patient with nonsyndromic bilateral and multiple impacted ...an 0.018 3 0.025-in preadjusted...

CASE REPORT

Patient with nonsyndromic bilateral and multipleimpacted teeth and dentigerous cysts

Yoshihito Ishihara,a Hiroshi Kamioka,b Teruko Takano-Yamamoto,c and Takashi Yamashirod

Okayama and Sendai, Japan

aAssisDentibAssocine,JapancProfeTohokdProfecine,JapanThe aproduSuppograntReprinSchooKita-KSubm0889-Copyrdoi:10

228

This article reports the successful treatment of a patient with the unusual occurrence of bilateral and multipledentigerous cysts of the premolars. One impacted mandibular premolar was moved by traction orthodontically.On the opposite side, the impacted premolar was autotransplanted after space was created through mesialmovement and hemi-sectioning of the neighboring molars. The impacted maxillary premolar was extracted.Miniscrews were additionally used for anchorage reinforcement to prevent unintended counteractions and solvethe problem of space management after autotransplantation. We also reviewed the clinical implications of thediagnosis, planning, and treatment of cyst-associated impacted teeth in young adult patients. (Am J OrthodDentofacial Orthop 2012;141:228-41)

Dentigerous cysts are major pathologic entitiesfound in dentistry, accounting for approximately24% of all true cysts in the jaws.1,2 The cyst

involves the odontogenic epithelia of impactedpermanent teeth, supernumerary teeth, odontomas,and, rarely, deciduous teeth.3-5 The mandibular thirdmolars are the most frequently involved, althoughthere is a marked incidence among mandibular secondmolars and premolars.6 Even though the developmentof cysts can be responsible for structural changes inthe bone,7 root resorption,8 and abnormal dentitiondevelopment, there is usually no pain or discomfortassociated with a cyst. Therefore, dentigerous cysts arefrequently discovered when radiographs are taken toinvestigate delayed tooth eruption, a missing tooth, or

tant professor, Department of Orthodontics, Graduate School of Medicine,stry and Pharmaceutical Sciences, Okayama University, Okayama, Japan.ciate professor, Department of Orthodontics, Graduate School of Medi-Dentistry and Pharmaceutical Sciences, Okayama University, Okayama,.ssor and chair, Division of Orthodontics and Dentofacial Orthopedics,u University Graduate School of Dentistry, Sendai, Japan.ssor and chair, Department of Orthodontics, Graduate School of Medi-Dentistry and Pharmaceutical Sciences, Okayama University, Okayama,.uthors report no commercial, proprietary, or financial interest in thects or companies described in this article.rted by the Japan Society for the Promotion of Science in the form ofs-in-aid for scientific research.t requests to: Yoshihito Ishihara, Department of Orthodontics, Graduatel of Medicine and Dentistry, Okayama University, 2-5-1 Shikata-Cho,u, Okayama 700-8525, Japan; e-mail, [email protected], revised and accepted, February 2010.5406/$36.00ight � 2012 by the American Association of Orthodontists..1016/j.ajodo.2010.02.043

malalignment. Most dentigerous cysts are single lesions.Meanwhile, bilateral and multiple cysts are rare and oc-cur typically in association with syndromes such as clei-docranial dysplasia, Maroteaux-Lamy syndrome, andbasal cell nevus syndrome.9-11 In the absence of thesesyndromes, bilateral dentigerous cysts located in themandible are uncommon, with few reports.10

Regarding orthodontic treatment for an impactedtooth with a dentigerous cyst, conventional treatmenttechniques have some limitations, including the age ofthe patient, tooth depth, tooth inclination, and eruptionspace. In some cases, extraction of the impacted toothmight be required, especially in adult patients. The auto-transplantation of teeth has been widely used in ortho-dontics to provide a treatment alternative in manysituations, mainly in patients with a severe impaction,early loss of permanent teeth, or congenital aplasia.12

Recently, the orthodontic miniscrew has provided rigidanchorage, made treatment more efficient, and alsomade biologically permissible movements possible.13-15

As a consequence, miniscrew use could change thetreatment paradigm and extend the scope oforthodontic mechanotherapy.

In this article, we report the successful treatment ofa patient with the unusual occurrence of nonsyndromicbilateral and multiple dentigerous cysts, in which themandibular left first molar was severely damaged.Combined treatments, including extraction, marsupiali-zation, orthodontic traction, and tooth autotransplanta-tion, were performed for the impacted premolars.Additionally, miniscrews were used to bring the trans-planted premolar to a suitable position in the mandibu-lar arch for favorable occlusion. Our treatment included

mailto:[email protected]

Fig 1. Pretreatment facial and intraoral photographs.

Ishihara et al 229

a consideration of the guidelines for multiple dentiger-ous cysts associated with mandibular premolars andmolars in young adults.

DIAGNOSIS AND ETIOLOGY

A boy, aged 13 years and 8 months, was examinedin the outpatient clinic of Okayama University Hospitalin Japan. A general dentist pointed out the impactedpremolars and introduced him to us at Okayama Uni-versity Hospital in Okayama, Japan. He had a symmet-rical face, a straight profile, and an obtuse nasolabialangle. The patient’s medical history showed nothing

American Journal of Orthodontics and Dentofacial Orthoped

unusual. There were no associated syndromes. Intraoralexamination showed the remaining mandibular seconddeciduous molars, missing second premolars, a dia-stema, a deep overbite, and a unilateral scissors-biteinvolving the left second molar (Figs 1-3). Thepretreatment panoramic radiograph and computedtomography scan (Fig 3, C-E) showed a well-definedradiolucent area surrounding the crown of the unerup-ted mandibular second premolars, the mandibularleft third molar, and the maxillary right second premo-lar. The distal root of the mandibular left first molarwas rebsorbed due to a dentigerous cyst associated

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Fig 2. Pretreatment dental casts.

230 Ishihara et al

with the mandibular left second premolar (Fig 3, C).The maxillary left second premolar was congenitallyabsent.

When compared with Japanese norms, the cephalo-metric analysis showed a tendency toward a skeletalClass III relationship (ANB, 1.2�) with mandibular ex-cess (SNB, 82.0�; Ar-Go, 64.6 mm; Go-Me, 82.2 mm)(Table I), a low mandibular plane angle (Mp-FH,12.7�), and an acute gonial angle (106.9�).16 The man-dibular incisors were proclined relative to the mandib-ular plane (L1-Mp, 106.0�). The molar relationshipswere Class II on both sides. The maxillary dental mid-line was almost coincident with the facial midline,but the mandibular midline was shifted 2.0 mm towardthe left. The maxillary anterior teeth were spaced. Thepatient showed no significant symptoms of a temporo-mandibular disorder. The incisal path was unstableduring opening-and-closing jaw movements with a 6degrees-of-freedom jaw movement recording system(Gnathohexagraph system, version 1.31; Ono Sokki,Kanagawa, Japan). The interincisal distance on maxi-mal opening without pain was 37 mm. An occlusalforce of 776 N and an occlusal contact area of 16.6mm2 (Table II) were calculated on an occlusal-force

February 2012 � Vol 141 � Issue 2 American

recording system (Dental Prescale & Occluzer; FujiFilm, Tokyo, Japan).

TREATMENT OBJECTIVES

The patient was diagnosed with an Angle Class IImalocclusion, a skeletal Class III jaw-base relationship,and multiple dentigerous cysts associated with the sec-ond premolars and the mandibular left third molar.The principal objectives were to create spaces for the un-erupted mandibular second premolars, then bring theminto the arch by using appropriate surgical techniques,and achieve acceptable occlusion with ideal overjetand overbite. The outline of the initial treatment planis shown in Figure 3, F. The distal root of the mandibularleft first molar, which was rebsorbed by the dentigerouscyst, underwent hemi-sectioning. Concerning the man-dibular right second premolar, we selected orthodontictraction after marsupialization of the cyst. In contrast,the mandibular left second premolar was brought intothe arch by autotransplantation. A miniscrew was im-planted to move the hemi-sectioned mandibular left firstmolar mesially, isolate it from the impacted teeth, andproduce adequate space for autotransplantation of theimpacted mandibular left second premolar.

Journal of Orthodontics and Dentofacial Orthopedics

Fig 3. A, Pretreatment cephalometric radiograph; B, pretreatment cephalometric tracing (solid line)superimposed on a mean profilogram (dotted line); C, pretreatment panoramic radiograph; D,pretreatment computed tomography image of the maxilla; E, pretreatment computed tomographyimage of the mandible (the images represent consecutive slices taken from the level of impactedpremolars); F, schematic outline of the initial treatment plan.

Ishihara et al 231

American Journal of Orthodontics and Dentofacial Orthopedics February 2012 � Vol 141 � Issue 2

Table I. Cephalometric summary

Variable Mean Initial Pre-edgewise PosttreatmentAngular (�)ANB 3.2 1.2 1.1 0.5SNA 81.5 83.3 83.3 83.3SNB 78.2 82 82.2 82.8Mp-FH 28 12.7 10.6 9.5Gonial angle 120.9 106.9 105.4 105.9U1-FH 112.4 103.1 114.7 110.8L1-Mp 95.2 106 103.7 110.7IIA 124.2 134 131 129Occlusal plane 15.5 10.5 7.5 10.7

Linear (mm)S-N 72.2 76.3 77.7 77.7N-Me 135.7 135.4 136 137.3Me/NF 74.6 71 71.2 72.1Ar-Go 53.2 64.6 67.3 70.1Ar-Me 115.6 123.1 126.5 129Go-Me 76.6 82.2 84 86Overjet 3.3 4.1 4.3 2.2Overbite 3.3 6.7 6.1 3.8U1/NF 32.4 34.1 31.3 32U6/NF 26.2 26.7 26.3 27L1/Mp 48.9 45.4 46.4 44.5L6/Mp 37.5 38.1 38.9 40.2

Table II. Changes in occlusal force, contact area, andmaximummouth opening after orthodontic treatment

Occlusalforce (N)

Occlusal contactarea (mm2)

Maximunopening (mm)

Initial 776 16.6 37Pre-edgewisetreatment

589 13.7 49

Posttreatment 1153 24.4 48

232 Ishihara et al

TREATMENT ALTERNATIVES

One therapeutic method proposed for the treatmentof an impacted mandibular left second premolar is or-thodontic traction after marsupialization of the cyst.However, the mandibular left second premolar wasdeeply and closely positioned next to the mesial rootof the mandibular left first molar. Another alternativewas surgical removal of the impacted teeth and replace-ment with implants or conventional prosthodontics aftergrowth completion.17 This alternative is consideredmostly for extremely malpositioned impacted teeth inuncooperative patients. The patient had a desire to pre-serve his own teeth as much as possible. After a thoroughdiscussion, he decided to undergo autotransplantation.

TREATMENT PROGRESS

Initially, the mandibular second deciduous molars andimpacted maxillary second premolar were extracted. A re-movable plate was used to correct the deep overbite andscissors-bite of the maxillary left second molar (Fig 4).

Then a marsupialization procedure was performedfor the cyst-associated mandibular right second premo-lar. Five months after marsupialization of the cyst, or-thodontic traction of the impacted tooth was initiated.A force was applied from the mandibular lingual archwith an elastic chain. Eight months after orthodontictraction, a 0.018 3 0.025-in preadjusted edgewise ap-pliance was placed sectionally on both mandibular mo-lars (Fig 4). After leveling and alignment with 0.016-in


nickel-titanium archwires, a miniscrew was implantedbetween the mandibular left canine and the first premo-lar (Fig 4). After placement of the miniscrew, a ligaturewire was used to connect the miniscrew to the mandib-ular first premolar to reinforce anterior anchorage. Lin-gual buttons were bonded on the lingual side of themandibular left first premolar and the hemi-sectionedleft first molar. Elastic chains were tied both labiallyand lingually from the mandibular left first premolarto the hemi-sectioned left first molar for mesial move-ment of the hemi-sectioned tooth (Figs 4 and 5).

Six months after mesial movement, adequate spacewas obtained for autotransplantation (Fig 5), and theimpacted mandibular left second premolar was trans-planted into the space. The autotransplanted toothwas fixed for 3 months by placing a 0.0175-in Respondarchwire (Ormco, Glendora, Calif) between adjacentteeth. Occlusal adjustment was performed to preventtraumatic occlusion. By the end of the fixation period,endodontic treatment was started for the autotrans-planted teeth. The pre-edgewise stage of cephalometricevaluation and superimposed cephalometric tracingsshowed a decrease in the ANB angle with mandibulargrowth (Table I).

The second phase of treatment began at age 16 years6 months (Figs 6-9). An 0.018 3 0.025-in preadjustededgewise appliance was placed for all brackets andbands. Initial alignment was achieved with 0.016-innickel-titanium archwires. The leveling phase was com-pleted with 0.0163 0.022-in nickel-titanium archwires.The leveling stage took 4 months. Then stainless steelwires were positioned to coordinate both arch forms.Stainless steel 0.017 3 0.025-in archwires were placedfor detailing. The total second phase of the treatmentperiod was 19 months. After removing the edgewise ap-pliance, the maxillary and mandibular teeth were stabi-lized by using bonded and Begg-type retainers. Fixedprosthetic restoration was performed for the hemi-sectioned and autotransplanted teeth to resolve tooth-size discrepancies.

TREATMENT RESULTS

After removing thefixed appliance, the impactedman-dibular left second premolar was successfully aligned in


Fig 5. Intraoral photographs during mesial movement of the hemi-sectioned tooth: A, start of mesialmovement; B, 1 month after the start; C, 4 months after the start; D, 6 months after the start.

Fig 4. Progress of initial treatment.

Ishihara et al 233

the mandibular arch after autotransplantation (Figs 10-12). The unilateral scissors-bite involving the left secondmolar was improved. The posttreatment panoramic radio-graph showed good root parallelism and nomarked apicalroot resorption or marginal or vertical bone loss of theperiodontal tissues (Fig 12). Simultaneously, a Class II mo-lar relationship was established on the right side with idealoverjet and overbite (Figs 10 and 11).

The posttreatment cephalometric evaluation anda superimposed cephalometric tracing showed that the


mandibular incisors were slightly inclined labially (L1-Mp,110.7�) and intrudedby1.9mm (Fig 13, Table I). The dentalmidlines were almost coincident with the facial midline.

When we evaluated jaw movements after treatment,we found that the interincisal distance on maximalopening without pain had increased to 49 mm. Boththe occlusal force and the occlusal contact area alsoincreased (Table II). With a 6 degrees-of-freedom jawmovement recording system, a smooth and stable mouthopen-and-close incisal path was achieved during


Fig 6. Pre-edgewise facial and intraoral photographs.

234 Ishihara et al

maximum open-and-close jaw movements (Fig 14, B).Additionally, increases in the condylar movement onboth sides during maximum mouth opening and closingwere observed.

Acceptable occlusion and a satisfactory facial profilewere maintained during the 2-year retention phase,although a minor dental midline deviation occurred(Fig 15). The patient and his parents were satisfiedwith the results of the treatment.

DISCUSSION

Various surgical approaches and orthodontic tech-niques have been suggested for the treatment of


impacted teeth. A dentigerous cyst can cause displace-ment or resorption of adjacent teeth, infection, andpathologic jaw fracture. Regarding the treatment of animpacted tooth with a dentigerous cyst in preadolescentchildren, removal of the entire cyst or marsupialization isthe main treatment to preserve the cyst-associated toothand promote its eruption. Orthodontic traction of theimpacted tooth has often been performed after marsupi-alization if needed.18,19 Meanwhile, surgical removal ofthe tooth (extraction) is the usual treatment for cyst-associated impacted teeth in adult patients. This casereport describes the management of a cyst-associatedimpacted tooth in a young adult.


Fig 7. Pre-edgewise dental casts.

Ishihara et al 235

Orthodontic treatment for the impacted tooth usu-ally includes opening an adequate space with a fixed ap-pliance to provide a more favorable alignment.Miniscrews have shown considerable potential asa straightforward alternative to simplify complicatedcases for orthodontists. Miniscrews have gained wideracceptance for absolute anchorage during various typesof tooth movement.13-15 We also used a miniscrew fororthodontic anchorage to solve the problem of spacemanagement and prevent some unintendedcounteractions, such as lingual tipping of the incisorsand aggravation of the dental midline deviation.Without miniscrews, it might have been difficult tocorrect the interincisal relationship and the dentalmidline in this patient. Therefore, our report alsodemonstrates the usefulness of miniscrews in thetreatment of cyst-associated impacted teeth.

Regarding both functional and esthetic aspects, fixedprosthodontic restoration such as a conventional bridge,a resin-bonded bridge, or a solitary implant is the treat-ment of choice when a single tooth is lost. Furthermore,tooth autotransplantation has become another methodof treating certain orthodontic complaints.12 Autotrans-plantation is one of the treatment alternatives for replac-ing a missing tooth when a donor tooth is available.20 An


advantage of this option is that it is a natural tooth ratherthan a prosthesis or an implant. Additionally, toothtransplantation allows dentofacial development andmaintains the alveolar bone volume. The success ratehas been reported to be excellent if the donor teeth aretransplanted before root formation is complete. Inaddition, some studies have reported that autotransplan-tation is a reliable method leading to a favorable progno-sis for donor teeth with closed apices if the teeth areendodontically treated.21,22 However, the long-termprognosis is questionable, with a potential for root re-sorption.23 In this patient, a cyst-associated tooth withcompleted root formation was autotransplanted and or-thodontically moved after the initial healing period. Asfor the impactedmandibular left secondpremolar, ortho-dontic traction after marsupialization is another recom-mended treatment to preserve the cyst-associated toothand promote its eruption. The successful eruption ofdentigerous cyst-associated premolars is associatedwithmany factors, including the age of the patient, toothposition, dental root formation, and mesiodistal angula-tion.24,25 Hyomoto et al25mentioned that the angulationand the position of the impacted mandibular premolar inthe alveolar bone were significant factors for eruption.The teeth showed less than an 80� tooth axis angulation


Fig 9. Superimposed cephalometric tracings show changes from pretreatment to pre-edgewisestages: A, sella-nasion plane at sella; B, palatal plane at ANS; and C, mandibular plane at menton.

Fig 8. A, pre-edgewise cephalometric radiograph; B, pre-edgewise cephalometric tracing (solid line)superimposed on a mean profilogram (dotted line); C, pre-edgewise panoramic radiograph.

236 Ishihara et al

February 2012 � Vol 141 � Issue 2 American Journal of Orthodontics and Dentofacial Orthopedics

Fig 10. Posttreatment facial and intraoral photographs.

Ishihara et al 237

or were less than 9mmdeep in bone. Based on the resultsof their study, it might be appropriate to consider the au-totransplantation of an impactedmandibular left secondpremolar by using the same criteria.

The autotransplanted tooth functioned over the 2years of retention. Jonsson and Sigurdsson26 stated that35 of 40 (87.5%) autotransplanted teeth reacted normallyto orthodontic treatment, and the transplantation did notaffect the long-term prognosis. However, further observa-tion of the autotransplanted tooth is required because thelong-term stability after treatment is unknown.


CONCLUSIONS

We treated a young adult patient with nonsyn-dromic bilateral and multiple dentigerous cysts. Thisreport demonstrates several combined treatmentapproaches for a cyst-associated impacted toothincluding the use of miniscrews, extractions,marsupialization, orthodontic traction, and toothautotransplantation.

We thank Joji Fukunaga for performing the surgicaltreatment.


Fig 11. Posttreatment dental casts.

238 Ishihara et al

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2. Aziz SR, Pulse C, Dourmas MA, Roser SM. Inferior alveolar nerveparesthesia associated with a mandibular dentigeneous cyst. JOral Maxillofac Surg 2002;60:457-9.

3. Miller CS, Bean LR. Pericoronal radiolucencies with and withoutradiopacities. Dent Clin North Am 1994;38:51-61.

4. Takagi S, Koyama S. Guided eruption of an impacted secondpremolar associated with a dentigerous cyst in the maxillary si-nus of a 6-year-old child. J Oral Maxillofac Surg 1998;56:237-9.

5. Mintz S, Allard M, Nour R. Extraoral removal of mandibular odon-togenic dentigerous cysts: a report of 2 cases. J Oral MaxillofacSurg 2001;59:1094-6.

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8. Gulbranson SH,Wolfrey JD, Raines JM,McNally BP. Squamous cellcarcinoma arising in a dentigerous cyst in a 16-month-old girl.Otolaryngol Head Neck Surg 2002;127:463-4.

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12. Andreasen JO, Paulsen HU, Yu Z, Bayer T, Schwartz O. A long-termstudy of 370 autotransplanted premolars. Part II. Tooth survivaland pulp healing subsequent to transplantation. Eur J Orthod1990;12:14-24.

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14. Roberts WE, Marshall KJ, Mozsary PG. Rigid endosseous implantutilized as anchorage to protract molars and close an atrophic ex-traction site. Angle Orthod 1990;60:135-52.

15. Kuroda S, Katayama A, Takano-Yamamoto T. Severe anterioropen-bite case treated using titanium screw anchorage. AngleOrthod 2004;74:558-67.

16. Wada K, Matsushima K, Shimazaki S, Miwa Y, Hasuike Y, Sunami R.An evaluation of a new case analysis of a lateral cephalometricroentgenogram. J Kanazawa Med Univ 1981;6:60-70.

17. Cronin RJ Jr, Oesterle LJ. Implant use in growing patients:treatment planning concerns. Dent Clin North Am 1998;42:1-34.

18. Clauser C, Zuccati G, Barone R, Villano A. Simplified surgical ortho-dontic treatment of dentigerous cyst. J Clin Orthod 1994;28:103-6.


Fig 12. A,Posttreatment cephalometric radiograph;B, posttreatment cephalometric tracing (solid line)superimposed on a mean profilogram (dotted line); C, posttreatment panoramic radiograph.

Fig 13. Superimposed cephalometric tracings show changes from pre-edgewise to posttreatmentstages: A, sella-nasion plane at sella; B, palatal plane at ANS; and C, mandibular plane at menton.

Ishihara et al 239


Fig 14. Condylar and jaw movements with a 6 degrees-of-freedom jaw movement recording system:A, pretreatment; B, posttreatment. C, Condyle; M, molar; I, incisor; R, right side; L, left side.

240 Ishihara et al

19. Sain D, Hollis WA, Togrye AR. Correction of superiorly displacedimpacted canine due to a large dentigerous cyst. Am J Orthod Den-tofacial Orthop 1992;102:270-6.

20. Schwartz O, Rank CP. Autotransplantation of cryopreserved toothin connection with orthodontic treatment. Am J Orthod Dentofa-cial Orthop 1986;90:67-72.

21. Lundberg T, Isaksson S. A clinical follow-up study of 278 au-totransplanted teeth. Br J Oral Maxillofac Surg 1996;34:181-5.

22. Tsurumachi T, Kakehashi Y. Autotransplantation of a maxillarythird molar to replace a maxillary premolar with vertical root frac-tures. Int Endod J 2007;40:970-8.


23. Paulsen HU. Autotransplantation of teeth in orthodontic treat-ment. Am J Orthod Dentofacial Orthop 2001;119:336-7.

24. Yahara Y, Kubota Y, Yamashiro T, Shirasuna K. Eruption predictionof mandibular premolars associated with dentigerous cysts. OralSurg Oral Med Oral Pathol Oral Radiol Endod 2009;108:28-31.

25. Hyomoto M, Kawakami M, Inoue M, Kirita T. Clinical conditionsfor eruption of maxillary canines and mandibular premolars asso-ciated with dentigerous cysts. Am J Orthod Dentofacial Orthop2003;124:515-20.

26. Jonsson T, Sigurdsson TJ. Autotransplantation of premolars topremolar sites. A long-term follow-up study of 40 consecutive pa-tients. Am J Orthod Dentofacial Orthop 2004;125:668-75.


Fig 15. Postretention facial and intraoral photographs.

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patient with nonsyndromic bilateral and multiple impacted ...an 0.018 3 0.025-in preadjusted...

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