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Parental

Response

to Repeat

Testing

of Infants

with ‘False-Positive’

Results

in a Newborn

Screening

Program

James

R. Sorenson,

PhD, Harvey

L. Levy, MD,

Thomas

W. Mangione,

PhD, and Stephen

J. Sepe,

MPH

From the Boston University School of Medicine, School of Public Health, Massachusetts Metabolic Screening Program, and Center for Survey Research, Boston

ABSTRACT. Forty-five percent of the parents of 60

in-fants being retested in a newborn screening program for

metabolic disorders understood that their infant was

undergoing retesting because the first test result was

abnormal. Fifty-five percent had incorrect or incomplete information, believing that retesting was routine, or that mistakes had been made in the original testing procedure, or they report being told nothing specific about the

test-ing. Parents who were aware that the initial test was

abnormal were no more anxious or depressed while

wait-ing for the repeat test results than other parents. At a

second interview after learning the normal results of the repeat test, both those parents informed of the initial abnormal result as well as those not informed were less

anxious and depressed. However, 36% of the parents of

these normal infants reported concern about the health

of their infant because of the repeat testing. This concern was not related to a parent’s knowledge that the initial test result was abnormal, but was greater in parents

reporting that they had not received sufficient informa-tion about the screening/testing process and its

signifi-cance for the health of their infant. Pediatrics 1984;

73:183-187; newborn screening program, parental

re-sponse to repeat testing, testing.

Routine newborn screening for a variety of

dis-orders is conducted in many parts of the wonld.13

Such screening is designed to diagnose diseases

such as phenylketonunia within the first few days

after birth when the infant is clinically normal and

when treatment can prevent the usual clinical

course of an untreated disease.4

Newborn screening tests identify three groups of

infants: those who do not have a disease, those who

Received for publication Feb 1, 1982; accepted March 29, 1983.

Reprint requests to (J.R.S.) Boston University School of

Medi-cine, School of Public Health, 80 East Concord St, Boston, MA

02118.

PEDIATRICS (ISSN 0031 4005). Copyright © 1984 by the

American Academy of Pediatrics.

do, and those whose test results are outside the

accepted normal range, but not so aberrant as to

indicate a disease. These “suspicious” cases require

a repeat test to distinguish the rare infant who has

a disease (true positive) from the far more common

case in which the initial suspicious result is due to

a transient biochemical on hormonal perturbation

(false positive).

Requests for repeat specimens are usually made

after an infant has been released from the hospital.

Parents take the infant either to their physician on

a clinic for the repeat test. As virtually all of these

infants will have a normal result on the second test,

there is concern that telling parents the initial

result was elevated, ambiguous, or that there might

be a problem will lead to unwarranted anxiety,

depression, or worry among parents.

There have been no studies that assess directly

the nature and extent of parental reactions to

re-peat newborn screening. Therefore, a study was

designed to assess (1) parental understanding of

the reason for repeat testing of their infant; (2)

parental response to the repeat test as a function

of their understanding of the reason for the retest;

and (3) the frequency of parental concern resulting

from such repeat screening.

PROCEDURES AND METHODS

Subjects for this study came from the

Massachu-setts Newborn Screening Program.5 This program,

which tests for a variety of disorders, consists of

multiple tests performed on filter paper specimens

of blood and urine from newborn infants.

Addi-tional specimens are requested when a result in one

of these initial tests is considered to be abnormal.

These requests are made through a letter from the

State Laboratory Institute to the physician or clinic

(2)

primary specimen. (For extremely elevated results

a telephone call may precede the letter. This study

excluded such cases.) This letter explicitly states

that the initial result was abnormal, that this

sug-gests the possibility of a disorder (the specific

dis-order is identified and briefly described), and that

an additional specimen is necessary to rule out a

true positive result. This letter states that the

ab-normal test result is very likely a transient

bio-chemical on hormonal perturbation.

The physician on clinic to whom this letter is

sent is responsible for contacting parents,

explain-ing the need for the repeat test, arranging for the

additional specimen, and reporting the results of

the retest to parents. If test results on the repeat

specimen are abnormal, the infant is referred for

further evaluation. If the repeat test result is

non-mal, the initial result is considered a false positive.

Between October 1978 and June 1979, there were

68 families in which the initial newborn test result

was suspicious and a repeat specimen was

re-quested. Parents were sent a letter by the research

group after they had taken their infant to a

physi-cian or clinic for the repeat test. The letter stated

that a study was being conducted to assess parental

experiences with newborn screening, that the

ne-search group knew only that a repeat specimen was

requested, and that participation was voluntary and

confidential.

Data collection involved two parental interviews,

the first after collection of the specimen for the

repeat test but before parents knew the results, and

the second after the parents learned the results of

the repeat test. If the repeat test result was

abnor-mal, the family was excluded from the study.

Interviews were conducted on the telephone

ex-cept in cases in which respondents requested a

personal interview. Twenty percent of the

inter-views were conducted in person. Analysis of data

collected via telephone compared with data from

personal interview revealed no significant

differ-ences in respondent characteristics or study

van-ables. In all cases, interviews were conducted with

the parent who took the newborn to the physician

on clinic for the repeat test.

Information was collected on parental age,

edu-cation, race, understanding of the reason for the

repeat test, and knowledge of the disorder involved.

Parental psychological status was assessed with the

Multiple Affect Adjective Checklist (MAACL),6 a

132-item test that measures anxiety and depression

through respondent assessment of his on hen feeling

states. The form used in the study was the “today”

or state form, which is intended to assess

psycho-logical state rather than more permanent

person-ality traits.

RESULTS

Of the 68 parents eligible during the study period,

60 (88%) participated; two refused to participate,

and six did not participate because of family illness

on our inability to establish contact. Fifty-five of

the participants were the mother and five the father

of the retested newborn.

First Interview

The presumptive disorders for which repeat

spec-imens were requested are as follows: galactosemia,

33%; maple syrup urine disease, 20%;

homocysti-nunia, 20%; phenylketonunia, 20%; histidinemia,

3%; iminoglycinunia, 2%; and cystinunia, 2%.

Spec-imens requested for suspicion of congenital

hypo-thynoidism were excluded because this aspect of the

screening program had just begun.

In 63% of cases, some special action, such as a

call from the physician or clinic, was taken to notify

parents of the need for a specimen (Table 1). In the

remaining cases a routine procedure, usually a

well-baby visit, was utilized. (Massachusetts physicians

may not have contacted parents for an immediate

retest, knowing that if the initial result was highly

elevated they [the physician] would have received

a call from the State Laboratory.)

The reasons for the repeat test that parents

ne-port being given by their provider are listed in Table

2. Forty-five percent of the parents reported being

told that the initial test result was abnormal

whereas 55% reported being told something else.

Although 25% knew the precise reason for the

repeat test, namely that the first test result was

elevated, less than one third, 32%, reported being

told something at variance with what them provider

was told by the state laboratory.

The 60 parents discussed the repeat test with

various acquaintances while waiting for results:

spouse, 75%; parent, 30%; health professional, 25%;

friend, 21.7%; and sibling, 13.3%. Although not

shown, approximately 60% of those discussing the

repeat test with their spouse indicated that anxiety

about their infant’s health was a reason for the

discussion. In 24% of contacts with nonspouses,

anxiety was mentioned as a reason for the

discus-sion.

TABLE 1. Method Used to Inform Parent of

Repeat Test (N = 60)

Need for

Method %

Special effort

Doctor’s/clinic secretary or nurse called or

sent postcard 36%

Doctor/clinic called parent 27%

Routine office/clinic visit

Other

63

(3)

TABLE 3. Mean Parental Knowledge

Multiple Affect Adjective Checklist of Initial Test Results

(MAACL) Anxiety and Depression Scores by Interview and

Psychological Dimension

1st Interview 2nd Interview Mean Difference Scores

1st to 2nd Interviews Total Population . (5) 4.5 .000 (4.8) Anxiety

1st Test Other Reason for

Abnormal Repeat Test

(1) (2) Pt

10.7k 9.9 NS

(3.6) (4.4) 1st Test Abnormal (3) 6.2 (3.3)

Other Reason for

Repeat Test

(4) Pt

5.3 NS

(3.1)

Depression 18.7 16.8 NS

(4.5) (5.5) 15.6 (4.9) 14.0 NS (5.8) 2.8 .001 (6.4)

N 27 32 25 32 58

* Values shown in columns 1 to 5 are means; SD is shown in parentheses.

t

Determined t test, independent samples.

:1:Determined by t test, dependent samples.

At the time of the first interview, 81% of the

parents believed that the repeat test would show

their infant to be healthy, whereas the remainder

said they did not know what the test would show.

None of the parents believed that their infant would

be found to have a disorder.

A comparison between parents who understood

that the first test result was abnormal and the other

parents revealed no significant differences in

dis-cussion of the repeat test with various people, nor

in beliefs about the actual health status of their

infant.

At the time of the first interview (Table 3,

col-umns 1 and 2), there were no significant differences

in either anxiety or depression between parents who

understood that the initial test results were

abnon-mal and those who did not have this information.

Analysis also revealed that parents informed

about the need for a repeat test by a special contact

were no more or less likely than parents informed

during a routine visit to discuss the repeat test with

others, to be uncertain about the health of their

infant, on to be more anxious or depressed.

Second Interview

The second interview took place within 14 days

after the parents were informed that results of the

TABLE 2. Reasons Providers Gave Parents

Test (N = 59)

for Repeat

Parent Told %

Initial test result abnormal 45

1st sample elevated 25%

1st sample ambiguous 12%

May be a “problem” 8%

Other reason 55

Told nothing specific 23%

Repeat testing routine 19%

Mistake made on 1st test 10%

1st test lost 3%

repeat test were normal. Twelve of the 60 parents

were not informed of the repeat test results by their

physician on clinic; presumably they were told that

they would not hear from their provider unless

there was a problem. These parents were

inter-viewed by the researchers 2 to 4 weeks after their

infant was retested.

Forty-two percent of the parents who learned the

results of the repeat test did not wait for their

physician to call. Rather, they initiated the contact.

Very few parents reported discussion of the repeat

test results with spouses, friends, or relatives; 15

parents did discuss the results with a professional

other than their regular provider, usually for the

purpose of obtaining information.

As shown in Table 3 (columns 3 and 4), at the

second interview parents who understood that the

initial test result was abnormal were no more

anx-ious or depressed than parents who had a different

understanding of the reason for the repeat test. An

examination of Table 3 data also shows significant

reductions in anxiety and depression among both

groups of parents from the first to the second

interview (column 5). However, there was concern

on doubt about the health of the infant among some

parents. When asked about this, 64% ofthe parents

said they had no concern, but 36% had concern,

with about half of these expressing a great deal of

concern. This concern was not related to whether

on not the parent knew that the initial test result

was abnormal. Moreover, this specific concern

about the health of the infant was not related to

the more general assessments of parental anxiety

and depression. In addition, parents expressing

doubts about the health of their infant at the second

interview were not necessarily those who reported

these doubts at the first interview, suggesting that

these concerns developed during the repeat testing

(4)

Analysis of data implies that the absence of

in-formation by parents about the screening/testing

process, as distinct from the repeat test result, may

have contributed to such concerns. For example,

parents expressing doubt about their infants’ health

after the screening process, compared with those

who did not, were more likely to say that not enough

information about the testing process had been

provided by their physician or clinic (50% v 22%,

P < .05). Also, they were more likely to want to

talk to someone about both the health of their

infant (40% v 8%,

P

< .01) and about the results

of the repeat test (80% v 20%, P < .01). Moreover,

parents who reported being told nothing specific

about the repeat screening program at the first

interview, were at the second interview somewhat

more likely than other parents to want to talk with

someone about the results of the repeat test (73%

V 31%, P = .06). They also tended to be less satisfied

with their screening experience than other parents

(31% v 59%, P < .05). A similar conclusion

con-cerning the negative effect of too little information

by participants was reached after an analysis of a

Tay-Sachs screening program.7

Finally, 78% of the parents reported that they

were satisfied or very satisfied with the services

provided by the screening laboratory, and an almost

equal number (72%) were satisfied with their

doc-ton’s or clinic’s services. Of those expressing

dissat-isfaction, most said that the screening program took

too long to report results and that their physician

on clinic had not supplied enough information.

DISCUSSION

The results reported here should be viewed with

certan study limitations in mind. First, we relied

upon parental, not provider reports, to determine

the information that parents obtained about the

screening/testing process and results. We suspect,

however, that there is very high correspondence

between what providers told parents and what

par-ents report they were told, in large part because of

the recency and the importance of the screening!

testing events for these new parents. Even if there

are occasional differences, however, the results

ne-ported here remain important, for they document,

at a minimum, the relationship between parental

beliefs and information about the screening/testing process and their response to this experience.

Second, the assessment of parental anxiety and

depression is based on self-reported feeling states.

It is possible that some parents may have felt it

necessary to be less than candid in reporting their

feelings or that psychological processes, such as

denial, may have influenced self-reports. Although

the instrument used has demonstrated reliability

and validity, a clinically and behaviorally focused

assessment might have permitted more subtle

ex-amination of parental psychological status.

With these limitations in mind, we believe that

the present study provides support for one of the

recommendations of the Committee for the Study

of Inborn Errors of Metabolism of the National

Academy of Sciences.8 This committee, after

delib-erating the pros and cons of disclosing initially

aberrant test results to parents before retesting the

infant, recommended disclosure. They did so largely

on the belief that parents have a night to be fully

informed about all aspects of the care and

treat-ment of their infant. There was concern,

neverthe-less, that disclosure of such results, virtually all of

which turn out to be false positives, would

psycho-logically upset parents. The present study

demon-stnates that disclosure of such initial aberrant

screening results does not appear to entail any

significant risk of making patients anxious or

de-pressed. More specifically, the data show that

pan-ents informed of the initial abnormal results,

com-pared with those not informed, were no more

anx-ious on depressed, nor did they have more concerns

about the health of their newborn, either while

waiting to learn the repeat test results or after

learning them.

The present study also demonstrates, however,

that having an infant retested can lead to parental

concern. Even when normal retest results are

re-ported, if parents have not received adequate

infon-mation about the screening/testing process and its

implications for their infants, then some concern

about the health status of the newborn may result.

For some parents, knowledge of the results of

screening tests constitutes insufficient information. A more complete understanding of the screening/

testing process is required. Such information, in

providing knowledge about the potential health

problems and hence the necessity of netesting, may

enable parents to put the repeat test and its results

in perspective.

We cannot determine from the present study

whether the concerns expressed by the parents will

last a week, a month, or longer, non can we tell from

this study whether the doubts about their infant’s

health will lead parents to seek special cane or view

their infant as being at nisk for special health

prob-lems.9 What this study does demonstrate is (1)

informing parents that an initial screening test

result is abnormal did not psychologically upset

them and (2) providing information about the

proc-ess of newborn screening/testing, rather than only

the results, can reduce the chance that parents will

develop concerns about their infant’s health

(5)

ACKNOWLEDGMENT

This work was supported, in part, by a contract from

the Division of Family Health Services, Massachusetts Department of Public Health, Boston.

REFERENCES

1. Levy HL: Genetic screening. Hum Genet 1973;4:1-104

2. Levy HL, Mitchell ML: Regional newborn screening for hypothyroidism. Pediatrics 1979;63:340-342

3. Mamunes P: Neonatal screening tests. Pediat Clin North Am 1980;27:733-751

4. McBean MS: The problems of parents of children with

phen-ylketonuria, in Bickel H, Hudson FP, Woolf LI (eds):

Phen-ylketonuria and Some Other Inborn Errors of Amino Acid Metabolism. Stuttgart, Gang Thieme Verlag, 1971, pp

280-282

5. Levy HL: Neonatal screening for inborn errors of metabolism.

Clin Endocrinol Metal, 1974;3:153-166

6. Zukerman M, Rubin B: Manualfor the Multiple Affect

Adjec-tive Checklist. San Diego, CA, Educational and Industrial

Testing Service, 1965

7. Childs B, Gordis L, Kaback MM, et al: Tay-Sachs screening:

Social and psychological impact. Am J Hum Genet 1976;28:550-558

8. Committee for the Study of Inborn Errors of Metabolism,

National Academy of Sciences: Genetic Screening, Programs, Principles, and Research. Washington, DC, National

Acad-emy of Sciences, 1975, pp 259-260

9. Levy JC: Vulnerable children: Parents’ perspectives and the use of medical care. Pediatrics 1980;65:956-963

LATE AT NIGHT THOUGHTS

We have always known that childhood is the period in which new languages

as well as one’s own can be picked up quickly and easily. The facility disappears

in most people around the time of adolescence, and from then on the acquisition

of a new language is hand, slogging labor. Children are gifted at it, of course.

But it requires a different order of respect to take in the possibility that children

make up languages, change languages, perhaps have been carrying the

respon-sibility for evolving language from the first human communication to twentieth

century speech. If it were not for the children and their special gift we might

all be speaking Indo-Eunopean or Hittite, but here we all are, speaking several

thousand different languages and dialects, most of which would be

incompre-hensible to the human beings on earth just a few centuries back.

Submitted by Student

From Lewis Thomas: Late at Night Thoughts on Listening to Mahler’s Ninth Symphony. New York,

(6)

1984;73;183

Pediatrics

James R. Sorenson, Harvey L. Levy, Thomas W. Mangione and Stephen J. Sepe

Newborn Screening Program

Parental Response to Repeat Testing of Infants with `False-Positive' Results in a

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1984;73;183

Pediatrics

James R. Sorenson, Harvey L. Levy, Thomas W. Mangione and Stephen J. Sepe

Newborn Screening Program

Parental Response to Repeat Testing of Infants with `False-Positive' Results in a

http://pediatrics.aappublications.org/content/73/2/183

the World Wide Web at:

The online version of this article, along with updated information and services, is located on

American Academy of Pediatrics. All rights reserved. Print ISSN: 1073-0397.

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