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Table of Contents   
CASE REPORT  
Year : 2019  |  Volume : 30  |  Issue : 6  |  Page : 960-963
Recurrent ameloblastoma 24 years after hemimandibulectomy: A case report and review of literature


Department of Oral and Maxillofacial Surgery, Tamil Nadu Government Dental College and Hospital, Chennai, Tamil Nadu, India

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Date of Submission21-Jun-2017
Date of Acceptance21-May-2018
Date of Web Publication14-Jan-2020
 

   Abstract 


Ameloblastoma is a benign, locally aggressive epithelial odontogenic tumor with a high recurrence rate. The management of ameloblastoma has always been controversial and an enigma to the surgeons. Literature suggests that 50% of the recurrences occur during first 5 years after the primary surgery, and the recurrence rate following a radical approach such as a segmental resection is 4.6%. The reasons for recurrence after a radical approach can be multifactorial such as remaining stumps, soft tissues, or intraoperative contamination. The purpose of this case report is to emphasize the fact that a recurrence even after 24 years is possible in spite of a radical segmental resection, and hence, a continuous follow-up of the patient is needed and to highlight the fact that the possibility of malignant ameloblastoma or ameloblastic carcinoma should be ruled out when dealing with such ameloblastomas recurring after a long period after a radical primary surgery.

Keywords: Ameloblastoma, radical approach, recurrent

How to cite this article:
Balasubramaniam S, Jayaraman B, Thirunavukkarasu R, Kamalakaran AK. Recurrent ameloblastoma 24 years after hemimandibulectomy: A case report and review of literature. Indian J Dent Res 2019;30:960-3

How to cite this URL:
Balasubramaniam S, Jayaraman B, Thirunavukkarasu R, Kamalakaran AK. Recurrent ameloblastoma 24 years after hemimandibulectomy: A case report and review of literature. Indian J Dent Res [serial online] 2019 [cited 2020 Jul 8];30:960-3. Available from: http://www.ijdr.in/text.asp?2019/30/6/960/275866



   Introduction Top


Ameloblastoma, a benign odontogenic tumor, was first described by Broca and named by Churchill.[1] It occurs more frequently in the angle and ramus area with no definite sex predilection and mostly diagnosed between the second and sixth decades. The cells of origin of ameloblastoma seem to be epithelial cells that express amelogenin.[2] Although benign, it is locally more aggressive. Ameloblastomas can be classified clinically as unicystic, multicystic/solid, desmoplastic, and peripheral or extraosseous.[3] The intraosseous tumors have high recurrence rate. The management of ameloblastoma has always been controversial and an enigma to the surgeons. Various treatment modalities have been advocated for the management of ameloblastoma ranging from simple enucleation to radical procedures such as resection.[4] However, the factors such as size of the lesion, anatomical location, histological variant, and anatomical involvement should be considered in formulating a treatment modality for the management of ameloblastoma [5] rather a fixed treatment protocol.

In this paper, a case of huge ameloblastoma which has recurred after 24 years even after a radical treatment has been discussed depicting the highly recurrent and aggressive nature of this benign tumor.


   Case Report Top


A 60-year-old male patient was referred to the department of oral and maxillofacial surgery with the complaint of swelling in the lower jaw for 7 months. The history of the patient revealed that he had undergone segmental resection of the left mandible without any reconstruction 24 years earlier. The past surgical reports suggested that it was an ameloblastoma. Extraoral examination revealed a swelling of size 11 cm × 8 cm in the left submandibular region with a segment of mandible missing on the left side. The extensions of the swelling were from the left submental to angle region. The consistency was firm, and it was nontender [Figure 1]a. On intraoral examination, a swelling of size 5 cm × 3 cm extending distally to the left mandibular canine till the retromolar area was seen. The swelling was firm in consistency with a normal overlying mucosa [Figure 1]b. Orthopantomography revealed a segment of mandible missing on the left side with a condylar stump remaining along with a part of a K-wire [Figure 2]a. The computed tomography showed a well-defined hypodense lesion in the left mandibular region with a condylar stump remaining [Figure 2]b and [Figure 2]c. An incisional biopsy was performed, and the histopathologic report was suggestive of ameloblastoma [Figure 3]a.
Figure 1: (a) Firm asymptomatic swelling in the left submandibular region. (b) Intraoral presentation of the lesion

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Figure 2: (a) Orthopantomography showing resected left mandible with remaining condyle, coronoid, and a part of K-wire. (b) Computed tomography coronal view showing hypodense lesion in the left submandibular region. (c) Computed tomography showing resected left mandible with remaining condyle, coronoid, and a part of K-wire

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Figure 3: (a) Incisional biopsy histopathology (H and E, ×10) showing follicles of odontogenic islands characterized by peripheral layer of tall columnar cells with and without reversed polarized hyperchromatic nuclei enclosing central cells ranging in appearance from stellate to acanthomatous and granular. (b) Postoperative histopathology (H and E, ×10) consistent with incisional biopsy histopathology

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Surgical excision of the lesion along with the removal of the condylar stump and K-wire was planned under general anesthesia. Risdon's incison was made on the left side, layerwise dissection was done and the lesion was identified and excised in toto after releasing from the soft tissue adhesions [Figure 4]a and [Figure 4]b and the Condylar stump and the K wire were also removed [Figure 5]a and [Figure 5]b. The wound was closed layer wise with absorbable sutures. Postoperative recovery of the patient was uneventful, and the specimen was sent for histopathologic examination. The postoperative histopathologic report [Figure 3]b was consistent with the preoperative report [Figure 3]a confirming the diagnosis of ameloblastoma showing follicles of odontogenic islands characterized by peripheral layer of tall columnar cells with and without reversed polarized hyperchromatic nuclei enclosing central cells ranging in appearance from stellate to acanthomatous and granular [Figure 3]b. The patient is disease free after a 3-year follow-up [Figure 6].
Figure 4: (a) Identification of the lesion. (b) Excised lesion in toto

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Figure 5: (a) Resected condyle and coronoid. (b) Part of K-wire removed

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Figure 6: Postoperative extraoral view

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   Discussion Top


Ameloblastoma, a benign odontogenic tumor, represents 10% of all jaw tumors.[6] It is most frequently encountered in the mandibular posterior region and usually diagnosed between 30 and 60 years. In the current case, taking into account the site and a history of the treated case of ameloblastoma, a provisional diagnosis of ameloblastoma was made. Despite a clinical and radiographic suspicion in favor of ameloblastoma, a biopsy is always mandatory in such extensive lesions to exclude a malignancy.

The recurrence rate after curettage is 50%–90%.[7] Conventional ameloblastomas tend to infiltrate intact cancellous bone at the periphery of the lesion before bone resorption actually becomes evident clinically and this could be one of the reasons for the recurrence after conservative approaches. The multicystic ameloblastoma has more aggressive behavior and a greater tendency to recur than the unicystic, and hence, it was presumed that the primary lesion could have been a multicystic ameloblastoma.

Radical surgeries such as marginal resection and segmental resection with the inclusion of adjacent soft tissues still have 5%–15% chances of recurrence.[8] The recurrent lesion could arise from the remaining stumps, soft tisues or by intraoperative contamination during the primary surgery or a combination of all the above.[9] In the reported case since the condylar stump was remaining, it could be considered as one of the causes for recurrence.

Al-Bayaty et al.[10] and To et al.[8] in their individual case reports on recurrent ameloblastoma reported that tumor seeding during surgery could be cited as the cause for recurrence which again could be a possibility in the current case. Literature suggests that 50% of the recurrences occur during first 5 years after the primary surgery.[11] In contrary to this, Mainenti et al.[12] reported a case of recurrent ameloblastoma 33 years after hemimandibulectomy. Siar et al.[13] in their analysis on a retrospective study on 340 cases of ameloblastoma in Malaysian population reported that the mean period of recurrence was 7.3 years with a range of 1–29 years. Hong et al.[14] in a retrospective study of 239 patients with ameloblastoma reported a recurrence rate of 4.5% after segmental resection, 11.6% after marginal resection, and 29.3% after conservative treatment modalities. The present case wherein a recurrence has been reported 24 years after a segmental resection is a clear evidence of the aggressive and recurrent nature of this benign lesion.

Another important aspect to be reviewed when encountering recurrent ameloblastomas is to exclude the possibility of a metastatic malignant ameloblastoma and ameloblastic carcinoma. The lung is the most common site of metastasis (80%).[15] According to Van Dam et al.,[16] the average time from the diagnosis of primary to metastasis is 18 years. In the present case, the patient reported with a recurrence after 24 years; hence, this was also taken into consideration, and the patient was subjected to preoperative and the postoperative chest radiographs after 2-year follow-up, and a thoracic opinion was obtained, and no signs metastasis was detected ruling out a malignant ameloblastoma, and histologically, there were no signs of malignancy in favor of an ameloblastic carcinoma.


   Conclusion Top


The possibility of an iatrogenic tumor cell implantation during the surgical procedures should be emphasized. An intraoperative seeding of the tumor cells is strongly suspected as the reason for recurrence in the reported case in spite of a radical treatment of the primary lesion. The most important but difficult aspect in the management of ameloblastoma is a close follow-up of the patient because a recurrence even after 24 years is possible despite a radical primary treatment and hence a continuous follow-up, radical approach along with avoidance of intraoperative tumor seeding is required to avoid recurrence in this locally aggressive highly recurrent benign tumor.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initial will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Reichart PA, Philipsen HP, Sonner S. Ameloblastoma: Biological profile of 3677 cases. Eur J Cancer B Oral Oncol 1995;31B: 86-99.  Back to cited text no. 1
    
2.
Kumamoto H, Yoshida M, Ooya K. Immunohistochemical detection of amelogenin and cytokeratin 19 in epithelial odontogenic tumors. Oral Dis 2001;7:171-6.  Back to cited text no. 2
    
3.
Barnes L, Eveson JW, Reichart PA, Sidransky D, editors. World Health Organization Classification of Tumours. Pathology & Genetics. Head and Neck Tumours. World Health Organization. International Agency for Research on Cancer. Lyon: IAC Press; 2005.  Back to cited text no. 3
    
4.
Müller H, Slootweg PJ. The ameloblastoma, the controversial approach to therapy. J Maxillofac Surg 1985;13:79-84.  Back to cited text no. 4
    
5.
Sampson DE, Pogrel MA. Management of mandibular ameloblastoma: The clinical basis for a treatment algorithm. J Oral Maxillofac Surg 1999;57:1074-7.  Back to cited text no. 5
    
6.
Becelli R, Carboni A, Cerulli G, Perugini M, Iannetti G. Mandibular ameloblastoma: Analysis of surgical treatment carried out in 60 patients between 1977 and 1998. J Craniofac Surg 2002;13:395-400.  Back to cited text no. 6
    
7.
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. 7
    
8.
To EW, Tsang WM, Pang PC. Recurrent ameloblastoma presenting in the temporal fossa. Am J Otolaryngol 2002;23:105-7.  Back to cited text no. 8
    
9.
Martins WD, Fávaro DM. Recurrence of an ameloblastoma in an autogenous iliac bone graft. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;98:657-9.  Back to cited text no. 9
    
10.
Al-Bayaty HF, Murti PR, Thomson ER, Niamat J. Soft tissue recurrence of a mandibular ameloblastoma causing facial deformity in the temporal region: Case report. J Oral Maxillofac Surg 2002;60:204-7.  Back to cited text no. 10
    
11.
Olaitan AA, Arole G, Adekeye EO. Recurrent ameloblastoma of the jaws. A follow-up study. Int J Oral Maxillofac Surg 1998;27:456-60.  Back to cited text no. 11
    
12.
Mainenti P, Oliveira GS, Moraes HM, Blumer Rosa LE. Recurrent ameloblastoma after 33 years of hemimandibulectomy – Case report. Appl Cancer Res 2008;28:37-40.  Back to cited text no. 12
    
13.
Siar CH, Lau SH, Ng KH. Ameloblastoma of the jaws: A retrospective analysis of 340 cases in a Malaysian population. J Oral Maxillofac Surg 2012;70:608-15.  Back to cited text no. 13
    
14.
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. 14
    
15.
Verneuil A, Sapp P, Huang C, Abemayor E. Malignant ameloblastoma: Classification, diagnostic, and therapeutic challenges. Am J Otolaryngol 2002;23:44-8.  Back to cited text no. 15
    
16.
Van Dam SD, Unni KK, Keller EE. Metastasizing (malignant) ameloblastoma: Review of a unique histopathologic entity and report of Mayo clinic experience. J Oral Maxillofac Surg 2010;68:2962-74.  Back to cited text no. 16
    

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Correspondence Address:
Dr. Saravanan Balasubramaniam
Department of Oral and Maxillofacial Surgery, Tamil Nadu Government Dental College and Hospital, Chennai - 600 003, Tamil Nadu
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijdr.IJDR_345_17

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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