International Journal of Pediatric Otorhinolaryngology 73 (2009) 16911695

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International Journal of Pediatric Otorhinolaryngology

journal homepage: www.elsevier.com/locate/ijporl

Newborn hearing screening on infants at risk

Christine Ohl, Liliane Dornier, Cecile Czajka, Jean-Claude Chobaut, Laurent Tavernier *
ENT Department, University Hospital of Besancon, France

A R T I C L E I N F O

A B S T R A C T

Article history:
Received 8 June 2009
Received in revised form 24 August 2009
Accepted 26 August 2009
Available online 30 September 2009

Objectives: This article presents the results of newborn hearing screenings on infants at risk of hearing
impairment at the French University Hospital of Besancon from 2001 to 2007.
Materials and methods: All newborns at risk of hearing impairment were tested according to the method
recommended by the Joint Committee on Infant Hearing (JCIH): a two-step automated oto-acoustic
emissions (AOAE) program, completed by an auditory brainstem response (ABR) for the positive
diagnosis of hearing impairment. The screening started with AOAE on the third day of life, at the earliest.
If one or both ears did not have AOAE, the infant was re-tested at which time, should the AOAE again be
positive, ABR was performed. When the ABR threshold was 40 dB or more, the infant was referred to an
audiologist specialized in infant deafness for diagnosis conrmation and management.
Results: Over the period, 1461 infants were screened, among whom 4.55% were diagnosed as deaf or
hard of hearing. Nearly 10% of the infants were lost to follow up. Forty-six children had a sensorineural
hearing impairment, of which 34 were bilateral and were managed before the age of 6 months. The risk
factors for sensorineural hearing loss were (in order of statistical signicance): severe birth asphyxia;
neurological disorder; syndromes known to be associated with hearing loss; TORCH (toxoplasmosis,
rubella, cytomegalovirus, herpes) infections; family history of deafness; age at the time of screening; and
the association of 2 or more risk factors. However, birth weight inferior to 1500 g and premature birth
before the 34th week of pregnancy did not show a statistically signicant inuence on sensorineural
hearing loss. Craniofacial anomalies (mostly cleft palate and ear aplasia) were a signicant factor for
conductive hearing loss.
Conclusion: Our selected hearing screening on infants at risk allowed 60 deaf children access to early
management. However, too many children were lost to follow up; which revealed that better
information regarding risk of hearing loss must be provided to parents and paramedics and universal
newborn screening needs to be performed. The most important result of this study is that in a population
of hearing impaired children, with an impairment incidence close to what is commonly reported, the
association of several risk factors proves to be a signicant additional risk factor for hearing impairment.
2009 Elsevier Ireland Ltd. All rights reserved.

Keywords:
Sensorineural hearing loss
Infant
Screening
Risk factors

1. Introduction
In the general population, 1 newborn every 5001000 births
presents permanent hearing impairment [1], a greater incidence
than the incidence of diseases routinely screened at birth [2]. In
certain higher risk populations, this incidence could increase
10- to 50-fold [1].
A disease must fulll certain requirements before it is
systematically screened at birth: high incidence; serious disease
(life or function threatening); and existence of an accepted
treatment which improves the prognosis [3]. Thus, in France,

* Corresponding author at: Service dORL, Centre Hospitalier Universitaire, 3,

Boulevard Fleming, 25030 Besancon Cedex, France. Tel.: +33 3 81 66 82 38;
fax: +33 3 81 66 84 79.
E-mail address: ltavernier@chu-besancon.fr (L. Tavernier).
0165-5876/$ see front matter 2009 Elsevier Ireland Ltd. All rights reserved.
doi:10.1016/j.ijporl.2009.08.027

congenital hypothyroidism (1/4000 births), phenylketonuria (1/

15,000), congenital adrenal hyperplasia (1/12,000), drepanocytosis
(1/4000), and cystic brosis (1/4000) have been routinely screened
at birth for many years, despite lesser frequency than permanent
hearing loss [1].
The risk factors of hearing loss in neonates, which were rst
documented in 1994 and then revised in 2000 by the Joint
Committee on Infant Hearing (JCIH) [4], are: premature birth
(gestational age 34 weeks); low birth weight (<1500 g); children
from hearing impaired families; TORCH infections; neurological
disorder; hyperbilirubinemia; craniofacial anomalies; syndromes
known to be associated with hearing loss; and severe birth
asphyxia (APGAR < 7 at 5 min). Other risk factors have been tested,
such as maternal drug abuse, persistent high pulmonary pressure
[1], intra-ventricular haemorrhage [5], high C reactive protein
levels [6] but were not proven to be signicant. Between 3 and 5%
of the at-risk newborns suffer a permanent hearing loss.

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C. Ohl et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 16911695

Hearing impaired children present delays in language learning

and general development [7], which could only have been
prevented by early diagnosis and management; while ears are
mature at birth, auditory cortex and neural connections only
develop with acoustic stimuli [8].
A number of Universal Newborn Hearing Screenings (UNHS)
have recently been put in place in France and abroad [9] with good
results: increased verbal reasoning skills and language were
noticed when children were diagnosed and rehabilitated before
the age of 12 months [7,8,10].
Hearing screening (based on auditory brainstem response
(ABR) and behavioral audiometry) on infants at risk has been
performed for some time at the University Hospital of Besancon. In
April 2001, we began a new method of screening based on the
association of automated oto-acoustic emissions (AOAE) and ABR,
which optimize the management of hearing impaired children.
While we were reviewing the outcomes of this type of
screening, the French National Consultative Committee for life
and health sciences (Comite consultatif national dethique pour les
sciences de la vie et de la sante) issued a statement, which,
although skeptical on UNSH, is, however, supportive of screening
infants at risk [11].
2. Materials and methods
The selected population included every newborn presenting
one or more of the risk factors dened by the JCIH [4]: premature
birth (gestational age 34 weeks); low birth weight (<1500 g);
hyperbilirubinemia; children from hearing impaired families;
craniofacial anomalies; syndromes known to be associated with
hearing loss; and those hospitalised in the neonates unit of the
University Hospital of Besancon (level III maternity hospital with
an associated neonatal intensive care unitfor births at risk). The
causes for hospitalisation were in utero infection and severe birth
asphyxia and neurological troubles (hypotonia, meningitis, etc.).
The screening consisted of three steps (Fig. 1):
 Detection of bilateral AOAE as early as the third day of life, up to
several weeks, depending on the general status of the child (out
of incubator, extubation, etc.). Each ear was tested twice for
increased reliability.
 In cases where the test was positive (i.e. AOAE not found) in one
or both ears, a second AOAE test was performed, at the earliest 2
days after the rst test, depending on the tests circumstances
(agitated child and loud breathing) and the time before
discharge.

Fig. 1. Protocol of screening. AOAE, automated oto-acoustic emissions; ABR,

auditory brainstem response; +, positive screening (i.e. absence of AOAE); ,
negative screening (i.e. presence of AOAE); N: normal ABR; A: abnormal ABR.

 If the second AOAE test was positive for one or both ears, ABR was
performed within 4 weeks (during hospitalisation or after
discharge in an outpatient clinic).
If thresholds were abnormal on ABR (i.e.: superior to 40 dBHL),
the child and his parents were sent to an ENT doctor specialized in
child hearing loss for diagnosis conrmation and management.
AOAE were tested during infants natural sleep (still in their
bed) by qualied bio-medical staff using an Echoscreen1 with a
20 s time response. For presence of AOAE the response was PASS,
and FLACK when absent. If ABR was performed while infants were
still hospitalised, the same realisation conditions applied. If an
infant had previously been discharged, ABR took place within the
audiology laboratory of the ENT ward. All tests were performed by
the same staff.
During a childs hospitalisation the parents were approached by
pediatricians and the bio-medical staff who offered information on
the risks of newborn permanent hearing loss and discussed
screening tests which were then performed, unless parents
refused.
If the child was discharged before the second AOAE had been
performed, a note was written on the childs health record,
recommending parents consult with an ENT unit and complete the
screening test series. In the event the child was transferred to
another hospital, the transfer note included the same recommendation.
Statistical analysis was conducted with Epi InfoTM 2000
software (version 3.4.1, CDC Atlanta, GA). Khi square test, exact
test of Fisher or MannWhitney test were used, following usual
conditions of application. Signicance was set at 0.05.
3. Results
Between April 2001 and March 2007, 1464 infants (687 females
and 777 males, from approximately 28,000 births) were screened
at the University Hospital of Besancon. Three children died during
the screening period and were thus excluded. After the rst test,
538 children (37%) tested positive (i.e. no AOAE) on at least one ear
(Fig. 2): 261 were bilateral (18%) and 277 were unilateral (19%).
Four hundred and thirty-two were re-tested for AOAE, of which:
156 children (10.6% of the screened population) again tested
positive (90 bilateral, 66 unilateral), and among whom 120 were
tested for ABR: whereby 60 were subnormal or normal
(<40 dBHL). Thirty-six children were lost to follow up, i.e. 23%
of the children for whom AOAE was not found in either test).

Fig. 2. Results of the hearing screening on infants at risk. LTF: lost to follow up.

C. Ohl et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 16911695

Table 1
Characteristics of the hearing impairment screened.
Hearing loss

CHL

Table 3
Risk factors comparison between sensorineural hearing loss and normal hearing
infants.

SNHL

Unilateral

Bilateral

Total

Medium
Severe
Profound

6
2
0

6
0
0

12
2
0

Total

14

Unilateral

1693

Bilateral

Total

9
2
1

23
2
9

32
4
10

12

34

46

NH and CHL
(n = 1273)

SNHL: sensorineural hearing loss; CHL: conductive hearing loss.

Sixty children (i.e. 4.55% of the children who went through the
entire three-step screening process) were diagnosed with hearing
loss, 46 of whom were diagnosed with sensorineural hearing loss
(Table 1). If we extrapolate these results to the children lost to
follow up, an extra 37 hearing losses (2.05%), including 28
sensorineural hearing losses (21 bilateral) would likely have been
diagnosed.
There is no statistically signicant difference in sex ratios
between hearing impaired (deaf or hard of hearing) (603 (47.9%)
females658 males) and good hearing children (27 (45%) females
33 males). A comparable sex ratio is found in sensorineural hearing
impaired children: 20 (43.5%) females26 males.
There was a statistically signicant difference between the
average weight at birth of children with sensorineural hearing loss
(SNHL) (1655 g) and those without (1980 g) (p = 0.01). Gestational
age at birth was statistically correlated to birth weight with a 0.70
correlation coefcient and a [0.670.73] 95% condence interval.
The difference in average weight between premature infants
(1628 g) and non-premature infants (2749 g) was statistically
signicant (p < 10 3). Thus, gestational age at birth (premature
newborns) and birth weight are conrmed not to be independent
variables.
The age difference at the time of screening between sensorineural hearing impaired children (35.6 days) and the other
children (19.3) was statistically signicant (p = 6  10 4).
The deafness and hearing impairment risk factors of the
children within this study are presented in Table 2: 15 neurological
disorders (4 meningitis, 1 encephalitis, 1 lack of corpus callosum
with intra-ventricular haemorrhage, 1 white substance damage, 5
neonatal seizures, 2 neurological neonatal suffering and 1
hydrocephaly); 20 craniofacial anomalies (8 cleft palates, 1
choanal atresia, 3 major ear aplasias, 6 minor ear aplasias and 2
non-specied facial malformations); 13 syndromes known to be
associated with hearing loss (1 Townes-Brocks syndrome, 1
Klinefelter syndrome, 1 trisomy 21, 1 cri du chat syndrome, 1
CHARGE syndrome, 6 Pierre Robin syndromes and 2 non-specied
syndromes); and 10 TORCH infections (9 CMV and 1 herpes).

SNHL
(n = 46)

Severe birth asphyxia

Neurological disorders
Syndromes associated
with hearing loss
Craniofacial anomalies
Family deafness
TORCH

12 (0.9%)
9 (0.7%)
9 (0.7%)

4 (8.7%)
6 (13.0%)
4 (8.7%)

2  10
5  10
7  10

18 (1.4%)
14 (1.1%)
6 (0.5%)

2 (4.3%)
3 (6.5%)
4 (8.7%)

0.15
0.02
2  10

Number of risk factors

0
1
2

343 (26.9%)
643 (50.5%)
287 (22.5%)

9 (19.6%)
12 (26.1%)
25 (54.3%)

<10

3
6
4

NH: normal hearing; CHL: conductive hearing loss; SNHL: sensorineural hearing
loss.

A last risk factor, which does not appear in Table 2 was

hyperbilirubinemia, which was found on only two occassions and
was not taken into account for statistical analyses.
Statistical analysis revealed correlations between the four
groups of babies (normal hearing (NH), conductive hearing loss
(CHL), unilateral and bilateral SNHL) for each risk factor, except in
premature children and children with a birth weight under 1500 g
(Table 2).
Risk factor presence within a given group of children (NH, CHL,
unilateral or bilateral SNHL) was tested against the prevalence of
that risk factor in the each of the remaining groups. No statistical
difference was found in risk factor prevalence between unilateral
and bilateral SNHL. Presence of only one risk factor craniofacial
anomalies was statistically different between NH (1.0%) and CHL
(37.5%) children (p = 6  10 7).
We also conducted a set of tests between unilateral and
bilateral SNHL children against NH and CHL children wherein the
presence of each risk factor was statistically signicantly different,
except for craniofacial anomalies, as shown in Table 3.
Finally, associations of two or more risk factors signicantly
increase the risk of bilateral hearing impairment; infants with two
(or more) risk factors were more often bilaterally hearing impaired
than infants with a single risk factor, respectively 6.1 and 1.6%
(p < 10 3).

4. Discussion
Newborn hearing screening has already demonstrated great
benets in children who are hard of hearing. Indeed, children who
are diagnosed and rehabilitated earlier, show better language and

Table 2
Risk factors prevalence in the studied population.
NH
(n = 1259)

CHL
(n = 14)

Unilateral SNHL
(n = 12)

Bilateral SNHL
(n = 34)

Weight at birth <1500 g

Premature birth
Severe birth asphyxia
Neurological disorders
Syndromes associated with hearing loss
Craniofacial anomalies
Family deafness
TORCH

283
862
12
8
8
13
13
5

3
5
0
1
1
5
1
1

5
7
0
0
2
0
1
2

12
21
4
6
2
2
2
2

(35.3%)
(61.8%)
(11.8%)
(17.6%)
(5.9%)
(5.9%)
(5.9%)
(5.9%)

0.14
0.06
<10
<10
<10
<10
0.03
<10

Number of risk factors

0
1
2

341 (27.1%)
635 (50.4%)
283 (22.5%)

5 (14.5%)
16 (32.6%)
18 (52.9%)

<10

(22.5%)
(68.5%)
(1.0%)
(0.6%)
(0.6%)
(1.0%)
(1.0%)
(0.4%)

(21.4%)
(35.7%)
(7.1%)
(7.1%)
(35.7%)
(7.1%)
(7.1%)

2 (14.3%)
8 (57.1%)
4 (28.6%)

NH: normal hearing; CHL: conductive hearing loss; SNHL: sensorineural hearing loss.

(41.7%)
(58.3%)

(16.7%)
(8.3%)
(16.7%)

4 (33.3%)
1 (8.3%)
7 (58.3%)

5
5
5
5

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C. Ohl et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 16911695

better behavioral skills at the age of 5 than children diagnosed later

[7].
In our department, we have performed NHS on infants at risk
(as dened by JCIH in 2000) since 2001. Over that time, 4.55% of
screened children were diagnosed with hearing impairment;
similar to the ndings of several other studies on infants at risk
[1,5,6,12,13], regardless of the protocol used.
AOAE is the screening test of choice because it is: fast and easy
to use at a neonate bed; non-invasive; and highly sensitive and
reproducible [14]. The main disadvantage of AOAE, however, is
that it is not capable of detecting retro cochlear deafness to which
newborns at risk are susceptible (even though its prevalence is
relatively low, at 2.1% of permanent hearing loss in children) [15].
As a result, many screenings now include an automated ABR test,
from which the referral rate is 3.2% [16]. Such tests are only
marginally longer and slightly more expensive than AOAE.
However, at the time we dened our screening test, automated
ABR was not yet available nor had proven its superiority to AOAE.
In this study, the referral rate after the two-step protocol was
10%, higher than many studies on universal screenings with AOAE
protocols [12,15,17,18]; however, this type of screening is usually
meant for bilaterally absent OAE, therefore contributing to lower
referral rates [12]. However, in studies on infants at risk, the
referral rate with AOAE can increase up to 12% [19] when ABR is
performed as a third step, allowing the identication of false
positive results in the test (indeed 50% of ABRs were normal).
Among all tested risk factors, severe birth asphyxia, neurological troubles, syndromes known to associate hearing loss and in
utero infection by TORCH, were present (statistically signicant) in
all four groups of infants (Table 2) and are a reection of global
health. Age at the time of the rst AOAE, also a risk factor for
hearing impairment (p < 103), is a further reection of infants
global health status, given we had to wait until infants were in a
stable condition before we could test them.
For children with a family history of permanent childhood
sensorineural hearing loss, the difference between all groups was
signicant, although it was the least signicant risk factor;
probably due to its low prevalence in our population (Table 2).
Hyperbilirubinemia, however, had too low a prevalence for
statistical analysis.
Concerning craniofacial anomalies, no difference was shown
between SNHL and the normal hearing group (Table 3), whereas
there was a signicant difference between CHL and the normal
hearing group, which is expected as most of the 18 children with
craniofacial anomalies had cleft palate (8) or ear aplasia (3 major, 6
minor) which imply conductive hearing loss.
Prevalence of birth weight inferior to 1500 g and premature
birth (34 weeks), which were correlated, was not signicantly
different in all four groups of infants, probably due to the high
proportion of premature births in the global population (68%). This
was also found in other studies [6] and explains why, in those
studies, the population was recruited only among premature
births: to eliminate bias and solely describe the other risk factors
[12].
Nevertheless, our results are broadly comparable to results
found in the literature [1,46,12,13]; except for the nonsignicance of craniofacial anomalies as already mentioned.
Prevalence of the different etiologies observed in our study, also
corroborate what is generally described in older hearing impaired
children [2022] with the exception of a higher rate of premature
and low birth weight infants in our population (most likely a bias of
our neonatal screening, which included all premature newborns)
and the high literature rate of congenital rubella (which is not
present at all in our population). The later is explained by
successful rubella vaccination campaigns over the years. As
etiologies remain the same over the years, newborn screenings

seem even more pertinent. Indeed, all causes diagnosed early lead
to a better management of impaired hearing children.
The association of more than one risk factor, which is extremely
frequent (27% of the studied population), also appeared to be an
additional risk factor for hearing loss (8% of hearing impairment
among children with more than one risk factor). This nding rarely
appeared in the literature to date [23,24], and if hypothesized was
never statistically proven, despite appearing here to be a relevant
risk factor (p < 10 3).
Our study also had a high proportion of children lost to follow
up (almost 10% of the children tested at least once), which may be
explained by different reasons:
 A number of children were transferred to regional hospitals as
soon as they were in a stable condition, despite the screening
protocol being incomplete. It is probable though, that some of
these children completed the remaining screening protocol, as
suggested in the transfer note, however we were not informed of
the results.
 Lack of information with the parents, who, therefore, were not
aware enough of the consequences of SNHL and the benets of
early management; a contributing factor would be that pediatric
and ENT units are not in the same wing of our hospital.
 Children discharge earlier than expected, which prevented us
from completing our screening protocol.
The hearing impaired children who slipped through our screening (potentially 37 children, 2.5%) could have had delayed diagnosis
and management as a result. This extrapolation is within the range of
that noted by other studies [16,18,2527]. However, this rather large
extrapolation, does not take into account further hearing evaluation
which may have been performed in the discharge hospitals where
additional care could have been provided.
Newborn hearing screening of infants at risk must however be
improved. This may be achieved by giving better information to
staff in charge of newborns (for example pediatricians and
paramedics), who must know the risk factors and the benet
children can get from early diagnosis and management. Parents
also need to be better informed, and not only at the time of the
screening, for universal newborn screenings, information should
ideally be given before birth. Finally, babys health records should
be more consistently used and appropriately lled in and red by
parents and medical staff.
Organization could also be improved by, for example,
integrating other screening tests such as Guthrie Test, to locate
lost to follow up children.
Several studies also demonstrate that newborn hearing screening does not lead to higher maternal anxiety nor negatively
impacts motherinfant relationships [27]. Because worse hearing
outcomes are associated with a 2 months after birth delayed
screening, UNHS should be performed on every newborn as early
as possible, i.e. in maternity hospitals before discharge.
Early hearing screening has already proven benets in verbal or
behavioral skills management of hearing impaired children, but
also has economic benets; a recent study showed that, after 10
years, the cost of a UNHS would be lower than the overall
management cost of hearing impaired children later diagnosed
[16,26].
In 2007, the French health high authority (Haute Autorite de
Sante) had released a position statement recommending initiation
of UNHS programs in hospitals and evaluation of its benets and
costs, acknowledging that hearing impairment is a public health
concern [8]. In 2008, the French National Consultative Committee
for life and health sciences gave a less positive opinion in
consideration of an alternative ethical approach [11] following
requests by deaf or hard of hearing adults and psychologists. The

C. Ohl et al. / International Journal of Pediatric Otorhinolaryngology 73 (2009) 16911695

committee emphasized the difference between two categories of

children screened: children from normal hearing families, whose
parents are anxious to limit and even restore their childs hearing;
and children from hearing impaired families, who do not
approach hearing impairment as a handicap and fear unnecessary
overmedication. The committee also insisted on increasing
parents information, which may not have been properly delivered
during short stays in maternity hospitals. We therefore believe
parents information should be provided: before birth; as a nationwide campaign; and probably during birth preparation.
While the committee does not provide supporting evidence, it is
concerned that early screening could have a negative impact on
parentinfant relationships, despite scientic studies suggesting the
contrary (i.e. early screening does not alter parentinfant relationships) as mentioned earlier. The committee is, however, in
agreement with screening infants at risk of hearing impairment.
It is obvious to screen and then follow high risk babies until normal
hearing is restored or surgery is performed (if required) however,
our primary goal must be to expand screening to the general
population who would not otherwise have been tested. Additionally,
a UNHS program would enable us to reach those cases where
medical staff are not or incompletely informed of the risk factors.
5. Conclusion
The incidence of hearing impairment diagnosed following the
newborn screening test, as specied above, is comparable to the
incidence rate commonly reported in the literature.
Although the use of AOAE gives satisfying results, since it is
reliable and easy to use, we must remain attentive that it does not
identify retro cochlear hearing losses in this at risk population.
The percentage of children lost to follow up is not a result of our
protocol but, rather, due to a lack of parents and pediatric teams
knowledge of the risk factors leading to hearing loss or impairment
and the benets of early diagnosis and management.
The association of several risk factors, rarely mentioned and not
statistically evidenced in the literature to date, proves here to be a
signicant additional risk factor for hearing impairment.
The importance of selective screening for infants at risk is
recognized and supported world-wide. However, we believe such
acceptance should only be taken as a precursor to Universal
Newborn Hearing Screening; this will be achieved by spreading
knowledge of the benets of early intervention and encouraging
the development of structures required to implement such a
protocol.
Acknowledgment
The authors want to express thanks to Anne Chaurand, the biomedical agent who performed all automated OAE and ABR of this
study and lled in meticulously the screening records.
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