Newborn Screening Knowledge and Attitudes Among Midwives and Out-of-Hospital-Birth Parents

Abstract

Midwifery and nursing are collaborative partners in both education and practice. Understanding needs and barriers to clinical services such as newborn screening is essential. This study examined knowledge and attitudes of midwives and out-of-hospital-birth parents about newborn blood spot screening (NBS). Descriptive and cross-sectional surveys were distributed to midwives and out-of-hospital-birth patients from birth center registries and the Utah Health Department of Vital Records. Seventeen midwife surveys (response rate: 17%) and 113 parent surveys (response rate: 31%) were returned. Most midwives and out-of-hospital-birth parents reported satisfactory knowledge scores about NBS. Only 5% of parents (n = 6) did not participate in NBS. Most midwives reported that NBS is important and encouraged patients to consider undergoing NBS. Some concerns included the lack of education for both midwives and out-of-hospital patients and the trauma and accuracy of the heel prick soon after birth. Both midwives and out-of-hospital-birth parents expressed a need for improved NBS education. Additional studies are needed to ascertain whether this trend is seen with similar populations throughout the United States, to further elucidate the factors that drive NBS nonparticipation, and to develop educational resources for midwives and their patients.

Healthcare provider and parental support for newborn blood spot screening (NBS) is crucial for ensuring NBS participation.¹ Newborn blood spot screening is a population-based screening system that identifies infants who have certain genetic and metabolic disorders and provides additional follow-up services. Its success is dependent on rapid responses from all stakeholders (ie, parents, providers, state health departments, and laboratories) throughout the screening process.² Initially, NBS was screened only for phenylketonuria (PKU) but has since expanded to include multiple conditions (although some providers still use the nonpreferred term “PKU test” to refer to NBS). Newborn blood spot screening is recognized as one of the most successful public health programs in the United States over the past century.³ During a newborn’s first few days of life, blood is drawn from a heel stick; the blood spots are dried on filter paper, sent for analysis, and typically only the positive results are returned to parents.

Based on a nationwide effort, most state NBS programs adopted the Recommended Uniform Screening Panel that currently consists of 31 primary conditions. Many state programs conduct only 1 heel prick to obtain newborn blood specimens⁴). Utah, however, collects 2 newborn screening specimens, which are tested for 39 disorders. The first screen is usually done between 24 hours and 5 days of life, or right before discharge from the hospital. The second screen is done between 7 and 28 days of age, usually at the 2-week well-child check with the newborn’s pediatrician. It is expected that NBS will continue to expand the number of screens both in Utah and nationally as screening capabilities enable testing for a progressively larger set of uncommon conditions.¹

Public attitudes toward NBS demonstrate high support for NBS but low awareness.^5,6 This is, in part, because of the mandatory nature of NBS and current brochure-based educational efforts (brochures about NBS are given, together with a number of other materials, to new parents at the hospital immediately after the birth of the newborn). Previous studies examining parental knowledge of and attitudes about NBS programs found that 59% of new parents were unsure or did not know what blood spots were in the context of NBS.⁷ Additional studies also demonstrated similar results among healthcare providers. For example, one study revealed that providers saw their primary role as information givers, but many reported that they lacked knowledge, training, or time to give adequate explanations about NBS.⁸

Unfortunately, a gap in the literature remains regarding the knowledge and attitudes of out-of-hospital (OOH)-birth parents and providers about NBS. As the national trend of OOH births—defined as births that occur at home and at birthing centers—continues to rise, understanding, knowledge, and attitudes among these groups will be important for providing access to this public health program.⁹ Out-of-hospital births in the United States increased by 80.2% from 2009 to 2014, from 32 596 to 58 743; home births increased by 77.3%; and births in freestanding birthing centers increased by 79.6%.¹⁰ In 2014, 63.8% of OOH births were home births, 30.7% were in freestanding birthing centers, and 5.5% were in other places—physicians’ offices or clinics. In Utah, the rate of OOH births is double the national average.¹¹ Anecdotally, the rates of NBS participation are lower for OOH birth parents than for parents who have hospital births.^5,7,12,13 The reasons for declining NBS participation rates for OOH births remain largely unexplored, both nationally and within Utah. In Utah, parents can decline NBS for religious reasons but must submit a signed form to the Utah Department of Health NBS program. There are a variety of protocols in other states; some states allow no exemptions, and some states allow parents to decline NBS for any reason.

The importance of collaborative relationships between midwives and nursing is important for ensuring evidence-based practice during maternity care.^14,15 This is even more important for immediate pediatric care of the newborn. Newborn screening is a critical component of ensuring the health and safety of newborns. As many nursing programs also include midwifery clinical tracks, understanding the challenges and needs of midwives and OOH birth parents can help address these barriers as well as facilitators for ensuring that this public health program is offered adequately to all parents.¹⁶ However, there is still limited information about factors that affect collaboration between midwives and other healthcare professionals, including nurses, and more research is needed to better understand barriers and facilitators.

In this descriptive study, we aimed to help fill this gap in the literature and describe OOH birth parents’ and midwives’ knowledge, attitudes, and decisions about NBS. Because the OOH birth rate for the state of Utah is double the national average and the NBS nonparticipation rate for OOH is higher than the national average nonparticipation rate, this study provided opportunities to understand the extent to which barriers to NBS participation exist for midwives and their clients. The conceptual framework that guided this research was the social ecological model, which seeks to understand how interpersonal and environmental factors interact to determine behaviors, decisions, and facilitators that promote public health participation.^17,18

METHODS

Institutional review board approval was obtained prior to any research activity. A consent cover letter was included in the mailed packets that were sent by the birth center and the Utah Department of Health. Both quantitative and qualitative survey items (ie, open-ended questions) were included in the questionnaire.

Setting and sample

A birth center in Salt Lake City that births approximately 300 babies per year and employs 4 midwives was chosen as the initial site for survey distribution. The birth center’s clinical staff distributed the surveys and consent letters to midwives and clients via mail in March 2016. However, although 100 surveys were distributed, fewer than 20 were returned, and most were incomplete. To increase the response rate, employees at the Utah Department of Health mailed out invitations and consent letters that included the online survey link to approximately 400 OOH birth parents and 100 midwives. Individuals were randomly chosen to participate from the Vital Records database, and to increase response rate, a $2 incentive was included with each survey invitation. In Utah, there are approximately 100 midwives registered with the Utah Department of Health. Although there were approximately 1100 OOH births in Utah in 2016, funding limitations precluded mailing survey invitations and consent letters to additional parents and midwives. A total of 113 parent surveys and 17 midwife surveys were completed; 33 surveys were returned unopened (potential participants had either moved or their address was no longer valid). The response rate was 31% for the parent group and 17% for the midwife group. The response rate for both paper-based and online surveys in this study was consistent with outcomes from other studies.¹⁹

Instruments

Two descriptive cross-sectional surveys were modified from previously published surveys in the medical literature. The 28-question survey for midwives was modified from a validated survey for midwives about knowledge, attitudes, and practices regarding newborn hearing screening²⁰ and took approximately 10 minutes to complete. The midwife survey also consisted of 5-point Likert-style questions, open-ended free-response questions, and multiple-choice questions that listed 3 or more options. Survey items for parents consisted of 42 questions from a validated survey about knowledge and attitudes toward NBS.²¹ The parent survey also consisted of 5-point Likert-style questions, open-ended free-response questions, multiple-choice questions with 3 or more options, and true/false questions that also included a “do not know” option. Both modified surveys were piloted and refined among genetic counselors, public health experts, and bioethicists for readability prior to data collection. The differences in the length of the knowledge surveys between the 2 groups were because of time constraints for healthcare providers, and as such, the midwife survey contained 6 knowledge questions, whereas the parent survey contained 13 knowledge questions. Likewise, the parent survey was longer than the midwife survey overall. Knowledge scores for midwives and parents were compared by taking the percentage of knowledge questions answered correctly and averaging the scores for each group separately.

Data analysis

Descriptive statistics were used to analyze the quantitative data. Individual knowledge items were summed to identify the number of correct answers for both the midwives and the parents. A content analysis was used to analyze the qualitative data.^22,23 Two independent coders applied open coding to the participants’ responses and all of the codes were generated by both reviewers from the participants’ own words.^24,25 For example, if a participant wrote “they were unaware about NBS,” then this was coded as “unaware about NBS.” The codes developed by the 2 independent reviewers were compared and no major discrepancies were identified.

The codes were then linked together to form categories based on degree of similarity. Only categories that were repeated most often by the participants were reported. This approach to qualitative descriptive research has been well supported in the literature and accurately captures self-reported participant experiences by using data and codes that closely reflect the actual statements of the participants.^26,27

RESULTS

Most parents (94%) participated in NBS; only 6 parents (5%) did not participate. One parent (1%) did not know whether his or her child had participated in NBS or not. Of those who did not participate, 4 declined NBS and 2 reported that NBS was not offered. Limited demographic data were collected from midwives to promote more anonymity and encourage participation. Of the midwives, only 1 participant self-reported working in the home setting with the remaining participants reporting working in hospitals and/or birth centers. Parental demographic questions (gender, age, ethnicity, and so forth) were not included in order to promote anonymity and encourage greater participation among the home births group. The average score for correct parental responses to knowledge questions about NBS was 62% (8/13 questions correct); midwives scored higher than parents (75% correct; 10/13 questions). Additional questions were asked of the parents and midwives about attitudes and experiences with NBS (see Tables 1 and 2).

Table 1.

Out-of-hospital-birth parents′ experiences with and attitudes toward newborn blood spot screening

Survey questions	Parents
I have previously heard about NBS	97% yes
I heard about NBS before this pregnancy	77% yes
I heard about NBS from my experience with previous children	68% yes
I heard about NBS from my midwife	67% yes
I heard about NBS from another healthcare provider	23% yes
I heard about NBS from a prenatal birthing class	17% yes
I heard about NBS from the Internet	17% yes
I heard about NBS from family or friends	11% yes
I heard about NBS from professional education	9% yes
I heard about NBS from newspapers	2% yes
I heard about NBS from magazines or television	1% yes
I know a great deal about NBS	42% mostly agree
	24% disagree
	25% not sure
	9% completely agree
How informed about NBS were you before your baby had NBS?	48% very informed
	36% somewhat informed
	9% a little informed
	5% not at all informed
	(2% did not answer)
I have enough information about NBS	54% mostly agree
	31% completely agree
	7% mostly disagree
	6% unsure
	(2% did not answer)
Do you agree that NBS should be mandatory?	55% no
	29% yes
	6% no opinion/unsure
	(10% did not answer)
Should a parent be able to refuse NBS for personal beliefs?	70% yes
	15% no
	15% no opinion/unsure
Should a parent be able to refuse NBS for religious beliefs?	72% yes
	13% no
	15% no opinion/unsure

Abbreviation: NBS, newborn blood spot screening.

Table 2.

Midwives′ demographics and experiences with and attitudes toward newborn blood spot screening

Survey questions	Midwives′ responses
Regularly receive NBS results	82% yes
Either “familiar” or “very familiar” with NBS	100% yes
I know what to do upon receiving an abnormal NBS result	88% agree
Basic midwifery training experiences prepared me to meet the needs of infants with NBS disorders	24% agree
On-the-job training prepared me to meet the needs of infants with NBS disorders	88% agree
Utah Department of Health training is a resource for NBS knowledge	71% agree
On-the-job training is a resource for NBS knowledge	76% agree
Professional journals, hospital Continuing Nursing Education (CNE) programs, andphysicians/OB-GYNs are resources for NBS knowledge	4% agree
It is important to have NBS done on all newborns	76% agree
Gender	100% female
Practice mainly in metropolitan area	89%
Practice mainly in small city	6%
Practice mainly in rural area	5%
Average age	51 y
Average length of midwifery practice	21 y
Average number of newborns birthed/year	96 newborns

Abbreviation: NBS, newborn blood spot screening.

Data about experiences and attitudes were captured from responses to Likert questions and open-ended questions. Overall, 63% of parents were “very supportive” and 26% were “moderately supportive” of the NBS program. One participant reported that she and her partner supported the NBS program because they “feel that it is good to screen for some of these diseases, especially those which might be prevented if detected during screening.” Similarly, another parent supported NBS because “the benefits . . . outweighed any of the negatives.” When asked whether NBS caused them to worry, 90% of parents responded “no” and 10% responded “yes.” For participants who responded “yes” to this question, 2 representative quotes included “[NBS] is not always accurate. It can show an abnormality when there is none and vice-versa,” and “These tests are usually not that helpful anyway—too many false positives.”

Despite the mostly positive parental attitudes toward the NBS program, many parents wrote that NBS should not be mandatory. Only 29% stated that NBS should be mandatory; 55% of parents said that they did not agree that NBS should be mandatory, and 16% were unsure. The most common reasons participants disagreed with mandating NBS included (a) traumatic experiences with NBS, (b) the potential inaccuracy of the results, and (c) concern over what would be done with the residual dried blood spots. For example, one parent wrote, “The pricking of the heel and who would receive the information, and causing unnecessary pain to [her] just born child!”

For participants who thought about or declined NBS, the most common reasons apart from those mentioned previously included (a) lack of information about NBS, (b) perceived government impingement, and (c) uncertainty about storage of and access to the data. For example, one participant wrote, “We do not agree with how the information collected during the screening is stored, and that others are allowed access to that information. We also do not agree with forced screening in any way.” Another parent wrote, “The child belongs to the parent, not the State.”

Similar questions were posed to the midwives in this study. When asked about the importance of NBS for all newborns, 24% of the midwives responded “somewhat important” or “unimportant,” while 76% responded “important” or “very important.” The following is one representative comment:

I believe midwives should be very supportive of the NBS. It is a part of protecting the newborn. Many of my clients would not have their babies screened if I did not encourage and educate them on the importance of this screening. Midwives should promote and encourage this testing for their clients’ newborns.

Midwives were also asked about their role in promoting NBS and/or follow-up after screening. Most of the responses were focused on the educational needs of clients and the prioritization of informed consent. For example, one midwife said, “As a health care provider my role is to inform and educate. Once clients understand the purpose of NBS, they usually opt to participate in NBS.” Another participant agreed: “I think we should support [NBS] and encourage patients to do it and follow up with any abnormal [result with] their pediatrician.” However, despite the stated support for NBS, midwives reported that it might conflict with their philosophy toward child birth. The following is one comment that captured this perspective:

I know the law says only parents of an organized religion whose teachings are opposed to it may refuse, but a hallmark of midwifery is that parents may refuse anything and their child’s healthcare. If a parent is truly informed, I don’t feel that right should be trampled on, even though I realize there will inevitably be some damaged babies as a result. It’s the age-old freedom vs. safety debate.

Midwives expressed various opinions about the amount of worry caused by NBS and the level of confidence they had in the accuracy of the screening results. Although there was broad support for NBS in general, about 25% of midwives agreed with the statement, “NBS causes excessive anxiety and/or concern among parents” (65% said that they did not believe that NBS causes excessive worry, and 10% were unsure). One midwife described a source of worry about NBS among her clients:

Some of my clients are concerned that the Federal Government is gathering DNA data on their child for alternative motives. Nothing we have explains what is done with the cards after the testing is done, or if the results of the tests and private information are shared with any agencies other than the one [doing the] testing. Also, the process of requesting that the cards be burned and completely destroyed, not just set under ultraviolet light, is difficult. The result is to waive the test rather than get it done.

In terms of confidence in the accuracy of NBS results, 53% of the midwife participants were “very confident,” whereas 35% of the midwives were “somewhat confident” and 12% were “unsure.” One midwife who chose “somewhat confident” cited “lots of false positives” as a reason for her lowered confidence.

There were several other obstacles to NBS participation reported by midwives. One issue was the cost of the NBS kit. One participant stated, “I think the kits are too expensive and that if the state is going to require the test be performed, then they should provide the kits free of charge.” Furthermore, some midwives also desired more training on the how to perform the heel-prick test and wanted to know which reimbursement codes to use for performing the screen.

When respondents prioritized their highest informational needs, replies included learning more about guidelines for informing families about positive screening results, early intervention options, useful contacts for more information, and education on the genetics of inherited disorders.

DISCUSSION

This was one of the few studies in the United States that assessed experiences with, knowledge of, and attitudes toward NBS on the part of both OOH birth parents and midwives. To date, most of the research literature has been focused on attitudes toward consent and informed decision making for hospital births.^28–30 The success of NBS depends on the entire population participating to ensure that all infants are screened, and that affected infants are identified and receive proper follow-up treatment. Therefore, collaboration between state health departments and midwives is critical to ensure that NBS is offered and properly conducted. The results of this study highlight not only high support for but also experiences and educational needs about NBS for OOH birth parents and these providers.

Parents who choose to give birth outside a hospital are a small minority of the population and tend to hold views about the overmedicalization of pregnancy and birth.³¹ However, the outcomes of this research are consistent with other research literature in that both parents and providers are highly supportive about NBS.^32,33 Midwives reported in this research that their roles were information givers and emphasized the importance of informed consent (ie, explaining risks/benefits of NBS). These data are similar to other studies on providers’ attitudes toward NBS to promote informed decision making about NBS.^34,35 One interesting finding that emerged from the study was the focus on the role of midwives in helping parents create the pregnancy and birth experience they want; thus, if a parent declines NBS, the midwife is expected to honor that parental decision. Newborn blood spot screening nonparticipation may be the result of factors other than lack of knowledge; however, some of the parental responses in this study are consistent with responses from in-hospital-birth parents described in other literature, in that the heel prick can be an emotionally traumatic experience for the parents.³² Parents may not necessarily decline NBS because of lack of education or based on personal values but to avoid causing pain to their newborn. Therefore, additional practices to minimize pain should be included in education delivered to providers by health departments.

Support for the NBS program by OOH birth parents (89%) was similar to what was found in previous studies on parental attitudes toward NBS programs in that most agreed that NBS should be conducted (86%⁵; although most participants did not agree with the mandatory nature of NBS). Some midwives also reported that they questioned the accuracy of the results and how the outcomes of NBS would be stored. Furthermore, there were some concerns over the storage and research use of residual dried blood spots. In Utah, the results from NBS (not the actual blood spots) are stored until the child is 22 years of age. This includes all demographic data, screen tests results, confirmatory test results, nursing notes, and other information that documents the child’s condition and status; this portion of the card is kept separately from the blood spots. After 22 years, all records are destroyed. After all necessary diagnostic testing has been completed, parents may also request the destruction of the dried blood spots sample within 90 days. Providing midwives with sufficient education to help answer these parental concerns in order to provide adequate and accurate information for parental decision making is critical for health departments. However, midwives must also communicate with health departments about these concerns and needs in order to promote collaborative partnerships.

Other barriers to NBS participation that were reported for both midwives and OOH birth parents were similar to other results from in-hospital births. First, more training is needed on how to accurately conduct NBS and how to address parental concerns about the pain of the heel prick. Obtaining sufficient blood during the heel prick without touching the heel to the filter paper has required additional training efforts from state health departments for providers.¹³ Second, both parents and midwives asked for more information about NBS and for this information to be delivered before the birth of the newborn. A recent randomized controlled trial that compared prenatal education with the current practice of in-hospital postnatal education found increased parental support for NBS and higher knowledge in the prenatal education group.²⁹ Third, providing background information on how NBS was developed to provide equal access to everyone in the population may help address parental concerns around government intrusion in healthcare. Both midwives and parents reported worries over the accuracy of the results, so providing more detailed information about how NBS is conducted, as well as rates of positives, false positives, and false negatives, may increase confidence in the approach (currently, these data are not given to healthcare providers). Finally, desire for more education and training on how NBS works within a system and how to manage follow-up for positive results was expressed in this study. More outreach efforts may be needed by both health departments and midwives about these educational needs and how they may be incorporated into training and education of midwives in nursing schools.

Limitations to the study include a small sample drawn from only 1 state. Another potential limitation is that those who participated in the survey might be fundamentally different from nonparticipants; for example, midwives and OOH birth parents who choose NBS might be more likely to participate in the study than those who opt out. This sample bias would lessen the external validity and generalizability of this study. A major strength of the research was the ability to study multiple factors and obtain both quantitative and qualitative data. Further studies will be useful for determining whether public health measures might be effective in increasing NBS participation and knowledge and therefore lowering NBS nonparticipation rates. These preliminary results can also help generate hypotheses, and the study can serve as a pilot for future research.

CONCLUSION

Additional studies are required to more clearly determine what barriers to NBS exist among OOH birth parents and how to address these concerns for midwives and their clients throughout the nation. Because there has yet been no study published in the literature that compares midwives’ and OOH birth parents’ experiences with NBS, this study provides a valuable framework that can be utilized in future studies on a larger, more diverse population. If NBS programs do not actively engage the growing midwifery and OOH birth communities, NBS participation rates are likely to decrease in these populations. Already, the research literature documents that NBS noncompletion rates and inaccurate clinical collection rates are higher in OOH births than in hospital births. Newborn blood spot screening nonparticipation—whether because of lack of knowledge, lack of resources, misinformation, or mistrust of the medical community—can lead to not identifying children with disorders tested for by NBS, which jeopardizes the national efficacy of population screening. Although each of these disorders is individually rare, their cumulative incidence is relatively high—estimated at being from 1 in 1500 to 1 in 5000 live births.³⁶ This study highlights the need to improve collaboration between midwives and state health departments, to tailor education and training resources for midwives and their OOH birth clients, and to open dialogue with parents to address their concerns about NBS in order to reduce mortality and morbidity from NBS-related disorders. In general, more research is needed to understand facilitators and barriers of collaboration with midwives and other healthcare professionals.¹⁴ State NBS programs must therefore make concerted efforts to understand their own OOH birth population’s educational, financial, and training needs. Because each state implements its NBS protocol differently, state-specific midwifery education efforts are needed to ensure that midwives are properly provided with the tools they require to inform their clients about NBS. If NBS programs are unsuccessful in ensuring NBS participation among OOH birth populations, the effectiveness of one of the most successful public health programs in our nation’s history will decline.

How might this affect nursing practice?

Out-of-hospital births are increasing, and additional educational efforts are needed to ensure that clinicians who serve this population have adequate education and the resources needed for their clients to make informed decisions about participation in NBS. Nursing is a collaborative partner and educator for midwifery. Parents who choose to give birth out of the hospital demonstrated limited knowledge about newborn screening and expressed the need to improve educational outreach efforts about the importance of newborn screening. Midwives are critical partners in the newborn screening system, and this research revealed high support for newborn screening, but more outreach and training by health departments and nursing schools is needed to ensure continued collaboration and accurate clinical collection of blood spots.