Letter to the Editor

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TABLE 2. Results of Second Hearing Screening on Universal Basis

952 LE1TERS TO THE EDITOR

Hearin g Loss? Totals

Yes No Test Positive Screen Fail n Row % Column % 3997 47.89 100.00 4 348 52.11 3.30 8345 6.15 Test Negative Screen Pass n Row % Column % 0 0.00 0.00 127 421 100.00 96.70 127 421 93.85 Totals n Row % 3997 2.94 131 769 97.06 135 766.00

withholding known effective therapeutic intervention for

hearing loss;

2. It would bedifficult toimplement on thebasis of “informed consent” (since parents of such children could not possibly be informed as to the nature of the impact ofwithholding intervention);

3. It might be difficult to support on an ethical basis (witness some

of the negative reactions to the placebo study of Bretlau and

colleagues, 1981);6

4. It may place the investigators and their affiliated “deep-pock-ets” in a medically-legal difficult position. For example, one could envision a scenario whereby early intervention is shown

to provide some benefit. Then, the parents of those children for

whom either a placebo or no intervention was given might

interpret the absence of intervention to constitute a significant

infringement on their child’s abifity to pursue happiness (a

right guaranteed by the Constitution) and could take the

inves-tigators to task-asserting any academic, behavioral, social, or

emotional difficulties thenceforth encountered are the result of

the absence of early intervention, given no absolute proof to the contrary-for a rather not-so-scientific compensation).

Under those conditions, the pure scientist may be viewed not so

much as a person who enhances the knowledge that benefits

society, but may be viewed as something less than a hero for using children as guinea pigs in scientific experiments.

Bess and Paradise have raised some thought-provoking

per-spectives on universal neonatal hearing screening. We do not

believe that considerate caution should preclude the establish-ment of a national goal. In addition, we believe that the absence of

incontrovertible evidence detailing the benefits of any clinical

treatment approach should not prevent the clinician from using

the best available evidence to date (with all its imperfections

recognized) to formulate the most appropriate course of action for

the care of patients in conjunction with his best clinical judgment.

MICHAEL J. M. Rinri, Pi-iD

Department of Audiology

Loyola University Medical Center

Maywood, IL

GREGORY J.MATZ, MD

Department of Otolaryngology

Loyola University Medical Center Maywood, IL

REFERENCES

1. Bess FH, ParadiseJL. Universal screening for infant hearing impairment: not simple, not risk-free, not necessarily beneficial, and not presently justified. Pediatrics. 199493:330-334

2. Teele DW, Klein JO, Chase C, Menyuk P, Rosner BA, and the Greater Boston Otitis Media Study Group. Otitis media in infancy and intellec-teal ability, school achievement, speech, and language and age 7 years.

Iinfect Dis. 1990;162:685-694

3. HallJW, Kileny PR, Ruth RA, Peters-KripalJ. Newborn auditory screen-ing with Algo-1 vs. conventional auditory brainstem. Paper presented at the American Speech-Language-Hearing Association Convention 1987, New Orleans, LA

4. Statistical Abstracts of the United States 1993-The National Data Book.

United States Department of Commerce 1993, ISBN: 0-16-0420474 5. Raffin MJM, Matz GJ. Research design for early intervention. Audiology

Today 1994. In press

6. ThomsenJ, Bretlau P.Toss M,Johnsen NJ. Placebo effects in surgery for M#{233}ni#{232}re’sDisease. Arch Otolaryngol. 1981;107:271-277

To the

Editor.-The importance of early identification of hearing loss is

sum-marized in the report issued by the United States (US) Department

of Health and Human Services,’ entitled “Healthy People 2000.”

The goal is to reduce the average age at which children with

significant hearing impairment are identified to no more than 12

months. The report states:

The future of a child born with significant hearing impairment

depends to avery large degree on early identification (ie, audio-logical diagnosis before 12 months of age) followed by

immedi-ate and appropriate intervention. If hearing-impaired children

are not identified early, it is difficult, ifnot impossible, for many

of them to acquire the fundamental language, social, and

cogni-tive skills that provide the foundation for later schooling and success in society. When early identification and intervention occur, hearing-impaired children make dramatic progress, are

more successful in school, and become more productive

mem-bers of society. The earlier intervention and habilitation begins,

the more dramatic the benefits (p. 460).

On the average, children in the US with severe to profound

hearing impairment are not identified until 24 to 30 months of age, while children with milder, yet nonetheless, significantly

detri-mental hearing losses frequently are not identified until they are 5

to 6 years of age.’ It is also alarming that only about 3% of all

children born in the US participate in any type of newborn

hear-ing-screening program.3 Thejoint Committee on Infant Hearing is

in the process of revising their position on infant screening. Their

1990 Position Statement recommended hearing screening only for

infants who exhibit one or more of ten risk factors for hearing loss.4 However, recent research has revealed that as many of half

of all children with bilateral severe-to-profound hearing losses

have not exhibited any of the high-risk factors.54

The September 28, 1993 draft of the Joint Committee on

Infant Hearing Position Statement recognizes these findings

and states:7

Because normal hearing is critical for speech and oral

lan-guage development as early as the first six months of life, hearing loss ininfants must be identified before three months

of age. To acquire access to most infants, the Joint Committee

on Infant Hearing recommends screening infants prior to

hospital discharge from the newborn nursery.

... a method of hearing screening must be able to identify infants with hearing losses of 30 dB HL and greater in one or both ears, in the frequency region important for speech

recog-mtion (approximately 500 through 4000 Hz). Of the various

approaches to newborn hearing screening currently available, two physiologic measures [auditory brainstem response (ABR)

and otoacoustic emissions (OAE)l most nearly achieve this goaL

In addition to the National Institutes of Health Consensus

Statement in support of universal hearing screening,8 it is clear

that many professionals from different disciplines now believe the

time has come to support universal hearing screening. Much of the

recent support has come as a result of studies showing that OAEs

are capable of identifying infants with hearing loss of

approxi-mately 30 dB HL and greater.93

I have personal admiration for Doctors Bess and Paradise and their long-standing contributions to our understanding of effects

of hearing disorders on children. However, I must express concern

at Viet Nam:AAP Sponsored on September 1, 2020

www.aappublications.org/news

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LETFERS TO THE EDITOR 953

with their conclusions about the validity, predictive value, cost,

practicability, and efficacy of using evoked otoacoustic emissions

(EOAEs) for mass screening for infant hearing impairment, which

led them to write the article entitled “Universal Screening for

Infant Hearing Impairment: Not Simple, Not Risk-Free, Not

Nec-essarily Beneficial, and Not Presently Justified.” ‘

Doctors Bess and Paradise make some assumptions regarding

prevalence of hearing loss, sensitivity, and specificity of TEOAE

measures, use of the ABR for the second screen, and data taken

from “laboratory” conditions for testing newborns under the

heading of “Validity and Predictive Value.” I respectfully disagree

with their assumptions.

Although prevalence of bilateral profound hearing loss in

chil-dren has historically been reported as I to 1.3/1000, our popula-tion of interest is unilateral/bilateral mild to profound

season-neural hearing loss where the prevalence is estimated to be 1 .5 to

6/1000.13.15.16 For mathematical examples I will use the

preva-lence of 5.95/1000 reported by White and Behrens (1993).’

They also reported that sensitivity of TEOAEs approaches

100%, based on behavioral audiological evaluations as the

“gold standard.”

Recent data on specificity is much better than reported in early studies.’7 The Rhode Island Hearing Assessment Project utilizes

EOAE as the initial infant screen and has now screened over

23 000 newborns since February 1990. With their experience,

soft-ware and hardware modifications, and improved clinical

tech-niques, specificity has improved to above 90% for the initial

screen. Their latest technique includes an immediate recheck for

collapsed canals or ears occluded with vernix caseosa for the

initial EOAE screen. When the initial screening result is “fail” the

recheck includes replacing the disposable tip on the probe and

rescreening immediately. This procedure yielded a fail rate of only

4.9% for 1223 infants tested at Women’s and Infants Hospital

(November through December 1993, K. R. White and B. R. Vohr,

personal communication, 1993-1994).’ The average fail rate for

other hospitals in the Rhode Island Project varies, depending on

the length of screening experience, but averages 8.4%. These fail

rates cover all births regardless of when they were discharged

from the hospital and includes the categories of both “fail” and

“partial pass.” Their screening procedures are accomplished in

nurseries with background noise levels of 45 to 60 dBA, not

unrealistic “laboratory” type sound treated rooms. Usually it is

the infants own noise (breathing, etc) that prolongs the test time beyond I minute. However, if other sounds are problematic, a quiet isolette with a lid reduces the ambient noise levels to 45 to 50

cIBA. In addition, the “fail” rate for the second EOAE screens

accomplished 2 to 6 weeks later is only 9% of those failing the

initial screen. Hospital nursery employees such as trained

assis-tants or clerks perform the screening before discharge.

Audiolo-gists interpret test results from computer files and schedule

no-checks and follow-up.

As an example of a three-step screening program to meet the

goals of thejoint Committee, consider the following cost estimates

using fail rates and prevalence of hearing loss from the Rhode

Island project (based on 1000 live births).

Procedure Number Charge ($) Total ($)

First OAE screen 1000 30.00

(Fail rate 8%)

Second OAE screen 80 30.00

(Fail rate (10%)

30000.00

2400.00

Test battery on eight infants per 1000 who

screens:

fail both OAE

. Threshold ABR including sedation

. Audiologic pediatric evaluation

. High frequency tympanometry

. ENT consult

$270.00 54.00 21.00 101.00

Total Charge $446.00

Total newborn screens = $32 400 + 8

diagnostics (3568)

Cost per newborn for screens and

diagnostic: $35 968/1000

Cost per infant with sensonneural

hearing loss: $35 968/5.95

$5 968.00

s.oo

= $6 045.00

In their article Bess and Paradise calculated the cost to screen 4 million live births at approximately 200 million dollars. The yield

would be 4000 hearing-impaired children at a cost of $50 000 per

identified child. We would estimate the cost to be 144 million to identify 23 800 hearing-impaired children at a cost of $6045 per identified child (only 12% of the Bess and Paradise estimate).

The comments of Bess and Paradise about efficacy are

surpris-ing. First they admit that “Much theoretical understanding,

into-itive belief, and clinical experience argue in favor of efficacy.”

Then they conclude that, “Nonetheless no direct evidence

dem-onstrates conclusively that intervention appropriate by current

standards results in more good than harm to the child and the family.” if they truly believe that current evidence is not

suffi-ciently “conclusive” for universal newborn screening, why then, is

it “conclusive” enough for screening at risk infants? Are we less

concerned about “harm” to families of at risk children than to

families of children not at risk? Interested readers may wish to

review the artide by Kuhl et al17 in addition to the eight

publica-tions supporting early intervention cited by Bess and Paradise.

If one accepts the premise that early identification of hearing

loss and appropriate amplification and language stimulation is

valuable, one way to measure its value would be in educational savings. If efficacy is measured in terms of statistical outcomes,

then indeed, it may be difficult to show that student “X’ will score

better than student “Y’ later on in life on some standard test.

However, placement of students in the educational system is

based on their performance, and if even a small percentage of

children born with hearing loss can reach standard classroom

placement, then the overall savings to society will be sufficient to

pay for the program.

The following example is offered to show how educational cost

savings might offset the costs of universal screening. The cost to educate one child K-12 in a regular dassroom is about $44 000 in

Rhode Island. The cost for a hearing-impaired child in a

self-contained dass is $126 000 and $429 000 in a residential school. By dividing the cost to identify each heating-impaired child by the cost difference between education in a regular versus

self-con-tamed classroom and multiplying the quotient by 100, one can

estimate the percent of early identified hearing-impaired children

that would need to be educated in regular classrooms in place of

self-contained classrooms for the universal screening/diagnostic program to pay for itself in educational savings. For example, $6045 #{247}[$126 000 - $44 000] x 100 = 7.4%. As a second example, if

only 2% of children identified early could be educated in

self-contained classes rather than residential schools, the identification

program would pay for itself.

The views expressed here, I believe, are in concert with

clini-anna who have extensive hands-on experience with testing

in-fants, children, and adults with EOAES. We have found EOAE

measures to yield frequency-specific data and to be simple, quick, noninvasive, objective, sensitive, and cost-efficient. Accurate

mea-surements do not require highly-skilled personnel. Results ofmass

screening have demonstrated success in identifying large numbers of infants with impaired hearing.

Clearly we have reached the point where the usefulness of

OAFS as a universal newborn hearing screening tool should be

considered. The questions that remain pertain to refining the

method, optimizing the procedures, expanding the availability of

services, and the development of models of service delivery to

match national demographics.

MARTIN S. ROBINETrE, PiD

Mayo Clinic

Rochester, NY 55905

REFERENCES

1. US Department of Health and Human Services Healthy People 2000: National Health Promotion and Disease Prevention Objectives. Washington, DC: Public Health Service; 1990

2. Commission on Education of the Deaf. Towards Equality: Education ofthe Deaf. Washington, DC: US Government Printing Office; 1988 3. Bess FH, Hall JW. Screening Children for Auditory Function. Nashville,

TN: Bill Wilkerson Center Press; 1992

4. Joint Committee on Infant Hearing (1990). Position statement. ASHA. 199133(suppl 5):3-6

5. Elssmann SF, Matkin ND, Sabo MP. Early identification of congenital sensorineural hearing impairment. Hear 1. 1987;40:13-17

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954 LE1TERS TO THE EDITOR

6. Mauk GW, White KR, Mortensen LB. Behrens TR. The effectiveness of screening programs based on high-risk characteristics in early identifi-cation of hearing impairment. Ear Hear. 1991;12:312-319

7. Draft: Tuesday, September 28, 1993. Joint Committee on Infant Hearing

1993 Position Statement, pp. 1-11

8. NIH Consensus Statement. Early identification of Hearing impairment in infants and Young Children. March 1-3, 1993;11:1-24

9. Bonfils P, Uziel A, Pujol R. Screening for auditory dysfunction in infants by evoked oto-acoustic emissions. Arch Otolaryngol Head Neck Surg. 1988;114:887-890

10. Stevens J, Webb H, Hutchinson J, Connell J, Smith M, Buffin J. Click evoked otoacoustic emissions compared to brainstem electric response.

Arch Dis Child. 1989;64:1105-1111

11. Stevens J,Webb H, Hutchinson J, Connell J, Smith M, Buffin J. Click evoked otoacoustic emissions in neonatal screening. Ear Hear. 1990;11: 128-133

12. Kennedy C, Kimm L, Dees D, Evans P. Hunter M, Lenton 5, Thornton A. Otoacoustic emissions and auditory brainstem responses in the newborn. Arch Dis Child. 1991;66:1124-1129

13. White KR, Behrens TR, eds. The Rhode Island Hearing Assessment Project:

implicationsfor Universal Newborn Hearing Screening. Seminars in Hearing. 1993;14:1-119

14. Bess FH, Paradise JL. Universal screening for infant hearing

impairment: not simple, not risk-free, not necessarily beneficial, and not

presently justified. Pediatrics. 199493:330-334

15. Watkin P, Baldwin M, McEnery G. Neonatal at risk screening and the identification of deafness. Arch Dis Child. 1991;66:1130-1135

16. Parving A. Congenital hearing disability-epidemiology and identification: acomparison between two health authority districts. mt IPediatr Otolaryngol. 199327:29-46

17. Kuhi PK, Williams KA, Lacerda F,Stephens KN, Lindbloom B. Linguis-tic experience alters phonetics perception in infants by six months of age. Science. 1992255:606-608

To the

Editor.-In their recent Pediatrics commentary entitled “Universal

Screening for Infant Hearing Impairment: Not Simple, Not

Risk-Free, Not Necessarily Beneficial, and Not Presentlyjustified,” Bess

and Paradise argue that “universal screening is ill-considered and

at this time ill-advised.” Bess and Paradise state their general objections to the National Institutes of Health (NIH) Consensus

Statement on Early Identification of Hearing Impairment, and

then express their concerns about the rationale for early

interven-tion for infant hearing impairment and about current infant

screening tests. Bess and Paradise conclude that current

tech-mques for infant hearing screening are too costly and are

unac-ceptable to clinidans, and that the auditory brainstem response

(ABR) has questionable reliability and validity as an infant hearing screening procedure. Their arguments, however, are not based on

direct clinical experience in hearing screening, nor do they cite published clinical studies to support their opinions.

Within the past 15 years, we have each implemented

ABR-based hearing screening programs of infants at risk for hearing loss, and we have periodically published our clinical

experienc-es.’8 Over 14 875 neonates and infants have undergone hearing

screening in our combined programs. For at least 786 of these infants, a serious hearing impairment was identified and con-firmed by diagnostic audiometry. These infants received the ben-efits of early intervention.9 Our collective clinical experience does

not support several of the opinions expressed by Bess and

Para-dine. First, ABR techniques, including conventional and

auto-mated, are readily “accepted” by most clinicians. Secondly, over-referral rates fall far short of the 100-to-I statistic cited by the NIH Consensus Statement, and quoted by Bess and Paradise. For ex-ample, even in the intensive care nursery setting, which is not the

ideal environment for hearing screening, and with the intensive

care nursery population, which is characterized by a 4 to 5% prevalence of significant hearing loss, our collective ABR failure rates are on the order of 8%. That is, only two infants are referred

for every hearing-impaired infant. Failure rates for automated

ABR in the well-baby population are 5% or less.’0 Finally, the

charge for automated ABR hearing screenings are not inconsistent with hospital charges for other relatively minor procedures.

Despite their stated concerns about hearing screening with

ABR, Bess and Paradise conclude in the last sentence of their

commentary that hearing screening of at risk infants with an

automated ABR technique is practical and cost-effective. We

con-cur wholeheartedly. This important point should not be lost in the dialogue and debate generated by the NIH Panel recommendation of universal hearing screening. Bess and Paradise recommend reliance on the high-risk register to identify infants with hearing impairment. It is important to keep in mind, however, the well-appreciated limitation of the high-risk register. Namely, the

ma-jority of children with significant hearing impairment are not

identified with the high-risk register.

J.MICHAEL Dtsusas, Pi-iD

Department of Otorhinolaryngology

University of Oklahoma Health Sciences Center

Oklahoma City, OK

JAMES W. HALL ifi, PiiD

Division of Hearing and Speech Sciences

School of Medicine

Vanderbilt University

Nashville, TN

Joi-mi T. JACOBSON, Pi-iD

Eastern Virginia Medical College

Norfolk, VA

PAUL R. KILFNY, PHD

School of Medicine

University of Michigan

Ann Arbor, MI

Roc A. Run-i, Pi-iD

University of Virginia Health Sciences Center

Charlottesville, VA

REFERENCES

1. Dennis JM, Sheldon R, Toubas P. McCaffee MA. Identification of hear-ing loss in the neonatal intensive care unit population. Am I Otol. 19845:201-205

2. Hall Ill JW. HandbookofAuditory Evoked Responses. Needham, MA: Allyn

& Bacon; 1992

3. Hall ifi JW, Kripal JP, Hepp T. Newborn hearing screening with audi-tory brainstem response: measurement problems and solutions. Semin Hear. 198815-33

4. Hall ifi JW, Prentice CH. Newborn hearing screening with auditory brainstem response(ABR): experience with 1982 versus l99OJoint Com-mittee Risk Criteria. In: Bess FH, Hall Ill JW, eds. Screening Children for Auditory Function. Nashville, TN: Bill Wilkerson Press; 1992:145-162 5. Jacobson F, Morehouse CR, Johnson MJ. Strategies for infant auditory

brainstem response assessment. Ear Hear. 19823:263-270

6. Jacobson JT, Jacobson C, Spahr R. Automated ABR and conventional ABR screening techniques in high-risk infants I Am Acad Audio!. 1990;

I:187-195

7. Kileny PR. New insights on infant ABR hearing screening. Scand Audio!. 1988;(Suppl 30):81-88

8. Ruth PA, Dey-Sigman S, Mills JA. Neonatal ABR hearing screening. Hear J.198538:39-45

9. Levitt H, McGarr N. Speech and language development in hearing-impaired children. In: Bess PH, ed. Hearing Impairment in Children. York, PA: York Press; 1988:375-388

10. Davis S. Hearing Screening at Baptist Memorial Hospital, Memphis, TN. Paper presented at Universal Infant Hearing Screening National Seminar; November 13, 1994; Nashville, TN

To the

Editor.-In March 1993, the Consensus Panel of the National Institutes of

Health (NIH) Consensus Development Conference on Early

Iden-tification of Hearing Impairment in Infants and Young Children

recommended universal infant hearing screening and reinforced the goal of early intervention for infants with hearing loss. In an

editorial commentary, Bess and Paradise opposed the

recommen-dation for universal infant hearing screening and disparaged the

benefit and effectiveness of early intervention.’ I am concerned

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1994;94;952

Pediatrics

Martin S. Robinette