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and coagulation factors in children with cerebral palsy. Ann Neurol. 1998;44:665– 675

5. Debus O, Koch HG, Kurlemann G, et al. Factor V Leiden and genetic defects of trombophilia in childhood porencephaly. Arch Dis Child. 1998;78:F121–F124

6. Mercuri E, Cowan F, Rutherford M, Acolet D, Pennock J, Dubowitz L. Ischaemic and haemorrhagic brain lesions in newborns with seizures and normal Apgar scores.Arch Dis Child.1995;73:F67–F74

7. Cowan FM, Pennock JM, Hanrahan JD, Manji KP, Edwards AD. Early detection of cerebral infarction and hypoxic ischemic encephalopathy in neonates using diffusion-weighted magnetic resonance imaging. Neu-ropediatr.1994;25:172–175

8. Barmada MA, Moossy AJ, Shuman RM. Cerebral infarcts with arterial occlusion in neonates.Ann Neurol.1979;6:495–502

9. Koelfen W, Freund M, Konig S, Varnholt V, Rohr H, Schultze CH. Results of parenchymal and angiographic magnetic resonance imaging and neuropsychological testing of children after stroke as neonates.Eur J Pediatr.1993;152:1030 –1035

Universal Neonatal Hearing Screening

To the Editor.—

Dr Paradise’s commentary appearing in the March 1999 issue of Pediatricsraises to my mind 2 questions regarding universal neo-natal hearing screening: Is this an appropriate time to institute universal neonatal screening? Would it be more appropriate in-stead to institute a 2-step screening process consisting of a high-risk questionnaire followed up with neonatal screening when indicated?1

In general, decisions regarding the selection of screening tech-niques have largely been made by the test developers or specialty groups, such as the American Speech-Language-Hearing Associ-ation, and the rationale for their decisions is often unknown. However, since society is either benefiting from the test or paying

the costs of errors, decisions regarding the use of the screening tests should reflect the values of society. This point is of particular significance, as some states have already mandated universal neo-natal hearing screening. Therefore, it would behoove health pro-fessionals making such decisions to incorporate societal values in the decision-making process. One approach to achieve this is through an approach called decision analysis.2,3

Screening tests generally produce 4 outcomes: A, correct iden-tification of individuals with the problem; B, overreferrals; C, underreferrals, and D, correct identification of those individuals who are free of the problem. Figure 1 illustrates these outcomes and their relationship to each other.

Decision analysis enables one to determine for any 1 or more screening procedures the costs and benefits of the 4 outcomes and to sum them to obtain an overall picture useful in making com-parisons with alternative screening procedures. The analysis en-tails first the determination of the costs and benefits for individ-uals falling in each of the 4 cells. For instance, the costs and benefits (factors) associated with each of the cells is illustrated in Fig 2.

Although the costs of screening, rescreening, diagnosis, and treatment may not be so difficult to obtain from the literature, the economic gain is more difficult to assess in view of the fact that about 30% of those with sensory neural hearing loss may have concomitant developmental disabilities such as mental retardation and cerebral palsy.4Factors such as intrinsic gain, parental anxi-ety, and the value of identifying normal individuals can be ob-tained by sampling a cross-section of society with questionnaires. The second step is to sum the positive and negative factors in each of the 4 cells. The third step is review the literature for large-scale clinical trials of a test such as one for universal neonatal hearing screening to determine the relative frequency with which subjects fall into each of the 4 cells. The fourth and final step, giving a subtotal for each cell, is to multiply the probability obtained in step 3 times the sum of factors obtained in step 2. In the case of

Fig 2. Costs and benefits.

Fig 1. Screening outcomes.

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universal neonatal hearing screening, the results of this step may be surprising because the relative low cost of the procedure may be offset by the 90⫹% of overreferrals. The subtotals for the 4 cells are then summed to obtain the overall benefit of that particular screening procedure. Having completed such an analysis for uni-versal neonatal hearing screening, one can conduct similar analy-ses for alternative procedures such as that proposed by Paradise and others.

Although the use of decision analysis provides an excellent approach to the selection of screening tests, there are certain limiting factors that may make the selection of certain procedures unfeasible, even though results indicate that the greatest gain can be obtained by using one particular procedure. Examples of such limiting factors are: 1) availability of diagnostic and treatment services (alluded to by Paradise); 2) affordability of services; 3) probability that follow-up diagnosis and treatment will occur; and 4) an overreferral rate that is unacceptable to diagnosticians. One might also question the use of economic criteria in making screen-ing decisions. When financial resources are finite, there are few alternatives in deciding how to allocate health dollars.

In summary, a large clinical trial such as proposed by Paradise should be undertaken at this time. If so, decision analysis could be used evaluating the results of various screening options. In the meantime, it would be wise to curtail any further legislation to make universal neonatal hearing screening mandatory.

William K. Frankenburg, MD, MSPH

Departments of Pediatrics and Preventive Medicine University of Colorado School of Medicine

Denver, CO 80237-5075

REFERENCES

1. Paradise JL. Universal newborn hearing screening: should we leap before we look?Pediatrics.1999;103:670 – 672

2. Hammond KR, Aldeman L. Science and human judgment. Science. 1976;194:389 –396

3. Hershey JC. Consequence evaluation in decision analytic models of medical screening, diagnosis and treatment.Methods of Information in Medicine.1974;13:197–203

4. Van Naarden K, Decoufle´ P, Caldwell K. Prevalence and characteristics of children with serious hearing impairment in metropolitan Atlanta, 1991–1993.Pediatrics.1999;103:570 –575

To the Editor.—

Dr Paradise raises important issues with respect to the identi-fication of infants with congenital hearing impairment that pedi-atricians should carefully consider. Dr Paradise suggests that most affected infants can be detected through screening of high-risk newborns. The evidence from a number of studies suggests oth-erwise. Only about half of all affected infants have 1 or more risk factors at birth.2–5Moreover, not all infants with risk factors will necessarily be identified through targeted screening. For example, only 60% of infants in the United Kingdom with congenital hear-ing impairment who have known risk factors (neonatal intensive care unit, family history, craniofacial abnormality), one third over-all, are identified through targeted hearing screening.6

Dr Paradise proposes an “alternative, comparably large-scale national effort” to evaluate screening of high-risk infants and increased public and professional awareness as alternatives to universal newborn hearing screening (UNHS). Evidence of this kind is already available from the United Kingdom, where the majority of health districts use both targeted newborn hearing screening and a developmental check of all infants’ hearing at 6 to 9 months of age. The median age of entry into intervention in these districts is above 18 months of age.6Intervention by age 6 months is important to ensure normal language development.7 The UK experience, including a randomized, controlled trial, is that programs using UNHS get the majority of children into intervention by 6 months of age, at a cost per case detected only slightly higher than with targeted screening.6,8

We agree with Dr Paradise that attention must be paid to providing adequate facilities and professional services for fol-low-up of children identified with hearing impairment. We ac-knowledge the importance of the issue of parental concern regard-ing positive screenregard-ing results and recommend that programs monitor and address these concerns. We also agree that a public

awareness program to help identify infants with hearing loss is important to detect infants lost to follow-up and those who have a late-onset or progressive hearing loss.

We encourage pediatricians to use the framework provided by Paradise in formulating their positions on newborn hearing screening. At the same time, on the basis of the available evidence, we concur with the AAP policy statement9that supports hospitals and communities in their efforts to develop and implement uni-versal hearing detection and intervention programs. In so doing, these programs should address the important questions and con-cerns that Dr Paradise raises.

Scott Grosse, PhD Mike Adams, MD June Holstrum, PhD Kim Van Naarden, MPH Coleen Boyle, PhD

National Center for Environmental Health Centers for Disease Control and Prevention Atlanta, GA 30341

REFERENCES

1. Paradise JL. Universal newborn hearing screening: should we leap before we look?Pediatrics.1999;103:670 – 672

2. MMWR. Serious hearing impairment among children aged 3–10 years—Atlanta, Georgia, 1991–1993. MMWR Morb Mortal Wkly Rep. 1997;46:1073–1075

3. Mauk GW, White KR. Giving children a sound beginning: The promise of universal newborn screening.Volta Rev.1995;97:5–32

4. Mutton P. Early identification of deaf babies.Lancet.1998;352:1951–1952 5. Stein LK. Factors influencing the efficacy of universal newborn hearing

screening.Hearing Loss in Children.1999;46:95–105

6. Davis A, Bamford J, Wilson I, et al. A critical review of the role of neonatal hearing screening in the detection of congenital hearing im-pairment.Health Technol Assessment.1997;1(10)

7. Yoshinaga-Itano C, Sedey AL, Coulter DK, Mehl AL. Language of early-and later-identified children with hearing loss. Pediatrics. 1998;102: 1161–1167

8. Wessex Universal Neonatal Hearing Screening Trial Group. Controlled trial of universal neonatal screening for early identification of perma-nent childhood hearing impairment.Lancet.1998;352:1957–1964 9. American Academy of Pediatrics, Task Force on Newborn and Infant

Hearing. Newborn and infant hearing loss: detection and intervention. Pediatrics.1999;103:527–530

In Reply.—

I appreciate Dr Frankenburg’s comments, and particularly his endorsement of Fred Bess’ and my view1,2that deciding for or against universal neonatal hearing screening should await the results of a large-scale randomized trial. Dr Frankenburg’s cost-analysis approach is germane and interesting; to his list would need to be added other potential untoward consequences of false-positive identification, as we have discussed previously2: disrup-tion of parent-infant bonding, unfavorable “labeling,” unneces-sary diagnostic tests, and inappropriate therapeutic procedures. In addition, agreed-upon dollar values for many of the positive and negative factors in such a cost analysis will not be easy to come by. Dr Grosse and colleagues cite 4 references3– 6as indicating that only about half of all infants with congenital hearing impairment have 1 or more risk factors (as listed on the Joint Committee on Infant Hearing high-risk register [HRR]7) at birth. None of the 4 references contain primary data; each refers, in turn, to other reports. In one of those reports,8indeed only 48% of congenitally hearing-impaired children had a high-risk factor (as listed on an early [1982] version of the HRR9), but 33% had been intensive care nursery (ICN) graduates. The authors did not indicate how much overlap existed between the 2 subgroups, but surely the percent-age who fell into either the then-current HRR category or the ICN category, or both, would have exceeded 50%. In another of the reports,10involving 6- to 9-year-old children with educationally significant sensorineural hearing loss, 50% of the children had had a high-risk factor (again as listed on the 1982 version of the HRR9), but the authors indicated that including ICN admission as a risk factor would have raised that value to 63%. If, as seems likely, the study population included certain children with later-onset or

LETTERS TO THE EDITOR 617

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acquired hearing loss whose loss could not have been detected as neonates, exclusion of those children from the denominator would have resulted in an even higher percentage classifiable as HRR/ ICN, particularly if one referred to the most recent (1994) version of the HRR.7Finally, in the United Kingdom study11cited later by Dr Grosse and colleagues, which involved a total of 53 781 new-borns, 70% (37/53) of the infants eventually identified as having moderate to profound bilateral sensorineural hearing loss had high-risk factors and/or were in special/ICNs. I stand by my statement that most of the infants with sensorineural hearing impairment detectable in the newborn period will be found by screening only newborns who meet HRR criteria and/or are ad-mitted to an ICN.12

The statement that “intervention by age 6 months is important to ensure normal language development” refers to the study by Yoshinaga-Itano and colleagues,13 limitations of which I have discussed previously.14The study consisted of a nonrandomized comparison, some of its findings seem counterintuitive, and its authors considered its results suggestive rather than indicative of a cause-and-effect relationship.

Dr Grosse and colleagues express agreement with concerns that Bess and I have raised about adequacy of facilities and follow-up, about the need for greater public awareness, and particularly about the impact of false-positive identifications. Unfortunately, those concerns appear to be receiving little more than lip service in the drive to implement universal screening.

Once again, I refer the interested reader to a previously pub-lished exchange of correspondence (Pediatrics. 1994;94:948 –963) that provides a comprehensive summary of arguments for and against universal newborn hearing screening. A more recent ex-change (Pediatrics. 1999;104:351–355) adds new details. It seems likely that the arguments will continue to be voiced until such time as the issue is settled one way or the other by an appropri-ately designed and executed study or, less rationally and far more expensively, by the cumulative weight of favorable or unfavorable experience.

Jack L. Paradise, MD

Department of Pediatrics University of Pittsburgh School of Medicine and

Children’s Hospital of Pittsburgh Pittsburgh, PA 15213-2583

REFERENCES

1. Bess FH, Paradise JL. Universal screening for infant hearing impairment: not simple, not risk-free, not necessarily beneficial and not presently justified.Pediatrics.1994;93:330 –334

2. Bess FH, Paradise JL. Reply to letters concerning universal screening for infant hearing impairment.Pediatrics.1994;94:959 –963

3. MMWR. Serious hearing impairment among children aged 3–10 years—Atlanta, Georgia, 1991–1993. MMWR Morb Mortal Wkly Rep. 1997;46:1073–1076

4. Mauk GW, White KR. Giving children a sound beginning: the promise of universal newborn hearing screening.Volta Rev.1995;97:5–32 5. Mutton P. Early identification of deaf babies.Lancet.1998;352:1951–1952 6. Stein LK. Factors influencing the efficacy of universal newborn hearing

screening.Pediatr Clin North Am.1999;46:95–105

7. Joint Committee on Infant Hearing. 1994 position statement.Pediatrics. 1995;95:152–156

8. Elssmann SF, Matkin ND, Sabo MP. Early identification of congenital sensorineural hearing impairment.Hear J.1987;40:13–17

9. Joint Committee on Infant Hearing. 1982 position statement.Pediatrics. 1982;70:496 – 497

10. 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

11. Wessex Universal Neonatal Hearing Screening Trial Group. Controlled trial of universal neonatal screening for early identification of perma-nent childhood hearing impairment.Lancet.1998;352:1957–1964 12. Paradise JL. Universal newborn hearing screening: should we leap

before we look?Pediatrics.1999;103:670 – 672

13. Yoshinaga-Itano C, Sedey AL, Coulter DK, Mehl AL. Language of early-and later-identified children with hearing loss. Pediatrics. 1998;102: 1161–1171

14. Paradise JL. Reply to letters concerning universal newborn hearing screening: should we leap before we look?Pediatrics.1999;104:354 –355

Cardiopulmonary Resuscitation in Very Low

Birth Weight Infants

To the Editor.—

We read with interest the recent manuscript entitled “Cardio-pulmonary Resuscitation in the Very Low Birth Weight Infant: The Vermont Oxford Network Experience.”1The authors should be commended for pursuing a complex clinical issue. However, there are significant deficiencies in study design and data analysis that limit the interpretation and significance of the data.

Briefly, as background information, the birth of an infant is associated with abrupt cessation of the fetomaternal circulation with subsequent rapid and profound physiologic changes involv-ing both the cardiac and respiratory systems. Failure of either system to adapt will result in cardiorespiratory compromise and the need for resuscitation. Two cardinal events appear critical in the genesis of compromised cardiorespiratory adaptation and the subsequent need for intensive resuscitation (CPR). These events include failure to establish a functional residual capacity (FRC) and the presence of impaired placental gas exchange as evidenced by profound fetal acidemia (umbilical cord arterial pH ⱕ7.00 [2,31]). Failure to establish a FRC may be secondary to ineffective or improper ventilation or an abnormal underlying pulmonary state, eg, pulmonary hypoplasia, pulmonary immaturity, etc. In-deed, we previously documented the prominent association be-tween improper or ineffective ventilatory support and the need for CPR.4The presence of asphyxia may be suspected when there is an associated sentinal event, eg, abruptio placentae, but often is clinically inapparent in the delivery room.5Recovery after CPR is in part influenced by the cause of the cardiorespiratory compro-mise, the duration of the precipitating event, the establishment of effective ventilation, and restoration of spontaneous circulation (ROSC) with establishment of coronary perfusion pressure.

To briefly illustrate the importance of the above issues, we present data on infants⬍1500 g birth weight who received CPR (n⫽21), ie, chest compression only (n⫽17) and additionally epinephrine (n⫽4) for the years 1996 –1998. It should be noted that in cases of uncertain prognosis (birth weight⬍600 g) CPR is not pursued in our delivery room (DR) in all cases. Moreover, prophylactic surfactant is not administered in the DR. The per-centage of infants receiving CPR by birth weight is shown in Table 1.

The percentage of infants requiring chest compressions is com-parable to the Vermont Oxford Network experience. However, the percentage of infants requiring epinephrine is markedly less. Cur-rent National Resuscitation Program (NRP) guidelines are fol-lowed in our institution; however, we stress the importance of ventilation in ROSC.

This may in part explain the less frequent use of epinephrine. The perinatal characteristics and short-term outcome of the 21 infants are illustrated in Table 2.

The data highlight several important points that are crucial when assessing the efficacy of CPR including: 1) the duration of CPR which was significantly longer for infants who died, 270⫾ 231 versus 43⫾32 seconds (P⫽.0004) for all survivors, and even shorter for those survivors without severe IVH 30⫾12 seconds; 2) lethal conditions incompatible with life which included pulmo-nary hypoplasia, complex heart disease (unknown in the DR) and

TABLE 1. DR-CPR by Birth Weight

BW (G) n DR-Cardiac

Compression %

Epinephrine %

501–750 80 7.5% (1.25%)

751–1000 103 6.7% 0

1001–1250 146 2.7% 0

1251–1500 196 2.0% 3 (1.5%)

DR-CPR indicates delivery room resuscitation; BW, birth weight; G, grams.

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DOI: 10.1542/peds.106.3.616

2000;106;616

Pediatrics

William K. Frankenburg

Universal Neonatal Hearing Screening

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DOI: 10.1542/peds.106.3.616

2000;106;616

Pediatrics

William K. Frankenburg

Universal Neonatal Hearing Screening

http://pediatrics.aappublications.org/content/106/3/616

located on the World Wide Web at:

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

the American Academy of Pediatrics, 345 Park Avenue, Itasca, Illinois, 60143. Copyright © 2000

has been published continuously since 1948. Pediatrics is owned, published, and trademarked by

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Figure

Fig 1. Screening outcomes.
TABLE 1.DR-CPR by Birth Weight

References

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