treatment of skeletal open-bite malocclusion with ...treatment of skeletal open-bite malocclusion...

CASE REPORT

Treatment of skeletal open-bite malocclusionwith lymphangioma of the tongue

Chooryung J. Chung,a Soonshin Hwang,b Yoon-Jeong Choi,c and Kyung-Ho Kimd

Seoul, Korea

FromScienYonseaAssisbResidcClinidProfeReprinSevere-maiSubm0889-Copyrdoi:10

Lymphangioma of the tongue causesmassive tongue enlargement, leading to difficulties in swallowing andmas-tication, speech disturbances, airway obstruction, and skeletal deformities such as open-bite malocclusion.Early reduction of tongue volume improved the excessive open bite in a young girl, but it was not sufficient toredirect the original hyperdivergent growth pattern. Orthodontic camouflage treatment was therefore rendered.Long-term evaluation after tongue-reduction surgery and orthodontic treatment is presented. (Am J OrthodDentofacial Orthop 2012;141:627-40)

An open bite develops from a combination ofdental, skeletal, and environmental factors.1,2

Although simple open bites caused by dentalfactors are relatively easy to correct with favorableoutcomes, open bites with skeletal components areusually more difficult to treat with less stable results.2,3

Environmental factors, including aberrant neuromuscularfunction of the lip or tongue, abnormal tongue posture,and airway obstruction, can also increase the risk ofopen bite.1,2,4,5 Therefore, proper identification of theetiology and careful diagnosis are important in achievingoptimal treatment results. Here, we report the long-termoutcome of an open-bite malocclusion with macroglossiadue to tongue lymphangioma and hyperdivergent growthtreated with serial tongue-reduction surgeries and ortho-dontic camouflage treatment.

DIAGNOSIS AND ETIOLOGY

A girl, aged 3 years 5 months, came to the Depart-ment of Orthodontics, Gangnam Severance Dental Hos-pital, Yonsei University, in Seoul, Korea, seekingorthodontic treatment. She had been diagnosed withlymphangioma of the tongue. Macroglossia caused herto drool and have breathing difficulty. She also had

the Department of Orthodontics, Gangnam Severance Hospital, Oralce Research Center, Institute of Craniofacial Deformity, College of Dentistry,i University, Seoul, Korea.tant professor.ent.cal assistant professor.ssor.t requests to: Kyung-Ho Kim, Department of Orthodontics, Gangnamance Hospital, 211 Eonjuro, Gangnam-gu, Seoul, 135-720, Korea;l, [email protected], April 2010; revised and accepted, July 2010.5406/$36.00ight � 2012 by the American Association of Orthodontists..1016/j.ajodo.2010.07.029

a dental history of abnormal tongue posture. She hada convex profile with increased lower facial height andsevere anterior open bite (�13 mm). The deciduoussecond molars were the only teeth that were occluding.Several carious lesions were present in the maxillary arch(Figs 1 and 2). The panoramic radiograph showeda supernumerary tooth between the maxillary centralincisors. Lateral cephalometric analysis confirmeda skeletal Class II open bite with increased lower facialheight. The posteroanterior cephalometric radiographshowed slight mandibular asymmetry but otherwisewas within normal limits (Figs 3 and 4, Table).

The primary cause of the open bite was evidently re-lated to the macroglossia of the tongue due to lymphan-gioma.6-8 Lingual lymphatic malformation is frequentlylocalized in the anterior two thirds of the tongue andenlarges to a great extent after an episode of upperrespiratory tract infection.9 As the tongue swells, it cancause difficulties in swallowing and mastication, speechdisturbances, airway obstruction, and deformities ofmaxillofacial structures.10,11 Surgical removal is thetreatment of choice when the lymphangioma interfereswith esthetics or function.11,12 Spontaneous regressionof anterior open bite after glossectomy has beenreported in several patients,8,13 and self-correction hasalso been described throughout the growth period.14

We believed therefore that a decrease of the anterioropen bite could be expected after reduction of thetongue volume in our patient. However, the effects ofa glossectomy on the vertical skeletal pattern or changesduring facial growth are not yet fully understood.

TREATMENT OBJECTIVES

The main treatment objectives were to regain properfunction and to improve the occlusion early as possible.

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Fig 1. Initial photographs, age 3 years 5 months.

Fig 2. Initial dental casts, age 3 years 5 months.

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For these goals, reduction of excess tongue volume wasof paramount importance. If the hyperdivergent growthpattern remained, growth modification followed bycamouflage or surgical orthodontic treatment wouldalso be necessary to achieve ideal esthetics and function.

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TREATMENT ALTERNATIVES

To improve perioral function and to correct the openbite caused by the enlarged tongue, surgical excision ofthe lesion and tongue-reduction surgery were indicated.Spontaneous changes in occlusion after tongue reduction

Journal of Orthodontics and Dentofacial Orthopedics

Fig 3. Cephalometric and panoramic radiographs, age 3 years 5 months.

Fig 4. Cephalometric tracing, age 3 years 5 months.

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Table. Cephalometric analysis

Age

Initial Pretreatment Posttreatment 1-year retention

3 y 5 mo 12 y 1 mo 13 y 10 mo 14 y 10 moSNA (�) 84.5 78.0 76.0 77.0SNB (�) 74.5 73.5 72.0 73.0ANB (�) 10.0 4.5 4.0 4.0Wits (mm) �2.5 3.0 �1.5 �3.0Gonial angle (�) 112.0 135.5 135.5 135.0SN-MP (�) 55.0 55.5 55.5 54.0FMA (�) 45.8 45.3 46.3 46.2Posterior facial/anterior facial height 52.3 54.7 55.4 55.3Ramus height (mm) 31.9 41.4 45.6 46.4Overbite depth indicator 52.8 55.5 58.3 64.5U1 to SN (�) 96.0 120.0 98.5 102.0IMPA (�) 83.0 66.5 72.0 76.0

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and continued eruption of the permanent dentition wereexpected, so careful follow-up of growth was plannedbefore considering any active orthodontic treatment.Surgical extraction of the supernumerary tooth was alsoadvised before the eruption of the maxillary centralincisors.

If the hyperdivergent growth pattern remained dur-ing the follow-up period, several methods for growthmodification could be considered. In patients withlong-face syndrome combined with a Class II skeletalgrowth pattern, a high-pull headgear or a maxillarysplint could be used to prevent excessive vertical growth.Another option would be to use functional appliancescombined with a high-pull headgear for maximumvertical growth control.

Based on the severity of the vertical skeletal problemsand the amount of remaining open bite after growth ob-servation and modification, camouflage treatment witha fixed appliance accompanied by either nonextractionor extraction or surgical correction could be consideredto achieve ideal esthetics and function.

After a series of tongue surgeries, the patient and theparents were clearly reluctant to accept additional surgi-cal treatment. Therefore, growth modification withheadgear during the growth period was emphasized,followed by orthodontic camouflage treatment.

TREATMENT PROGRESS AND RESULTS

The patient had several tongue-reduction surgeriesbefore she was 9 years old. Progressive open-bite closurecould be seen after the tongue-reduction surgeries; thisimplied that the macroglossia was a major etiologic fac-tor for the extreme open bite. The supernumerary toothwas also surgically extracted.

By the time the patient was 10 years 4 months old, allpermanent teeth except the maxillary and mandibular


right canines had erupted. The skeletal age of the patientreached stage 3 of the skeletal maturity index of Fish-man15 at this point. Since a hyperdivergent growth pat-tern was evident even after the decrease in open bite,orthopedic growth modification with a maxillary splintand high-pull headgear was attempted before thegrowth peak started at skeletal maturity stage 4. Therewas dental midline deviation to the right (2 mm) witha lack of canine space on the right side, leaving bothmaxillary and mandibular canines impacted (Fig 5). Con-sidering the amount of space needed for the canineswith correction of the midline and the large overjet(8.0 mm), first premolar extractions were indicated.The maxillary and mandibular right first premolarswere extracted first to guide the canines to erupt alongwith continuous use of the high-pull headgear. How-ever, the patient did not comply well with the appliance,and there was little orthopedic effect, if any. Cephalo-metric superimposition throughout the period con-firmed the persistent hyperdivergent skeletal growthpattern (Fig 6).

At 12 years 1 month of age, the patient was reex-amined for the second phase of orthodontic treatment.She had a long face, mentalis muscle strain, right shiftof the dental midlines of both arches, and a large overjetof 8.5 mm (Fig 7). According to the dental cast analysis,there were a 1-mm arch-length discrepancy in the max-illary arch and a 6.5-mm discrepancy in the mandibulararch (Fig 8). Cephalometric analysis indicated that thepatient was skeletal Class II with a hyperdivergentgrowth pattern and proclination of the maxillary inci-sors. The posteroanterior radiograph showed a right shiftof the dental midline in both arches. According to thehand-wrist radiograph, the patient was in stage 8 ofskeletal maturity, and menarche had already occurred(Figs 9 and 10, Table).


Fig 5. Intraoral photographs and panoramic radiograph at age 10 years 4months. Canines on the rightside are impacted.

Fig 6. Cephalometric superimpositions from 3 to 12 years, showing the results of tongue reduction.

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Fig 7. Pretreatment photographs, age 12 years 1 month.

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The treatment plan for the second phase of ortho-dontic treatment was established. Additional extractionsof the maxillary and mandibular left first premolars wereplanned for correction of the midline, alignment, andoverjet. A high-pull headgear was advised to controlthe remaining vertical growth. The patient once againdid not cooperate with the high-pull headgear, and ver-tical growth control was compromised. After premolarextraction, fixed appliances were bonded. Both archeswere aligned, and space closure and midline correctionwere continued. After 1 year 9 months of orthodontictreatment, overbite and overjet improved, resulting inClass I molar and canine relationships. Lip closure be-came much more natural after orthodontic treatment,but the mentalis muscle strain still remained (Figs 11and 12). Maxillary anterior proclination was corrected,and the dental and facial midlines were coincident.From the hand-wrist radiograph, radius fusion hadstarted, indicating that the growth was almost complete(Figs 13 and 14). According to the superimposition, the


open bite had been successfully managed with thecamouflage treatment; however, vertical growthcontrol showed limited results because of the lack ofpatient cooperation (Fig 15).

After treatment, maxillary and mandibular fixed re-tainers were bonded, and removable circumferentialretainers were used full time for the next 6 months.The occlusion remained stable after 1 year of retentionwithout any relapse (Figs 16 and 17). Occlusal forcemeasured by using the prescale system had alsoincreased after treatment and even during theretention period indicating functional improvement(Fig 18).16 Vertical growth still continued throughoutthe retention period, but overjet and overbite were main-tained (Fig 19).

DISCUSSION

Open bite develops by interaction of many etiologicfactors, andmalfunction of the tonguemight be a primaryreason for a skeletal open bite.1,2,17,18 For this patient,


Fig 9. Pretreatment cephalometric, panoramic, and hand-wrist radiographs.

Fig 8. Pretreatment dental casts.

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Fig 10. Pretreatment cephalometric tracing.

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lymphangioma of the tongue was a major factor thatcaused the open bite; tongue-reduction surgeries assistedin the progressive open-bite closure. Lymphangioma isa benign tumor of the lymphatic vessels.19,20 A majorityof cases are congenital, and 95% of the tumors occurbefore age 10. Lymphangioma of the tongue is a rarecondition, but it is reported to be the most commoncause of congenital macroglossia.7 According to ourlong-term evaluation, the early reduction of the tonguevolume was effective in the improvement of the excessiveopen bite, but it was not sufficient to redirect the originalhyperdivergent growth pattern.

Many reports have mentioned difficulty in treatingskeletal open bites in association with long-face syn-drome, which involves a dolichocephalic facial form,high mandibular plane angle, hyperdivergent skeletalgrowth pattern, and anterior open bite.21,22 Ourpatient demonstrated the typical traits of a Class IIskeletal pattern and long-face syndrome. Facial patternis reported to develop even before the first molar erupts.Also, patients with an open bite and increased lowerfacial height are reported to have earlier onset of pu-berty.23 This indicates that early orthopedic interventionis necessary for favorable craniofacial growth, andrandomized clinical trials have also reported the effec-tiveness of treatment in the mixed dentition.1,2

Therefore, attempts were made to redirect the growth


pattern before and during puberty. However, it wasunfortunate that we could not determine the effect ofearly growth modification treatment using high-pullheadgear, because of the lack of patient compliance.

The mandibular growth pattern should also be a con-sideration for favorable treatment results. The condylesof open-bite subjects are reported to grow posteriorly,leading to clockwise rotation of the mandible. Therefore,the second phase of orthodontic treatment involving ex-tractions should be postponed until after puberty to pre-vent unnecessary extrusion of the posterior teeth andfurther bite opening.24 In this patient, orthodontic treat-ment involving fixed appliances and premolar extrac-tions was postponed until after she had menarche toprevent further clockwise rotation of the mandible. Ifthe patient had cooperated with the high-pull headgear,treatment results could have been more favorable.

Skeletal open bite with excessive vertical growth canbe a challenge to the practitioner and patient alike, sincelong-term stability is a major concern regardless of thetreatment modality.25,26 The final occlusion was wellmaintained regardless of the remaining vertical growthin this patient. Improvement in function indicated bythe gradual increase in occlusal force could be taken asa positive sign of stability. However, considering thepatient’s skeletal age, minor growth might still remain,and it requires careful follow-up.


Fig 11. Posttreatment photographs, age 13 years 10 months.

Fig 12. Posttreatment dental casts.

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Fig 13. Posttreatment cephalometric, panoramic, and hand-wrist radiographs.

Fig 14. Posttreatment cephalometric tracing.

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Fig 15. Cephalometric superimposition before and after phase 2 treatment.

Fig 16. One-year retention photographs, age 14 years 10 months.

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Fig 17. One-year retention dental casts.

Fig 18. One-year retention lateral and panoramic radiographs and changes in occlusal force.

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Fig 19. Cephalometric superimposition, ages 3 to 14 years.

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CONCLUSIONS

Macroglossia caused by lymphangioma can be a ma-jor cause of an open-bite malocclusion. Thoroughevaluation of the etiology and its correction, properdiagnosis, and timely intervention produced a favorablefunctional outcome.

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American Journal of Orthodontics and Dentofacial Orthoped

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19. Rice JP, Carson SH. A case report of lingual lymphangioma pre-senting as recurrent massive tongue enlargement. Clin Pediatr(Phila) 1985;24:47-50.

20. White MA. Lymphangioma of the tongue: report of case. ASDC JDent Child 1987;54:280-2.


21. Denny JM, Weiskircher MA, Dorminey JC. Anterior open bite andoverjet treated with camouflage therapy. Am J Orthod DentofacialOrthop 2007;131:670-8.

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