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  Table of Contents  
LETTER TO THE EDITOR
Year : 2014  |  Volume : 51  |  Issue : 4  |  Page : 592-593
 

Giant ameloblastoma: A rarity


1 320 Field Hospital, 99 APO., Trivandrum, Kerala, India
2 Department of Preventive and Community Dentistry, Ahmedabad Dental College, Ahmedabad, Gujarat, India
3 Department of Oral Pathology and Microbiology, College of Dental Science, Bhubaneswar, Odisha, India
4 Department of Oral Pathology and Microbiology, Vaidik Dental College and Research Centre, Daman, India

Date of Web Publication1-Feb-2016

Correspondence Address:
Dr. K Choudhary
320 Field Hospital, 99 APO., Trivandrum, Kerala
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0019-509X.175298

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How to cite this article:
Choudhary K, Shah H, Panda S, Gandhi S. Giant ameloblastoma: A rarity. Indian J Cancer 2014;51:592-3

How to cite this URL:
Choudhary K, Shah H, Panda S, Gandhi S. Giant ameloblastoma: A rarity. Indian J Cancer [serial online] 2014 [cited 2019 Aug 24];51:592-3. Available from: http://www.indianjcancer.com/text.asp?2014/51/4/592/175298


Sir,

The aim of this letter was to report a gigantic ameloblastoma in the anterior mandibular region, which is very rare considering the location as well as gigantic size owing to the advanced diagnostic and treatment aspect of ameloblastoma.

The ameloblastoma is a true neoplasm of odontogenic epithelial origin. It is the second most common odontogenic neoplasm, and only odontoma out numbers it in reported frequency of occurrence. [1],[2],[3] It can enlarge and invade the whole jaw and adjacent tissues.

However, until now, giant ameloblastoma has received very limited attention and reported in the literature. The patient reported here was the case with the biggest ameloblastoma in anterior mandible reported in the literature.

We are reporting a case of giant ameloblastoma in a 65-year-old female patient who reported to our hospital for treatment of large swelling in anterior portion of mandible. She noticed the swelling since 10 years, but she did not seek any consultation. The swelling has been slowly increasing to reach the maximum present size of 14 cm × 11 cm in greatest dimension clinically [Figure 1]. Swelling involves anterior portion of mandible extending up to angle region on both side of mandible. No period of accelerated growth or decrease in size of the swelling was reported by the patient. The discomfort due to huge size was the principle complaint. The mucosa over the swelling appears stretched but it was otherwise normal. The skin at the inferior border of swelling was fixed to the underlying bone. The swelling was hard in consistency with mild irregularities. Intraorally labial vestibule in the anterior mandibular region is obliterated. A provisional diagnosis of ameloblastoma was made.
Figure 1: Clinical image showing swelling of 14 cm × 8 cm in lower face

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Postero anterior mandibular view shows large multilocular lesion involving whole body of mandibleextending upto angle of mandible bilaterally [Figure 2]. Incision biopsy was taken and sent for histopathological examination. Microscopic examination hematoxylene and eosine stained section shows ameloblastic follicles consisting of peripheral ameloblast like cells and central stellate reticulum like cells in a background of moderately collagenous connective tissue stroma. Some of the follicles are showing squamous metaplasia keratin in the center [Figure 3]. Final diagnosis of acanthomatou sameloblastoma was given. Resection of the segment of mandible done and size of resected specimen was 14 cm × 11 cm × 8 cm [Figure 4]. Immediate surgical reconstruction was done using non-vascular free fibula graft and reconstruction plate. Patient is on continuous follow-up and no recurrence was noted after 1 year of follow-up.
Figure 2: Large multilocular radiolucency is seen in the anterior mandibular region, extending from the body of the mandible bilaterally to angle of mandible

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Figure 3: Ameloblastic follicles with squamous metaplasia. Some follicles show cystic degeneration, ×10 mangnification

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Figure 4: Gross specimen

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Giant ameloblastoamas are rarely reported in present literature due to timely sought treatment. Michael, et al. in 2010 reported a recurrent ameloblastoma of size 24 cm × 19 cm × 15 cm in diameter, diagnosed as intracranial extension of giant multicystic maxillo-mandibular ameloblastoma. Intracranial extension of the mass through the eroded squamous temporal bone, into the right middle cranial fossa was observed, with mass effect on the right lateral ventricle and septum pellucidum. The ipsilateral Sylvian fissure, basal cisterns, and the cerebral sulci were all effaced, and it was case of recurrent amelobalstoma.[4] Except this, Hughes, et al. from the United States of America described a giant ameloblastoma that measured 15.2 cm × 11.4 cm × 12.0 cm with no intracranial extension.[5] Dunn, et al. reported a giant ameloblastoma, which measured 17 × 15 × 13 on computed tomogram but no intracranial extension was observed.[6] Kyriazis, et al. reported a 73-year-old Negro woman with an aggressive ameloblastoma. From its original site in the left maxilla, the tumor extended upward, destroyed the base of the skull, and infiltrated the brain, replacing the temporal lobe of the brain.[7] Case reported by Mallick, et al. Extended into para-pharyngeal space to infracrinal space causing erosion of the base of skull and petrous temporal bone, reaching into cranial cavity. No evidence of brain parenchymal involvement was noted. Right side cervical lymphadenopathy was also present.[8] In literature search we did not find any thorasic and abdominal cavity extension of ameloblastoma. In Indian population, no case of intracerebral extension of ameloblastoma had been reported. No case has been reported exclusively involving anterior mandible extending up to angle of mandible bilaterally. Furthermore, it is the maximum size ameloblastoama involving anterior mandible and extending bilaterally reported in Indian population best of our knowledge based on literature search.

However, cases have been reported in lower socio-economical group patients. Giant ameloblastoma often described in African term as "second head" due to delay in presentation. This delay in presentation is due to a combination of poor socio-economic background and beliefs of the patients. Treatment of this condition is usually a challenge to the surgeon in a developing country with minimal surgical facilities.[9] In India alsomain reason behind this delay is lack of awareness and unavailability of tertiary care centre in remote places. Long duration of lesion may be a reason for acanthomatous change as reported in our case. It has been reported that repeated intervention and long standing ameloblastoma are the causes of metastatic ameloblastoma. However, in our case the patient is healthy post-operatively. In conclusion, all patients diagnosed with giant and long standing ameloblastoma should be screened for metastatic ameloblastoma and any other recurrence.

 
  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.
Neville BW, Damm DD, Allen CM, Bouquot JE. Odontogenic Tumor. Oral and Maxillofacial Pathology. 2nd ed. Philadelphia: WB Saunders; 2002. p. 611-9.  Back to cited text no. 2
    
3.
Kessler HP, Schwartz-Dabney C, Ellis E 3rd. Recurrent left mandibular enlargement. J Contemp Dent Pract 2003;3:127-37.  Back to cited text no. 3
    
4.
Michael AN, Eyituoyo MO, Mubarak ME, Olabode VO. Intracranial extension of giant multicysticmaxillomandibularameloblastoma: A case report. Internet J Radiol 2010;12:1-4.  Back to cited text no. 4
    
5.
Hughes CA, Wilson WR, Olding M. Giant ameloblastoma: Report of an extreme case and a description of its treatment. Ear Nose Throat J 1999;78:568-74.  Back to cited text no. 5
    
6.
Dunn JL, Olan WJ, Bank WO, Narang AK, Schwartz AM. Giant ameloblastoma: Radiologic diagnosis and treatment. Radiographics 1997;17:531-6.  Back to cited text no. 6
    
7.
Kyriazis AP, Karkazis GC, Kyriazis AA. Maxillary ameloblastoma with intracerebral extension. Report of a case. Oral Surg Oral Med Oral Pathol 1971;32:582-7.  Back to cited text no. 7
    
8.
Mallick JA, Ali SA. Ameloblastoma: Management and review of literature. J Pak Med Assoc 2002;52:488-90.  Back to cited text no. 8
    
9.
Obisesan BA, Adeyemo WL, Ladele BS. Management of giant ameloblastoma in an African environment-report of 2 cases. Pakistan Oral Dental J 2004;24:3-6.  Back to cited text no. 9
    


    Figures

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



 

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