functional and fixed orthodontic treatment in a child with cerebral palsy

CASE REPORT

Functional and fixed orthodontic treatmentin a child with cerebral palsy

Hakan Necip _Iscan,a Gamze Metin-G€ursoy,b and Selin Kale-Varlikc

Ankara, Turkey

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Cerebral palsy is a permanent neuromuscular motor disorder that results from injury in the developing brain dur-ing the prenatal or postnatal period. Common functional and craniofacial problems related to cerebral palsyinclude impaired swallowing, chewing, and speech; maxillary transverse deficiency; excessive anterior facialheight; and Class II malocclusion. This article reports the treatment of a 12-year-old girl with ataxic cerebralpalsy; she had a dental and skeletal Class II malocclusion, maxillary transverse deficiency, and severe crowdingin both arches. Treatment included rapid maxillary expansion with simultaneous functional therapy and fixedorthodontic extraction therapy in a period of 2 years 3 months. Vertical control was maintained by a vertical chin-cap. An acceptable occlusion and improvements in facial esthetics, speech, and oral function were achieved.(Am J Orthod Dentofacial Orthop 2014;145:523-33)

Acommon definition of cerebral palsy (CP) is a dis-order of movement and posture caused by adefect or lesion in the immature brain.1 A collab-

orative group called Surveillance of Cerebral Palsy inEurope has classified CP into 4 main types: spastic,ataxic, dyskinetic, and mixed.2 The most importantcauses of CP are premature birth,3 oxygen reduction inthe developing brain, and periventricular leukomalacia.Other possible causes include infections of the brain,toxemia of pregnancy, cerebral dysgenesis, kernicterus,poisoning with certain drugs and heavy metals, andhead trauma.4,5 CP was reported to be related to ClassII maloclusion.4,6 Rosenbaum et al7 and Franklin et al3

found that even though increased overjet and overbitewere common in CP patients, malocclusion prevalencewas similar to that of the healthy population. Intraoralproblems often seen in children with CP include poororal hygiene, periodontal disease, dental caries, bruxism,trauma, protrusion of the maxillary anterior teeth, exces-sive overjet and overbite, incompetent lips, openbite, and unilateral crossbite.3,4,7-10 Facial muscle

the Department of Orthodontics, Faculty of Dentistry, Gazi University,a, Turkey.rer.arch assistant.ciate professor.thors have completed and submitted the ICMJE Form for Disclosure oftial Conflicts of Interest, and none were reported.ss correspondence to: Gamze Metin-G€ursoy, Biskek cad. 1. Sok. No: 4,emek/Ankara, Turkey; e-mail, [email protected].

itted, revised and accepted, June 2013.5406/$36.00ight � 2014 by the American Association of Orthodontists./dx.doi.org/10.1016/j.ajodo.2013.06.025

impairments or habits related to jaw, lip, and tonguemovements in patients with CP can result in sucking,chewing, swallowing, and speech problems; drooling;tongue thrust; and temporomandibular disorders.4,11-13

Functional appliance therapy guides skeletal growthand modifies the patient's neuromuscular behavior,especially masticatory muscle activity.14-17 However,some have reported that children with neuromusculardiseases such as CP could not be treated successfullywith functional appliances.18 In a review of the literature,we found no study or case report regarding the benefitsand effects of functional or fixed orthodontic appliancetherapy in children with CP. The aim of this case reportwas to present an acceptable orthodontic treatment,including functional appliance treatment, of a patientwith ataxic CP.

ETIOLOGY AND DIAGNOSIS

A 12-year-old girl was referred to the orthodonticdepartment of Gazi University in Ankara, Turkey, fordiagnosis and treatment. Her parents' main complaintswere her severe crowding, buccally positioned maxillarypermanent canines, and unpleasant smile (Fig 1). Herparents reported that the pregnancy and parturitionwere normal, and she was born in the 40th week ofgestation. When she was 50 days old, after a regularfeeding session, she vomited and aspirated while asleep.She was quickly hospitalized. During this period, shesuffered from high fever. Subsequently, she was evalu-ated and treated by a multidisciplinary team includinga pediatrician, a physiotherapist, a psychologist, and aneurologist. Her diagnosis was ataxic CP and mental

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http://dx.doi.org/10.1016/j.ajodo.2013.06.025

Fig 1. Pretreatment facial and intraoral photographs.

524 _I scan, Metin-G€ursoy, and Kale-Varlik

retardation (IQ, 35-49). She had problems with balance,coordination, and speech; orthopedic problems; andstrabismus. Her walking and talking abilities were de-layed until 7 years of age because of her 76% physicaldisability.

In her oral examination, severe maxillary transversedeficiency with a transverse discrepancy,19 bilateral pos-terior crossbite, Angle Class II Division 1 malocclusionwith an excessive overjet and a normal overbite(Table), fractured permanent maxillary central incisors,and severe crowding in both dental arches were found(Fig 1). The dental cast analysis showed severe arch-length deficiencies in both arches (Fig 2). The handand wrist radiograph showed that her skeletal age was14 years with a remaining growth potential of only1.7% according to the method of Greulich and Pyle20

andMP3 union skeletal growth stage according to Graveand Brown.21 Evaluation of the panoramic radiographshowed no congenitally missing teeth, and no periapical

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pathologies, root abnormalities, or resorption was noted(Fig 3). The pretreatment cephalometric analysis showeda skeletal Class II relationship, a steep mandibular planeangle, reduced effective mandibular length, excessiveupper and lower anterior facial heights, a vertical facialgrowth pattern, and increased articular and steepocclusal plane angles. Both maxillary and mandibularincisor positions were acceptable (Table).

TREATMENT OBJECTIVES

The etiology could have been a combination of in-herited growth pattern, CP, and weakness of the buccalmusculature. The main objectives of the treatment wereto (1) expand the maxillary arch to favor the tonguemovements and overcome the swallowing, speech, andexcessive drooling problems; correct the bilateral poste-rior crossbites and also obtain a spontaneous forwardpositioning of the mandible by eliminating the

Journal of Orthodontics and Dentofacial Orthopedics

Table. Cephalometric analyses at pretreatement, endof phase 1, end of phase 2, and 1 year of retention

Pretreatment Phase1 Phase 2 PostretentionS-N (mm) 64.8 65.6 65.7 65.8NSAr (�) 122.5 120.5 122.2 122.1SArGo (�) 152.9 149.1 144.7 148ArGoMe (�) 131.4 134.9 136.7 132.9Sum of posterior angles

(NSAr + SArGo +ArGoMe) (�)

406.8 404.5 403.6 403

SN-GoGn (�) 44 41.9 41.1 40.7N-ANS (perp HP)

(mm)57 56.2 56.1 55.7

ANS-Me (perp HP)(mm)

65 66.1 66.5 65.9

SNA (�) 78 78 77.8 78.2SNB (�) 73 74.4 74.3 74.1ANB (�) 5 3.7 3.5 4.2Midface length

(Co-A) (mm)77.8 78.6 79.3 80.1

Mandibular length(Co-Gn) (mm)

111.9 115.4 115.6 116.4

U1-SN (�) 100.9 90.8 87 91.7U1-NA (mm) 3.6 2.4 0.7 1.5U1-NA (�) 23.3 12.7 9.2 13.5IMPA (�) 81.6 82.7 81.7 85.5L1-NB (mm) 2.9 3.1 3 3.6L1-NB (�) 21.2 21.5 19.6 22.6Transverse

discrepancy (mm)�8 2 0 �2

Arch length deficiencyMaxilla (mm) �15 �10 0 0Mandible (mm) �12 �12 0 0Overjet (mm) 7.5 5 2 4Overbite (mm) 1.5 1.5 2 2

Perp, Perpendicular; HP, horizontal plane.

_I scan, Metin-G€ursoy, and Kale-Varlik 525

functional interferences caused by maxillary constric-tion; (2) correct the Class II molar and canine relation-ships and the convex facial profile with functionaltherapy, which might serve as myofunctional therapyto achieve better masticatory muscle activity and stimu-late the forward growth of the mandible with the re-maining growth potential of 1.7%; (3) control thevertical growth; and (4) solve the arch-length discrep-ancy and align both dental arches by extracting the 4first premolars to achieve a better occlusion.

TREATMENT ALTERNATIVES

One benefit of maxillary arch expansion is that spaceis gained for better tongue movements; this can lead toimprovements in speech and swallowing and help elim-inate excessive drooling problems.22 The advantages offunctional therapy include improved masticatory muscleactivity, lessening of impaired chewing problems, andimproved esthetic appearance; these changes wouldprovide physiologic and psychologic advantages forthe patient and her parents. The severe mandibular

American Journal of Orthodontics and Dentofacial Orthoped

crowding would not permit a nonextraction treatmentin the mandibular arch. The only alternative treatmentmodality was orthognathic surgery, but we decided notto postpone the presurgical treatment, which consistedof rapid maxillary expansion (RME) and fixed appliances.

TREATMENT PROGRESS

Treatment was carried out in 3 phases: (1) RME andfunctional therapy with a modified Twin-block appli-ance used simultaneously with RME and an extraoralvertical chincap appliance; (2) fixed appliance treatmentwith the extraction of the 4 first premolars and an ex-traoral vertical chincap appliance; and (3) retention. Allclinical procedures were carried out by the second author(G.M.-G.). The diagnostic, radiographic, and clinical rec-ords before and after these 3 stages were collected andevaluated.

Inphase 1, treatment beganwithRME,whichwasusedin 2 stages because of the severe narrowness of themaxil-lary arch. The first RME appliancewas an acrylic cap splinttype with a frame constructed with 0.8-mm round stain-less steel hard wire, activated at 1 turn per day until 8 mmof widening was achieved in a period of 1 month 13days.23 Next, an acrylic cap splint type of RME appliance,modified by the first author (H.N._I.) to serve as the upperpart of a Twin-block appliance, was bonded to use forfunctional therapy and also for further expansion of themaxillary arch to overcorrect by 5 mm; this took about 1month.24 Although the RME was discontinued after 13mm of widening, with an overcorrection of the transversediscrepancy, the appliancewas not debonded andwas leftin themouth for retention and to serve as the upper part ofthe Twin-block appliance for 3 months. Total durationwith the Twin-block therapy was 4 months. The patientwas asked to wear a vertical chincap applying 400 g offorce beginning from the first RME appliance to the endof the Twin-block therapy to (1) assist the anterior rota-tion of the mandible, (2) control the vertical increase ofthe posterior dentoalveolar and anterior facial heights,and (3) stabilize the mandible in forward and closed posi-tion by assisting themaxillary andmandibular bite-blocksof the Twin-block appliance to interlock at an angle of45�.25 Total duration of phase 1 of treatment was 6months 25 days. The acrylic splint-type RME appliancewas debonded, and a maxillary Essix plate (DENTSPLYLtd, Surrey, United Kingdom) was placed. At the end ofthis phase, the mandibular position and occlusion werenot stable because of crowding, and a mandibular shiftto the right was observed. Fixed appliance therapy inphase 2 of treatment would solve this problem.

In phase 2, the maxillary permanent first molars werebanded, and a transpalatal archwas inserted in the palatal

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Fig 2. Pretreatment dental casts.

Fig 3. Pretreatment cephalometric, panoramic, and hand-wrist radiographs.


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Fig 4. Facial and intraoral photographs at the end of phase 2 treatment.


tubes of these teeth to maintain the intermolar width.26

After the extraction of the 4 first premolars, the mandib-ular first molars were banded, and all the other teeth werebonded with 0.018-in slot Roth brackets (Masterbrackets, Roth prescription; American Orthodontics, She-boygan, Wis). The maxillary and mandibular arches wereleveled using 0.013-in copper-nickel-titanium and0.016-in, 0.018-in, 0.016 3 0.022-in, and 0.017 30.025-in nickel-titanium and archwires within 6months.The vertical chincapwas used until the end of the levelingphase. Then approximately 5mmof extraction space wasleft in the maxilla, and no space was left in the mandible.This space was closed using chain elastics on the 0.01630.022-in stainless steel archwire in the maxillary arch.The lower midline shift caused by the relapse of theRME was corrected with a transpalatal arch and inter-maxillary elastics. Because the patient could not attendher appointments for the next 3 months because of herorthopedic surgery, the fixed appliances were removed


without correction of the second-order positions of theincisors and any settling procedures. Total duration forthis phase (fixed appliance treatment) was 1 year 5months 22 days (Figs 4-6).

In phase 3, retention was started by full-coveragethermoplastic appliances (Essix retainers) in both themaxilla and the mandible with full-time wear exceptduring meals for 6 months and then half days for thenext 6 months.27 At the end of the retention period of1 year, the postretention records were collected(Figs 7-9).

TREATMENT RESULTS

The total treatment time was 2 years. During thisperiod the patient had 2 surgeries for strabismus. Atthe end of phase 1 of treatment, favorable tongue move-ments and speech changes were observed, and the swal-lowing and excessive drooling problems were improved


Fig 5. Dental casts at the end of phase 2 treatment.

Fig 6. Cephalometric and panoramic radiographs at theend of phase 2 treatment.



dramatically. At the end of treatment, the functional andesthetic objectives and a Class I molar relationship wereachieved. The posttreatment intraoral photos (Fig 4) anddental casts (Fig 5) show that transverse dimensions,molar and canine relationships, overjet, and overbitewere corrected. The occlusion showed acceptable inter-digitation and canine guidance. No root resorption orpathologic problems were observed at the end of treat-ment in the posttreatment cephalometric and panoramicradiographs (Fig 6). The cephalometric analysis at theend of phase 1 and the superimposition of the pretreat-ment and posttreatment tracings of phase 1 of treatment(Fig 10) showed that the increases of the upper and lowerdentoalveolar and anterior facial heights and the poste-rior rotation of the mandible were hindered. Thus, thefacial profile was improved by the forward mandibularautorotation, the maxillary and mandibular sagittal rela-tionships were corrected, and all results were stable dur-ing phase 2 of treatment (Table). During the retentionperiod, superimposition of the posttreatment and 1-year postretention tracings (Fig 11) showed a mildmaxillary anterior rotation resulting in relapse of theClass II canine and molar relationships.

DISCUSSION

Medical and dental interventions in children withataxic CP who have control and coordination impair-ments in jaws, lips, and tongue movements resulting


Fig 7. Facial and intraoral photographs at 1 year posttreatment.


in sucking, chewing, swallowing, drooling, tonguethrust, and speech problems are challenging for bothparents and clinicians.3,4,7,11 Because many of theseproblems relating to oral functions might be due tothe narrow maxillary arch and the retruded mandible,and taking into account the psychologic benefits thatmight be gained, we decided not to postpone RMEand fixed appliance treatment to the patient's laterstages of growth.22 However, it has been stated that chil-dren with neuromuscular diseases such as CP cannot betreated successfully with functional appliance therapy.18

Although the drooling problems in children with CP havebeen treated with Castillo Morales appliances or theirmodifications, no study or case report was found onfunctional or orthodontic treatment in subjects withCP.28,29 Weakness of the masticatory muscles inpatients with CP was reported.30,31 In contrast to thesefindings on muscle weakness in patients with CP, someauthorities have found that oral muscle activities arenot impaired, even in subjects who have global motor


involvement, and there is no difference inelectromyographic activity of oral muscles in differenttypes of CP. Some researchers have suggested thatgeneral motor involvement of these patients does notdirectly affect oral muscle performance.32,33 Althoughcontrol and coordination impairments in jaw, lip, andtongue movements were observed in our patient, wethought that functional treatment with a Twin-blockappliance supported by a vertical chincap after RMEwould help to achieve improvements in oral and perioralfunctions and chewing problems. The most dramaticchanges of the overall treatment were achieved at theend of phase 1, which lasted for 6 months 25 days.The maxilla was expanded by 13 mm, whereas forwardgrowth of the mandible was stimulated to reach Class Imolar and canine relationships with a modified Twin-block appliance working simultaneously with RME. Atthe end of phase 1, more space was gained for bettertongue movements, resulting in improvements of swal-lowing, speech, and excessive drooling. The advantages


Fig 8. Dental casts at 1 year posttreatment.

Fig 9. Cephalometric and panoramic radiographs at 1year posttreatment.



of functional therapy, such as better masticatory muscleactivity and improvement in impaired chewing prob-lems, were achieved. However, the superimpositions ofthe pretreatment and postphase 1 treatment showedthat the effect of the vertical chincap could not be de-nied; it inhibited the vertical growth of the posteriordentoalveolar structures and the posterior and backwardrotation of the mandible during the functional treat-ment (Fig 10).25 Effective mandibular length wasincreased, the mandibular plane angle was decreased,and the vertical growth pattern of the face was inhibited(Table). These changes were stable during the fixedappliance treatment, except for the saddle angle, whichincreased to its pretreatment value after phase 1 treat-ment. The maxillary and mandibular incisors were re-tracted during phase 2 treatment, but they tended torelapse in the retention period (Fig 11).

The most frequent symptoms of CP are increasedmuscle activity such as spasticity, dystonia, increasedreflexes, and loss of function such as weakness and lossof fine motor skills. Increased muscle activity mightshow favorable properties for treatment, although weak-ness and loss of fine motor skills can have negativefeatures.34 Increased muscle activity is due to hypersen-sitivity of the tonic stretch reflex pathways in patientswith CP.35,36 The effect of the activator on masticatorymuscle activity, which is used to modulate mandibulargrowth, has been reported extensively in previous


Fig 10. Superimpositions relating to pretreatment (black line), phase 1 (green line), and phase 2(red line).

Fig 11. Superimposition of the cephalometric radiographs before (black line) and after (green line) theretention period (phase 3).


studies.15-17 In subjects with CP, the tonic stretch reflexresponses are not present in lip or tongue muscles,37

but they are present in the jaw-closing muscles.38 There-fore, we suggest that in our CP patient, the tonic stretchreflex responses in the jaw-closingmuscles supported the


functional (Twin-block) therapy despite the loss of finemotor skills and weakness. This weakness could havebeen compensated by muscle overactivity. Furthermore,our patient had never needed medication to treatincreased muscle activity because her ataxic type of CP



did not cause severe loss of her fine motor skills. Hence,wewere encouraged to use functional treatment. Finally,further electromyographic studies and results in CP pa-tients are necessary to further comment on this issue.

CONCLUSIONS

Although no studies or case reports were found oneither fixed or functional orthodontic treatment effectsand benefits in children with CP, some studies showedpositive developments in children treated orthodonti-cally.39 However, no information on the kind oforthodontic therapy was found. Chadwick et al40 havetreated children with learning difficulties by fixed ortho-dontic therapy. They have claimed that although ortho-dontic treatment can provide both functional andesthetic improvements, it might not be the ideal. Eventhough our patient's treatment had to be finished soonerthan the time required because of her general healthproblems, both the patient and her family were satisfied.As a result, we affirm that orthodontic therapy isextremely effective in children with CP when it is appliedat the right time for each particular patient.

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functional and fixed orthodontic treatment in a child with cerebral palsy

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