“Whole Body” Mobility After One Year of Intraoral ApplianceTherapy in Children with Cerebral Palsy and Moderate EatingImpairment

Erika G. Gisel, PhD, OTR,1 Stephane Schwartz, DDS, MsD,2 Andrea Petryk, BSc, OT,1 Dianna Clarke, BSc, OT,1

and Hubert Haberfellner, MD31School of Physical and Occupational Therapy, McGill University, Montreal, Quebec,2Faculty of Dentistry, McGill University,Montreal, Quebec,3University Children’s Hospital, Innsbruck, Austria

Abstract. The reciprocal influence of body postures onthe oral structures, but also of the oral structures on bodypostures, has been proposed by clinicians and is takeninto consideration when treating children with poor pos-tural control and moderate to severe eating impairments.However, this relationship has not been rigorously in-vestigated. The purpose of this study was to documentthe possible relationships among oral-motor, postural,and ambulatory control. Ambulatory skills [exclusive useof wheelchair (w/c) vs w/c and ambulation], posturalcontrol when sitting, “pathologic” reflexes, and lip andtongue posture were recorded before and after one yearof therapy with an intraoral appliance (ISMAR) in 20children with cerebral palsy and moderate eating impair-ment. Significant improvement occurred in sitting (head–trunk–foot control) following one year of ISMARtherapy. Ambulatory status also significantly improvedabove the level of maturation. Half of the childrenshowed marked improvement in oral posture, i.e., theirresting mouth posture was closed rather than open. Theseresults support an hypothesis of interaction between oralstructures and postural control of the “whole body.” Fur-ther studies are needed to determine the controls of sucha relationship.

Key words: Sitting assessment — Postural control —Mobility — Eating — Food textures — Facial expres-sion — Deglutition — Deglutition disorders

The importance of positioning children with cerebralpalsy and poor postural control has long been recog-nized. Interaction with the physical [1–6] and social en-vironment [7,8] is thereby facilitated. However, only fewstudies address the significance of positioning for feed-ing [7,9,10]. A question not yet examined is whether onefunctional sitting position is sufficient for many differenttasks, or whether there may be a number of functionalpositions, each unique to a given task.

Morris and Klein [9] discuss the importance ofpostural adjustments to the facilitation of feeding. Theysuggest that the entire body, from the head to the feet,must be considered and that changes in one part of thebody will affect other parts, either supporting or imped-ing feeding. Casaer et al. [11] showed that postural con-trols are present in the neonate, but that they vary withbehavioral state and positioning and seem to be taskspecific, i.e., they change with feeding. Our heightenedawareness of the role of postural support/control hasevolved from our studies on the efficacy of oral motortherapies in children with CP and eating impairments[12–17]. In this domain, it has become clear that thesafety of ingestion is dependent on proper positioning ofthe head, neck, and trunk [18,19]. The effect of changesin positioning is immediate. Aspiration can be minimizedor eliminated when the position of the head, neck, andtrunk are adjusted to the child’s oral-motor skills. Thelong-term effects of using an optimal position on thehealth of children with severe feeding impairments arecurrently under investigation [20].

The influence of body postures on the oral struc-tures and of oral structures on body postures has beeninvestigated. Movements of the oral structures affect theelectromyographic activity of the neck musculature [21].

Supported by National Health Research and Development Program No.6605-4086-59.Correspondence to:Erika G. Gisel, Ph.D., School of Physical & Oc-cupational Therapy, McGill University, 3654 Drummond St., Montre-al, Quebec, H3G 1Y5, Canada. E-mail: egisel@po-box.mcgill.ca

Dysphagia 15:226–235 (2000)DOI: 10.1007/s004550000032

© Springer-Verlag New York Inc. 2000

Effortful head lifting from a prone position is associatedwith an open mouth posture in neonates [11]. Openmouth posture, with drooling and tongue thrust, in thechild with CP is perceived as a distortion of facial ex-pression and may constitute one of the most severehandicaps the child with CP experiences [22]. This ab-normal open mouth posture is associated with depressionof the jaw, contributes to loss of food and liquid duringmeals, and generates occlusal disorders [23]. Hypotonic-ity of the lips and jaw further aggravate the open mouthposture [24]. Haberfellner et al. [14] reported that oneyear of intraoral appliance (ISMAR) therapy resulted insignificant improvement in jaw stability. Thus, greaterjaw stability may provide the basis for a more closedmouth posture and may have an effect on the perceptionof a more normal facial expression. Such changes infacial expression will be reported in this article.

Another benefit of intraoral appliance therapywas significant improvement in functional feeding skillsin children with moderate dysphagia [14]. We showedpreviously [25] that changes in functional feeding skillsare associated with the ability to eat more challengingfood textures, i.e., a change from pureed and hashedfoods to soft cooked solids. While this change was astated goal in our earlier study [25], in the presentstudy it was an expected outcome following one year ofISMAR therapy. Hence, no specific instructions weregiven to caregivers, but their intuitive reaction to thechild’s improved feeding skills was to change the foodtextures offered. These changes were documented.

We described the reciprocal relationship betweenpostural control and ingestive skills of children with CPand moderate dysphagia. Evidence for a “whole-body”relationship, as suggested by Morris and Klein [9], fur-ther comes from developmental studies by Touwen [26]that showed a significant correlation between the devel-opment of sitting and walking. Therefore, the purpose ofour study was to explore this “whole-body” relationshipamong oral-motor, postural, and ambulatory control. Wepropose that improvements in oral-motor skills will beassociated with improvements in postural control andambulatory skills following one year of intraoral appli-ance therapy in children with cerebral palsy and moder-ate dysphagia.

Method

Sample

Twenty children (11 girls and 9 boys) participated in this study. Theyranged in age from 4.2 to 13.1 years with a mean age of 8.3 ± 0.9 yearsat the onset of the study. All children had a diagnosis of cerebral palsywith tetraparesis and moderate motor impairment [27]. At the onset ofthe study, 14 children used a wheelchair exclusively as their means of

transportation. Four children used a wheelchair for long distance trans-port but began to use assistive devices such as tricycles, walkers, andhand-holding for ambulation indoors. One child had no wheelchair butused a tricycle, and 1 child was ambulatory. Eleven of the 20 childrenwere fully dependent in their daily living activities, including feeding,while the remaining 9 needed partial assistance. Fourteen wore diapersregularly, and the remaining 6 were able to indicate when they neededto go to the bathroom. As a group, children ate a solid texture within 1SD of the norm of the Gisel video assessment (GVA) [28,29], but were2 SD above the norm for puree. Children as a group were 1.5 SD belowthe mean in weight-for-age, and skinfolds (triceps, subscapular) werebelow the 50th centile-for-age. All children were able to breathethrough their nose [30].

The state of dentition of the sample was as follows: 8 children,(6 boys and 2 girls) were in primary dentition. Seven children (3 boysand 4 girls) were in early mixed dentition, meaning that the four firstpermanent molars as well as the eight permanent incisors had erupted,but the primary canines and primary molars were still in place. Twochildren (girls) were in late mixed dentition, meaning that most of theremaining primary canines and molars were replaced by the secondaryteeth, but some primaries were still in place. Finally, 3 children (girls)had their permanent dentition. None of the children had other oralappliances or orthodontic work done during the course of this study.

In terms of cognitive development, children varied from no tosevere impairment, based on their ability to respond consistently toquestions which required a yes or no answer related to personal needssuch as eating, drinking, and toileting. Ten children received medica-tion to control seizures and ten did not. Commitment by parents/caregivers to long-term appliance treatment was essential. Thus, care-givers that had shown consistency in other aspects of long-term care fortheir child were approached to participate in this study. Children wererecruited from special schools of the greater Montreal area betweenFebruary 1994 and February 1996. Both institutional and parental con-sent was obtained before entry into the study.

Study Design

Children were randomized to immediate ISMAR treatment (InnsbruckSensoriMotor Activator and Regulator; group A,n 4 10) or control(standard rehabilitation at school, group B,n 4 10). Parents wereinformed that all children in the control group would enter the ISMARtreatment after the control period (6 months) and receive the sameintervention as group A, only later. All children received 12 months ofISMAR treatment. Phase I of treatment (6 months) focused on jawstabilization and phase II (6 months) on oral structure mobilization. Thefollowing abbreviations will be used for the time intervals: control, M-6to M0; phase I, M0 to M6; and phase II, M6 to M12.

All children received their ISMAR at month 0. Following awear-adjustment period (see Protocol below), children began phase I assoon as they had reached 20 minutes of unassisted, continuous wear ofthe ISMAR.

Protocol

Children in both groups underwent the same series of assessments atM0, M6, and M12; only group B received its first assessment at M-6.

Testing in the School Environment

Postural/Ambulatory Skill MeasurementsThese tests focus on quality and duration of head and limb control andfrequency of pathologic movements of the axial skeleton and extremi-

E.G. Gisel et al.: Oral Appliance Therapy and Whole-Body Mobility 227

ties. All tests are reliable in the 0.87–0.91 range [4]. The term “patho-logic movements” (reflexes) is used loosely and is professional jargon.The movements described are tonic reflexes, existing on a continuumfrom normal to abnormal, and are part of the postural repertoire. Thesemovements (reflexes) do not dominate posture under normal conditionsbut may be uncontrolled (i.e., pathologic) in children with cerebralpalsy. The established terminology is used in this article.

Postural skills were scored from a video recording focusing onthe child seated in his/her wheelchair, secured only with a lap belt.Each body side was recorded for 5 minutes. Postural control was mea-sured with the Sitting Assessment Scale [4]. Postural control is ex-pressed on a scale of 1 (none), 2 (poor), 3 (fair), to 4 (normal) anddetermined for the head, trunk, foot, arms, and hands. For manipula-tion, children were given four toys to manipulate in standard fashion.Data were computed for the two body sides and averaged. Duration ofhead control was scored over the 5 minutes of recording of each bodyside by determining how long the child could hold the head erect.

Counts of pathologic movements of the legs, knees, and head/back and the asymmetric tonic neck reflex (ATNR) were taken of thetwo body sides and averaged [4]. To elicit the ATNR, children wereoffered 5 small spoonfuls of applesauce with the spoon approachingfrom a 45° angle to the body midline, so that the head had to be turned.Another 5 spoonfuls were offered approaching the mouth from thebody midline.

Careful note was also taken of children’s ambulatory status ateach time point. Function was categorized as using wheelchairs exclu-sively or using wheelchairs for long-distance travel but assistive de-vices, such as tricycles, walkers, canes, or hand-holding, for shortdistances.

Facial ExpressionVideo photography was continued after postural testing and performedin standard fashion during the administration of five standard foodtextures [28,29]. Children were offered 5 spoonfuls of puree (unsweet-ened applesauce), 2 viscous textures (5 bites of fruit gelatin and 5raisins), and 2 solid textures (5 cereal rings and 5 bites of wheatbiscuit). For the facial analysis, only the three most advanced texturesa child was able to eat were analyzed, e.g., applesauce, raisins, andwheat biscuit, or applesauce, fruit gelatin, wheat biscuit. Raisins andthe wheat biscuit are the more advanced viscous texture and solidtexture, respectively. The analysis of facial expression focused on theposition of the lips: (1) open, (2) semiopen, and (3) closed, and theposition of the tongue: (1) resting beyond the lower lip, (2) resting onlower lip, (3) resting on incisal edge of teeth, and (4) resting behind theteeth. Higher values indicate a more normal position. We chose threeframes for each of the three textures for a total of nine frames. We alsochose frames before the first bite, before the third bite, and after thefifth bite.

Food Textures of the Lunch MealCareful note was made of all the foods caregivers sent to school forlunch and the textures recorded according to the definitions in theappendix. All foods were weighed before and after the meal. Theweight of the food remaining after the meal was subtracted from theinitial weight to determine the amount ingested. Children also lose foodduring ingestion. The amount is difficult to determine accurately be-cause some spills on bibs and some falls on the table or the floor. Also,spilled liquids evaporate over the course of the meal. For this reason,we felt that the chosen approach was as close to an accurate measure-ment as possible. The weight of each food was then expressed as apercent of the total (solids and liquids). This allowed us to determinethe changes in any of the textures eaten before and after treatment. Thecaloric value of the meal was determined using the ESHA Diet Anyl

Can IBM 1.1 program. Caloric consumption per kilogram of bodyweight was used as a standardized measure to follow intake over theone year of the study.

Testing at the Dental Clinic

All procedures pertaining to the evaluation, preparation, and follow upof ISMARs were performed in the dental clinic of the Montreal Chil-dren’s Hospital at M-6, M0, M6, and M12. They consisted of extraoraland intraoral photographs, dental casts which were subsequently putinto articulation, fabrication of ISMARs, and fitting. These procedureshave been described in detail by Haberfellner et al. [14].

Wear Adjustment PeriodFollowing fitting, children wore the appliance daily, increasing thelength of time until 20 minutes of continuous wear was reached. Onaverage, children took 128 ± 34 (SE) days. At this point ISMAR wearwas switched from day- to night-time wear [31,32].

Treatment Phase IPhase I of treatment began following the adjustment period. The ap-pliance was not worn when children had colds and needed to breathethrough the mouth. Treatment was resumed as soon as nasal breathingwas reestablished. Also, ISMARs were not worn during meals. Onechild did not complete phase I because the parents withdrew the childfrom the study.

Treatment Phase IIChildren were again evaluated after phase I (M6). Goals were deter-mined for each child according to his/her needs (e.g., tongue tip eleva-tion to the upper alveolar ridge, tongue lateralization, lip pursing, lipretraction). Grooves were drilled into the lingual part of the occlusalshelves or beads were attached to different loci to elicit tongue move-ments. Treatment compliance was described by Haberfellner et al. [14].

Data Analyses

Data ManipulationFirst, Sitting Assessment scores for each child were aggregated intotwo domain-specific (axial skeleton:head, trunk, and foot control;upper extremity:arm and hand function) average scores. Next, theirgroup scores were calculated for each of the two domains and thetreatment period. For these measures, differences between values ob-served in each child at subsequent visits were then calculated and thegroup-specific mean changes were estimated. Means for the four patho-logic reflexes (combined) were calculated for individual children andthen mean values were estimated for each group and treatment period.For the analysis of facial expression (lip posture, tongue position), dataof individual children are presented at M-6, M0, and M12. This choicewas based on the observation of wide variations between children. Twoindependent observers analyzed the tongue and lip postures of eachchild. Pearson correlations were performed to determine the interraterreliability of the results. Means for each food/liquid texture (% byweight) were computed in each of the groups and each time point.

Statistical MethodsData on children in each of the two study groups were analyzed bothseparately and jointly. Analyses focusing on group B only were usefulto compare changes observed during active treatment with “spontane-ous” changes occurring in the same group during the control phase

228 E.G. Gisel et al.: Oral Appliance Therapy and Whole-Body Mobility

(M-6 to M0). Separate within-group analyses allowed us to also assessthe comparability of treatment effects across the two groups and toinvestigate reasons for potential discrepancies. In contrast, the mainadvantage of joint analysis of the two groups was to enhance precisionof the estimated treatment effects and increase power of statistical tests.Pairedt-tests were used to test the hypothesis that the mean differencebetween subsequent values of a given measure was different from zero.Independent groupt-tests were used for between-groups analyses.

To investigate children’s ambulatory skills before and aftertreatment, a binary variable was defined (exclusive use of wheelchairvs. walking with or without an assistive device). The main motivationwas that limited sample size did not allow us to model more than twocategories of outcome. Analysis was performed using three time peri-ods (M-6 to M0, M0 to M6, M6 to M12) with a linear time contrast anddata pooled from groups A and B. Generalized Estimating Equation(GEE) analyses were performed to test the significance of the changesin walking status over time while taking into account the dependence ofsubsequent within-child observations [33]. A significance level of 0.05was used for all hypothesis tests.

Results

Postural and Ambulatory Skills

Table 1 illustrates postural control of our children whilesitting. There was a significant improvement in the con-trol of the axial skeleton (head, trunk, foot control com-bined,p 4 0.043) in group B between M0 and M12, aswell as a marginally nonsignificant improvement whenthe two groups were combined (A + B,p 4 0.071).Significant improvement was found in upper-extremitycontrol (arm and hand function combined) between M0and M12 in the combined group (A + B,p 4 0.049).Overall, group B performed in the mid “fair” range,showing a small decline during the control period butimprovement during the treatment phases. Group A per-formed in the mid “poor” to “fair” range, showingmarked improvement during phase I but no furtherchange during phase II of treatment. Further inspectionof the data showed no differences between the right andleft body sides on any of the postural control measures.Control of head posture (Table 2) was marked by a small(2%) decline between M-6 and M0 in group B, and an8% decline in the combined group (A + B) from M0 toM6, with contributions from both group A and group B.Analysis of differences between time intervals showedsignificant improvement in the duration (in seconds) ofhead control between M6 and M12 in the combinedgroup (Table 2,p 4 0.031). However, this representsonly a catch-up of the loss during M0 to M6 comparedwith the control period.

Table 3 illustrates the clinical evidence of changein mobility/ambulatory status. In group B at M-6, 4 chil-dren used wheelchairs and assistive devices. One child,designated as (+1) in Table 3, had no wheelchair but useda tricycle at M-6 and a walker at M0. He is again des-

ignated as (+1) at M0. There was a significant decreaseover time in the number of children who used wheel-chairs exclusively (p 4 0.028). The significance of thechange over time was confirmed by the general test ofassociation using Cochran–Mantel–Haenzel statistics (p4 0.018). In contrast, no evidence of changes was foundin group B during the control period (M-6 to M0), as theresults of both tests were definitely nonsignificant forGEE analysis (p 4 0.30) and McNemar (p4 0.31) test.

Facial Expression

Figure 1 illustrates lip posture and tongue position in oneof our subjects before and after intraoral appliancetherapy. In the frontal view (A), the typical open mouthresting posture with tongue protrusion over the teeth,better noted in the profile view (B), is well illustrated.Following one year of intraoral appliance therapy, thetongue can be observed resting behind the teeth (C), andthe most frequent resting posture of the lips is a closedmouth posture (D).

Figure 2 illustrates children’s changes in lip pos-ture and tongue position. The M-6 data point could notbe ascertained reliably. Of the ten children of group B,four had a complete data set and four an incomplete dataset on lip posture; two children were not cooperative andcould not be tested. Taking these limitations into ac-count, the mean changes in the complete data set (n 4 4)during the control period from M-6 to M0 were +0.11(puree), +0.17 (solid), and −0.10 (viscous), or +0.06 forthe three textures combined. A change during treatmentof +0.20 was regarded as clinically meaningful. A 20%change was chosen based on earlier results from ourISMAR studies that showed significant changes (14.5 ±3.8%) in functional feeding skills [14]. Thus, a 20%change was considered a conservative estimate for aclinically meaningful change. In each group at least halfof the children showed clinically meaningful improve-ment in lip posture [n 4 6 (A), n 4 5 (B)], whereas theother half either did not reach the 0.20 level of change (n4 2) or registered a small loss [n 4 1 (A), n 4 5 (B)].Overall, there were few changes in tongue position ingroup B because most children already held their tonguebehind the teeth. One child (No. 18) had a habit of play-ing with her tongue. Thus, her decrease reflects a mo-ment when she engaged in such play. When she wascalm, her tongue always rested behind her teeth. In groupA, tongue position also was unchanged with the excep-tion of child No. 5 who showed marked (0.80) improve-ment. His tongue thrust was very severe at the onset ofthe study and ISMAR therapy had a positive effect on hisresting tongue position.

E.G. Gisel et al.: Oral Appliance Therapy and Whole-Body Mobility 229

Interrater Reliability

Scores for lip posture and tongue position were corre-lated between the two observers (Table 4). Correlationcoefficients ranged from 0.89 to 0.99, indicating veryhigh agreement between observers (p < 0.0001). More-over, Table 4 shows that the mean ratings of the twoobservers were almost identical, further indicating highconsistency of their evaluations.

Food Textures Consumed

Table 5 illustrates the distribution of food textures con-sumed, expressed as a proportion (by weight) of liquids

Table 1. Sitting controla in children with cerebral palsy and moderate eating impairment before and after one year of intraoral appliancetherapy (ISMAR)b

M-6 M0 M6 M12

B A B A B A B

Axial skeletonHead 3.5 2.6 3.4 2.9 3.5 2.9 3.7

± 0.2 ± 0.4 ± 0.3 ± 0.4 ± 0.2 ± 0.4 ± 0.2Trunk 3.4 2.7 3.3 3.0 3.6 2.9 3.8

± 0.3 ± 0.4 ± 0.3 ± 0.4 ± 0.2 ± 0.4 ± 0.1Foot 3.8 3.2 3.8 3.4 3.9 3.4 4.0

± 0.1 ± 0.2 ± 0.1 ± 0.2 ± 0.1 ± 0.2 ± 0.0

Upper extremityArm 3.6 2.4 3.2 3.0 3.4 3.2 3.7

± 0.2 ± 0.3 ± 0.3 ± 0.2 ± 0.2 ± 0.3 ± 0.2Hand 3.2 2.5 3.0 2.7 3.0 2.6 3.2

± 0.2 ± 0.2 ± 0.3 ± 0.3 ± 0.3 ± 0.2 ± 0.3

aPostural control was scored on a 4-point scale where 04 no control, and 44 full control.bData represent mean ± SE; group An 4 10, group Bn 4 10. Scores are means of the right and left body side [4].

Table 2. Postural controla and “pathologic” reflexesb in children with cerebral palsy and moderate eating impairment before and after one year ofintraoral appliance therapy (ISMAR)c

M-6 M0 M6 M12

B A B A+B A B A+B A B A+B

Postural controlHead (s) 284.2 271.3 278.6 275.0 237.4 266.4 252.7 265.9 282.3 274.5

± 6.3 ± 16.0 ± 16.7 ± 11.3 ± 20.6 ± 15.2 ± 12.7 ± 15.2 ± 9.0 ± 11.8Axial skeleton 3.6 2.8 3.5 3.2 3.1 3.7 3.4 3.1 3.8 3.5

± 0.2 ± 0.2 ± 0.2 ± 0.2 ± 0.3 ± 0.1 ± 0.2 ± 0.3 ± 0.1 ± 0.2Upper extremityd 3.4 2.5 3.1 2.8 2.8 3.2 3.0 2.9 3.5 3.2

± 0.2 ± 0.2 ± 0.3 ± 0.2 ± 0.3 ± 0.3 ± 0.3 ± 0.2 ± 0.2 ± 0.2

Pathologic reflexesPathologic movements 0.89 1.28 0.48 0.88 0.63 0.45 0.53 0.86 0.51 0.68

± 0.28 ± 0.61 ± 0.20 ± 0.46 ± 0.35 ± 0.18 ± 0.19 ± 0.25 ± 0.19 ± 0.22% children with pathologic movements 0.70 0.70 0.50 0.60 0.44 0.60 0.52 0.89 0.60 0.73

± 0.15 ± 0.15 ± 0.17 ± 0.11 ± 0.17 ± 0.16 ± 0.18 ± 0.11 ± 0.16 ± 0.10

aPostural control: axial skeleton is expressed as a mean of head, trunk, and foot control (see Methods).b“Pathologic” movements are expressed as a mean of 4 reflexes (frequency of occurrency) counted during 5 min (see Methods).cData represent mean ± SE.dUpper extremity is expressed as a mean of arm and hand control (see Methods).

Table 3. Number (%) of children using a wheelchair exclusively (w/c)or wheelchair and ambulatory devices (w/c+) before and after one yearof intraoral appliance therapy (ISMAR)

M-6 M0 M12

w/c+ w/c w/c+ w/c w/c+ w/c

Group Aa — — 1b (10) 9 (90) 5 (56) 4 (44)Group B 4 + 1 (50) 5 (50) 4 + 1 (50) 5 (50) 5 (50) 5 (50)

Total 5 (50) 5 (50) 6 (30) 14 (70) 10 (53) 9 (47)

aOne child was withdrawn from group A after 6 months.bThis child was ambulatory throughout the study and was not using awheelchair.

230 E.G. Gisel et al.: Oral Appliance Therapy and Whole-Body Mobility

and solids (total4 100%). We assumed that these tex-ture distributions were similar to what children were of-fered at home because no requests were made for “spe-cial” food textures to be sent to school.

Liquid intake represented about 35% of total in-take in both groups of children. It consisted mostly ofthin liquids in group B and thin and heterogeneous liq-uids (mixture of thin liquids and soft solids) in group Aat the onset of the study (M0). A change in group A topredominantly thin liquids occurred at M6.

Pureesconstituted 13–23% of total food texturesconsumed. This percentage did not change over time ineither group A or B. Of note is that group A received atleast 5% more purees throughout the study than group B.This is consistent with our observation that the oral-motor skills were poorer in group A than in B [14]. Theconsistency of purees varied from thin and runny to thickand cohesive depending on children’s oral-motor skills.

The most marked changes were noted in the per-centage ofsoft solidsconsumed. No change was noted in

group B during the control period (17.1% M-6 vs. 15.1%M0), followed by a 10% increase during phase I (15.0%M0 vs. 25.4% M6). This is consistent with the significantimprovement in biting and chewing described during thesame period [14]. Although group A also experiencedimprovement during the M0 to M6 phase, a decreasefollowed in the M6 to M12 phase. Thus, this change was

Fig. 1. Photograph of a child before(A, B) and 12 months after treat-ment(C, D). Note the typical open mouth posture(A), with the tongueresting on the incisal edge of the teeth(B). C Tongue position behindthe teeth;D lips closed.[With permission fromJ Dent Child 66(3):180–187, 1999]

Fig. 2. Change in lip posture and tongue position between month 0 andmonth 12 of individual children with cerebral palsy and moderate eat-ing impairment. A change of 0.20 can be regarded as clinically mean-ingful for lip posture and tongue position. Child No. 9 was withdrawnfrom the study after 6 months.

Table 4. Comparison of scoring lip posture and tongue position be-tween two independent observers

Lip posture Tongue position

M0mean

M12mean

M0mean

M12mean

Observer 1 1.99 2.12 3.59 3.66SD ± 0.44 ± 0.47 ± 0.56 ± 0.45Observer 2 2.06 2.22 3.59 3.63SD ± 0.35 ± 0.37 ± 0.65 ± 0.51Correlationa 0.89 0.89 0.93 0.99pb <0.001 <0.001 <0.001 <0.001

aPearson correlation coefficient between the corresponding scores ofthe 2 observers.bp value for testing the null hypothesis that the 2 observers’ scoringsare independent (correlation coefficient4 0).

E.G. Gisel et al.: Oral Appliance Therapy and Whole-Body Mobility 231

not maintained, in contrast to group B. The increase inthe percent (15.0% vs. 25.4%) of soft solid textures wasachieved through a concurrent decrease in hashed tex-tures in group B (28.0% M0 vs. 14.7% M6). Group Ashowed a smaller gain of 5% between mashed (granularpuree) and hashed (finely cut) textures in phase II (M6 toM12). Finally, group B showed a very small gain infoods constituting a “regular table diet.” None of thechildren in group A received regular table diet foods.

Estimation of caloric intake from this one meal(lunch) indicated considerable variation from one testmeal to the next (M-6 to M12) within individual chil-dren. However, as a combined group (A + B), caloricintake per kilogram of body weight remained stable(Table 6). An effort was made to increase the caloricdensity of meals in children in group A who were morethan 2 SD below the mean in weight-for-age. This in-crease is reflected in the change between M0 and M6 andwas sustained at M12.

Discussion

Postural Control and Ambulation

Significant improvement occurred in group B in the con-trol of the axial skeleton during the 12 months of thestudy. This improvement was also noted clinically. Chil-

dren needed fewer adaptations on ambulation equipment,resulting in removal of back supports on tricycles or achange from use of a tricycle to ambulation with awalker. Our data further showed a significant changeover time in the number of children who began ambu-lating that could not be attributed to maturation alone.Literature on the development of body stabilization inspace suggests that stability arises from the hip, given asupport surface, and progresses to the shoulders and fi-nally to the head [36]. Hence, the progression of ourchildren is consistent with these findings.

Control of head posture, measured in a seatedposition, while children were manipulating small toys

Table 5. Distribution of food textures (%)a consumed by children with cerebral palsy and moderate eating impairment before and after one year ofintraoral appliance therapy (ISMAR)b

Group Time

Solid Liquid

Heterogeneous TotalPuree Mashed Hashed Soft Regular Thin Nectar Thick

B M-6 14.2 0.0 30.0 17.1 1.0 33.8 2.0 0.0 2.0 100.0± 5.2 ± 0.0 ± 6.9 ± 6.5 ± 0.7 ± 5.5 ± 2.0 ± 0.0 ± 2.0 —

B M0 18.7 0.0 28.0 15.0 2.0 33.9 2.2 0.0 0.2 100.0± 6.1 ± 0.0 ± 8.3 ± 5.8 ± 1.5 ± 3.7 ± 2.2 ± 0.0 ± 0.2 —

A 21.5 19.4 19.0 3.3 0.0 22.9 0.0 0.0 14.0 100.0± 9.1 ± 10.3 ± 10.9 ± 3.1 ± 0.0 ± 4.8 ± 0.0 ± 0.0 ± 8.8 —

A + B 20.0 9.2 23.7 9.5 1.1 28.7 1.2 0.0 6.8 100.0± 7.5 ± 7.6 ± 9.4 ± 5.0 ± 1.1 ± 4.5 ± 1.6 ± 0.0 ± 6.3 —

B M6 13.3 3.0 14.7 25.4 3.2 37.0 1.9 0.0 1.6 100.0± 4.0 ± 3.0 ± 7.6 ± 7.6 ± 2.2 ± 6.4 ± 1.8 ± 0.0 ± 1.6 —

A 19.7 19.2 12.4 9.4 0.0 33.8 0.0 0.0 5.6 100.0± 8.0 ± 8.4 ± 6.4 ± 5.0 ± 0.0 ± 5.1 ± 0.0 ± 0.0 ± 5.3 —

A + B 16.4 10.7 13.6 17.8 1.7 35.5 1.0 0.0 3.5 100.0± 6.1 ± 6.5 ± 6.8 ± 6.8 ± 1.6 ± 5.7 ± 1.3 ± 0.0 ± 3.7 —

B M12 16.1 0.0 22.3 26.6 3.1 29.9 0.0 2.1 0.0 100.0± 5.5 ± 0.0 ± 10.7 ± 8.5 ± 1.9 ± 4.7 ± 0.0 ± 2.1 ± 0.0 —

A 23.3 13.8 17.7 4.7 0.0 33.8 6.7 0.0 0.0 100.0± 5.7 ± 7.1 ± 6.8 ± 4.2 ± 0.0 ± 4.8 ± 6.4 ± 0.0 ± 0.0 —

A + B 19.5 6.5 20.1 16.2 1.6 31.8 3.2 1.1 0.0 100.0± 5.5 ± 5.2 ± 8.9 ± 7.5 ± 1.5 ± 4.7 ± 4.4 ± 1.5 ± 0.0 —

aFor definition of textures see Appendix.bData represent mean ± SD of intake by weight (liquids + solids + 100%).

Table 6. Estimated mean caloric intake (kcal/kg body weight) fromone meal for children with cerebral palsy and moderate eatingimpairmenta

M-6 M0 M6 M12

Group A — 19.7 24.3 24.6±10.0 ±11.4 ± 8.1

Group B 21.5 21.4 20.9 16.3± 5.6 ± 7.8 ± 8.3 ± 9.1

Group A + B 21.5 20.5 22.5 20.2± 5.6 ± 8.7 ± 9.7 ± 9.4

aData represent mean ± SD.

232 E.G. Gisel et al.: Oral Appliance Therapy and Whole-Body Mobility

showed a small decline (8%) during phase I but a catch-up (8%) during phase II of treatment. Overall, the meanduration of head control was 92% of the total time mea-sured, and the decline observed between M0 and M6may be a result of the small increase in the number ofchildren showing pathologic movements in group B be-tween M0 and M6. All postural control measures showedimprovement over time in both groups, and, therefore,they could not account for this decrease in the duration ofhead control. Furthermore, we did not find any differ-ences between the right and left body sides on any of thepostural control measures. Hence, we do not attribute toomuch significance to this change because children inboth groups “caught up” to the M0 level in phase II ofour study.

The significant changes in postural control andambulation from M0 to M12 in this study occurred aftersignificant changes in jaw stabilization and oral-motorskills were achieved between M0 and M6 [14]. Further-more, the significant improvement in the axial skeleton(head–trunk–feet) control and upper extremity controlfrom M0 to M12 lend some support to the postulatedreciprocal influences among oropharynx, shouldergirdle, and trunk [21]. It also supports the proposed“whole-body” association between oral-motor and pos-tural controls [9,35]. While this is a first attempt at dem-onstrating such an association following a therapeuticintervention, Dowling [36,37] noticed difficulties anddelay in ambulatory development in children with esoph-ageal atresia. The only factor he found that could accountfor these problems was the deprivation of nutritive suck-ing (development of oral-motor skills) these children ex-perienced in their early development (personal commu-nication). Beratis et al. [38] reported similar results. Cau-sal relationships for such a whole-body hypothesis aredifficult to establish in a human model because experi-mental deprivation of feeding to determine motor out-comes would be unethical. Animal models may be moreappropriate to work out the details of the developmentaland neuromuscular mechanisms of control.

Role of Positioning for FeedingSome studies on feeding interventions in children witheating impairment acknowledge the importance of posi-tioning for feeding, but did not examine the effects ofpositioning on the safety of swallowing [39,40]. Morerecent studies used radiologic techniques (i.e., videofluo-roscopy) to examine the safety of swallowing [18,19,41].Many base treatment recommendations on the radiologicfindings [42–44], but in only one study were severelyaffected children followed for 3–9 months after position-ing and food texture changes [42]. Less coughing andchoking during meals was reported, and caregivers ex-

pressed satisfaction with these changes [42]. Other stud-ies demonstrated that following proper positioning, feed-ing improved immediately. This was due to better foodretention, rather than a change in oral-motor skills [2,7].We have suggested that better food/liquid retention fol-lowing intraoral appliance therapy may have contributedto children’s ability to maintain their growth trajectory ata time when a weight “lag down” was expected [27].

It is well documented that children may sufferfrom frequent chest infections and even repeated pneu-monias when there is chronic food aspiration [19,45,46].Improvement in postural control as well as in jaw sta-bility following intraoral appliance therapy may contrib-ute to safer feeding [14]. Caregivers must be made awarethat the best position for feeding may be different frompositions for hand manipulations such as school activi-ties and playing with toys or computers.

Facial Expression

Better lip posture, i.e., closed mouth posture, was ob-served after 12 months of intraoral appliance therapy in11 (58%) of our children, suggesting a normalization oftheir facial expression. In the remaining 8, 4 children (2each in groups A and B) were able to communicate andmay have had better resting lip postures from the begin-ning. The other 4 children had moderate to severe cog-nitive impairments that may have contributed to theirlack of change in resting lip posture. Richardson [47]described reactions of nonhandicapped people towardpersons with a disability; they may be more formal, moreanxious, and over controlled in their behavior. Therefore,normalizing facial expression may have an important im-pact on the social development of a child because facialexpressions affect the way people approach others[8,47].

Our study concentrated on moderately impairedchildren. Only five were able to communicate either ver-bally or through sign language. Improvements in socialdevelopment may be more apparent in mildly affectedchildren who have the ability to communicate more ef-fectively. However, even severely impaired children en-gage in social activity when given the opportunity. Facialdeformity and reduced facial mobility is often equatedwith mental delay by teachers and family members[22,32,48]. Hence, children with cerebral palsy who havephysical but no cognitive impairments may not be en-couraged to reach their full potential in school and inactivities of daily living. Thus, normalizing a child’s fa-cial expression may open up new opportunities for par-ticipation and integration into society.

There were no changes in tongue position duringthe 12 months of the study. We found this surprising.

E.G. Gisel et al.: Oral Appliance Therapy and Whole-Body Mobility 233

Tongue position is important because one sees thetongue when the mouth is open and associates this pos-ture with disability despite the fact that the tongue maybe resting behind the teeth, as was the case with many ofour children. By assuming a more normal resting posi-tion of the mouth (lips closed), the tongue becomes au-tomatically hidden. From a physiological point of view,bringing the tongue back into the mouth places it in aposition which allows training for more efficient feeding(phase II of intervention) [14] through practice of morerefined movements such as lateralization, axial tipping,or lifting of the tongue tip.

Finally, we were surprised by the very noticeablechanges in facial expression in some of our children.Although we were aware of these effects based on 25years of experience with ISMARs, there had been noapriori intention to study this aspect of intervention.Nonetheless, it was felt that ana posterioriexplorationof these changes would be worthwhile. One of the limi-tations of this exploration is that the existing data werenot gathered for this purpose. Therefore, our analysisfrom existing video documentation may not have beenideal. However, a prospective approach to study this phe-nomenon is now justified.

Food Textures

Clinically meaningful changes in food textures offeredby caregivers were achieved during one year of intraoralappliance therapy. At the same time, children received adiet that was adequate in calories and that permitted themto maintain their established growth trajectory [14]. Theclinical significance of progressing to age-appropriatefood textures may not have been sufficiently appreciatedor it may have even been overlooked in the past. Wepropose that the more solid food textures force the childwith a moderate eating impairment to use oral-motorskills such as biting and chewing (which are not usedwith purees). That may reinforce the development of jawstability/control [14]. Thus, the change from puree tosolid feeding may contribute to the development of pos-tural controls through the associations discussed above.It further highlights the interdependence of body parts assuggested in the whole-body hypothesis and underlinesthe importance of oral feeding for children whenever it issafe and developmentally appropriate.

Acknowledgment.We are grateful to Drs. P. McKinley and M. Abra-hamowicz for critical comments on previous drafts of the manuscript.This research was supported through a grant by the National HealthResearch and Development Program #6605-4086-59. The authors ac-knowledge the generous support of Mrs. S. Desrocher in facilitating therecruitment of children as well as the support of occupational therapistsin the school system. Special thanks are also extended to all the teach-ers who enthusiastically supported our followup of children within theirclassrooms.

Appendix: Definitions of Food Textures

Solid Food

Puree: put through a blender, yogurt, puddingFork mashed: vegetable, meat, starchHashed: finely cut vegetables, meat, starchSoft table diet: blended with some sauce, vegetable (soft

cooked), meat (fish, chicken, hashed beef), starch(soft cooked pasta, rice, potatoes; soft breads,muffins, cakes), dairy (soft cheeses)

Regular table diet: vegetable (raw, crispy, cooked aldente), meat (beef, chicken), starch (crusty bread,baked potatoes)

Liquid Food

Thin: water, juice, milkNectar: apricot, pear juiceThick: with a thickening agent, liquid-type pudding

Heterogeneous Food

Thin liquid with solid pieces which requires simulta-neous drinking of liquid and chewing of solid.

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