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 Table of Contents  
CASE REPORT
Year : 2017  |  Volume : 9  |  Issue : 4  |  Page : 272-275

Conservative accession toward treatment of plexiform ameloblastoma in an 11-year-old girl


1 Department of Pedodontics and Preventive Dentistry, Seema Dental College and Hospital, Rishikesh, Uttarakhand, India
2 Department of Periodontolog, Seema Dental College and Hospital, Rishikesh, Uttarakhand, India

Date of Web Publication1-Dec-2017

Correspondence Address:
Avantika Tuli
Department of Pedodontics and Preventive Dentistry, Seema Dental College and Hospital, Rishikesh - 249 201, Uttarakhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJDS.IJDS_50_17

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  Abstract 

Ameloblastoma is the common form of aggressive benign tumor of the jaws, but it is rare in childhood. The treatment of ameloblastoma is controversial. Surgical treatment of ameloblastoma in children follows the principles of the clinical and pathological aspects of the tumor and poses a special problem due to the incomplete growth of the jaws. With a unicystic ameloblastoma, the procedure of choice is a conservative approach. This paper describes the conservative treatment of a plexiform unicystic ameloblastoma in a child involving curettage of the tumor and the extraction of four teeth under general anesthesia, with a good prognosis of the case.

Keywords: Ameloblastoma, plexiform, unicystic


How to cite this article:
Tuli A, Chandra S, Sehgal M, Tuli AS, Varshney M. Conservative accession toward treatment of plexiform ameloblastoma in an 11-year-old girl. Indian J Dent Sci 2017;9:272-5

How to cite this URL:
Tuli A, Chandra S, Sehgal M, Tuli AS, Varshney M. Conservative accession toward treatment of plexiform ameloblastoma in an 11-year-old girl. Indian J Dent Sci [serial online] 2017 [cited 2019 Sep 17];9:272-5. Available from: http://www.ijds.in/text.asp?2017/9/4/272/219625


  Introduction Top


Ameloblastoma, a benign but locally aggressive tumor of odontogenic epithelium, is the most frequent odontogenic tumor of the mandible and maxilla. Ameloblastomas are typically classified as multicystic/solid, unicystic and peripheral/extra osseous ameloblastomas.[1] Ameloblastoma can occur in all age groups, but the peak incidence is in the third and fourth decades of life and occurrence in childhood is rare. Ameloblastoma is generally asymptomatic and presents as a slowly enlarging facial swelling.[2] Radiographically, it appears as a unilocular radiolucent area with a well-defined margin or with a multilocular aspect, often in the shape of soap bubbles or a honeycomb. Plexiform unicystic ameloblastomas do not exhibit the exact histological criteria for ameloblastoma have therefore been considered by some pathologists to be a hyperplastic epithelial proliferation rather than ameloblastomas. Several therapeutic modalities have been proposed, including curettage, marsupialization, cryosurgery, surgical excision, electrocautery, sclerotherapy, and radiotherapy.[3]

Thus, the challenges in the management of this tumor are to provide complete excision, in addition, to reconstruct the bony defect, to provide the patient with reasonable cosmetic and functional outcome. With this in mind, this paper is aimed at describing the management of a plexiform ameloblastoma of in an 11-year-old child.


  Case Report Top


An 11-year-old female patient was referred to the department of Pedodontics and Preventive Dentistry, Seema Dental College and Hospital, Rishikesh with the complaint of swelling in lower left back region of jaw for 1½ year. The patient presented with a history of swelling on the lower left side of the face which was initially small in size and increased gradually with time. Sometimes, it was associated with pain which was intermittent in nature. No history of ulceration was seen. Medical history was noncontributory. The patient visited the dentist 3–4 days back and antibiotics and analgesics were prescribed for the same.

On extraoral examination, asymmetry of face with diffuse swelling was present on the left side [Figure 1]. Color of the swelling was same as that of skin. The swelling extended from 2 to 3 cm below the ala-tragus line up to the lower border of mandible. Mediolaterally, it extends from corner of mouth to posterior border of mandible. On palpation, there was no rise in temperature and was hard in consistency and nontender in nature.
Figure 1: Pre-operative extraoral photograph showing marked swelling on left side of face

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Intraoral examination revealed that the swelling extended from the distal of 33–36 and was fluctuant in nature [Figure 2].
Figure 2: Pre-operative intraoral photograph

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The preoperative orthopantomogram (OPG) showed a radiolucency extending from 33 to the mid portion of ascending ramus and coronoid process [Figure 3]. Radiolucency was multilocular, well demarcated with corticated margins. Teeth number 37 and 38 are pushing toward ascending ramus. Developing 38 was seen almost near to the base of coronoid process.
Figure 3: Pre-operative orthopantomogram showing a radiolucency extending from 33 to the mid portion of ascending ramus and coronoid process

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Due to the patient's age and the biological behavior of the tumor over the 1 year period, the procedure was performed under general anesthesia [Figure 4]. Before surgical intervention under GA, blood investigations, and chest X-ray were advised to the patient.
Figure 4: Patient being prepared for dental treatment under general anesthesia

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The treatment plan of curettage with the extraction of four adjacent teeth to generate a margin of safety including two erupted and enucleation of two unerupted teeth was decided [Figure 5].
Figure 5: Incision followed by enucleation and complete marsupilization of lesion

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The excised tissue was sent for histopathological analysis [Figure 6]. An OPG was taken after the surgery to asses the complete removal of the tumour [Figure 7]. Histopathology revealed the excised tissue having strands and cords of odontogenic epithelium, forming a complex branching pattern. The cells had uniform elongated nuclei. The intervening tissue was loose, with stellate reticulum like epithelium. The supporting stroma was vascular and showed many dilated congested blood vessels. No increased mitotic activity or cytoplasmic atypia was seen [Figure 8]a and [Figure 8]b.
Figure 6: Lesion excised from operative site

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Figure 7: Post-operative orthopantomogram taken after surgery with iodoform gauze at treatment site

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Figure 8: Slide magnification of excised tissue- under (a: ×10, b: ×40) magnification

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The surgical wound was then treated with Carnoy's solution. The use of Carnoy's solution was to decrease chances of recurrence after conservative surgical treatment of ameloblastoma followed by bismuth iodoform paraffin paste dressing was placed in the cystic cavity, and the soft tissues were closed to allow healing by secondary intention.

The patient has been in follow-up since the day of the surgery. After 3 months, an OPG was taken which showed bone deposition on the affected site [Figure 9] and [Figure 10]. Clinically, the swelling on the left side of the mandible had subsided, and patient is asymptomatic [Figure 11]. Prosthetic rehabilitation has been planned for the patient in subsequent follow-up visits to improve the patient's mastication and esthetics.
Figure 9: Post-operative photograph of the patient after 6 days

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Figure 10: Post-operative orthopantomogram of the patent after 3 months reveals bone deposition

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Figure 11: Post-operative photograph of the patient 3 months after surgery

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  Discussion Top


Ameloblastomas are classified into three variants that must be differentiated because they have different treatments and prognoses: solid or multicystic (86% of cases), unicystic (13% of cases), and peripheral (1% of cases) types. Among all of the intraosseous types, the multicystic ameloblastoma is the most aggressive variant whereas the peripheral ameloblastoma has a less aggressive biologic behavior, with a favorable prognosis.[1] Furthermore, its age of presentation varies with changing locations, but age range usually remains within standard accepted age group of second to the seventh decade.[4],[5] The youngest case of ameloblastoma reported was at the age of 2 years.[6]

Lau and Samman [7] in 2006 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.[7] In addition, Reddy and Rao reported that there was no recurrence in 6-year-old child who was treated by extraction of deciduous teeth followed by enucleation and Carnoy's solution application with the follow-up of 2 years.[8] In another case by Scariot et al. of 9-year-old having unicystic ameloblastoma was treated with curettage and the extraction of two premolar mandibular teeth which resulted in no recurrence as well as excellent postoperative function and aesthetics.[3] Hong et al. in a retrospective analysis of 239 patients with ameloblastomas of the jaws reported recurrences of 4.5% in patients treated by segmental resection or maxillectomy, 11.6% in a patient treated by resection with bone margin, and 29.3% treated with conservative treatment (enucleation, curettage and marsupialization).[9]

In the present case, complete curettage and enucleation of the whole of the lesion was done that was extending toward coronoid process with the removal of permanent teeth and their buds in anticipation that they could be the site of recurrence in future.


  Conclusion Top


Ameloblastoma is a benign, but locally invasive odontogenic tumor with a high rate of recurrence. Most studies showed that the prognosis for ameloblastoma is dependent upon the method of surgical treatment rather the histologic type of tumor. Conservative timely intervention and conservative surgical treatment should be the first choice for treating ameloblastomas in children followed by Carnoy's solution application that may improve treatment outcome and prevents complications associated with larger resection.

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

1.
Pereira CM, Carneiro DS, Silva AF, Costa CC. Surgical treatment of maxillary solid ameloblastoma by weber-fergusson incision and immediate prosthetic rehabilitation. Int J Prosthet Dent 2011;2:19-23.  Back to cited text no. 1
    
2.
Adeyemo WL, Bamgbose BO, Ladeinde AL. Surgical management of ameloblastomas: Conservative or radical approach. A critical review of the literature. Oral Surg 2008;1:22-7.  Back to cited text no. 2
    
3.
Scariot R, Silva RV, Felix WS. Conservative treatment of ameloblastoma in child: A case report. Balti Dent Maxillofac J 2012;14:33-6.  Back to cited text no. 3
    
4.
Kannepady SK, Sakri SB, Nanditha S. Unicystic ameloblastoma in a young female: A case report and review of literature. World J Dent 2011;2:263-7.  Back to cited text no. 4
    
5.
Menezes JD, Imada TS, Bernini GF. Recurrence of multicystic ameloblastoma: Case Report. Landmark Res J Med Sci 2014;1:27-30.  Back to cited text no. 5
    
6.
Chaudhary Z, Krishnan S, Sharma P, Sharma R, Kumar P. A review of literature on ameloblastoma in children and adolescents and a rare case report of ameloblastoma in a 3-year-old child. Craniomaxillofac Trauma Reconstr 2012;5:161-8.  Back to cited text no. 6
    
7.
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. 7
    
8.
Reddy SK, Rao GS. Unicystic ameloblastoma in 6 year old and its significance. J Oral Maxillofac Surg 2011;2:363-6.  Back to cited text no. 8
    
9.
Hong J, Yun PY, Chung IH, Myoung H, Suh JD, Seo BM, et al. Long-term follow up on recurrence of 305 ameloblastoma cases. Int J Oral Maxillofac Surg 2007;36:283-8.  Back to cited text no. 9
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8], [Figure 9], [Figure 10], [Figure 11]



 

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