Hyperplastic dental follicle or peri-follicle fibrosis, is. Hyperplastic Dental Follicle Review of Literature and Report of Two Cases in One Family

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1 Puliction Hyperplstic Dentl Follicle Review of Literture nd Report of Two Cses in One Fmily Chun Xio SUN 1, Crig RIRIE 1, Jeffrey M HENKIN 1 Hyperplstic dentl follicle is n extremely rre lesion. The prctitioner should e le to differentite it from dentigerous cyst. The present rticle will review relted literture nd report on two cses in one fmily. A 12-yer-old white femle nd her 15-yer-old rother were referred exposure of unerupted cnines. No systemic diseses or syndromes were present. Intr-orl exmintions were unremrkle, except the sence of ll eight cnines. Rdiogrphic exmintions reveled impcted cnines with ech unerupted tooth surrounded well-demrcted rdiolucency, which pssed eyond the cementoenmel junction. The teeth were surgiclly exposed nd tissue specimens surrounding the unerupted teeth were nlysed histologiclly. Histology reveled firous connective tissue with res demonstrting some ground sustnce nd multiple odontogenic epithelil rests. Some surfces were prtilly lined reduced enmel epithelium. A dignosis of hyperplstic dentl follicle ws mde. Sometimes, it is difficult to differentite hyperplstic dentl follicle from odontogenic firom, oth simple nd centrl ms. A correct dignosis should e sed on clinicl, rdiogrphic nd histologicl findings. Key words: hyperplstic dentl follicle, unerupted cnine Hyperplstic dentl follicle or peri-follicle firosis, is n unusul lesion often confused with odontogenic firom 1,2. Grdner ws the first one who ttempted to clrify these similr lesions 1. He pointed out tht three different lesions my resemle ech other. The first one is hyperplstic dentl follicle, which is minly composed of firous connective tissue nd wvy collgen fires. It my contin strnds nd rests of odontogenic epithelium. Clcified mterils my e present in the connective tissue, which is then clled clcifying hyperplstic dentl follicle, nd ws first descried Sndler et l 2. 1 Deprtment of Periodontics, Lom Lind University School of Dentistry, Lom Lind, CA, USA. Corresponding uthor: Dr Jeffrey M. Henkin, Advnced Eduction Progrm in Periodontics nd Implnt Surgery, Lom Lind University School of Dentistry, Tylor St., Lom Lind, CA 92354, USA. Tel: ; Fx: E-mil: jhenkin@llu.edu The second m of the lesion hs een clled simple odontogenic firom. This lesion resemles hyperplstic dentl follicle histologiclly, ut differs in its size nd loction. The simple odontogenic firom is usully much lrger thn hyperplstic dentl follicle, nd is invrily ssocited with crowns of unerupted teeth, which is not lwys the cse with odontogenic firom. The third lesion hs een nmed centrl odontogenic firom (WHO type). This lesion hs some unique histologicl fetures such s dysplstic dentin, cementumlike clcified tissues nd mrked epithelil prolifertion. The lst two lesions re considered true neoplsms, wheres the hyperplstic dentl follicle is considered hmrtomtous lesion. Multiple hyperplstic dentl follicles is n extremely rre condition. To dte, there re only six cses reported in the literture (Tle 1) 2-6. None of these reported ny symptoms, nd they minly ffect unerupted posterior teeth, which ccounts 30 out of 36 teeth involved. All The Chinese Journl of Dentl Reserch 71

2 Puliction Tle 1 Reported cses of multiple hyperplstic dentl follicles. Authors Ptients Teeth ffected Defective ffected teeth Sun, et l (current report) 15-yer-old white mle 6, 11, 22, 27 mentioned Clcifiction No 12-yer-old white femle 6, 11, 22, 27 Wlker, et l (reference 6)* 6-yer-old femle T mentioned Foci of irregulr clcifiction Gomez, et l (reference 4) 15-yer-old mle 1, 2, 5, 6, 7, 11, 12, 15, 22, 27, 32 Unremrkle Type A nd B clcifiction in firous soft tissue nd dystrophic clcifiction in the pulp tissue nd dentine mtrix Grdner, et l (reference 5) 26-yer-old white mle 17, yer-old mle 7 teeth mentioned Type A clcifiction dominnt Lukinm, et l (reference 3) 24-yer-old mle 18, 20 Hypoplstic enmel nd defective crowns nd roots Bsophilic, concentric, clcified odies, type A clcifiction Sndler, et l (reference 2) 15-yer-old lck mle 1, 2, 13, 15, 16, 17, 18, 20, 21, 28, 29, 31, 32 Unremrkle Aundnt clcified mterils in the whorled res of the connective tissue nd in the hylinised tissue surrounding the odontogenic epithelium * The dignosis ws hyperplstic dentl follicle with n incipient denomtoid odontogenic tumour Fig 1 Pnormic rdiogrphs of the 12-yer-old femle ptient 6 months fter tooth 13 extrction. Rdiolucency round ech unerupted cnine ws well demrcted. Fig 2 Rdiogrphs showed rdiolucencies pssed eyond the cementoenmel junction (CEJ). Peripicl rdiogrphs of teeth 22 (right) nd 27 (left) re shown. Rdiolucencies pssed eyond the CEJ nd no ovious resorption ws noticed on djcent teeth. 72 Volume 13, Numer 1, 2010

3 of these cses presented with clcifiction. In the present report, the uthors present two cses; ech hs four unerupted cnines ssocited with non-clcified hyperplstic dentl follicles, nd occur in the sme fmily. Puliction Cse reports In Jnury 2009, 12-yer-old Cucsin femle ws referred to the Deprtment of Periodontics, Lom Lind University School of Dentistry exposure of unerupted teeth 6, 11, 22 nd 27. The ptient hd unerupted tooth 13 extrcted n orl surgeon 6 months prior to the present consulttion. There were no ssocited systemic diseses or syndromes present. Introrl exmintion reveled tht teeth 6, 11, 22 nd 27 were not cliniclly present, nd the ptient ws undergoing orthodontic tretment. Swelling on the uccl sides of teeth 22 nd 27, depressile on plption, were detected. A pnormic rdiogrph showed uniloculr, well-circumscried rdiolucency round the crowns of ech impcted tooth (Fig 1). Teeth 1 nd 16 were still ming while teeth 17 nd 32 ppered to e congenitlly missing. Peripicl rdiogrphs (Fig 2) nd cone em 3-D imges (dt not shown) showed tht the rdiolucencies were eyond the cementoenmel junction. Impcted teeth (6, 11, 22 nd 27) were surgiclly exposed vi closed flp technique to preserve kertinised tissue, nd undnt dense firotic tissues were found surrounding the impcted teeth, extending into the res of the roots (Fig 3). The tissues round the impcted cuspids were collected nd sent to privte lortory pthologicl nlysis. Under light microscopy, the pthologist found considerle ground sustnce nd multiple odontogenic epithelil rests, with some exhiiting juxt-epithelil hylinistion. The histologicl dignosis ws hyperplstic dentl follicles (peri-folliculr firosis) without clcifiction (Fig 4). Three months lter, the ptient s 15-yer-old rother ws referred to the deprtment of periodontics the sme reson. This ptient hd lmost identicl clinicl findings s his younger sister, except tht ll of his third molrs were ming. Following his surgery, the pthologicl dignosis ws lso reported s hyperplstic dentl follicle (Fig 5). Discussion The most frequently impcted teeth re mndiulr third molrs, followed mxillry cnines, mndiulr cnines, premolrs nd incisors. The incidence of mxillry cnine impction rnges from 1 to 3%, nd pltl impction is more often oserved thn lil impction c Fig 3 Exposure of impcted tooth 27. A closed flp technique ws used: flp ws elevted (), outer lyer of one ws removed nd hyperplstic dentl follicle ws seen surrounding the impcted tooth (), surrounding tissue ws removed nd tooth exposed (c), gold chin ws onded onto exposed tooth surfce (d), flp closed nd 1 week postopertive photo (e nd f, respectively). The Chinese Journl of Dentl Reserch 73

4 d e Puliction (the rtio is pproximtely 2:1 to 6.6:1.) 7,8. The etiology of tooth impction is still not very cler. Possile primry cuses re the rte of root resorption of deciduous teeth, trum of the deciduous tooth ud, disturnces in tooth eruption sequence, vilility of spce in the rch, rottion of tooth uds, premture root closure nd cnine eruption into the cleft re in persons with cleft plte 9. The proposed secondry cuses re norml muscle pressure, ferile disese, endocrine disturnces nd vitmin D deficiency 9. The norml pericoronl rdiolucency is considered to e in the rnge of 2 to 3 mm 10, nd there is no definitive explntion of the mechnism cusing hyperplstic dentl follicle. Kim et l reported tht the expression of severl mtrix metlloproteinses is down-regulted in hyperplstic dentl follicles; moreover, severl collgen genes re up-regulted 11. Previous reports hve descried only mle ptients (Tle 1). The present report is the first cse report involving permnent teeth in femle ptient. This is lso the first report of hyperplstic dentl follicle involved in only cnines. To the uthors knowledge, this report is lso the first one to report two cses in silings, which suggests tht the condition my hve genetic predilection. Tooth impction cused hyperplstic dentl follicle my e ssocited with defective tooth mtion 3, nd it lso my e ssocited with other symptoms, such s melogenesis imperfect, rough hypoplstic type nd gingivl hyperplsi 12. It is very importnt dentists to recognise these possile ssocited lesions. Hyperplstic dentl follicle my involve just one tooth or my e present in multiple teeth. Sometimes, it is difficult to differentite hyperplstic dentl follicle from odontogenic tumours, especilly when clcifiction is present. Clcifiction ws estimted to e present in one-third of dentl follicles 13,14. The finl dignosis must consider the clinicl, rdiogrphic nd histologicl fetures of the lesion. f Acknowledgements The uthors thnk Dr Gretchen Folk, n orl pthologist, Scripps Orl Pthology Service, Sn Diego, CA, providing the photomicrogrphs. The uthors report no conflict of interest relted to the present cse report. 74 Volume 13, Numer 1, 2010

5 > > Puliction Fig 4 Histologicl oservtion with hemtoxylin nd eosin (H&E) stining. Aundnt firous collgen tissues interspersed with some ground sustnces (rrowheds in ), nd odontogenic epithelil rests (rrows in ). Some surfces were prtilly lined reduced enmel epithelium (red rrows in ). No clcifiction ws found in either section. Originl mgnifiction 20. Fig 5 The rdiogrphs nd histology of the 15-yer-old mle ptient. This ptient hd lmost identicl lesions s his sister descried in Figures 1 to 3. Pnormic rdiogrph (), 1 yer fter extrction of deciduous teeth in impcted cnine positions. Histology () with H&E stining t mgnifiction 20. References 1. Grder DG. The centrl odontogenic firom: n ttempt t clrifiction. Orl Surg Orl Med Orl Pthol 1980;54: Sndler HJ, Nerssin RR, Ctldo E, Pocheit S, Dyl Y. Multiple dentl follicles with odontogenic firom-like chnges (WHO type). Orl Surg Orl Med Orl Pthol 1988;66: Lukinm PL, Hietnen J, Anttinen J, Ahonen P. Contiguous enlrged dentl follicles with histologic fetures resemling the WHO type of odontogenic firom. Orl Surg Orl Med Orl Pthol 1990;70: Gomez RS, Silv EC, Silv-Filho EC, Cstro WH. Multiple clcifying hyperplstic dentl follicles. J Orl Pthol Med 1998;27: Grdner DG, Rdden B. Multiple clcifying hyperplstic dentl follicles. Orl Surg Orl Med Orl Pthol Orl Rdiol Endod 1995;79: Wlker LM, Wood AJ, McDonld A, Crpenter W. Unerupted mndiulr second primry molr with n unusul histopthologicl finding: cse report. J Dent Child 2004;71: Grover PS, Lorton L. The incidence of unerupted permnent teeth nd relted clinicl cses. Orl Surg Orl Med Orl Pthol 1985;59: Jco H. The etiology of mxillry cnine impctions. Am J Orthod 1983;84: Bishr SE, Kommer DD, McNeil MH, Montgno LN, Oesterle LJ, Youngquist HW. Mngement of impcted cnines. Am J Orthod 1976;69: Mesgrzdeh AH, Esmilzdeh H, Adolrhimi M, Shhmfr M. Pthosis ssocited with rdiogrphiclly norml folliculr tissues in third molr impctions: clinicopthologicl study. Indin J Dent Res 2008;19: Kim SG, Kim MH, Che CH, Jung YK, Choi JY. Downregultion of mtrix metlloproteinses in hyperplstic dentl follicles results in norml tooth eruption. BMB Rep 2008;41: Roqueert D, Chmpsur A, Gil del Rel P, Prsd H, Rohrer MD, Pintdo M et l. Amelogenesis imperfect, rough hypoplstic type, dentl folliculr hmrtoms nd gingivl hyperplsi: report of cse from Centrl Americ nd review of the literture. Orl Surg Orl Med Orl Pthol Orl Rdiol Endod 2008;106: Kim J, Ellis GL. Dentl folliculr tissue: misinterprettion s odontogenic tumors. J Orl Mxillofc Surg 1993;51: Surez PA, Btskis JG, El-Nggr AK. Don t confuse dentl soft tissues with odontogenic tumors. Ann Otol Rhinol Lryngol 1996;105: The Chinese Journl of Dentl Reserch 75