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An Official Publication of the Indian Association of Oral and Maxillofacial Pathologists

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Year : 2022  |  Volume : 26  |  Issue : 2  |  Page : 273-276

Granular cell ameloblastoma as a solitary peripheral growth after twenty years of segmental resection of the mandible: A rare case of recurrence

1 Department of Oral Pathology and Microbiology, Nair Hospital Dental College, Mumbai, Maharashtra, India
2 Department of Oral and Maxillofacial Surgery, Nair Hospital Dental College, Mumbai, Maharashtra, India

Date of Submission17-Jan-2022
Date of Decision24-Mar-2022
Date of Acceptance28-Mar-2022
Date of Web Publication28-Jun-2022

Correspondence Address:
Rajiv S Desai
Department of Oral Pathology and Microbiology, Nair Hospital Dental College, Mumbai - 400 008, Maharashtra
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/jomfp.jomfp_24_22

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The ameloblastoma is a slowly growing, locally invasive, benign epithelial odontogenic neoplasm of the jaws with a high rate of recurrence if not removed adequately. We report an interesting case of granular cell ameloblastoma, which presented as a solitary, peripheral, soft tissue growth 20 years after initial segmental resection of the left mandible. The basal layer of oral mucosa could be the possible source of peripheral ameloblastoma in our case. In order to reduce the chances of recurrence, we suggest to incorporate mucosal stripping along with the conventional treatment as a mandatory rather than an elective procedure while treating ameloblastoma.

Keywords: Ameloblastoma, granular cell ameloblastoma, recurrence

How to cite this article:
Fukate CA, Bansal SP, Andrade NN, Desai RS. Granular cell ameloblastoma as a solitary peripheral growth after twenty years of segmental resection of the mandible: A rare case of recurrence. J Oral Maxillofac Pathol 2022;26:273-6

How to cite this URL:
Fukate CA, Bansal SP, Andrade NN, Desai RS. Granular cell ameloblastoma as a solitary peripheral growth after twenty years of segmental resection of the mandible: A rare case of recurrence. J Oral Maxillofac Pathol [serial online] 2022 [cited 2022 Aug 14];26:273-6. Available from: https://www.jomfp.in/text.asp?2022/26/2/273/348748

   Introduction Top

The ameloblastoma is the most common benign intraosseous epithelial odontogenic neoplasm predominantly affecting the posterior mandible. It is a slow growing, locally invasive tumour characterized by expansion and a tendency for recurrence if not removed adequately.[1] Although histological variants have no prognostic implication, the six main histological types include (i) follicular, (ii) plexiform, (iii) acanthomatous, (iv) granular, (v) basaloid, and (vi) desmoplastic forms.[2]

We report an unusual case of granular cell ameloblastoma (GCA) which presented as a peripheral, soft tissue growth in the left posterior mandible of a 40-year-old female patient, 20 years after the primary segmental resection of the left mandible. Our case also highlights the importance of life-time follow-up for a patient with ameloblastoma.

   Case Report Top

A 40-year-old female patient reported to our department with a chief complaint of gradually increasing painless mass in the left mandibular posterior region since 1 month. The patient gave the past history of surgical removal of the ameloblastoma involving the left mandibular posterior region in 2001. Patient's medical, social, and family history was non-contributory.

Extra-oral examination showed slight facial asymmetry with left mandibular deviation. No evidence of draining sinus, pus discharge, or cervical lymphadenopathy was observed [Figure 1]a. Intra-orally, a 2 cm × 1 cm solitary, nodular growth in the left mandibular edentulous posterior region was observed. The overlying mucosa was erythematous and fissured [Figure 1]b. On palpation, the growth was non-tender, sessile, and firm in consistency. Based on the clinical examination, differential diagnosis of reactive lesions like pyogenic granuloma, peripheral giant cell lesion, and fibrous hyperplasia was considered. Keeping previous history of surgical removal of the ameloblastoma in mind, due consideration to recurrent ameloblastoma was also kept in mind.
Figure 1: (a) Extra-oral view showing left deviation of the chin. (b) Intraoral growth in the edentulous left mandibular posterior region

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A panoramic radiograph showed the presence of radio-opaque bone plate extending from the left symphysis to the left condylar region suggestive of a previous segmental resection of the left mandible followed by reconstruction of the left mandible with metallic bone plate [Figure 2]. No evidence of lytic lesion was observed in the area corresponding to the clinical lesion.
Figure 2: The panoramic radiograph showing a well-defined characteristic radio-opacity in the form of bone plate on the left side of mandible extending from the mandibular symphysis to left condylar region

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Surgical excision of the mass was performed under general anaesthesia. Grossly the tumour was white in colour, ovoid in shape, measuring about 2.5 cm × 1.5 cm in size. The cut surface was smooth, white in colour, and firm in consistency [Figure 3].
Figure 3: Gross specimen appearing ovoid soft tissue mass measuring 2 cm × 1.5 cm. Consistency of the specimen was firm. Inset: solid, smooth, and whitish cut surface

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Microscopic examination of the haematoxylin and eosin stained sections showed fibrous connective tissue exhibiting numerous odontogenic epithelial islands with the peripheral tall columnar cell revealing nuclear reversal of polarity, resembling ameloblast-like cells [Figure 4]. Centre of the islands showed stellate-reticulum-like cells with numerous large eosinophilic rounded or polyhedral granular cells. The final diagnosis of peripheral GCA was rendered [Figure 5]. The post-operative healing was uneventful. However, the patient was lost to follow-up.
Figure 4: Histopathology showing numerous odontogenic epithelial islands composed of peripheral tall, columnar, ameloblast-like cells, and centrally placed stellate reticulum-like cells with coarse granular eosinophilic cells (Haematoxylin & eosin stain 4×)

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Figure 5: Histopathology showing stellate-reticulum like cells along with numerous large eosinophilic rounded or polyhedral granular cells (Haematoxylin & eosin stain 10×)

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   Discussion Top

The GCA is a rare variant accounting for only 5% of all ameloblastoma and found as an admixture with other histologic patterns, mostly the follicular variant.[3],[4] Microscopically, GCA is characterised by the groups of granular cells, which have abundant cytoplasm filled with eosinophilic granules that resemble lysosomes, both ultrastructurally and histochemically. The acquisition of granular cell phenotype has been attributed to an ageing or degenerative change in long-standing lesions like ours.[5],[6] GCA may rarely behave in a malignant fashion giving rise to regional or distant metastasis.[7],[8] Extra-oral soft tissue recurrent ameloblastoma as a granular cell variant 41 years after extirpation of a follicular type ameloblastoma has been reported in the literature.[9]

The present case is different from the previous reports of ameloblastoma because the tumour occurred as a solitary intra-oral soft tissue growth 20 years after the first surgery and did not show intra-osseous component or malignant alteration after such a long period.[10],[11]

The differential diagnosis of GCA includes granular cell odontogenic tumour, granular cell tumour and congenital epulis, all of which have different clinical presentation and biological behaviours.[12]

Immunohistochemically, the granular cells are found to be positive for cytokeratin, CD68, lysozymes, and alpha -1-antichymotrypsin, indicating epithelial origin and lysosomal aggregation.[8],[13] Various researchers have demonstrated a link between the neuroectoderm, ameloblastoma, and normal oral ectoderm, suggesting the possible origin of ameloblastoma from the basal layer of the oral mucosa.[14],[15] Occurrence of GCA as a peripheral growth 20 years after the first surgery favours the hypothesis that the basal cell layer of oral mucosa could be the sought out culprit for recurrence in our case. In view of the above, we recommended the stripping of the overlying mucosa along with the conventional treatment to reduce the chance of recurrence. The present case also emphasizes the life-time follow-up for the patient with ameloblastoma.

Author contributions

R.D. and N.A have been involved in drafting the manuscript.

S.B. and C.F. revised it critically. All the authors have read and approved the final manuscript.

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 initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

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Conflicts of interest

There are no conflicts of interest.

   References Top

El-Naggar AK, Chan JK, Grandis JR, Takata T, Slootweg PJ. World Health Organization Classification of Head and Neck Tumours. 4th ed. Lyon, France: IARC Press; 2017.  Back to cited text no. 1
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. 2
Hartman KS. Granular-cell ameloblastoma. Oral Surg Oral Med Oral Pathol 1974;38:241-53.  Back to cited text no. 3
Kameyama Y, Takehana S, Mizohata M, Nonobe K, Hara M, Kawai T, et al. A clinicopathological study of ameloblastomas. Int J Oral Maxillofac Surg 1987;16:706-12.  Back to cited text no. 4
Nikitakis NG, Tzerbos F, Triantafyllou K, Papadimas C, Sklavounou A. Granular cell ameloblastoma: An unusual histological subtype report and review of literature. J Oral Maxillofac Res 2011;1:e3.  Back to cited text no. 5
Nasu M, Takagi M, Yamamoto H. Ultrastructural and histochemical studies of granular-cell ameloblastoma. J Oral Pathol 1984;13:448-56.  Back to cited text no. 6
Takahashi K, Kitajima T, Lee M, Iwasaki N, Inoue S, Matsui N, et al. Granular cell ameloblastoma of the mandible with metastasis to the third thoracic vertebra. A case report. Clin Orthop Relat Res 1985;197:171-80.  Back to cited text no. 7
Kumamoto H, Ooya K. Immunohistochemical and ultrastructural investigation of apoptotic cell death in granular cell ameloblastoma. J Oral Pathol Med 2001;30:245-50.  Back to cited text no. 8
Okubo H, Yagishita H, Koike T, Kurita H, Kurashina K. Recurrent ameloblastoma as a granular cell variant 41 years after extirpating follicular tumor: Report of a case. Asian J Oral Maxillofac Surg 2010;22:159-63.  Back to cited text no. 9
Rapidis AD, Andressakis DD, Stavrianos SD, Faratzis G, Arnogiannaki-Liappi N, Lagogiannis GA, et al. Ameloblastomas of the jaws: Clinico-pathological review of 11 patients. Eur J Surg Oncol 2004;30:998-1002.  Back to cited text no. 10
Gardner DG. Critique of the 1995 review by Reichart et al. of the biologic profile of 3677 ameloblastomas. Oral Oncol 1999;35:443-9.  Back to cited text no. 11
Mirchandani R, Sciubba JJ, Mir R. Granular cell lesions of the jaws and oral cavity: A clinicopathologic, immunohistochemical, and ultrastructural study. J Oral Maxillofac Surg 1989;47:1248-55.  Back to cited text no. 12
Dina R, Marchetti C, Vallania G, Corinaldesi G, Eusebi V. Granular cell ameloblastoma. An immunocytochemical study. Pathol Res Pract 1996;192:541-6.  Back to cited text no. 13
Gagari E, Deligianni E, Antoniades DZ, Goode R. Neuroendocrine markers in ameloblastoma-An immunohistochemical study of thirty two cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:194.  Back to cited text no. 14
Nagamalini BR, Suneela S, Narayan TV, Shreedhar B, Mohanty L, Shenoy S, et al. Origin of ameloblastoma from Basal cells of the oral epithelium-establishing the relation using neuroectodermal markers. J Clin Diagn Res 2014;8:ZC447.  Back to cited text no. 15


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


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