Sindrome Down Habilitacion Fisica

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doi: 10.2522/ptj.20110061

Originally published online August 18, 20112011; 91:1463-1477.PHYS THER.

Tiernan and Joseph E. HornyakDale A. Ulrich, Amy R. Burghardt, Meghann Lloyd, ChadSyndrome: A Randomized TrialTwo-Wheel Bicycle for Children With DownPhysical Activity Benefits of Learning to Ride a

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Physical Activity Benefits of Learning

to Ride a Two-Wheel Bicycle forChildren With Down Syndrome:A Randomized TrialDale A. Ulrich, Amy R. Burghardt, Meghann Lloyd, Chad Tiernan,Joseph E. Hornyak

Background. People with Down syndrome (DS) display consistent patterns ofphysical inactivity. If these sedentary behaviors continue over extended periods of

time, there will be negative health consequences.

Objective. The objective of this study was to investigate the physical activity andhealth-related outcomes of teaching children with DS to ride a 2-wheel bicycle.

Design. This study was a randomized intervention in which the control groupwaited 1 year to receive the intervention.

Setting. This intervention study was conducted in a community setting.

Participants. The participants were children who were 8 to 15 years of age andwho had been diagnosed with DS.

Intervention. The participants were randomly assigned to an experimentalgroup (bicycle intervention) or a control group (no intervention).

Measurements. Measurements were obtained in the month before the inter-vention (preintervention), at 7 weeks after the intervention, and at 12 months afterthe preintervention measurement for all participants.

Results. The results indicated no group differences at the preintervention session.Fifty-six percent of the participants in the experimental group successfully learned toride a 2-wheel bicycle during the 5-day intervention. Analysis showed that partici-pants who learned to ride spent significantly less time in sedentary activity at 12months after the preintervention measurement and more time in moderate to vigor-

ous physical activity than participants in the control group. Body fat appeared to bepositively influenced over time in participants who learned to ride.

Limitations. It is unknown how frequently the children in the experimentalgroup rode their bicycles after the intervention.

Conclusions. Most children who are 8 to 15 years of age and who have beendiagnosed with DS can learn to ride a 2-wheel bicycle. Learning to ride can reducetime spent in sedentary activity and increase time spent in moderate to vigorousphysical activity, which may influence the health and functioning of these children.

D.A. Ulrich, PhD, Center on Phys-ical Activity & Health in PediatricDisabilities, School of Kinesiology,University of Michigan, 1402

Washington Heights, Ann Arbor,MI 48109-2013 (USA). Address allcorrespondence to Dr Ulrich at:[email protected].

A.R. Burghardt, MS, Departmentof Psychiatry, School of Medicine,University of Michigan.

M. Lloyd, PhD, Faculty of HealthSciences, University of OntarioInstitute of Technology, Oshawa,Ontario, Canada.

C. Tiernan, MS,Center on PhysicalActivity & Health in Pediatric Dis-

abilities, School of Kinesiology,University of Michigan.

J.E. Hornyak, MD, PhD, Depart-ment of Physical Medicine &Rehabilitation, School of Medi-cine, University of Michigan.

[Ulrich DA, Burghardt AR, LloydM, et al. Physical activity benefitsof learning to ride a two-wheelbicycle for children with downsyndrome. Phys Ther. 2011;91:14631477.]

2011 American Physical Therapy

AssociationPublished Ahead of Print: August

18, 2011Accepted: May 16, 2011Submitted: February 23, 2011

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Recent research has shown thatengaging in physical activity isa primary avenue of prevention

and intervention for combating obe-sity in all populations.1 Physical inac-

tivity and obesity also are growingproblems in people with intellectualdisabilities.26 People with disabili-ties are less likely to be active thanthe general population, resulting inan increased strain on families andthe health care system.3,7,8 The phys-ical activity of people with disabili-ties has been identified as a healthpriority by Cooper et al9 as well as in

the Surgeon Generals report onimproving the health of people withmental retardation.3 These reports

recommended that professionalsdevelop methods to promote physicalactivity and reduce sedentary behav-iors in people with disabilities.2,3

Down syndrome (DS) is a common

genetic disorder that is identified

before or soon after birth and thatcarries the certainty of developmen-tal delays. In the United States, DSoccurs approximately 1.36 times inevery 1,000 live births.10 Trisomy 21

is the most common cause; the extracopy of chromosome 21 results ingene overexpression. This genotypeis present in approximately 95% ofpeople with DS. The gene overex-pression leads to a highly complexphenotype in which both physiolog-ical and cognitive developments aresignificantly altered.11 Children withDS also experience significant delays

in the onset of early motor milestonesand display qualitative differences inmovement patterns in comparison

with children who show typicaldevelopment (TD).1218 Additionally,children with DS have higher rates ofoverweight and obesity.12,19

Research on physical activity and its

relationship to health in children

with DS is extremely limited. Onestudy20 examined the physical activ-ity patterns of 30 sibling pairs (chil-dren who had DS and a sibling whohad TD and was closest in age)

through parent report. The resultsindicated that parents perceivedtheir children with DS to be lessphysically active than their siblings

with TD.20 Another study21 investi-gated the physical activity levels ofchildren with DS (mean age7.1

years) and siblings with TD throughthe use of accelerometers. Theresults indicated that over a 7-day

period, the average length of a boutof vigorous physical activity was sig-nificantly shorter in the children

with DS.21

Most children with DS eventuallylearn a basic repertoire of motorskills; however, they seem to fall fur-ther behind their peers as they age.

One culturally normative skill thatoften is never learned is riding a2-wheel bicycle.12,18,22 Bicycle ridingin childhood provides many oppor-tunities for engaging in physicalactivity as well as extended opportu-

nities for social interactions withfamily members, peers, and commu-nity members. Menear22 found that

parents valued bicycle riding as askill that could increase opportuni-ties for their children to participatein the community and interact withpeers.

Recent research has clearly demon-strated the negative impact of seden-tary behaviors on health.2325 Chil-

dren and adolescents with DS areless active than their peers withTD.2022,26 However, despite the evi-dence regarding the passive natureof children with DS,26 little researchhas been conducted on designingand testing interventions to decreasesedentary behaviors in people withDS and increase the time that they

spend in physical activity.27

The Bottom Line

What do we already know about this topic?

Children and youth with Down syndrome display a very stable pattern of

physical inactivity, which gets worse with age.

What new information does this study offer?

Less than 10% of children with Down syndrome who are 8 to 15 years of

age can ride a 2-wheel bicycle. This intervention study demonstrated,

over a 5-day period, that a majority of children with Down syndrome can

learn to ride a 2-wheel bike if given intensive, individualized instruction.

Those children who learned to ride demonstrated a significant decrease

in time spent in sedentary activity 12 months after training and a reduc-

tion in subcutaneous fat.

If youre a patient or caregiver, what might thesefindings mean for you?

If you or someone you care for has Down syndrome, your physical

therapist may advise you to seek out a bicycle training program such as

that conducted by the Lose the Training Wheels organization and, after

you have learned, to ride your bike as often as you can. Your physical

therapist also may encourage you to learn other activities to increase your

daily physical activity.

Bicycle Intervention and Down Syndrome

1464 f Physical Therapy Volume 91 Number 10 October 2011



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The aim of our randomized interven-tion study was to test the hypothesisthat increasing the physical activityand motor skill repertoire of children

with DS by one functional, culturally

normative activity valued by mostfamilies could have a positive impacton patterns of physical activity asso-ciated with health and functioning.Our expectation was that with avail-able training technology and individ-ualization of training, we could teachmost children with DS to ride a2-wheel bicycle and, as a result, thatthose who learned to ride would sig-

nificantly decrease the time spent insedentary activity (SED) and increasethe time spent in moderate to vigor-

ous activity (MVPA). We also antici-pated that a decrease in time spent inSED and an increase in time spent inMVPA might decrease body fat or atleast prevent it from being increasedover a 12-month follow-up period.

This is the first intervention studyfounded on the principles ofdynamic systems theory28 in aneffort to change patterns of physicalinactivity in children. We are treatingthe motor and physical skill reper-

toire in children with DS as a poten-tial control parameter (ie, an inabilityto ride a 2-wheel bicycle). Our inter-

vention is designed to help childrenscale up their repertoire with ahighly valued activity (riding a2-wheel bicycle) and, as a result,shift the children away from moresedentary behaviors.

MethodWritten informed consent from par-

ents and assent from children wereobtained. A total of 72 children whohad DS and who were 8 and 15 yearsof age were enrolled and randomlyassigned to either an experimental(EXP) group (bicycle intervention)or a control (CON) group (no inter-

vention) (the CON group waited 1year to receive the intervention).

Participants were recruited throughcontact with and presentations to 5large organizations for parents of

children with DS in Michigan, Ohio,and northern Indiana. According toparent report during recruitmentand child report during initial datacollection, none of the participants

could ride a 2-wheel bicycle at entryinto the study. Most parents claimedthat they had been trying for years toteach their children how to ride a2-wheel bicycle and had basicallygiven up trying.

Figure 1 shows the flow of partici-pants (N81) through the study.Children were excluded from the

study if they had any medical condi-tions (eg, uncontrolled seizures) thatdid not allow physical exertion at a

moderate level or if they exhibitedsevere behavioral issues duringscreening or measurements that pro-hibited them from being able to ben-efit from instruction within a com-munity setting. Eleven children were

not included in the final analysisbecause of parent withdrawal orattrition at follow-up. Of the 61remaining children, 34 remained inthe EXP group and 27 remained inthe CON group.

For all participants, regardless ofgroup, measurements were obtained

in the month before the intervention(preintervention), at 7 weeks afterthe intervention (post-1), and at 1

year after the preintervention mea-surement (post-2). Measurementsincluded leg strength, standing onone leg for balance, height, weight,skinfolds, and physical activity.These measurements were selected

primarily because they representedperformance and health-related areasthat could be affected by participa-tion in physical activity. This selec-tion appears to conform to the con-cepts included in the InternationalClassification of Functioning, Dis-

ability and Health (ICF) model.29

We hypothesized that participation

in and acquisition of the skill of bicy-cle riding by children with DS could

reduce SED, increase leg strength,and reduce body fat.

Leg StrengthIsometric knee extensor muscle

strength was assessed with a manualmuscle tester (MMT) (LafayetteInstrument Co, Lafayette, Indiana).The participant was seated on a table

with the knee at 120 degrees, as mea-sured with a goniometer. The MMT

was placed on the distal anteriorshank approximately 7.6 cm (3 in)above the ankle. The participant wasencouraged to provide maximal

force against the MMT for 5 seconds;peak force was recorded in kilo-grams. Three trials were performed,

and the peak value was used for eachleg. Isometric knee flexor musclestrength was assessed with the par-ticipant in a prone position on thetable with the knee flexed to 20degrees off the table. The MMT was

placed on the lower portion of theposterior side of the leg approxi-mately 7.6 cm above the ankle.Three trials were performed, and thepeak value was used for each leg.Research staff received training with

the MMT and had to demonstrateconsistency across several trials and

with 2 testers experienced in the

professional use of the MMT. Theresearch coordinator observed the 2testers during the formal testing toensure that consistent procedures

were being implemented.

Standing BalanceStanding balance was assessed byasking the participant to place the

hands on the hips and stand on onefoot. Each participant was allowed tocontinue until 60 seconds wasreached or the standing foot was dis-placed, the hands were removedfrom the hips, or the raised legtouched the floor. Time was mea-sured to the nearest tenth of a sec-ond. Three trials were repeated for

each leg, and the peak value wasused. The research coordinator arbi-trarily observed the testing station to



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ensure that the testers were scoringthe performances consistently. Inthe event that the scores recorded bythe coordinator and the tester dif-fered by 0.5 second or more, the trial

was repeated.

Height, Weight, and Skinfolds

Height and weight were measuredwith the child wearing no shoes.Height was measured to the nearesttenth of a centimeter with a portablestadiometer (SECA S-214) (SecaCorp, Hanover, Maryland). Weight

was measured to the nearest tenth ofa kilogram (Health O Meter H-349KLdigital scale) (Pelstar LLC, Alsip, Illi-

nois). Skinfold measurements weretaken by a physician who had many

years of experience with skinfold

measurements involving children.Skinfold measurements were takenat the triceps muscle and calf withLange skinfold calipers (Beta Tech-nology, Santa Cruz, California). Thecalipers were calibrated before eachof the 3 measurement sessions. Allmeasurements were taken on the

right side of the body and to thenearest tenth of a millimeter inaccordance with standardized proce-dures.30 Two measurements wereobtained at each site, and the aver-age was used. A regression equation

was used to calculate the percentageof body fat.

Physical ActivityPhysical activity was assessed for 7days with Actical accelerometers

(Phillips Respironics Inc, Bend, Ore-gon). The Actical monitor was pro-grammed to sample at intervals of 15seconds. Participants and their par-ents were instructed on how to wearthe monitor (placing it on the righthip just above the iliac crest with anelastic belt) and were told that chil-

dren should wear the monitor for allactivities except swimming, shower-ing or bathing, and sleeping. Theyalso were given a log to record times

when the monitor was not worn.Because of the limited number ofaccelerometers available, each phys-ical activity measurement wasobtained as close as possible to the 3

measurement time points describedabove.

Assessed forEligibility(N=81)

Enrollment(n=72)

Randomization

Excluded (n=9)

Not meetinginclusion criteria(n=9)

CONAllocated and waiting 1 year to

be trained (n=36)

EXPAllocated and trained (n=36)

Lost to Follow-upDue to moving out of area (n=2)

Analyzed(n=19)Did not learn to ride andwere excluded from finaloutcome analysis (n=15)

Analyzed (n=27)

Lost to Follow-upDue to parent loss of interest during wait (n=7)

Exhibited behavior problems during measurement (n=2)

Figure 1.

Flow of participants through the study. EXPgroup that received bicycle training, CONgroup that waited to receive bicycletraining.





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InterventionThe bicycle training intervention

was provided by the Lose the Train-

ing Wheels organization (Goddard,Kansas) and consisted of 5 consecu-tive days of individual instruction for75 minutes per day. Five days oftraining is currently the only optionprovided by the Lose the Training

Wheels organization (see Appendixfor a description of the basic trainingmanual). Trained staff with profes-sional experience with children whohad cognitive constraints and DS

were used. The intervention was set

up in a summer camp format inwhich the participants came into thetraining facility for 75 minutes and

then returned home until the nextday. Specially engineered adaptedbicycles provided by the Lose theTraining Wheels organization wereused for the initial training (Fig. 2shows an example of an adaptedbicycle). The adapted bicycles weretailored throughout the training tothe participants individual needs on

the basis of their level of riding skill.The adapted bicycles have a series of8 roller wheels (Fig. 3). The roller

wheels taper progressively fromroller 1 to roller 8. The first and sec-ond roller wheels afford riders withmany opportunities to pedal contin-uously without fear of falling and tolearn to stop and place their feet

firmly on the floor. The seventh andeighth roller wheels are the mosttapered on the ends; by the time

riders have advanced to these rollerwheels, they have demonstratedgood control of the adapted bicycle.The floor manager continuouslyobserves each rider during the train-

ing session to determine when therider is ready for a change in theroller wheel. To make this decision,the floor manager looks primarily atthe speed of pedaling, whether rid-ers are beginning to lean into acurve, and whether riders are begin-ning to relax the joints in their armsand begin to use the handle bars toturn and control the bicycle. Most

riders begin the training by freezingthe degrees of freedom in their arms(locking their arms) to better control

the bicycle and because they antici-pate falling. Once they succeed incontinuous pedaling around thetraining facility for 10 minutes ormore without falling, they begin torelax the joints in their arms. The

adapted bicycle also has a handleattached to the rear for the trainer touse, when necessary, as a promptingsystem to facilitate continuous ped-aling or leaning into turns or to stopthe rider quickly to prevent a fall.

The goal is to progress to a standard2-wheel bicycle.

As the childs performance improvedover the 5 days of training, he or shereceived training on bicycles thatbecame progressively more similarto a standard 2-wheel bicycle withthe training handle still attached. Par-ents were encouraged to bring thechilds own bicycle to the trainingprogram by the last day to assist the

child in making a successful transi-tion to his or her own bicycle forriding at home. Participants weretested on the final day of the inter-

vention to measure their success inriding independently on a 2-wheelbicycle for a minimum of 9 m (30 ft).Riders were positioned on their2-wheel bicycles, and a distance of

9 m was marked in front of them. Onthe basis of the measurements takenon the final day, participants in the

EXP group were classified as havinglearned to ride a 2-wheel bicycle(EXP-L group) or not having learnedto ride a 2-wheel bicycle.

Data ReductionFor the physical activity measure-ments, data were used if the partici-pant wore the monitor for a mini-mum of 10 hours per day for aminimum of 4 days out of each of the

7-day monitoring periods. One of the4 days also must have been a week-end day. These criteria have been

established in the literature as sug-gested guidelines for including validdata points.31 The data were reducedto assign the activity count per15-second epoch into the followingcategories: SED and MVPA. We alsosummarized the data by calculatingthe average activity counts per min-ute (AVGMIN). We used the cutoff

points (adjusted for 15-secondepochs) outlined by Puyau et al31

because they were established withActical accelerometers in childrenwho were in the same age range andhad TD. At the time of data collec-tion and reduction, no knownregression equations had been devel-oped for children with DS; thus, we

used cutoff points established forchildren who were in the same agerange but had TD.

Figure 3.Series of rollers for adapted bicycle.

Figure 2.Adapted bicycle.



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Data AnalysisA mixed-model analysis was per-formed with SPSS version 18.0 (SPSSInc, Chicago, Illinois). Performing aretrospective analysis, we defined

the EXP group as including only par-ticipants who did learn to ride a2-wheel bicycle (EXP-L group) to elu-cidate the effects of successfullylearning to ride after the intervention(defined by riding independently aminimum of 9 m). We did notinclude in the final outcome analysisany participants in the EXP group

who did not learn to ride. Therefore,

19 participants in the EXP-L groupand 27 participants in the CONgroup were included in the outcome

analysis. Sex and age at entry into thestudy were covariates, and group(EXP-L and CON) and time (preinter-

vention, post-1, and post-2) weremain effects in the model. The inter-actions of group and time also were

examined to determine whether onegroup performed better than theother group over time. The depen-dent variables tested were knee flex-ion and extension strength in theright and left legs, body mass index

(BMI), percentage of body fat, stand-ing balance on the right and left legs,

AVGMIN, and number of minutes

per day spent in SED and MVPA. Forthe activity variables, we co-varied

whether or not the days were week-end days or weekdays. The signifi-cance level was set at .05. Post hocanalysis was performed with Bonfer-roni corrections.

Role of the Funding Source

Partial funding for this research wasprovided by the National Down Syn-drome Society, the Steelcase Founda-tion, the Lyle Foundation, and theDown Syndrome Association of

Western Michigan. Doctoral stu-dents were funded in part by a lead-ership training grant(H325D070081) awarded to Dr

Ulrich by the US Office of SpecialEducation Programs, US Departmentof Education. The Lose the Training

Wheels organization provided accessto their fleet of adapted bicycles andmonitored the bicycle trainingprotocol.

ResultsThe average ages at entry into thestudy were 12.0 (SD1.9) years forthe CON group and 12.4 (SD2.2)

years for the EXP-L group. Therewere 11 boys and 16 girls in the CONgroup and 9 boys and 10 girls in theEXP-L group. The race or ethnicitybreakdown of the convenience sam-ple was as follows: 24 white, 2 Afri-

can American, and 1 other in theCON group and 16 white, 1 His-panic, and 2 other in the EXP-L

group.

No significant group differenceswere found in any of the variablesmeasured at preintervention. Theresults of the mixed-model analysis

indicated a significant group timeinteraction (F2,853.71, P.028) forBMI.A post hocanalysis showed thatthe CON group had a significantincrease in BMI at post-2 relative tothat at the baseline (P.001). In

addition, main effects of group werefound for knee flexion in the rightand left legs. The EXP-L group had

significantly greater knee flexion inthe right leg (F4.491,38, P.041)and left leg (F5.381,38, P.026)overall than the CON group. How-ever, no significant group timeinteractions were observed. Theonly other significant result was themain effect of the group timeinteraction for the percentage of

body fat (F

3.172,84, P

.047). Apost hoc analysis showed that over-all, the EXP-L group had significantlydecreased percentages of body fat atpost-1 and post-2 (P.004 and

P.006, respectively). The raw dataand P values for the main effect ofthe group time interaction areshown in the Table.

For the physical activity data, signif-icant group time interactions were

found for SED (F3.812,611,P.023), MVPA (F5.222,622,P.006), and AVGMIN (F5.552,620,P.004). There was no significantdifference in total activity monitor

wear time between the groups. Therange of activity monitor wear timesacross groups and measurement ses-sions was 12.8 to 13.6 hours per day.

A post hoc analysis of time spent inSED showed a significant decrease atpost-1 (P.001) and post-2 (P.001)relative to preintervention for theEXP-L group. The EXP-L group alsospent significantly less time in SED at

post-1 (P.035) and post-2 (P.004)than the CON group (Fig. 4). Aposthocanalysis showed the EXP-L group

spent significantly more time inMVPA at post-2 than the CON group(P.023). A post hoc analysis alsoshowed that the CON group spentmore time in MVPA at preinterven-tion than at post-1 (P.001) and

post-2 (P.004); meanwhile, theEXP-L group spent significantly moretime in MVPA at post-2 than at post-1(P.009). A post hoc analysisshowed that the EXP-L group hadsignificantly higher AVGMIN than

the CON group at both post-1(P.023) and post-2 (P.004). Forthe CON group, AVGMIN signifi-

cantly decreased from baseline topost-1 (P.004) and baseline topost-2 (P.039), whereas for theEXP-L group, AVGMIN significantlyincreased from baseline to post-2(P.018) (Fig. 5).

DiscussionIn preparation for conducting the

present intervention study, we sur-veyed 298 families who lived inMichigan, Ohio, and Indiana and

who had a child with DS in the agerange of 8 to 15 years; according toparent report, 9.7% of the children inthis age range could ride a 2-wheelbicycle. The average age of thisgroup was 12.5 years. These data

support the view that learning toride a 2-wheel bicycle is a challeng-ing task for this population. The





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results of the present study demon-strate that 56% of the total partici-pants in the EXP group learned toride a 2-wheel bicycle with 75 min-utes of individualized training per

day for 5 consecutive days. Theseresults and those of other studieslend support for this method of train-ing people with DS32,33 to ride a bicy-cle. Many of the participants whohad DS and who learned to ride arenot necessarily prepared to ride theirbicycles on busy city streets. Bicycleriding can be done for exercise and

recreation in many locations, includ-ing parks, bicycle paths, large park-ing lots, local neighborhood side-

walks, and school playgrounds.Parents were encouraged to pro-mote bicycle riding and continuedpractice in riding. At post-2, accord-ing to parent report, none of the par-

ticipants in the CON group, who didnot receive the bicycle training,could demonstrate the ability to ridea 2-wheel bicycle.

When all of the participants in theEXP and CON groups were com-pared, the physiological and motormeasurements of leg strength, BMI,

percentage of body fat, and one-legbalance were not significantlyaffected over the 1-year period after

an intervention aimed at teachingchildren with DS to ride a 2-wheelbicycle. This finding should not besurprising given that 44% of the totalparticipants in the EXP group did not

learn to ride a 2-wheel bicycle inde-pendently within the 5-day interven-tion and, therefore, were function-ing more like participants in theCON group. Because of this result,participants in the EXP group whodid not learn to ride were excludedfrom any outcome analysis. Giventhe variability in cognitive develop-

ment in people with DS, some of theparticipants who did not learn toride may have simply needed more

Table.Analysis of Outcome Variables by Groupa and Timeb

Variable

Preintervention Post-1 Post-2

Pc

X (SD) Effect

Size,

Cohend

X (SD) Effect

Size,

Cohend

X (SD) Effect

Size,

CohendEXP-LGrou p CON Group EXP-LGroup CON Group EXP-LGroup CON Grou p

Knee extension

strength, right

leg, kg

15.8 (10.1) 12.9 (6.6) 0.35 19.7 (9.8) 15.9 (5.4) 0.50 15.0 (4.0) 12.3 (4.5) 0.64

Knee extension

strength,

left leg, kg

15.4 (10.3) 13.5 (6.3) 0.23 20.1 (10.2) 15.4 (5.9) 0.58 14.7 (4.0) 12.0 (4.9) 0.61

Knee flexion

strength,

right leg, kg

13.6 (11.2) 11.1 (4.3) 0.32 17.5 (9.1) 12.6 (4.1) 0.74 15.0 (4.4) 12.0 (5.1) 0.63

Knee flexion

strength,

left leg, kg

13.3 (8.3) 11.3 (4.7) 0.31 17.2 (11.2) 12.2 (4.4) 0.64 13.6 (4.0) 10.2 (4.4) 0.81

Balance, right leg, s 4.7 (4.8) 3.9 (2.7) 0.21 7.0 (7.8) 5.1 (5.4) 0.24 7.3 (8.3) 4.5 (4.0) 0.45

Balance, left leg, s 5.3 (4.9) 4.5 (6.8) 0.14 6.6 (5.7) 5.1 (6.1) 0.25 8.3 (8.8) 4.1 (3.3 0.69

Body mass index,

kg/m224.3 (5.5) 23.0 (4.8) 0.25 24.3 (5.5) 23.3 (4.8) 0.19 24.6 (4.7) 24.3 (5.2) 0.06 .028

Percentage of body

fat

36.7 (14.5) 32.1 (13.6) 0.33 31.5 (12.5) 28.2 (11.1) 0.28 30.5 (9.8) 29.5 (9.3) 0.10 .047

Time spent in

sedentary

activity, min

531.7 (101.1) 537.1 (104.7) 0.05 473.0 (155.5) 522.8 (141.8) 0.34 456.6 (139.1) 527.6 (139.1) 0.63 .023

Time spent in

moderate to

vigorous activity,

min

39.2 (23.7) 46.9 (29.2) 0.29 36.5 (25.8) 34.8 (18.0) 0.08 48.7 (26.2) 39.7 (23.8) 0.36 .006

Average activity,counts/min

284.6 (124.8) 314.2 (154.0) 0.21 304.5 (140.6) 270.7 (112.0) 0.27 352.5 (134.2) 290.8 (132.8) 0.46 .004

a Experimental group, consisting of participants who learned to ride a 2-wheel bicycle (EXP-L group), and control group, consisting of participants whowaited 1 year to receive the intervention (CON group).b Before the intervention (preintervention), 7 weeks after the intervention (post-1), and 1 year after the preintervention measurement (post-2).c For the group time interaction effect.





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days of practice and training. This

conclusion is supported by the gen-eral observation that several riders inthe EXP group were close to meeting

the criterion of 9 m on the final dayof the intervention but did notachieve it. In the future, this hypoth-esis could be tested by providingaccess to training bicycles for a lon-ger period than the standard 5 days.

We observed that fear of fallingappears to be a major constraint in

teaching people with DS to ride a2-wheel bicycle. This observationwas based on the behavior of most ofthe participants while pedaling dur-ing day 1 or 2 of training and is sup-ported by recent motor controlresearch involving people who haveDS and who have concluded thattheir major goal in movement is to

move safely.34 This fear can be a fac-tor in why most children and youths

with DS do not learn how to ride a

2-wheel bicycle. However, all par-

ticipants in the EXP group madeobservable improvements in theirability to ride over the 5 days of the

intervention. These improvementswere frequently noted in commentsmade by parents during each day oftraining.

Analysis of data from the EXP-Lgroup and the CON group showed atrend for both BMI and percentage ofbody fat to be potentially affected

over the 1-year follow-up period. Onthe basis of effect size statistics(Table), the EXP-L group displayedhigher BMIs and percentages of bodyfat at the preintervention measure-ment, but this difference disap-peared over 12 months (post-2). Thisfinding warrants additional researchinto whether learning to ride a bicy-

cle may enhance physical health byimproving body composition.

The most meaningful result of thepresent study appears to be that theEXP-L group reduced the averageamount of time spent in SED per dayby 75 minutes. With any standard of

evaluation, this reduction in SED isamazing and, if continued, shouldhave a positive impact on futurehealth. The results also demon-strated that, compared with the CONgroup, the EXP-L group significantlyincreased the time spent in MVPA atpost-2. This pattern was reflected inthe AVGMIN variable. This differ-ence cannot be explained by

increased age, given that physicalactivity typically decreases withincreased age.35 Interestingly, the

research staff received unsolicitedcommunications from multiple par-ents whose children were in theEXP-L group, suggesting that the par-ents perceived that their children

were less fearful and more motivated

to try other physical and sports activ-ities after overcoming their fear offalling and learning to ride a 2-wheelbicycle. These parental perceptionsneed to be validated in futureresearch.

The improvement in physical activ-ity levels could help explain the

trend exhibited in percentage ofbody fat. Increased physical activitycan contribute to decreased bodymass, which was a trend found in theEXP-L group. It is not clear why timespent in MVPA decreased in theCON group during this time,although the literature on children

with TD shows a decline in physical

activity after the onset of puberty, soit is possible that this phenomenon isreflected in youths with DS as well.36

However, this phenomenon wouldaffect both the EXP-L group and theCON group equally. The decrease intime spent in MVPA by both groupsat post-1 could have been a seasonaleffect because post-1 measurements

generally occurred in August andSeptember, when outside tempera-tures during the day were high and

Figure 4.Average time (minutes) spent in sedentary activity by participants in the experimentalgroup who learned to ride a 2-wheel bicycle (EXP-L group) and participants in thecontrol group (who waited 1 year to receive the intervention) (CON group) acrossmeasurements. Prebefore the intervention (preintervention), Post-17 weeks afterthe intervention, Post-21 year after the preintervention measurement.





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schools were resuming classes. Insummary, the physical activityresults of the present study indicatethat participating in an adapted bicy-cle training program has the poten-

tial to decrease time spent in SEDand increase time spent in MVPA orat least stave off its decline withagea factor that is especiallyimportant for the overall health ofchildren with DS.

The results of the present study pro-vide support for our hypothesis thatincreasing the physical activity skill

repertoire with a functional, cultur-ally normative activity valued bymost families and children could

have a positive impact on physicalactivity associated with health andfunctioning. Increasing time spent inMVPA by children and youths is asignificant challenge faced by practi-tioners around the world,35 and chil-

dren with DS are less likely to engagein physical activity than their peers

with TD.20,21 The results of the pres-ent study demonstrate that learninghow to ride a 2-wheel bicycle mayincrease or maintain time spent in

MVPA by children with DSa prom-ising idea. Interestingly, a reductionin time spent in sedentary behaviors

is emerging as the front line of actionin promoting physical activity in chil-dren and youths. Therefore, thedemonstration of a significantdecline in time spent in sedentarybehaviors in the present study is amajor milestone in determining strat-egies for promoting physical activityand reducing sedentary behaviors in

children and youths with DS. Addi-tional research is needed to deter-mine whether the effects are main-tained over longer periods of time.The present study appears to serveas an example of an interventionfounded on the concepts included inthe ICF model. Acquiring skills inother physical activities, such as

dance, swimming, and martial arts,also should improve participation,functioning, and health-related out-

comes. Future physical activity inter-

ventions also must involve parents tohelp maximize the frequency of rid-ing for children with DS and to eval-

uate how parental support for phys-ical activity mediates the frequencyof riding and important health-related and functional outcomes.

LimitationsThis training study had several limi-tations. The first limitation was thatnot all of the participants in the EXP

group learned to ride a 2-wheel bicy-cle and therefore were not includedin the final outcome analysis; this fac-tor reduced the statistical power.Second, some of the parents whosechildren were randomly assigned tothe CON group decided that they didnot want to wait 1 year for theirchildren to receive training and

dropped out of the study. In futureresearch, families assigned to theCON group should bepaid for wait-

ing; the compensation could be used

to access an alternative intervention(not bicycle training) to help meetthe needs of their children while

they wait to receive the bicycle train-ing intervention. The third limitation

was the lack of a follow-up measureof the frequency of riding by theparticipants over the 12-monthfollow-up period. This measurecould have helped us explain whysome participants who learned toride displayed more or fewer bene-

fits in the long term. The final limi-tation relates to the method of wear-ing the Actical physical activitymonitor. We elected to follow thestandard protocol of wearing themonitor on the right hip. In retro-spect, wearing the monitor on thehip most likely meant that much ofthe increased physical activity result-

ing from bicycle riding was missedbecause the participants were sittingdown (on the bicycle seat) while

Figure 5.Average time (minutes) spent in moderate to vigorous activity by participants in theexperimental group who learned to ride a 2-wheel bicycle (EXP-L group) and partici-pants in the control group (who waited1 year to receive the intervention) (CON group)across measurements. Prebefore the intervention (preintervention), Post-17 weeksafter the intervention, Post-21 year after the preintervention measurement.





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they rode. However, the Actical isthe accelerometer of choice for mea-suring physical activity over anentire day, even though it is not asaccurate at capturing activities such

as biking and swimming (because ofwater).37 To capture actual differ-ences in riding behaviors, a secondaccelerometer could be placed onthe ankle to monitor changes in legactivity due to riding; however,much more research is needed to

validate this procedure.

On the basis of the results of our

randomized intervention study, itappears that the principles ofdynamic systems theory28 can serve

as a foundation for changing patternsof physical inactivity in children

with DS and children without DS.Future research is needed to identifyother potential control parametersthat are modifiable with therapy and

training in an effort to shift childrenaway from stable patterns of physicalinactivity. We have provided supportfor increasing the skill repertoire,but other factors also must be con-sidered and tested; these factors

include leg strength, endurance,confidence, and psychosocial factorsthat may improve with the increased

socialization that occurs withincreased participation in physicalactivities within the community.

ConclusionThe majority of children who haveDS and who are older than 8 years ofage can learn to ride a 2-wheel bicy-cle given appropriate individualized

training. More clinical and educa-tional settings should make an effortto teach people with DS to ride a2-wheel bicycle, to develop strate-gies for monitoring and reinforcingthe frequency of riding, and to dis-tribute information to parents onlocations within their community toride. Therapists also should encour-

age organizations for parents of chil-dren with DS (especially the DADs[Dads Appreciating Down Syn-

drome] group) to organize bicycleriding groups and to encourage rid-ers to train and enter the bicycle rid-ing events in the Special Olympics.The Special Olympics will increase

the availability of riding events onlyif more participants learn to ride.Therapists should provide parents

with methods for increasing theirchilds leg strength before enrollingin a bicycle training program. Partic-ipants who did not learn to ride fre-quently spent more time off the bicy-cle during the intervention sessionsand stated that their legs were too

tired to continue. Leg fatigue mayhave been present, but low levels ofmotivation were observed in many

of the participants who were 8 or 9years of age and who used leg fatigueas an excuse to get off the bicycle.Participants who spent most of the75 minutes available for training onthe bicycle practicing generally

learned to ride in 5 days.

Dr Ulrich, Dr Lloyd, Mr Tiernan, and DrHornyak provided concept/idea/researchdesign. All authors provided writing anddata collection. Ms Burghardt provided data

analysis. Dr Ulrich and Ms Burghardt pro-vided project management. Dr Ulrich pro-vided fund procurement. Dr Lloyd providedconsultation (including review of manuscriptbefore submission).

All methods used in this study wereapproved by theUniversity of Michigan Insti-tutional Review Board.

Partial funding for this research wasprovidedby the National Down Syndrome Society,the Steelcase Foundation, the Lyle Founda-tion, and the Down Syndrome Association of

Western Michigan. Doctoral students werefunded in part by a leadership training grant

(H325D070081) awarded to Dr Ulrich by theUS Office of Special Education Programs, USDepartment of Education. The Lose theTraining Wheels organization providedaccess to their fleet of adapted bicycles andmonitored the bicycle training protocol.

Trial registration: Australian New ZealandClinical Trials Registry (ANZCTR) identifier:

ACTRN12611000808943.

DOI: 10.2522/ptj.20110061

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children: cohort study.BMJ. 1999;319:1039.3 Closing the gap: a national blueprint for

improving the health of individuals withmental retardation. In: Surgeon GeneralsConference on Health Disparities andMental Retardation. Rockville, MD:Office of Surgeon General, US Departmentof Health and Human Services; 2002.

4 The Surgeon Generals Call to Action toPrevent and Decrease Overweight andObesity 2001.Rockville, MD: Office of theSurgeon General, US Department ofHealth and Human Services; 2001.

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vention and Promotion, Centers for Dis-ease Control and Prevention, US Depart-ment of Health and Human Services; 1996.

7 Draheim CC, Williams DP, McCubbin JA.Prevalence of physical inactivity and rec-ommended physical activity incommunity-based adults with mental retar-dation. Ment Retard. 2002;40:436444.

8 Centers for Disease Control and Prevention.Physical activity among adults with a disabil-ity: United States, 2005.MMWR Morb Mor-tal Wkly Rep.2007;56:10211024.

9 Cooper RA, Quatrano LA, Axelson PW,et al. Research on physical activity andhealth among people with disabilities: aconsensus statement. J Rehabil Res Dev.1999;36:142.

10 Centers for Disease Control and Preven-tion. Improved national prevalence esti-mates for 18 selected major birth defects:United States, 19902001. MMWR MorbMortal Wkly Rep. 2006;24:1301.

11 Wishart JG. The development of learningdifficulties in children with Downs syn-drome. J Intellect Disabil Res. 1993;37:389403.

12 Block ME. Motor development in childrenwith Down syndrome: a review of theliterature. Adapt Phys Activ Q. 1991;8:179209.

13 Carr J. Mental and motor development in

young mongol children.J Ment Defic Res.1970;14:205220.

14 Frith U, Frith CD. Specific motor disabili-ties in Downs syndrome. J Child PsycholPsychiatry. 1974;15:293301.

15 Ulrich DA, Ulrich BD, Angulo-Kinzler RM,Yun J. Treadmill training of infants withDown syndrome: evidence-based develo-pomental outcomes.Pediatrics. 2001;108;e84.

16 Morris AF, Vaughan SE, Vaccaro P. Mea-surements of neuromuscular tone andstrength in Downs syndrome children. JMent Defic Res.1982;26(pt 1):4146.





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19 Rubin SS, Rimmer JH, Chicoine B, et al.Overweight prevalence in persons withDown syndrome. Ment Retard. 1998;36:175181.

20 Sharav T, Bowman T. Dietary practices,physical activity, and body-mass index in aselected population of Down syndromechildren and their siblings. Clin Pediatr.1992;33:341344.

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22 Menear K. Parents perceptions of healthand physical activity needs of childrenwith Down syndrome. Downs Syndr ResPract. 2007;12:6068.

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Appendix.Bicycle Training Manuala

This is a manual addressing the critical training guidelines used by researchers during 2-wheel bicycle interventions foryouth with Down syndrome and autism spectrum disorder. The challenge of learning to ride a standard 2-wheel bicycleis to attain effective internal feedback needed for balance while riding a bicycle that initially is perceived to be

unpredictable and unstable.b This sense of instability activates ineffective defense responses. These same responses arereinforced when using training wheels and lead to the acquisition of a counterproductive motor plan. In turn, when thetraining wheels are removed, the riders response to the bicycles action is opposite of the motor plan that is necessaryto maintain control of the bicycle. The method developed in this manual is designed to teach individuals the correctmotor plan for riding a 2-wheel bicycle through a series of bicycle adaptations and individualized training, while atthe same time reducing their fear of falling. In addition, it is recognized that similarities exist in patterns of learningamong individuals with and without disabilities; however, individuals with disabilities may require additional timeand specialized teaching strategies to have success.b This training incorporates innovative teaching techniques andspecialized equipment designed to facilitate 2-wheel bicycle riding success for individuals with disabilities.b

Research Study Information

Training Details

1. One or 2 months prior to intervention, measurements of physical activity using Actical physical activity monitorsare recorded.

2. One or 2 days prior to intervention, measurements encompassing areas of intellect, physical attributes, psycho-

social attributes, balance, and strength are recorded for each individual participating in bike camp.

3. Bike camp runs for 1 week, with 5 successive training sessions of 75 minutes each. Five sessions are held eachday for 7 to 8 riders each.

4. Each participant is paired with one trainer, and they remain paired for the duration of camp.

5. The facility used needs to have a spacious flat floor area with no or few obstacles.

6. Two to 3 months following intervention, measurements of physical activity using Actical physical activity

monitors are repeated.

7. Approximately 1 year following intervention, measurements encompassing areas of physical activity, physicalattributes, psychosocial attributes, balance, and strength are repeated.

8. Trainers are required to interact with the riders guardian to help identify methods used at home to motivate therider and to manage the riders behavior.

Specialized Equipment

Adapted Bicycle

The method to providing stability while learning involves mechanically modifying the bike so as to mitigate itsinstability.b The purpose is to begin with a stable, specially designed adapted bicycle and to incrementally progressto a 2-wheel bicycle. The progression can be altered to meet the individual needs of the riderb:

1. A rear handle is fitted to the bike so that the trainers can better protect and guide the participant.

2. The seats are typically larger in size and positioned lower so that the participants feet are flat on the ground whileseated on the bike.

3. The special rollers are fitted on the rear of the bike in place of a standard wheel. The rollers facilitate a movementthat is similar to a 2-wheel bike, a simulation that training wheels cannot offer, minimizing the likelihood of falling.

4. The rollers prevent quick tipping by changing the effect of gravity on the bicycle. The roller has a wide, crowned

surface, unlike the narrow surface of a standard bicycle wheel. When the bike tips on this wide, crowned surface,the mass of the rider is lifted so that the bike tips more slowly. c

(Continued)





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Appendix.Continued

5. Roller wheels taper as they move up in difficulty from level 1 to level 8. These rollers aid in the progression ofskills leading to riding a standard 2-wheel bicycle.

6. The drive train is fitted with a fixed wheel, meaning there are no pedal brakes and the pedals continuously rotateforward with or without force being applied.

Hand BrakesHandle

Seat

RollerFixed Wheel

Riser

Pedal Shank

Bicycle With Adaptations

Adapted Bicycle With Rollers Standard 2-Wheel Bicycle With Spotting Handle

(Continued)





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Appendix.Continued

7. The pedal shank is shorter, which, in turn, reduces the range of motion in the hip needed to complete a pedalrotation, minimizing loss of balance.

8. A riser is added to the handlebars to promote an upright, seated position while riding.

9. Hand brakes are used as the primary method of braking. Not having a foot brake minimizes the functions requiredfor the riders foot, allowing the rider to concentrate on pedaling forward. The hand brake is attached to the rear

wheel of the bicycle only.

Training Team Roles

Floor Manager

During each camp, an experienced trainer assumes the role of floor manager. The floor manager has the respon-sibility of managing the training team and the teaching progression of each participant. The floor manager also isactive in assisting the training team with communication with guardians. Specifically, the floor manager is respon-sible for the following:

1. Keeps logs of participants skill level, equipment use, and progression.

2. Assesses and records participants ability level.

3. Determines when participant is ready to progress to higher levels.

4. Provides trainers with teaching tips.

Bike Technician

During each camp, an experienced trainer assumes the role of bike technician. The bike technician has theresponsibility of maintaining the adapted bikes and making adjustments to equipment such as adjusting handlebars,fitting the seat, and changing rollers. The bike technician also rides with the participant on a tandem bicycle, whichallows the participant to experience how to correctly turn, brake, and pedal fast. Specifically, the bike technicianis responsible for the following:

1. Adjusts bikes to meet each individuals physical needs.

2. Maintains bikes.

3. Adjusts bikes to higher levels.

4. Rides with participant on tandem bike.

Trainer

During each camp, a trainer is paired with an individual rider and is responsible for training that same individual forthe entire duration of camp. Trainers have experience working with individuals with disabilities, and many have

teaching or physical therapy experience. Trainers need to communicate with the participant and guardian tounderstand the unique learning style of that participant. With this information, the trainer is able to individualize thetraining strategy to meet the needs of the participant he or she is working with:

1. Trainers are paired with the same participant throughout camp.

2. Trainers have experience working with individuals with disabilities or teaching experience.

3. Trainers individualize their training strategy to meet the unique needs of each participant.

4. Trainers communicate with guardians to educate them on the training process so they can supplement continuedtraining at home, monitor their childs riding, and encourage continued practice of the skills learned at camp.

(Continued)





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Appendix.Continued

Individualized Training Strategies

Each trainer is responsible for developing a training strategy to meet the unique learning needs of each individual.

Specifically, the trainer typically individualizes training using many of the following techniques:

1. Maintain minimal talking.

2. Reduce fear.

3. Eliminate falling.

4. Increase focus.

5. Guide steering.

6. Lean bike into turns.

7. Increase riding time, motivate individual to continue to pedal.

8. Reinforce positive behaviors.9. Communicate with participant, guardians, and other team members.

10. Supplement communication as needed through ASL or picture vocabulary.

Training Progression

The adapted bicycles are designed to do the teaching. The training team facilitates the process. The participants learnto control and maintain the bicycle through their physical and visual experiences while riding, rather than throughreliance on explanations and demonstrations.b Individuals, especially those with disabilities, learn more effectively

with an experiential approach.d The training progression is designed to advance participants through a series ofadapted equipment that can be individualized, providing experience and continued success at a pace that isappropriate for each rider. The training progression is outlined in the following:

Sequence of Training Progression

1. Roller Bike Levels 18 Tandem bike ride with bike technician Typically implemented for first 34 sessions

2. Straight Launch

Ride 2-wheel bike straight and practice braking with trainer spotting Typically implemented in fourth or fifth session

3. Brake Box

Stationary braking practice while bike is immobilized Typically implemented when rider struggles with braking during the straight launch trial

4. Launch

Ride 2-wheel bike for a longer distance on a straight or curved path and practice braking with trainer spotting Typically implemented after successful straight launch with braking

5. Self-Launch

Practice self-starting the bike without trainer assistance Practice self-launch, maneuvering and braking without trainer assistance Typically implemented after much demonstrated success with launch, braking, and maneuvering

a The trainingmanual should not be used or reproduced without permission from Lose the Training Wheels organization (http://losethetrainingwheels.org/contact.html).b Klein RE, McHugh E, Harrington SL. Adapted bicycles for teaching riding skills. Teaching Exceptional Children. 2005;6:5056.c Fiske B. Father of intervention. Bicycling.2005;11:3335.dWilson C. PIPSSPlayground Intervention Program for Social Skills. In: Proceedings of the 13th International Symposium on Adapted Physical Activity; July2001; Vienna, Austria. 2001:197201.



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doi: 10.2522/ptj.20110061Originally published online August 18, 2011

2011; 91:1463-1477.PHYS THER.Tiernan and Joseph E. HornyakDale A. Ulrich, Amy R. Burghardt, Meghann Lloyd, ChadSyndrome: A Randomized TrialTwo-Wheel Bicycle for Children With DownPhysical Activity Benefits of Learning to Ride a

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