Multiple tooth anomalies in a nonsyndromic patient with class II division 2 malocclusions: A case report and a literature review

Transcription

1 Cse Report Multiple tooth nomlies in nonsyndromi ptient with lss II division 2 mlolusions: A se report nd literture review E Ismn, O Ismn 1, AM Aktn 1, E Cifti 1, T Topuoglu 2 Deprtments of Orthodontis nd 1 Dentomxillofil Rdiology, Fulty of Dentistry, Gzintep University, Gzintep, Southestern Antoli, 2 Deprtment of Orthodontis, Fulty of Dentistry, Zirve University, Gzintep, Turkey Astrt Reports in the literture out the rniofil hrteristis of ptients with lss II division 2 mlolusions show lot of different ptterns ompnied y pltlly displed upper inisors, ongenitl missing teeth, polydistem, fusion, germintion, tooth imption, peg shped lterl inisors, persistent teeth, hypodonti, persistent deiduous teeth, trnspositions, nd supernumerry teeth. The following se report fouses on the desription of the linil hrteristis oserved on ptient with very unusul onjuntion of dentl nd skeletl nomlies mentioned ove, s well s literture review on the relted issues. Extr intr orl exmintions, rdiogrphi evlutions, orthodonti onsulttion, nd reviewing the literture onluded tht this nonsyndromi ptient tht refused to reeive ll dentl tretment pprohes is speil with its uniqueness. Key words: Angle lss II, fused teeth, mlolusion, overite, tooth normlities Dte of Aeptne: 10-Ot-2014 Introdution Clss II division 2 mlolusions my our in vriety of dentolveolr mlolusion strutures. Geneti inlintion my ply n importnt role mong these differentitions. Dentl nomlies in numer, size, position, or struture re lso importnt uses tht n led to mlolusions. Geneti or environmentl ftors, suh s drug emryopthy, fetl lohol exposure, or hyper/hypovitminosis in pregnnt mother, re thought to e the etiology of the nomlies. It my e esy to sy tht the etiology of the two onditions n e onsidered to e nested phenomenon. Bsdr et l. reported tht lss II division 2 mlolusions re losely ssoited with some types of ongenitl tooth nomlies, suh s missing teeth, peg shped lterls, trnspositions, or imptions. [1] These mlolusions, whih ould e seen either on their own or with vrious ompnying disorders, my tully e polygeni nd dditive in nture through omined expression of genetilly ssigned ntomil omponents. However, previous studies hve not exmined how mny dentl nomlies ould e seen in one ptient. [1] For this reson, this se study presents very unusul omintion of multiple dentl nomlies, inluding germintion, peg shped teeth, ongenitlly missing teeth, geminted impted nine, persistent teeth, nd polydistem with lss II division 2 skeletl nomly in 15 yer old oy. Cse Report A helthy 15 yer old young oy ws referred to our lini due to toothhe in the lower right nine region. Informed onsent given y the institution ws signed y the ptient s prents. The ptient hd no systemi disorders, nd there ws no syndrome history in his medil or Quik Response Code: Aess this rtile online Wesite: Address for orrespondene: Dr. E Işmn, Deprtment of Orthodontis, Fulty of Dentistry, Gzintep University, 27310, Gzintep, Turkey. E mil: erenismn@hotmil.om DOI: / PMID: Nigerin Journl of Clinil Prtie Jul-Aug 2015 Vol 18 Issue 4 563

2 fmily kground. During the extr orl nd intr orl exmintions [Figure 1 h], some skeletl nd dentl nomlies suh s deep ite, polydistem, missing teeth, lss II molr nd nine reltionships, peg shped lterls, nd fusion/germintion phenomenon were oserved. After detiled rdiogrphi exmintion inluding pnormi, ephlometri, postero nterior nd peripil rdiogrphs, vrious dentl nomlies, suh s hypodonti, gemintion, peg shped teeth, persistent deiduous teeth, nd impted geminted teeth were notied [Figure 2 ]. During linil oservtion, it ws notied tht the ptient hd tendeny for lss II division 2 mlolusion. The si fetures of this mlformtion inlude the lss II molr reltionship of the dentl rhes omined with retrolintion of the upper inisors nd exessive inisl overite [Figure 1d nd f]. The gonil ngle seemed to e low, resulting in rther squre fil profile [Figure 1]. The liomentl groove ws pronouned eneth the lower lip, evoking nd strengthening the dignosis of this lss II division 2 mlolusion. For this reson, onsulttions with liniins from the orthodontis deprtment were done nd fter proper orthodonti evlution inluding ephlometri nlysis [Figure 3], it ws found tht the ptient hd norml sgittl mxillry position (SNA = 86.9) nd retruded mndiulr position (SNB = 78.0). Aording to the vertil evlution, the ptient hd low ngle pttern (Sn GoGn = 26.4) s well s lower fil height (ANS Me = 57.1). These outomes onfirmed tht these dentl nomlies were ssoited with skeletl lss II division 2 mlolusion with redued lower fil height [Figure 3]. The ril sttus of the ptient ws white usin. For tht reson, the Europen ephlometri norms were sujeted for the ptient. [2] Rdiogrphi exmintion reveled tht the mndiulr permnent entrl nd lterl inisors were ilterlly ongenitlly missing [Figure 4]. In ddition, the right nd left mndiulr third molrs nd the left mxillry third molr were lso found to e ongenitlly missing [Figure 4 nd ]. Beuse seven ongenitlly missing teeth were determined in one ptient, this ondition ould e termed s eing oligodonti, ording to the literture. [3] The right mxillry permnent nine nd the seond premolr teeth were oserved s impted rdiogrphilly [Figure 5]. An irregulr ilterl morphology of the mndiulr permnent nines, with greter mesiodistl rown width developmentl oluso gingivl slots on the ul nd lingul surfes, ws found to e geminted [Figures 4 nd 6 ]. A vitlity test ws performed on the geminted teeth, nd the right mndiulr nine ws found to e nonvitl, the other teeth were found to e vitl, lthough the teeth were dignosed s eing ries free. Gingivitis relted to ll the teeth ws deteted, espeilly round the geminted teeth. The mxillry permnent right nine ws lso found to e geminted. The geminted tooth on the upper jw ws found to e impted, d e f g Figure 1: Extr-orl photogrphs of the ptient. () Anterior fil view. () Anterior smiling view. () Profile view. (d) Right dentl view (note tht the impted tooth is visile on the gingivl mrgin.) (e) Frontl dentl view. (f) Left dentl view. (g) Olusl view of the upper dentition (note the polydistem). (h) Olusl view of the lower dentition 564 Nigerin Journl of Clinil Prtie Jul-Aug 2015 Vol 18 Issue 4 h

3 Figure 2: () Pnormi rdiogrph of the ptient. () Antero-posterior rdiogrph of the ptient. () Lterl ephlometri rdiogrph of the ptient Figure 3: Cephlometri tring of the ptient Figure 4: () Rdiogrph of the mndiulr nterior re (showing the sene of mndiulr entrl nd lterl inisors ilterlly). () Rdiogrph presenting the lkness of the right mndiulr third molr. () Rdiogrph exhiiting the lkness of the left mndiulr nd mxillry third molr Figure 5: Rdiogrphi view of the impted mxillry permnent right nine nd seond premolr Figure 7: Anterior nd lterl intr-orl views of the ptient (note the ilterlly peg-shped mxillry lterl inisors, left mxillry nine nd right mxillry entrl teeth nd polydistem) Figure 6: Intr-orl photogrphs of the ptient. () Olusl view. () Left intr-orl view. () Right intr-orl view rdiogrphilly [Figure 5]. The mxillry lterl inisors, ilterlly, the right entrl inisor, nd the left nine were found to e peg shped teeth. Polydistem ws notied relted to the peg shped teeth, linilly [Figure 7 nd ]. The right mndiulr primry entrl nd lterl inisors, the left mndiulr primry entrl inisor, nd the right mxillry primry nine were persistent [Figures 4 nd 5]. The inisl edges of the persistent inisors were eroded euse of the deep ite, lthough their roots were either intt or minimlly resored [Figures 6 nd 4]. Nigerin Journl of Clinil Prtie Jul-Aug 2015 Vol 18 Issue 4 565

4 The dignosis outomes were summrized to the ptient nd ll the multi disipliner tretment pln inluding deprtments of periodontology, onservtive tretment, surgery, orthodonty, nd prostheti tretment ws delred to the prents. However, the fmily s soil seurity ws not suitle for detiled pproh like this nd esides they were living fr wy from the university lini nd trveling severl times would ost lot of money whih this fmily might not fford; therefore, the prents refused to tke required tretment. For this reson, the follow up reords nd pre post tretment evlution ould not e performed. Disussion There re numer of studies out lss II division 2 mlolusions in the literture tht show mny different forms tht re ompnied y nomlies, suh s displed upper inisors, ongenitl missing teeth, polydistem, fusion, germintion, tooth imption, peg shped lterl inisors, hypodonti, persistent deiduous teeth, trnspositions, nd supernumerry teeth. [1] However, the present study imed to present more extreme se of lss II division 2 mlolusion with multiple omintion of rre dentl nomlies, suh s ilterl geminted lower nines, n impted geminted nine, oligodonti, persistent teeth, polydistem, peg shped lterl inisors nd nines, nd genesis of the third molrs ssoited with lss II division 2 skeletl pttern. Aording to our est knowledge, no sientifi literture hs previously reported on similr ssoition mong these types of dentl normlities in single ptient. In the study of Bsdr et l., [1] the uthor suggested tht there is strong ssoition etween lss II division 2 mlolusion nd ongenitl genesis. He lso stted tht with the exeption of the third molrs, the sene of ll other teeth ws seen t lest 3 times more often in individuls hving lss II division 2 mlolusion thn in the generl puli. In nother study, it ws onluded tht there were signifint rtes of dentl normlities in orthodonti ptients; however, there ws no ssoition etween orthodonti mlolusion types nd dentl nomlies, exept for imption. [4] Defets in ertin genes nd etiologi ftors in the pre nd postntl periods re thought to e responsile for dentl nomlies. [5] Although geneti kground plys n importnt role in skeletl lss II division 2 mlolusion nd some dentl nomlies suh s ongenitl missing teeth, peg shped lterls, trnspositions, nd imptions, it ws found tht minimum numer of dentl nomlies ws reported in skeletl lss II division 2 mlolusion ording to the results of the study y Uslu et l. [4] However, in this urrent se report, multiple dentl nomlies were presented with skeletl lss II division 2 mlolusion. Fmilil ourrene of lss II/division 2 deep ite hs een ttested in severl literture pulishments inluding twin nd triplet reports y Kloeppel, [6] Mrkovi [7] nd Litt nd Nielsen [8] nd pedigree studies y Korkhus [9] nd Truner. [10] Pek et l. [11] delred tht these studies points to indisputle geneti influene, proly of n utosoml dominnt type with inomplete penetrne, s ritil element in the formtion nd expression of Angle s lss II/division 2 deep ite mlolusion. They onluded tht inheritne of omplex olusl vrition suh s II/2 deep ite mlolusion my tully e polygeni nd dditive in nture, through omined expression of genetilly determined ntomil omponents, rther thn eing the effet of single ontrolling gene for entire olusl mlformtion. Regrding this se report, the uthors thought tht some isolted nonsyndromi geneti prolems suh s single nuleotide polymorphism or multiple muttions in genes responsile with the formtion of teeth or mxill mndiulr skeletl omplex would hve een found if the ptient hd greed to tke the relted tests. Future se reports should etter fous on the genotypes of the ptient. Hypodonti is the lk of dentition in whih one or more (up to five) teeth re ongenitlly missing, wheres the sene of more thn five teeth is known s oligodonti. [3] The prevlene of hypodonti rnges from 0.3% to 11.3% in the literture; however, oligodonti is rther less pprent, nd it is not s ommon s hypodonti. [12 15] It n e seen in syndromi ptients s well s nonsyndromi individuls. Severl studies dislosed tht seven genes re urrently known to hve potentil for using nonsyndromi oligodonti: PAX9 [16] (pired ox gene 9), MSX1 [17] (musle segment homeoo 1), EDA [18] (etodysplsin A), AXIN2 [16] (xis inhiition protein), NEMO [19] (NF kpp B essentil modultor), KRT17 [20] (kertin 17), nd EDARADD [16] (EDAR ssoited deth domin). These types of norml inidents ompnying y lss II division 2 mlolusion were known to e infrequent. In this report, seven permnent teeth, inluding the third molrs (3), the entrl inisors (2), nd the lterl inisors (2), were ongenitlly missing nd ould e lssified s oligodonti. Therefore, the present se is differentited from the previously pulished studies relted to lss II division 2 mlolusion euse it inluded nonsyndromi oligodonti. [1] The inidene of gemintion ours when two teeth originte from one tooth ud nd onsequently, the ptient hs igger tooth, ut the numer of teeth is norml. [21,22] This n lso use imption due to the lrger volume of the rown of the geminted tooth. [23] In ddition, it n e sid tht the gemintion phenomenon ommonly ours in the nterior mxillry region. [24] In the present se, the impted right mxillry nine ws identified s eing geminted due to the mxillry lotion. It did not indue 566 Nigerin Journl of Clinil Prtie Jul-Aug 2015 Vol 18 Issue 4

5 n inrese in the numer of teeth in the rh. Moreover, it hd lrger tooth pperne, nd it ws found to e impted. In ontrst to gemintion, fusion rises through the union of two normlly seprted tooth germs when the ptient ppers to hve missing tooth. However, when norml tooth ud unites to supernumerry tooth germ, it ould lso e desried s fusion. [21,22] Fusion is more frequently seen in the nterior mndiulr region. [24] In the present se, ilterl mndiulr permnent geminted or fused teeth were found linilly nd rdiogrphilly. This ondition n e termed s ilterl fusion sine the nterior mndile lotion of the teeth used derese in the numer of teeth in the rh. On the other hnd, the missing teeth seen in the mndile is ertinly not n evidene of the fusion phenomenon. Among these types of ptients, the numer of teeth is lso norml; thus, distinguishing etween gemintion nd fusion might e rther strenuous or un hievle. [25] In order to overome the diffiulty in the dignosis nd terminology of these types of ses, the term doule or twinning tooth ould e introdued. [24,26,27] These onditions, gemintion or fusion, re very unusul ses in permnent dentition. Moreover, the ft of eing ilterl or multiple mkes them reltively rre. [26,27] Throughout the literture, no studies were notied regrding the geneti kground of tooth fusion s well s gemintion. It is ovious tht future studies should e onentrting on the geneti sis of tooth prolems espeilly fusion nd gemintion. Aording to our most reent knowledge, no sientifi or well ordered pper hs een formerly puliized in the literture out n equivlent onnetion of multiple dentl normlities in single ptient. This kind of imption, tooth genesis, nd peg shped teeth re thought to e responsile for the polydistem seen in the ptient in the present se. This ondition n use estheti, orthodonti, nd periodontl prolems for prtitioners. [28] When teeth dimensions re out of their norml rnge nd smller thn usul, the term mirodonti is used to define the se. Until now, three types of mirodonti hve een hrterized s follows: True generlized mirodonti, reltive generlized mirodonti, nd mirodonti involving single tooth. Mxillry lterl inisors, whih re ffeted y mirodonti nd whih re ommonly lled peg shped lterls, re one of the most well known forms of this phenotype. A peg shped inisor hs remrkle derese in dimeter, extending from the ervil re to the inisl order. [1,2] However, mirodonti of the mxillry nd mndiulr entrl inisors is lso omprtively rre ondition. [29] In the urrent se, the ptient hd ilterl mirodonti peg shped mxillry lterl inisors long with unilterl mxillry peg shped entrl inisor s n unusul ondition. This unommon peg shped tooth nomly is found ommonly on the mxillry lterl inisor; other tooth types re rrely influened y n nomly suh s mndiulr entrl inisor ondition. [29] Although the impted teeth, espeilly the mxillry nine teeth, were found to e losely ssoited with lss II division 2 mlolusion ses in the literture, [1] some of the uthors demonstrted no reltionship etween imption nd mlolusion. [4] In the present se, the right mxillry nine tooth ws found to e impted, nd the imption ws loted on the pexes of the right mxillry lterl nd entrl inisors, rdiogrphilly. The impted nine hd lrge pulp hmer nd mesiodistl dimeter, nd it ppered to e geminted tooth. In ddition, the ptient hd right mndiulr primry entrl nd lterl inisors, left mndiulr primry entrl inisor, nd right mxillry primry s persistent teeth. Persistent teeth seen on mndiulr primry entrl nd lterl inisors were epted s eing reltively rre in the literture. [30] Moreover, previous studies showed tht the most ommon reson for the persistene of primry teeth ws the ongenitl sene of permnent suessor teeth, followed y imption, norml position, nd lte eruption of suessor teeth. In ordne with the literture, the urrent se showed ongenitl sene of permnent suessor teeth nd no or miniml root resorption relted to the suessor teeth. [30 32] Conlusion The omintion of dentl normlities s seen in this se presumly points to unknown geneti ftors. This se presents rre omintion of multiple dentl nomlies in nonsyndromi ptient, whih is different from formerly reported ses. In ddition, the se highlights n ssoition etween lss II division 2 mlolusion nd vrious dentl nomlies suh s pltlly displed upper inisors, ongenitl missing teeth, polydistem, fusion, germintion, tooth imption, peg shped lterl inisors, persistent teeth, hypodonti, persistent deiduous teeth, trnspositions, nd supernumerry teeth. Clinilly, suh ses re importnt nd id in our ility to dvne our understnding of the different types of nomlies tht might e seen in linil prtie. Dentists infrequently enounter suh multiple dentl normlities ses, nd lerer understnding of the normlities my e useful for true dignose nd etter linil mngement. Cses of nonsyndromi ptients with multiple dentl nomlies re intriguing, nd new reserh studies re needed to enlighten the etiology. The uthors delre tht there is no onflit of interests regrding the pulition of this pper. Referenes 1. Bsdr EK, Kiokpsoglou M, Stellzig A. The Clss II Division 2 rniofil type is ssoited with numerous ongenitl tooth nomlies. Eur J Orthod 2000;22: MNmr JA Jr. A method of ephlometri evlution. Am J Orthod 1984;86: Lmmi L, Hlonen K, Pirinen S, Thesleff I, Arte S, Nieminen P. A missense Nigerin Journl of Clinil Prtie Jul-Aug 2015 Vol 18 Issue 4 567

6 muttion in PAX9 in fmily with distint phenotype of oligodonti. Eur J Hum Genet 2003;11: Uslu O, Akm MO, Evirgen S, Ceei I. Prevlene of dentl nomlies in vrious mlolusions. Am J Orthod Dentofil Orthop 2009;135: Frdi A, Kondylidou Sidir A, Bhour Z, Prisis N, Tsirlis A. Inidene of impted nd supernumerry teeth rdiogrphi study in North Greek popultion. Med Orl Ptol Orl Cir Bul 2011;16:e kloeppel W. Close ite olusion in twins. Fortshr Kieferorthop 1953;14: Mrkovi MD. At the rossrods of orl fil genetis. Eur J Orthod 1992;14: Litt RA, Nielsen IL. Clss II, division 2 mlolusion. To extrt or not extrt? Angle Orthod 1984;54: Korkhus G. Investigtions into the inheritne of orthodonti mlformtions. Dent Re 1930;50: Truner R. Leitfden der Prtishen Kieferorthopdie. Berlin: Verlg die Quintessenz; p Pek S, Pek L, Ktj M. Clss II Division 2 mlolusion: A heritle pttern of smll teeth in well developed jws. Angle Orthod 1998;68: Rølling S. Hypodonti of permnent teeth in Dnish shoolhildren. Snd J Dent Res 1980;88: Rose JS. A survey of ongenitlly missing teeth, exluding third molrs, in 6000 orthodonti ptients. Dent Prt Dent Re 1966;17: Hvikko K. Hypodonti of permnent teeth. An orthopntomogrphi study. Suom Hmmslk Toim 1971;67: Dvis PJ. Hypodonti nd hyperdonti of permnent teeth in Hong Kong shoolhildren. Community Dent Orl Epidemiol 1987;15: Bergendl B, Klr J, Steksén Bliks C, Norderyd J, Dhl N. Isolted oligodonti ssoited with muttions in EDARADD, AXIN2, MSX1, nd PAX9 genes. Am J Med Genet A 2011;155A: Chishti MS, Muhmmd D, Hider M, Ahmd W. A novel missense muttion in MSX1 underlies utosoml reessive oligodonti with ssoited dentl nomlies in Pkistni fmilies. J Hum Genet 2006;51: Ayu M, ur Rehmn F, Ysinzi M, Ahmd W. A novel missense muttion in the etodysplsin A (EDA) gene underlies X linked reessive nonsyndromi hypodonti. Int J Dermtol 2010;49: Ku CL, Dupuis Girod S, Dittrih AM, Bustmnte J, Sntos OF, Shulze I, et l. NEMO muttions in 2 unrelted oys with severe infetions nd onil teeth. Peditris 2005;115:e Gss JK, Wilson NJ, Smith FJ, Lne EB, MLen WH, Rytin E, et l. Stetoystom multiplex, oligodonti nd prtil persistent primry dentition ssoited with novel kertin 17 muttion. Br J Dermtol 2009;161: Le Gll M, Philip C, Aoudhrm G. Orthodonti tretment of ilterl geminted mxillry permnent inisors. Am J Orthod Dentofil Orthop 2011;139: Brook AH, Winter GB. Doule teeth. A retrospetive study of geminted nd fused teeth in hildren. Br Dent J 1970;129: Segur Ege JJ, Jiménez Ruio A, Ríos Sntos JV, Velso Orteg E. Dens evgintus of nterior teeth (tlon usp): Report of five ses. Quintessene Int 2003;34: Guimrães Crl LA, Firoozmnd LM, Dis Almeid J. Doule teeth in primry dentition: Report of two linil ses. Med Orl Ptol Orl Cir Bul 2008;13:E Trumn A, Silermn SL. Triple teeth: Cse reports of omined fusion nd gemintion. ASDC J Dent Child 1988;55: Budd CS, Reid DE, Kulild JC, Weller RN. Endodonti tretment of n unusul se of fusion. J Endod 1992;18: Delny GM, Goldltt LI. Fused teeth: A multidisiplinry pproh to tretment. J Am Dent Asso 1981;103: Uheonye IJ, Tokuno AA. Prevlene of peg shped lterls in South Western Nigeri: A omprison of field nd lini findings. Int J Dent Si 2010;8: Chnhl HP, Nndll B. Coexistent peg shped mndiulr entrl inisors long with mxillry lterl inisors A rre se. Int J Orl Mxillof Pthol 2012;3: Aktn AM, Kr I, Sener I, Bereket C, Celik S, Kirty M, et l. An evlution of ftors ssoited with persistent primry teeth. Eur J Orthod 2012;34: Polder BJ, Vn t Hof MA, Vn der Linden FP, Kuijpers Jgtmn AM. A met nlysis of the prevlene of dentl genesis of permnent teeth. Community Dent Orl Epidemiol 2004;32: Beker A, Gillis I, Shpk N. The etiology of pltl displement of mxillry nines. Clin Orthod Res 1999;2:62 6. How to ite this rtile: Ismn E, Ismn O, Aktn AM, Cifti E, Topuoglu T. Multiple tooth nomlies in nonsyndromi ptient with lss II division 2 mlolusions: A se report nd literture review. Niger J Clin Prt 2015;18: Soure of Support: Nil, Conflit of Interest: None delred. 568 Nigerin Journl of Clinil Prtie Jul-Aug 2015 Vol 18 Issue 4