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Preterm Birth: Causes, Consequences, and Prevention (2007)

Chapter: Appendix D A Systematic Review of Costs Associated with Preterm Birth

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Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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D
A Systematic Review of Costs Associated with Preterm Birth

John A. F. Zupancic1

The costs of prematurity have been the subject of close scrutiny for more than 2 decades. During that time, there have been dramatic advances in therapeutic interventions, improvements in mortality, and significant inflation in medical care costs. Although the neonatal intensive care phase is most prominent in discussions of these costs, the sequelae of prematurity constitute a chronic disease with significant long-term implications. This appendix has two parts: the first examines the literature on costs associated with the initial hospitalization for prematurity, whereas the second examines longer-term economic factors.

To evaluate the quality and applicability of cost-of-illness studies, it is important to have agreed-upon definitions of certain technical terms. There are, for example, several different categories of cost that might be important to stakeholders. Direct medical costs arise from resources directly consumed by patients. In the context of this review of costs of initial hospitalization, these include costs for the hospital bed and support services (“accommodation”), ancillary services (pharmacy, radiology, laboratory, and respiratory care services), and professional fees. In contrast to these direct costs, overhead costs are those expenses that are incurred in running the institution but that are not directly attributable to a particular patient.

1

John A. F. Zupancic MD, ScD, Department of Pediatrics, Harvard Medical School, Boston, Massachusetts.

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

These include overhead expenditures for housekeeping, administration, and the physical plant. Costs to patients include nonmedical direct costs (such as the cost of transportation to the hospital, meals, and child care for other siblings). Finally, there are costs related to lost productivity, also referred to as indirect costs. In the short run, these are the lost contributions of labor by the parents; in the long run, they also include reduced work alternatives for the patient.

The particular choice of which costs to include is dependent on the perspective of the stakeholder who will be using the information. For example, hospital administrators will be most interested in the direct medical costs that affect hospital financial operations and less interested in wage losses by parents. However, in valuing the financial burden of prematurity to society, it is vitally important to understand the full extent of costs, regardless of the individuals to whom they accrue. In the absence of such information, policy makers may make decisions that appear to be fiscally desirable but in fact simply shift costs to another, unmeasured facet of care.

Studies that measure costs on their own without reference to the effectiveness of an intervention are known as “cost-of-illness studies.” When costs are combined into a single metric with a measure of effectiveness, such as life years saved, the study is referred to as a “cost-effectiveness analysis.” When patient preferences for particular outcomes are used as the measure of effectiveness, as they are in quality-adjusted life years, the study is a known as a “cost-utility analysis.” Both cost-effectiveness and cost-utility analyses express costs and effects as a ratio measure. In contrast, cost-benefit analyses measure effects in monetary terms and subtract this value from the cost of the intervention to determine value for money.

METHODS

Population Description

Although much attention has been focused on infants at the borderline of viability, the large volume of children at higher gestational ages may drive the overall financial impact of prematurity. Therefore, broad inclusion criteria were maintained for the review and encompass children in the following categories: (a) premature, defined as birth at less than 37 weeks of gestational age, and (b) low birth weight, defined as a birth weight of less than 2,500 grams.

Although the assessment of efficiency is a critical component of technology assessment, the focus of this appendix is on the absolute financial burden of prematurity and low birth weight rather than on the value for money of treatment modalities. Therefore, only costing studies are included and cost-effectiveness, cost-utility, and cost-benefit studies are excluded,

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

except when the population measurement of pertinent costs was a central component. In some cases cost descriptions of cohorts alongside trials of efficacy of therapeutic interventions are included, but these are restricted to control groups to optimize external validity.

Two factors have significant impacts on the generalizability of the findings from economic studies. First, the therapies offered in the field of neonatal intensive care and their resulting outcomes have changed substantially in the past 2 decades. In the late 1980s, there was a dramatic decrease in the rate of mortality because of prematurity with the introduction of surfactant replacement therapy, a therapy subsequently shown repeatedly to have important cost implications. Because the underlying structure of care has changed, this problem is difficult to overcome. For the review of short-run costs, therefore only studies reporting on cohorts after 1990 are included to optimize the external validity of the results and avoid bias against lower birth weight in incremental cost comparisons. Since follow-up care of infants following discharge has changed to a lesser degree, reports of long-run costs are included if the cohort was born after 1980. In a few cases, cohorts were assembled with patients from before and after 1980; these studies are included if >50 percent of the cohort met the inclusion date criterion.

Similarly, substantial effort has been made to assess the costs of neonatal intensive care in other countries. In some cases, as in the United Kingdom, Europe, and Canada, health care delivery and financing may have certain similarities that allow extrapolation of the results to the U.S. population. However, studies from middle-income and developing countries were excluded on the grounds that both outcomes and health care delivery cost structures are too different to interpret the applicability of the results in the U.S. context.

Data Sources and Search Strategies

The following databases were searched for candidate articles: Medline (1990 to 2005), electronic abstracts from the annual meetings of the Society for Pediatric Research (1998 to 2005), Econlit (1990 to 2005), and the Proquest dissertation and theses full-text index (1990 to 2005). The Science citation index was searched for articles citing any of the first authors of included studies. The bibliographies of other systematic or informal reviews were scanned (17). The websites of organizations known to be involved in similar research were also scanned; these included the websites of the March of Dimes, the Alan Guttmacher Institute, and the Centers for Disease Control and Prevention. The search strategies were intentionally broad to ensure optimal sensitivity. The intersection of the exploded MeSH headings “infant, newborn” and “costs and cost analysis” was used for Medline

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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searches. For other databases, the keywords “econom*” OR “cost*” AND “infant” were used. The titles and abstracts of the articles retrieved with these strategies were manually screened to determine whether they met the inclusion criteria described above.

Cost Projections

Cost estimates were inflated to 2005 constant dollars by using both the medical component of the consumer price index (8) and the hospital producer price index (9). Currencies were converted to U.S. dollars by using purchasing power parities (10). Per-patient estimates of cost were projected to the U.S. birth cohort by multiplying by the number of infants in a given gestational age or birth weight category, according to the 2002 natality file of the National Center for Vital Statistics (11). State-level population cost estimates were projected to the U.S. birth cohort by dividing by the ratio of the state’s birth cohort to the total number of births nationally for 2002 (the most recent year for which such statistics were available).

YIELD OF SEARCH STRATEGY

The manual screening of titles and abstracts from the initial literature searches yielded 170 articles. Of these, the majority were eliminated because they were review articles or economic evaluations of interventional studies, were based on data from middle-income or developing countries, were studies at the neonatal intensive care unit level not stratified by birth weight or gestational age, or had cohort dates prior to 1990 for short-run cost reports or 1980 for long-run cost reports. Descriptions and methodological assessment of the short-run (1227) and the long-run (15, 27–41) cost studies retrieved are shown in Tables D-1 and D-2, respectively.

METHODOLOGICAL LIMITATIONS OF STUDIES RETRIEVED

Studies of Initial Hospitalization

The methodological quality of the short-run cost studies since 1990 was variable. Only five of the reports involved the collection of data alongside prospective research projects (12, 16, 23, 24, 27), whereas the remainder were retrospective analyses of administrative and clinical data collected for other purposes. In some cases, particularly those analyses involving statelevel data in California, there was a preexisting process for checking the integrity of data through reabstraction (18, 25). However, the accuracy of the smaller secondary data sets was not reported in the publications.

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

TABLE D-1A Studies of Costs During Initial Hospitalization

Paper

Date of Cohort

Location of Cohort

Type of Cohort

Sample Size

Currency

Currency Date

Adams (13)

1996

US

Convenience sample

Total: 12,125

Normal preterm: 456

Extreme preterm: 513

Normal full term: 9,179

USD

Not specified

Brazier (14)

Not specified

UK

Hospital-specific

38

UK pounds

Not specified

Chollet (15)

1989-1991

US

Convenience sample

Total: 58,904

Normal preterm: 946

Extreme preterm: 986

Normal full term: 44,041

USD

Not specified

Doyle (16)

1997

Australia

Geographic (state)

233

Australian dollar

1997

Giacoia (17)

1983-1984

US

Hospital-specific

167

USD

Not specified

Gilbert (18)

1996

California

Geographic (state)

25-38 weeks: 147,224

500-3000 grams: 458,366

USD

Not specified

Kilpatrick (19)

1990-1994

California

Hospital-specific

138

USD

Not specified

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Luke (20)

1991-1992

Illinois

Hospital-specific

Twins: 111

Singletons: 106

USD

Not specified

Marbella (21)

1989-1994

Wisconsin

Geographic (state)

Premature: 26,668

Full term: 368,955

USD

Not specified

McLoughlin (22)

Not specified

UK

Hospital-specific

109

UK pounds

Not specified

Rogowski (23)

1993-1994

US

25self-selected hospitals

3,288

USD

1994

Rogowski (24)

1997-1998

US

29 hospitals in QI collaborative

6,797

USD

1998

Schmitt (25)

2000

California

Geographic (state)

518,697

USD

2003

St John (26)

1989-1992

Alabama

Hospital-specific

958

USD

Not specified

Tommiska (27)

1996-1997

Finland

Geographic (regional hospital)

<1000 g: 105

Euro

1997

Victorian Infant Collaborative Study Group (12)

1991-1992

Australia

Geographic (state)

429

Australian dollar

1992

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

TABLE D-1B Studies of Costs During Initial Hospitalization

Paper

Gestational Age

Birth Weight

Costs Included

Cost vs Charges

Data Sources

Uncertainty

Adams (13)

Not specified (premature and extreme preterm defined by ICD9)

Not specified

Hospital; professional

Paid claims

Claims database

Not specified

Brazier (14)

Not specified

Not Specified

Travel

Costs

Parent interview

Not specified

Chollet (15)

All

All

Hospital; professional

Billed charges

Claims database

Not specified

Doyle (16)

Not specified

500-999 grams

Hospital

Cost(ventilator and non-ventilator days, inflated from 1987 values)

Prospective research database

Sensitivity analysis

Giacoia (17)

Not specified

All

Travel

Costs

Parent interview

Statistical

Gilbert (18)

25-38 weeks

500-3000 grams

Hospital

CCR (hospital-specific)

State-level linked vital statistics and discharge records

Not specified

Kilpatrick (19)

24-26 weeks

Not specified

Hospital

CCR (hospital-specific)

Hospital chart review and billing database

Not specified

Luke (20)

All

All

Hospital

Not specified

Hospital bills; hospital chart

Statistical

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Marbella (21)

Not specified (premature defined as all prematurity-related DRG)

All

Hospital

Charges

State-level linked vital statistics and discharge records

Not specified

McLoughlin (22)

22-37

<2500 g

Travel

Costs

Parent interview

Not specified

Rogowski (23)

Not specified

501-1,500 grams

Hospital

CCR (department-specific)

Hospital financial data

Not specified

Rogowski (24)

Not specified

501-1,500

Hospital

CCR (department-specific)

Hospital financial data

Not specified

Schmitt (25)

All

All

Hospital

CCR (hospital-specific)

State-level linked vital statistics and discharge records

Statistical

St John (26)

24-32 vs 33-42

Not specified

Hospital; professionl

CCR (hospital-specific)

Hospital chart review and billing database

Not specified

Tommiska (27)

≥22weeks

<1000 grams

Hospital; nonmedical direct; productivity losses

Cost (derivation not specified)

Hospital cost database and national ELBW registry; familyquestionnaire at 2 years

Statistical and sensitivity analysis

Victorian Infant Collaborative Study Group (12)

Not specified

500-999 grams

Hospital

Cost (ventilator and nonventilator days, inflated from 1987 values)

Prospective research database

Sensitivity analysis

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

TABLE D-2A Studies of Costs Following Discharge from Initial Hospitalization

Paper

Date of Cohort

Location of Cohort

Type of Cohort

Sample Size

Currency

Currency Date

Broyles (28)

1988-1996

Texas

Hospital-specific

388 (control group of RCT)

USD

1997

Chaikand (29)

1987-1988

US

National

6,788

USD

1989-1990

Chollet (15)

1989-1991

US

Convenience sample

Total: 58,904

Normal preterm: 946

Extreme preterm: 986

Normal full term: 44,041

USD

Not specified

Gennaro (30)

1990-1994

US

Hospital-specific

224

USD

Not specified

Lewit (31)

1988

US

National

35,000

USD

1988

McCormick (32)

1983-1984

Pennsylvania

Hospital-specific

VLBW: 32

Controls: 34

USD

Notspecified

Medstat (41)

2001

US

Convenience sample

Total: 28,958

All preterm: 3,214

Normal full term: 15,795

USD

2004

Petrou (33)

1970-1993

UK

Geographic (regional)

>37weeks: 226,120

32-36 weeks: 11,728

28-31 weeks: 1,346

<28weeks: 500

UK Pounds

1998-1999

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Petrou (34)

1978-1988

UK

Geographic (regional)

>37weeks: 90,236

32-36 weeks: 4,485

28-31 weeks: 596

<28weeks: 241

UK Pounds

1998-1999

Pharaoh (35)

1979-1981

UK

Geographic (regional)

109

UK Pounds

1984

Rogowski (36)

1986-1987

California

Geographic (state)

Inception: 887

Survived: 591

USD

1987

Rolnick (37)

1993, 1995

Minnesota

Hospital-specific (single health plan)

2500-4499: 1203=1500-2499: 38

USD

Notspecified

Roth (38)

1990-1991

Florida

Geographic (state)

120,533

USD

2001

Stevenson (39)

1980-1981

UK

Geographic (regional)

Total: 641

<1,000: 20

1,000-1,500: 153

1,501-2,000: 468

UK Pounds

1979

Stevenson (40)

1980-1981

UK

Geographic (regional)

Total: 52

<1,000: 9

1,000-1,500: 25

1,501-2,000: 18

UK Pounds

1979

Tommiska (27)

1996-1997

Finland

Geographic (regional hospital)

<1000 g: 105

>36weeks: 75

Euro

1997

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

TABLE D-2B Studies of Costs Following Discharge from Initial Hospitalization

Paper

Gestational Age

Birth Weight

Costs Included

Cost vs Charges

Data Sources

Uncertainty

Discounting

Broyles (28)

Not specified

<1000 or <1,500 and ventilated except drugs

Hospital; outpatient

CCR (department-specific)

Prospective research database

Statistical

3%

Chaikand (29)

Not specified

<2,500

Education

Costs

NCHS Child Health Supplement to National Health Interview Survey

Statistical

2%

Chollet (15)

All

All

Hospital; professional

Billed charges

Claims database

Not specified

Not specified

Gennaro (30)

Not specified

<2,500

Wages; transportation; unreimbursed med

Costs

Interviews

Statistical

Not specified

Lewit (31)

All

All

Hospital; education

Charges

National Medical Expenditure Survey

Not specified

Not specified

McCormick (32)

Not specified

<1,500

Hospital; outpatient MD; transportation; childcare

Charges

Diary; interviews

Statistical

Not specified

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Medstat (41)

Not specified

All

Hospital; outpatient medical; drugs

Paid claims

Marketscan proprietary database of 100 payers

Not specified

Not specified

Petrou (33)

All

Not Specified

Hospital

Costs

Linked vital statistics and National Health Service records; NHS financial returns

Statistical

Not specified

Petrou (34)

All

Not Specified

Hospital

Costs

Linked vital statistics and National Health Service records; NHS financial returns

Statistical

Not specified

Pharaoh (35)

Not specified

<1,500

Hospital; outpatient medical

Costs

Prospective research database

Not specified

5%

Rogowski (36)

Notspecified

<1,500

Hospital; outpatient MD except drugs

Inpatient: CCR (department-specific)

Outpatient: charges

Medicaid claims data

Not specified

Not specified

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Paper

Gestational Age

Birth Weight

Costs Included

Cost vs Charges

Data Sources

Uncertainty

Discounting

Rolnick (37)

Not specified

>1,500

Hospital; Outpatient MD except drugs

Charges

Claims database

Statistical

Not specified

Roth (38)

Not specified

All

Education

Costs

Linked birth and school records

Statistical

Not specified

Stevenson (39)

Not specified

<2,000

Hospital; outpatient MD; drugs

Costs

Medical records; MD interview

Not specified

6%

Stevenson (40)

Not specified

<2,000

Hospital; outpatient MD; drugs

Costs

Medical records; MD interview; department of education

Not specified

6%

Tommiska (27)

≥22weeks

<1000 grams

Hospital; nonmedical direct; productivity losses

Cost (derivation not specified)

Hospital cost database and national ELBW registry; family questionnaire at 2 years

Statistical and sensitivity analysis

Not specified

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

The inconsistent level of regionalization in neonatal care delivery makes the nature of the cohort critically important for cost analyses of prematu-rity. Six of the reports (12, 16, 18, 21, 25, 27) involved cohorts that were geographically based at the state or regional level. Others used claims data from nationally representative insurance organizations (13, 15) or data from a large number of self-selected hospitals in a neonatal quality improvement network (23, 24). Although the latter data do not guarantee freedom from selection bias, the large number of patients and hospitals involved makes such bias less likely than the likelihood of selection bias with the two small single-center studies (20, 26). In most cases, the inclusion criteria were adequately described; however, three studies defined gestational-age exclusively by diagnosis-related group (DRG) codes (13, 15, 21) rather than by direct observation of patient information. Because there is some discretion in the use of some DRG codes (specifically, “normal preterm” and “preterm with problems”), generalization of the findings to a larger population is more difficult.

Because of the very low incidence of extreme prematurity and the marked variability and skew of cost data, it is also important that studies have adequate sample sizes. As these studies were targeted toward the estimation of costs rather than inference, it is difficult to define an appropriate sample size a priori. The U.S. state-level geographic studies (18, 21, 25), insurance-claims studies (13, 15), and neonatal-network studies (23, 24) all based cost estimates on at least 1,000 premature infants. In contrast, the hospital-specific studies (19, 20, 26) and non-U.S. studies (12, 16, 27) examined as few as 105 premature infants, a sample size that is unlikely to yield useful confidence intervals, especially when the data are projected to national levels.

Earlier cost-of-illness studies in neonatalogy often relied on hospital charges, as these were easily available in administrative databases. However, the use of charges as a measure of economic cost may distort cost estimates because charges reflect a hospital’s decisions regarding markups and internal cost transfers. Information on actual costs may be derived from proprietary cost accounting systems or from conversion factors known as cost-to-charge ratios, which are mandated for the reporting of cost information to federal agencies. Two studies in the current review relied only on billed charges (15, 21), one relied on paid insurance claims (13), two relied on older costing studies corrected for inflation (12, 16), and two referred to costs but did not specify the means of derivation (20, 27). The remaining six studies used Medicare cost to charge ratios and are likely to be more comparable to each other.

Given the issues of variance in cost data and the number of comparisons made between groups of infants in the costing studies, it is particularly surprising that they relied almost entirely on point estimates without any

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

quantification of statistical or parameter uncertainty. Statistical uncertainty was reported in only three studies (20, 25, 27), and sensitivity analyses were performed in three studies (12, 16, 27).

Because almost any medical care involves a net expenditure of resources, it is important that the costs of prematurity be viewed as incremental on the medical costs for neonatal care had the premature infant been delivered later in gestation. Five of the studies did not include a cohort of infants delivered at full term (12, 16, 19, 23, 24).

In regionalized systems of neonatal care, infants are often transferred to other hospitals for higher levels of care or to less intensive care settings for a period of further maturation following the resolution of acute cardiorespiratory instability. Failure to include posttransfer costs would lead to underestimates of the cost of care, whereas failure to include transferred infants would affect the external validity of the estimates and possibly overestimate treatment costs, as retrotransferred infants are more stable. Three studies explicitly noted that transferred infants were excluded (1921), two explicitly included them (15, 25), and the remainder did not specify the inclusion or exclusion of transferred infants.

Several authors also excluded nonsurviving infants from cost estimates (18, 21). One article that did measure the costs for nonsurviving infants observed that they represented 9 percent of total costs (27). Because nonsurviving infants have shorter lengths of stay, they are likely to be less costly (24, 26), even with increased per diem resource utilization due to their higher illness acuity. Including only the costs accruing to survivors will yield underestimate, as resources must also be invested in nonsurvivors to yield a surviving infant.

The currency date was not provided in seven of the articles; when this information was necessary for calculations, the most recent year of the cohort was assumed.

Studies of Costs Following Discharge from the Initial Hospitalization

The longer-run costing studies were similarly methodologically variable. Several of these limitations overlapped with those of the short-run studies.

Most of the studies were secondary analyses of data collected for other purposes. One study was a randomized trial with prospective data collection (28), six used linked governmental vital statistics and administrative or survey databases (27, 29, 31, 33, 34, 38), four used commercial or governmental claims databases (15, 36, 37, 41), three used clinical data (35, 39, 40), and two collected primary data through patient interviews (27, 30, 32). For preexisting data sets, validation was generally not described.

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

The majority of long-run studies were from broadly representative cohorts. These geographically based cohorts were at the regional (27, 3335, 39, 40), state (36, 38), and national (29, 31) levels. Only six smaller studies used convenience or hospital-specific samples (15, 28, 30, 32, 37, 41).

There were, however, other problems with generalizability. Six of the 16 long-run studies were from countries other than the United States (27, 3335, 39, 40). Of note, these were all geographical cohorts and represented the only studies in the review with assessment of direct medical costs beyond 2 years of age. Generalizability was also compromised by the broad time range of the studies. As noted above, the literature search was restricted only to studies conducted after 1980, given that postdischarge care is not as technologically sophisticated and therefore not as rapidly changing. This decision also allowed the inclusion of longer periods of follow-up. The cohort dates of these studies, however, ranged from 1980 to 2001; and the degree of comparability or applicability of earlier studies to the current context is unclear. Most studies did specify the birth weight and gestational age inclusion criteria for the samples, which allowed projection of the results for health policy. Studies that relied on claims databases used DRG designations, which must be applied with a degree of discretion, thus limiting external validity (15, 41).

In contrast to the studies of initial neonatal hospitalization, the sample sizes of most of these studies were adequate, possibly because of the greater reliance on administrative data. Four of the 16 studies had sample sizes of >20,000 infants (15, 31, 33, 34). Although these were heavily weighted toward full-term controls, the sizes of the subsamples of infants born preterm still comprised several hundred to several thousand infants. Only five studies had sample sizes of <150 infants (27, 30, 32, 35, 40). Although the confidence intervals around the cost estimates are consequently broad, most of these studies involved direct patient interviews or questionnaires and yielded unique information that would otherwise not have been obtainable.

Ten of the studies stated that they used costs derived from cost-to-charge ratios or other cost-accounting systems. None of the authors conducted primary costing research, and direct validation of preexisting cost-accounting systems was not presented; however, in at least some of the studies, these appear to be well-established and accepted systems (33, 34). The validity of patient-reported costs is equally difficult to ascertain. None of the studies presenting data from patient interviews, diaries, or questionnaires provided validation of these sources (27, 30, 32).

Uncertainty was again assessed inconsistently. Seven of the studies presented no information on sampling or parameter uncertainty, either through descriptive statistics or sensitivity analysis (15, 31, 35, 36, 3941).

The assessment of costs for premature infants over the longer term is a

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

more complex exercise, requiring the assembly of data from multiple sources. In some studies, this led to a large number of assumptions regarding the applicability of data, the choice of unit costs, and natural history (29, 31). Although these assumptions were well explained, it is very difficult to know the extent to which they are valid.

Unlike the assessment of initial hospital costs, the study of postdischarge resource utilization ideally tracks costs over several years. Most individuals have a positive rate of time preference; that is, they prefer to receive benefits sooner rather than later and to defer negative factors such as costs. To take account of this preference, economists apply a discount rate to reduce the weight of future investments in a stream of costs over time. This is variably set at 0 to 5 percent, with a 3 percent rate generally being considered reasonable for application in the base case. Only 5 of the 16 studies in the present review applied a discount rate (28, 29, 35, 39, 40).

CONTENT LIMITATIONS OF RETRIEVED STUDIES

Studies of Initial Hospitalization
Cost Categories

Cost categories in the studies of initial hospitalization costs for prematurity were variable. Only one study included both direct nonmedical costs and productivity losses (27). Only three studies included professional fees as a component of direct medical costs (13, 15, 26). The inclusion of overhead costs could be inferred in most cases but was usually not specified.

Only one author disaggregated costs into their component parts, in two studies. Rogowski showed that for infants with birth weights of <1,500 grams, accommodation costs represented most of the total cost of care (72 percent). Of the remaining ancillary costs, 22 percent was for respiratory therapy, 24 percent was for laboratory work, 7 percent was for radiology, and 16 percent for pharmacy (24).

Maternal Costs

This review was not specifically targeted to maternal costs, but these were measured in several of the studies reviewed (13, 15, 18, 20, 21). This is an important potential confounder, as the marginal contribution of maternal care may introduce bias in the comparison of costs for various gestational ages. For example, for infants born at from 25 to 29 weeks gestation, maternal costs were approximately 8 percent of the neonatal costs, for infants born at from 30 to 34 weeks gestation they were 32 percent, for infants born at from 37 to 38 weeks gestation they were 192 percent (18).

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Corresponding U.S. population projections were as follows: for infants born at 25 to 29 weeks gestation, $100 million; for infants born at 30 to 34 weeks gestation, $429.5 million; and for infants born at 37 to 38 weeks gestation $1.94 billion. Adams et al., found that maternal costs were $7,451 for those with full-term infants, $10,626 for those with healthy preterm infants, and $11,508 for those with extreme preterm infants (13). Chollet et al. found that maternal costs were $7,850 for the full-term DRG, $13,017 for the healthy preterm DRG, and $14,815 for the extreme preterm DRG (15). Thus, maternal costs vary inversely with birth weight, in parallel with neonatal costs, but to a lesser degree.

The articles that do include maternal costs list only the cost for the delivery admission; other costs are often associated with premature delivery (closer monitoring, repeat admissions, etc.) that may not be captured and would inflate the costs of the delivery itself. In an exception to this pattern, Schmitt et al. showed that, for pregnancies that resulted in births of infants less than 1,500 grams, prenatal hospitalizations accounted for $6.4 million of a total of $49.6 million of maternal hospital expenditures in California (25). Infertility treatments were also not captured in the studies reviewed.

Birth Weight and Gestational Age Cohorts

The majority of studies reported only on very low birth weight infants (in some cases with full-term controls). Although the per-infant cost is significantly lower for moderately preterm infants, the much higher incidence of moderate prematurity results in significant population costs. Schmitt et al. showed that infants with birth weights of 1,500 to 2,500 grams had total projected costs of $2.46 billion nationally (25). St John et al. projected that the costs for infants born at 30 to 34 weeks of gestation were $2.29 billion (26).

No studies determined the incremental contribution of low birth weight after controlling for gestational age.

Financial Burden on Minorities

Although certain ethnic and racial groups experience an increased burden of prematurity, the cost studies reviewed here did not specifically examine the issue of which populations are associated with the initial hospitalization costs.

Financial Burden on Payers

The studies reviewed did not provide specific disaggregations of the sources of payment for care for prematurity. Two of the studies relied on

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

claims databases from large third-party payers. Chollet et al. calculated that prematurity accounts for $4.7 billion to employer-sponsored health insurance plans, beyond the costs for normal-term deliveries (15). Older studies suggested that the newborn period is a major source of uncompensated care (42), accounting for as much as 19 percent of the direct delivery and uncompensated care dollars allocated to maternity and neonatal care (43). The March of Dimes estimates that 11 percent of newborns covered by employer health plans are born premature and that $7.4 billion (in 2002 dollars) in hospital charges was billed to private insurers (44).

Studies of Costs Following Discharge from the Birth Hospitalization
Cost Categories

Thirteen of 16 studies included direct medical costs (15, 27, 28, 3137, 3941). However, three of these included only rehospitalization costs and omitted the costs of outpatient physician visits or pharmaceuticals (31, 33, 34). Several of the studies failed to disaggregate the costs of rehospitalization from outpatient care (35, 37) or the costs of initial hospitalization from those of postdischarge care (31).

Educational costs were assessed in only four studies (27, 29, 31, 38). Two of these dealt with preschool costs (27, 31), one dealt with kindergar-ten costs (38), and two dealt with educational costs beyond kindergarten (29, 31). None followed an incidence cohort of infants longitudinally through school or assessed costs throughout the school spectrum by a consistent methodology.

Parental out-of-pocket expenditures were assessed in five studies (14, 17, 22, 27, 30). All of these included travel costs. Only one, non-U.S. study examined parental wage losses (27). None of the studies retrieved examined the lost productivity of the child.

Only one study attempted to ascertain costs in the direct medical, educational, and parental out-of-pocket expense categories (27).

Time Horizon

Only four of the studies tracked costs beyond 5 years of age. Despite the availability of cohorts reporting clinical follow-up to young adulthood (45, 46), there is almost no information on the cost implications of prematurity beyond early childhood.

Birth Weight and Gestational Age Cohorts

In contrast to the studies of initial hospitalization, most studies of

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

longer-term costs included infants across the birth weight and gestational age spectra.

Comorbid and Resulting Conditions

The studies retrieved did not make any attempt to attribute postdischarge costs to particular neonatal or postnatal conditions other than birth weight or gestational age. Thus, limited information is available on the proportion of rehospitalization or outpatient costs that are associated with chronic lung disease, retinopathy of prematurity, or cerebral palsy. There are, however, estimates of the costs of certain of these (4752) conditions separately, without the prematurity denominator afforded by the studies reviewed here. Studies of premature infants with chronic lung disease, for example, show that the costs for infants with admissions for respiratory syncytial virus infection are significantly higher; however, the proportion of costs for the entire birth cohort cannot be derived directly (51, 52). Similarly, although costs are available for cerebral palsy, most children with this condition were not born prematurely; the extent to which the costs for cerebral palsy drive the longer-term costs for premature infants is not known.

CHARACTERIZATION AND ESTIMATES OF COSTS OF INITIAL HOSPITALIZATION

Direct Medical Costs

Despite the heterogeneity of methods, designs, and sources, estimates of the per-patient cost are moderately consistent. The second column of Table D-3 shows the per-patient costs derived from the studies, expressed in 2005 U.S. dollars. For infants with birth weights of 500 to 999 grams, cost estimates ranged from $67,027 to $221,450, with four of six estimates in the range $84,847 to $126,380 per infant.

There are several potential sources of variability in cost estimates. First, the studies were performed with cohorts spanning a 15-year period. The characteristics of the adjusters for differences over time are therefore significant. The results are reported with both the medical component of the consumer price index (CPI), which relies on charges and may therefore overestimate charges, and the hospital producer price index (PPI), which used the actual net costs of resources, purchased by both public and private sources. As shown in Table D-3, use of these indices had a minimal effect on the results, suggesting that the source of variation lies elsewhere. A second potential source of variability in estimates is in the geographic diversity of the cohorts. International comparisons are facilitated to some extent by

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

TABLE D-3 Direct Medical Costs During Initial Hospitalization

Study

Per Patient Cost

Per Patient Cost - 2005 Dollars Using Medical CPI

Per Patient Cost - 2005 Dollars Using Hospital PPI

Adams (13)

Normal preterm: 10,416

Normal preterm: 14,634

Normal preterm: 13,506

 

Extreme preterm: 49,933

Extreme preterm: 70,151

Extreme preterm: 64,744

 

Normal full-term: 1,139

Normal full-term: 1,600

Normal full-term: 1,477

Chollet (15)

Normal preterm: 15,363

Normal preterm: 27,827

Normal preterm: 22,430

 

Extreme preterm: 55,424

Extreme preterm: 100,389

Extreme preterm: 80,919

 

Normal full-term: 2,376

Normal full-term: 4,303

Normal full-term: 3,469

Doyle (16)

500-999: 81,956

500-999: 73,920

500-999: 68,801

 

Aust dollars per live birth

 

 

Gilbert (18)

25 week survivor: 202,700

25 week survivor: 284,775

25 week survivor: 262,825

 

30 week survivor: 46,400

30 week survivor: 65,188

30 week survivor: 60,163

 

38 week survivor: 1,100

38 week survivor: 1,545

38 week survivor: 1,426

Kilpatrick (19)

24 week survivor: 294,749

24 week survivor: 447,851

24 week survivor: 405,210

 

25 week survivor: 181,062

25 week survivor: 275,111

25 week survivor: 248,918

 

26 week survivor: 166,215

26 week survivor: 252,552

26 week survivor: 228,506

Luke (20)

25-27 weeks: 195,254

25-27 weeks: 329,292

25-27 weeks: 285,070

 

28-30weeks: 91,343

28-30weeks: 154,048

28-30weeks: 133,361

 

31-34 weeks: 18,367

31-34 weeks: 30,976

31-34 weeks: 26,816

 

39-42 weeks: 2,230

39-42 weeks: 3,761

39-42 weeks: 3,256

Marbella (21)

“premature survivor”: 16,973

“premature survivor”: 25,789

“premature survivor”: 23,334

 

3000-4500 gram: 1,300

3000-4500 gram: 1,975

3000-4500 gram: 1,787

Rogowski (23)

501-1500 grams: 49,457

501-1500 grams: 75,147

501-1500 grams: 67,992

 

501-1000 grams: 83,176

501-1000 grams: 126,380

501-1000 grams: 114,347

 

24-26 weeks: 95,560

 

 

 

27-29 weeks: 61,724

 

 

 

30-32 weeks: 35,106

 

 

Rogowski (24)

501-1500 grams: 53,316

501-1500 grams: 70,633

501-1500 grams: 68,043

 

501-1000 grams: 90,015

501-1000 grams: 119,251

501-1000 grams: 114,879

 

24-26 weeks: 101,638

 

 

 

27-29 weeks: 62,960

 

 

 

30-32 weeks: 34,258

 

 

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

US Population Cost Projection— 2005 Dollars Using Medical CPI

US Population Cost Projection— 2005 Dollars Using Hospital PPI

Per-Patient LOS

Per-Patient Ventilator Days

Per-Patient Maternal Costs

<37 weeks: 7.04 billion

<37 weeks: 6.50 billion

Normal preterm: 6.1

N/A

Normal preterm: 10,626

<28 weeks: 2.06 billion

<28 weeks: 1.90 billion

Extreme preterm: 19.9

 

Extreme preterm: 11,508

37-41 weeks: 5.17 billion

37-41 weeks: 4.77 billion

Normal full-term: 1.9

 

Normal full-term: 7,452

<37 weeks: 13.38 billion

<37 weeks: 10.78 billion

N/A

N/A

Normal preterm: 13,017

<28 weeks: 2.95 billion

<28 weeks: 2.38 billion

 

 

Extreme preterm: 14,815

37-41 weeks: 13.9 billion

37-41 weeks: 11.20 billion

 

 

Normal full-term: 7,850

500-999 grams:

500-999 grams:

500-999:

500-999:

N/A

1.94 billion

1.81 billion

82.1 per live birth

34.4 per live birth

 

<37 weeks: 4.62 billion

<37 weeks: 4.26 billion

25 week survivor: 92

N/A

25 week survivor: 7,500

30-34 weeks: 1.87 billion

30-34 weeks: 1.73 billion

30 week survivor: 30.4

 

30 week survivor: 7,200

500-1499 grams: 2.82 billion

500-1499 grams: 2.60 billion

38 week survivor: 1.8

 

38 week survivor: 2,500

500-2499 grams: 3.50 billion

500-2499 grams: 3.23 billion

 

 

 

24 week survivor: 1.79 billion

24 week survivor: 1.62 billion

24 week survivor: 120

24 week

N/A

25 week survivor: 1.28 billion

25 week survivor: 1.16 billion

25 week survivor: 86

survivor: 76

 

26 week survivor: 1.36 billion

26 week survivor: 1.23 billion

26 week survivor: 80

25 week

 

 

 

 

survivor: 58

 

 

 

 

26 week

 

 

 

 

survivor: 45

 

25-27 weeks: 5.25 billion

25-27 weeks: 4.55 billion

25-27 weeks: 71.2

N/A

25-27 weeks: 84,892

28-30weeks: 4.76 billion

28-30weeks: 4.12 billion

28-30weeks: 39.0

 

28-30weeks: 81,971

31-34 weeks: 4.35 billion

31-34 weeks: 3.77 billion

31-34 weeks: 11.8

 

31-34 weeks: 23,759

39-42 weeks: 8.7 billion

39-42 weeks: 7.54 billion

39-42 weeks: 3.1

 

39-42 weeks: 7,573

<37 weeks: $8.16 billion

<37 weeks: 7.38 billion

“premature survivor”: 14.1

N/A

N/A

3000-4500 grams: $5.85 billion

3000-4500 grams: 5.29 billion

3000-4500 gram: 1.7

 

 

501-1500 grams: 3.93 billion

501-1500 grams: 3.56 billion

501-1500 grams: 49

N/A

N/A

501-1000 grams: 2.89 billion

501-1000 grams: 2.61 billion

501-1000 grams: 75

 

 

 

 

24-26 weeks: 82

 

 

 

 

27-29 weeks: 58

 

 

 

 

30-32 weeks: 39

 

 

501-1500 grams: 3.69 billion

501-1500 grams: 3.55 billion

501-1500 grams: 47

N/A

N/A

501-1000 grams: 2.72 billion

501-1000 grams: 2.62 billion

501-1000 grams: 73

 

 

 

 

24-26 weeks: 79

 

 

 

 

27-29 weeks: 55

 

 

 

 

30-32 weeks: 36

 

 

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Study

Per Patient Cost

Per Patient Cost— 2005 Dollars Using Medical CPI

Per Patient Cost— 2005 Dollars Using Hospital PPI

Schmitt (25)

500-999 grams: 205,218

500-999 grams: 221,450

500-999 grams: 222,104

 

500-1499 grams: 136,456

500-1499 grams: 147,249

500-1499 grams: 147,684

 

>2,500 grams: 1,647

>2,500 grams: 1,777

>2,500 grams: 1,783

St John (26)

24 week survivor:145,892

24 wk survivor: 246,044

24 wk survivor: 213,002

 

30 week survivor: 37,569

30 week survivor: 63,359

30 week survivor: 54,851

 

40 week survivor: 1,127

40 week survivor: 1,900

40 week survivor: 1,645

Tommiska (27)

<1000 gram survivor:

<1000 gram survivor: 105,662

<1000 gram survivor: 90,338

 

70,290 Euro

Full term: 774

Full term: 662

 

Full term: 515 Euro

 

 

Victorian Infant

500-999 grams: 53,655

500-999 grams: 67,865

500-999 grams: 58,752

Collaborative Study

Aust dollars per live birth

 

 

Group (12)

 

 

 

Weighted averages calculated from data in articles to correspond to weight categories above.

the Organisation for Economic Co-operation and Development purchasing power parity, although this accounts only for differences in price and not differences in practice style. Within the United States, neonatal care is fairly homogeneous, so it is likely that price differences will outweigh geographic practice style differences. An adjustment for geographic differences was not made because of the difficulty in defining the location of care for most of the cohorts, which were often either at the state level or at institutions in several states. Finally, variability arises because of the authors’ choices of which types of costs to include. Unfortunately, most authors did not disaggregate costs into components, thus making an assessment of the contributions of these choices to the cost estimates difficult to quantify. Table D-3 presents neonatal costs separately from maternal costs, showing a greater degree of variability for the former.

Because it is difficult to fully address variability in prices, an attempt was also made to describe resource utilization independent of its price. Table D-3 shows the length of stay (LOS) and the number of days of assisted ventilation for the studies reviewed. Although not all studies reported these measures, the variability in the direct markers of resource utilization appears to be somewhat less than that for cost. The LOSs for infants with birth weights of 500 to 999 grams ranged from 64 to 106 days in six studies, with four studies showing LOSs in the narrow range of 73 to 82 days.

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

US Population Cost Projection 2005 Dollars Using Medical CPI

US Population Cost Projection 2005 Dollars Using Hospital PPI

Per-Patient LOS

Per-Patient Ventilator Days

Per-Patient Maternal Costs

500-999 grams: 7.70 billion

500-999 grams: 7.70 billion

500-999 grams: 64.4

N/A

500-999 grams: 11,609

500-1499 grams: 5.06 billion

501-1499 grams: 5.06 billion

500-1499 grams: 47.9

 

500-1499 grams: 12,183

>2,500 grams: 6.58 billion

>2,500 grams: 6.58 billion

>2,500 grams: 2.3

 

>2,500 grams: 3,378

< 37 weeks: 9.92 billion

< 37 weeks: 8.59 billion

N/A

N/A

N/A

30-34 weeks: 3.86 billion

30-34 weeks: 3.34 billion

 

 

 

37-42 weeks: 7.34 billion

37-42 weeks: 6.35 billion

 

 

 

500-999 grams: 2.41 billion

500-999 grams: 2.06 billion

<1000 gram

N/A

N/A

37-41 weeks: 2.50 billion

37-41 weeks: 2.14 billion

survivor: 106

 

 

 

 

Full term: N/A

 

 

500-999 grams: 1.55 billion

500-999 grams: 1.34 billion

500-999 grams:

500-999 grams:

N/A

 

 

77 per live birth

23 per live birth

 

Studies that reported both birth weight- and gestational age–specific costs show that these are consistent. Gilbert et al. give a per-patient cost of $202,700 for a survivor born at 25 weeks gestation and a per-patient cost of $224,400 for a survivor born with a birth weight of 500 to 750 grams (18). For survivors born at 30 weeks gestation, the per-patient cost is $46,400, whereas for a survivor with a birth weight of 1,250 to 1,499 grams, the per-patient cost is $51,900. These authors excluded infants whose birth weights and gestational ages did not match; the birth weight-specific costs may therefore be less biased, as the gestational age on vital statistics records is more often likely to be inaccurate. Across all studies, under the assumption that DRG Code 387 for extreme prematurity corresponds to gestational ages of <28 weeks, costs ranged from $70,451 to $100,389, similar to those for the corresponding birth weight range of 500 to 999 grams.

When the costs were projected to the U.S. population, the range for infants born at <37 weeks gestation was $4.62 billion to $13.38 billion, with the latter estimate derived from a study based on charges (15). For infants with birth weights of 500 to 999 grams, the costs ranged from $1.53 billion to $5.06 billion, with the range for the U.S. population being $2.72 billion to $5.06 billion.

Full-term controls had per-patient costs of $734 to $4,303. If the stud-

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

ies that used charges or failed to specify this were excluded, the range for the U.S. population was $1,545 to $1,900. Corresponding population projections were $2.37 billion to $6.58 billion.

The previously noted inverse relationship between costs and gestational age or birth weight was observed in all studies. The marked right skew of the cost data was also noted in those studies that reported means and medians (2325, 27).

Direct Nonmedical Costs and Lost Productivity

Table D-4 describes studies that assessed parental out-of-pocket expenditures and lost productivity. In the single study that examined both non-medical direct costs and productivity losses, parental mean costs before discharge for extremely low birth weight infant were €2,755, or 4 percent of total costs (27). These were composed of 64 percent travel costs, 30 percent lost earnings, and 6 percent accommodation. An earlier interview study of travel costs for mothers of 109 low-birth-weight infants in Britain showed that 36 percent of families traveled more than 21 miles, that 88 percent of families visited the infants daily, and that the median total expenditure was between £101 and £200 (in 1990 United Kingdom pounds sterling), with a maximal expenditure of £1,000 (22). More recently, Gennaro observed the families of 224 infants born at less than 37 weeks of gestational age discharged from a single center between 1990 and 1994 (30). That study did not detail the productivity losses during the initial hospitalization. The average mother in that study used 4 weeks of maternity leave during the initial hospitalization. Out-of-pocket expenses averaged $433, with the largest category being transportation.

CHARACTERIZATION AND ESTIMATES OF COSTS FOLLOWING DISCHARGE FROM THE BIRTH HOSPITALIZATION

Direct Medical Costs

Estimates of direct medical resource utilization and costs following discharge from the birth hospitalization are given in Table D-5. There was a greater degree of heterogeneity compared with that in the studies of initial hospitalization, in large degree because of the various time horizons and costs included. For this reason, it is problematic to provide a single estimate or range of estimates for these costs. As for short-run costs, costs and resource utilization were inversely related to gestational age. In addition, in studies with longer follow-ups, the majority of the costs accrued in the first year of life. In studies that measured both outpatient and rehospitalization costs, the latter accounted for the majority of costs in the first year of life,

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

TABLE D-4 Parental Out of Pocket Expenses and Lost Productivity During Initial Hospitalization

Paper

Parental Wages

Lost Productivity of Child

Child Care

Other

Travel

Brazier (14)

N/A

N/A

N/A

N/A

Per visit: 3-17

Per week: 27.98

Gennaro (30)

N/A

N/A

N/A

Total initial hosp 2,500: 433

<2,500: 271

Giacoia (17)

N/A

N/A

N/A

N/A

Total: 250

Per visit: 25

McLoughlin (22)

N/A

N/A

N/A

N/A

Median: Approx 100 (from figure)

Tommiska (27)

<1000: 827a

N/A

N/A

Accomodation <1,000: 165a

Total initial hospital <1,000: 2,755

<1,000: 1763a

aCalculated from data in article.

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

TABLE D-5 Direct Medical Expenses Following Discharge from the Initial Hospitalization

Paper

Time Horizon

Rehospitalization (cost)

Outpatient MD, Drug and Other (cost)

Broyles (28)

1 year adjusted age

<1,500: 6,982

<1,500: 2,931

Chollet (15)a

2 years

(1 year detail)

Not separated from initial hospitalization

Normal preterm: 4,463

Extreme preterm: 6,329

Normal full-term: 2,243

Other preterm: 4,195

Lewit (31)

15 years of age

<1,500

<1 year (not separated from initial hospitalization)

3-5 years: 290

6-10 years: 470

N/A

McCormick (32)

1 year of age

<1,500: 8,250

Term: 900

MD visits

1,500: 564

Term: 232

Other Outpatient:

<1,500: 1,311

Term: 63

Medstat (41)

1 year of age

Preterm: 35,034

Normal full-term: 1,210

Preterm: 6,576

Normal full-term: 1,620

Petrou (33)

5 years of age

Year 1:

>37 weeks: 297

32-36 weeks: 2,016

28-31 weeks: 7,272

<28 weeks: 10,630

Total (inc birth hosp):

>37 weeks: 1,333

32-36 weeks: 4,378

28-31 weeks: 14,059

<28 weeks: 13,639

N/A

Petrou (34)

10 years of age

Including birth hosp:

>37 weeks: 1,659

32-36 weeks:7,394

28-31 weeks:17,751

<28 weeks: 17,820

N/A

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Post-Discharge Total

Rehospitalization (mean or median)

Rehospitalization (mean total days)

Rehospitalization (LOS)

Outpatient Visits (Clinic, MD, ER)

<1,500: 9,913

<1,500: 0.7

<1,500: 7.6

N/A

<1,500:

Clinic: 6.4

ER: 1.9

Total: 8.2

Not separated from initial hospitalization

N/A

N/A

N/A

N/A

<1,500

<1 year (not separated from initial hospitalization)

3-5 years: 290

6-10 years: 470

N/A

N/A

N/A

N/A

<1,500: 10,139

Term: 1,179

N/A

N/A

N/A

<1,500: 18.5

Term: 9.3

Preterm: 41,611

Normal full-term: 2,831

Preterm: 1.3

Normal full-term: 1.1

N/A

Preterm: 16.8

Normal full-term: 2.3

Preterm: 8.9

Normal full-term: 5.9

Year 1:

>37 weeks: 297

32-36 weeks: 2,016

28-31 weeks: 7,272

<28 weeks: 10,630

Total (inc birth hosp):

>37 weeks: 1,333

32-36 weeks: 4,378

28-31 weeks: 14,059

<28 weeks: 13,639

>37 weeks: 2

32-36 weeks: 2

28-31 weeks: 1

<28 weeks: 1

>37 weeks: 6.3

32-36 weeks: 16.2

28-31 weeks: 48.7

<28 weeks: 49

N/A

N/A

Including birth hosp:

>37 weeks: 1,659

32-36 weeks: 7,394

28-31 weeks: 17,751

<28 weeks: 17,820

>37 weeks: 0.75

32-36 weeks: 1.48

28-31 weeks: 2.54

<28 weeks: 1.9

Including birth hosp:

>37 weeks: 6.2

32-36 weeks: 18

28-31 weeks: 58.2

<28 weeks: 59.2

N/A

N/A

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Paper

Time Horizon

Rehospitalization (cost)

Outpatient MD, Drug and Other (cost)

Pharaoh (35)

4 years of age

N/A

N/A

Rogowski (36)

1 year of age

<1,000: 12,800

<1,500: 5,290

<1,500: 870

Rolnick (37)

1 year postdischarge

N/A

N/A

Stevenson (39)c

8-9 years of age

<1,000: 421

1,001-1,500: 433

1,501-2,000: 270

<1,000: 876

1,001-1,500: 793

1,501-2,000: 430

Stevenson (40)d

8-9 years of age

<1,000: 1,861

1,001-1,500: 1,439

1,501-2,000: 570

<1,000: 1,617

1,001-1,500: 1,447

1,501-2,000: 1,149

Tommiska (27)e

2 years of age

Year 1:

<1,000: 12,185

control: 225

Year 2:

<1,000: 2,575

control: 195

Year 1:

<1,000: 1,995

control: 295

Year 2:

<1,000: 1,420

control: 315

aMothers and infants combined

bCalculated from data in article

cPer infant with no disability

dPer infant with disability

eTotal cost includes nonmedical (See Tables D-5 and D-6)

with a range of 54 to 60 percent of the costs in the non-U.S. studies and 70 to 86 percent of the costs in the U.S. cohorts.

Petrou examined factors associated with higher rehospitalization costs (33). These included maternal conditions, such as hospitalization, age >35 or <20 years, or smoking; perinatal factors, such as instrumental delivery or delivery complications; demographic factors, such as lower socioeconomic status; and prematurity.

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

Postdischarge Total

Rehospitalization (mean or median)

Rehospitalization (mean total days)

Rehospitalization (LOS)

Outpatient Visits (Clinic, MD, ER)

<1,500: 2,620b

N/A

N/A

N/A

N/A

<1,500: 6,160

<1,000: 1.8

1,000-1,249: 1.9

1,250-1,499: 2.1

<1,500: 5.5

<1,000: 13.6

1000-1,249: 9.6

1,250-1,499: 9.8

N/A

2,500-4,499: 2,919

1,000-2,500: 5,938

N/A

N/A

N/A

N/A

<1,000: 1,297

1,001-1,500: 1,226

1,501-2,000: 699

N/A

N/A

N/A

N/A

<1,000: 3,475

1,001-1,500: 2,886

1,501-2,000: 1,719

N/A

N/A

N/A

N/A

Year 1:

<1,000: 20,390

control: 1,415

Year 2:

<1,000: 13,955

control: 1,205

Year 1:

<1,000: 1.8

control: 0.1

Year 2: <1,000:1.0

control: 0.1

Year 1:

<1,000: 8.4

control: 0.3

Year 2:

<1,000: 4.2

control: 0.4

N/A

Year 1:

<1,000: 7.1

control: 2.9

Year 2:

<1,000: 6.5

control: 3.8

Educational Costs

Details of studies assessing educational costs are given in Table D-6. As noted above, the assessment of educational costs was inconsistent with respect to both the study methodology and the age at the time of the assessment. After controlling for medical, economic, and social factors, Chaikind and Corman found a 49 percent relative risk for special education placement in low-birth-weight infants (29). This translated to a net present value

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

TABLE D-6 Educational Expenditures Following Discharge from Initial Hospitalization

Paper

Pre-School Age

School Age

Chaikand (29)

N/A

<2500

Age 6-15: 1,240

(370.8 million total/yr)

Lewit (31)

<1500 Preschool: 290

<1500

Age 6-10: Special Ed 150

Age 6-15: Grade Rep 45

Pharaoh (35)

N/A

<1,500: 4,211

Roth (38)

N/A

Kindergarten

<1,000: 6979

1,000-1,499: 5,740

1,500-2,499: 4,870

>2,500: 4,375

Tommiska (27)

“Special Day Care”

Year 1:

<1,000: 0

control: 0

Year 2:

<1,000: 1,285

control: 0

N/A

Clements

Early intervention

(0-3 years cumulative)

<28 weeks: 7,182

28-30 weeks: 5254

31-33 weeks: 2,654

34-36 weeks: 1,321

37-39 weeks: 770

>40 weeks: 613

N/A

of $1,240 (in 1988 U.S. dollars) per low birth weight infant, or $370 million per year for special education costs alone. Roth et al. found that infants with birth weights of <1,000 grams had kindergarten costs 60 percent higher than those for normal birth weight controls (38). However, the authors noted that low levels of maternal education and poverty accounted for more than three quarters of the total excess costs of kindergarten, whereas low birth weight accounted for only 1 percent of the total excess costs because of the relative numbers of children with these predisposing conditions.

Early intervention costs may also be significant. A recent study of statelevel data in Massachusetts assessed costs from birth to age 3 years for early

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

intervention services by gestational age. Infants born at less than 28 weeks of gestation had early intervention costs of $7,182 (in 2003 U.S. dollars), whereas the early intervention costs were $613 for those delivered at 40 weeks of gestation or more (53).

Direct Nonmedical Costs and Lost Productivity

Table D-7 shows the costs associated with postdischarge parental out-of-pocket expenses and lost productivity. Tommiska et al. documented firstyear parental wage losses of €5,990 for infants with birth weights of <1,000 grams, whereas the loss was €880 for controls. These costs increased to €8,175 in the second year for the parents of children born with low birth weights (27). Similar data are not available for the United States.

SUMMARY OF COST STUDIES FOR PREMATURITY

Several themes emerged from this systematic review of cost-of-illness studies:

  1. Most studies had significant methodological limitations. These included a reliance on administrative data sets without adequate checks on data validity, small sample sizes and possible selection bias, a lack of quantification of uncertainty through the use of descriptive statistics or sensitivity analysis, the use of charges or inadequately described costing, the lack of a control group, and a failure to discount appropriately for longer studies.

  2. All cost estimates in the literature omitted at least some potentially important components of costs and are therefore likely to underestimate true resource use. These omissions included professional fees, hospital costs for transferred infants, costs for nonsurviving infants, and out-of-pocket costs for parents. There is almost no information on the lost earnings of parents and no information on the productivity losses of the infants themselves. Maternal costs for the delivery hospitalization and the costs of antenatal admissions were omitted by most studies. The analysis of infants and mothers separately risks both an underestimation of the costs of prematurity and the possibility of missed shifting of costs between the two groups. Very few studies have addressed educational costs, and these do not provide adequate information across the spectrum of school age and disability.

  3. Most authors did not disaggregate costs into their components, making it problematic to target the sources and predictors of high costs.

  4. Studies confirmed the previously noted inverse relationship of maternal, neonatal, and postdischarge costs with birth weight and gestational age.

Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
×

    TABLE D-7 Out-of-Pocket Expenses and Lost Productivity Following Discharge from Initial Hospitalization

    Paper

    Parental Wages

    Lost Productivity of Child

    Child Care

    Other

    Travel

    Tommiska (27)

    Year 1:

    <1,000: 5,990

    control: 880

    Year 2:

    <1,000: 8,175

    control: 595

    N/A

    N/A

    “Home Aid”

    Year 1:

    <1,000: 0

    control: 0

    Year 2:

    <1,000: 255

    control: 85

    Year 1:

    <1,000: 75

    control: 15

    Year 2:

    <1,000: 85

    control: 15

    McCormick (32)

    N/A

    N/A

    <1,500: 563

    Term: 1,082

    N/A

    <1,500: 180

    Term: 23

    Gennaro (30)

    N/A

    N/A

    N/A

    Non-reimbursed Health

    <2,500: 69

    Total Out of Pocket

    <2,500: 445

    N/A

    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
    ×
    1. Although the per-patient cost for moderately preterm infants is lower than that for extremely preterm infants, the larger numbers of these infants results in population costs of similar magnitudes in both groups. This has important implications for the setting of policy, as interventions that ameliorate moderate prematurity may be as cost-effective as the more difficult interventions that would eliminate extreme prematurity.

    2. The time horizon for economic follow-up is inadequate. Very few studies have followed infants beyond 5 years, and none have documented the implications of prematurity in young adulthood, despite the availability of appropriate cohorts.

    3. Most of the studies with longer time horizons or more comprehensive methods of cost ascertainment have been from countries other than the United States. The appropriateness of generalizing these cost estimates to the United States is uncertain.

    RECOMMENDATIONS FOR POLICY AND FURTHER RESEARCH

    The findings summarized above have direct implications for policy and research:

    1. Because policy makers are likely to accept the dollar estimates of the costs of illness without adequately assessing the quality of the underlying studies, it is important that the peer review process identify the more significant methodological limitations, such as the use of unadjusted charges or the lack of assessment of uncertainty.

    2. In light of the prominence of the economic implications on health policy decision making for this population, it is essential that a U.S. study with a societal perspective be performed. This should include a comprehensive assessment of productivity losses and parental out-of-pocket expenditures in the U.S. context.

    3. Studies with longer time horizons should be undertaken and should preferably use an incidence approach rather than a prevalence approach to costing. At a minimum, these should extend well into school age. The implications of prematurity as young adults enter the work force should be examined, as should lifetime labor force implications.

    4. Because of the interactions between socioeconomic status and outcome, costing studies should include specific analyses of the increased burden on particular racial and ethnic populations.

    5. The focus of short-run costing studies should be shifted toward a perinatal perspective, in which the unit of analysis is the maternal-infant dyad. This is now feasible with improved data linkage.

    6. Attention should be directed toward the economic implications of moderate prematurity, in addition to extreme prematurity.

    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
    ×
    1. A longitudinal analysis of the economic implications of prematurity to the educational system should be undertaken.

    2. A comprehensive longitudinal study of the long-term economic implications of prematurity should be undertaken.

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    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
    ×
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    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    Page 716
    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    Page 717
    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    Page 718
    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    Page 719
    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    Page 720
    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    Page 721
    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    Page 722
    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    Page 723
    Suggested Citation:"Appendix D A Systematic Review of Costs Associated with Preterm Birth ." Institute of Medicine. 2007. Preterm Birth: Causes, Consequences, and Prevention. Washington, DC: The National Academies Press. doi: 10.17226/11622.
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    The increasing prevalence of preterm birth in the United States is a complex public health problem that requires multifaceted solutions. Preterm birth is a cluster of problems with a set of overlapping factors of influence. Its causes may include individual-level behavioral and psychosocial factors, sociodemographic and neighborhood characteristics, environmental exposure, medical conditions, infertility treatments, and biological factors. Many of these factors co-occur, particularly in those who are socioeconomically disadvantaged or who are members of racial and ethnic minority groups.

    While advances in perinatal and neonatal care have improved survival for preterm infants, those infants who do survive have a greater risk than infants born at term for developmental disabilities, health problems, and poor growth. The birth of a preterm infant can also bring considerable emotional and economic costs to families and have implications for public-sector services, such as health insurance, educational, and other social support systems.

    Preterm Birth assesses the problem with respect to both its causes and outcomes. This book addresses the need for research involving clinical, basic, behavioral, and social science disciplines. By defining and addressing the health and economic consequences of premature birth, this book will be of particular interest to health care professionals, public health officials, policy makers, professional associations and clinical, basic, behavioral, and social science researchers.

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