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Year : 2015  |  Volume : 11  |  Issue : 4  |  Page : 1033

Ameloblastic fibroma with ghost cell differentiation and calcification: A unique case report

Department of Oral and Maxillofacial Pathology, Sri Gobind Tricentenary Dental College, Gurgaon, Haryana, India

Date of Web Publication15-Feb-2016

Correspondence Address:
Manpreet Arora
Department of Oral and Maxillofacial Pathology, Sri Gobind Tricentenary Dental College Dental College, Gurgaon - 122 505, Haryana
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/0973-1482.150436

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 > Abstract 

The ameloblastic fibroma (AF) is an uncommon odontogenic tumor that may present an aggressive behavior and may have potential for malignant transformation. Ghost cell differentiation within AF is extremely rare. There are only seven cases in English literature in which ghost cells are found in AF but all these previously reported cases were associated with typical calcifying odontogenic cyst. Here, we present a unique case in 31/2-year-old child with solid lesion which comprised odontogenic epithelium strands, islands, and myxoid ectomesenchyme with focal areas of ghost cell differentiation and calcification associated with neoplastic epithelium.

Keywords: Ameloblastic fibroma, calcification, COC, ghost cells

How to cite this article:
Arora M, Dave A, Shetty V, Saluja P. Ameloblastic fibroma with ghost cell differentiation and calcification: A unique case report. J Can Res Ther 2015;11:1033

How to cite this URL:
Arora M, Dave A, Shetty V, Saluja P. Ameloblastic fibroma with ghost cell differentiation and calcification: A unique case report. J Can Res Ther [serial online] 2015 [cited 2020 Jan 29];11:1033. Available from: http://www.cancerjournal.net/text.asp?2015/11/4/1033/150436

 > Introduction Top

Ameloblastic fibroma (AF) is a rare odontogenic tumor with both neoplastic epithelial and ectomesenchyme components, but with no dental hard tissues.[1] If there is dentin or enamel formation, the lesion is classified as ameloblastic fibro-dentinoma (AFD) or ameloblastic fibro-odontoma (AFO), respectively.[1] Even more rarely, AFs may be combined with other odontogenic tumors, such as ameloblastomas and calcifying odontogenic cyst (COC). AFs containing ghost cells such as those in COCs are even a rarer event. To our knowledge, there are seven cases of AF with ghost cells differentiation. These previously mentioned cases are typical COCs with AF having cystic-solid lesions. Here, we report a case of solid AF with ghost cell differentiation and calcifications in neoplastic epithelium but with no features of COC as previously reported.

 > Case Report Top

A 3½-year-old male patient was referred with the chief complaint of swelling of left side of face. Patient's parents noticed the swelling when the child was 1 year old. They applied some herbal medication with hot fermentation which gave some temporary relief. Swelling started increasing rapidly since 1½ year. The past medical and family history was not relevant. Delivery of child was normal with no history of medication during pregnancy. On clinical examination, child built was thin and was well-oriented with no systemic findings. Extraoral examination reveals facial asymmetry with extraoral swelling on left side of face [Figure 1]. Nasal septum was deviated toward right side. Left submandibular lymph nodes were palpable. Intraoral examination revealed a non-tender diffuse hard swelling of 6 × 6 cm. All teeth were present with displaced 51 61 62 63 64 and mobile 65. Overlying mucosa was ulcerated. Panaromic radiograph showed a multilocular radiolucent lesion in left maxilla [Figure 2]. A 3D computed tomography was done which revealed ill defined osteolytic lesion with extensive spread of the tumor mass involving maxillary sinus, nasal septum, and alveolar process [Figure 3]. The clinical diagnosis of ameloblastoma was given. Incisional biopsy was sent to department of Oral and maxillofacial pathology SGT Dental College. Microscopic examination showed a typical AF with epithelial strands buds and island with peripheral tall columnar cells and stellate reticulum-like cells in the center distributed in the hypercellular immature fibrous tissue [Figure 4]a. The final diagnosis of AF was given. Surgical excision of the mass was done under GA and the specimen was submitted. Grossly the excised specimen was solid [Figure 5]. Microscopic examination of this specimen in addition to the above-mentioned features revealed cystic region lined by epithelium having basal tall columnar cells with palisaded nuclei and superficial stellate reticulum like cells. Ghost cells differentiation just like those seen in COC and areas of calcifications were seen within the epithelium [Figure 4]b[Figure 4]c,[Figure 4]d.
Figure 1: Clinical picture showing swelling on left maxilla

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Figure 2: Panoramic radiograph revealing multilocular radiolucency in left maxilla with displacement of teeth

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Figure 3: A 3D computed tomography image shows destruction of left maxilla

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Figure 4: (a) Photomicrograph showing islands and strands of odontogenic epithelium and cellular mesenchyme tissue resembling dental papilla (original Magnification ×20) (b) Lining epithelium with ameloblastic differentiation and stellate reticulum like cells (original Magnification ×10) (c) Typical ghost cells in ameloblastic lining epithelium (original Magnification ×20) (d) Calcifications within stellate reticulum-like cells

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Figure 5: The excised lesion consisting of solid soft tissue mass

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 > Discussion Top

The AF is a mixed odontogenic tumor composed of odontogenic ectomesenchyme resembling dental papilla with epithelial strands and nests similar to the dental lamina and enamel organ, but with no dental hard tissue.[1] Occurrence of ghost cells in AF as those seen in COC is even a rarer event.[2],[3],[4],[5] So far, there have been only seven cases in English literature but they all had COC and the component of AF existed in cystic wall or some solid area [Table 1]. Grossly, the lesion described in our case was solid with characteristic features of AF. Interestingly, in few areas ameloblastic epithelium with ghost cell features and calcifications were manifested.
Table 1: The reported cases of calcifying odontogenic cyst with ameloblastic fibroma[6]

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Ghost cells are essentially requisite for the diagnosis of odontogenic ghost cell tumor (OGCT) and COC, although they are neither unique to nor pathognomonic of these lesion.[6] But they had been occasionally reported to associate with other odontogenic tumors such as odontomas,[7],[8] ameloblastomas,[9] adenomatoid odontogenic tumors,[10] and AF.

The biologic mechanism causing such a unique combination is not readily apparent. The possible pathogenic mechanisms would seem to be either a collision of two separate lesions or a transformation of one lesion to another.[11] The collision of two separate tumors seems unlikely in this case because there was no cystic capsule and lining of typical COC.

The COC has been accepted as benign odontogenic cyst since it was first described by Gorlin and associates [12] in 1962. In 1971 the WHO decided that this lesion should be classified as an odontogenic tumor.[13] The pathogenesis of COC remains unknown. Praetorius believes that it is not the well-defined separate entity that it seemed to be when it was first described. He has shown that at least four types of odontogenic tumor are known to occur in combination with the COC. These are the ameloblastoma, the odontoameloblastoma, the fibro-odontoma, and the complex odontome.[11] Shear [14] has started that it is an intriguing question whether those COCs which have features of the other odontogenic tumors and cysts develop those secondarily or whether they are themselves secondary phenomena in pre-existing odontogenic tumors or cysts.Altini and Farman [11] believed that the development of the COC components is a secondary event within the pre-existing odontogenic tumor.

Because in our case, the ghost cells associated with neoplastic epithelium were found in few areas and lesion did not show other features of COC, the histogenesis and pathogenesis are worth further investigation. Whether AF with ghost cell differentiation may behave differently from AF or COC alone and the prognosis for this kind of combined lesion is still unclear. Although the treatment for COC and AF is usually conservative, the treatment for reported AF with COC includes enucleation, excision, and resection. Long-term follow-up data and additional cases are still needed to clarify the clinical significance of these lesions.

 > Acknowledgement Top

We thank Department of Oral Surgery, SGT dental college Gurgaon for providing the clinical and radiographic details.

 > References Top

Barnes L, Eveson JW, Reichart P, Sidransky D. World Health Organization classification of tumors. Pathology and genetics of Head and Neck Tumors. Lyon: IARC Press; 2005. p. 208-309.  Back to cited text no. 1
Lin CC, Chen CH, Lin LM, Chen YK, Wright JM, Kessler HP, et al. Calcifying odontogenic cyst with ameloblatic fibroma: Report of three cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;98:451-60.  Back to cited text no. 2
Yoon JH, Kim HJ, Yook JI, Cha IH, Ellis GL, Kim J. Hybrid odontogenic tumor of calcifying odontogenic cyst and ameloblastic fibroma. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;98:80-4.  Back to cited text no. 3
Praetorius F, Hjorting-Hansen E, Gorlin RJ, Vickers RA. Calcifying odontogenic cyst. Range, variations and neoplastic potential. Acta Odontol Scand 1981;39:227-40.  Back to cited text no. 4
Farman AG, Smith SN, Nortje CJ, Grotepass FW. Calcifying odontogenic cyst with ameloblastic fibro-odontoma: One lesion or two? J Oral Pathol 1978;7:19-27.  Back to cited text no. 5
Moleri AB, Moreira LC, Carvalho JJ. Comparative morphology of 7 New cases of calcifying odontogenic cyst. J Oral Maxillofac Surg 2002;60:689-96.  Back to cited text no. 6
Han PP, Nagatsuka H, Siar CH, Tsujiqiwa H, Gunduz M, Tamamura R, et al. A pigmented calcifying cystic odontogenic tumor associated with compound odontoma: A case report and review of literature. Head Face Med 2007;3:35.  Back to cited text no. 7
Matsuzaka K, Inoue T, Nashimoto M, Takemoto K, IshiKawa H, Asaka M, et al. A case of an ameloblastic fibro-odontoma arising from a calcifying odontogenic cyst. Bull Tokyo Dent Coll 2001;42:51-5.  Back to cited text no. 8
Ide F, Obara K, Mishima K, Saito I. Ameloblastoma ex calcifying odontogenic cyst (dentinogenic ghost cell tumor). J Oral Pathol Med 2005;34:511-2.  Back to cited text no. 9
Zeitoun IM, Dhanrajani PJ, Mosadomi HA. Adenomatoid odontogenic tumor arising in a calcifying odontogenic cyst. J Oral Maxillofac Surg 1996;54:634-7.  Back to cited text no. 10
Altini M, Farman AG. The calcifying odontogenic cyst. Eight new cases and a review of literature. Oral Surg Oral Med Oral Pathol 1975;40:751-9.  Back to cited text no. 11
Gorlin RJ, Pindborg JJ, Odont, Clausen FP, Vickers RA. The calcifying odontogenic cyst--A possible analogue of the cutaneous calcifying epithelioma of Malherbe. An analysis of fifteen cases. Oral Surg Oral Med Oral Pathol 1962;15:1235-43.  Back to cited text no. 12
Pindborg JJ, Kramer IR, Torlont H. Histological typing of odontogenic tumors, jaw cysts and allied lesions. Geneva, Switzerland: World Health Organization; 1971. p. 28-35.  Back to cited text no. 13
Shear M. Cyst of the oral regions. Bristol: John Wright and Sons; 1976. p. 59-66  Back to cited text no. 14


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

  [Table 1]


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