Nationwide Outcomes After Thoracoscopic Versus Open Resection of Congenital Pulmonary Airway Malformations in Newborns

Abstract

Purpose:

Elective resection of congenital pulmonary airway malformations (CPAM) has been debated for decades and varies significantly between individual surgeons. However, few studies have compared outcomes and costs associated with thoracoscopic and open thoracotomy approaches on a national level. This study sought to compare nationwide outcomes and resource utilization in infants undergoing elective lung resection for CPAM.

Materials and Methods:

The Nationwide Readmission Database was queried from 2010 to 2014 for newborns who underwent elective surgical resection of CPAM. Patients were stratified by operative approach (thoracoscopic versus open). Demographics, hospital characteristics, and outcomes were analyzed using standard statistical tests.

Results:

A total of 1716 newborns with CPAM were identified. Elective readmission for pulmonary resection was performed in 12% (n = 198), with 63% of resections completed at a different hospital than the newborn stay. Most resections were thoracoscopic (75%), compared to only 25% via thoracotomy. Infants treated with thoracoscopic resection were more often male (78% versus 62% open, P = .040) and were older at the time of resection. Patients who had an open thoracotomy experienced a higher rate of serious complications (40% versus 10% thoracoscopic, P < .001), including postoperative hemorrhage, tension pneumothorax, and pulmonary collapse. Readmission costs were higher for infants treated via thoracotomy (P < .001).

Conclusion:

Thoracoscopic lung resection for CPAM is associated with lower cost and fewer postoperative complications than thoracotomy. Most resections are performed at different hospitals than the place of birth, which may affect long-term outcomes from single institutional studies. These findings may be used to address costs and improve future evaluations of elective CPAM resections.

Introduction

Congenital pulmonary airway malformations (CPAM) are a mass of abnormal and nonfunctional fetal lung tissue that forms during anomalous lung development, occurring with an estimated incidence between 1 in 8000 and 1 in 35,000 live births.¹ These malformations can involve any portion of the lung anatomy, including the trachea (type 0 CPAM), which when involved is incompatible with life.² Many patients who present for possible surgical resection have lower airway lesions involving the bronchi, bronchioles, and/or alveoli. These malformations have been found to increase the risk of infection, malignancy, and pulmonary hypoplasia with resultant pulmonary hypertension.²

Although definitive treatment involves surgical resection, the optimal timing for resection of asymptomatic CPAM has been debated by surgeons for decades. Numerous studies have supported earlier resection given the higher rate of complications associated with delaying surgical treatment, including pneumonia and malignancy.^3,4 Despite this, others argue against asymptomatic lung resection given the risks associated with surgery and the potential for overtreatment.^5,6

With the advent of thoracoscopic resection techniques, few studies have compared the outcomes and costs associated with open thoracotomy and thoracoscopic resections in the newborn population on a national level. Previous studies demonstrate that thoracoscopic resections are associated with improved outcomes when compared to open thoracotomy with regard to length of hospital stay, duration of pleural drainage, and postoperative pulmonary function test scores.^7,8 Thoracoscopic resection also provides the benefits associated with a minimally invasive approach, including improved cosmetic outcomes and a faster recovery.⁹ However, barriers to the widespread use of minimally invasive approaches include the low operative volume of this procedure in neonates at many centers, as well as the potential risk of controlling hemorrhage during thoracoscopic dissection.^9,10

This study sought to compare nationwide outcomes and resource utilization in infants undergoing elective lung resection for CPAM. We hypothesize that elective thoracoscopic pulmonary resection may be associated with fewer postoperative complications and lower overall costs for the treatment of CPAMs in newborns compared to thoracotomy.

Materials and Methods

The Nationwide Readmissions Database (NRD), a part of the Healthcare Cost and Utilization Project (HCUP), comprises information from 15 million admissions over 22 states.¹¹ The NRD contains data from ∼17 million discharges annually and is weighted to estimate roughly 36 million discharges nationally. These data contain demographics, diagnosis, and procedure codes from the International Classification of Disease, Ninth Revision Clinical Modification (ICD9-CM), hospital characteristics, and length of stay (LOS) for each patient encounter. A single hospitalization data sample is created in the NRD when patients have same-day events such as discharge and readmission to a different hospital, or transfers between different hospitals.

Readmissions across subsequent calendar years or different states are not recorded, as the NRD receives annual data from individual State Inpatient Databases. In addition, patients admitted in December are not recorded such that a 30-day readmission rate can be computed. Despite these elements, the NRD remains the most accurate and comprehensive dataset of hospital readmissions data in the United States, making it the ideal database for analyzing nationwide readmissions.

The NRD was queried from 2010 to 2014 to identify newborn patients <1 year old using ICD9-CM codes associated with newborn delivery (supplemental codes V30-39) with a diagnosis of congenital cystic lung (748.4). Patients undergoing pulmonary resection were identified using procedure codes for open (33.1, 32.4, 32.49) or thoracoscopic (32.20, 32.30, 32.41, 34.21) approaches. Patient demographics, medical comorbidities, hospital factors, and outcomes were analyzed. Readmission was defined as admission within 30 days and within the same calendar year of index discharge (overall readmission). ICD9-CM diagnosis and procedure codes are provided by the NRD for index hospitalization and readmissions.

The 2010–2014 study period was selected as it is the most recent contiguous dataset in the NRD utilizing the ICD9 diagnosis code schema before the NRD's change to utilizing ICD10 coding in 2015, and recent work has demonstrated inaccuracy in comparing these coding schemas against each other.¹² Results were weighted for national estimates as per HCUP utilizations guideline. This retrospective comparative analysis was deemed exempt from review by the Institutional Review Board.

Continuous variables are reported as mean ± standard deviation for parametric data or median (interquartile range) for nonparametric data and analyzed using Student's t-test or Mann–Whitney U test, respectively. Categorical variables were compared among groups using chi-squared analysis or Fisher's exact test, as appropriate. IBM SPSS Statistics version 28 (International Business Machines Corp, Armonk, NY, USA) was utilized for statistical analysis. Statistical significance was defined as P < .05.

Results

Cohort demographics

There were 1716 newborns with CPAM identified during the study period (Table 1). The majority (64%) were male with a median age of 5 [4–7] months at the time of surgery. One-fourth of the cohort was born before 37 weeks gestational age, and 20% had concomitant major congenital cardiac anomalies. Eighteen percent (n = 308) underwent resection, the majority (n = 198) of which were performed upon elective readmission. Thoracoscopic approach was utilized more frequently (56%) than open thoracotomy (44%) for pulmonary resection, and the majority of patients were routinely discharged home (88%). Most patients were treated at large (78%), metropolitan teaching hospitals (87%), and most patients utilized public insurance (48%).

Table 1.

Characteristics of Infants Undergoing Surgical Management for Congenital Pulmonary Airway Malformations

Characteristics	n (%)
All patients	1716 (100)
Age (months)^a	5 [4–7]
Sex
Male	1093 (64)
Female	623 (36)
Lung resection admission
Index	125 (7)
Readmission	198 (12)
Lung resection type	308 (18)
Thoracoscopic	172 (56)
Open	137 (44)
Index LOS^a	17 [2–17]
Index LOS >7 days	625 (36)
Index admission mortality	65 (4)
Associated diagnoses and comorbidities
Premature (<37 weeks gestational age)	427 (25)
Major cardiac anomalies	345 (20)
Patent ductus arteriosus	245 (14)
Respiratory distress syndrome	255 (15)
Median household income quartile
First (lowest)	371 (22)
Second	463 (27)
Third	520 (30)
Fourth (highest)	359 (21)
Primary expected payer
Public^b	814 (48)
Private	748 (44)
Self-pay/other	143 (8)
Discharge disposition
Home (routine)	1509 (88)
Home with home health care	90 (5)
Transfer to short term hospital	40 (2)
Skill nursing facility	^*
Unknown	^*
Hospital ownership
Not-for-profit	1345 (78)
Public	339 (20)
Other	32 (2)
Teaching status
Metropolitan teaching	1499 (87)
Metropolitan nonteaching	208 (12)
Nonmetropolitan	^*
Hospital bed size
Large	1346 (78)
Medium	273 (16)
Small	96 (6)

Cells marked with an asterisk (^*) represent actual values censored from publication in accordance with the Health care Cost and Utilization Project Data Use Agreement.

Data presented as median [interquartile range].

Medicaid.

LOS, length of stay.

Readmission characteristics of newborns with CPAMs

Overall, 19% of patients were readmitted during the study period. The majority of readmissions (65%) were elective, and most patients were readmitted for pulmonary resection (61%) (Table 2). One-third of readmissions occurred within 30 days of the index hospitalization. Fifty-one percent of patients were readmitted to hospitals different than the newborn admission. For those not undergoing elective resection, the readmission rate was 8%, of which 4% were for respiratory complications such as pneumonia or apparent life-threatening events.

Table 2.

Readmission Characteristics of Newborns Undergoing Surgery for Congenital Pulmonary Airway Malformations

Characteristics	n (%)
Readmission type^a
Elective	212 (65)
Unplanned	113 (35)
Readmitted within 30 days	106 (6)
Readmitted within 30 days DH^a	46 (43)
Overall readmission	325 (19)
Overall readmission DH^a	167 (51)
Elective resection	198 (19)
Excluding elective resections
Readmitted within 30 days	84 (6)
Overall readmission	113 (8)
Respiratory (pneumonia, ALTE)	60 (4)
Feeding issue/FTT	20 (1)
Dehydration/electrolyte disorder	29 (2)

Denominator calculated as percent of readmission.

ALTE, apparent life-threatening event; DH, different hospital; FTT, failure to thrive.

Index and readmission characteristics of newborns undergoing elective CPAM resection during readmission

There were 198 patients who underwent elective readmission for pulmonary resections (Tables 3 and 4). The majority of newborns receiving elective resection were treated via thoracoscopic (75%) approach. Those treated by thoracoscopic CPAM resection were more often male (78% versus 62% open) and slightly older at the time of resection (138 versus 128 days open), both P < .05. Patients treated by thoracoscopic resection had a significantly higher rate of private insurance (82% versus 49% open) and were more frequently from the top household income quartiles (64% versus 48% open), both P < .001. All patients in the open thoracotomy group were treated at large (100%) hospitals with no difference in hospital teaching status in either group.

Table 3.

Index Admission Characteristics of Infants Receiving Elective Surgical Treatment for Congenital Pulmonary Airway Malformations on Readmission

Characteristics	Thoracoscopic n = 148 (75%)	Open n = 50 (25%)	P
Age at resection (days)^a	138 [133–206]	128 [69–203]	.009
Sex			.040
Male	115 (78)	31 (62)
Female	33 (22)	19 (38)
Additional diagnoses
Major cardiac anomalies	21 (14)	12 (24)	.126
Other circulatory anomalies	20 (13)	0 (0)	.005
Prematurity	20 (14)	11 (22)	.153
LBW, VLBW, or ELBW	15 (10)	11 (22)	.050
Respiratory distress syndrome of newborn	25 (17)	^*	.166
Insurance type			<.001
Public^b	26 (18)	26 (51)
Private	112 (82)	24 (49)
Median household income quartiles			<.001
Bottom half	52 (35)	26 (52)
Top half	96 (65)	24 (48)
Hospital ownership			.043
Not-for-profit	43 (29)	22 (44)
Public	96 (65)	28 (56)
Other	^*	0 (0)
Hospital bed size			<.001
Large	111 (75)	50 (100)
Medium	17 (12)	0 (0)
Small	20 (14)	0 (0)
Teaching status			.398
Metropolitan teaching	124 (84)	40 (78)
Metropolitan nonteaching	24 (16)	11 (22)

Bolded values represent statistical significance set at P < 0.05.

Cells marked with an asterisk (^*) represent actual values censored from publication in accordance with the Health care Cost and Utilization Project Data Use Agreement.

Data presented as median [interquartile range].

Medicaid.

ELBW, extremely low birth weight; LBW, low birth weight; VLBW, very-low birth weight.

Table 4.

Readmission Characteristics of Infants Receiving Elective Surgical Treatment for Congenital Pulmonary Airway Malformations on Readmission

Characteristics	Thoracoscopic n = 148 (75%)	Open n = 50 (25%)	P
Days to readmission (RA)^a	137 [127–192]	168 [67–201]	.558
Readmission within 30 days	12 (8)	11 (22)	.013
Overall readmission DH	94 (64)	31 (62)	.867
Readmission complication
Major complication^b	14 (10)	20 (40)	<.001
Any complication^c	51 (35)	41 (80)	<.001
Other infections	0 (0)	0 (0)	n/a
Pneumonia	0 (0)	0 (0)	n/a
Readmission cost^a	$17,686 [$14,608–$27,609]	$21,325[$12,128–$48,979]	.081
Readmission to DH Cost^a	$17,087 [$13,073–$22,824]	$48,979 [$15,783–$66,274]	<.001

Bolded values represent statistical significance set at P < 0.05.

Data presented as median [interquartile range].

Bleeding, pulmonary collapse, tension pneumothorax.

Pneumothorax, air leak, subcutaneous emphysema, or other major complication.

DH, different hospital.

There was no difference in time to readmission between either group (Table 4). Those treated by an open approach were more frequently readmitted within 30 days (22% versus 8% thoracoscopic) for their resection. The majority of both the thoracoscopic group (64%) and open (62%) group were readmitted at a hospital different than their index hospitalization. Serious readmission complications, including bleeding, pulmonary collapse, and tension pneumothorax, were significantly higher among patients receiving open resections (40%) compared to the thoracoscopic group (10%), P < .001. Readmission costs were similar, although costs of readmission to a different hospital were higher with an open approach.

Discussion

Overall, this analysis represents one of the largest studies to examine nationwide outcomes of CPAM resection in newborns. Most resections were performed thoracoscopically during elective readmissions, with the majority at hospitals different than that of their newborn hospitalizations. Newborns from families in the top half of median household incomes and those with private insurance more frequently received thoracoscopic resection compared to open thoracotomy. Those who underwent open pulmonary resections had a higher rate of complications, including hemorrhage, pulmonary collapse, and tension pneumothorax. Patients who underwent open procedures incurred higher overall readmission costs.

Given the risks associated with congenital lung lesions, including respiratory compromise, recurrent pulmonary infections, and malignancy, surgical resection remains the mainstay of treatment for these lesions even when patients are asymptomatic.^3,13–15 Historically, thoracotomy was the traditional approach due, in part, to its effectiveness in achieving a complete resection and that it does not necessitate lung isolation.¹⁶ However, substantial morbidity is associated with this approach, with a growing body of evidence identifying long-term musculoskeletal postoperative complications in children, including chest wall deformities, scoliosis, and asymmetry of the thoracic wall and shoulder girdle.^17,18 Rothenberg first reported success with thoracoscopic lobectomy in infants, citing advantages over the traditional open approach, including reductions in postoperative pain, recovery time, and length of stay.¹⁹ Subsequent series have added evidence for improved cosmetic outcomes over open and muscle-sparing thoracotomy incisions, as well as a decreased incidence of long-term musculoskeletal sequelae.^17,20–22

Thoracoscopic approaches were utilized more frequently than thoracotomy for elective pulmonary resection of CPAMs in this nationwide sample. Although the complication rate, including bleeding, pulmonary collapse, and tension pneumothorax, was somewhat higher than those reported in other series, the overall trend of fewer complications in the thoracoscopic group is in-line with previous reports.^{9,17,21,23,24} One reservation cited by those favoring thoracotomy is the potential risk of being unable to obtain appropriate vascular control during resection compared to open surgery.^10,25 However, this study found that the postoperative bleeding rate was higher among patients receiving thoracotomy compared to thoracoscopic resection, which is also in line with recent series demonstrating reduced blood loss associated with thoracoscopic approaches.²⁶

Alternatively, it may be that patients with more medical comorbidities or with lesions less amenable to thoracoscopic approaches were selected for thoracotomy. However, we observed a similar rate of characteristics such as major cardiac anomalies, low birth weight, and newborn respiratory distress syndrome between both groups. Overall, these data support the national trend toward increased utilization of thoracoscopy for elective resection of CPAMs in newborns and is in line with recent multi-institution studies.^22,24,27 Interestingly, the majority of both surgical cohorts in this study were readmitted to a hospital different than their newborn stay to undergo resection. Given the low incidence of these lesions and operations,¹ this finding represents an important consideration for future single-institution studies of CPAM that should carefully examine longitudinal follow-up patterns of their newborn patients with congenital lung lesions.

Socioeconomic inequalities have been associated with differences in overall outcomes and time to initial disease presentation for many surgical diseases.^28–31 Disparities in the utilization of thoracoscopic compared to open resection existed in this newborn population. Patients from low-income households received a significantly higher rate of open thoracotomy resections compared to their counterparts from high-income households. Patients who underwent open surgeries experienced a higher number of complications during their operative hospitalization. Although limited studies have examined socioeconomic disparities for pediatric patients undergoing thoracic surgeries, other series have demonstrated correspondingly poor outcomes in adult patients from lower-income households undergoing pulmonary resections for oncologic indications.^32,33 One pediatric study by Wagenaar et al. demonstrated that payer status was associated with in-hospital survival rates and resource utilization among children receiving open and thoracoscopic resection of pediatric lung malformations utilizing the Kids' Inpatient Database.²⁵

Although there were no inpatient mortalities in either group in our study to examine in relationship to payer status, a significantly higher ratio of patients with private insurance were treated thoracoscopically. Furthermore, patients treated via thoracoscopic approaches were more frequently from the top 50% of median household incomes compared to their counterparts receiving thoracotomy. Patients treated by thoracoscopic approaches incurred lower readmission costs, which aligns with literature in adult studies supporting lower overall hospitalization costs associated with thoracoscopy utilization.³⁴ Given the higher rate of postoperative complications and readmission costs observed in newborns receiving thoracotomy in this study, surgeons should be cognizant of the total management costs and resource allocation associated with selecting individual treatment strategies, especially in groups from low-income households or with public or self-pay insurance status.

There were several significant limitations in the present study. A major limitation of this and other retrospective administrative database studies is the lack of granular detail related to individual patients' and surgeons' operative details. For example, previous studies have demonstrated that operative time is inversely correlated with individual surgeon experience with thoracoscopy and is significantly associated with clinical outcomes.^9,21 Furthermore, patient characteristics such as weight at the time of surgery as well as the anatomic location of the surgery can affect perioperative outcomes and influence operative approach.^35–37 Recent improvements in prenatal ultrasound have allowed for antenatal diagnosis of CPAM and other congenital lesions, which can influence the timing of surgery and treatment in the postnatal period.⁶ Given that this study utilized ICD-9 diagnosis codes, it was not possible to evaluate these factors or identify if patients were symptomatic at the time of resection.

Another significant limitation of this study was the age of the study period chosen (2010–2014), which may underestimate the increased use of thoracoscopy for this operation. After 2014, the NRD converted from the ICD9-CM to ICD10-CM coding schema for the 2015–2019 dataset releases. The ICD10-CM coding system uses a substantially higher number of diagnosis codes, which can raise concern for miscoding and prevents accurate comparison of data samples using different coding schemas.¹² Furthermore, delays in surgical timing and utilization of minimally invasive approaches due to the COVID-19 pandemic incurred widespread downstream effects on pediatric patients access to surgery services from 2019 to the present, which has not been characterized in patients undergoing CPAM resection.^38–40 As a result, the 2010–2014 time period was selected as the most recent, contiguous dataset releases using one coding schema without confounding effects due to the COVID-19 pandemic.

Given the low incidence of CPAM cases and patients undergoing elective resection, this constrained our ability to perform more complex multivariate models to identify prognostic factors associated with resection type. As with any retrospective data repository, the NRD may include errors correlated with data collection and entry as well as incorrect ICD-9 coding. In addition, patients cannot be followed across subsequent calendar years or when readmitted in different states. However, previous HCUP reports cite that <5% of all patients are readmitted across other states.¹¹

Conclusions

This study is one of the largest series to examine nationwide outcomes of elective CPAM resection in newborns. Most patients are undergoing elective resection at different hospitals than the newborn hospitalization. Thoracoscopic resection is associated with lower complication rates and cost. Although thoracoscopy is more common, there are socioeconomic disparities in its utilization. Ultimately, this information should be utilized by surgeons to address costs, minimize utilization disparities, and improve future single-institution studies of elective CPAM resections.

Footnotes

Authors' Contributions

Study Conception and Design: C.T.H., J.E.S., E.A.P., and C.M.T. Acquisition of Data: C.T.H., J.P.P., and C.M.T. Analysis and Interpretation of Data: C.T.H., C.M.T., J.E.S., and E.A.P. Drafting of article: C.T.H., S.C.C., and C.M.T. Critical Revision of article: All authors.

Disclosure Statement

No competing financial interests exist.

Funding Information

No funding was received for this article.

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