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dentistry journal

Case Report Uprighting an Impacted Permanent Mandibular First Associated with a Dentigerous Cyst and a Missing Second Mandibular Molar—A Case Report

Konstantina Tsironi 1,* , Emmanouil Inglezos 1, Emmanouil Vardas 2 and Anastasia Mitsea 3 1 Posidonos 14, Imia square, Voula, 16673 Athens, Greece 2 Clinic of Hospital Dentistry, Dental School, National and Kapodistrian University of Athens, Thivon 2 Goudi, 11527 Athens, Greece 3 Department of Oral Diagnosis and Radiology, Dental School, National and Kapodistrian University of Athens, Thivon 2 Goudi, 11527 Athens, Greece * Correspondence: [email protected]; Tel.: +30-698-682-7064

 Received: 3 April 2019; Accepted: 21 May 2019; Published: 27 June 2019 

Abstract: The purpose of this paper is to present a case of an impacted mandibular first molar associated with a dentigerous cyst and a missing in an 11-year-old girl that was treated with combined surgical and orthodontic procedures. After clinical and radiographic evaluation, marsupialization of the cyst was decided, and a molar attachment was bonded on the buccal side of the impacted molar as a part of a full orthodontic treatment with fixed appliances. After 18 months of orthodontic traction, the molar was moved to a more advantageous position, and new bone apposition was observed on the site of the cystic lesion. Histological examination confirmed a dentigerous cyst. The molar was left to erupt spontaneously for 14 more months. A functional occlusion was finally achieved. An interdisciplinary approach proved to be an effective modality in treating a large dentigerous cyst associated with a deeply impacted first mandibular molar, presenting many advantages, such as new bone apposition and patient comfort.

Keywords: first molar uprighting; impacted molar; dentigerous cyst; unerupted ; missing second mandibular molar

1. Introduction can be defined as tooth retention due to an obstacle in the eruption path or—less commonly—due to an ectopic position of the tooth germ [1]. Tooth impaction is relatively common (prevalence of 17%) [2], third molars being the most commonly affected teeth, followed by maxillary canines, mandibular , and mandibular canines. The prevalence rate of impaction for the mandibular second molar ranges from 0.06 to 0.3%, and for the mandibular first molar, the prevalence described is <0.01% [3]. Impaction of the first molar is a serious problem that needs to be addressed in order to achieve functional occlusion and facial harmony. In cases of non-treatment, it can cause a decrease in the vertical dimension of the lower , , extrusion of the antagonist, root resorption in the adjacent teeth, or formation of a dentigerous cyst [4,5]. Dentigerous cysts are odontogenic cysts that originate by separation of the follicle from around the of an unerupted tooth [6]. Generally, they are associated with the crowns of impacted or unerupted or, less frequently, with an , a developing tooth, or a deciduous tooth [7,8]. The incidence of dentigerous cysts in the general population has been estimated at 1.44 cysts for every 100 unerupted teeth [9], comprising the second most common (14 to 24%) of all odontogenic cysts [10,11]. Most often, dentigerous cysts present no clinical symptoms and are detected during routine radiographic examination. In some cases, gum swelling or sensitivity,

Dent. J. 2019, 7, 63; doi:10.3390/dj7030063 www.mdpi.com/journal/dentistry Dent. J. 2019, 7, x FOR PEER REVIEW 2 of 10 Dent. J. 2019, 7, 63 2 of 10 detected during routine radiographic examination. In some cases, gum swelling or sensitivity, tooth mobility, and displacement of adjacent teeth may be observed if the cyst reaches large dimensions tooth(>2 cm mobility, in diameter) and displacement or if it gets infected of adjacent [6,12]. teeth Radiographically, may be observed dentigerous if the cyst reaches cysts are large characterized dimensions (by>2 a cm symmetric, in diameter) well-circumscribed or if it gets infected radiolucent [6,12]. Radiographically, lesion, most often dentigerous unilocular, surrounding cysts are characterized the crown byof an a symmetric,unerupted tooth well-circumscribed [12,13]. Differential radiolucent diagnosi lesion,s from most other often cysts, unilocular, such as radicular surrounding cysts and the crownodontogenic of an keratocysts, unerupted toothor from [12 tumors,,13]. Di suchfferential as ameloblastoma, diagnosis from calcifyi otherng cysts, epithelial such odontogenic as radicular cyststumor, and and odontogenic odontogenic keratocysts, fibroma, is necessary or from tumors, through such histopathologic as ameloblastoma, evaluation calcifying [14]. Dentigerous epithelial odontogeniccysts are generally tumor, treated and odontogenic surgically either fibroma, by enucleation, is necessary marsupialization, through histopathologic or by decompression evaluation [14 of]. Dentigerousthe cyst via cystsfenestration are generally [15–17]. treated This surgicallycase report either describes by enucleation, a conservative marsupialization, surgical approach or by decompressioncombined with oforthodontic the cyst via treatment fenestration of [15an– 17impacted]. This case first report mandibular describes molar a conservative associated surgicalwith a approachdentigerous combined cyst in an with adolescent. orthodontic treatment of an impacted first mandibular molar associated with a dentigerous cyst in an adolescent. 2. Case Presentation 2. Case Presentation An 11-year old female came to the clinic after her parents complained of missing lower left teeth. No painAn 11-yearor previous old female discomfort came was to the reported. clinic after The her overall parents patient’s complained dental of and missing physical lower health left teeth. was Nogood pain with or previousnon-specific discomfort general wasmedical reported. history The an overalld no contra-indication patient’s dental and to dental physical treatment. health was A goodsigned with informed non-specific consent general from medicalthe patient’s history mother and no was contra-indication obtained before to dentalthe patient treatment. participated A signed in informedthe study. consent from the patient’s mother was obtained before the patient participated in the study. Extraoral examination revealed a symmetric face with no deficitdeficit in the lower left part of the the face. face. Intraoral examinationexamination revealed revealed a a Class Class II II incisor relationship relationship and and a Class a Class II molarII molar relationship relationship from from the rightthe right side side in a latein a mixedlate mixed . dentition. At the At left the side, left theside, first the mandibular first mandibular molar wasmolar clinically was clinically absent, andabsent, the and overlying the overlying mucosa wasmucosa normal was in normal color and in color texture. and The texture. adjacent The deciduous adjacent deciduous second molar second had amolar large had amalgam a large restoration amalgam restoration with no signs with of no secondary signs of caries.secondary caries. The panoramicpanoramic radiographic radiographic examination examination (PanRad) (PanRad) revealed revealed the presencethe presence of six permanentof six permanent molars inmolars the upper in the jawupper and jaw five and permanent five permanent molars molars in the lowerin the jawlower (Figure jaw (Figure1). From 1). the From size the of size the teeth,of the theteeth, stage the of stage the root of formation,the root formation, the location the of thelocati teethon buds,of the and teeth the buds, angulation and the of the angulation impacted of molar, the itimpacted was assumed molar, that it was the impactedassumed that tooth the was impacted the first tooth mandibular was the molar, first mandibular and the adjacent molar, tooth and bud the wasadjacent the mandibular tooth bud thirdwas the molar. mandibular A well-circumscribed third molar. unilocularA well-circumscribed radiolucent lesion unilocular in the radiolucent body of the mandiblelesion in the was body noticed, of the associated with was the noticed, crown a ofssociated the vertically with the impacted crown mandibularof the vertically left first impacted molar. Themandibular roots of left the impactedfirst molar. molar The roots were of completely the impacted developed molar were with completely closed apexes. developed The cephalometric with closed X-rayapexes. confirmed The cephalometric a skeletal Class X-ray II malocclusionconfirmed a skeletal (Figure2 Class). The II clinical malocclusion diagnosis (Figure was dentigerous 2). The clinical cyst associateddiagnosis was with dentigerous the impacted cyst molar. associated with the impacted molar.

Figure 1. Pretreatment panoramic radiograph showing an impacted mandibular left molar associated with aa rather rather large, large, well well circumscribed, circumscribed, unilocular unilocula radiolucentr radiolucent lesion. Thelesion. root The is almost root fullyis almost developed. fully developed.

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FigureFigure 2. 2. Pretreatment PretreatmentPretreatment cephalometric cephalometric radiograph radiograph showing showing a askeletal skeletal Class Class II II profile. profile. profile.

TheThe main main objectives objectives objectives of of of the the the treatment treatment treatment plan plan plan were were were to to toeliminate eliminate eliminate the the the cystic cystic cystic lesion lesion lesion and and and establish establish establish a a functionalfunctionala functional occlusion. occlusion. occlusion. The The The latter latter latter should should should include include include expansion expansion expansion of of ofthe the the upper upper upper arch, arch, arch, leveling leveling leveling and and and alignment alignment ofof both bothboth arches, arches,arches, closure closure ofof of anyany any residual re residualsidual spaces spaces spaces of of missingof missing missing teeth, teeth, teeth, and and establishmentand establishment establishment of a functionalof of a a functional functional molar molarmolarrelationship. relationship. relationship. After After takingAfter taking taking into consideration into into consideration consideration the age the the of age age the of patient,of the the patient, patient, the missing the the missing missing mandibular mandibular mandibular left molar left left molarmolarand her and and occlusal her her occlusal occlusal status, status, status, a combined a a combined combined surgical-orthodontic surgic surgical-orthodontical-orthodontic approach approach approach was decided was was decided decided upon. upon. upon. Both lowerBoth Both lowerlowerdeciduous deciduous deciduous molars molars molars were were extractedwere extracted extracted under under under local loca loca anesthesia.l lanesthesia. anesthesia. Two Two Two metal metal metal bands bands bands were were were cemented cemented cemented to theto to thethemaxillary maxillary maxillary first first first permanent permanent permanent molars, molars, molars, and and and a Quad-Helixa a Quad-Helix Quad-Helix appliance appliance appliance was was was inserted inserted inserted in in thein the the palatal palatal palatal tubes tubes tubes of ofofthe the the bands. bands. bands. EightEight Eight monthsmonths months later,later, later, allall all mandibularmandibular mandibular permanent permanent premol premolars premolarsars had hadhad fully fullyfully erupted, erupted,erupted, and and a anew new panoramicpanoramic examination examination showed showed that that the the impacted impacted molar molarmolar was waswas still stillstill at atat the thethe same samesame height heightheight (Figure (Figure(Figure 3).3 3).).

Figure 3. Panoramic radiograph after the extraction of the mandibular deciduous molars with the Figure 3. Panoramic radiograph after after the extraction of the mandibular deciduous molars with the Quad-helix appliance. Quad-helix appliance.

FixedFixed orthodontic orthodontic appl appliancesappliancesiances (MBT, (MBT, Dynaflex DynaflexDynaflex®®, ,10403, 10403 10403 International International International Plaza Plaza Plaza Drive, Drive, Drive, St.Ann., St.Ann., St.Ann., MO, MO, MO, USA)USA) were were were placed placed placed in in inthe the the mandibular mandibular mandibular arch. arch. arch. After After After thr threeee threemonths months months of of active active of orthodontic active orthodontic orthodontic treatment, treatment, treatment, initial initial alignmentalignmentinitial alignment was was achieved, achieved, was achieved, and and exposure exposure and exposure of of the the impa of impa thectedcted impacted tooth tooth was toothwas performed. performed. was performed. A A muccoperiostal muccoperiostal A muccoperiostal flap flap waswasflap raised wasraised raised under under under local local localanesthesia, anesthesia, anesthesia, and and anda a communicati communicati a communicationonon was was was established established established between between between the the the cystic cystic cystic cavity cavity cavity andand the the oral oral cavity. cavity. A A specimen specimen of of the the cyst cyst was was se se sentntnt for for biopsy. biopsy. The The crown crown of of the the impacted impacted tooth tooth was was exposed,exposed, and and a a conventional conventional molar molar attachment attachment wa was sbonded bonded on on the the buccal buccal side side perpendicular perpendicular to to the the

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Dent.exposed, J. 2019, 7, andx FOR a PEER conventional REVIEW molar attachment was bonded on the buccal side perpendicular4 of 10 to the long axis of the second molar with a self-etching adhesive. To upright the second molar, elastic traction longwas axis applied. of the second molar with a self-etching adhesive. To upright the second molar, elastic tractionHistopathological was applied. examination confirmed the diagnosis of a dentigerous cyst. HistopathologicalAfter two months examination of orthodontic confirmed extrusion, the diagnosis radiographic of a dentigerous examination cyst. showed a significant reductionAfter two in themonths size of theorthodonti cystic cavityc extrusion, and a radiographic more favorable examination position ofshowed the first a significant molar (Figure 4). reductionOrthodontic in the traction size of continuedthe cystic cavity for 16 and more a more months, favorable while theposition upper of dental the first arch molar was (Figure also treated 4). with Orthodonticfixed orthodontic traction appliances.continued for Eighteen 16 more months months, after while the the surgical upper procedure, dental arch the was first also molar treated had taken witha very fixed favorable orthodontic position appliances. in the Eighteen dental arch, months thus after debonding the surgical of the procedure, fixed appliances the first wasmolar decided had due takento the a very patient’s favorable unwillingness position in tothe continue dental arch with, thus the debonding fixed appliance of the treatment. fixed appliances The previously was decided impacted due to the patient’s unwillingness to continue with the fixed appliance treatment. The previously molar was left to erupt spontaneously, while the upper left molars were stabilized in order to prevent impacted molar was left to erupt spontaneously, while the upper left molars were stabilized in order over-eruption (Figure5). to prevent over-eruption (Figure 5).

FigureFigure 4. 4.PanoramicPanoramic radiograph radiograph two months two months after afterthe surgical the surgical exposure exposure of the offirst the permanent first permanent mandibularmandibular molar. molar. Note Note the thereduction reduction in size in sizeof th ofe former the former cystic cystic cavity cavity and the and more the more favorable favorable axis axis of of the impacted tooth. tooth.

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Figure 5. Immediately after debonding of the fixed orthodontic appliances of both dental arches. The Figure 5. Immediately after debonding of the fixed orthodontic appliances of both dental arches. mandibular left first molar was left to erupt spontaneously. Note that the maxillary molars are The mandibularattached to one left another first molar via a was stainless-steel left to erupt wire spontaneously. in order to prevent Note further that the eruption maxillary of the molars first are attachedmaxillary to one molar. another via a stainless-steel wire in order to prevent further eruption of the first maxillary molar.

The patient reported for follow-up appointments every two months, and finally, 14 months after the debonding of the fixed appliances, the molar had fully erupted (Figure6). A stable occlusion was achieved, both arches were well aligned, and there were no residual spaces. Dent. J. 2019, 7, x FOR PEER REVIEW 6 of 10

The patient reported for follow-up appointments every two months, and finally, 14 months after Dent. J. 2019, 7, 63 6 of 10 the debonding of the fixed appliances, the molar had fully erupted (Figure 6). A stable occlusion was achieved, both arches were well aligned, and there were no residual spaces.

Figure 6. 14 months after debonding of the fixed orthodontic appliances. The first mandibular left Figure 6. 14molar months has fully after erupted debonding and is in occl ofusion the fixedwith the orthodontic maxillary first appliances.molar. The first mandibular left molar has fully erupted and is in occlusion with the maxillary first molar.

Radiographic examination revealed an almost vertical position of the mandibular first molar and normal trabecular bone surrounding the previously impacted tooth (Figure7). Dent. J. 2019, 7, x FOR PEER REVIEW 7 of 10

Dent. J. 2019, 7, 63 7 of 10 Radiographic examination revealed an almost vertical position of the and normal trabecular bone surrounding the previously impacted tooth (Figure 7).

Figure 7. Panoramic radiograph 14 months after the debonding of the fixed orthodontic appliances Figure 7. Panoramic radiograph 14 months after the debonding of the fixed orthodontic appliances and 32 months after the marsupialization of the dentigerous cyst. and 32 months after the marsupialization of the dentigerous cyst.

3. Discussion Discussion For the treatment of the dentigerous cyst, th thee possible alternatives were enucleation, marsupialization, or or decompression of the cystcyst viavia fenestration.fenestration. Marsupialization Marsupialization is is a a rather conservative treatment treatment modality modality for for the the treatment treatment of dentigerous of dentigerous cysts, cysts, as it decreases as it decreases the risk the of riskjaw fractureof jaw fracture and nerve and nervedamage damage [18]. [Moreover,18]. Moreover, it is itthe is thetreatment treatment of ofchoice choice when when the the treatment treatment plan plan involves preservation of of the impacted tooth, in co contrastntrast with enucleation, where the involved tooth is extracted. Also, Also, marsupializati marsupialization,on, when when compared compared to to decompression decompression of of the the cyst cyst via fenestration, has the advantage of promoting the spontaneous toot toothh eruption of the tooth that is associated with the cyst cyst [19]. [19]. However, However, in in patients patients over over 10 10years years old, old, spontaneous spontaneous eruption eruption does does not often not oftenoccur, occur, thus orthodonticthus orthodontic treatment treatment is almost is almost always always necessary necessary in order in order to toguide guide the the involved involved tooth tooth to to its its final final position inin the the dental dental arch arch in casesin cases that thethat tooth the istooth not extractedis not extracted with the cystwith [20 the]. Severalcyst [20]. orthodontic Several orthodonticmodalities have modalities been described have been to described guide the to eruption guide the of eruption an impacted of an tooth, impacted including tooth, segmentalincluding segmentalsprings, diverse springs, spring diverse designs, spring elastic designs, traction, elastic or even traction, mini implants or even [21 mini–23]. implants Both segmental [21–23]. springs Both segmentaland diverse springs spring designsand diverse require spring complex designs mechanics require and complex may cause mechanics tissue irritationand may tocause the patient. tissue irritationInstead, elastic to the tractionpatient. canInstead, be quite elastic acceptable traction fromcan be the quite patient acceptable and would from be the adequate patient inand our would case, beas theadequate long axis in our of the case, impacted as the toothlong axis did notof th presente impacted severe tooth angulation. did not Minipresent implants severe canangulation. be quite Minihelpful implants in the uprightingcan be quite process, helpful but in duethe uprighting to their cost process, and the but surgical due to process their cost involved, and the the surgical use of processthis technique involved, was the rejected. use of this technique was rejected. Impacted first first molars molars associated associated with with a a dentigerous dentigerous cyst cyst are are very very rare, rare, thus thus little little information information is availableis available in the in the dental dental literature. literature. Moreover, Moreover, agenesis agenesis of the of thelower lower left second left second molar molar is also is alsoa very a veryrare entityrare entity [24]. Usually, [24]. Usually, dentigerous dentigerous cysts are cysts associat are associateded with impacted with impacted teeth, mandibular teeth, mandibular third molars third beingmolars the being most the commonly most commonly affected aff teeth,ected teeth,followed followed by the by permanent the permanent maxillary canine and andthe permanentthe permanent maxillary maxillary third third molar molar [7]. [ 7Less]. Less commonly, commonly, it itcan can occur occur in in central central , incisors, yet yet the occurrence of suchsuch aa cystcyst withwith aa permanentpermanent first first molar molar is is still still uncommon uncommon and and comprises comprises about about 1.1% 1.1% of

Dent. J. 2019, 7, 63 8 of 10 all dentigerous cysts [25]. Male patients are more frequently affected, especially during the second and the third decade of life [6]. It is known that dentigerous cysts may present complications such as pathological fractures [26–28] or transformation, either malignant or ameloblastic. Even though malignant transformation is rare, such cysts should be either enucleated or marsupialized [29,30]. In our case, even though there was lack of any symptoms, the cyst size and the location made the decision for early treatment necessary. Histological examination was considered essential to confirm the clinical and the radiographical diagnosis. The clinical and radiological criteria for selecting the appropriate treatment modality relate to the following: cyst size and location, dentition involved and stage of the root development of the involved tooth, the position of the involved tooth in the bone and relation to the adjacent teeth or anatomical structures and age of the patient [31]. Combination of enucleation and complete removal of the involved tooth is preferred in cases involving a single tooth impaction. For instance, this is usually the treatment of impacted third molars in adults as they have no function; however, when other teeth are impacted, removal of the tooth is not often in the patient’s best interests or desire, especially in children as removal of such teeth may have functional, esthetic as well as psychological consequences [15]. In our case, the cyst was marsupialized because the associated tooth was vital in order to achieve a functional occlusion and an esthetic result. Combined orthodontic and surgical treatment was effective in preventing not only nerve damage but also periodontal breakdown of the impacted tooth and the adjacent . The marsupialization allowed for some bone filling of the residual cavity as the cyst decompressed [15,28,32]. Different treatment modalities such as surgical repositioning and extraction of the impacted tooth induce a higher risk of complications. Surgical repositioning or transplantation may provoke root resorption, ankylosis, or even necrosis and therefore should be the treatment of choice only in cases where orthodontic treatment is contraindicated [33,34]. Extraction of the impacted tooth to let the non-impacted molars erupt does not always have the optimized result, as the other molars may incline towards the extraction site [35]. In our case, extraction would have had destructive effects for the occlusion, as the second molar was missing. Cooperation of the patient is fundamental when marsupialization is the treatment of choice, as oral hygiene and long-term patience is crucial for treatment success. The orthodontic appliances can irritate the adjacent mucosa and cause pain to the patient [36]. The fact that elastic traction was used for the uprighting of the molar until it reached the gingival margin made it easier and more convenient for the patient in order to achieve good oral hygiene.

4. Conclusions We showed cyst marsupialization together with orthodontic molar uprighting to be effective in treating a dentigerous cyst associated with an impacted mandibular first molar. Such an interdisciplinary approach makes treatment easier, greatly reduces the risk of postsurgical complications, and appears to be advantageous in terms of the periodontal health of adjacent teeth. In addition, it is a conservative surgical approach that is generally preferred by the patients, as it preserves the function and the esthetic values and also prevents the adolescent from psycho-social trauma associated with . However, such an approach does require close cooperation—not only among practitioners but also between the patient and the practitioners—in order to monitor the healing of the lesion, the maintenance of adequate oral hygiene, and the compliance of the patient with the long-term evaluation of the uprighting of the tooth in its final position.

Author Contributions: Conceptualization, K.T., E.I. and E.V.; methodology, E.I. and E.V.; software, E.I. and A.M.; validation, K.T., E.I., E.V. and A.M.; formal analysis, K.T.; investigation, K.T.; resources, K.T. and A.M.; data curation, K.T.; writing-original draft preparation, K.T.; writing-review and editing, K.T. and A.M.; visualization, K.T., supervision, A.M.; project administration; E.I. and E.V. Funding: This case report received no external funding. Dent. J. 2019, 7, 63 9 of 10

Conflicts of Interest: The authors declare no conflict of interest.

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