for 1% of all oral tumours and cysts of the jaw and approximately 10%–12% of odontogenic tumours overall.[1],[2],[3] An ameloblastoma is a locally invasive odontogenic neoplasm with a rare ability to metastasise if not excised entirely. Ameloblastomas typically occur within the third or fourth decade of life and are considered a rarity in the paediatric patient population, accounting for approximately 10%–15% of all reported cases.[3],[4],[5] A rare variant of the ameloblastoma, which usually occurs in younger patients, is the unicystic ameloblastoma (UCA). Treatment modalities for ameloblastomas include segmental/marginal resection or a conservative approach, i.e. marsupialisation or enucleation with peripheral ostectomy with or without adjunctive therapy.

Treatment of ameloblastoma in adults generally requires resection to counter the aggressive nature of this tumour, particularly for the solid and multicystic types. In children and adolescents, however, treatment of ameloblastomas is complicated by three factors: (1) continuing facial growth and different bone physiology (more cancellous bone, increased bone turnover and reactive periosteum) as well as the presence of unerupted teeth; (2) difficulty in the initial diagnosis and (3) predominance of the UCA. All these factors pose some special problems owing to concerns about the effects of resection and reconstruction on maxillofacial development.[1],[6],[7] Although there is consensus that ameloblastoma has to be treated aggressively to avoid recurrence, the applicability of an initial radical, extensive surgical procedure in children poses a dilemma. We report on the treatment outcome of a paediatric patient who presented with a UCA in the body-ramus region of the mandible.


  Materials and Methods Top


An 11-year-old boy was referred from a peripheral centre with swelling over the right side of his face. The parents noticed that swelling was slowly growing, and the boy complained of mild pain occasionally. Clinically, there was an unremarkable extraoral swelling with mild tenderness over the right body region of the mandible.

On intraoral examination, there was unrestricted mouth opening with a swelling in the posterior mandible extending from the distal of the first molar up to the retromolar area with an unerupted second molar. Mild lingual and buccal expansion was seen. Tenderness was observed during palpation of the soft tissue around this tooth; however, no other signs of inflammation were noted [Figure 1]. Panoramic radiographic investigation showed a well-circumscribed, unilocular radiolucent lesion extending from the distal root of the first molar to the ramus region. A partially formed lower right second molar was displaced to the inferior border and a partially formed third molar was displaced towards the posterior ramus area of the mandible [Figure 2].
Figure 1: Preoperative photograph showing minimal expansion

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Figure 2: Preoperative OPG showing the lesion. OPG: Orthopantomography

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The differential diagnosis included a dentigerous cyst, odontogenic keratocyst and UCA. An incisional biopsy was performed, and histopathologic examination of the tissue was suggestive of ameloblastoma [Figure 3].
Figure 3: Histopathological picture

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Considering the age and growth potential of the patient, a decision of conservative treatment was made. Surgical treatment consisted of enucleation of the entire lesion, together with removal of the resorbed first molar, displaced lower right second and third molar through an intraoral surgical window. A peripheral ostectomy was then performed. The wound was closed primarily [Figure 4] and [Figure 5]. Before discharge, a course of antibiotics, analgesics and mouthwashes for a week was prescribed. The operative site was reviewed after 1 week, oral hygiene habits were reinforced.
Figure 4: Intraoperative photographs

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Figure 5: Preoperative OPG. OPG: Orthopantomography

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Panoramic radiographs were taken immediately postoperatively and at 6-month interval to observe bony infill into the wound. The patient last underwent review in July 2019, and a panoramic investigation confirmed complete bone remodelling and no sign of recurrence of the lesion [Figure 6] and [Figure 7].
Figure 6: Postoperative OPG at 6 months showing complete consolidation of bone. OPG: Orthopantomography

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Figure 7: Postoperative clinical photograph

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  Discussion Top


Ameloblastoma is rare in the young population, with only 8.5%–15% of all ameloblastomas in Western countries.[1],[8],[9] The Asian and the African reports show a higher percentage, ranging from 14.6% to 25%.[10],[11] In study by Andrade et al.[12] at an Indian institution accounted for 17.5% of ameloblastomas in the paediatric group. Ameloblastomas are broadly classified into four types: conventional (solid/multicystic), unicystic, extraosseous or peripheral and metastasising.[13] Robinson and Martinez[14] first described the UCA radiographically as being a cystic unilocular lesion that responds better to conservative management. Ackermann et al.[15] further subclassified the UCA as follows: Type I (simple or luminal), in which the tumour is confined to the lining epithelium of the cyst, type II (intraluminal), in which the nodular proliferation of the neoplastic epithelium projects into the lumen and type III (mural or intramural), in which epithelium in either a follicular or plexiform pattern invades the connective tissue wall. This classification is thought to have a direct bearing on the biological behaviour, treatment and prognosis of UCAs.[15] Biopsy remains the gold standard for histopathologic diagnosis. Incisional biopsy of a large cystic lesion associated with an impacted tooth in the ramus-angle region of the mandible (especially when associated with an impacted tooth such as in our patient) is mandatory to exclude lesions such as dentigerous cysts, odontogenic keratocysts or even conventional ameloblastomas and for typing of UCAs. In our case, the incisional biopsy could not ascertain the subtype of ameloblastoma.

The systematic review by Lau and Samman[16] showed that when it comes to treating UCAs, resection resulted in the lowest recurrence rate (3.6%). Despite the high success rate of resection for UCAs, more conservative treatment to minimise morbidity and to optimise quality of life is generally favoured. Although a UCA is inherently less aggressive than its solid counterpart and should therefore respond to less aggressive treatment modalities, simple enucleation may not be appropriate. The use of enucleation alone was shown to result in the highest recurrence rate (30.5%). Pogrel and Montes[17] also concluded that simple enucleation alone (with an unexpectedly high recurrence rate of up to 60%) has no role in the management of the intraosseous ameloblastoma. Enucleation followed by application of Carnoy solution resulted in a recurrence rate of 16%.[12] The recurrence rate for UCAs treated by marsupialisation with additional surgical procedures (enucleation or resection) was 18%.[18] The mean period to recurrence has been reported to be around 5 years, suggesting that all patients should be followed long-term.[2],[7]

Type III UCAs are considered aggressive as the conventional ameloblastoma, with a high potential to recur, so the commonly adopted treatment approach has been to resect the lesion followed by reconstruction with vascularised or non-vascularised tissues. Although conservative treatment options including enucleation with curettage (with or without the adjunctive treatment of cryotherapy and/or chemical or thermal cauterisation), chemotherapy and radiation therapy, have been recommended in the literature, however, they are seldom adopted.[16],[18],[19]

The occurrence of ameloblastoma in children alters craniofacial growth and development and often results in psychological and social disorders.[20] Treatment planning for ameloblastoma in a child must take into consideration the age of the patient, type and size of the lesion and reconstruction of the defect thereafter. Treatment of ameloblastomas in paediatric patients is not only aimed at restoring function and aesthetics but also must consider the potential impact of the treatment and reconstruction on craniofacial growth. In addition, consideration must be given to whether the dissection is going to be subperiosteal or supraperiosteal, together with the post-ablative grafting options in cases requiring resections. With subperiosteal dissection, the periosteum is left intact with only the pathologic lesion removed, thus increasing the body's ability to form new bone, especially in children.[20],[21] The grafting options include autogenous bone grafts (considered the gold standard for reconstruction but limited by the amount of bone obtainable from the donor sites), allografts and xenografts, as well as the addition of synthetic substitutes, with or without bone-enhancing or bone-forming proteins such as bone morphogenetic proteins.[2],[21],[22],[23]

In the present case, the lesion itself was very large and would ordinarily have entailed a mandibular segmental resection from the lower right first molar to the ramus region. This approach would have required a second surgery for reconstruction of the segmental defect. In an attempt to avoid the need for these extensive reconstructions, our patient was treated conservatively with enucleation and peripheral ostectomy of the lesion. Our patient was followed every 3–6 months for 2 years and had complete decompression and bone regeneration in the defect, with no sign of recurrence. The intact periosteal layer and the high osteogenic potential in our patient appear to have contributed to the complete filling of the cavity with bone within 2 years. The complete regeneration of bone in the large cavity precluded the need for resection of the mandible and subsequent bone graft. This modified enucleation approach could perhaps be considered a treatment option in the management of large UCAs in paediatric patients, particularly in cases without cortical or periosteal breakthroughs. Aggressive treatment should be reserved for recurrent cases. Published reports now recommend a relatively conservative treatment like enucleation with peripheral ostectomy in the first instance, reserving a more aggressive therapy for any recurrence considering the growth potential and capacity for bone regeneration in children.[6],[19]

Long-term follow-up should be ensured, which must include both clinical examination and frequent post-surgical radiographic examination, which would significantly assist in the early detection of late recurrence. A yearly follow-up (using radiography and biopsy if required) for at least 10 years is planned for this patient owing to the potential risk of late recurrence of the tumour in adulthood.


  Conclusion Top


Although the dictum for treatment of ameloblastoma is to treat it aggressively. In paediatric patients, considering the age and growth potential, the conservative treatment seems a viable option with good results. Further studies are required to validate these promising results of conservative treatment of UCAs in children.

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

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Payne SJ, Albert TW, Lighthall JG. Management of ameloblastoma in the pediatric population. Oper Tech Otolaryngol Head Neck Surg 2015;26:168-74.  Back to cited text no. 3
    
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Bansal S, Desai RS, Shirsat P, Prasad P, Karjodkar F, Andrade N. The occurrence and pattern of ameloblastoma in children and adolescents: An Indian institutional study of 41 years and review of the literature. Int J Oral Maxillofac Surg 2015;44:725-31.  Back to cited text no. 5
    
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Seintou A, Martinelli-Kläy CP, Lombardi T. Unicystic ameloblastoma in children: Systematic review of clinicopathological features and treatment outcomes. Int J Oral Maxillofac Surg 2014;43:405-12.  Back to cited text no. 7
    
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Keszler A, Dominguez FV. Ameloblastoma in childhood. J Oral Maxillofac Surg 1986;44:609-13.  Back to cited text no. 8
    
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WrightJM, Soluk Tekkesin M. The WHO classification of odontogenic lesions: A summary of the changes of the 2017 (4th) edition. J Istanb Univ Fac Dent 2017;34:10-30.  Back to cited text no. 13
    
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Robinson L, Martinez MG. Unicystic ameloblastoma: A prognostically distinct entity. Cancer 1977;40:2278-85.  Back to cited text no. 14
    
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Ackermann GL, Altini M, Shear M. The unicystic ameloblastoma: A clinicopathological study of 57 cases. J Oral Pathol 1988;17:541-6.  Back to cited text no. 15
    
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Lau SL, Samman N. Recurrence related to treatment modalities of unicystic ameloblastoma: A systematic review. Int J Oral Maxillofac Surg 2006;35:681-90.  Back to cited text no. 16
    
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Pogrel MA, Montes DM. Is there a role for enucleation in the management of ameloblastoma? Int J Oral Maxillofac Surg 2009;38:807-12.  Back to cited text no. 17
    
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Haq J, Siddiqui S, McGurk M. Argument for the conservative management of mandibular ameloblastomas. Br J Oral Maxillofac Surg 2016;54:1001-5.  Back to cited text no. 18
    
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Kalaskar R, Unawane AS, Kalaskar AR, Pandilwar P. Conservative management of unicystic ameloblastoma in a young child: Report of two cases. Contemp Clin Dent 2011;2:359-63.  Back to cited text no. 19
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Johnson J, Jundt J, Hanna I, Shum JW, Badger G, Melville JC. Resection of an ameloblastoma in a pediatric patient and immediate reconstruction using a combination of tissue engineering and costochondral rib graft: A case report. J Am Dent Assoc 2017;148:40-3.  Back to cited text no. 20
    
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