ARTICLEPEDIATRICS Volume 139 , number 2 , February 2017 :e 20160789

Outcomes From a Hearing-Targeted Cytomegalovirus Screening ProgramMarissa L. Diener, PhD, a Cathleen D. Zick, PhD, a Stephanie Browning McVicar, AuD, CCC-A, b Jill Boettger, MS, CCC-A/SLP, b Albert H. Park, MDc

abstractBACKGROUND AND OBJECTIVES: Cytomegalovirus (CMV) is the most common congenital infection

and nongenetic cause of congenital sensorineural hearing loss in the United States. Utah

was the first state to pass legislation mandating CMV screening for newborns who fail

newborn hearing screening (NBHS). The study objective was to present outcomes of

hearing-targeted CMV screening and determine factors predicting CMV screening.

METHODS: We used Utah Department of Health HiTrack and Vital Records databases

to examine CMV screening from 509 infants who failed NBHS in the 24 months after

implementation of the Utah legislation. Multivariate logistic regression analyses were

conducted to identify predictors of compliance with CMV screening and diagnostic hearing

evaluation.

RESULTS: Sixty-two percent of infants who never passed hearing screening underwent CMV

screening. Fourteen of 234 infants tested within 21 days were CMV positive; 6 (42.9%)

had hearing loss. Seventy-seven percent of eligible infants completed a diagnostic hearing

evaluation within 90 days of birth. Compliance with CMV screening was associated with

sociodemographic factors, time since the law was enacted, and NBHS protocol. Infants born

after the legislation showed greater odds of achieving timely diagnostic hearing evaluation

than infants born before the law.

CONCLUSIONS: Incorporating CMV screening into an established NBHS program is a viable

option for the identification of CMV in infants failing NBHS. The addition of CMV testing can

help a NBHS program attain timely audiological diagnostics within 90 days, an important

early hearing detection and intervention milestone.

Departments of aFamily and Consumer Studies and cSurgery, Division of Otolaryngology–Head and Neck

Surgery, University of Utah, Salt Lake City, Utah; and bUtah Department of Health, Salt Lake City, Utah

Dr Diener conceptualized the study, drafted parts of the initial manuscript, helped analyze the

data, and reviewed and revised the manuscript; Dr Zick conceptualized the study, drafted parts of

the initial manuscript, carried out the initial analyses, and reviewed and revised the manuscript;

Dr McVicar designed the data collection instruments, coordinated and supervised data collection

at the Department of Health, extracted the Early Hearing Detection and Intervention (EHDI) and

birth certifi cate data, and critically reviewed the manuscript; Ms Boettger coordinated and

supervised data collection at the Department of Health, extracted the EHDI and birth certifi cate

data, and critically reviewed the manuscript; Dr Park provided substantial contributions to the

conception and design of the study, facilitated the acquisition of data between HiTrack and Vital

Records, and critically reviewed the manuscript; and all authors approved the fi nal manuscript as

submitted.

DOI: 10.1542/peds.2016-0789

Accepted for publication Nov 17, 2016

Address correspondence to Marissa Diener, PhD, 225 South 1400 East, Room 228 AEB, Department

of Family and Consumer Studies, University of Utah, Salt Lake City, UT 84112. E-mail: marissa.

diener@fcs.utah.edu

To cite: Diener ML, Zick CD, McVicar SB, et al. Outcomes

From a Hearing-Targeted Cytomegalovirus Screening

Program. Pediatrics. 2017;139(2):e20160789

WHAT’S KNOWN ON THIS SUBJECT: Cytomegalovirus

(CMV) is the most common congenital infection

and nongenetic cause of congenital sensorineural

hearing loss in the United States. Utah was the

fi rst state to introduce statewide hearing-targeted

CMV screening. Other states have considered

implementing this approach.

WHAT THIS STUDY ADDS: Results from the Utah

statewide hearing-targeted CMV program and

diagnostic hearing evaluation data are presented.

These data can inform public health campaigns and

policymakers considering legislation on congenital

CMV.

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Cytomegalovirus (CMV) is the most

common congenital infection, with

a prevalence of 0.3% to 1.2% in

industrialized nations, and is the

most common nongenetic cause of

congenital sensorineural hearing

loss (SNHL) in the United States. 1 – 3

Although the exact magnitude is

unclear, 6% to 30% of pediatric

hearing loss may be attributed to

congenital CMV (cCMV). 2, 4 – 7

Congenital CMV often goes

undetected at birth because most

newborns are “asymptomatic”

with no obvious clinical signs or

symptoms other than a possible

failed newborn hearing screen

(NBHS), although 10% to 15% of

these asymptomatic infants have

or will develop SNHL. 8, 9 For the

purposes of this study, infants with

no clinical symptoms other than

isolated hearing loss were considered

asymptomatic because the only

way to identify them was through

CMV screening or hearing screening

followed by CMV screening. Targeted

screening of infants for cCMV based

on a failed NBHS (hearing targeted

early CMV screening; HT-CMV) is

1 approach for early detection of

asymptomatic cCMV. Although the

cost for CMV screening is higher than

cost estimates for other newborn

screening tests, few infants are

tested in an HT-CMV approach,

making the CMV screening process

more similar to diagnostic testing

than to screening. As such, CMV

screening appears to be relatively

low cost compared with diagnostic

testing, although the impact on

health outcomes have not yet been

examined. 10, 11

In 2013, Utah became the first

state to enact a CMV public health

initiative on CMV education and

testing (UCA 26-10-10). 12 The first

provision of the law charged the Utah

Department of Health with creating

an education program about birth

defects associated with and ways

to prevent cCMV. This program

has an approximate annual budget

of $30 000 and targets women of

childbearing ages, as well as child care

and health care providers. The second

provision mandates that all infants

who fail their NBHS be tested for CMV

within the first 3 weeks of life unless

a parent declines the test. The 21-day

period is required to differentiate

cCMV from postnatally acquired CMV,

which is not associated with childhood

hearing loss. 1, 13, 14 The associated Rule

(R398-4) further clarifies eligibility

criteria for CMV screening as those

infants who fail both their inpatient

and return outpatient screening

or those infants who fail their first

hearing screening if it occurs after

age 14 days. The legislation was

designed to identify asymptomatic

infants most at risk for hearing loss:

those who fail NBHS.15, 16 In special

populations where NBHS(s) cannot

be accomplished before 21 days of age

(such as those in the NICU), testing

for cCMV is left to the discretion of the

medical practitioner(s) caring for the

newborn.

The Utah law is serving as a model

for actions in other states. In 2015,

Connecticut enacted legislation that

mirrors Utah’s mandatory testing

provision for newborns. 17 Texas,

Tennessee, Hawaii, and Illinois

enacted legislation to increase

cCMV educational efforts; Illinois

now provides information on

testing options for cCMV and early

intervention. 18 –21 The results of

Utah’s work are timely and have

implications for national public

policy. The goal of this study is to

present the results from the first

2 years after Utah’s legislation and

determine which sociodemographic

and health care characteristics are

related to compliance for timely CMV

screening and diagnostic hearing

evaluation.

METHODS

Data

The data for the current study were

drawn from 2 sources: the Utah

Department of Health’s Early Hearing

Detection and Intervention (EHDI)

Tracking and Data Management

System (HiTrack) and the Utah Vital

Records database for births that

occurred in the 24 months before

and after the implementation of the

law on July 1, 2013. The number of

infants who failed their NBHS and

the number of infants eligible who

actually underwent CMV screening

were determined from Utah’s EHDI

HiTrack database. Out of 103 868

births, 705 newborns (0.7%) failed

their inpatient and subsequent NBHS

or failed their first hearing screen

after age 14 days and were eligible

for CMV screening according to

the legislation. Nationally, ∼1.6%

of infants failed their final NBHS

in 2013. 22 Potential factors that

may influence successful early

CMV screening were obtained from

Vital Records. Sixteen records were

dropped because of missing data

on mother’s education and thus,

689 infants had vital records data.

Not included in the current study

were infants in special populations

such as those in the NICU who had

NBHS after 21 days. Thirteen infants

with symptomatic cCMV (ie, those

who showed clinical symptoms at

birth that led to CMV screening such

as thrombocytopenia, petechiae,

hepatomegaly, splenomegaly,

intrauterine growth restriction,

hepatitis, or central nervous system

involvement) were all in the NICU

and not included in the current

study because the focus of the

legislation was on infants who failed

NBHS. The study was approved by

the Institutional Review Boards at

Primary Children’s Hospital, the

University of Utah, and the Utah

Department of Health.

Statistical Methods

Descriptive statistics on the

sociodemographic characteristics

of mothers and the hearing

screening that led to CMV screening

eligibility and the national EHDI

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PEDIATRICS Volume 139 , number 2 , February 2017

3-month diagnostic audiologic

follow-up milestone attainment

were examined. Multivariate logistic

regression analyses, appropriate for

dichotomous outcomes, assessed

what characteristics were linked

to the likelihood of the infant

undergoing (1) CMV screening by 3

weeks of age and (2) an audiological

diagnostic evaluation by 3 months of

age. The events-to-variable ratio was

>10 for all analyses involving logistic

regression. The odds ratios (ORs)

generated from such regressions

can be interpreted as the effect of

being in a specific category relative

to the reference group on the odds

of the outcome in question, holding

all other covariates constant. 23

Collinearity diagnostics undertaken

as part of the multivariate analyses

revealed no causes for concerns

regarding multicollinearity among

the independent variables.

RESULTS

Descriptive Statistics

Figure 1 shows the various CMV

screening patterns for those who

were eligible for CMV screening

according to the HT-CMV legislation

because of an inpatient and

outpatient failed screen or an initial

failed screen after 14 days. One

hundred eighty infants passed a

subsequent hearing screen and were

not referred for CMV screening (and

excluded from subsequent analyses).

Fourteen (6.0%) of the 234 infants

tested within 21 days of birth were

positive, and 6 (42.9%) of those had

confirmed hearing loss. The type

of CMV test varied and is indicated

in Fig 1. Saliva samples for CMV

screening were taken at least 60

minutes after breastfeeding. 24 Seven

(8.8%) of the 80 infants tested after

21 days were CMV positive, and 3

(42.9%) of those had confirmed

hearing loss.

Table 1 provides descriptive

information on the mothers and their

infants born both before and after

the legislation who never passed a

NBHS. Mothers whose infants failed

NBHS (and were referred for CMV

screening after the legislation) were

more likely than the population

of all births to be younger than 29

years of age, not married, have less

than a college education, have the

birth covered by Medicaid, have a

3

FIGURE 1CMV testing fi gures for Utah births between July 1, 2013, and June 30, 2015. a One infant who passed NBHS had a twin who never passed a NBHS. Subsequently, both infants were screened for CMV and found to be CMV positive. The twin with normal hearing who passed the hearing screening was not included in the analyses, however. CHL, conductive hearing loss; PCR, polymerase chain reaction.

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nonhospital birth, and live away from

the urban Wasatch Front.

Figure 2 illustrates the 90-day

diagnostic hearing evaluation

patterns for the infants who failed

their NBHS(s) after the law. After

the legislation, 77% of all infants

received timely diagnostic hearing

evaluation (compared with 56% in

the 24 months before the legislation).

After the law, among the infants

who received a CMV test, 272 of 314

(86.6%) had the diagnostic hearing

evaluation within 90 days, compared

with 120 of 195 (61.5%) infants

who did not receive a CMV test (see

Fig 3). After the legislation, 54%

of the infants who had diagnostic

hearing evaluations after 90 days

had comorbidities (eg, chronic otitis

media with or without cleft lip and

palate) that delayed final audiological

determination.

Among all infants who completed a

diagnostic hearing evaluation after

the legislation, 218 (50.2% of 434

infants) were found to have had

normal hearing, whereas 215 (49.5%

of 434 infants) were confirmed

to have a hearing loss. Eighty-five

infants had SNHL, 19 mixed, 87

conductive hearing loss, 5 auditory

neuropathy spectrum disorder, and

19 undetermined. Eighty-five percent

of infants diagnosed with SNHL had

CMV screening completed.

Multivariate Analyses

Multivariate analyses examined the

sociodemographic and health care

variables associated with achieving

CMV screening compliance ( Table 2)

and the 3-month hearing diagnostic

target ( Table 3). We examined

the sociodemographic and NBHS

protocol predictors of infants

completing CMV screening within 21

days of birth.

The multivariate analyses showed

an OR of 0.23 (confidence interval

[CI] 0.11–0.51), indicating the odds

of CMV screening was 77% lower

for infants who received 2 screens,

at least 1 of which occurred after

14 days of birth (n = 9 infants; 10%

of those who had 2 screens with at

least 1 after 14 days) compared with

those infants who underwent the

recommended 2 screenings within

14 days of birth (n = 162; 66% of

those who had 2 screens within 14

days of birth). The odds of an infant

getting CMV screening for those born

in the first 6 months after the law

was enacted were 64% lower than

those born after the first 6 months

(OR = 0.36, CI = 0.23–0.57). The odds

of an infant getting CMV tested were

1.84 times higher (CI = 1.17–2.89)

for infants born to a mother with a

4

TABLE 1 Descriptive Information Regarding the Study Populations in Utah, 2011–2015

Variables July 2013–June 2015 July 2011–June 2013

Did Not Pass All Hearing

Screensa

All Births, % Did Not Pass All Hearing

Screensa

All Births, %

n % n %

Total 509 569

Mother’s age, y

≤29 271 53.24b 59.64 360 63.27 61.37

>29 238 46.76b 40.36 209 36.73 30.63

Mother’s education

Less than bachelor’s degree 367 72.10b 67.70 408 71.70 69.33

Bachelor’s degree or higher 142 27.90b 32.30 161 28.30 30.67

Mother’s ethnicity

Non-Latina 436 85.66 85.19 481 84.53 85.52

Latina 73 14.34 14.81 88 15.47 14.48

Mother’s marital status

Not married 124 24.36b 18.78 129 22.67b 18.42

Married 385 75.64b 81.22 440 77.33b 81.58

Birth covered by Medicaid

No 356 69.94b 74.01 394 69.24 72.54

Yes 153 30.06b 25.99 175 30.76 27.46

Nonhospital birth

No 457 89.78b 96.77 505 88.75b 96.30

Yes 52 10.22b 3.23 64 11.25b 3.13

Resides in urban, Wasatch Front

No 155 30.45b 24.89 197 34.62b 24.71

Yes 354 69.55b 75.11 393 65.38b 75.29

Number of siblings

0 147 28.88 32.06 180 31.63b 32.39

1–2 258 50.69 47.90 228 40.07b 47.41

3+ 104 20.43 20.03 137 24.08b 20.20

a Excludes infants who eventually passed a NBHS.b Signifi cantly different from the Utah birth population based on χ2 tests (P < .05).

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PEDIATRICS Volume 139 , number 2 , February 2017

bachelor’s degree or higher relative

to those with less than a bachelor’s

degree. The location of the infant’s

birth was also associated with the

odds of undergoing CMV screening

within 21 days of birth; the odds of

an infant receiving CMV screening

were 69% lower (OR = 0.31, CI =

0.15–0.63) for infants born outside a

hospital compared with those born in

a hospital.

In Table 3, we present the logistic

regression OR estimates of factors

associated with eligible infants

undergoing a diagnostic hearing

evaluation by 3 months of age for all

infants born 24 months before and

24 months after the legislation was

enacted. Two multivariate models are

estimated. The first model adjusts for

the different CMV screening patterns

with the reference group being

infants born before the CMV law

went into effect. The second model

provides a strict test of the law’s

impact on the likelihood of complying

with the 3-month diagnostic hearing

evaluation regardless of the pattern

of CMV screening.

Column 3 in Table 3 reveals that

timely diagnostic hearing evaluation

rates improved after the legislation

from 56% to 77%. Model 2 shows

that the odds of an infant receiving a

timely diagnostic hearing evaluation

were 2.75 times higher (CI =

2.08–3.64) for those infants born

after the law compared with those

born before the legislation. Model

1 demonstrates that the pattern

of CMV screening was related to

the odds of undergoing a timely

diagnostic evaluation. Although all

5

FIGURE 2Three-month diagnostic hearing evaluation fi gures for Utah infants who failed their NBHS and were asymptomatic for CMV for the 24-month period after enactment of the law (July 2013–June 2015). a Infants whose diagnostic evaluations were still in process had received at least 1 diagnostic evaluation but had not yet received a confi rmed diagnosis.

FIGURE 3Numbers of infants who completed diagnostic hearing evaluations within 90 days by CMV testing status.

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infants were more likely to undergo

timely diagnostic evaluation after

the legislation compared with those

born before the law, the magnitude

of the OR varied by CMV screening

pattern. Infants who underwent

CMV screening within 21 days were

>5 times as likely to have timely

diagnostic testing done relative to

those born before the CMV screening

legislation was enacted (OR = 5.46,

CI = 3.52–8.48). In contrast, the effect

size, although still positive, is smaller

for infants who were tested after 21

days and infants who did not undergo

CMV testing. These differences in

magnitude across the groups may

reflect a “compliance effect” in which

parents who comply with the CMV

testing also comply with the 3-month

diagnostic hearing evaluation

recommendation.

Models 1 and 2 reveal that several

sociodemographic factors were

also linked to the odds of meeting

the diagnostic recommendations.

Mothers with college degrees and

married mothers had greater odds

of completing diagnostic testing by

3 months of age relative to mothers

with less education and unmarried

mothers. Births covered by Medicaid

were more likely to comply with

the 3-month diagnostic testing

recommendation than those births

not covered by Medicaid. Conversely,

the odds of an infant completing

timely diagnostic testing were lower

for those who had a nonhospital

birth compared with infants born in

the hospital. The odds of an infant

completing timely diagnostic testing

were also lower for children with

siblings compared with only children.

DISCUSSION

This study is the first to assess the

implementation of a statewide

HT-CMV screening of infants after

the enactment of CMV legislation.

Fourteen infants were identified as

CMV-positive within 21 days after

birth; 6 had hearing loss. It is highly

likely that these asymptomatic

cCMV infected children would not

have been diagnosed at a later

time because of the difficulty

distinguishing postnatally acquired

6

TABLE 2 Associations of Maternal and Infant Factors With Follow-up CMV Testing Among Infants Who Never Passed a NBHS, July 2013–June 2015 (N = 509)

Variables Undergoing CMV Testing Within

21 Days

Relative Risk (95% CI)

n % Crude Adjusteda

Total 234 45.97

Mother’s age, y

≤29 125 46.13 Reference Reference

>29 109 45.80 0.99 (0.70–1.40) 0.96 (0.63–1.48)

Mother’s education

Less than bachelor’s degree 152 41.42 Reference Reference

Bachelor’s degree or higher 82 57.75 1.93 (1.31–2.86) 1.84 (1.17–2.89)

Mother’s ethnicity

Non-Latina 206 47.25 Reference Reference

Latina 28 38.36 0.70 (0.42–1.15) 0.69 (0.39–1.21)

Mother’s marital status

Not married 53 42.74 Reference Reference

Married 181 47.01 1.19 (0.80–1.79) 0.91 (0.55–1.52)

Birth covered by Medicaid

No 168 47.19 Reference Reference

Yes 66 43.14 0.85 (0.58–1.24) 0.99 (0.63–1.57)

Nonhospital birth

No 222 48.58 Reference Reference

Yes 12 23.08 0.32 (0.16–0.62) 0.31 (0.15–0.63)

Resides in urban, Wasatch Front

No 64 41.29 Reference Reference

Yes 170 48.02 1.31 (0.90–1.92) 1.37 (0.89–2.11)

Number of siblings

0 72 48.98 Reference Reference

1–2 122 47.29 0.82 (0.57–1.18) 0.94 (0.60–1.48)

3+ 40 38.46 0.54 (0.34–0.85) 0.90 (0.49–1.68)

Birth date

January 1, 2014–June 30, 2015 197 51.30 Reference Reference

July 1, 2013–December 30, 2013 37 29.60 0.46 (0.31–0.68) 0.36 (0.23–0.57)

NBHSs

2 screens ≤14 d old 162 48.07 Reference Reference

1 screen and referred for testing 10 58.82 1.54 (0.57–4.15) 1.59 (0.57–4.49)

2 hearing screens >14 d old 9 17.65 0.23 (0.11–0.49) 0.23 (0.11–0.51)

3 hearing screens 53 50.96 1.12 (0.72–1.74) 1.12 (0.69–1.82)

a Data were adjusted for the variables in the table. The adjusted model χ2 = 66.46 (P < .01).

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CMV from cCMV after 3 weeks of

age from urine or saliva. Alternative

approaches to cCMV diagnosis at a

later date by using archived neonatal

dried blood spot (DBS) polymerase

chain reaction testing may be an

appropriate testing strategy for

infants with hearing loss, depending

on the sensitivity of the DBS assay

used and length of storage of DBS

neonatal Guthrie cards. 25, 26

Identification of asymptomatic

CMV-positive children provides

opportunities for focused

surveillance. Fowler and others have

shown that cCMV positive infants are

at risk for delayed onset, fluctuating,

and progressive SNHL. 7 – 9, 16 This

risk has been reported to be 8% to

15% or higher for asymptomatic

cCMV.16, 27 Repeated hearing testing

of cCMV children also provides an

opportunity to detect changes in

hearing thresholds and provide

earlier intervention. Furthermore,

HT-CMV screening may improve

the diagnosis of symptomatic cCMV

infected infants. Future research

should examine the hypothesis that

HT-CMV screening improves the

diagnosis of not only asymptomatic

but also symptomatic cCMV infants.

Infants born after the CMV legislation

were significantly more likely

to undergo diagnostic hearing

evaluation by 3 months of age than

infants born before the HT-CMV

legislation. Educational campaigns

and urgency surrounding CMV also

may have increased awareness of

the importance of timely diagnostic

hearing evaluation. Delays in the

diagnosis and treatment of hearing

loss are a major challenge for

universal NBHS programs. Thus,

HT-CMV screening has implications

not just for CMV infected but for

all infants who are hard of hearing.

Several studies have demonstrated

improved language outcomes when

children who are hard of hearing

7

TABLE 3 Associations of Maternal and Infant Factors With Follow-up Diagnostic Hearing Evaluation by 3 Months of Age Among Infants Who Failed Their

NBHSs, July 2011–June 2015 (N = 1078)

Variables Diagnostic Hearing Evaluation

by 3 Months

Relative Risk (95% CI)

n % Crude Model 1 Adjusteda Model 2 Adjustedb

Total 712 66.05

Mother’s age, y

≤29 406 64.34 Reference Reference Reference

>29 306 68.46 1.20 (0.93–1.56) 1.33 (0.96–1.85) 1.31 (0.95–1.81)

Mother’s education

Less than bachelor’s degree 487 62.84 Reference Reference Reference

Bachelor’s degree or higher 225 74.86 1.71 (1.27–2.29) 1.53 (1.08–2.17) 1.62 (1.15–2.29)

Mother’s ethnicity

Non–Latina 604 65.87 Reference Reference Reference

Latina 108 67.08 1.06 (0.74–1.51) 1.39 (0.94–2.07) 1.33 (0.90–1.96)

Mother’s marital status

Not married 145 57.31 Reference Reference Reference

Married 567 68.73 1.64 (1.23–2.19) 1.97 (1.38–2.81) 1.96 (1.38–2.78)

Birth covered by Medicaid

No 495 66.00 Reference Reference Reference

Yes 217 66.16 1.01 (0.77–1.33) 1.39 (1.01–1.91) 1.41 (1.02–1.93)

Nonhospital birth

No 662 68.81 Reference Reference Reference

Yes 50 43.10 0.34 (0.23–0.51) 0.37 (0.24–0.57) 0.33 (0.22–0.50)

Resides in urban, Wasatch Front

No 213 60.51 Reference Reference Reference

Yes 499 68.73 1.44 (1.10–2.87) 1.27 (0.95–1.71) 1.34 (1.01–1.79)

Number of siblings

0 240 73.39 Reference Reference Reference

1–2 339 69.75 1.07 (0.79–1.44) 0.70 (0.50–0.98) 0.71 (0.59–1.99)

3+ 133 55.19 0.57 (0.41–0.80) 0.42 (0.27–0.65) 0.40 (0.26–0.62)

CMV testing status

Born before CMV testing mandate enacted 320 56.24 Reference Reference Reference

Born after the CMV testing mandate

enacted: no CMV test

120 61.54 1.25 (0.89–1.74) 1.46 (1.02–2.07)

Born after the CMV testing mandate

enacted: CMV test after 21 d

66 82.50 3.67 (2.01–6.68) 3.63 (1.95–6.74)

Born after the CMV testing mandate

enacted: CMV test within 21 d

206 88.03 5.73 (3.73–8.78) 5.46 (3.52–8.48)

Born after CMV testing mandate enacted 392 77.01 2.61 (2.00–3.41) 2.75 (2.08–3.64)

a Data were adjusted for the variables in the table. The adjusted model χ2 = 132.08 (P < .01).b Data were adjusted for the variables in the table. The adjusted model χ2 = 161.66 (P < .01).

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are diagnosed and treated before 6

months of age. 28 – 30

We found parents of infants

who complied with the CMV

testing recommendations were

more likely to comply with the

recommendation of having the

infant undergo a diagnostic

evaluation in a timely manner. This

finding suggests that educational

efforts and systems should target

CMV noncompliers; identifying

infants who failed

to be tested for CMV in a timely

manner may be important for

improving timely diagnostic

hearing rates by targeting this

hard-to-reach group.

For those interested in improving

CMV screening compliance, our

multivariate analysis demonstrated

several factors that were associated

with CMV screening success.

Lower maternal education and

nonhospital births were associated

with lower odds of CMV screening.

Those infants who did not have 2

hearing screens before 14 days of

age had lower odds of receiving

timely CMV screening. Continuing

education of health care providers

and parents on the importance

of early CMV screening is critical

given that testing after 21 days

cannot differentiate cCMV and

CMV acquired postnatally. Focused

efforts to improve awareness of the

importance of CMV screening among

midwives who often are involved

with nonhospital births should

improve screening success.

Both universal CMV screening and

HT-CMV screening approaches have

advantages and disadvantages.

Universal screening is optimal

for identifying all cCMV-infected

newborns, including the 10% to 15%

of asymptomatic cCMV-infected

infants who will develop hearing

loss after birth and those with

nonspecific symptoms who may

not be otherwise diagnosed. 16, 27, 31

Yet universal screening is a massive

undertaking, and costs are unknown.

An HT-CMV approach piggybacks

onto an ongoing NBHS program

that can facilitate implementation

and lower costs. Assuming the 1.5%

referral rate nationally, a more

modest 60 000 newborns would

need to be tested each year via this

HT-CMV approach. 22 On the other

hand, targeted CMV screening misses

asymptomatic infants who have

normal hearing at birth but will later

develop CMV-related hearing loss

and those infants with nonspecific

clinical symptoms who may not

receive CMV screening.

Rates of timely diagnostic

evaluation may also be

improved by focusing on several

sociodemographic groups who are

at-risk for lower compliance rates.

Specifically, larger families, single

mothers, mothers without a college

degree, those who had nonhospital

births and those not covered by

Medicaid were all at relatively

greater risk of not meeting the

3-month diagnostic testing

recommendation. Compliance

with the diagnostic testing

recommendations may be improved

by targeting educational and

outreach efforts to these groups.

One limitation of the study is the

inability to examine data that were

not reported to the Utah Department

of Health. Thus, we may have

overestimated noncompliance for

both CMV and audiological evaluation

if the health care provider did not

provide the information to the

Department of Health, although

multiple efforts were made to obtain

CMV testing information through

other means. The CMV Rule states

that medical providers must report

results of the CMV screening within

10 days of receiving results. The

Department of Health provides

EHDI and CMV Mandate reports

for each hospital and midwife so

that organizations can identify

their compliance rate and improve

performance.

CONCLUSIONS

A statewide HT-CMV approach can

identify cCMV-infected children.

The Utah program, being the first

in the nation, serves as a model on

which other states can build their

programs. Fourteen infants were

recognized who likely would not

have otherwise been identified

with cCMV over 2 years of testing.

Because lower maternal education,

nonhospital births, and NBHS that

occurred after 14 days of age were

associated with lower rates of

successful CMV screening, a directed

approach toward maternal and

midwife outreach, and educating

screeners may improve the success

of this and other programs. Timely

CMV screening rates increased

substantially from the first 6 months

to the subsequent 18 months after

the legislation, indicating successful

uptake of the screening program.

Statewide HT-CMV screening also

significantly improved the 3-month

diagnostic hearing evaluation rate.

Furthermore, the study identified

several sociodemographic and

CMV screening patterns related

to 3-month diagnostic hearing

evaluation that can be used to target

educational and outreach efforts

going forward.

ABBREVIATIONS

CI:  confidence interval

cCMV:  congenital

cytomegalovirus

CMV:  cytomegalovirus

DBS:  dried blood spot

EHDI:  Early Hearing Detection

and Intervention

HT-CMV:  hearing targeted early

cytomegalovirus

screening

NBHS:  newborn hearing screen

OR:  odds ratio

SNHL:  sensorineural hearing loss

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PEDIATRICS Volume 139 , number 2 , February 2017

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FINANCIAL DISCLOSURE: The authors have indicated they have no fi nancial relationships relevant to this article to disclose.

FUNDING: No external funding.

POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential confl icts of interest to disclose.

COMPANION PAPER: A companion to this article can be found online at www. pediatrics. org/ cgi/ doi/ 10. 1542/ peds. 2016- 3837.

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Albert H. ParkMarissa L. Diener, Cathleen D. Zick, Stephanie Browning McVicar, Jill Boettger and

Outcomes From a Hearing-Targeted Cytomegalovirus Screening Program

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