PEDIATRICS Vol. 122 No. 4 October 2008, pp. e884-e890 (doi:10.1542/peds.2008-0606)
ARTICLE |
Preauricular Skin Tags and Ear Pits Are Associated With Permanent Hearing Impairment in Newborns
a Speech and Hearing Center, Chaim Sheba Medical Center, Tel Hashomer, Israel
b Department of Communication Disorders, Sackler Faculty of Medicine, Tel-Aviv University, Tel Hashomer, Israel
c Department of Neonatology, Edmond and Lily Safra Children's Hospital, Sackler Faculty of Medicine, Tel-Aviv University, Chaim Sheba Medical Center, Tel Hashomer, Israel
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ABSTRACT |
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 TOP ABSTRACT MATERIALS AND METHODSRESULTSDISCUSSIONCONCLUSIONSREFERENCES |
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OBJECTIVES. Our goals were to (1) study the prevalence of hearing impairment in a large cohort of infants with preauricular skin tags or ear pits and compare it with that among all other newborns participating in our universal newborn hearing screening program during the same period and (2) evaluate the effectiveness of transient evoked otoacoustic emissions as a hearing-screening tool in this population.
PATIENTS AND METHODS. During the study period of 7.5 years, 68484 infants were screened for hearing impairment, of whom 637 (0.93%) had preauricular skin tags and/or ear pits. The population was divided into 3 groups: (1) a low-risk group for hearing impairment; (2) a high-risk group for hearing impairment; and (3) a very high-risk group for hearing impairment. The screening results and audiological follow-up for these infants were examined retrospectively.
RESULTS. A significantly higher prevalence of permanent hearing impairment was found among infants with preauricular skin tags or ear pits (8 of 1000), compared with infants without tags or pits (1.5 of 1000). In the low-risk group, the prevalence was 3.4 of 1000, compared with 0.5 of 1000 in infants with and without preauricular tags or pits, respectively. In the high-risk group, the prevalence was 77 of 1000, compared with 20 of 1000 in infants with and without preauricular tags or pits, respectively. The odds ratio for hearing impairment associated with preauricular skin tags and/or ear pits after adjusting for level of risk group was 4.9. All infants diagnosed with permanent hearing impairment, with the exception of 1 with late-onset impairment, were detected by in-hospital transient-evoked otoacoustic emissions screening.
CONCLUSIONS. Infants with preauricular skin tags or ear pits are at increased risk for permanent hearing impairment. Transient-evoked otoacoustic emissions were found to be an effective hearing-screening tool in this population.
Key Words: preauricular skin tags • ear pit • isolated • hearing impairment • newborns
Abbreviations: CFA—craniofacial anomaly • PSEP—preauricular skin tags and/or ear pits • BOR—branchio-oto-renal • HI—hearing impairment • CHL—conductive hearing loss • UNHS—universal newborn hearing screening • TEOAE—transient evoked otoacoustic emission • JCIH—Joint Committee on Infant Hearing • SNHL—sensory neural hearing loss • ABR—auditory brainstem response • OR—odds ratio • ST—skin tag • EP—ear pit • CI—confidence interval
Minor craniofacial anomalies (CFAs) commonly found in newborn infants include preauricular skin tags and/or ear pits (PSEP) anterior to the external ear.1,2 The reported incidence of such preauricular anomalies ranges from 0.3% to 5% depending on the geographical location of the described population.3–11 These preauricular malformations usually appear in isolation and are considered by some authors to be of minor clinical importance.3,5 PSEP can, however, be associated with other major CFAs, including auricle and ear-canal malformations and/or genetic syndromes (eg, Treacher-Collins, Goldenhar, or branchio-oto-renal [BOR] syndromes) that themselves may be associated with hearing impairment (HI).3,7,12–14
Limited and conflicting data have previously been reported regarding the incidence of permanent HI associated with isolated PSEP.3,4,8,11,12 Some studies have reported a very high incidence of 13% to 17%,3,4 whereas others have reported no cases of permanent HI.11,12
PSEP associated with other CFAs and/or associated syndromes have been reported to be highly associated with conductive or mixed HI.3,15,16 For example, an incidence of 100% HI was reported in 10 children with PSEP and other CFAs and/or syndromes, with most exhibiting conductive hearing loss (CHL).3
Today it is widely accepted that universal newborn hearing screening (UNHS) is the first important step toward early identification and habilitation of infants with HI.17–22 Transient evoked otoacoustic emissions (TEOAEs) offer a reliable, fast, noninvasive, and easy-to-perform method for UNHS.23–26 Since 1997, a UNHS program has been in operation at the Chaim Sheba Medical Center. All infants are tested by using TEOAEs as a first-stage hearing screening before their discharge from the hospital.
The aims of this present cross-sectional study, therefore, were twofold: (1) to evaluate the prevalence of HI in a cohort of infants with PSEP with and without additional risk factors for HI and to compare it to that in all other newborn infants participating in our UNHS program during the same period; and (2) to evaluate the effectiveness of TEOAEs as a screening tool in this population.
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MATERIALS AND METHODS |
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TOPABSTRACTMATERIALS AND METHODSRESULTSDISCUSSIONCONCLUSIONSREFERENCES |
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Subjects
The cohort of this cross-sectional study was drawn from the population of all infants born at the Chaim Sheba Medical Center from January 1, 1997, through June 30, 2004. All infants were cared for in the well-infant nursery, in the intermediate care unit, or in the NICU, and all participated in the UNHS program.
The entire population was divided into 3 groups: (1) a group at low risk for HI, including infants with or without PSEP and no additional risk factors for HI according to the Joint Committee on Infant Hearing (JCIH) 1994/2000 position statements18,19; (2) a group at high risk for HI with or without PSEP, including infants with additional risk factors for HI on the basis of the JCIH 1994/2000 position statements18,19 (eg, family history of permanent childhood sensory neural hearing loss [SNHL]; in utero infection; a NICU stay of 
48 hours; hyperbilirubinemia at a serum level requiring exchange transfusion, etc) and excluding major auricular deformities and/or associated syndromes; and (3) a group at very high risk for HI with or without PSEP, including only those infants with major auricular deformities (eg, ear microtia, meatal atresia) and/or associated syndromes (eg, Goldenhar or BOR syndromes).
Hearing-Screening Protocol
All infants born at our medical center participate in our UNHS program, which uses TEOAEs as the first step in in-hospital screening. All infants at risk for HI, including infants with PSEP, take part in the special screening protocol for high-risk infants. The protocol includes an auditory brainstem response (ABR) test within 1 month after discharge and audiological follow-ups until the age of 3 years, in accordance with the 1994/2000 JCIH position statements.18,19 The protocol for our UNHS program, conducted in 3 stages, was described in detail in a previous report.27 It should be noted that infants at low risk for HI were screened before discharge by using the TEOAE screening test, followed by an ABR test at the outpatient clinic only if they failed in-hospital screening.
Hearing Impairment
HI was defined as unilateral or bilateral SNHL or conductive or mixed hearing loss of >25 dB HL in the 500- to 4000-Hz frequency region. Permanent HI was defined as sensory-neural, mixed, or structural conductive.28 The diagnosis was based on elevated ABR thresholds for air-conduction click and tonal stimuli with or without elevated ABR thresholds for bone-conduction click stimuli, as well as elevated behavioral thresholds in the absence of otitis media, ruled out on the basis of otoscopic examination by ear, nose, and throat specialists. Transient HI was defined on the basis of the above-mentioned elevated air-conduction ABR and behavioral thresholds, in the presence of ABR thresholds for bone-conduction click stimuli within the normal range (no more than 15 dB nHL), and in the presence of otitis media confirmed by otoscopic examination.29 HI severity was determined on the basis of work by Clark30: mild HI (26- to 40-dB HL), moderate HI (41- to 55-dB HL), moderately severe HI (56- to 70-dB HL), severe HI (71- to 90-dB HL), and profound HI (>90-dB HL).
Data Analysis
The odds ratio (OR) for HI associated with PSEP was calculated by using logistic regression adjusted for level of risk (by including this variable in the model as a covariant). A P value of <.05 was considered significant.
The research was approved by the ethics committee of the Chaim Sheba Medical Center.
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RESULTS |
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TOPABSTRACTMATERIALS AND METHODSRESULTSDISCUSSIONCONCLUSIONSREFERENCES |
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During a 7.5-year period, a total of 68484 infants were born at our medical center. Of these, 637 infants were born with either unilateral or bilateral PSEP (prevalence: 0.93%, as shown in Table 1). Specifically, 496 infants were born with preauricular skin tags (STs) (prevalence: 0.73%), of whom 454 had isolated STs and 42 had STs with additional risk factors for HI. One hundred thirty-two infants were born with ear pits (EPs) (prevalence: 0.19%), of whom 125 had isolated EPs and 7 had EPs with additional risk factors for HI. Finally, 9 infants were born with preauricular STs and EPs (prevalence: 0.01%), of whom 8 had isolated PSEP and 1 had PSEP with additional risk factors for HI.
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The group at low risk for HI included 65051 infants, of whom 587 infants had isolated PSEP and no other risk factors for HI. The high-risk group included 3406 infants, of whom 39 infants had PSEP and other risk factors for HI. The group at very high risk for HI with major auricular deformities and/or facial deformities included 27 infants, of whom 11 had PSEP and major auricular deformities (eg, ear microtia, meatal atresia) and/or associated syndromes (Goldenhar or BOR syndrome).
PSEP as Isolated Entities: Low-Risk Group
The screening results for the 587 infants with isolated PSEP and no other risk factors for HI (low-risk group) are presented in detail in Fig 1. Of the infants who failed the TEOAE screening tests, 2 infants were diagnosed with permanent HI. One child with ST in the right ear was identified with bilateral moderate SNHL (0.17%), and 1 child with STs in the right ear was identified with bilateral moderate permanent CHL (0.17%). Thus, the prevalence of permanent HI in infants with isolated PSEP and no other risk factors for HI (low-risk group) was found to be 0.34%. After the ABR test at the follow-up visit, 20 infants (3.5%) were diagnosed with transient CHL (10 infants with unilateral CHL and 10 with bilateral CHL). Six additional infants (1.0%) presented abnormal ABR results despite normal ABR thresholds and normal TEOAEs. Seventy-two infants were given further follow-up by using behavioral audiometry until the age of 7 to 36 months. Two-hundred forty-nine infants did not show up for behavioral audiometry follow-up.
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PSEP and Additional Risk Factors for HI: High-Risk Group
The characteristics of the 39 infants who had PSEP and additional risk factors for HI (high-risk group) are summarized in Table 2.
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All infants underwent an in-hospital TEOAE screening test and an ABR test at the outpatient clinic. Of these, 30 (77%) passed the screening successfully. Of the 9 infants who failed TEOAE testing, 7 infants returned for a third TEOAE and an ABR test. Two infants were lost to follow-up. At the end of this stage, 3 additional infants passed the tests, and 4 infants were referred for a full audiological evaluation.
After the diagnostic evaluation, 3 (7.7%) infants were found to have permanent HI. One premature infant with bilateral ST was diagnosed with severe SNHL in the right ear. Another extremely premature infant with a left EP had bilateral mild-to-moderate SNHL. The third, a term asphyxiated infant with a right ST, had bilateral moderate SNHL. Three (12.5%) additional infants with transient CHL were identified.
PSEP With Major Auricular Deformities and/or Associated Syndromes: Very High-Risk Group
Eleven infants were born with PSEP and major auricular deformities and/or associated syndromes. Specifically, 3 infants were born with isolated ear microtia (2 unilateral and 1 bilateral), 3 infants were born with isolated unilateral meatal atresia, 4 infants were born with ear microtia (2 unilateral and 2 bilateral) as part of Goldenhar syndrome, and 1 infant was born with BOR syndrome.
All infants underwent in-hospital TEOAE screening. Of these, only 2 (18%) infants passed the screening successfully. Nine (82%) infants failed the TEOAE test and were referred for a full audiological evaluation. At the end of this stage, all 9 (82%) infants were found to have permanent HI. The 2 infants who had passed the TEOAE screening successfully were further monitored. One child with BOR syndrome was diagnosed with late-onset bilateral SNHL at the age of 2 years. The other child was diagnosed with transient CHL. Thus, the prevalence of permanent HI in infants with PSEP and major auricular deformities and/or associated syndromes (very high-risk group) was found to be 91%.
The characteristics of infants from all risk groups with permanent HI are listed in Table 3.
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Prevalence of HI in Infants With and Without PSEP
The prevalence of HI in the different study groups of infants with PSEP (n = 637) was further compared with all other newborns without PSEP born at our center, during the same period, who took part in our UNHS program (n = 67847) (Table 4). The very high-risk group, in which HI is obvious, was excluded from additional analysis.
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The OR for HI associated with PSEP in all infants before adjusting for level of risk (low and high risk) was 5.4 (95% confidence interval [CI]: 2.21–13.44; P < .001). After adjusting for level of risk, the OR for HI associated with PSEP was 4.9 (95% CI: 1.94–12.4; P < .001).
The majority of hearing-impaired infants (12 of 15 infants) were found to have STs rather than EPs. Table 5 presents the laterality of HI in comparison with the side and type of PSEP.
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No cases of auditory neuropathy, defined as infants who successfully passed TEOAE screening but had severely abnormal or absent ABR, were identified in our PSEP cohort.
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DISCUSSION |
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TOPABSTRACTMATERIALS AND METHODSRESULTSDISCUSSIONCONCLUSIONSREFERENCES |
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Our study shows an elevated prevalence of HI in a large cohort of infants with isolated PSEP compared with a control group of all other newborns participating in the hospital's UNHS program during the same period. HI was found to be
7 times more common among low-risk infants with PSEP compared with infants without PSEP and 4 times more common among high-risk infants. TEOAE screening was found to be an effective hearing-screening tool in infants with PSEP; all infants but 1 (with late-onset HI) diagnosed with permanent HI were detected by in-hospital hearing screening. The prevalence of PSEP found in our population (0.93%) is in accordance with the generally reported incidence of 0.3% to 1.0%.3–6,9–11 The prevalence of preauricular STs in the present study (0.73%) corresponds to the previously published incidence of 0.4% to 0.6%.3–5,9,10,31 The relatively low prevalence of preauricular EPs found in our present study (0.19%) is lower than the 0.9% to 5% previously reported in other studies6–8 but is in accordance with a report on a trend toward decreased prevalence of preauricular EPs in Israel.31
As in other reports, bilateral preauricular STs occurred in 6% of the cases, and bilateral preauricular EPs occurred in 20% of the cases.1,6,7 In accordance with a previous report, preauricular EPs tended to occur more commonly on the right side,32 whereas a higher tendency for preauricular STs in the left ear was not observed in the present study. Thus, it seems that our cohort is representative of the population of PSEP.
In our study, the combined prevalence of HI in infants with PSEP in the low- and high-risk groups was found to be significantly higher than that of infants without PSEP (0.8% vs 0.15%) but not as high as previously reported (13%–17%).3,4,15 Our results contradict other studies that reported no cases of permanent HI in infants with isolated PSEP.11,12 The differences in the prevalence rates reported in the literature may be related to several factors. One important factor is the criteria selection for the different populations reported in the various studies. For example, a 13% incidence of SNHL was reported in a cohort of 178 infants with isolated PSEP.3 A large proportion of these hearing-impaired infants, however, had a familial history of HI, which increases the risk for HI. As previously suggested by François et al,12 there may be 2 different types of PSEP: isolated cases in infants without any other risk factors and those associated with a familial history of HI. It should be noted that in our present study, only 5 infants with PSEP had a known familial history of HI, and none of them were diagnosed with HI. Other factors that may be related to this inconsistency include different definitions of HI, different audiological evaluations to confirm the diagnosis of HI, and variations in cohort size and length of follow-up.
PSEP are formed during embryogenesis and are related to the development of the auricle during the sixth week of gestation.1,2 The auricle is formed from the first and second branchial arches, which develop into 6 hillocks.2,33,34 The most accepted theory attributes the development of EPs to an incomplete or defective fusion of the 6 hillocks.6,8,35 Thus, PSEP are expected to be related to CHL rather than to SNHL. In our study, 1 infant with isolated PSEP but no other risk factors for HI was diagnosed with a permanent bilateral moderate CHL. This finding is in accordance with that from a previous study in which 4 of 5 hearing-impaired infants with isolated PSEP were reported to have a mild-to-moderate CHL.15 However, the other infant with isolated PSEP diagnosed in our study had bilateral moderate SNHL. Cases of SNHL have been reported in infants with isolated PSEP and no other risk for HI.4,15 Although the inner ear does not originate from the same embryonal tissue as the external ear, multiple ear malformations may be possible because of the proximity of these tissues and because the inner ear and external ear develop at the same time of embryogenesis.36 Thus, isolated PSEP can possibly be a sign not only of abnormalities at the level of the external and/or middle ear but also of the inner ear.
As expected, a high prevalence of HI was found among infants with PSEP and additional risk factors for HI (high-risk group), as determined by the JCIH (1994/2000)18,19 and, moreover, in infants with PSEP and major auricular deformities and/or associated syndromes (very high-risk group). These results are in accordance with those of Kankkunen and Thiringer,3 who reported an incidence of 100% HI in 10 children with PSEP and other CFAs and/or syndromes.
In our study, a prevalence of 3.5% of transient CHL was diagnosed in the cohort. An earlier study on the audiological assessment of infants with PSEP reported a higher prevalence of 9.5% of CHL caused by secretory otitis media.12 The differences between the 2 studies may be related to the fact that in the earlier study, hearing status was assessed at a later stage (8–18 months), whereas in our study the prevalence refers only to the outcome of the in-hospital hearing screening and not to other transient CHL cases that may have developed later in childhood. Our results are in accordance with a previous report of a prevalence of 2% of transient CHL cases detected by TEOAEs in newborns participating in a UNHS program.37
TEOAE screening was found to be a reliable screening method for early in-hospital detection of HI in newborn infants with PSEP. All infants but 1 who were diagnosed with HI failed TEOAE screening tests and were diagnosed at a very early stage. The only child who passed the TEOAE screening test developed late-onset HI at the age of 2 years and was later diagnosed with BOR syndrome.
This retrospective study has several limitations. The hearing of infants with PSEP was assessed by using TEOAEs as well as ABR and additional behavioral follow-up, as recommended by the JCIH high-risk registry (1994/2000).18,19 All infants in the control group were screened by using TEOAEs before discharge as part of our UNHS program and monitored further only if they failed the in-hospital screening or had any risk factors for HI. Thus, most of the infants in the control group had only TEOAE screening and were not monitored further. This limitation could have missed infants in the control group with an abnormal ABR despite normal TEOAEs. Another limitation is the length of follow-up of the study group in comparison to the control group. As a result, infants with late-onset permanent HI as well as transient CHL caused by otitis media may have been missed. It should be noted, however, that in Israel all infants are routinely screened at the age of 7 months in well-infant clinics, and those infants identified with significant HI that requires amplification and habilitation are generally referred to 1 of the national rehabilitation centers for the hard of hearing. To the best of our knowledge, none of the infants who were lost to follow-up were referred to 1 of these rehabilitation centers.
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CONCLUSIONS |
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TOPABSTRACTMATERIALS AND METHODSRESULTSDISCUSSIONCONCLUSIONSREFERENCES |
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It is well established that major CFAs are an important risk factor for HI. Nevertheless, minor anomalies such as PSEP, although detected at birth, are often disregarded. The findings of our study provide additional evidence that isolated PSEP constitute a risk factor for HI. Therefore, infants who exhibit isolated PSEP should be routinely screened for HI immediately after birth. TEOAE screening was found to be an effective screening tool in these infants.
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ACKNOWLEDGMENTS |
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We are grateful to Ester Shabtai and Valentina Boyko for statistical analysis and the National Rehabilitation Centers for the Hard-of-Hearing for help in follow-up.
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FOOTNOTES |
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Accepted Jun 10, 2008.
Address correspondence to Daphne Ari-Even Roth, PhD, Chaim Sheba Medical Center, Speech and Hearing Center, Tel Hashomer 52621, Israel. E-mail: rothd{at}post.tau.ac.il
The authors have indicated they have no financial relationships relevant to this article to disclose.
| What's Known on This Subject Limited and conflicting data have previously been reported regarding the incidence of permanent HI associated with isolated PSEP.
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| What This Study Adds Infants with PSEP are at excess risk for permanent HI (up to 6-fold). TEOAEs were found to be an effective hearing-screening tool in this population.
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