LETFERS
TO
THEEDITOR
957
if the savings in social expenses could not finance the screening,you have to ask firstly, who would refuse treatment of a patient
with a 10% chance of healing, and secondly, how do you estimate
the value of a better quality of life?
Finally, it would have been valuable if Bess and Paradise would
have put forward alternative constructive suggestions to improve
early identification and thereby diminish the uncertainty of the
statistical effect of early intervention. An example of an alternative
approach would be to compare two health authority districts as
suggested by Parving.4
THE EUROPEAN CONCERTED AcTIoN ON OTOACOUSTIC
EMissIoNs
F. Grandori
(Italy)
L.
Collet
(France)
D. Kemp (Great Britain)
G. Salomon
(Denmark)
K.
Schorn(Germany)
A. R. D. Thornton (Great Britain)
REFERENCES
1. Eggermont, Bock. Acta Otolaryngol. 1986;(suppl 429)
2. Kuhl et al. Science. 1992;255:606-608
3. Kok et al. Audiology. 199332:213-224
4. Parving A. mntIPediat Otolaryngol. 199327:29-46
To the
Editor.-We believe that universal newborn hearing screening is
a
no-cessity. Data indicate that the High
Risk
Register currently used inmany hospitals and recommended by Bess and Paradise only
identifies about 50% of congenital hearing impairments. Causing
further consternation is the fact that the average age of
identifica-tion of hearing impairment in the United
States
remains
at 3 yearsof age. We know that 1/1000 children are born deaf and about
7/1000 have bilateral hearing impairments in the mild to severe
range. Adding hearing loss with late onset, hearing loss associated
with otitis media, and hearing loss affecting one ear or the high
frequency ranges, about 14/1000 school
age children
have hearingloss significant enough to affect their learning and academic
achievement.
We also know that the less substantial the hearing loss is, the
later the identification is likely to be. Because
these
children
do
demonstrate some hearing ability, it is difficult for parents and
other observers to behaviorally assess
true hearing
status. Yet it isthe children with hearing losses other than profound deafness
who respond best to early intervention (hearing aids, parent home
programs, auditory, language and speech therapy, special
pro-school experiences). When the hearing loss is identified early (by
3 months of age), and appropriate intervention is implemented,
these children have the opportunity to learn language and com-munication skills very much like their normal hearing peers.
The financial implications of intervention and educational
ser-vices further supports early identification. Consider the following points:
I. Intervention services can start immediately upon identification of a hearing impairment; these services are usually available within
local communities and often at no direct cost or minimal
cost to
families.
2. Language is essential to learning but it is the primary barrier to
learning for children with hearing impairments; the earlier intervention starts, the better the prognosis for language com-petence, and hence the better the opportunity for participating in regular education classes.
3. Hearing losses that are in the mild to moderate range
are usually
the last to be identified, yet respond best to intervention; these children have the potential to be educated in regular education classes with minimal support.4. Deafchildren can be educated in regular classes with the services of an educational-interpreter if they possess ago-appropriate language skills.
5. With age-appropriate language abilities, children
who
are deafand
hard of hearing can be employed and be contributing members
of the community.
6. Early intervention is a bargain; medical
costs increase
with longer duration before identification; education costs increasesignifi-cantly
with
intensity of services. Asan example,
the
chart
below
compares
regulareducation
and
special
education costfor
educatingColorado
childrenwith hearing
impairments
fordifferent
levels
of
intensityof
services, including early inter-vention (birth through 3); thesedata
clearly
illustrate
the
sub-stantial cost increaseas more
services
are
required for a child. Itshould
be obvious
thatwe either
pay a little bit early on
ina
child’s life
(cost of
screening and early identification services)or pay more
later(more intense
educational
services
due to
the child’s delays-see Table).TABLE. Regular Education Versus Special Education in
Terms
of Cost
Placement
Annual
Cost
Excess Cost/Yr
Regular Education* $4064.75
(No special education services)
Itinerant/Consultative $5767.55 +702.80
(Usually
up to
5 h/wk of specialeducation
services)
Resource $6397.55 +2332.80
(Usually up to 20 h/wk of
special education services)
Self-Contained $12 389.75 +8325.00
(More than 20
h/wk
of
specialeducation
services)
Preschool
$8 193.98 +2129.23(Special
education preschool 3 h/day)Residential
$31 139.00 +27 074.25(Placement
at Colorado
School
for the Deaf and Blind)Early
Home
Intervention
Programt
$2600.00(In home
family intervention-90 min/wk)* Non-special education student cost based on 1993 state average
per pupil
operating
revenue.t
Home
Intervention
Program-Colorado
Department
of Health,
Handicapped
Children’s
Program
(Birth-2).Of
note also is
that it isnot possible
to
attach a monetary valueto
everything.The family
stress
and
frustration
thatexists
whencommunication between a
child
and the parents
is impaired
can-not be easily measured.
Yet
knowing a child’s problemsand
needs asearly
as possible
minimizes
the
anxiety that affects thisrela-tionship.
In summary,
we feel that the benefits
of
screeningall infants
for
hearing impairment far exceed
the problems
identified
in the Bessand Paradise
article.
Contrary
to the authors’ beliefs, data do exist thatsupport
decreasedcosts
to
societywith
early
identification and intervention.Excunv
BOARD OF THE EDUCATIONAL AUDIOLOGYASSOCIATION
Pegy
Von
Almen
Laurie
Allen
Tammie
Adkins
Karen
Anderson
Tricia
Blake-Rahter
Kris
English
Cheryl
DeConde
Johnson,
EdD
Weld
County
School
DistrictGreely,
CO
80631To the
Editor.-Neonatal
hearing screening hascome to
the forefront of interestwithin
our professional
communities
and
among the generalpub-lic, particularly
over the
last year. Identifying childrenwith
hear-ing loss at the earliest
age possible,
however,
has been
a
long-standing tenet of audiologists, pediatricians, otolaryngologists,at Viet Nam:AAP Sponsored on September 1, 2020
www.aappublications.org/news
958
LETI’ERS
TO THE
EDITOR
and early childhood specialists, based both on knowledge of
do-velopmental processes and abundant clinical experiences. Indeed,
the identification of conditions present in early childhood that can result in long-term disabilities are critical components of current
legislative (Individuals with Disabilities Education Act [IDEA],
Part H Public Law 102-119 [formerly 99-457])’ and public health
initiatives (Healthy People 2000).2 Subsequent intervention has
been shown to positively influence outcome when such programs
are initiated in early life.3 Moreover, congenital or early-onset, sensory or neurologic disorders that result in long-term
develop.-mental disabilities are known to be more costly to society when
such deficits are first detected at older ages. Thus, the concept of
hearing screening programs designed to identify debilitating
hear-ing loss in early childhood is well founded. Both the National
Institutes of Health (NIH) Consensus Statement” and the Joint
Committee on Infant Hearing (JCIH) Position Statements
(current-ly in revision) reflect this challenge of the identification of hearing loss as early in life as possible.
Recently, in their commentary in Pediatrics entitled “Universal Screening for Infant Hearing Impairment: Not Simple, Not Risk-Free, Not Necessarily Beneficial, and Not PresentlyJustified,” Bess
and Paradise6 raised important and relevant questions regarding
the concept of mass screening for hearing loss in the neonatal
period. Their letter was motivated by the Consensus Panel
State-ment that resulted from the NIH Conference of March 1 through
3, 1993. Among other recommendations, the Panel advised that universal hearing screening of all newborns be implemented; pref-erably before hospital discharge, but no later than 3 months of age. Specifically, the Panel recommended that evoked otoacoustic emissions (EOAE) be used as the first-level screening procedure, followed by auditory brainstem response (ABR) screening of all
infants who failed the EOAE screen.
Bess and Paradise articulated numerous practical and fiscal
concerns about these recommendations. Moreover, Bess and
Par-adise argued that insufficient empirical evidence exists to support
the sweeping recommendations for universal neonatal hearing
screening and a two-stage screening protocol. They further argued that neither the instrumentation currently available, nor the
test-ing and follow-up protocols suggested met the rigorous
prereq-uisites necessary for instituting any screening initiative. Bess and Paradise specifically questioned: 1) the simplicity, efficiency, reli-ability and predictive value of the screening devices/protocols
recommended; 2) the availability, accessibility, and efficacy of
treatment options; 3) the provisions for ensuring compliance with
follow-up for infants who failed screening; 4) whether thorough
cost analyses of the recommendations had been completed a priori;
and most importantly, 5) whether neonatal hearing screening was
“risk-free” when measured in human terms (the unnecessary wor-rying of parents/caregivers in cases of false-positive outcomes).
As the three pediatric audiologists who served as members of
the Planning Committee for the NIH Consensus Development
Conference, we deem it important to address the comments of
Bess and Paradise in light of our participation in, and perceptions
of, the NIH Conference and the subsequent Consensus Statement.
The body of evidence presented by numerous respected scientists
and
clinicians over the 2-day period that led to the development of the NIH Position Statement was clear. The following facts appear, then and now, irrefutable: 1) our current models for the identifi-cation of potentially-disabling hearing loss in infancy and early childhood are inadequate. For example, the use of the High-Risk Registers for the identification of infants and young children atrisk for hearing loss followed by the direct auditory screening of
those so designated7 identifies only about 50% of young children
who have congenital or early-onset permanent hearing loss;8’ 2)
except for the neonatal period, our current health-care delivery
system provides us no other opportunity to pursue an organized,
direct screen of hearing until children enter the public school system-for most, at about 5 to 6 years of age; 3) although delayed
language and speech development are the primary indicators of a
hearing disability, most parents and primary care providers do
not suspect heating loss as a possible etiology. Most primary care
providers lack sufficient information regarding normal communi-cation development and early-onset hearing loss, neither do the
majority of providers systematically evaluate speech, language or
hearing during routine well-baby visits;’2’4 4) in the United States,
the average age when permanent hearing disabilities are
identi-fied remains at about 18 months for severe to profound bilateral
impairments and as late as 60 months for unilateral impairments
or bilateral losses of lesser degree.’ Although the prevalence of
bilateral profound hearing loss has been historically reported as
1:1000 live births,
the
target population also includes mild to moderate losses/unilateral losses. For these degrees of permanent impairment, prevalence estimates are higher (1.5 to 6 per 1000 births’5’6). Thus, our goal remains to identify these children beforeschool age and the onset of costly academic failure and/or the
need for special service provision; 5) instrumentation (eg, EOAE
and automated ABR) is available and currently undergoing rapid
technological improvement, thereby affording simple, relatively risk-free, and cost-effective screening device options for clinicians pursuing the initiation of neonatal hearing screening programs.
We agree
with Bess and Paradise that experience and researchwith well-devised and implemented mass neonatal hearing
screening programs is needed before many important questions
can be thoroughly addressed. Some of these authors’ concerns,
such as treatment efficacy, however, will likely not be amenable to
empirical study because of ethical and moral concerns. We cannot
imagine knowingly withholding intervention from a group of
young children with hearing loss as part of a planned, prospective, case-control investigation designed to determine the age and au-diometric variables that significantly influence communicative/
developmental outcome. It would seem unlikely that an
Institu-tional Review Board or more importantly, an informed parent,
would agree to deny intervention (amplification, aural
habilita-tion, medical monitoring and parent counseling) to a young child
identified as having a permanent hearing loss. The risk-benefit
ratio for such a protocol would preclude such a circumstance. The
exact “formula” for determining which children may suffer
signif-icant, long-standing consequences will likely be determined
by the
synergistic interaction of the specific degree, configuration, type and age of onset of the hearing loss, as well as the specific set of intrinsic and extrinsic factors each individual
child
brings to this unique developmental equation.Simply viewed, the NIH Consensus Panel had two choices:
maintain the status quo or set a vision for the future. The Panel, composed of both professionals and the lay public, opted for the latter. Further, the Consensus Panel determined from the data
presented
that
current technology would allow the direct andaccurate screening of hearing in very young children. One could
argue that the cause might have been better served if the Panel
had
been less specific about the test protocol and timelines,
forexample, while placing more emphasis on the development of
sito-specific innovative
alternative
approaches to the earlyidenti-fication of hearing loss. Hindsight not withstanding, the NIH
Consensus Panel took an important, decisive and knowingly con-troversial position on newborn hearing screening.
The importance of Bess and Paradise’s critical commentary on
this important topic is appreciated. Their article has challenged professionals to re-examine positions, long-held beliefs, and cur-rent screening initiatives. We also believe, however, that such commentary should not dissuade professionals from aggressively pursuing the vision of universal early identification and
amelio-ration of hearing loss. This goal must be maintained as one of our
most important clinical challenges. To suggest otherwise would be
to deny that optimal child development is facilitated by the rich
and abundant input provided through hearing.
JUDITH
S.
GRAVEL,PHD
Albert
Einstein
College
of Medicine
and
the
Montefiore
Medical
Center
Bronx,
NY
ALLAN
0.
DIEioi,PHD
Indiana
University
School
of Medicine
Indianapolis, IN
NOEL
D.
MATKIN,PHD
Children’s Hearing Clinic
University
of Arizona
Tucson,
AZ
REFERENCES
I. Early intervention programs for infants and toddlers with handicaps:
final regulations. Fed Reg. 198954:(#119)26306-26348
2. Healthy People 2000. United States Department of Health and Human Services, Public Health Service. Healthy People 2000: National Health
at Viet Nam:AAP Sponsored on September 1, 2020
www.aappublications.org/news
LETFERS
TO THE EDITOR
959
Promotion and Disease Prevention Objectives. Washington, DC: US Gov-ernment Printing Office; 1990
3. Infant Health and Development Program. Enhancing the outcomes of low-birth-weight, premature infants. A multisite, randomized trial.
JAMA. 1990;263:3035-3042
4. NIH Consensus Panel Report. Newsletter. Ambulatory Pediatr Assoc. 1993;29:18-28
5. Joint Committee on Infant Hearing. 1990 Position Statement. AAP News.
April 1991;7:6,14. Reprinted in Policy Reference Guide, 6th edition. Elk Grove Village, IL: American Academy of Pediatrics; 1993:343-346 6. Bess FH, Paradise J. Universal screening for infant heating impairment:
not simple, not risk-free, not necessarily beneficial, and not presently
justified. Pediatrics. 1994;93:330-334
7. American Speech-Language-Heating Association. Guidelines for
audio-logic screening of newborn infants who are at-risk for heating
impair-ment. ASHA. 1989;31:89-92
8. Mahoney TM, Eichwald JG. The ups and “Downs” of high-risk hearing screening: The Utah statewide program. Semin Hear. 1987;8:155-163
9. Epstein 5, Reilly JS. 5ensorineural hearing loss. Pediatr Clin North Am. 198936:1501-1520
10. Stein L, Clark 5, Kraus N. The hearing-impaired infant: patterns of identification and habilitation. Ear Hear. 1983;4:232-236
1 1. Elssmann S. Matkin N, Sabo M. Early identification of congenital
sen-sorineural hearing impairment. Hear
J.
19879:13-1712. Shah C, Chandler D, Dale R. Delay in referral of children with impaired
hearing. Volta Rev. 1978;80:207
13. Kenworthy 01, Triggs E, Perrin J, Bess F. Current-screening practices of primary care physicians. Paper presented at the Conference on Otitis Media and Development: Screening, Referral and Treatment. 1987;
Vanderbilt University, Nashville, TN
14. Diefendorf AO, Weber BA. Identification of hearing loss: programmatic and procedural considerations. In: Roush J, Matkin N, ads. Infants and Toddlers with Hearing Loss: Family-Centered Identification, Assessment and
Intervention. Baltimore, MD: York Press; 1994:43-64
15. White KR, Behrens TR, ads. The Rhode Island Hearing Assessment Project: implications for universal newborn hearing screening. Semin
Hear. 1993;14:l-119
16. Watkin P, Baldwin M, McEnery G. Neonatal at risk screening and the
identification of deafness. Arch Dis Child. 1991;66:1130-1135
In
Reply.-We are pleased that our challenge’ of the National Institutes of
Health Consensus Conference recommendation2 that all infants be
screened for hearing impairment within the first 3 months of life,
and preferably before discharge from the newborn nursery, has
generated so much response. We thank Dr Miller for his approv-ing comments and particularly for his suggestions concerning
screening-related research. We also welcome the provocative
ro-joinders from the proposed screening program’s advocates, first,
because in answering their various criticisms we hope to shed
further light on key component issues, and second, because
heightened attention in these pages to the problem of hearing loss
in young infants can only lead to heightened general vigilance and therefore earlier case detection.
OVERVIEW OF THE ISSUES
Unarguably, all infants with handicapping degrees of hearing
impairment should ideally be identified as early as possible.
How-ever, it is important to emphasize at the outset that universal
screening within the first 3 months of life will identify relatively
few infants with hearing impairment who would not have been
identified by screening all newborns who meet high-risk-register
criteria3 and/or are admitted to an intensive-care nursery (HRR/
ICN infants). On the other hand, the added cost of universal
screening-in monetary terms and, more importantly, in harm done-could be immense.
Collectively, the respondents advocating an early infant
screen-ing program have underrated the problems it would pose in
implementation and follow-through, overstated its potential
ben-efits, and virtually ignored its indirect costs and risks.
Regarding implementation andfollow-through, the advocates have
glossed over complex issues involving in-hospital personnel ro-quirements and logistics, particularly in the face of the rapidly
growing, if not virtually universal practice of discharging
new-borns within 24 to 48 hours of birth. They have also largely failed
to address procedures for fulfilling
standard
screening-program
requirements, particularly 1) assuring in advance the availability
of
adequate resources-facilities, personnel,and
financing-foraccomplishing recommended interventions; 2) establishing and
maintaining mechanisms for maximizing and monitoring
compli-ance; and 3) educating and counseling parents of infants with
false-positive test results and evaluating the near- and long-term impact of those results.
In projecting benefits, the advocates have, variously, 1)
broad-ened the definition of handicapping hearing loss to include
chil-dren with milder degrees of sensorineural loss (in whom
unto-ward developmental effects, and
the
effectiveness of earlyintervention, are uncertain) thereby arriving at higher estimates of
prevalence than the 0.1% (1:1000) rate we used in calculating
expected outcomes of the recommended screening protocol; 2)
neglected to distinguish between cases in which hearing loss is
present at birth and therefore would be potentially detectable by
the screening program proposed, and cases in which hearing loss
develops only later and therefore would not be detectable by
screening early in infancy; 3) failed to note that relatively early
identification is already being accomplished in many locales; 4)
projected more optimistic estimates of the validity of the proposed
screening protocol than are justified
by a comprehensive
overviewof experience
to date;
5) failed to make clear the differences to beexpected
in cost-yield relationships between screening HRR/ICN infantsand
screening
healthy,
non-HRR/ICN infants; and 6)as-sumed a greater degree of certainty about the efficacy of early
intervention for infants with hearing impairment than is justified by available evidence.
Regarding
costs andrisks, the respondents
advocating
universal
screening have failed to confront the fact that, of the hundreds of thousands of initial test failures by infants each year that wouldresult
from
the proposed
screening
program,
over
95% would be falso-positives by even the most optimistic estimates of testvalid-ity. Thus, few of the advocates mentioned, and none
acknowl-edged as substantial concerns, the quantum of needless parental
anxiety, potentially harmful labeling, and other psychological
dis-turbance
that the proposed
program might generate among fam-iies of normally-hearing infants. Finally, none of the advocates adverted to another, more consequential risk of universal infant screeningthat
we cited
in our commentary,’ namely, the risk of unnecessaryor
harmfuldiagnostic
procedures
or treatments
car-ried out on children.INDWIDUAL ISSUES
Here we address, summarizing for brevity, the various
argu-ments
advanced
by the respondents
advocating
universalscreen-ing. Parentheses indicate by whom the respective arguments were
advanced.
Prevalence
of Hearing
Loss
Respondent argument: Ifone includes mild to moderate unilateral and bilateral sensorineuraihearing loss, the overallprevalence may be as high
as 0.6%, rather than the 0.1% rate that we used (Gravel et al; Hall;
Northern; Robinette). Including mild to moderate loss is justified by
studies showing speech and language delays secondary to the mild conductive loss that accompanies otitis media (Vohr).
Reply:
Trueprevalence
is poorly
understood.
Prevalence
rates
willvary depending
on the population
tested,the
typeand degree
of hearing
loss, the tests used
to measure
hearing,
and the ages at
which
the hearing
tests
were
administered.
As noted
previously,
the higher
estimates
of prevalence
cited
include
cases
in which sensorineuralloss would
not yet have
developed
or beendetect-able during
early
infancy.
Regarding mild
to moderate
hearing
loss,
studies
attributingspeech
and language
delays to mild conductive loss secondary tootitis media
have
had
majorlimitations
and cannot
be
consideredconclusive.46
For
that reason, and becausethe
developmental
effects
of milder
degreesof sensorineural
loss also are
uncertain, it seems inappropriate at present to include infants with such loss inthe target
population
of any screening
program.
Regarding moderate to profound unilateral and bilateral
sen-sorineural
loss-for
which
we agree screening would bejustified
ifthe screening
protocol
gave
satisfactory
outcomes-prevalence
inthe ICN
is around 2 to 4%,7whereas
estimates
of prevalence
in
the well-baby
nursery
range
from 0.05 to
0.l%,”ie, about
#{188}othe
prevalence
in the ICN.
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1994;94;957
Pediatrics
Judith S. Gravel, Allan O. Diefendorf and Noel D. Matkin
Letter to the Editor
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Pediatrics
Judith S. Gravel, Allan O. Diefendorf and Noel D. Matkin
Letter to the Editor
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