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CASE REPORT Table of Contents   
Year : 2007  |  Volume : 18  |  Issue : 4  |  Page : 218-221
Desmoplastic ameloblastoma in Indians: Report of five cases and review of literature


1 Department of Oral and Maxillofacial Pathology, Meenakshi Ammal Dental College and Hospital, Chennai, Tamilnadu, India
2 Department of Oral Pathology, Purvanchal Institute of Dental Sciences, Gorakpur, UP, India

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Date of Submission20-Sep-2006
Date of Decision15-Apr-2007
Date of Acceptance20-Apr-2007
 

   Abstract 

In the recent World Health Organization classification of odontogenic tumours, desmoplastic ameloblastoma has been characterized as a variant of ameloblastoma, with specific clinical, radiographical, and histological features. Till date, 145 cases have been reported in Japanese, Chinese, Malaysian, Western, and African populations, with very few cases described in Indians. Here, we report five cases in the Indian population. The male to female ratio was 3:2. The mean age at diagnosis was 33.2 years. Four of the tumours were located in the maxilla, in the anterior premolar region. The lone mandibular tumour was located anteriorly, crossing the midline. Three of the tumours had a mixed radiologic appearance with poorly defined borders. Unilocular change was seen in one of them. Two tumours presented as unilocular radiolucencies with specks of radiopacities and well-circumscribed borders. Histologically, irregular odontogenic islands, with a stretched-out 'kite-tail' appearance, were seen in a dense desmoplastic stroma. The peripheral layer of the epithelial islands was made up of flattened cells and the inner core was made up of spindle-shaped and, in some instances, squamous-shaped cells. In two cases, odontogenic epithelium in the form of follicles, typical of solid/multicystic ameloblastoma, was seen and these were typed as 'hybrid' variants. All the cases were treated by resection.

Keywords: Ameloblastoma, desmoplastic, hybrid, India

How to cite this article:
Sivapathasundharam B, Einstein A, Syed RI. Desmoplastic ameloblastoma in Indians: Report of five cases and review of literature. Indian J Dent Res 2007;18:218-21

How to cite this URL:
Sivapathasundharam B, Einstein A, Syed RI. Desmoplastic ameloblastoma in Indians: Report of five cases and review of literature. Indian J Dent Res [serial online] 2007 [cited 2014 Oct 31];18:218-21. Available from: http://www.ijdr.in/text.asp?2007/18/4/218/35836
Ameloblastomas are common benign epithelial odontogenic tumours that are locally invasive in nature. [1] The most common histological patterns of ameloblastoma have been classified as follicular, plexiform, acanthamatous, granular cell, basal cell, desmoplastic, and keratomatous. [1]

The first detailed report on the desmoplastic variant of ameloblastoma (DA) in the English literature was given by Eversole in 1984 who called it an 'ameloblastoma with pronounced desmoplasia.' [2] However, Takigawa et al.[3] and Uji et al.[4] were the early ones to draw attention to this unusual variant, characterized by extensive stromal desmoplasia with small compressed nests and strands of odontogenic epithelium. Radiographically, this variant exhibited no features of conventional ameloblastoma. In 2001, Philipsen, Reichart, and Takata reviewed the 100 cases of DA reported in the literature from 1984 to 2001. [5] A further 45 cases have been reported since. [6],[7],[8],[9],[10],[11],[12],[13],[14],[15],[16],[17],[18],[19],[20] The growing knowledge regarding the clinico-radiographical presentation and pathology of DAs has led to its categorization as a distinct variant of ameloblastoma in the World Health Organization (WHO) classification of odontogenic tumours in 2003. Other variants are solid/multicystic ameloblastoma (SMA), unicystic ameloblastoma, and peripheral/extraosseous ameloblastoma. [21]

The review by Philipsen, Reichart, and Takata has described cases of DA reported from Japanese, Western, Chinese, Malaysian, and Black populations. [5] Very few studies have been done in the Indian population. [18] In this article, we present the clinico-pathological features of five cases of DA in Indians.


   Case Report Top


The clinical, radiographical, and histological findings of the five cases of DA are summarized in [Table - 1].

The male to female ratio was 3:2. The mean age at diagnosis was 33.2 years. Four of the tumors were located in the maxilla, in the anterior premolar region. The lone mandibular tumor was located anteriorly and crossed the midline.

Three of the tumors had mixed radiologic appearances, with poorly defined borders mimicking fibro-osseous lesions [Figure - 1]. Unilocular change was seen in one of them [Figure - 1]. Two tumors presented as unilocular radiolucencies with specks of radiopacities [Figure - 2]. Both these tumors had well-circumscribed borders. Even though the tumors were located in the alveolar region, resorption of teeth was not seen. In two cases, there was displacement of the teeth [Figure - 1],[Figure - 2].

Histologically, irregular odontogenic islands with a stretched-out 'kite-tail' appearance were seen in a dense desmoplastic stroma [Figure - 3]. The peripheral layer of the epithelial islands was made up of flattened cells and the inner core was made up of spindle-shaped and, in some instances, squamous-shaped cells. The typical peripheral columnar cells and inner stellate reticulum-like cells of SMA were absent. In two of the cases however, odontogenic epithelium in the form of follicles typical of SMA were observed, besides the irregular stretched-out epithelial islands in a desmoplastic background [Figure - 4]. These were typed as 'hybrid' variants of DA. All the cases were treated by resection.


   Discussion Top


Desmoplastic ameloblastoma (DA) has been characterized by WHO as a variant of ameloblastoma, with specific clinical, imaging, and histological features. [21]

DA is similar to SMA in age and gender distribution. The mean age of DA at the time of diagnosis is 42.9 years. The male to female ratio is 1:0.9. These data are based on only 109 cases [22] and more studies are needed to evaluate the true age and gender distribution of DA.

Previous reported cases of DA have shown this variant to have a strong predilection for the anterior premolar region of the jaws, occurring with equal frequency in both the maxilla and the mandible, much in contrast to SMA, which is more common in the posterior mandible. [22] All our cases showed this predilection for the anterior jaw, even though four cases occurred in the maxilla and only one in the mandible.

Wang et al. studied the radiologic appearance of 15 cases of DA and reported three distinct radiologic features: namely, unilocular formation, containing varying amounts of radiopaque islands or strands of material; multilocular destruction, containing irregular or line-like radiopaque areas; and mixed destruction, showing plexiform radiopaque material and unilocular change. [8] The third mixed pattern was noted in three of our cases, with the unilocular change seen in the lone mandibular tumor. SMA, in contrast, is usually a multi- or unilocular radiolucency. Two of our cases, though unilocular, showed specks of radiopacity, resembling the first pattern reported by Wang et al. Takata et al. attributed the mixed radiologic appearance and ill-defined margins of DA to its infiltrative nature. [23] They argued that the tumor infiltrates the bone marrow spaces and in the process, remnants of the original bone, either nonmetaplastic or nonneoplastic, remain in the tumor tissue. But this argument does not explain the absence of such bone spicules in SMAs. Philipsen et al. have suggested that the tumor islands induced proliferation of mesenchymal cells, resulting in desmoplasia and metaplastic bone formation (osteoplasia). [24] This argument again is not acceptable, as the same inductive potential is not observed in SMA.

Root resorption is a common finding in DA [22] but was not seen in any of our cases, though displacement of teeth was seen in two cases.

The histologic appearance of DA is characteristic and differs from SMA in the nature of the collagenous fibrous stroma and the morphology and constituent cell types of the odontogenic islands. [5],[22] The stromal connective tissue shows extensive dense collagenization with occasional areas of bone. The odontogenic islands are irregular and appear to be compressed or squeezed by the stromal tissue, giving rise to a stretched-out tail-like appearance. Neither the palisaded peripheral columnar cells with reversed polarity nor the inner stellate reticulum-like cells seen in SMA are observed in DA. This variant exhibits odontogenic islands made up of peripheral flattened, cuboidal, or squamous cells with rare foci of palisaded columnar cells and central hypercellular spindle or polygonal cells. Microcysts, either containing eosinophilic amorphous deposits or appearing empty, may occur centrally. In some instances, typical follicular tumor islands (as seen in SMA) surrounded by less dense collagenous stroma have been observed in an otherwise typical DA. This entity has been termed as a 'hybrid' variant. Three of our cases presented with features typical of DA. Two of our cases belonged to the hybrid variety.

Recently, Hirota et al. reported a case of DA with a few areas of characteristic compressed tumor islands along with a predominantly basal cell pattern of odontogenic cells, similar to the appearance in basal cell ameloblastoma. [17]

Even after two decades of the first report of DA, the cause for this peculiar histologic appearance is still unclear. Compared to the SMAs, various immunohistochemical studies have reported DA tumor cells as showing variable expression of S-100 protein and desmin, [25] marked immunoexpression of transforming growth factor-beta (TGF-b), [23] high expression of caspase-3 and Fas, [7] decreased expression of CK19, [26] and high expression of p63. [27] Compared to the stroma of SMA, the desmoplastic stroma of DA has been reported to show a strong positive reaction for collagen type VI (ruling out the scar tissue), [22] immunonegativity for tenascin, and strong immunopositive reaction for fibronectin and type 1 collagen. [20] Kishino et al.[6] demonstrated oxytalan fibres in the stromal tissue of one case, suggesting that the tumor had derived from the epithelial rest of Malassez in the periodontal membrane of a neighbouring tooth.

Desmoplasia of the stromal connective tissue in DA can be argued to be a maturation change in a SMA, as similar dense collagenization is seen during maturation of longstanding tumors. This argument can be supported by the existence of 'hybrid' tumors, more specifically in our two hybrid cases, wherein the follicles were present in a desmoplastic background. But the lesser frequency of DA in the posterior mandible compared to SMAs is then unanswered. Firstly, it is probable that the location of the tumor can influence the maturity of the lesion and, hence, the tumors in the anterior jaws may mature sooner than those occurring in the posterior mandible. Of significance in this context is the higher frequency of SMAs in the anterior jaws in Blacks, indicating the possible racial influence in the site predilection of ameloblastomas. It is also possible that the majority of these ameloblastomas could turn out to be desmoplastic variants if a careful histological review is carried out. Secondly, DA might not actually be a rare entity. Many hybrid lesions may have been misclassified, since the presence of typical ameloblastic islands in some areas could have warranted a diagnosis of SMA.

Ameloblastomas may thus be common in the anterior and posterior jaw and the tumors in the anterior jaw may mature early, explaining the unique site predilection, mixed radiolucent appearance, and histologic presentation of DAs.

 
   References Top

1.Kramer IRH, Pindborg JJ, Shear M. Histological typing of Odontogenic tumour. Springer-Verlag, World Health Organization: Berlin; 1992. p. 11-4.  Back to cited text no. 1      
2.Eversole LR, Leider AS, Hansen LS. Ameloblastoma with pronounced desmoplasia. J Oral Maxillofac Surg 1984;42:735-40.  Back to cited text no. 2  [PUBMED]    
3.Takigawa T, Matsumoto M, Sekine Y. A case report of ameloblastoma prolifertaed like epulis of maxilla (Article in Japanese). Nihon Univ Dent 1981;55:920-4.  Back to cited text no. 3      
4.Uji Y, Kodama K, Sakamoto A, Taen A. An ameloblastoma with interesting findings (Article in Japanese). Jpn J Oral Maxillofac Surg 1983;29:1512-9.  Back to cited text no. 4      
5.Philipsen HP, Reichart PA, Takata T. Desmoplastic ameloblastoma (including 'hybrid' lesion of ameloblastoma). Biological profile based on 100 cases from the literature and own files. Oral Oncol 2001;37:455-60.  Back to cited text no. 5      
6.Kishino M, Murakami S, Fukuda Y, Ishida T. Pathology of the desmoplastic ameloblastoma. J Oral Pathol Med 2001;30:35-40.  Back to cited text no. 6  [PUBMED]  [FULLTEXT]  
7.Kumamoto H, Kimi K, Ooya K. Immunohistochemical analysis of apoptosis-related factors (Fas, Fas ligand, caspase-3 and single-stranded DNA) in ameloblastomas. J Oral Pathol Med 2001;30:596-602.   Back to cited text no. 7  [PUBMED]  [FULLTEXT]  
8.Wang S, Chen X, Chen Y. Radiologic features of desmoplastic ameloblastoma (Article in Chinese). Zhonghua Kou Qiang Yi Xue Za Zhi 2001;36:253-5.   Back to cited text no. 8  [PUBMED]    
9.Beckley ML, Farhood V, Helfend LK, Alijanian A. Desmoplastic ameloblastoma of the mandible: Case report and review of literature. J Oral Maxillofac Surg 2002;60:194-8.  Back to cited text no. 9  [PUBMED]  [FULLTEXT]  
10.Wakoh M, Harada T, Inoue T. Follicular desmoplastic ameloblastoma with radiographic features of concomitant fibro-osseous and solitary cystic lesions. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2002;94:774-80.  Back to cited text no. 10  [PUBMED]  [FULLTEXT]  
11.Iida S, Kogo M, Kishino M, Matsuya T. Desmoplastic ameloblastoma with large cystic change in the maxillary sinus: Report of a case. J Oral Maxillofac Surg 2002;60:1195-8.  Back to cited text no. 11  [PUBMED]  [FULLTEXT]  
12.Mintz S, Velez I. Desmoplastic variant of ameloblastoma: Report of two cases and review of the literature. J Am Dent Assoc 2002;133:1072-5.  Back to cited text no. 12  [PUBMED]  [FULLTEXT]  
13.Durmus E, Kalayci A, Ozturk A, Gunhan O. Desmoplastic ameloblastoma in the mandible. J Craniofac Surg 2003;14:873-5.  Back to cited text no. 13  [PUBMED]  [FULLTEXT]  
14.Maresi E, Tortorici S, Orlando E, Burruano F. Desmoplastic ameloblastoma. Clinical and histopathological diagnostic criteria. Minerva Stomatol 2003;52:127-32.  Back to cited text no. 14      
15.Pillai RS, Ongole R, Ahsan A, Radhakrishnan RA, Pai KM. Recurrent desmoplastic ameloblastoma of the maxilla: A case report. J Can Dent Assoc 2004;70:100-4.   Back to cited text no. 15  [PUBMED]  [FULLTEXT]  
16.Asaumi J, Hisatomi M, Yanagi Y, Matsuzaki H, Choi YS, Kawai N, et al. . Assessment of ameloblastomas using MRI and dynamic contrast-enhanced MRI. Eur J Radiol 2005;56:25-30.  Back to cited text no. 16      
17.Hirota M, Aoki S, Kawabe R, Fujita K. Desmoplastic ameloblastoma featuring basal cell ameloblastoma: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;99:160-4.  Back to cited text no. 17  [PUBMED]  [FULLTEXT]  
18.Desai H, Sood R, Shah R, Cawda J, Pandya H. Desmoplastic ameloblastoma: Report of a unique case and review of literature. Indian J Dent Res 2006;17:45-9.  Back to cited text no. 18  [PUBMED]  Medknow Journal  
19.Adebiyi KE, Ugboko VI, Omoniyi-Esan GO, Ndukwe KC, Oginni FO. Clinicopathological analysis of histological variants of ameloblastoma in a suburban Nigerian population. Head Face Med 2006;2:42.   Back to cited text no. 19  [PUBMED]  [FULLTEXT]  
20. dos Santos JN, De Souza VF, Azevedo RA, Sarmento VA, Souza LB. 'Hybrid' lesion of desmoplastic and conventional ameloblastoma: Immunohistochemical aspects. Rev Bras Otorrinolaringol (Engl Ed) 2006;72:709-13.   Back to cited text no. 20      
21.Barnes L, Eveson JW, Reichart PA, Sidransky D, editors. World Health Organization classification of tumours. Pathology and genetics of head and neck tumours. IARC Press: Lyon; 2005. p. 284.  Back to cited text no. 21      
22.Reichart PA, Philipsen HP. Desmoplastic ameloblastoma. In : Odontogenic tumours and allied lesions. Quintessence Publishing Co Ltd: London; 2004. p. 69-74.  Back to cited text no. 22      
23.Takata T, Miyauchi M, Ogawa I, Kudo Y, Takekoshi T, Zhao M, et al. . Immunoexpression of transforming growth factor beta in desmoplastic ameloblastoma. Virchows Arch 2000;436:319-23.  Back to cited text no. 23      
24.Philipsen HP, Ormiston IW, Reichart PA. The desmo- and osteoplastic ameloblastoma. Histologic variant or clinicopathologic entity? Case reports. Int J Oral Maxillofac Surg 1990;48:1231-5.  Back to cited text no. 24      
25.Siar CH, Ng KH. Patterns of expression of intermediate filaments and S-100 protein in desmoplastic ameloblastoma. J Nihon Univ Sch Dent 1993;35:104-8.  Back to cited text no. 25  [PUBMED]    
26.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. 26  [PUBMED]  [FULLTEXT]  
27.Kumamoto H, Ohki K, Ooya K. Expression of p63 and p73 in ameloblastomas. J Oral Pathol Med 2005;34:220-6.  Back to cited text no. 27  [PUBMED]  [FULLTEXT]  

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Correspondence Address:
B Sivapathasundharam
Department of Oral and Maxillofacial Pathology, Meenakshi Ammal Dental College and Hospital, Chennai, Tamilnadu
India
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DOI: 10.4103/0970-9290.35836

PMID: 17938502

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    Figures

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