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Journal of Indian Society of Pedodontics and Preventive Dentistry Official publication of Indian Society of Pedodontics and Preventive Dentistry
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CASE REPORT
Year : 2014  |  Volume : 32  |  Issue : 3  |  Page : 251-254
 

Conservative management of unicystic ameloblastoma in a young child: A case report


1 Department of Paediatric and Preventive Dentistry, Bharati Vidyapeeth Dental College and Hospital, Navi Mumbai, Maharashtra, India
2 Department of Oral and Maxillofacial Surgery, Bharati Vidyapeeth Dental College and Hospital, Navi Mumbai, Maharashtra, India
3 Department of Paediatric and Preventive Dentistry, Modern Dental College and Research Centre, Indore, Madhya Pradesh, India

Date of Web Publication2-Jul-2014

Correspondence Address:
Sonal R Naidu
Department of Pedodontics and Preventive Dentistry, Bharati Vidyapeeth University Dental College and Hospital, Navi Mumbai - 400 614, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.135842

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   Abstract 

Unicystic ameloblastoma is a rare, benign, locally invasive odontogenic neoplasm of young age that shows clinical, radiographical or gross features of an odontogenic cyst, but it histologically shows typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal and/or mural tumor growth. A case of asymptomatic unicystic ameloblastoma in an 11- year-old-female, who was treated by surgical enucleation followed by Carnoy's solution application for 5 minutes, is presented. The present case report describes the clinical and radiological behaviors, its importance and complexity of a differential diagnosis and treatment protocol of lesions in the mandibular molar-ramus area considering the special problems in children.


Keywords: Carnoy′s solution, mandibular molar-ramus area, recurrence, surgical enucleation, unicystic ameloblastoma


How to cite this article:
Naidu SR, Hegde RJ, Devrukhkar VN, Patel AR. Conservative management of unicystic ameloblastoma in a young child: A case report. J Indian Soc Pedod Prev Dent 2014;32:251-4

How to cite this URL:
Naidu SR, Hegde RJ, Devrukhkar VN, Patel AR. Conservative management of unicystic ameloblastoma in a young child: A case report. J Indian Soc Pedod Prev Dent [serial online] 2014 [cited 2019 Aug 23];32:251-4. Available from: http://www.jisppd.com/text.asp?2014/32/3/251/135842



   Introduction Top


Ameloblastoma also known as solid or multicystic ameloblastoma is a relatively rare odontogenic epithelial neoplasm that has traditionally been characterized as persistent and aggressive in nature. These are benign lesions but locally invasive and usually originate in the mandibular molar-ramus area. Ameloblastoma has been classified into several histological subtypes including follicular, cystic, acanthomatous, plexiform, basal cell and granular cell. They are also classified as multicystic versus unicystic in which former is more aggressive and recurs more frequently than does latter. According to data in the literature, the disease occurs in all age groups but the peak incidence is in the third and fourth decades. [1] The tumor in young people is considered a rarity and it accounts for approximately 10-15% of all reported cases of ameloblastoma. [2]

Very little is known regarding the etiology of ameloblastoma. [3] Ameloblastoma is generally asymptomatic and presents as a slowly enlarging facial swelling. [4] However, the presence of the tumor may cause symptoms such as pain, malocclusion, the loosening of teeth or ulceration. [1] The difficulty in the diagnosis is not surprising, as the epithelium of a dentigerous cyst and that of ameloblastoma are derived from the same embryonic source; thus, biopsy remains the only method of confirming the diagnosis. [3]

The treatment of ameloblastoma is controversial and poses special problems in children. [1] Unicystic ameloblastoma is treated conservatively with decompression, enucleation and peripheral ostectomy as well as periodic long-term follow-up. A more aggressive surgical approach may be considered when the condition recurs more than twice or according to the patient's wishes. [1]

This case report describes the conservative treatment of a unicystic ameloblastoma in a young patient.


   Case Report Top


An 11-year-old female patient reported to the Department of Pedodontics and Preventive Dentistry, Bharati Vidyapeeth Dental College and Hospital, Navi Mumbai, with a chief complaint of swelling and intra-oral pus discharge from lower left back region. Patient was apparently alright two months back, when the swelling appeared asymptomatically and gradually increased to the present size. Patient was prescribed some medication from local dentist for swelling but was not resolved. On extra-oral examination, a single diffuse swelling was noticed on left side of face with no sinus or active discharge of pus. It was non-pulsatile, non-tender and firm in consistency on palpation [Figure 1] and [Figure 2]. On intra-oral examination, expansion of both cortical plates was seen associated with 36 and 37 regions [Figure 3].
Figure 1: Extra-oral swelling seen on left side of face

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Figure 2: Extra-oral swelling seen on left side of face

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Figure 3: Intra-orally expansion of cortical plates seen in 36 region

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Orthopantamograph revealed a large oval-shaped radiolucency attached along the distal root of 37 extending completely into the rami of the mandible [Figure 4]. Occlusal radiograph showed expansion of both the cortical plates associated with 36 and 37 [Figure 5].
Figure 4: Orthopantamograph showing a large oval-shaped radiolucency attached along the distal root of 37 extending completely into the rami of the mandible

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Figure 5: Occlusal radiograph showing expansion of both the cortical plates associated with 36, 37

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Fine needle aspiration cytology (FNAC) was done, which showed a blood tinged fluid on aspiration and was sent for investigation [Figure 6]. Other routine examinations were within normal limits. A provisional diagnosis of dentigerous cyst, OKC or ameloblastoma was made.

Under general anesthesia, enucleation and peripheral ostectomy was performed [Figure 7] and [Figure 8]. Impacted second permanent mandibular molar was extracted. Carnoy's solution was applied in the bone cavity for 5 minutes to prevent the recurrence. Bismuth iodoform paraffin paste (BIPP) dressing was placed in the cystic cavity, and the soft tissues were closed to allow healing by secondary intention. The surgical site healed without complications. The BIPP dressing was replaced by a smaller one on every subsequent third week. Histological analysis of surgical specimen confirmed it as unicystic ameloblastoma with intraluminal proliferations [Figure 9]. As the diagnosis carries a risk of recurrence, a long-term follow-up period was planned.
Figure 6: FNAC showing a blood tinged fl uid on aspiration

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Figure 7: Intra-oral marking for incision

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Figure 8: Enucleation and peripheral osteotomy done

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Figure 9: Surgical specimen for biopsy

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   Discussion Top


Unicystic ameloblastoma is categorized as a disorder of odontogenesis sharing common clinical and radiographical manifestations with other odontogenic lesions (dentigerous cyst, odontogenic keratocyst) making diagnosis difficult. Unicystic ameloblastoma is radiologically characterized by a unilocular aspect and it is less aggressive than the solid type, but has the potential for recurrence.

For any lesion with impression of cyst, or unicystic ameloblastoma on radiograph incision biopsy should be considered. Ackerman et al., and Isacsson and associates [5] recommend not to perform such incisional biopsy as they are not always representative to have proper diagnosis, as entire tissue must be included.

The treatment of ameloblastoma in children is complicated by three factors:

  1. Continuing facial growth, different bone physiology (greater percentage of cancellous bone, increased bone turnover and reactive periosteum) and presence of unerupted teeth;
  2. Difficulty in initial diagnosis and
  3. Predominance of the unicystic type of ameloblastoma. [6]


The treatment of ameloblastoma ranges from a conservative approach to radical resection. Radical resection of an ameloblastoma in children should be avoided. [7] Available treatment options include enucleation, followed by use of Carnoy's solution, marsupialization followed by enucleation, marginal resection and aggressive resection. [8] It is of general agreement that unicystic ameloblastoma in children should be conservatively treated to avoid potential complications associated with larger resection like facial deformity, masticatory dysfunction, abnormal jaw movements, abnormal psychological development and jaw growth. Secondly, plexiform type of unicystic ameloblastoma, which is more common in children, behaves less aggressively than follicular type supporting conservative treatment. [9]

In the present case report, unicystic ameloblastoma was conservatively treated followed by application of Carnoy's solution to decrease recurrence. Overall recurrences rate for unicystic ameloblastoma is 6.7-35.7% with an average of 7 years. Lau et al., reported that the recurrence rate of 3.6% for resection, 30.5% for enucleation, 16% for enucleation followed by Carnoy's solution application and 18% for marsupialization followed by enucleation. However, recurrence rate should also be evaluated based on the histological subtypes of unicystic ameloblastoma and accordingly follow-up should be planned. [10]


   Conclusio Top


Oral health care providers should be aware of the unilocular radiolucency of the jaws as this lesion could be unicystic ameloblastoma having strong propensity for recurrences. Conservative timely intervention and conservative surgical treatment should be the first choice for treating ameloblastoma in children followed by Carnoy's solution application that may improve treatment outcome and potential complications associated with larger resection. The treatment should be performed as soon as possible after diagnosis, in order to prevent the possible proliferation in adjacent tissues.

 
   References Top

1.Huang IY, Lai ST, Chen CH, Chen CM, Wu CW, Shen YH. Surgical management of ameloblastoma in children. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;104:478-85.  Back to cited text no. 1
    
2.Keszler A, Dominguez FV. Ameloblastoma in children. J Oral Maxillofac Surg 1986;44:609-13.  Back to cited text no. 2
[PUBMED]    
3.Daramola JO, Ajagbe HA, Oluwasanmi JO. Ameloblastoma of the jaws in Nigerian children: A review of sixteen cases. Oral Surg Oral Med Oral Pathol 1975;40:458-63.  Back to cited text no. 3
[PUBMED]    
4.Al-Khateeb T, Ababneb KT. Ameloblastoma in young jordanians: A review of the clinicopathologic features and treatment of 10 cases. J Oral Maxillofac Surg 2003;61:13-8.  Back to cited text no. 4
    
5.Isacsson G, Andersson L, Forsslund H, Bodin I, Thomsson M. Diagnosis and treatment of the unicystic ameloblastoma. Int J Oral Maxillofac Surg 1986;15:759-64.  Back to cited text no. 5
[PUBMED]    
6.Guerrisi M, Piloni MJ, Keszler A. Odontogenic tumors in children and adolescents. A 15-year retrospective study in Argentina. Med Oral Patol Oral Cir Bucal 2007;12:E180-5.   Back to cited text no. 6
    
7.Takahashi K, Miyauchi K, Sato K. Treatment of ameloblastoma in children. Br J Oral Maxillofac Surg 1998;36:453-6.  Back to cited text no. 7
    
8.Ueno S, Mushimoto K, Shirasu R. Prognostic evaluation of ameloblastoma based on histologic and radiographic typing. J Oral Maxillofac Surg 1989;47:11-5.  Back to cited text no. 8
    
9.Sampson DE, Pogrel MA. Management of mandibular ameloblastoma: The clinical basis for a treatment algorithm. J Oral Maxillofac Surg 1999;579:1074-7.  Back to cited text no. 9
    
10.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. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9]



 

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    Abstract
   Introduction
   Case Report
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