Health Care and Societal Costs of Bronchopulmonary Dysplasia Wannasiri Lapcharoensap, MD,* Henry C. Lee, MD MS, † Amy Nyberg, BS, ‡ Dmitry Dukhovny, MD MPH* *Department of Pediatrics, Oregon Health and Science University, Portland, OR † Department of Pediatrics, Stanford University, Stanford, CA ‡ March of Dimes NICU Family Support Coordinator, Helen DeVos Children’s Hospital, Grand Rapids, MI Education Gaps Bronchopulmonary dysplasia (BPD) continues to affect a large portion of very low-birthweight infants; therefore, knowledge of the short- and long-term economic impact of BPD is necessary to gain a broader perspective of the disease. Abstract Despite significant technological advances and increasing survival of premature infants, bronchopulmonary dysplasia (BPD) continues to be the most prevalent major morbidity in surviving very low-birthweight infants. Infants with BPD are often sicker, require longer stays in the NICU, and accumulate greater hospital costs. However, care of the infant with BPD extends beyond the time spent in the NICU. This article reviews the costs of BPD in the health-care setting, during the initial hospitalization and beyond, and the long-term neurodevelopmental impact of BPD, as well as the impact on a family caring for a child with BPD. Objectives After completing this article, readers should be able to: 1. Describe the short- and long-term costs for infants with bronchopulmonary dysplasic (BPD). 2. Describe the impact of BPD on the health-care system. 3. Compare the costs of infants and children with BPD to those without BPD. 4. Discuss the impact of BPD on the child’s neurodevelopment. 5. Estimate the burden of BPD on the family unit. INTRODUCTION Approximately 1 in 10 infants is born prematurely in the United States. (1) In the year 2015 alone, there were more than 55,000 very low-birthweight (VLBW, <1,500 g) infant births. Although overall survival rates are improving, the number AUTHOR DISCLOSURE Drs Lapcharoensap, Dukhovny, and Ms Nyberg have disclosed no financial relationships relevant to this article. Dr Lee has disclosed that he is the principal investigator on a grant from the Eunice Kennedy Shriver National Institute of Child Health and Human Development (R01 HD087425) that, in part, supports this research. This commentary does not contain a discussion of an unapproved/investigative use of a commercial product/device. ABBREVIATIONS BPD bronchopulmonary dysplasia GA gestational age RSV respiratory syncytial virus VLBW very low-birthweight Vol. 19 No. 4 APRIL 2018 e211 by guest on April 2, 2018 http://neoreviews.aappublications.org/ Downloaded from
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Health Care and Societal Costs ofBronchopulmonary Dysplasia
Wannasiri Lapcharoensap, MD,* Henry C. Lee, MD MS,† Amy Nyberg, BS,‡ Dmitry Dukhovny, MD MPH*
*Department of Pediatrics, Oregon Health and Science University, Portland, OR†Department of Pediatrics, Stanford University, Stanford, CA
‡March of Dimes NICU Family Support Coordinator, Helen DeVos Children’s Hospital, Grand Rapids, MI
Education Gaps
Bronchopulmonary dysplasia (BPD) continues to affect a large portion of very
low-birthweight infants; therefore, knowledge of the short- and long-term
economic impact of BPD is necessary togain abroader perspectiveof thedisease.
Abstract
Despite significant technological advances and increasing survival of
premature infants, bronchopulmonary dysplasia (BPD) continues to be the
most prevalent major morbidity in surviving very low-birthweight infants.
Infants with BPD are often sicker, require longer stays in the NICU, and
accumulate greater hospital costs. However, care of the infant with BPD
extends beyond the time spent in the NICU. This article reviews the costs of
BPD in the health-care setting, during the initial hospitalization and beyond,
and the long-term neurodevelopmental impact of BPD, as well as the impact
on a family caring for a child with BPD.
Objectives After completing this article, readers should be able to:
1. Describe the short- and long-term costs for infants with
bronchopulmonary dysplasic (BPD).
2. Describe the impact of BPD on the health-care system.
3. Compare the costs of infants and children with BPD to those without BPD.
4. Discuss the impact of BPD on the child’s neurodevelopment.
5. Estimate the burden of BPD on the family unit.
INTRODUCTION
Approximately 1 in 10 infants is born prematurely in the United States. (1) In the
year 2015 alone, there were more than 55,000 very low-birthweight (VLBW,
<1,500 g) infant births. Although overall survival rates are improving, the number
AUTHOR DISCLOSURE Drs Lapcharoensap,Dukhovny, and Ms Nyberg have disclosed nofinancial relationships relevant to this article.Dr Lee has disclosed that he is the principalinvestigator on a grant from the EuniceKennedy Shriver National Institute of ChildHealth and Human Development (R01HD087425) that, in part, supports thisresearch. This commentary does not contain adiscussion of an unapproved/investigativeuse of a commercial product/device.
ABBREVIATIONS
BPD bronchopulmonary dysplasia
GA gestational age
RSV respiratory syncytial virus
VLBW very low-birthweight
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remains an expensive therapy to administer and cost-
effectiveness for various populations remains in ques-
tion, though a stronger case can be made for preterm
infants with BPD. (23)
Beyond the Second Year: Rehospitalizations andOutpatient VisitsRehospitalization rates in patients with BPD continue to
be comparatively higher beyond the initial year with an
increased risk for rehospitalization and more frequent out-
patient visits. (13) Although most infants will not require
oxygen beyond 2 years of age, indicating clinical improve-
ment, up to 25% will continue to have respiratory com-
plications into young adulthood. (12)(24) Children and
adolescents with a history of BPD are more often diagnosed
with asthma (26.9% vs 11.8%; P<.0001) and psychiatric
illnesses (such as depression, anxiety, attention-deficit/
hyperactivity disorder) leading to increased health-care uti-
lization. (25) In a Quebec study comparing a preterm BPD
cohort and a preterm respiratory distress syndrome cohort
over 16 to 25 years, the costs of medical services and total
health-care costs per person annually was significantly
increased in the BPD cohort ($13,472 vs $10,719; P¼.02;
expressed in 2008 Canadian dollars). (25)
Durable Medical EquipmentIn a multicenter study, approximately one-third of infants
with BPD of any severity were sent home with oxygen
TABLE. Selected Studies Outlining the Resource Utilization and/or CostAssociated With BPD
REFERENCEYEARPUBLISHED POPULATION TIME HORIZON RESOURCES COSTS/CHARGES NOTES
Patel et al (26) 2016 N¼254VLBWSource:Prospectivesingle center,admitted to theNICU 2008-2012
Country: USA
Birthhospitalization
Although the LOSis not directlyreported, theauthors do pro-vide a cost perday and thus itcould be calcu-lated; addition-ally, there is areport onbreakdown ofdifferent ser-vices usedbetween the 2groups (seeTable 3 in Patelet al.)
Cost/charges: CostsCurrency: US dollars, 2014Median cost for BPD $269,004(IQR:204,606-331,552)compared to $117,078 (IQR90,496-162,017) (P<.001)
In an adjusted analysis, BPDincreased costs by$41,929 compared tonon-BPD patients
Source of costs: hospital dataand cost accounting sys-tem includes direct andindirect hospital costs
Facility fees: includedProfessional fees: included(based on physicianpayments)
Parent costs: not includedOther: N/A
Prospective cohortTable 3 of this article byPatel et al provides adetailed descriptionof the total costsand the individualsub-costs (e.g. hos-pital direct costs,physician costs, andNICU cost per day)
Adjustment: pro-pensity score forBPD, race/ethnicity,gender, gestationalage, and small forgestational age
Johnsonet al (7)
2013 N¼425VLBWSource: Singlecenter
Country: USA
Birthhospitalization
LOS:BPD: Mean 94–31days comparedto 46–19 daysw/o BPD(P<.001)
Cost/charges: CostsCurrency: US dollars, 2009Mean 103,151–43,482 com-pared to 44,465–23,300(P<.001)
BPD cost is $31,565 highercompared to no morbiditywhen adjusted for GA, sex,birthweight, race/ethnicity,and primary payer (P<.001)
Source of costs: Hospital dataand cost accounting sys-tem using direct hospitalcosts for each billable item
Facility fees: includedProfessional fees: Not includedParent costs: Not includedOther: N/A
Adjusted for birth-weight, GA, andsociodemographiccharacteristics
Continued
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NIS samples 20% ofUS hospital dis-charges and isweighted toapproximatepopulation
BPD identified usingthe ICD-9 code770.7
Limited to patients<1,500 g birth-weight and thosewho received non-invasive mechanicalventilation or con-tinuous invasivemechanical ventila-tion ‡96 hoursbased on procedurecodes
Both univariate andmultivariable analy-ses performed (4different modeladjustments weredone)
Goal of the study wasto assess trends overtime for incidenceof BPD, LOS, andcharges
Continued
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LOS was deter-mined in thisstudy, but notdirectly com-paring thosewith BPD tothose without(however, thereis LOS compar-ing normalnewborns topreterm/lowbirthweight)
Cost/charges: Costs (chargeswere converted to costsusing cost-to-chargeratios)
Currency: US dollars, 2001(presumed year, notexplicitly stated)
Mean 116,000 (95%CI¼102,200–129,900; me-dian¼102,000) for BPDcompared to 16,900 (95%CI¼15,200–18,600; me-dian¼6,300) without BPD
NIS samples 20% ofUS hospital dis-charges and isweighted toapproximatepopulation
Attempted to mini-mize the overlapfrom inter-hospitaltransfers during thebirth admission(80% of birthsinvolved no hospitaltransfer)
BPD identified usingthe ICD-9 code770.7
All patients who weredefined as preterm/low birthweight(<2,500 g) wereincluded in thisanalysis likelydecreasing themean/median costof hospitalization inthe non-BPD group
Limited to those whosurvived to firstbirthday, had fol-low-up care atKaiser, and did nothave any majoranomalies
Table 1 of the studyprovides additionalinformation onresource utilizationduring the birthhospitalization (eg,duration of ventila-tor and oxygen use;% of comorbiditiessuch as NEC, ROP,and IVH)
No risk factors identi-fied among patientswith BPD who wereand were notreadmitted
Continued
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Combination of hos-pital records aswell as parentalrecords andregional chargeswere used
Distribution of costs82% – hospital charges12% – physician fees5% – community hos-pital charges
1% – parentalexpenses
Interviews of parentsprovided parentalexpenses duringinfanthospitalization
Financial and emo-tional impact ofhome care was as-sessed via aquestionnaire
Home oxygen therapycosts were obtainedas average chargesfrom suppliers in theregion
The table breaks down some of the elements of costs and summarizes the economic burden. BPD¼bronchopulmonary dysplasia; ED¼emergencydepartment; GA¼gestational age; HCUP¼Healthcare Cost and Utilization Project; ICD-9¼International Classification of Diseases, Ninth Revision;IQR¼interquartile range; IVH¼intraventricular hemorrhage; LOS¼length of stay; N/A¼not available; NEC¼necrotizing enterocolitis; NHS¼ NationalHealth Service; NIS¼National Inpatient Sample; RDS¼respiratory distress syndrome; ROP¼retinopathy of prematurity; RSV¼respiratory syncytial virus;VLBW¼ very low birthweight.aRepresents the same cohort of patients with different focus and period of analysis.
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terventions), the return of investment calculus should
take into account the annual economic impact of 12,100
infants diagnosed with BPD (22% of VLBWs, low esti-
mate) per year in the United States and their lifetime
“cost” to those individuals and their families, health-care
system, and society.
ACKNOWLEDGMENTS
The content is solely the responsibility of the authors and does
not necessarily represent the official views of the National
Institutes of Health. Additionally, we would like to thank the
VanDis family for sharing their personal experience of having
infants in the NICU.
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Figure. Chris and Denise Van Dis with their twins, Natalie and Caleb, whowere born at 24 weeks’ gestation and had bronchopulmonary dysplasia.Click this link (https://youtu.be/TRGKWm2DBbA) to watch a 6-minutevideo of the parents’ account of their personal experience of bringinghome 2 former preterm infants with BPD.
American Board of PediatricsNeonatal-Perinatal ContentSpecification• Know the prognosis, long-term complications, and permanentsequelae of bronchopulmonary dysplasia.
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1. Your NICU is forming a multidisciplinary team specifically focused on the care of the verypreterm neonate. As you plan for resource allocation, you are reviewing studies on verylow-birthweight infants. Which of the following morbidities experienced by these infantshas been associated with the highest mean marginal cost?
2. A 25-week gestational age infant is nearing discharge after a prolonged initial NICU stay.The infant was diagnosed as having BPD at 36 weeks’ postmenstrual age, but is nowbreathing room air. As you counsel the parents on what to expect in the coming months,they ask about the possibility of rehospitalization. Which of the following statementsregarding preterm infants and hospital readmission is correct?
A. Respiratory illnesses are the number 1 reason for hospital readmissions in pre-mature infants, and patients with BPD are at higher risk for such readmissions.
B. Although prolonged NICU hospitalizations are not desirable, a benefit of a longhospital course in very preterm infants is that their risk of readmission within thefirst year is lower than that for term or late preterm infants.
C. Although preterm infants are just as likely to have illnesses that might lead tohospitalization, the rates of hospital readmission in the first year are similar to orless frequent than term infants, due to parental avoidance of the health-caresystem.
D. BPD is not an independent risk factor for increased readmission during the first yearafter birth.
E. The main reason for readmission in infants with BPD is patent ductus arteriosusligation.
3. As a premature infant is getting ready for discharge, you are counseling the parents aboutthe potential benefits of palivizumab for respiratory syncytial virus (RSV) prophylaxis.Which of the following statements regarding this practice is correct?
A. RSV has a consistent risk profile for both term and preterm infants during the firstyear, with similar rates of hospitalization during each month after birth.
B. Although RSV infection can be more serious in infants with BPD, hospitalization foran RSV infection is now rare, because of the growing efficacy of herd immunity fromvaccinations.
C. It is recognized that BPD is a major independent risk factor for RSV disease leadingto serious infections and hospitalizations.
D. Although RSV infection can cause serious illness in infancy, there are no associatedlong-term effects, other than recurrent wheezing which may last up to about 3years of age.
E. Palivizumab is recommended for all preterm infants of less than 34 weeks’ ges-tational age regardless of whether there was a history of BPD.
4. A female infant born at 25 weeks’ gestational age is now 2 years old and being seen in thefollow-up clinic. The infant had a history of BPD and went home receiving oxygen therapy,but oxygen therapy was discontinued since 6 months of age. She is now meetingdevelopmental milestones, with no hospitalizations in the past year. Which of thefollowing statements regarding this and similar patients is correct?
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A. This patient is not truly categorized as having BPD, because she does not requireoxygen at 2 years of age.
B. Children with a history of BPD are more often diagnosed with asthma and psy-chiatric illnesses such as depression and anxiety.
C. Rates of readmissions to the hospital and outpatient visits for patients with BPDbecome similar to the general population after the first year.
D. In most population-based cohort studies, up to 75% of patients diagnosed withBPD in the NICU will require oxygen again in childhood after they turn 2 years old.
E. By young adulthood (w18 to 22 years of age), the rates of respiratory complicationsin patients who had BPD are less than 5%.
5. An infant born at 25 weeks’ gestational age has BPD and is now 4 months old in the NICU.He continues to require oxygen therapy, with occasional need for positive pressureventilation. As you are discussing further management, you discuss the possibility oftracheostomy. Which of the following statements regarding supplemental oxygen andother therapies for patients with BPD is correct?
A. Although tracheostomy is a relatively common procedure in the NICU (w10% of allpatients), it is relatively less common for patients with BPD (w1% of infants withsevere BPD) because of the transient nature of the disease.
B. Tracheostomy may be a result of pulmonary disease dependent on positivepressure ventilation, consequences of prolonged intubation (such as subglotticstenosis, vocal cord paralysis, granulation tissue), or airway anomalies (such aslaryngotracheomalacia).
C. Most tracheostomies for this condition are short-lived, with median time todecannulation being 30 to 45 days, depending on the cohort.
D. Health-care utilization in the 5 years after tracheostomy placement is less than thatamong those who do not receive tracheostomy, due to the benefit of relativestability of airway management and decreased need for acute care hospitalization.
E. Although there is an added immediate cost for the procedure of tracheostomy,there is generally an overall cost savings in early infancy (w0 to 5 years of age) forboth direct and indirect costs, due to reduction in readmission after the procedure.
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Wannasiri Lapcharoensap, Henry C. Lee, Amy Nyberg and Dmitry DukhovnyHealth Care and Societal Costs of Bronchopulmonary Dysplasia
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