VOLUME 65 S MARCH 1980 #{149}NUMBER 3

PEDIATRICS Vol. 65 No. 3 March 1980 463

Pediatrics

Early Intervention for Infants with DownSyndrome: A Controlled Trial

Martha C. Piper, PhD, and I. B. Pless, MD

From the School of Physical and Occupational Therapy and Department ofEpidemiology and Health, McGill University, Montreal

ABSTRACT. The mental development of 37 infants with

Down syndrome, allocated either to an experimental or

control group, was assessed over a six-month period byan independent evaluator. The experimental group par-

ticipated in biweekly therapy sessions designed to stim-ulate normal development while the control group re-

ceived no intervention. The Griffiths Mental Develop-mental Scales were used to assess changes in the devel-

opmental status in the two groups, which were shown to

be equal initially on a variety of variables. No statistically

significant differences in mental development betweenthe experimental and control groups were found. The

early intervention regimen investigated in this study wasnot efficacious in altering the pattern of mental develop-

ment in those Down syndrome infants participating in

the program. Pediatrics 65:463-468, 1980; Down syn-drome, early intervention, infants, mental development.

Infant stimulation and training programs have

been developed as means to ameliorate the severity

of the mental handicap associated with Down syn-

drome. Unfortunately, the influence of such pro-

grams on the mental functioning of Down syndrome

children remains uncertain in spite of the fact that

infants with Down syndrome are easily identified at

birth, thereby permitting early intervention within

the first months of life.

Received for publication June 8, 1979; accepted July 16, 1979.

Reprint requests to (M.C.P.) School of Physical and Occupa-

tional Therapy, McGill University, 3654 Drummond St, Mon-

treal, Quebec H3G 1Y5.

PEDIATRICS (ISSN 0031 4005). Copyright © 1980 by theAmerican Academy of Pediatrics.

The contribution of early intervention programs

in deterring mental retardation in children defined

as being “at risk” for retardation at some later date

has been established.’3 However, it is still unclear

whether early intervention remediates retardation

when applied to children with diagnosed forms of

mental retardation, such as Down syndrome. The

distinction between these two populations of chil-

dren, those “at risk” for mental retardation and

those defined as mentally retarded, although essen-

tial when discussing the effect of early intervention,

is often overlooked.

Historically, the positive effect of early interven-

tion for children with Down syndrome was inferred

from comparisons ofthe development of institution-

alized children with that of children reared in the

home.4’9 However, the interpretation of these stud-

ies is extremely difficult since selective factors may

have produced comparison groups that are biased.’#{176}

Although more recent evaluations of early interven-

tion programs for these children report positive

findings, similar difficulties arise regarding the com-

parison groups used in several of these investiga-

tions.”’3 To date, only two evaluations of early

intervention for children with Down syndrome have

employed adequate control groups; but aside from

matching subjects on age, sex, and mental age,

neither study focused attention on other potential

factors that might influence development.’4’5

Moreover, the effect of intervention during infancy

was not evaluated in spite of evidence that mea-

sured intelligence declines as the child with Down

syndrome grows older.9”6”7

The purpose of this study was to evaluate the

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464 EARLY INTERVENTION FOR INFANTS WITH DOWN SYNDROME

efficacy of an early intervention program for infants

with Down syndrome. In order to rigorously exam-

me this question, we conducted a controlled trial by

using comparable experimental and control groups.

Changes in developmental status over a six-month

period were assessed with the Griffiths Mental De-

velopmental Scales.

METHODS AND MATERIALS

Design

A total of 37 infants with Down syndrome, all

under 24 months of age, participated in a pretest,

posttest experiment in which one group (n = 21)

received treatment in an early intervention program

for a six-month period while a control group (n =

16) received no such treatment. Allocation to the

two groups was determined by the date of referral

to the program; infants entering the program be-

tween July and December were placed in the treat-

ment group while those referred between March

and June were allocated to the control group and

received no treatment throughout the summer

months. Those enrolling either in January or Feb-

ruary received treatment but were not included in

the trial because the summer holiday period pre-

vented them from having six months of continuous

therapy. In order to obtain an adequate number of

subjects, we repeated this procedure over a two-

year period, from the fall of 1976 through the fall of

1978.

Program

The intervention program consisted of center-

based biweekly therapy sessions of one hour’s du-

ration designed to encourage the child’s acquisition

of successive developmental levels. Activities to

stimulate normal development, such as rolling, sit-

ting, reaching, or speaking, were demonstrated and

taught to the parent. In addition, a set of written

instructions was given for the parent to follow at

home between sessions. The staff consisted of two

special educators, one child care worker, one social

worker, one occupational therapist, and one physi-

cal therapist. Upon acceptance, a child was assigned

to one staff member who then became the primary

therapist.

Scores on the Griffiths Mental Development

Scales were gathered at admission into the study

and again at its conclusion. Outcome was defined in

terms of the change scores (post minus prescores)

on this measure of mental development.

Measures

To assess the comparability of the experimental

and control groups, we collected the following data

upon admission into the study: chronologic age, sex,

maternal age, birth weight, number of siblings, pres-

ence or absence of congenital heart disease, type of

residential care, status of the home environment as

assessed by the Home Observation for Measure-

ment of the Environment Inventory,’8 and pre-

scores on the Griffiths Scales. It was judged that

any of these variables might affect the performance

of the infants in the trial.

The Home Observation for Measurement of the

Environment Inventory’8 (HOME) identifies those

features in an infant’s environment most likely to

influence development. The nature of the home as

measured by this Inventory, has been shown to be

a good predictor of a normal infant’s intelligence.’9

While no studies of retarded children with this

measure have been reported, we believed that the

quality of a retarded infant’s home environment

could influence the efforts of an intervention pro-

gram.

The HOME Inventory uses 45 items to assess six

categories of stimulation available to the infant:

emotional and verbal responsivity of the mother;

avoidance of restriction and punishment; organiza-

tion of the physical and temporal environment;

provision of appropriate play materials; maternal

involvement; and opportunities for variety in daily

activities. Scoring is based partly on observation

and partly on answers to a semistructured inter-

view. A composite score, as well as scores for each

of the six categories, is obtained. A social worker

who did not know either the design of the study or

the group to which the infant was assigned admin-

istered the Inventory in the home with the child

awake,

The Griffiths Mental Development Scales�#{176}’2’

were selected as the principal outcome measure

because these scales were initially designed to en-

able a detailed differential diagnosis of mental sta-

tus in handicapped children in addition to measur-

ing general ability in normal infants. Furthermore,

this scale provides developmental quotients for five

skill areas, locomotor, personal-social, hearing and

speech, hand and eye, and performance, as well as

an overall measure of mental development. Each

subscale consists of 52 items from birth to 2 years

of age. Unlike the more commonly used Bayley

Scales of Infant Development,22 all five skills are

equally represented at all ages thereby providing a

detailed profile of any child at any age.23 In addition,

whereas the Bayley Scales render information in

only two areas, motor and mental, the Griffiths

Scales provide information for five specific areas.

These features, we believed, would permit a more

sensitive evaluation of the developmental progress

of a handicapped infant. Testing was performed by

a psychologist hired exclusively for this investiga-

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ARTICLES 465

tion who was kept unaware of the group status of

the subjects and of the basic design of the study.

The same psychologist administered both pre- and

post-measures to all children.

RESULTS

Discriminant analyses were performed to deter-

mine the linear combination of variables that best

characterizes the differences between the groups. A

stepwise analysis was first performed to confirm

that the two groups, experimental and control, were

comparable. The means, standard deviations, and

univanate F-ratios for all 19 variables are presented

in Table 1. Only one variable, a HOME Inventory

subscale, discriminated between the two groups (P

= .04), the experimental group having a significantly

higher score on this subscale.

The means, standard deviations, and univariate

F-ratios for the six change scores (post minus pre-

score) on the five subscales and total developmental

quotient of the Griffiths Mental Development

Scales are shown in Table 2. As expected, due to

advancing chronologic age, the mean developmen-

tal quotients in both groups for all subscales de-

dined over the six-month period. However, the

experimental group decreased less than the control

group in only two of the six subscales (hand and

eye, performance). For the remaining four sub-

scales, the control group’s quotients declined less

than those of the experimental group. For example,

the total development quotient in the experimental

group declined an average of 7.33 points over the

six months whereas the control group declined 5.94

points. However, no statistically significant differ-

ences between the two groups were found for any

of the individual change scores.

All six change scores were then employed simul-

taneously in a discriminant analysis to differentiate

between the experimental and control groups. The

derived discriminant function had a X6 of 6.057

with an associated p value of .417. It is concluded

that the combination of the six change scores failed

to discriminate significantly between the two

groups.

Following this, a stepwise discriminant analysis

was performed with the six change scores to deter-

mine the linear combination of the outcome van-

ables that best characterized the difference between

the groups. In order to adjust for the initial differ-

ence in the home environments between the two

groups, the HOME Inventory PLAY subscale was

entered into this analysis on the first step. Following

this adjustment, none of the change scores of the

developmental quotients provided a significant ad-

ditional contribution to the discriminant.

DISCUSSION

There is no statistical evidence to support the

notion that early intervention, as provided in this

trial, was efficacious in remediating retardation in

TABLE 1 . Subject Characteristics of Experimental and Control Groups

Characteristic Experimental (n = 21) Control (n = 16) P Value

Mean SD Mean SD

Child and Family

Chronologic age (mo) 9.33 6.26 8.43 5.79 NSSex (M = 1, F = 2) 1.52 0.51 1.62 0.50 NSMaternal age (yr) 30.43 7.11 29.81 6.00 NSBirth weight (gm) 2,949.10 607.50 2,990.00 568.25 NSNo. of siblings 0.95 0.97 0.81 0.93 NSCongenital heart disease (no = 1, yes = 2) 1.33 0.48 1.38 0.50 NSResidential care (natural = 1, foster = 2) 1.14 0.36 1.06 0.25 NS

HOME*Emotional and verbal responsivity of the mother 8.67 2.42 7.62 2.92 NSAvoidance of restriction and punishment 6.86 0.79 6.69 0.70 NS

Organization of the physical and temporal environ- 4.57 1.12 4.50 1.26 NSment

Provision of appropriate play materials 5.43 2.48 3.62 2.73 .04

Maternal involvement with the child 3.00 1.90 1.94 1.84 NS

Opportunities for variety in daily activities 2.67 1.20 2.38 1.02 NS

Griffiths Scales

Prescore/total quotient 79.38 9.67 78.88 16.60 NSPrescore/locomotor quotient 79.05 10.27 81.25 21.68 NSPrescore/personal-social quotient 84.62 11.95 83.56 19.36 NS

Prescore/hand and eye quotient 76.24 14.42 76.76 18.51 NS

Prescore/performance quotient 72.17 14.88 74.75 17.57 NS

Prescore/hearing and speech quotient 84.57 1 1.54 83.44 15.48 NS

*Home Observation for Measurement of the Environment Inventory.

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466 EARLY INTERVENTION FOR INFANTS WITH DOWN SYNDROME

TABLE 2. Outcome Variables (Post minus Prescores) forGroups

Experimental and Control

Experimental(N=21)

Control(N=16)

P Value

Total developmental quotient

MeanSD

-7.337.79

-5.948.86

NS

Locomotor developmental quotient

MeanSD

-8.8113.31

-2.389.56

NS

Personal-social developmental quotient

MeanSD

-8.1914.36

-7.5610.15

NS

Hand and eye developmental quotientMean

SD

-2.71

12.24

-3.12

16.36NS

Performance developmental quotientMean

SD

-5.71

15.00-9.56

15.84NS

Hearing and speech developmental quotientMean

SD

-11.00

15.64

-7.56

12.85

NS

infants with Down syndrome. The performance of

the experimental group was not superior to the

control group in any of the areas of development

assessed by the Griffiths Mental Development

Scales even after adjustment for the initial differ-

ence between the two groups on the home environ-

ment variable.

The negative findings of this trial are in sharp

contrast to others reported in the literature,”’5

each of which suggests some beneficial effects from

early intervention. Several possible explanations for

this discrepancy should be considered. Because we

did not assess development over the same six-

month period for both groups, a seasonal bias may

be present. Infants allocated to the experimental

group received intervention throughout the fail and

winter months whereas control subjects were as-

sessed for developmental change occurring during

the spring and summer. Accordingly, the experi-

mental infants may have had a slight disadvantage

associated with the timing of their assessments. For

example, some minor illnesses such as colds and flu,

are more prevalent in winter than summer, which

in turn mxy interfere with developmental progress.

Moreover, winter in Montreal is not conducive for

taking young children out-of-doors, whether for

play, shopping, or social visits.

It is also possible that a longer period of interven-

tion would have produced different results. Except

for the study by Aronson and Fallstrom,’#{176} however,

there is no other published evaluation for a period

greater than six months. But, because previous

investigations have dealt with preschoolers rather

than infants, the association between outcome and

length of treatment may be different in our trial

than in others; intervention may be efficacious in

infancy only when applied for a period greater than

six months.

In addition, the positive effect of infant interven-

tion may only be detectable with maturation, al-

though this explanation is not supported by the

experience with culturally disadvantaged children.

Bronfenbrenner3 found the positive effects of inter-

vention diminished following discontinuation of

treatment.

With the documented decline of developmental

quotients over time, the age of the child with Down

syndrome at the time of intervention may also be

a critical factor. Carr24 found that the greatest de-

dine in developmental quotients took place be-

tween 6 and 10 months of age. Because the mean

age of the infants in our trial was nine months, it is

possible that intervention was initiated at a time

when the natural decrease in intelligence was oc-

curring at a rapid rate. Accordingly, arguments can

be made for the necessity to either intervene more

aggressively or for the futility of treatment during

this period.

Similarly, more intensive intervention, regardless

of age, may be necessary although none of the

earlier studies reporting positive results were based

on more intensive treatment than that of the pro-

gram examined in this study. Nonetheless, the in-

teraction of amount of treatment with the age of

the child may necessitate a different approach for

infants than that usually employed with preschool

children.

The actual site of the therapy may also be an

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ARTICLES 467

important factor. The controversy surrounding

home versus center-based treatment is not new. In

this study, parents brought their infants to the

center and received instruction in an activity pro-

gram to be carried out in the home between treat-

ment sessions. In all other intervention studies, with

the exception of that of Hayden and Haring,’2 the

therapist conducted the training sessions in the

infant’s home. However, routine home visiting is

not only expensive in time and personnel, but also

is often not conducive for teaching.� Moreover,

because parents of handicapped children are fre-

quently isolated, the regular weekly visit may be-

come more social than educational. Although in

terms of “support” this may be of great importance,

it may nonetheless interfere with the productivity

of a therapy session. Bronfenbrenner3 in his review

of early intervention programs for culturally disad-

vantaged children, strongly advocated frequent

home visits by a therapist to foster the mother’s

role in the treatment. While home-based programs

may facilitate this parent-child interaction more

successfully than center-based approaches, parent

participation may be more important than the site

of the treatment.

Finally, we did not attempt to assess compliance

with the prescribed regimens. The issue of compli-

ance as it pertains to therapy for handicapped chil-

dren is a complex one; immediate gains from treat-

ment are seldom seen and parents may become

discouraged and discontinue treatment. If parents,

for whatever reason, fail to carry out the recom-

mended program, the problem is one of ineffective

delivery rather than the technique itself. Our con-

cern was not primarily with the intervention tech-

niques per se but rather with assessing the effec-

tiveness of an early intervention program as a

whole.

Assuming that none of these issues were of suf-

ficient importance to detract from the main find-

ings, it should, nonetheless, be stressed that the

results of this study are only based on one mea-

sure-the Griffiths Mental Development Scales.

Although it may be that the use of another assess-

ment instrument would have been more sensitive

in detecting developmental advances, it is impor-

tant to note that the Griffiths Scales were employed

as the outcome measure in several earlier interven-

tion studies where positive findings were

“4,5

Obviously the objectives of early intervention

programs encompass other areas besides the reme-

diation of a child’s retardation. The acceptance of

the child into the family structure, the resolution of

guilt feelings, and the establishment of realistic

expectations are additional reasonable goals. We

limited the focus of our study, however, to the effect

of early intervention on mental retardation. This by

no means rules out the value of other components

of this or other infant stimulation programs al-

though these also remain to be proven objectively.

The failure to demonstrate benefits for the

treated group in this study is disappointing for those

who believe such therapy is an effective method for

minimizing the retardation in Down syndrome. Al-

though other possible explanations exist for these

results, the findings clearly suggest that the efficacy

of this form of early intervention is doubtful. We

conclude, with some minor reservations, that the

particular early intervention regimen investigated

was not efficacious in altering the pattern of mental

development in those Down syndrome infants par-

ticipating in the program.

ACKNOWLEDGMENT

This research was supported in part by a grant fromthe National Health Research and Development Pro-

gram, Health and Welfare, Canada.

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ENDORPHINS AND SHOCK

B endorphin is likely released during shock states and may contribute to

hypotension. Naloxone, in an animal model, rapidly reverses endotoxin-induced

hypotension and also prevents its occurrence. The same findings are noted in

experimental hypovolemic shock. The low toxicity of naloxone and its effect on

shock in experimental animals makes it an attractive agent. Naloxone may be

efficacious in septic shock and in hypovolemic shock.

Comment: Animals only, so far, but these are fantastic data and raise wonderful

possibilities. The soon to come primate data will be exciting to see.

R.H.R.

Abstracted from J. W. Holaday et al: Naloxone reversal of endotoxin hypotension suggests role of

endorphins in shock (Nature 275:450, 1978); A. I. Faden, et al: Opiate antagonists: A role in thetreatment of hypovolemic shock (Science 205:317, 1979).

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1980;65;463PediatricsMartha C. Piper and I. B. Pless

Early Intervention for Infants with Down Syndrome: A Controlled Trial  

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Early Intervention for Infants with Down Syndrome: A Controlled Trial  

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