Background. In 2000, the American Academy of Pediatrics (AAP) Task Force on Newborn Screening published a blueprint for the future of newborn screening that included recommendations for information provided to parents about screening.
Objectives. To evaluate the completeness of educational material provided by newborn-screening programs and to measure the reading level and complexity of the material.
Methods. Telephone survey of newborn-screening programs (n = 51) followed by content analysis of educational material.
Results. All 51 programs responded (response rate: 100%); 47 of these programs made educational material available. None of the material included all elements recommended in the blueprint. Benefits of screening (98%) and how parents would be notified of results (87%) were included more often than the risks of screening (19%), possibility of a false-positive result (13%), importance of (34%) and how to respond to (28%) a positive result, and the storage and use of residual samples (11%). The median readability grade level was 10. Grade-level complexity of the material was not associated with completeness according to the AAP criteria.
Conclusions. Parent educational materials for newborn-screening programs do not meet the standard recommended by the AAP, and there are important variations between programs in the information provided to parents. Continuing research is needed to measure progress toward the goals outlined within the blueprint and to assess how these changes impact the care provided through newborn-screening programs.
Newborn screening has led to dramatic reductions in morbidity and mortality for a broad range of rare conditions through early detection and treatment. There is currently no federally mandated minimum set of conditions for inclusion in newborn-screening panels, and the operations of and resources for newborn-screening programs are determined at the state level. This results in wide variations in newborn-screening programs, such as how parents and physicians are informed about newborn-screening results and the methods and resources available for follow-up and treatment for children with abnormal screens.1
In 2000, the American Academy of Pediatrics (AAP) Task Force on Newborn Screening published a “blueprint” for the future of newborn screening to address concerns about variability among the newborn-screening programs and to develop a strategy to resolve ongoing and emerging challenges.2 This comprehensive report identified several key factors for successful screening programs. Some of these factors, such as expansion of public health infrastructure, establishing advisory committees, and implementing program surveillance and research, will take time to develop. Other factors, such as providing parents with comprehensive educational material about newborn screening, could be accomplished sooner through modification of existing materials.3
The AAP blueprint listed 7 recommendations for what should be presented in parent educational material: (1) the benefits of screening; (2) the potential risks of the screening test; (3) how parents will be informed of screening results; (4) the possibility of a false-positive test result; (5) the importance of responding to a positive test result; (6) how to respond to a positive test result; and (7) the screening program's policy for sample storage and use of stored samples. In addition, the task force stated the importance of ensuring that the material is written at an appropriate literacy level.
We are unaware of any previous study that has focused on the content and complexity of educational material produced by screening programs for parents. The objectives of this study were to evaluate the completeness of educational material provided by newborn-screening programs according to expert panel recommendations and to measure the reading level and complexity of the material.
Collection of Educational Material
In January 2004, the coordinators of each US newborn-screening program (n = 51) were contacted by telephone and asked to provide any standardized program materials used for educating new or expectant parents about newborn screening. We limited data collection to materials written in English and focused on the newborn-screening program; we excluded material on particular diseases only (eg, brochure on phenylketonuria). This study was approved by the University of Michigan Medical School Institutional Review Board.
A standardized data-collection instrument for content evaluation was developed and pilot tested for ease of use. Each program's educational materials were evaluated to determine if they included full, partial, or no coverage of information related to the 7 components recommended by the AAP Task Force on Newborn Screening (benefits of screening, potential risks, how parents will be informed of results, the possibility of a false-positive result, the importance of responding to a positive result, how to respond to a positive test result, and the screening program's policy for sample storage and use). For the risks associated with screening, we looked for statements regarding the direct risk of screening (eg, pain) and considered the harm of false-positive results separately along with statements about the possibility of false-positive results. In this evaluation we also considered factors recommended for inclusion by other expert panels: descriptions of all conditions being screened, the likelihood of the conditions occurring, and the process and implications of screening refusal.4–6 The presence of information regarding costs associated with screening was also considered, because the majority of programs charge a fee for newborn-screening tests.1 During the abstraction process, representative quotations were recorded to illustrate the quantitative findings. One investigator (K.E.F.) abstracted all data. To ensure reliability of the data abstraction, a 10% sample of the material was independently abstracted by another investigator (A.R.K.). Interrater reliability was assessed with the κ statistic.
Readability of the material was measured by using the SMOG grade formula, a previously validated measure of the complexity of patient-education material.7 The formula involves tallying all words with ≥3 syllables in 30 selected sentences (3 groups of 10 consecutive sentences from the beginning, middle, and end of the document). Then, the square root of that total is obtained and its integer calculated. Finally, the number 3 is added to the integer to obtain the grade level of the document. Length and format of the educational material (eg, single-page, bifold, or trifold brochure, booklet), a secondary measure of material complexity, was noted for each program's materials.
Univariate and selected bivariate frequencies were used to summarize the findings. In addition to frequency counts for each component included in the study, we also calculated the total number of the 7 AAP Task Force on Newborn Screening recommendations included in the materials. Readability grade level was categorized into ≤9th, 10th to 11th, and ≥12th grade. The χ2 test statistic was used to measure the strength of association in the bivariate analyses; P < .05 was considered statistically significant. Stata 8.2 (Stata Corp, College Station, TX) was used for all statistical analyses.
Availability of Educational Material
We contacted all 51 newborn-screening coordinators (response rate: 100%). All but 1 program reported that standardized educational material is available for parents about newborn screening (98%). However, 3 of the programs did not make the material available because they were in the process of revising or printing their materials. Thus, subsequent results are based on the materials available from 47 newborn-screening programs. There was high interrater reliability (κ = 0.94) in the coding for the 10% subsample of reabstracted materials.
Material Inclusion of AAP-Recommended Items
None of the program materials contain >5 of the 7 elements recommended by the AAP. Overall, 5 materials (11%) have 5 items, 22 (47%) have 3 or 4 items, and 20 (42%) have ≤2 of the elements (Table 1). Additional detail regarding each AAP-recommended item follows.
Benefits of Screening
Nearly all program materials (n = 46) contain a statement about the benefit of screening, and we found very little variation in the content of these messages. Materials primarily cite the benefit of early detection and treatment (eg, “If identified early with newborn screening testing, some of these disorders can be treated before they cause serious health problems”).
Direct Risks of Screening
Only 9 programs (19%) discuss the direct risks associated with screening. Of those, 5 present the risk of pain or infection. The other 4 materials cover risks of screening by stating that there is no significant risk to the infant. For example, 1 parent brochure states: “It makes a lot of sense to have the test done because there are no side effects and it's perfectly safe.”
How Parents Will Be Informed of Screening Results
Materials from 41 programs (87%) communicate how parents would be notified, but there was variation in the emphasis on parental responsibility. Of the 41 programs, 15 mention that parents would be notified if there were a problem but encourage families to ask their child's physician about the results. A similar number (n = 14) let parents know that they would be notified only if a problem was suspected, whereas 10 advise parents to ask their child's health care provider for results. Only 2 state that families would be notified regardless of the result.
Possibility and Potential Harm of False-Positive Result
Only 6 of the materials (13%) explicitly mention that false-positive results are possible (eg, “The screening tests are very sensitive so truly affected infants will not be missed, and thus some nonaffected infants will have falsely positive results”). In addition, only 3 mention any risks associated with false-positive results (eg, stress, inconvenience).
Although not specifically mentioning false-positive results, 33 materials (70%) address the potential need for retesting or the associated uncertainty about results. Materials discuss the need for retesting by offering reasons why an additional sample might be requested (n = 17), such as the initial sample being collected too early or containing an inadequate amount of blood for testing. A similar number of materials (n = 16) address parental uncertainty by stating that the need for retesting “does not necessarily mean that your infant has one of the conditions.”
Importance of Responding to a Positive Test Result
Materials from 16 programs (34%) provide a specific reason (eg, “timely treatment if a problem is detected”) regarding the importance of following up after an abnormal result. However, 29 programs (62%) do not provide a rationale but simply state that responding quickly is important.
How to Respond to a Positive Test Result
Materials from 34 programs (72%) do not tell parents what they should do in the event of an abnormal test result. However, 13 of them (28%) contain specific information (eg, “If you are asked to have your infant retested, please bring your infant back to the birth facility or your infant's doctor as soon as possible”).
Storage and Use of Stored Samples
Information regarding the policies for storage and use of residual samples are mentioned in the materials from 5 programs. Most of them mention that samples are stored securely (n = 3); the length of time programs store specimens ranged from 90 days to 21.5 years. Materials state that stored samples may be used for research (n = 3) or are not used for any other purpose (n = 2) without the request or prior written consent of parents.
Other Recommended Content
Description of the Conditions on the Screening Panel
All but 1 of the materials provide information about the conditions detected with the newborn-screening test; 9 materials (19%) provide a list or examples of conditions, whereas 37 (79%) offer a full description of each condition. In describing each condition, materials typically present details about prevalence, disease course, and treatment.
Likelihood of Having the Conditions
Materials from 22 programs (47%) present prevalence data to communicate the specific risk of each disorder. A qualitative statement (eg, “rare”) about the risk of the various conditions on the screening panel is presented in 13 materials (28%). There is no quantitative or qualitative information on the risk of the conditions in 12 of the materials (25%).
All of the materials (n = 47) discuss outcomes if the conditions are not treated. Of these, 8 provide a general statement (“mental retardation, growth failure and even death”). The others present outcomes specific for individual conditions. Similarly, nearly all (n = 43) discuss the types of treatments, either generally (“special diets and medicines”) or specific to individual conditions (“treatment with biotin supplementation”).
Process for Refusing Newborn Screening
Of the 47 programs that submitted materials for analysis, 5 do not permit refusal of screening for any reason and therefore were excluded from this analysis. Among states that permit refusal of newborn screening, 23 materials (55%) mention a parent's right to refuse screening. Among those that include the option of refusing, few (n = 6) also discuss the implications of doing so (eg, “Your hospital, doctor and the clinic staff are not responsible if your infant develops problems from these disorders”).
Cost of Screening
Only 11 materials (23%) mention the costs associated with screening. Of those, 2 indicate that testing is free, 4 mention that there is a charge for testing but do not specify the amount, 3 specify the cost of screening (ranging from $47 to $54), and 2 do not mention whether there are costs associated with the required screening tests but indicate that there is no extra cost for expanded or optional newborn-screening tests. In addition, approximately half of the materials (n = 6) that mention costs indicate that the fees are typically covered by third-party payers, and 4 of the materials provide information about who to contact if an individual has no insurance and is unable to pay. The potential for incurring other ancillary charges associated with newborn screening (eg, “Your health care provider may charge a fee to collect the specimen”) is mentioned by 2 of the program materials.
The median SMOG readability grade level of materials was 10. Overall, 12 of the materials (25%) were written at the 7th- to 9th-grade level, 28 (60%) were written at the 10th- to 11th-grade level, and 7 (15%) were written at the ≥12th-grade level. Materials that describe all conditions on the screening panel (n = 37) were more likely to be of greater complexity compared with those that only list conditions or give examples (n = 9) of conditions (P = .03). For materials that list conditions, 67% were written at the 7th- to 9th-grade level, and 22% were at the 10th- to 11th-grade level. For materials with a full description of conditions, 16% were written at the 7th- to 9th-grade, and 68% were at the 10th- to 11th-grade level.
Materials from 27 programs (58%) present newborn-screening information in a bifold or trifold format; 18 (38%) use a larger booklet, and 2 (4%) present the information in a single page. Document size was not associated with readability scores (P = .12).
The complexity of the material, measured as either readability or size, was not associated with completeness according to the AAP criteria (P = .12 and .98, respectively).
Most, but not all, newborn-screening programs have educational material available for families. None of this material meets all the criteria advocated by the AAP or other expert panels. In general, most materials highlight the benefits of screening and compulsory nature of testing in similar ways.
Although most materials present information on how parents will be informed about the results of screening, we found important differences in the expected role of parents in learning these results, ranging from families being notified regardless of the result to parental inquiry being emphasized. Some of these differences likely arise from programmatic differences.8
Only 13 of the programs informed parents about what steps should be taken in the event of a positive screening result. Typically, this was limited to a description of the need for retesting. Because programs make a concerted effort to quickly identify and track children with positive results and because the child's primary care physician is often responsible for notifying parents of the result, it could be argued that informing parents about the necessary steps after a positive test in the educational material provided at the time of screening is unnecessary. However, providing this information might decrease parental anxiety by emphasizing that an abnormal screen does not confirm disease and might enhance timely follow-up by underscoring the need for quick follow-up testing.
Most materials were written at the ≥10th-grade level. According to the National Adult Literacy Survey, almost half of adults in the United States have limited or extremely limited reading skills, and other studies demonstrate that more than one third of English-speaking patients have difficulty reading at the 10th-grade level.9–11 Additionally, the National Work Group on Literacy and Health recommends that material be written at or below the sixth-grade level to increase the likelihood that patient-education materials be read and understood.12 Providing parents with comprehensive educational material about newborn screening at a lower reading grade level might be challenging; however, among the material we reviewed, those that were more complete (ie, covered more of the AAP-recommended elements) did not have a higher reading level than those that were less complete.
As long as newborn-screening programs are managed at the state level, program variations will persist. The impact of the variations in the information provided by these programs is unclear; the association between messages in educational materials and compliance with retesting or parental anxiety over false-positive results has not been explored. In addition, we are unaware of any studies that have assessed what information parents need about newborn screening and how to provide this education most effectively. Similarly, no studies have addressed other sources of screening information for parents, such as conversations with health care providers.
Our study did not examine the context in which these written materials are presented to parents. A survey found that newborn-screening programs typically do not provide educational materials directly to parents but rather distribute them to hospitals and primary care providers, who are expected to share them with parents.1 Although there have been efforts to encourage information sharing with parents during the prenatal period, much of the responsibility for educating parents falls to individual hospitals and birthing centers after delivery. The postpartum period is a challenging time for new parents, and it is unclear how much information they can retain. Therefore, the Task Force on Newborn Screening recommended that prospective parents receive information about newborn screening during the prenatal period, preferably during a routine third-trimester prenatal care visit.2 A survey in 2000 found that some newborn-screening programs (13 of 51) had policies to encourage or require parent notification about newborn screening during the prenatal period.8 Despite evidence that policies regarding the timing of parental education exist, work is still needed to document if and when parents actually receive newborn-screening materials.
Our study focused on the written material presented to parents on newborn screening. Therefore, elements lacking in the written materials may be presented verbally to parents during encounters with prenatal and pediatric care providers. Additional work should be done to assess provider knowledge of newborn-screening programs and current patient-education practices. By ensuring provider coverage of these issues or updating program materials to include all AAP Task Force on Newborn Screening–recommended elements, parents can be provided with the information needed to understand the purpose of newborn screening and the meaning and implications of a positive result.
Parent education represents only one component of the AAP Task Force on Newborn Screening blueprint; the failure of educational material to meet the blueprint recommendations does not necessarily imply that there has not been important programmatic change to address other issues. This study, however, emphasizes the need to measure progress toward the goals outlined within the blueprint and to assess how these changes impact the care provided through newborn-screening programs.
- Accepted September 3, 2004.
- Address correspondence to Alex R. Kemper, MD, MPH, MS, Child Health Evaluation and Research Unit, Division of General Pediatrics, University of Michigan, 6E08 300 N Ingalls Building, Ann Arbor, MI 48109-0456. E-mail:
No conflict of interest declared.
- ↵United States General Accounting Office. Newborn Screening: Characteristics of State Programs. Report No. GAO-03-449. Washington, DC; 2003. Available at: www.gao.gov/new.items/d03449.pdf. Accessed April 19, 2004
- ↵Task Force on Newborn Screening. Serving the family from birth to the medical home. A report from the Newborn Screening Task Force convened in Washington DC, May 10–11, 1999. Pediatrics.2000;106(2 pt 2) :383– 427
- ↵President's Commission for the Study of Ethical Problems in Medicine and Biomedical and Behavioral Research. Screening and Counseling for Genetic Conditions: The Ethical, Social, and Legal Implications of Genetic Screening, Counseling, and Educational Programs. Washington, DC: US Government Printing Office; 1983
- National Institutes of Health-Department of Energy Working Group on Ethical, Legal, and Social Implications of Human Genome Research. Promoting safe and effective genetic testing in the United States: final report of the Task Force on Genetic Testing. 1997. Available at: www.genome.gov/10001733. Accessed April 19, 2004
- ↵US National Research Council Committee for the Study of Inborn Errors of Metabolism. Genetic Screening: Programs, Principles, and Research. Washington, DC: National Academy of Sciences; 1975
- ↵Kim S, Lloyd-Puryear MA, Tonniges TF. Examination of the communication practices between state newborn screening programs and the medical home. Pediatrics.2003;111(2) . Available at: www.pediatrics.org/cgi/content/full/111/2/e120
- ↵Kirsch IS, Jungeblut A, Jenkins L, Kolstad A. Adult Literacy in America: A First Look at the Findings of the National Adult Literacy Survey. Washington, DC: National Center for Education Statistics; 2002. Available at: http://nces.ed.gov/pubs93/93275.pdf. Accessed April 19, 2004
- Copyright © 2005 by the American Academy of Pediatrics