Marsupialization of a dentigerous cystfollowed by orthodontic traction of tworetained teeth: A case report

Ivano Maltoni 1, Manuela Maltoni 1, Giorgia Santucci 1, Fabio Ramina 2, Luca Lombardo 2, Giuseppe Siciliani 2

1. Private practice, Viale Alfredo Oriani, 1, 47100 Forlì, Italy2. Postgraduate School of Orthodontics, University of Ferrara, via Luigi Borsari, 46,

44121 Ferrara, Italy

Correspondence:Fabio Ramina, via Luigi Borsari, 46, 44121 Ferrara, Italy.fabio.ramina@student.unife.it

Introduction

The dentigerous cyst (or follicular cyst) is defined as an epithe-lial-lined pathologic space that encloses the crown of animpacted, embedded, unerupted or developing tooth. The cyst,which is attached to the tooth at the cementoenamel junction, iscaused by fluid accumulation either between the reducedenamel epithelium and the enamel, or between the layers of

the enamel organ. The accumulation of fluid associated with thedegradation of the follicle cells causes an increase in osmoticpressure and causes the lesion to enlarge [1]. This is the mostcommon type of developmental odontogenic cyst, making upabout 20% of all epithelium-lined cysts of the jaws [2]; it isestimated that about 10% of impacted teeth can form a dentig-erous cyst [3]. The non-keratinized stratified squamous cellsforming the inner layer of the lesion are capable of producing

Available online: 22 April 2019

KeywordsOrthodonticsFollicular cystUnerupted teethImpacted teethMarsupialization

Summary

This case report discussed a combined surgical-orthodontic rescue of two impacted teeth in a largedentigerous cyst by the means of fixed orthodontic appliances. After careful evaluation of the 3Dradiographic exams, extraction of the deciduous elements was carried out, followed by marsupi-alization and orthodontic traction of the impacted teeth. Surgical procedures, pre- and posttreat-ment records and orthodontic biomechanical evaluations are discussed.

Mots clésOrthodontieKyste folliculaireDents non évoluéesDents inclusesMarsupialisation

Résumé

Ce cas clinique a montré le sauvetage orthodontico-chirurgical de deux dents incluses dans delarges kystes folliculaires dentifères au moyen d'un appareil multibague. Après une évaluationradiologique précise tridimensionnelle, l'avulsion des dents temporaires a été réalisée, suivied'une marsupialisation et traction orthodontique des dents incluses. Le protocole chirurgical,l'analyse des documents pré- et post-traitement et de la biomécanique utilisée ont été discutés.

tome 17 > n82 > June 2019https://doi.org/10.1016/j.ortho.2019.03.019© 2019 CEO. Published by Elsevier Masson SAS. All rights reserved.

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International Orthodontics 2019; 17: 365–374

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metaplastic changes and progressing to more aggressive lesionssuch as odontogenic keratocysts, ameloblastoma, mucoepider-moid carcinoma, or squamous cell carcinoma [4]. Althoughdentigerous cysts can be associated with any unerupted tooth,most often they involve mandibular third molars (65% of allcases) [2]. Other frequent sites include maxillary canines, max-illary third molars, and mandibular second premolars [5]. Den-tigerous cysts rarely involve unerupted deciduous teeth [6], butthey may also involve unerupted supernumerary teeth or odon-tomas [7]. They are diagnosed most frequently in patientsbetween 10 and 30 years of age and there is a slight malepredilection [8]. This cyst is usually asymptomatic and discov-ered incidentally by routine radiographic examination; however,pain may occur when it becomes infected. After a long period oftime, it is likely to cause significant bone resorption and corticalexpansion, resulting in teeth displacement, particularly in thecyst-associated tooth. The radiographic appearance of a dentig-erous cyst is a well-circumscribed unilocular pericoronal radio-lucency (usually more than 5 mm) associated with an uneruptedtooth [9].The treatment for dentigerous cysts depends on their sizeand location. Moreover, the clinician should also considerthe patient's age, the teeth involved, and the involvementof other anatomic structures as basic criteria for choosingthe appropriate treatment modality [10]. A common treat-ment for dentigerous cysts is surgical enucleation of thelesion, together with removal of the unerupted tooth. If

eruption of the involved tooth is considered feasible, thenpartial removal of the cystic wall may be performed, leav-ing the tooth in place; in other cases, an orthodontictreatment is needed to assist eruption [11]. Marsupializa-tion or decompression is considered a more conservativeintervention with respect to the crowns of the developingpermanent teeth, generally preferred in children and in thecase of large dentigerous cysts. The aim of marsupializationis to relieve intracystic pressure through an accessory cavity,and to reduce the size of both the lesion and the bonedefect. It is also believed that a shorter decompressionperiod is needed for young patients. The cyst can thenbe removed at a later date, with a less extensive surgicalprocedure [2,12].

Case reportPatient evaluationThe patient (M. M., female, 13y 5m) presented at our practice,after being referred by her general dentist for the treatment of acystic-like formation, incidentally, discovered during the execu-tion of a panoramic radiograph; the lesion appeared to cause theretention of upper right canine and second premolar (13 and 15,FDI notation).She had already undergone a series of radiographic evaluationsby her previous dentist: two panoramic radiographs (performedat 10y 10m and 13y 1m) and a cone beam TC (12y 11m)(figure 1); those exams showed the evolution of the lesion,

Figure 1Radiographic exams showing the gradual enlargement of the cyst. On the left: first (upper) and second panoramic radiograph (lower),performed at 10y 10m and 13y 1m, respectively. On the right: cone beam CT, performed at 12y 11m, showing the extent of thelesion involving the two retained teeth

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displacing the retained teeth in the depth of the maxillary bonecavity.At the time of this visit (figure 2), there were neither symptomsnor noticeable facial asymmetries. Her medical history was non-contributory.Intraoral mucosa appeared to have no pathological signs. Thedental examination revealed the absence of 13 and 15, andthe presence of the deciduous 53 and 55, the latter presentingan occluso-mesial composite restoration. The lower midlinewas slightly deviated to the right, and there was a substantialanterior deep bite. The antero-posterior arch relationshipshowed a bilateral I molar and canine class. Both archesshowed moderate crowding. All elements showed physiolog-ical periodontal probing and no mobility at clinical manipula-tion; the thermal tests of vitality were positive for all dentalelements.A lateral cephalogram was advised (figure 3, table I).

DiagnosisThe radiographic images revealed a well-circumscribed radiolu-cent lesion with a well-defined radiopaque margin located inthe right side of the upper jaw. The upper right permanentcanine and second premolar were severely displaced and

impacted. The corresponding deciduous teeth (53 and 55) didnot present any radiographically noticeable root resorption.The tentative diagnosis of a dentigerous cyst involving twopermanent teeth was made. Other possibilities included a dif-ferential diagnosis of an inflammatory cyst, a keratocyst and acystic ameloblastoma.

Treatment planThe patient's general dentist suggested the enucleation of thecyst, together with the removal of the retained upper canineand premolar, under general anaesthesia.Our treatment plan was different, and included the marsupi-alization of the lesion, followed by the recovery of the retainedteeth and their orthodontic rehabilitation within the dentalarch.

Treatment progressAt the age of 13y 7m the first surgery was performed.Preoperatively, the patient rinsed with 0.2% chlorhexidinemouthwash. The deciduous canine and second molar teethwere extracted under local anaesthesia (articaine with1:100,000 epinephrine). After the elevation of a mucoperiostalflap, the socket of the 55 was used to establish a communica-tion between the cyst and the oral cavity. The cyst was deroofed

Figure 2Initial records (13y 5m)

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and its lining was sutured to adjacent oral mucosa with 4 silksuture stitches (Ethicon 4.0) (figure 4). A 3 cm long, 3 mm wideuniversal polyethylene intravenous infusion tube (Med + SMedical Solutions) was inserted into the cavity to secure thecyst decompression [13]. The tube was settled with a stainless-

steel ligature wire (0.1000 Krugg ligature) to a sectional0.019 � 0.025 SS connected to bands on 16 and 14 (figure 5).The excised lining was sent to the pathology laboratory forhistopathologic examination: the result confirmed the diagno-sis of dentigerous cyst.

Figure 3Pretreatment lateral cephalogram (left) and cephalometric analysis (right)

TABLE IPretreatment cephalometric analysis.

Pretreatment Normal value

Sagittal skeletal relation

Maxillary position (S-N-A) (8) 84.1 82.0 � 3.5

Mandibular position (S-N-Pg) (8) 79.2 80.9 � 3.4

Sagittal law relation (A-N-Pg) (8) 4.9 1.6 � 1.5

Wits appraisal (mm) 4.4 �1.0 � 1.0

Vertical skeletal relation

Maxillary inclination (S-N/ANS-PNS) (8) 9.0 8.0 � 3.0

Mandibular inclination (S-N/Go-Gn) (8) 25.2 33.0 � 6.0

Vertical jaw relation (ANS-PNS/Go-Gn) (8) 16.2 25.0 � 5.0

Dento-basal relations

Maxillary incisor inclination (I-ANS-PNS) (8) 117.8 110.0 � 6.0

Mandibular incisor inclination (I-Go-Gn) (8) 95.3 94.0 � 6.0

Mandibular incisor compensation (I-A-Pg) (mm) �2.9 1.0 � 2.3

Dental relations

Overjet (mm) 3.7 2.5 � 2.5

Overbite (mm) 3.7 2.5 � 2.0

Interincisal angle (U1/L1) (8) 128.4 130.0 � 6.0

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After a healing time of 8 days, the stitches were removed.The patient was instructed to irrigate the cystic space dailywith normal saline by a syringe for a period of 6 months.After this period, a new panoramic and a periapical radio-graph were executed, where a reduction of the cystic lumenwas detectable, together with the distal drifting of the 13,causing the transposition of the element with respect to 14(figure 6).After 1 month, Damon Q passive self-ligating fixed appliances(Ormco, Orange, California) were bonded, with a pair of open-coil springs used to maintain the upper arch perimeter.After 9 months from the marsupialization, a second surgery wasperformed under local anaesthesia (Articaine with 1:100,000epinephrine). In fact, after a muco-periosteum flap was ele-vated in correspondence to the involved teeth, access to thecrown of the horizontally and mesially impacted 15 wasachieved. An orthodontic Damon Q self-ligating bracket wasthen applied and subsequently linked to the archwire by meansof appropriately modelled ligature that gave the initial ortho-dontic traction (figure 7). The traction was obtained by an elasticligature (Elastic Thread light TP Orthodontics, Inc.) replacedevery 2 weeks, while the upper arch was used as an orthodonticanchorage.The standard Damon archwire sequence was applied, untilalignment of the erupted upper elements was obtained;13 months after the first surgery, a new OPT was performed(figure 8) on which the surgical disinclusion of the transposed13 was programmed.The third surgery was then realized under local anaesthesia. Afull-thickness palatal flap was performed in order to expose thecrown of 13, where an orthodontic button was bonded. After

Figure 4a–b: appearance of the tissues after first surgery: a: cystmarsupialization and extractions of the deciduous canine andsecond molar; b: decompression tube and sectional wire in place

Figure 5Panoramic and periapical radiograph showing the transposition of 13

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that, a metal ligature emerging from the flap was applied, and4 reabsorbable sutures were applied (Vicryl TM 4-0 Atraloc TM).The metal ligature was then connected to interincisal area of the0.014 � 0.02500 NiTi archwire by the means of an elastic traction(figure 9).After 6 months, the tooth appeared to be erupted in the arch in apalatal position. The button was subsequently replaced with thecorresponding dedicated vestibular bracket. Subsequently, the0.019 � 0.025 SS archwire was bent and a palatal loop wascreated in the canine region (figure 10). The aim of this loop wasto exercise the appropriate traction direction in order to main-tain proper force direction until the canine was guided into itsfinal position within the alveolar bone.The traction related to the loop continued until full engage-ment of the archwire into the bracket of the canine becamepossible.The orthodontic devices were removed after 2 years and9 months and fixed lingual retainers were placed on theanterior teeth. All the elements included in the cyst appearedto be recovered in the alveolar bone and a satisfactory align-ment was achieved. The arch form did not allow a perfectsymmetry since the presence of the cyst prevented an ade-quate development of the dento-alveolar bone, especially inthe site of the canine prominence: this lack of support bonegenerated a slight compensative tipping of the canine, linkedto a limited deviation of the upper dental midline (figure 10,table II).

DiscussionIn the treatment of a dentigerous cyst, the benefits ofmarsupialization include a gradual decrease in the cysticcavity, preservation of oral tissues, maintenance of the vital-ity of dental elements, prevention of tooth extraction, pre-vention of lesions to the adjacent anatomic structures(inferior alveolar nerve, maxillary sinus, nasal cavity), pre-vention of mandible fracture and a decrease in the risk of

Figure 7Orthopantomogram (OPT) showing the progress of the extrusion of 15 and the position of transposed 13

Figure 6a–b: appearance of the tissues after second surgery; a: positionof the metal ligature between the archwire and the bracket on15; b: sutures and archwire in place

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recurrence. Proper maintenance and careful cleaning andrinsing of the cystic area from debris stagnation is importantto induce a more favourable tissue response and to preventinfection and alythosis [14–17].Lack of clinically useful evidence regarding the following tootheruption complicates the therapeutic decision [18].

Figure 8a–b: third surgery: a: exposure of crown; b: initial traction by themeans of elastic and metal ligature

Figure 9Position and shape of the canine loop

TABLE IIPretreatment and posttreatment cephalometric analysis.

Pretreatment Posttreatment Normalvalue

Sagittal skeletal relation

Maxillary position(S-N-A) (8)

84.1 84.2 82.0 � 3.5

Mandibular position(S-N-Pg) (8)

79.2 80.1 80.9 � 3.4

Sagittal law relation(A-N-Pg) (8)

4.9 4.1 1.6 � 1.5

Wits appraisal (mm) 4.4 3.4 �1.0 � 1.0

Vertical skeletal relation

Maxillary inclination(S-N/ANS-PNS) (8)

9.0 7.6 8.0 � 3.0

Mandibular inclination(S-N/Go-Gn) (8)

25.2 24.2 33.0 � 6.0

Vertical jaw relation(ANS-PNS/Go-Gn) (8)

16.2 16.7 25.0 � 5.0

Dento-basal relations

Maxillary incisor inclination(I-ANS-PNS) (8)

117.8 122.4 110.0 � 6.0

Mandibular incisorinclination (I-Go-Gn) (8)

95.3 95.2 94.0 � 6.0

Mandibular incisorcompensation (I-A-Pg) (mm)

�2.9 �2.2 1.0 � 2.3

Dental relations

Overjet (mm) 3.7 4.3 2.5 � 2.5

Overbite (mm) 3.7 1.9 2.5 � 2.0

Interincisal angle(U1/L1) (8)

128.4 123.2 130.0 � 6.0

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Many cases previously reported have already shown goodresults in preserving cyst-associated teeth when marsupializa-tion only is performed [19–23]. Previous studies supported theeffectiveness of cyst marsupialization, considering whether thepatient is older or younger than 10 years old, axis inclination ofthe tooth, root maturity and space availability as predictiveindicators of further tooth eruption [18,24]. In contrast, otherauthors [25,26] found that these factors are insignificant and donot affect tooth eruption.However, tooth eruption might not always occur after mar-supialization: it has been reported that the eruption rateafter marsupialization ranges from 31 to 89.4% [25–28]. Forthose cases where no eruption occurs, a period of 100 days

after marsupialization is suggested as critical time fordeciding the possibility of using orthodontic traction: ifthere is enough space for eruption, orthodontic tractionof the cyst-associated permanent tooth should not be initi-ated earlier than 3 months after marsupialization of a den-tigerous cyst in preadolescents [29].Orthodontic traction of an impacted tooth has often been per-formed after marsupialization in patients with a large cyst, acyst-associated tooth with a matured root, or an ectopic eruptedtooth [22,30–33]. Our case seemed to belong to this category,considering that the two retained teeth were unable to spon-taneously erupt after marsupialization. As a matter of fact,6 months following the marsupialization, the angulation of

Figure 10Final records at the end ofthe treatment

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the second premolar was still on a "collision course'' with thecanine root; likewise, the transposition of the canine with re-spect to the first premolar was worsening. Thus, the lonemarsupialization was not enough to guarantee the eruptionof the retained teeth: therefore, an orthodontic treatmentwas set out in order to guide the impacted teeth into theirnormal position.The final decision of performing a marsupialization was madeafter considering the aesthetic and functional effect that adifferent kind of surgery (enucleation of the lesion and of thetwo retained teeth) would have on such a young patient.Considering that, the large cyst would have left a large cavitywithin the maxillary bone that would have hampered thefollowing implant-prosthetic rehabilitation. We must also

remember the young age of the patient that would not havepermitted an immediate implant approach: this would have ledto the necessity of a series of partial prostheses for a long periodof time.

ConclusionThe combination of marsupialization with orthodontic extrusionseems to be an efficient protocol able to preserve cyst associatedeeply impacted teeth, promoting their eruption and bonehealing in preadolescent patients.

Disclosure of interest: the authors declare that they have no competinginterest.

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