Case Report Dentinogenic Ghost Cell Tumor of the Peripheral Variant Mimicking Epulis

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1 Interntionl Dentistry Volume 2010, Article ID , 5 pges doi: /2010/ Cse Report Dentinogenic Ghost Cell Tumor of the Peripherl Vrint Mimicking Epulis Uddipn Kumr, 1 Hitesh Vij, 2 Ruchiek Vij, 2 Jitin Khrnd, 3 IN Aprn, 4 nd Rghu Rdhkrishnn 5 1 Deprtment of Orl Pthology nd Microiology, Hzrig College of Dentl Sciences nd Hospitl, Hzrig , Jhrkhnd, Indi 2 Deprtment of Orl Pthology, Institute of Dentl Studies nd Technologies, Modingr, Uttr Prdesh , Indi 3 Deprtment of Orl Pthology, Shree Bnkey Bihri Dentl College, Msuri, Ghzid , Indi 4 Deprtment of Prosthodontics, Mnipl College of Dentl Sciences, Mnipl University, Mnipl , Indi 5 Deprtment of Orl Pthology nd Microiology, Mnipl College of Dentl Sciences, Mnipl University, Mnipl , Indi Correspondence should e ddressed to Rghu Rdhkrishnn, rghu.rdhkrishnn@gmil.com Received 29 August 2010; Accepted 10 Octoer 2010 Acdemic Editor: Ahmd Wseem Copyright 2010 Uddipn Kumr et l. This is n open ccess rticle distriuted under the Cretive Commons Attriution License, which permits unrestricted use, distriution, nd reproduction in ny medium, provided the originl work is properly cited. Dentinogenic ghost cell tumor (DGCT) is n uncommon loclly invsive odontogenic tumor regrded y mny s vrint of clcifying odontogenic cyst. The peripherl vrint of this clinicl rrity ppers s well-circumscried mss mimicking nonspecific gingivl enlrgement. Microscopic ppernce of odontogenic epithelium dmixed with focl res of dentinoid formtion nd sheets of ghost cells giving the definitive dignosis of dentinogenic ghost cell tumor imply tht microscopic exmintion is compulsory for ny gingivl mss. Vn Gieson histochemicl stin further confirmed the nture of dentinoidlike mteril. A complete workup of cse of peripherl dentinogenic ghost cell tumor is presented in this pper nd the current concept s well s the pprisl of literture is presented. 1. Introduction Clcifying odontogenic cyst (COC) is unique jw lesion, first recognized s distinct entity y Gorlin et l. [1] nd hence the eponym Gorlin cyst. Pretorius et l. [2] clssified them into the cystic type (Type I) nd the solid type (Type II). The solid vrint of COC (Type II) is rre nd is designted s dentinogenic ghost cell tumor (DGCT), lthough the first description of the solid vrint ws given y Fejerskov nd Krogh s clcifying ghost cell odontogenic tumor [3]. DGCT is chrcterized microscopiclly y melolstom-like odontogenic epithelil prolifertion, presence of ghost cells, nd dentinoid-like mteril [4]. Due to its diverse histologicl picture, severl terms hve een used y different uthors to descrie this lesion such s dentinogenic ghost cell tumor [2], clcifying ghost cell odontogenic tumor [3], kertinizing melolstom [5], cystic clcifying odontogenic tumor [6], peripherl odontogenic tumor with ghost cell kertiniztion [7], dentinomelolstom [8], melolstic dentinom [9], epithelil odontogenic ghost cell tumour [10], nd odontogenic ghost cell tumor [11]. The term DGCT is commonly used, nd the peripherl vrint of this neoplstic entity is rre; only few reports with clinicl, rdiogrphic, nd histologic documenttion cn e found in the English literture. A report of peripherl dentinogenic ghost cell tumor (PDGCT) nd chrcteriztion of dentinoid mteril using Vn Gieson specil stin for the confirmtion dds new dimension to the dignosis of DGCT. 2. Cse Report A 40-yer-old mle ptient reported to the dentl clinics t Mnipl College of Dentl Sciences, Mnipl University, with complint of missing teeth. Clinicl exmintion disclosed swelling mesuring out 5 mm in dimeter in the region

2 2 Interntionl Dentistry c d Figure 2: Solid wellcircumscried encpsulted mss () showing melolstomtous islnds of odontogenic epithelium () nd ghost cells (c) with overlying epithelium (d) [Hemtoxylin nd Eosin, 4]. Figure 1: A nontender, persistent swelling in the region of lower left posterior region. of lower left premolrs which ws non tender, nd the over lying mucos ws norml in color. Cliniclly, provisionl dignosis of epulis ws mde (Figure 1). The lesion ws excised under locl nesthesi, nd the tissue ws sumitted for histopthologicl exmintion. Six months of followup exmintion did not show ny signs of recurrence. Histologicl exmintion reveled solid well-circumscried, encpsulted soft tissue mss surrounded y dense firous connective tissue covered y strtified squmous epithelium (Figure 2). The tumor mss reveled islnds of odontogenic epithelium resemling follicles of melolstom, consisting of columnr cells enclosing stellte reticulum like cells (Figure 3). These elements were ssocited with numerous ple, eosinophilic ghost cells with grnulr eosinophilic cytoplsm nd fint nucler outline (Figure 4). Few multinucleted gint cells of foreign ody type were evident t the periphery of the ghost cells in the connective tissue strom. Irregulr foci of tissue resemling dentin were oserved surrounding the odontogenic epithelil islnds, nd these res were tuulr nd t plces showed cellulr inclusions (Figure 5). Vn Gieson specil stin ws crried out to exmine the nture of the dentinoid-like mteril (Figure 6). The chrcteristic microscopic fetures nd the confirmtion of dentinoid-like mteril y specil stin contriuted to the dignosis of dentinogenic ghost cell tumor of the peripherl vrint. Figure 3: Amelolstomtous islnd of odontogenic epithelium with columnr sl cells () enclosing stellte reticulum like cells () [Hemtoxylin nd Eosin, 40]. 3. Discussion Figure 4: Ghost cells () nd surrounding dentinoid-like mteril () [Hemtoxylin nd Eosin, 20]. DGCT is distinct ut rre histologicl entity mong odontogenic ghost cell lesions which hve een recently clssified into the simple cystic type or COC; cysts ssocited with odontogenic hmrtoms or enign neoplsms; solid enign odontogenic neoplsm, which is sme s COC ut with dentinoid formtion, the DGCT; mlignnt odontogenic neoplsm-ghost cell odontogenic crcinom [8]. The peripherl dentinogenic ghost cell tumor (PDGCT), though distinct entity of odontogenic origin, is pprently rre. This rrity is due to the filure of its recognition s n isolted entity [12], nd mny of the cses of PDGCT hve een mistkenly dignosed s peripherl melolstom [13].

3 Interntionl Dentistry 3 Sl no. Reference Tle 1: Reported cses of peripherl dentinogenic ghost cell tumor. No. of cses Age in yers/sex Site Rdiogrphic fetures 1 Arms nd Howell [14] 1 13/M Extrosseous No significnt findings 2 Suk [15] 1 67/F Extrosseous No rdiogrphic chnges 3 Pretorius et l. [2] 2 52/M Mxillry left lterl incisor nd cnine region Slight erosion of underlying one 41/F Mndiulr nterior region Slight erosion of underlying one 4 Hirsherg et l. [16] 1 42/M Mndiulr left premolr region (lingul gingiv) No one involvement 5 McCltchey etl. [17] 1 57/M Mndiulr nterior region No one involvement 10/M Mndiulr centrl incisor 6 Buchner et l. [12] 3 53/F Mndiulr edentulous ridge 92/F Mndiulr edentulous cuspid-premolr region 7 Günhn et l. [18] 1 71/F Mxillry nterior region Slight erosion of underlying one 8 Ruenheimer et l. [19] 1 82/M Mndiulr right lveolr ridge (edentulous) No one involvement 9 Cstro et l. [20] 1 83/F Anterior ridge of edentulous mndile Cup shped resorption 10 Wong et l. [9] 1 71/M Mxillry cnine region Slight erosion eneth the growth 11 Iezzi et l. [21] 1 43/M Mxillry cnine region No one involvement 12 Ledesm-Montes et l. [22] 1 NS NS NS 13 Cndido et l. [23] 1 45/M Mndiulr cnine region No one involvement 14 Our cse 1 40/M Mndiulr premolr re NS: Not Specified. c Figure 5: Irregulr foci of dentine/osteo-dentine-like mteril () nd clcifying ghost cells () nd gint cells (c) [Hemtoxylin nd Eosin, 40]. Figure 6: Vn Gieson stins showing ghost cells stining yellow () nd surrounding dentinoid-like mteril stining pink () [Vn Gieson stin, 40]. The usul presenttion of the peripherl vrint is nodulr swelling on the edentulous lveolr mucos of denture werers, feture tht implictes trum/irrittion. This clinicl presenttion could led to the provisionl dignosis of epulis s ws true in our cse s well. Due to the derth of documented cses, it is difficult to typify the exct loction, ge, nd gender distriution of PDGCT. However sed on the existing literture it ppers to e more common in the cnine-premolr region, with predilection for the elderly ge group [2, 9, 12, 14 23] (Tle 1). Hirsherg et l. [16] indicted tht the tumor minly ffects the mxill nd occurs four times more commonly mong men. Contrry to this, Hong et l. [24] reported tht it minly occurs in the mndiulr edentulous mucos without ny sex predilection. This lesion seems to e less ggressive thn its centrl counterprt with no recurrences reported fter excision [12]. Bsed on this histologicl diversity, the origin of this lesion hs een ttriuted to cell rests of Serres or the surfce epithelium [25, 26]. The tumor is minly composed

4 4 Interntionl Dentistry of melolstom-like res of odontogenic epithelil islnds with vrying mount of ghost cells showing kertiniztion nd clcifiction. It my histologiclly show res similr to or my e ssocited with odontogenic tumors like complex nd compound odontomes, melolstic firoodontom, nd so forth. Ghost cells re thought to e trnsformed odontogenic epithelil cells, the mechnism of whose trnsformtion remins unknown [8]. In light microscope, they pper s enlrged, ovoid, or elliptoid epithelil cells, which re eosinophilic, usully with welldefined cell outlines ut my e lurred giving them fused ppernce. Histochemiclly, they re positive for kertin giving yellow fluorescence with Rhodmine B. Sometimes ghost cells my contin nucler remnnts in vrious stges of degenertion. The clcifiction which my occur in some of these ghost cells pper initilly s fine powdery/corse sophilic grnules nd lter s smll sphericl odies. Ultrstructurl studies hve shown tht these clcifictions represent dystrophic clcifiction [27]. Due to its iologicl ehvior COC, nd its vrints re thought to resemle enign odontogenic tumor rther thn cyst[6]. Previously, Arms nd Howell [14] hve estlished tht the term cyst does not imply in ll instnces s its clinicl fetures, growth ehvior, nd recurrence potentil re quite similr to those of melolstom. It hs lso een emphsized tht clcifictions found in COC nd CEOT re not feture of melolstom nd the comined occurrence of COC with other odontogenic lesions could e due to the multipotentility of odontogenic epithelium [4]. Further it is pointed out tht this lesion in ssocition with n odontom my result due to differentition nd degenertion of odontogenic epithelium [14]. Wht ppers s ssocited odontogenic tumor should e considered s n integrl prt of n entire lesion developing in the wll of COC [2]. In our cse s well, the presence of kertinizing ghost cells nd minerliztion ws n outstnding feture. The lesion did not exhiit ny cystic res, nd its fetures were consistent with those of DGCT. The clcifictions seen s dentoid, frequently descried in connection with msses of ghost cells, were chrcteristic finding of this lesion. These clcified res were thought to represent juxtepithelil osteoid formtion y grnultion tissue due to induction y ghost cells [14]. This view ws however negted lter s juxtepithelil osteoid nd dentinoid were oserved in res free of grnultion tissue nd ghost cells, s pointed outytjimetl.[28]. There re no reports or studies to clrify if this is n inductive effect or metplstic chnge in the connective tissue. In the present cse s well, the clcified mteril ws found either in res where the ghost cells nd connective tissue were in contct or in the connective tissue djcent or elow the sl cells, possily due to the inductive effects of ghost cells/epithelil cells [29]. Vn Gieson specil stin illustrted the collgenous nture of the dentinoid-like mteril. The histologicl nd histochemicl ttriutes of the dentinoid-like clcifiction descried in this peripherl odontogenic tumor est fitted with the dignosis of Peripherl Dentinogenic Ghost Cell Tumor. Conservtive ut ggressive locl resection hs een the tretment of choice with no recurrences found in ny ptient. However ptients with DGCT should remin in longterm follow-up [30]. Though numerous terminologies hve een suggested for this entity, we propose tht for lesion with the ove histologicl nd histochemicl chrcteristics, Peripherl Dentinogenic Ghost Cell Tumor est descries it nd should e the most preferred designtion. Arevitions DGCT: Dentinogenic ghost cell tumor COC: Clcifying odontogenic cyst PDGCT: Peripherl dentinogenic ghost cell tumor. References [1] R. J. Gorlin, J. J. Pindorg, F. P. Clusen, nd R. A. Vickers, The clcifying odontogenic cyst- possile nlogue of the cutneous clcifying epitheliom of Mlhere. An nlysis of fifteen cses, Orl Surgery, Orl Medicine, Orl Pthology, vol. 15, no. 10, pp , [2] F. Pretorius, E. Hjorting-Hnsen, R. J. Gorlin, nd R. A. Vickers, Clcifying odontogenic cyst. Rnge, vritions nd neoplstic potentil, Act Odontologic Scndinvic, vol. 39, no. 4, pp , [3] O. Fejerskov nd J. Krogh, The clcifying ghost cell odontogenic tumor or the clcifying odontogenic cyst, Orl Pthology, vol. 1, no. 6, pp , [4] Y. Tjim, S. Yokose, E. Skmoto, Y. Ymmoto, nd N. Utsumi, Amelolstom rising in clcifying odontogenic cyst: report of cse, Orl Surgery Orl Medicine nd Orl Pthology, vol. 74, no. 6, pp , [5] S. N. Bhskr, Orl surgery-orl pthology conference no. 13, Wlter Reed Army Medicl Center, Orl Surgery, Orl Medicine, Orl Pthology, vol. 19, no. 6, pp , [6] P. D. Freedmn, H. Lumermn, nd J. K. Gee, Clcifying odontogenic cyst. A review nd nlysis of seventy cses, Orl Surgery Orl Medicine nd Orl Pthology, vol. 40, no. 1, pp , [7] J. C. Vuletin, M. P. Solomon, nd L. P. Pertschuk, Peripherl odontogenic tumor with ghost-cell kertiniztion. A histologic, fluorescent microscopic, nd ultrstructurl study, Orl Surgery Orl Medicine nd Orl Pthology, vol. 45, no. 3, pp , [8] M. Sher nd P. Speight, Cysts of the Orl nd Mxillofcil Regions, Blckwell, Munksgrd, Dnmrk, 4th edition, [9]Y.K.Wong,S.C.Chiu,S.W.Png,ndJ.C.F.Cheng, Peripherl dentinogenic ghost cell tumour presenting s gingivl mss, British Orl nd Mxillofcil Surgery, vol. 42, no. 2, pp , [10] G. L. Ellis nd B. M. Shmookler, Aggressive (mlignnt?) epithelil odontogenic ghost cell tumor, Orl Surgery, Orl Medicine, Orl Pthology, vol. 61, no. 5, pp , [11] G. L. Ellis, Odontogenic ghost cell tumor, Seminrs in Dignostic Pthology, vol. 16, no. 4, pp , [12] A. Buchner, P. W. Merrell, L. S. Hnsen, nd A. S. Leider, Peripherl (extrosseous) clcifying odontogenic cyst. A review of forty-five cses, Orl Surgery Orl Medicine nd Orl Pthology, vol. 72, no. 1, pp , 1991.

5 Interntionl Dentistry 5 [13] T. Ngo, T. Nkjim, M. Fukushim, nd T. Ishiki, Alcifying odontogenic cyst: survey of 23 cses in the Jpnese literture, Mxillofcil Surgery, vol. 11, no. 4, pp , [14] A. M. Arms nd F. V. Howell, The clcifying odontogenic cyst. Report of four cses, Orl Surgery, Orl Medicine, Orl Pthology, vol. 25, no. 4, pp , [15] J. J. Suk Jr., Clcifying nd kertinizing odontogenic cyst, JournlofOrlSurgery, vol. 30, no. 12, pp , [16] A. Hirsherg, D. Dyn, nd I. Horowitz, Dentinogenic ghost cell tumor, Interntionl Orl nd Mxillofcil Surgery, vol. 16, no. 5, pp , [17] K. D. McCltchey, J. C. Stewrt, nd B. D. Ptterson, Dentinogenic ghost cell tumor presenting s gingivl mss, Annls of dentistry, vol. 47, no. 1, pp , [18] O. Günhn, A. Mocn, C. Cn, R. Kişnişci, A. Y. Aksu, nd R. Finci, Epithelil odontogenic ghost cell tumor: report of peripherl solid vrint nd review of the literture, Annls of dentistry, vol. 50, no. 2, pp. 8 48, [19] E. J. Ruenheimer, W. F. P. Vn Heerden, F. Sitzmnn, nd B. Heymer, Peripherl dentinogenic ghost cell tumor, Journl of Orl Pthology nd Medicine, vol. 21, no. 2, pp , [20]W.H.Cstro,M.C.FerreirDeAguir,ndR.S.Gomez, Peripherl dentinogenic ghost-cell tumor: cse report, Quintessence Interntionl, vol. 28, no. 1, pp , [21] G. Iezzi, C. Ruini, M. Fioroni, nd A. Pittelli, Peripherl dentinogenic ghost cell tumor of the gingiv, Periodontology, vol. 78, no. 8, pp , [22] C. Ledesm-Montes, R. J. Gorlin, M. Sher et l., Interntionl collortive study on ghost cell odontogenic tumours: clcifying cystic odontogenic tumour, dentinogenic ghost cell tumour nd ghost cell odontogenic crcinom, Orl Pthology nd Medicine, vol. 37, no. 5, pp , [23] G. A. Cndido, K. A. Vin, S. Wtne, nd E. F. Vencio, Peripherl dentinogenic ghost cell tumor: cse report nd review of the literture, Orl Surgery, Orl Medicine, Orl Pthology, Orl Rdiology nd Endodontology, vol. 108, no. 3, pp. e86 e90, [24] S. P. Hong, G. L. Ellis, nd K. S. Hrtmn, Clcifying odontogenic cyst. A review of ninety-two cses with reevlution of their nture s cysts or neoplsms, the nture of ghost cells, nd suclssifiction, Orl Surgery Orl Medicine nd Orl Pthology, vol. 72, no. 1, pp , [25] A. Buchner nd L. S. Hnsen, The histomorphologic spectrum of the gingivl cyst in the dult, Orl Surgery Orl Medicine nd Orl Pthology, vol. 48, no. 6, pp , [26] M. Altini nd A. G. Frmn, The clcifying odontogenic cyst. Eight new cses nd review of the literture, Orl Surgery Orl Medicine nd Orl Pthology, vol. 40, no. 6, pp , [27] J. P. Spp nd D. G. Grdner, An ultrstructurl study of the clcifictions in clcifying odontogenic cysts nd odontoms, Orl Surgery Orl Medicine nd Orl Pthology, vol. 44, no. 5, pp , [28] Y. Tjim, J. Ohno, nd N. Utsumi, The dentinogenic ghost cell tumor, Orl Pthology, vol. 15, no. 6, pp , [29] M. Junej nd J. George, Dentinogenic ghost cell tumor: cse report nd review of the literture, Orl Surgery, Orl Medicine, Orl Pthology, Orl Rdiology nd Endodontology, vol. 107, no. 5, pp. e17 e22, [30] G. Sun, X. Hung, Q. Hu, X. Yng, nd E. Tng, The dignosis nd tretment of dentinogenic ghost cell tumor, Interntionl JournlofOrlndMxillofcilSurgery, vol. 38, no. 11, pp , 2009.

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