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CASE REPORT
Year : 2020  |  Volume : 16  |  Issue : 3  |  Page : 661-664

Exophytic gingival growth of the maxillary canine region in a young individual: Extremely rare case report of peripheral dentinogenic ghost cell tumor


1 Department of Periodontics, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh, India
2 Department of Oral Pathology, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh, India
3 Department of Oral Surgery, Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi, Uttar Pradesh, India

Date of Submission16-Mar-2019
Date of Acceptance18-Oct-2019
Date of Web Publication11-Jun-2020

Correspondence Address:
Monika Bansal
Faculty of Dental Sciences, Institute of Medical Sciences, Banaras Hindu University, Varanasi - 221 005, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jcrt.JCRT_180_19

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 > Abstract 


The present case report is a case of peripheral dentinogenic ghost cell tumor (PDGCT), an extremely rare solid benign neoplastic variant of calcifying cystic odontogenic tumor of the gingiva mimicking clinically as pyogenic granuloma, fibroma, peripheral ossifying fibroma, and peripheral giant-cell granuloma. A 24-year-old male reported with painless, firm, solitary, sessile, smooth-surfaced, nonulcerative, nonpulsatile, well-defined swelling measuring ≈12 mm × 9 mm in the interdental gingiva of the teeth #13 and #14 extending to the mucogingival junction. Intraoral periapical radiographic showed a normal trabecular pattern with mild radiolucency without bony expansion, periapical lesion, and resorption of the adjacent teeth. The diagnosis was established by histopathologic examination. Very few cases of this entity have been documented in the literature. The present case report aims to document this rare entity and emphasizes on the fact that histopathological examination of every localized gingival growth should be included in the treatment planning to differentiate with other commonly found lesions.

Keywords: Calcifying odontogenic cyst, gingiva, odontogenic tumors, peripheral dentinogenic ghost cell tumor


How to cite this article:
Nahid R, Bansal M, Gupta K, Pandey S, Tiwari P, Agarwal R. Exophytic gingival growth of the maxillary canine region in a young individual: Extremely rare case report of peripheral dentinogenic ghost cell tumor. J Can Res Ther 2020;16:661-4

How to cite this URL:
Nahid R, Bansal M, Gupta K, Pandey S, Tiwari P, Agarwal R. Exophytic gingival growth of the maxillary canine region in a young individual: Extremely rare case report of peripheral dentinogenic ghost cell tumor. J Can Res Ther [serial online] 2020 [cited 2020 Aug 3];16:661-4. Available from: http://www.cancerjournal.net/text.asp?2020/16/3/661/286539




 > Introduction Top


Dentinogenic ghost cell tumor (DGCT) is an extremely rare solid benign neoplastic variant of calcifying cystic odontogenic tumor, first identified by Praetorius et al. 1981[1] and which has been reported for the first time in the literature in 1972 under the name “calcifying ghost cell odontogenic tumor” by Fejerskov and Krogh.[2] de Arruda et al. reviewed DGCTs and reported only 15 cases of peripheral or extraosseous in the gingiva or alveolar mucosa among a total of 55 cases of DGCT till date.[3] Hereby, we contribute the 16th case of peripheral DGCTs to the literature. The present case report aims to document an extremely rare case presenting on gingival tissue resembling clinically with the other reactive or inflammatory lesions that may lead to misdiagnosis and emphasizes the need for histological examination of every lesion.


 > Case Report Top


A 24-year-old male patient presented with a chief complaint of a lump in the upper right anterior region. The patient gave a history of painless slow-growing swelling over a period of 3 years without any treatment. Medical and family histories were noncontributory. The patient visited dentist 1 month back for extraction of #46. Extraoral examination revealed no lymphadenopathy and no temporomandibular joint abnormality, but facial asymmetry was present due to the presence of swelling in the right side.

Intraoral examination showed an oval, nonulcerative, solitary, sessile, smooth-surfaced swelling with confluent margins in the interdental gingiva of teeth #13 and #14 approaching to the mucogingival junction. The swelling was reddish-pink similar to the neighboring area measuring ≈12 mm × 9 mm [Figure 1]. On palpation, the swelling was firm, nontender, nonfluctuant, and nonpulsatile in nature. Adjacent teeth did not show mobility or displacement, but teeth #13 and #14 have probing depth of 6 mm and 7 mm only on the distobuccal and mesiobuccal surfaces, respectively. An intraoral periapical radiograph showed a normal trabecular pattern with mild radiolucency in the interdental area of related teeth without bony expansion, periapical lesion, and resorption of the adjacent teeth [Figure 2].
Figure 1: Intra-oral photograph showing an oval, nonulcerative, solitary, sessile, smooth-surfaced well-defined swelling with confluent margin in interdental gingiva of #13 and #14 teeth approaching to the mucogingival junction

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Figure 2: Intraoral periapical radiograph showing a normal trabecular pattern with mild radiolucency in the interdental area of teeth #13 and #14 with no bony expansion, no periapical lesion, and no resorption of the adjacent teeth

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Clinically and radiographically, differential diagnosis of pyogenic granuloma, fibroma, peripheral ossifying fibroma, and peripheral giant-cell granuloma was thought. Written informed consent was obtained from the patient. Wide local surgical excision was performed under local anesthesia because the lesion appeared more toward the benign nature. Erosion of the periosteum of the facial cortical plate was present. Mucoperiosteum flap was raised and mobilized from the adjacent area by giving periosteal incision and flap was sutured. Medications (antibiotics: amoxicillin (500 mg) and metronidazole (400 mg) three times daily for 7 days and analgesic: 50 mg diclofenac twice daily for 5 days) were prescribed. He was also instructed to rinse with 10 ml of chlorhexidine gluconate (0.2%) twice daily for 2 weeks. The patient was recalled after 1 week for suture removal. After 6-month follow-up, the site was healthy without any sign of local recurrence.

Excised tissue (≈12 mm × 8 mm) [Figure 3] was submitted for histopathologic examination and was stained with hematoxylin and eosin which showed parakeratinized stratified squamous epithelium of varying thickness overlying connective tissue stroma and the presence of odontogenic epithelium arranged in the form of island, strands, and cords (×4) [Figure 4]. Ghost cells were present in the central portion of tumor cells. Dentinoid material and calcifications were also seen scattered in the background of mild and generalized chronic inflammatory cells infiltration interspersed with dense bundles of collagen fibers (×10, ×40) [Figure 5] and [Figure 6]. Histopathological features were suggestive of peripheral DGCT (PDGCT).
Figure 3: Excised tissue measuring ≈12 mm × 8 mm submitted for histopathologic examination

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Figure 4: Peripheral dentinogenic ghost cell tumor (×4), hematoxylin and eosin stained sections showing overlying parakeratinized stratified squamous epithelium (A) and odontogenic epithelium arranged in the form of island, strands, and cords (B)

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Figure 5: Peripheral dentinogenic ghost cell tumor (×10) hematoxylin and eosin-stained sections showing odontogenic epithelium islands (A), ghost cells (B), and dentinoid material in the stroma (C)

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Figure 6: Peripheral dentinogenic ghost cell tumor (×40) hematoxylin and eosin-stained sections showing odontogenic epithelium islands (A), ghost cells (B), and dentinoid material in the stroma (C)

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 > Discussion Top


Calcifying odontogenic cyst (COC) comprises 1%–2% of all odontogenic cyst and tumors[4] and DGCTs, a type of solid variant of COC, are 11.5% among all COC.[5] PDGCTs are extremely rare and account for 13%–21% of all DGCTs.[6] Due to a dearth of documented cases, it is difficult to typify the exact location, age, and gender distribution of PDGCT. The demographics of current case are consistent with the previous reported cases except age. In the present case, the lesion was found in a young individual aged 24 years, whereas PDGCT has a predilection for older age group[7] that is contrary to our case.

Initially, inflammatory or reactive lesions, i.e., pyogenic granuloma, fibroma, peripheral ossifying fibroma, and peripheral giant-cell granuloma, were considered because clinically, the gingival lesion was asymptomatic, painless, slow-growing, sessile, firm, exophytic, and normal trabecular pattern with mild radiolucency was observed on radiograph. At the end, PDGCT was diagnosed by histologic examination due to the presence of odontogenic epithelium arranged in the form of island, strands, and cords. Ghost cells and dentinoid material were also present. These histologic and clinical features as well were in accordance with the reported findings of the WHO 2005[8] and Hong et al. 1991.[9] It has been reported that either radiographic change is not present in the peripheral tumors as in our case or saucerization of the bone may be present in ≈20%.[10]

Histopathological differential diagnoses considered for the present lesion were peripheral odontogenic fibroma (OF), peripheral ameloblastoma, and peripheral ameloblastic fibrodentinoma. Fibroblastic proliferation containing inactive odontogenic epithelial rests, with or without varying amounts of calcification, was the main histopathologic finding of OF. Peripheral ameloblastoma was excluded due to the presence of dentinoid or calcifications, as it does not show inductive changes in the form of dentinoid or calcifications, while peripheral ameloblastic fibroma was excluded due to the lack of dental papilla-like stroma. Thus, the lesion was diagnosed as a PDGCT, as it contains active odontogenic epithelium, ghost cells, and dysplastic dentin.[8] Due to the histological diversity, the origin of this lesion is suggested to be from the cell rest of Serres (remnants of dental lamina) or of surface epithelium.[8]

The lesion was surgically excised as the treatment of choice for PDGCTs includes conservative local resection with no recurrences postoperatively. However, long-term follow-up is suggested.


 > conclusion Top


PDGCT is an extremely rare case report of the gingiva mimicking clinically as pyogenic granuloma, fibroma, peripheral ossifying fibroma, and peripheral giant-cell granuloma. It emphasizes on histopathological examination of every localized gingival swelling to differentiate with other commonly found lesions. It should be bear in mind during the planning of treatment and prognosis of the lesion that this type of rare lesion may be encountered. Complete excision of the lesion should be recommended to prevent the chances of recurrence or metastatic transformation.

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 > References Top

1.
Praetorius F, Hjørting-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. 1
    
2.
Fejerskov O, Krogh J. The calcifying ghost cell odontogenic tumor-or the calcifying odontogenic cyst. J Oral Pathol 1972;1:273-87.  Back to cited text no. 2
    
3.
de Arruda JA, Monteiro JL, Abreu LG, de Oliveira Silva LV, Schuch LF, de Noronha MS, et al. Calcifying odontogenic cyst, dentinogenic ghost cell tumor, and ghost cell odontogenic carcinoma: A systematic review. J Oral Pathol Med 2018;47:721-30.  Back to cited text no. 3
    
4.
Günhan O, Erseven G, Ruacan S, Celasun B, Aydintug Y, Ergun E, et al. Odontogenic tumours. A series of 409 cases. Aust Dent J 1990;35:518-22.  Back to cited text no. 4
    
5.
Günhan O, Mocan A, Can C, Kişnişci R, Aksu AY, Finci R. Epithelial odontogenic ghost cell tumor: Report of a peripheral solid variant and review of the literature. Ann Dent 1991;50:8-11, 48.  Back to cited text no. 5
    
6.
Candido GA, Viana KA, Watanabe S, Vencio EF. Peripheral dentinogenic ghost cell tumor: A case report and review of the literature. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2009;108:e86-90.  Back to cited text no. 6
    
7.
McClatchey KD, Stewart JC, Patterson BD. Dentinogenic ghost cell tumor presenting as a gingival mass. Ann Dent 1988;47:31-2.  Back to cited text no. 7
    
8.
Philipsen HP, Riechart PA, Sciubba JJ, van der Waal I. Odontogenic tumors. In: Barnes L, Eveson JW, Reichart P, Sidransky D, editors. Head and Neck Tumours: Pathology and Genetics: World Health Organization Classification of Tumours. Lyon, France: IARC Press; 2005. p. 283-328.  Back to cited text no. 8
    
9.
Hong SP, Ellis GL, Hartman KS. Calcifying odontogenic cyst. A review of ninety-two cases with reevaluation of their nature as cysts or neoplasms, the nature of ghost cells, and subclassification. Oral Surg Oral Med Oral Pathol 1991;72:56-64.  Back to cited text no. 9
    
10.
Iezzi G, Rubini C, Fioroni M, Piattelli A. Peripheral dentinogenic ghost cell tumor of the gingiva. J Periodontol 2007;78:1635-8.  Back to cited text no. 10
    


    Figures

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



 

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