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Preterm Birth: Causes, Consequences, and Prevention
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.
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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,
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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 (1–7). 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
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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 (12–27) 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.
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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
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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
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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
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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
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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
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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
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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
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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
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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
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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.
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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
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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
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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:
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.
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.
Most authors did not disaggregate costs into their components, making it problematic to target the sources and predictors of high costs.
Studies confirmed the previously noted inverse relationship of maternal, neonatal, and postdischarge costs with birth weight and gestational age.
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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
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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.
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.
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:
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.
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.
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.
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.
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.
Attention should be directed toward the economic implications of moderate prematurity, in addition to extreme prematurity.
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A longitudinal analysis of the economic implications of prematurity to the educational system should be undertaken.
A comprehensive longitudinal study of the long-term economic implications of prematurity should be undertaken.
REFERENCES
1. Mugford M. The cost of neonatal care: reviewing the evidence. Soz Praventivmed. 1995;40(6):361–368.
2. Petrou S. Economic consequences of preterm birth and low birthweight. Br J Obstet Gynaecol. 2003 Apr;110(Suppl 20):17–23.
3. Petrou S, Davidson LL. Economic issues in the follow-up of neonates. Semin Neonatol. 2000 May;5(2):159–169.
4. Petrou S, Sach T, Davidson L. The long-term costs of preterm birth and low birth weight: results of a systematic review. Child Care Health Dev. 2001 Mar;27(2):97–115.
5. Richardson DK, Zupancic JA, Escobar GJ, Ogino M, Pursley DM, Mugford M. A critical review of cost reduction in neonatal intensive care. I. The structure of costs. J Perinatol. 2001 Mar;21(2):107–115.
6. Richardson DK, Zupancic JA, Escobar GJ, Ogino M, Pursley DM, Mugford M. A critical review of cost reduction in neonatal intensive care. II. Strategies for reduction. J Perinatol. 2001 Mar;21(2):121–127.
7. Zupancic JA, Richardson DK, Lee K, McCormick MC. Economics of prematurity in the era of managed care. Clin Perinatol. 2000 Jun;27(2):483–497.
8. U.S. Bureau of Labor Statistics. Consumer Price Index—All Urban Consumers—Medical Care (Series ID CUUR0000SAM). [Online]. Available: www.bls.gov/data [accessed August 1, 2005].
9. United States Bureau of Labor Statistics. Producer Price Index Industry Data—Hospitals (ID PCU622). 2005 [Online]. Available: www.bls.gov/data [accessed September 20, 2005].
10. Organization for Economic Cooperation and Development. Purchasing Power Parities for OECD Countries since 1980. 2005 [Online]. Available: www.oecd.org [accessed August 1, 2005].
11. Martin JA, Hamilton BE, Sutton PD, Ventura SJ, Menacker F, ML M. Births: Final Data for 2002. National Vital Stat Rep. 2003;52(10): 1-113.
12. The Victorian Infant Collaborative Study Group. Economic outcome for intensive care of infants of birthweight 500-999 g born in Victoria in the post surfactant era. J Paediatr Child Health. 1997 Jun;33(3):202–208.
13. Adams EK, Nishimura B, Merritt RK, Melvin C. Costs of poor birth outcomes among privately insured. J Health Care Finance. 2003 Spring;29(3):11–27.
14. Brazier L, Harper K, Marrington S. Hospital visiting costs. An exploratory study into travelling expenses incurred by parents with babies in a regional neonatal unit. J Neonat Nurs. 1995;1(2):29–31.
15. Chollet DJ, Newman JF Jr, Sumner AT. The cost of poor birth outcomes in employersponsored health plans. Med Care. 1996 Dec;34(12):1219–1234.
16. Doyle LW. Evaluation of neonatal intensive care for extremely low birth weight infants in Victoria over two decades. II. Efficiency. Pediatrics. 2004 Mar;113(3 Pt 1):510–514.
17. Giacoia GP, Rutledge D, West K. Factors affecting visitation of sick newborns. Clin Pediatr (Phila). 1985 May;24(5):259–262.
18. Gilbert WM, Nesbitt TS, Danielsen B. The cost of prematurity: quantification by gestational age and birth weight. Obstet Gynecol. 2003 Sep;102(3):488–492.
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19. Kilpatrick SJ, Schlueter MA, Piecuch R, Leonard CH, Rogido M, Sola A. Outcome of infants born at 24-26 weeks’ gestation. I. Survival and cost. Obstet Gynecol. 1997 Nov;90(5):803–808.
20. Luke B, Bigger HR, Leurgans S, Sietsema D. The cost of prematurity: a case-control study of twins vs singletons. Am J Public Health. 1996 Jun;86(6):809–814.
21. Marbella AM, Chetty VK, Layde PM. Neonatal hospital lengths of stay, readmissions, and charges. Pediatrics. 1998 Jan;101(1 Pt 1):32–36.
22. McLoughlin A, Hillier VF, Robinson MJ. Parental costs of neonatal visiting. Arch Dis Child. 1993 May;68(5 Spec No):597–599.
23. Rogowski J. Measuring the cost of neonatal and perinatal care. Pediatrics. 1999 Jan;103(1 Suppl E):329–335.
24. Rogowski J. Using economic information in a quality improvement collaborative. Pediatrics 2003 Apr;111(4 Pt 2):e411–e418.
25. Schmitt SK, Sneed L, Phibbs CS. The costs of newborn care in California: a populationbased study. Pediatrics. 2006;117:154–160.
26. St John EB, Nelson KG, Cliver SP, Bishnoi RR, Goldenberg RL. Cost of neonatal care according to gestational age at birth and survival status. Am J Obstet Gynecol. 2000 Jan;182(1 Pt 1):170–175.
27. Tommiska V, Tuominen R, Fellman V. Economic costs of care in extremely low birthweight infants during the first 2 years of life. Pediatr Crit Care Med. 2003 Apr;4(2):157–163.
28. Broyles RS, Tyson JE, Heyne ET, Heyne RJ, Hickman JF, Swint M, et al. Comprehensive follow-up care and life-threatening illnesses among high-risk infants: a randomized controlled trial. JAMA 2000 Oct 25;284(16):2070–2076.
29. Chaikand S, Corman H. The impact of low birthweight on special education costs. J Health Econ. 1991 Oct;10(3):291–311.
30. Gennaro S. Leave and employment in families of preterm low birthweight infants. Image J Nurs Sch. 1996 Fall;28(3):193–198.
31. Lewit EM, Baker LS, Corman H, Shiono PH. The direct cost of low birth weight. Future Child. 1995 Spring;5(1):35–56.
32. McCormick MC, Bernbaum JC, Eisenberg JM, Kustra SL, Finnegan E. Costs incurred by parents of very low birth weight infants after the initial neonatal hospitalization. Pediatrics. 1991 Sep;88(3):533–541.
33. Petrou S. The economic consequences of preterm birth during the first 10 years of life. Br J Obstet Gynaecol. 2005 Mar;112 Suppl 1:10–15.
34. Petrou S, Mehta Z, Hockley C, Cook-Mozaffari P, Henderson J, Goldacre M. The impact of preterm birth on hospital inpatient admissions and costs during the first 5 years of life. Pediatrics. 2003 Dec;112(6 Pt 1):1290–1297.
35. Pharoah PO, Stevenson RC, Cooke RW, Sandu B. Costs and benefits of neonatal intensive care. Arch Dis Child. 1988 Jul;63(7 Spec No):715–718.
36. Rogowski J. Cost-effectiveness of care for very low birth weight infants. Pediatrics. 1998 Jul;102(1 Pt 1):35–43.
37. Rolnick SJ, Jackson JM, O’Connor P, DeFor T. Impact of birthweight on healthcare charges within a managed care organization. Am J Manag Care. 2000 Dec;6(12):1289– 1296.
38. Roth J, Figlio DN, Chen Y, Ariet M, Carter RL, Resnick MB, et al. Maternal and infant factors associated with excess kindergarten costs. Pediatrics. 2004 Sep;114(3):720–728.
39. Stevenson RC, McCabe CJ, Pharoah PO, Cooke RW. Cost of care for a geographically determined population of low birthweight infants to age 8-9 years. I. Children without disability. Arch Dis Child Fetal Neonat Ed. 1996 Mar;74(2):F114–F117.
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40. Stevenson RC, Pharoah PO, Stevenson CJ, McCabe CJ, Cooke RW. Cost of care for a geographically determined population of low birthweight infants to age 8-9 years. II. Children with disability. Arch Dis Child Fetal Neonat Ed. 1996 Mar;74(2):F118–F121.
41. Thomson Medstat. The Cost of Prematurity to U.S. Employers. http://www.marchofdimes.com/prematurity/15341_15349.asp : March of Dimes; 2004.
42. Imershein AW, Turner C, Wells JG, Pearman A. Covering the costs of care in neonatal intensive care units. Pediatrics. 1992 Jan;89(1):56–61.
43. Long SH, Marquis MS, Harrison ER. The costs and financing of perinatal care in the United States. Am J Public Health. 1994 Sep;84(9):1473–1478.
44. March of Dimes. Impact on Business. http://www.marchofdimes.com/prematurity/15341_15349.asp: March of Dimes; 2005.
45. Hack M, Youngstrom EA, Cartar L, Schluchter M, Taylor HG, Flannery D, et al. Behavioral outcomes and evidence of psychopathology among very low birth weight infants at age 20 years. Pediatrics. 2004 Oct;114(4):932–940.
46. Saigal S, Pinelli J, Hoult L, Kim MM, Boyle M. Psychopathology and social competencies of adolescents who were extremely low birth weight. Pediatrics. 2003 May;111(5 Pt 1):969–975.
47. Darmstadt GL, Bhutta ZA, Cousens S, Adam T, Walker N, de Bernis L. Evidence-based, cost-effective interventions: how many newborn babies can we save? Lancet. 2005 Mar 12-18;365(9463):977–988.
48. Leader S, Yang H, DeVincenzo J, Jacobson P, Marcin JP, Murray DL. Time and out-of-pocket costs associated with respiratory syncytial virus hospitalization of infants. Value Health. 2003 Mar-Apr;6(2):100–106.
49. Stang P, Brandenburg N, Carter B. The economic burden of respiratory syncytial virusassociated bronchiolitis hospitalizations. Arch Pediatr Adolesc Med. 2001 Jan;155(1): 95–96.
50. Waitzman NJ, Romano PS, Scheffler RM. Estimates of the economic costs of birth defects. Inquiry. 1994 Summer;31(2):188–205.
51. Greenough A, Alexander J, Burgess S, Bytham J, Chetcuti PA, Hagan J, et al. Health care utilisation of prematurely born, preschool children related to hospitalisation for RSV infection. Arch Dis Child. 2004 Jul;89(7):673–678.
52. Greenough A, Cox S, Alexander J, Lenney W, Turnbull F, Burgess S, et al. Health care utilisation of infants with chronic lung disease, related to hospitalisation for RSV infection. Arch Dis Child. 2001 Dec;85(6):463–468.
53. Clemens K, Avadi F, Wilber N, Barfield WD. The cost of prematurity: birth to age three early intervention costs [abstract]. Pediatr Res., in press.
Representative terms from entire chapter:
increased access
