Home | About us | Editorial board | Ahead of print | Current issue | Archives | Search | Submit article | Instructions | Subscribe | Advertise | Contact us |  Login 
National Journal of Maxillofacial Surgery
 
Print this page Email this page Small font sizeDefault font sizeIncrease font size
Users Online: 4261
 


 
Table of Contents
CASE REPORT
Year : 2010  |  Volume : 1  |  Issue : 2  |  Page : 190-193  

Ameloblastoma relapse after 16 years of resection in symphysis of mandible sparing the bone graft


Department of Maxillofacial Surgery, H.P. Government Dental College/Hospital, Shimla, Himachal Pradesh, India

Date of Web Publication9-Apr-2011

Correspondence Address:
Rangila Ram
10/1 Rao Niwas, Jawahar Nagar Mandi, Himachal Pradesh
India
Login to access the Email id

Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0975-5950.79229

Rights and Permissions
   Abstract 

Ameloblastoma is a tumor derived from epithelium involved in odontogenesis. Although it is considered a benign tumor, its clinical behavior may be regarded as lying between benign and malignant. It is characterized by slow but persistent growth, local infiltration into adjacent tissues and recurrences; however, metastases are rare. Diagnosis mainly from tissue biopsy and characteristic finding on plain X-rays does assist in differentiating between types of ameloblastoma. The challenges in the management of this tumor are to provide complete excision as recurrence may occur in incomplete removal and also to reconstruct the bony defect in order to give reasonable cosmetic and functional outcome to the patient.

Keywords: Ameloblastoma relapse, epithelial odontogenic tumor, mandibular reconstruction


How to cite this article:
Ram R. Ameloblastoma relapse after 16 years of resection in symphysis of mandible sparing the bone graft. Natl J Maxillofac Surg 2010;1:190-3

How to cite this URL:
Ram R. Ameloblastoma relapse after 16 years of resection in symphysis of mandible sparing the bone graft. Natl J Maxillofac Surg [serial online] 2010 [cited 2021 Oct 27];1:190-3. Available from: https://www.njms.in/text.asp?2010/1/2/190/79229


   Introduction Top


Ameloblastoma is slow-growing, locally invasive, odontogenic tumor of the jaw with high recurrence rate if not treated adequately but with virtually no tendency to metastasize. [1] Ameloblastoma is the second most common odontogenic tumor. It exhibits no sex predilection and occurs over a wide age range. Most cases are diagnosed between 30 and 60 years, whereas the tumor is rare in age younger than 20 years. Geographic and racial differences have been described. [2],[3] Exact etiology of ameloblastoma is not known. Thus, the tumor conceivably may be derived from

  1. Cell rest of enamel organ, either remnants of dental lamina or remnant of Hertwigs sheath (cell rest of Malassez);
  2. Epithelium of odontogenic cyst (particularly dentigerous cyst); and
  3. Disturbances of developing enamel organ. [4]
The tumor occurs exclusively in the jaw and rarely in the sinonasal cavities. Approximately 80% occur in the mandible, with marked predilection for the posterior region. [5] This paper presents a case of a man who was operated in 1993, when he was 32 years old, for an ameloblastoma in the left body region of mandible. The surgical treatment was segmental resection of mandible; reconstruction of the defect was performed with a non-vascularised autologous bone graft of the iliac crest after 6 months of resection. After 16 years, ameloblastoma relapsed in the symphysis region without involving the graft.


   Case Report Top


A 32-year-old male patient reported to the Department of Oral and Maxillofacial Surgery in October 1993 with a chief complaint of swelling on the left side of lower jaw. The swelling was hard and had a duration of 6 months. It was gradually progressing in size and was associated with pain and paresthesia for the past 1 month. On I/O- Intraoral examination, the swelling extended from left lateral incisor upto second molar. There was more expansion of buccal cortex than lingual cortex. Expansion had caused the obliteration of buccal vestibule. On radiographic examination, well-defined multilocular radiolucency extending from lateral incisor to second molar was seen [Figure 1]. Incisional biopsy was done under local anesthesia. On h/p- histopathological examination, ameloblastoma was confirmed. Segmental resection of lesion with 1 cm of normal bone was done [Figure 2]. After 6 months of resection, reconstruction was done with nonvascularized iliac crest bone graft and stabilized with AO reconstruction plate [Figure 3]. The patient was kept under regular observation for a period of 6 months. In due course of time, mandibular prosthesis was given as replacement for missing teeth. No complications in the postoperative period were reported till 2008 [Figure 4].
Figure 1: Preoperative radiograph of ameloblastoma in November 1993

Click here to view
Figure 2: Immediate postoperative (January 1994) after resection

Click here to view
Figure 3: Postoperative (2 years after the reconstruction with iliac crest in 1996)

Click here to view
Figure 4: Postoperative OPG in 2006 after 12 years of resection

Click here to view


In December 2008, the patient reported to the department with a complaint of painless swelling in the labial vestibule, from the past 2 months, in relation to apical region of right lateral incisor and canine teeth. Aspiration revealed 2-3 ml of straw-colored fluid, after which the swelling subsided, which later recurred in 2 weeks. On radiographic examination [orthopantomogram (OPG)], multilocular radiolucency at the level of right symphysis region was seen with no involvement of the bone graft [Figure 5]. Because the lesion was completely contained within the healthy bone, marginal resection of the bone was done under local anesthesia with 1 cm margin [Figure 6]. On microscopic examination, recurrence of ameloblastoma was confirmed. Graft had consolidated markedly well with normal bone and to our surprise, relapse of the lesion was noticed on normal side and no recurrence was noticed on the bone graft [Figure 7]. The patient was kept under regular follow-up after every 3 months, no recurrence has been reported till date [Figure 8].
Figure 5: Postoperative OPG showing relapse in 2008

Click here to view
Figure 6: After marginal resection of relapse (right arrow) and butt joint between graft and normal bone (left arrow)

Click here to view
Figure 7: Intraoral view after marginal resection in 2010

Click here to view
Figure 8: After intraoral prosthesis

Click here to view



   Discussion Top


Ameloblastoma is a tumor with well-known propensity for recurrence. [6] Several factors that may influence the rate of recurrence have been identified. The first and the most important is clinicopathologic variant of tumor. It is generally accepted that there are three variants of the benign ameloblastoma, designated as solid or multicystic, unicystic and peripheral. [7],[8] The solid variety has the greatest propensity for local infiltration and therefore the highest potential for recurrence. [9] The second factor that should be considered is the anatomic site. [10] Up to 95% of ameloblastomas occur in the mandible. The dense cortical bone of the mandible prevents the tumor from spreading extensively for several years, although spread in the central cancellous bone is beyond the radiographic margins of the tumor. [11] The third factor contributing to recurrence is the adequacy of surgery. To ensure that lesion is completely removed, the anatomic extent of the tumor needs to be carefully assessed. The lesions that are completely intraosseous can be adequately assessed with standard radiography. Radiologically, the lesions are expansile, with thinning of cortex in the buccal-lingual plane. The lesions are classically multilocular cystic with a "soap bubble" or "honeycomb" appearance. Finally, the histological variant of the ameloblastoma has been suggested to be of prognostic significance in terms of recurrence. Treatment of ameloblastoma varies from enucleating and curettage to en bloc resection. [12] The treatment of choice depends on several factors. Multilocular ameloblastomas have higher recurrence rates than unilocular ones. Age is another important factor when considering the treatment options. The best treatment is still controversial. Since ameloblastomas infiltrate within the cancellous spaces more, the tumor margin goes beyond the apparent clinical and radiographic margin. The attempts to remove the tumor by curettage may leave small tumor islands in bone, which may later occur as recurrences. [13]

Marginal resection is the most common treatment approach; however, there are reports of 15% recurrence. It minimizes the mandible defect, but can only be used in selected cases. Because of the above factors, segmental resection in our case was done in the initial surgery. Foster et al, reported that vascularized bone flap can rebuild any defect extension, whereas bone grafts should have their use restricted to smaller defects that are shorter than 5 cm in length. The successful rate of graft does depend not only upon size, but also upon the contact surface of well-adjusted stumps, well-vascularized receiving bone margins, tight sealing of the oral mucosa, graft stillness with internal rigid fixation and maintenance of satisfactory dental occlusion. In our case, reconstruction was done after 6 months of resection with iliac crest bone graft and stabilized with AO-Osteosynthesis plate. [14] Removable partial denture was given to the patient to restore the esthetics and function after 6 months of reconstruction. After a gap of 15 years approximately, recurrence was noticed again but was seen on the other side of the bone; the graft and its junction was absolutely healthy. There are case reports of such recurrences of ameloblastoma after a due course of time, predominantly in grafts and also in the normal bone. But in our case, there was no involvement of graft, with the graft uptake remarkably well with no recurrence.


   Conclusion Top


Successful treatment is the one that renders an acceptable prognosis, causing minimal disfigurement and is based on the behavior and potential of the tumor. The result of this study shows that even after segmental resection and reconstruction with iliac crest, chances of recurrence for solid ameloblastoma of mandible are still there. So, more important is that long-term follow-up of at least >10 years for each case of ameloblastoma should be indicated.

 
   References Top

1.World Health Organization. WHO Classification of Tumour: Pathology and Genetics of Head and Neck Tumors. In: Barnes L, Eveson JW, Reichart P, editors. Lyon, France: IARC Press; 2005.  Back to cited text no. 1
    
2.Larsson A, Almeren H. The jaw: An analysis of a consecutive series of all cases reported to the Swedish Cancer registry During 1958-1971. Acta Pathol Microbiol Scand A 1978;86A;337-49.  Back to cited text no. 2
    
3.Sher M, Singh S. Age standardized incidence rates of ameloglastoma and dentigerous cyst on the Witwatersrand, South Africa. Community Dent oral Epidemiol 1978;6:195-9.  Back to cited text no. 3
    
4.World Health Organization. WHO Classification of Tumour: Pathology and 4. Levy SH. A text book of Oral Pathology. 4th ed, Chapter 4. Cyst and Tumour of Odontogenic Origin. Geneva: WHO; 2010. p .277.  Back to cited text no. 4
    
5.Chidzonga MM. Amelobla st oma in children. Zimbabwean experience. Oral Pathol Oral Radiol Endod 1996;81:168-70.   Back to cited text no. 5
    
6.Olaitan AA, Arole G, Adekey EO. Recurrent ameloblastoma of jaw; A follow-up study. Int J Oral Maxillofac Surg 1998;27:456-60.  Back to cited text no. 6
    
7.William TP. Management of ameloblastoma: A changing perspective. J Oral Maxifac Surg 1993;51:1064-70.  Back to cited text no. 7
    
8.Gardner DG. Controversies in the nomenclature, diagnosis and treatment of ameloblastoma. In: Worthington P, Evans JR, editors. Controversies in Oral and Maxillofac Surgery. Philadelphia, PA: Saunders; 1994. p. 362.  Back to cited text no. 8
    
9.Adekeye EO, Lavery KM. Recurrent ameloblastoma of the maxillofacial region. J Maxillofac Surg 1986;14:153-7.  Back to cited text no. 9
[PUBMED]    
10.Collings SJ, Harrison A. Recurrent Ameloblastoma? An historic case report and a review of literature. Br Dent J 1993;174:202-6.  Back to cited text no. 10
[PUBMED]    
11.Gardner DG, Pecak AM. The treatment of ameloblastoma based on pathologic and anatomic principle. Cancer 1980;46:2514-9.  Back to cited text no. 11
[PUBMED]    
12.Bataineh AB. The effect of preservation of the inferior and posterior borders on recurrence of ameloblastoma of the mandible. Oral Surg Oral Med Oral Path Oral Radiol Endod 2000;90:155-63.  Back to cited text no. 12
    
13.Montoro JR, Tavares MG, Melo DH, Franco Rde L, Mello-Filho FV, Xavier SP, et al. Mandibular ameloblastoma treated by bone resection and immediate reconstruction. Braz J Otorhinolaryngol 2008;74:155-7.  Back to cited text no. 13
[PUBMED]  [FULLTEXT]  
14.Baker A, McMahon J, Parmar S. Immediate reconstruction of continuity defect of mandible after tumour surgery. J Oral Maxillofac Surg 2001;59:1333-9.  Back to cited text no. 14
[PUBMED]  [FULLTEXT]  


    Figures

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


This article has been cited by
1 ÷n mandibulaśnin tekrarlayan ameloblastomu
Mithula NAIR,Vikram SHETTY,Vidya AJILA,Shruthi HEGDE
Cukurova Medical Journal. 2020; 45(2): 763
[Pubmed] | [DOI]
2 Radical management of aggressive lesions: a case report of recurrent ameloblastoma
Carlos Henrique de CARVALHO E SOUZA,Jessyca Leal Moura F…,Lucas Lopes Araķjo SOUSA,MŠrcia Socorro Da Costa BORBA,Simone Sousa Lob„o Veras BARROS,Maria C‚ndida de Almeida LOPES
RGO - Revista Gaķcha de Odontologia. 2015; 63(3): 327
[Pubmed] | [DOI]



 

Top
   
 
  Search
 
    Similar in PUBMED
   Search Pubmed for
   Search in Google Scholar for
 Related articles
    Access Statistics
    Email Alert *
    Add to My List *
* Registration required (free)  

 
  In this article
    Abstract
    Introduction
    Case Report
    Discussion
    Conclusion
    References
    Article Figures

 Article Access Statistics
    Viewed3251    
    Printed123    
    Emailed0    
    PDF Downloaded373    
    Comments [Add]    
    Cited by others 2    

Recommend this journal