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J O U R N A L O f s U R G ic A L R E sEAR C h ● JANUA R y 2021 (257) 379

e3 8 8

Available online at www.sciencedirect.com

ScienceDirect

journal homepage: www.JournalofSurgicalResearch.com

Relationships Between Hospital and Surgeon


Operative Volumes and Surgical Outcomes in
Hirschsprungs Disease

Jordan C. Apfeld, MD,a Richard J. Wood, MD,b Devin R. Halleran, MD,a


Katherine J. Deans, MD, MHSc,a,b Peter C. Minneci, MD, MHSc,a,b
and Jennifer N. Cooper, PhDa,*
a
Center for INNOVATION in PEDIATRIC PRACTICE, The RESEARCH Institute AT NATIONWIDE Childrens HOSPITAL, Columbus,
Ohio
b
DEPARTMEnt of Surgery, NATIONWIDE Childrens HOSPITAL, Columbus, Ohio

A R T I C L E I N F O A B S T R A C T

Article history:
BACKGROUND: The impact of surgical volume on outcomes in Hirschsprung’s disease (HD)
Received 18 February 2020
remains uninvestigated. We aimed to determine whether higher surgeon and hospital-
Received in revised form
level HD operative volumes are associated with improved surgical outcomes following
14 July 2020
primary surgery for neonatal HD.
Accepted 2 August 2020
MATERIALS AND methods: Neonates who underwent either an ostomy or pull-through (PT)
Available online xxx
procedure for HD before 60 d of life and a PT procedure by age 1 y were identified in
the Pediatric Health Information System (PHIS). Index admissions from January
Keywords:
2000 to September 2012 across 41 tertiary childrens hospitals were included. Surgeon and
Enterocolitis
hospital- level HD operative volume were defined as the average annual number of PT
Hirschsprung’s disease
procedures performed for HD in the 2 y preceding each included case. We examined the
Pediatric surgery
relationship between operative volumes and all-cause readmission, readmission for
Neonatology
Hirschsprung’s associated enterocolitis (HAEC), and rates of reoperation within 30 d and
Pull-through procedure
2 y.
Volume-outcome
Results: A total of 1268 infants were included. There were 218 patients (17.2%)
readmitted to the hospital within 30 d and 540 (42.6%) within 2 y. A total of 119 patients
(9.4%) had HAEC- related readmission within 30 d, and 271 (21.4%) had HAEC-related
readmission within 2 y. A total of 57 patients (4.5%) had a reoperation within 30 d and 129
(10.2%) within 2 y. In risk- adjusted analyses, there were no significant associations
between either surgeon or hos- pital HD operative volumes and readmission/reoperation
rates within 30 d or 2 y.
Conclusions: Neither surgeon nor hospital PT volumes were significantly associated
with readmission or reoperation rates for infants with Hirschsprung’s disease. Future
work is needed to evaluate whether operative volumes are associated with functional
outcomes following PT for HD.
ª 2020 Elsevier Inc. All rights reserved.
* Corresponding AUTHOR. Center for Surgical Outcomes Research and Center for Innovation in Pediatric Practice, The Research Institute at
Nationwide Childrens Hospital, 700 Children’s Drive, FB Suite 3A.3, Columbus, OH, 43205. Tel.: þ1614 355-4526; fax: þ1614 722-3544.
E-mail address: jennifer.cooper@nationwidechildrens.org (J.N. Cooper).
0022-4804/$ e see front matter ª 2020 Elsevier Inc. All rights
reserved. https://doi.org/10.1016/j.jss.2020.08.014
380 R C h ● J A NUARY 2 0 2 1 ( 2 5 7 ) 3 7 9 e3 8 8

Introduction with an International Classification of Diseases, ninth edition,


Clinical Modification (ICD-9-CM) diagnosis code for Hirsch-
Hirschsprung’s disease (HD) is a rare congenital anomaly (1 in sprung’s Disease (751.3), who were aged ≤60 d at the time of
5000 live births) associated with the failure of neural crest their admission for an ostomy procedure or pull-through
cells to migrate along the full length of the large intestine.1 procedure, and who were cared for at a hospital that began
The definitive treatment of Hirschsprung’s disease involves contributing inpatient data to PHIS at least 2 y before the
a surgical “pull-through” (PT) procedure, where the included admission. Primary surgeries for HD included pull-
diseased segment of distal intestine is removed, and healthy through (ICD-9-CM codes 48.41, 48.65, 48.49, 45.95, 48.40,
inner- vated intestine is brought down and connected above 48.43, 48.5, 48.50, 48.51, 48.52, 48.59, 48.42, 48.69, 48.61, 45.90,
the anal opening.2 Perioperative complications are common 45.92, 45.93, 45.94) and stoma/ostomy creation (ICD-9-CM
and include preoperative Hirschsprung’s-associated codes 46.03, 46.10, 46.11, 46.13, 46.20, 46.21, 46.23, 46.40,
enterocolitis (HAEC, estimated incidence 6 to 60%), 46.41, 46.43). We excluded patients who had both a pull-
postoperative HAEC (25%-37%), need for reoperation or through or total colectomy and an ostomy procedure docu-
repeat PT procedure (5%- 15%), and readmissions for other mented on the date of their initial HD-related surgery. We
reasons including inconti- nence (5%-25%) and constipation also excluded patients with particular data quality issues.
(10%-30%).3-13 Furthermore, long-term functional outcomes First, we excluded patients with duplicate MRNs or incorrect
have not been well-studied.14 Higher surgeon and hospital dates of service. We then excluded patients whose surgeon
operative volume have been associated with improved was missing a physician identification number or whose
outcomes in high-risk adult surgical procedures.15-17 surgeon did not have operative cases recorded in PHIS in at
Relationships between operative volumes and outcomes in least 22 of the preceding 24 mo. This latter exclusion was
neonatal and pediatric surgery remain an ongoing debate. If necessary to ensure that a surgeons annual PT volume in the
volume-outcomes relationships do exist, then local pediatric years pre- ceding each included case could be accurately
surgical subspecialization or regionaliza- tion of care may be determined. We did, however, include these latter two
warranted.18-25 However, given the rarity of congenital groups in calculations of hospital PT volumes.
surgical anomalies, future recommendations must take into
account how regionalization of care in the United States
could limit families’ access to care and could Definitions of hospital and surgeon volumes
limit pediatric surgical trainee case exposure. 26-29
Relationships between hospital or surgeon operative vol- Surgeon HD operative volume was defined as the average
ume and outcomes in neonatal and pediatric surgery have annual number of PT procedures performed by the surgeon
been reported in several studies examining surgical repair of in patients of any age during the preceding 2 y. This average
biliary atresia (BA), craniosynostosis, cleft lip, congenital dia- was taken over 2 y rather than 1 y in order to smooth out
phragmatic hernia (CDH), gastroschisis, pyloric stenosis, and year-to- year variation in volume while minimizing the
thyroidectomy.30-40 However, other studies of these same number of patients excluded due to their surgeons cases not
procedures, namely surgical repair of BA, CDH, and gastro- being identifiable in the PHIS database during the entire look-
schisis, and a recent study examining repair of esophageal back period.47 Hospital volume was defined similarly but at
atresia/tracheoesophageal fistula (EA/TEF), have failed to the hospital level.
show such a relationship. 32,40-47 Outcomes after Hirsch-
sprungs PT may depend on surgical and pathological exper-
Patient characteristics and outcomes
tise, as well as perioperative care, and therefore, may be
related to surgeon or hospital experience. 48-50 We aimed to
Evaluated baseline patient clinical, sociodemographic, and
determine whether surgeon or hospital-level HD operative
disease-specific characteristics were defined using ICD-9-CM
volumes are associated with surgical outcomes following
diagnosis and procedure codes, as well as Clinical Trans-
pull- through in infants with HD.
action Codes (CTC), which are unique to the PHIS and useful
for examining resource utilization. Preoperative factors of
Materials and Methods interest were birth-related characteristics (birthweight,
gestational age, fetal growth restriction/malnutrition), age at
Cohort identification surgery, associated congenital anomalies, and diagnoses
reflective of overall illness severity (mechanical ventilation,
We performed a multiinstitutional retrospective cohort study total parenteral nutrition [TPN], extracorporeal membrane
of infants with Hirschsprungs disease (HD) who underwent oxygenation [ECMO], etc). Postoperative outcomes included
operative treatment. The study cohort was identified using reoperation within 30 d or 2 y of PT surgery, all-cause read-
previously validated methods. 51 We included infants mission within 30 d or 2 y, and HAEC-related readmission
admitted by age 60 d during the period from January 2000 to within 30 d or 2 y. In a sensitivity analysis, these outcomes
September 2012. Data came from the Pediatric Health Infor- were evaluated at 4 y among a slightly smaller cohort whose
mation System (PHIS), a hospital discharge database con- outcomes could be tracked over this duration of time. ICD-9-
taining inpatient, observation, ambulatory surgery, and CM procedure and diagnosis codes used to identify primary
emergency department encounters at 52 tertiary childrens PT operations, reoperations, and HAEC-related readmissions
hospitals across the United States. 51,52 We selected infants are listed in the Online Tables 1e3.
Statistical analysis operative volume on patients surgical outcomes. For these
analyses, a Bonferroni correction for multiple comparisons
Associations between preoperative characteristics and out- was applied, such that P < 0.0083 was considered
comes were evaluated using chi-square or Fisher exact tests
statistically significant. Finally, two sensitivity analyses were
for categorical variables and Wilcoxon rank sum tests for
performed. First, to test whether outcomes changed over the
continuous variables. A multivariable logistic regression
study period, time in years was added to each multivariable
model was fit for each outcome of interest. These models
model as a continuous variable. Second, an indicator variable
included all preoperative characteristics that were present in
for whether
at least 10 patients and associated with any outcome at
a patient was treated at a hospital with a dedicated
P < 0.10 in bivariate analyses. In addition to patient charac- colorectal program was included in each multivariable
teristics, the multivariable models initially included surgeon
model, in place of hospital volume. However, because most
HD pull-through operative volume, then alternately included
of these programs began toward the end of or after the
hospital HD pull-through operative volume. Each of these
study period, and because surgeon identification numbers
volume variables was evaluated using restricted cubic splines
changed partway through the study period, we were able to
with quartile knots. We examined the potentially nonlinear
include only 47 patients treated at a children’s hospital that
relationship between surgeon or hospital operative volume
had an established colorectal center at the time. Therefore,
and the risk of each outcome in order to identify any
the study was underpowered to evaluate the impact of this
inflection points that could be used to categorize operative
structural hospital characteristic. SAS Enterprise Guide
volumes. Upon identifying no inflection points, both surgeon
version 7.11 (SAS Institute Inc., Cary, NC) was used for the
and hos-
statistical analyses.
pital operative volumes were dichotomized at their top
patient-level tertiles. As in previous studies, we created a Results
multi-level categorical variable that accounted for both sur-
geon and hospital-level operative volume. 39,47 A multinomial Our study cohort included 1268 patients treated across 41
logistic regression model that included all evaluated patient- tertiary children’s hospitals. Figure 1 describes our study
level preoperative characteristics was then used to estimate cohort selection. The baseline characteristics of the entire
each patient’s probability of having their surgery performed study cohort are shown in Table 1. Of note, 225 patients
by a lower or higher volume surgeon and at a lower or higher (17.7%) had a congenital cardiac anomaly, 93 (7.3%) had an
volume hospital. We then used an inverse probability of additional gastrointestinal anomaly, 152 (12.0%) had Down’s
treatment weighting using estimated propensity scores to syndrome, 189 (14.9%) had preoperative enterocolitis, 108
concurrently estimate the effects of both surgeon and (8.5%) had meconium ileus, and 190 (15.0%) had a
hospital documented

PHIS Database: Data from 41 hospitals*


Jan 2000 – Sept 2012

HD Diagnosis Code (751.3) +


Age at admission ≤60 days + Discharged at least 2 years aGer their hospital began contributing clinical and resource utilization data to PHIS
N = 3321

N = 840
No pull through procedure by age 1 yea
N = 2481

N = 277
N = 2204 No ostomy or pull through procedure by age 6

N = 41
Pull through or total colectomy AND ostomy procedure on firs

N = 2163
N = 23
Data quality issues (incorrect dates of service or dup

N = 2140

N = 60
Operated on by a surgeon without a surgeo

N = 2080
N = 812
Operated on by a surgeon who did not have evidence of having performed surgeries at the patient’s hospital during

N = 1268

Fig. 1 e Neonatal Hirschsprungs disease - cohort selection flow chart.


Table 1 e Characteristics of neonatal Hirschsprungs disease study cohort (n [ 1268).
Variable Median (IQR) or n (%)
Single stage procedure (no ostomy) 857 (67.6)
Age in days at first HD surgery 11 (7-23)
Age in days at pull through 24 (9-111)
Male 1007 (79.4)
Race
White 774 (61.0)
Black 216 (17.0)
Other/Unknown 278 (21.9)
Insurance status at first pull-through admission
Public insurance 620 (48.9)
Private insurance 480 (37.9)
Other 168 (13.3)
Birth weight* 3323.5 (2950-3690)
Gestational age at birth in total completed weeksy 39 (38-40)
Associated birth diagnoses documented before first pull through z
Prematurity 108 (8.5)
Slow fetal growth or fetal malnutrition 24 (1.9)
Any congenital anomaly (other than HD) 419 (33.0)
Cardiac anomalyx 225 (17.7)
Prior cardiac surgery 7 (0.5)
GI anomaly (other than HD) 93 (7.3)
Other structural anomaly 196 (15.5)
Down’s syndrome 152 (12.0)k
z
Associated other diagnoses documented before first pull through
Enterocolitis 189 (14.9)
Necrotizing enterocolitis (NEC) 17 (1.3)
Meconium aspiration 1 (0.1)
Meconium ileus 108 (8.5)
Bronchopulmonary dysplasia 34 (2.7)
Associated maternal diagnoses z
Perinatal infection 190 (15.0)
Multiple gestation 2 (0.2)
Maternal conditions that affected fetus or newborn 14 (1.1)
Maternal procedures that affected fetus or newborn 0 (0)
Maternal noxious influences 8 (0.6)
Pregnancy and labor complications 17 (1.3)
Resource utilization prior to pull-through procedure {
Any preop mechanical ventilation 247 (19.5)
Preop mechanical ventilation at the PT admission 83 (6.6)
Any preop TPN 942 (74.3)
Preop TPN at the PT admission 549 (43.3)
Any preop ECMO 0 (0)
Any preop blood transfusion 108 (8.5)
Preop blood transfusion at the PT admission 40 (3.2)
Any preop NICU stay 904 (71.3)
Preop NICU stay at the PT admission 538 (42.4)
Surgeon average annual HD pull-through volume in the previous 2 y 1.5 (1-2.5)
Hospital average annual HD pull-through volume in the previous 2 y 9.5 (7-13)
*
n ¼ 1108.
y
n ¼ 804.
z
Based on ICD-9-CM diagnosis codes at the first admission for pull through or any previous hospital admission to that hospital.
x
Cardiac anomalies include all coded cardiac defects, including those that did not require correction at birth.
k
There were 11 (0.9%) additional chromosomal anomalies.
{
Preop resource utilization is defined based on CTC codes for resources utilized on dates prior to the date of surgery as no time
information is available in PHI.
30 days of all-cause
30 days of all-cause
A 1 B 1
0.9 0.9
0.8 0.8

Probability
Probability 0.7 0.7
0.6 0.6
0.5 0.5
0.4 0.4

readmission within
readmission within

0.3 0.3
0.2 0.2
0.1 0.1
0 0

0 5 10 15 20 05101520253035
Surgeon volume Hospital volume
2 years of all-cause

2 years of all-cause
C 1
0.9
D 1
0.9
0.8 0.8
Probability

Probability
0.7 0.7
0.6 0.6
0.5 0.5
0.4 0.4
readmission within

readmission within
0.3 0.3
0.2 0.2
0.1 0.1
0 0

0 5 10 15 20 05101520253035
Surgeon volume Hospital volume

Fig. 2 e Relationship between the surgeon and hospital pull-through volumes and all-cause readmission with 30 d and 2 y,
respectively, for infants with Hirschsprungs disease.
30 daysof HAEC-related

30 daysof HAEC-related

A 1
0.9
B 1
0.9
0.8 0.8
0.7 0.7
0.6 0.6
Probability

0.5 0.5
Probability

0.4 0.4
0.3 0.3
0.2 0.2
readmission within

readmission within

0.1 0.1
0 0

0 5 10 15 20 05101520253035
2 yearsof HAEC-related
2 yearsof HAEC-related

Surgeon volume Hospital volume

C 1
0.9
D 1
0.9
0.8 0.8
0.7 0.7
Probability
Probability

0.6 0.6
0.5 0.5
0.4 0.4
readmission within
readmission within

0.3 0.3
0.2 0.2
0.1 0.1
0 0

0 5 10 15 20 05101520253035
Surgeon volume Hospital volume

Fig. 3 e Relationship between the surgeon and hospital pull-through volumes and HAEC-related readmission with 30 d and
2 y, respectively, for infants with Hirschsprungs disease.
A 1 B 1
0.9 0.9
0.8 0.8
within 2 yearsProbability of reoperation within 30 days Probability of reoperation

within 30 days Probability of reoperation


0.7 0.7
0.6 0.6
0.5 0.5
0.4 0.4
0.3 0.3
0.2 0.2
0.1 0.1
0 0

0 5 10 15 20 05101520253035
Surgeon volume Hospital volume

C 1
0.9 D 1
0.9
0.8 0.8

Probability of reoperation within 2 years


0.7 0.7
0.6 0.6
0.5 0.5
0.4 0.4
0.3 0.3
0.2 0.2
0.1 0.1
0 0

0 5 10 15 20 05101520253035
Surgeon volume Hospital volume

Fig. 4 e Relationship between the surgeon and hospital pull-through volumes and reoperation with 30 d and 2 y,
respectively, for infants with Hirschsprungs disease.

perinatal infection. At the time of HD pull-through, 247


and patients classified into four groups based on their sur-
infants (19.5%) had required mechanical ventilation, and 549
geon’s and hospital’s PT volumes, we found in propensity-
(43.3%) had required TPN at some point during their hospital
score-weighted analyses that there were no differences in
admis- sion or admissions at which HD-related surgery was
any evaluated outcomes across these four groups (Table 2).
performed.
In the sensitivity analysis following a smaller cohort (n ¼
The median annual surgeon PT volume was 1.5 procedures
1094) out to 4 y, we also did not find differences in any
(IQR 1-2.5), and the median annual hospital PT volume was 9.5
evaluated 4- year outcomes across these four groups (Online
procedures (IQR 7-13). Over two-thirds of infants (n ¼ 679,
Table 4). Finally, when either year of surgery or treatment at
67.7%) were treated by surgeons who had performed an a hospital with a pediatric colorectal program were added to
average of two or fewer HD pull-throughs per year during the the fitted multivariable models these variables, these
preceding 2 y. Over half of infants (n ¼ 649, 51.2%) were variables were not
treated at hospitals that had performed an average of less than
found to be statistically significant in any model (P > 0.10, data
10 HD pull-throughs per year during the preceding 2 y. A total not shown).
of 218
patients (17.2%) were readmitted to the hospital for any
reason within 30 d, and 540 patients (42.6%) were readmitted Discussion
within 2 y. A total of 119 patients (9.4%) had HAEC-related
readmission within 30 d, and 271 patients (21.4%) had HAEC- This study of more than 1000 infants with Hirschsprungs
related readmission within 2 y. A total of 57 patients (4.5%) disease found no association between surgeon or hospital
had a reoperation within 30 d, and 129 patients (10.2%) had a HD pull-through volumes and the postoperative outcomes of
reoperation within 2 y. reoperation, all-cause readmission, and HAEC-related read-
Risk-adjusted relationships were evaluated between the mission. This suggests that the established volume-outcome
surgeon and hospital PT volumes and all-cause readmission relationship previously shown in adult colorectal surgery
(Fig. 2); HAEC-related readmission (Fig. 3); and reoperation may not exist for neonates with HD, or at least not those in-
(Fig. 4) at 30 d and 2 y. There were no statistically significant fants who are treated at the moderate to large-sized tertiary
relationships between either surgeon PT volume or hospital children’s hospitals that contribute data to the PHIS. 53-57 The
PT volume and any outcome evaluated. When surgeon and lack of a volume-outcome relationship in HD aligns with past
hospital PT volumes were dichotomized at their top tertiles studies of complex neonatal surgical diseases like CDH, EA/
TEF, and gastroschisis.42-44,46,47,58 However, competing
Table 2 e Adjusted readmission and reoperation rates by the surgeon and hospital volume category.

P ¼ 0.76*
2-year reoperation

P ¼ 0.99y
studies on complex birth defects have demonstrated a
volume- outcome relationship, underscoring the
persistent need to identify, measure, and track
competencies in managing complex surgical
anomalies.35,36,40,59-61
These findings are consistent with those of another recent
10.0

12.0

10.8

12.8
study utilizing the PHIS, which assessed risk factors for recur-
rent HAEC-related admissions and repeated pull-through
30-day reoperation

P ¼ 0.76*

P ¼ 0.99y

pro- cedures and found no relationship between these


outcomes and either surgeon or hospital volume.62 However,
in contrast to this previous study, the present study assessed
nonlinear volume-outcome relationships and used
4.1

6.4

4.9

4.8

propensity score an- alyses to account for the probability of


treatment to allow concurrent estimation of the impact of
surgeon and hospital operative volumes. Second, the present
2-year HAEC- related readmission

P ¼ 0.24*

P ¼ 0.65y

study examined neonatal HD, thereby avoiding misclassifying


redo HD surgeries as primary HD surgeries for patients
treated at multiple hospi- tals. Third, in the present study,
surgeon, and hospital PT vol- umes were calculated as
patient-level time-dependent variables based on volumes in
22.1

26.0

18.0

19.5

the 2 y preceding each included patients PT. Such definitions


were appropriate and necessary because hospitals began
contributing to PHIS at different times throughout the study
Estimates shown are risk-adjusted (propensity score weighted) estimates of the percentages of patients with the outcome.
P-value across 4 categories for hospital and surgeon volume (hospital volume <12 and ≤ 12, surgeon volume <2 and ≤ 2).

period, and surgeon identification numbers changed midway


30-day HAEC- related readmission

P ¼ 0.19*

P ¼ 0.52y

the study period. Fourth, the present study relied on uniform


30-day and 2-year follow-up periods, whereas in the
previous study, average durations of follow-up varied
substantially across hospitals. Last, we included an expanded
list of infectious diagnoses in our definition of HAEC- related
7.7

10.4

8.7

14.3

readmissions, and such diagnoses were found in more than


25% of the identified HAEC-related readmissions.
Interestingly, studies examining some of the most com-
mon pediatric surgical procedures have reported
2-year all cause readmission

P ¼ 0.44*

P ¼ 0.32y

significant volume-outcome relationships, whereas, with


the exception of congenital heart disease, these
relationships are less clear for rare surgical congenital
anomalies.30,31,37,38,42,45,63 While studies show significant
regionalization for both common pediatric and rare
42.0

48.7

42.4

42.5

neonatal surgical procedures to high- volume centers,


Comparison of higher hospital volume to lower hospital volume groups.

further regionalization of complex neonatal surgery would


30-day all cause readmission

P ¼ 0.18*

potentially limit patients access to care and surgical


P ¼ 0.53y

trainees’ exposure to these procedures.18,19 Without


implementing additional “within-hospital” subspecialization,
it may not be feasible to increase surgeons disease-specific
caseload to a level that provides sufficient statistical power
16.0

20.1

14.0

21.6

for studies to demonstrate volume-outcome relationships


in rare neonatal surgical populations. Surgeon and hospital-
Hospital and surgeon volume category

level HD pull-through volumes appear to be quite low even


at moderate to large-sized tertiary childrens hospitals,
suggest- ing a lack of centralization or surgeon
Hospital volume <12

subspecialization in this procedure at most


institutions.18,19,27,28,64
These data can help to inform the ongoing ACS
Childrens Surgery Verification initiative, which aims, in
5) Surgeon volume > 2 (n ¼ 230)

part, to identify pediatric surgical procedures that may


benefit from further subspecialization.21,23,24 For neonatal
HD, there is currently no evidence, including from this
study, to justify transfers of neonates from one tertiary
*

childrens hospital to another based on hospital or surgeon


HD pull-through volumes as a proxy for experience.
However, this study provides general- izable estimates of
y
postoperative morbidity after HD pull- through, which
could be used to benchmark rates of read- mission or
reoperation after HD pull-through.
This study is not without limitations. First, due to our use
of administrative data, we were unable to incorporate Supplementary data
disease- specific clinical information on infants with HD in
our ana- lyses. For example, we could not definitively adjust Supplementary data to this article can be found online at
for the severity of disease or identify patients with long- https://doi.org/10.1016/j.jss.2020.08.014.
segment HD or total colonic aganglionosis (TCA), both of
which have been linked to higher rates of HAEC and other REFERENCES
complications.65,66 However, these higher-risk HD infants are
comparatively rare, and there is no clear indication that long-
1. Langer JC. Hirschsprung disease. Curr Opin PEDIATR.
segment dis- ease is selectively triaged to higher-volume
2013;25:368e374.
centers. Second, due to the types of hospitals that contribute 2. Teitelbaum DH, Cilley RE, Sherman NJ, et al. A decade of
to the PHIS, our findings are not necessarily applicable to experience with the primary pull-through for hirschsprung
smaller pediatric hospitals and units within general acute disease in the newborn period: a multicenter analysis of
care hospitals. Similarly, this study is not statistically powered outcomes. Ann Surg. 2000;232:372e380.
to differen- tiate outcomes between “high-volume” and 3. Haricharan RN, Seo J-M, Kelly DR, et al. Older age at diagnosis
of Hirschsprung disease decreases risk of postoperative
“super-volume” centers, such as those with dedicated
enterocolitis, but resection of additional ganglionated bowel
colorectal centers/sur- geons. Thus, this study contributes
does not. J PEDIATR Surg. 2008;43:1115e1123.
limited information to- ward answering the question of 4. El-Sawaf M, Siddiqui S, Mahmoud M, Drongowski R,
whether the centralization of surgical care for children with Teitelbaum DH. Probiotic prophylaxis after pullthrough for
HD would improve outcomes. We did evaluate the full range Hirschsprung disease to reduce incidence of enterocolitis: a
of surgeon and hospital vol- umes using cubic splines and did prospective, randomized, double-blind, placebo-controlled,
not detect inflection points at high volumes. However, multicenter trial. J PedIATR Surg. 2013;48:111e117.
determining whether centers with dedicated colorectal 5. Gosain A, Frykman PK, Cowles RA, et al. Guidelines for the
diagnosis and management of Hirschsprung-associated
programs have better outcomes than other childrens
enterocolitis. PEDIATR Surg Int. 2017;33:517e521.
hospitals would require additional analyses using more 6. Langer JC, Durrant AC, de la Torre L, et al. One-stage transanal
recent data. This study also lacked information on the Soave pullthrough for Hirschsprung disease: a multicenter
provider or institution-level factors besides operative volume experience with 141 children. Ann Surg. 2003;238:569e583
that may have impacted HD patient outcomes. Finally, this [discussion 83-85].
study was unable to identify reoperations that occurred at 7. Pen˜ a A, Elicevik M, Levitt MA. Reoperations in
hospitals other than where the initial PT occurred or to hirschsprung disease. J PEDIATR Surg. 2007;42:1008e1014.
8. Langer JC, Rollins MD, Levitt M, et al. Guidelines for the
assess functional outcomes, both of which will be better
management of postoperative obstructive symptoms in children
understood through enhanced prospective data gathering
with Hirschsprung disease. PEDIATR Surg Int.
initiatives.67-69 2017;33:523e526.
9. Dasgupta R, Langer JC. Evaluation and management of persistent
problems after surgery for Hirschsprung disease in a child. J PedIATR
GASTROENTEROL Nutr. 2008;46:13e19.
Conclusions 10. Han-Geurts IJ, Hendrix VC, de Blaauw I, Wijnen MH, van
Heurn EL. Outcome after anal intrasphincteric Botox injection
In conclusion, our analysis demonstrates that surgeon and in children with surgically treated Hirschsprung disease. J
hospital Hirschsprungs PT operative volumes are not signifi- PEDIATR GASTROENTErol Nutr. 2014;59:604e607.
cantly associated with rates of readmission or reoperation 11. Aworanti OM, McDowell DT, Martin IM, Quinn F. Does
after this procedure. Future studies evaluating the effects of functional outcome improve with time postsurgery for
hirschsprung disease? Eur J PEDIATR Surg. 2016;26:192e199.
volume on long-term functional outcomes and reoperations
12. Conway SJ, Craigie RJ, Cooper LH, et al. Early adult outcome of the
are needed. Duhamel procedure for left-sided Hirschsprung diseaseda
prospective serial assessment study. J PEDIATR Surg.
2007;42:1429e1432.
13. Huang W-K, Li X-L, Zhang J, Zhang S-C. Prevalence, risk factors,
Acknowledgment and prognosis of postoperative complications after surgery for
hirschsprung disease. J GASTROINTEST Surg.
Authors’ contributions: J.N.C. had full access to all of the data 2018;22:335e343.
in the study and takes responsibility for the integrity of the 14. Widyasari A, Pravitasari WA, Dwihantoro A, Gunadi. Functional
outcomes in Hirschsprung disease patients after transabdominal
data and the accuracy of the data analysis. JCA, RJW, DRH,
Soave and Duhamel procedures. BMC GASTROENTEROL.
KJD, and PCM all contributed to the following: study concept 2018;18.
and design, acquisition, analysis, or interpretation of data, 15. Birkmeyer JD, Siewers AE, Finlayson EVA, et al. Hospital volume
draft- ing of the article, critical revision of the article for and surgical mortality in the United States. N Engl J Med.
important intellectual content, and statistical analysis. 2002;346:1128e1137.
16. Birkmeyer JD, Stukel TA, Siewers AE, Goodney PP, Wennberg
DE, Lucas FL. Surgeon volume and operative mortality in the
Disclosure United States. N Engl J Med. 2003;349:2117e2127.
17. Finks JF, Osborne NH, Birkmeyer JD. Trends in hospital volume
The authors reported no proprietary or commercial interest and operative mortality for high-risk surgery. N Engl J Med.
in any product mentioned or concept discussed in this 2011;364:2128e2137.
article.
18. Barnhart DC, Oldham KT. Regionalization of children’s 42.
surgery. Ann Surg. 2016;263:1067e1068.
19. Salazar JH, Goldstein SD, Yang J, et al. Regionalization of
pediatric surgery. Ann Surg. 2016;263:1062e1066.
20. Referral to pediatric surgical specialists. PEDIATRics.
2014;133:350e356.
21. Children’s surgery verification pilot draft documents.
PEDIATRICS. 2015;135:e1538ee.
22. Stolar CJH. Best practice for infant surgeryda position statement
from the American Pediatric Surgical Association. J PEDIATR Surg.
2008;43:1585e1586.
23. Optimal resources for children’s surgical care v.1. American
College of Surgeons. 2015. Available at:
https://www.facs.org/ quality-programs/childrens-
surgery/childrens-surgery- verification/standards. Accessed
March 12, 2019.
24. Baxter KJ, Gale BF, Travers CD, Heiss KF, Raval MV.
Ramifications of the children’s surgery verification program for
patients and hospitals. J Am Coll Surg. 2018;226:917e924.e1.
25. Levels of neonatal care. PediATRICS. 2012;130:587e597.
26. Bruns NE, Shah MA, Dorsey AN, Ponsky TA, Soldes OS.
Pediatric surgery e a changing field: national trends in pediatric
surgical practice. J PEDIATR Surg. 2016;51:1034e1038.
27. Superina R. The shrinking landscape of pediatric surgery: is less
more? J PEDIATR Surg. 2018;53:868e874.
28. Carlisle EM, Rowell EE. Ethical challenges in regionalization of pediatric
surgical care. Curr Opin PEDIATR. 2019;31:414e417.
29. Al-Jazaeri A, Al-Shanafey S, Zamakhshary M, et al. The impact
of variation in access to care on the management of Hirschsprung
disease. J PEDIATR Surg. 2012;47:952e955.
30. Berry JG, Lieu TA, Forbes PW, Goldmann DA. Hospital
volumes for common pediatric specialty operations 2007;.
2007;161(1):38.
31. Evans C, Van Woerden HC. The effect of surgical training and
hospital characteristics on patient outcomes after pediatric
surgery: a systematic review 2011;. 2011;46(11):2119e2127.
32. McKiernan PJ, Baker AJ, Kelly DA. The frequency and outcome
of biliary atresia in the UK and Ireland. LANCET.
2000;355:25e29.
33. Wes AM, Mazzaferro D, Naran S, Hopkins E, Bartlett SP,
Taylor JA. Craniosynostosis surgery: does hospital case
volume impact outcomes or cost? PLAST Reconstr Surg.
2017;140:711e.
34. Wes AM, Mazzaferro D, Naran S, Bartlett SP, Taylor JA. Cleft-
palate repair: does hospital case-volume impact outcomes or cost?
PLASt Reconstr Surg. 2018;141:1193e1200.
35. Grushka JR, Laberge J-M, Puligandla P, Skarsgard ED. Effect of
hospital case volume on outcome in congenital
diaphragmatic hernia: the experience of the
Canadian Pediatric Surgery Network. J PEDIATR Surg.
2009;44:873e876.
36. Sacks GD, Ulloa JG, Shew SB. Is there a relationship between
hospital volume and patient outcomes in gastroschisis
repair? J PedIATR Surg. 2016;51:1650e1654.
37. Ly DP. Effect of surgeon and hospital characteristics on
outcome after pyloromyotomy 2005;. 2005;140(12):1191.
38. Safford SD, Pietrobon R, Safford KM, Martins H, Skinner MA,
Rice HE. A study of 11,003 patients with hypertrophic pyloric
stenosis and the association between surgeon and hospital
volume and outcomes 2005;. 2005;40(6):967e973.
39. Drews JD, Cooper JN, Onwuka EA, Minneci PC, Aldrink JH. The
relationships of surgeon volume and specialty with outcomes
following pediatric thyroidectomy. J PedIATR Surg.
2019;54:1226e1232.
40. Hong CR, Fullerton BS, Han SM, et al. Impact of disease-
specific volume and hospital transfer on outcomes in
gastroschisis. J PEDIATR Surg. 2019;54:65e69.
41. Bjørnland K, Hinna M, Aksnes G, et al. Outcome for biliary atresia
patients treated at a low-volume centre. SCAND J GASTROENTEROL.
2018;53:471e474.
Sømme S, Shahi N, McLeod L, Torok M, McManus B, Ziegler MM.
Neonatal surgery in low- vs. high-volume institutions: a KID inpatient
database outcomes and cost study after repair of congenital
diaphragmatic hernia, esophageal atresia, and gastroschisis. PediATR
Surg Int. 2019;35:1293e1300.
43. Harbert J, Hohmann S, Pillai S, et al. Case volume and
outcomes of congenital diaphragmatic hernia surgery in
academic medical centers 2015;. 2015;32(09):845e852.
44. Dubrovsky G, Sacks GD, Friedlander S, Lee S. Understanding
the relationship between hospital volume and patient
outcomes for infants with gastroschisis. J PEDIATR Surg.
2017;52:1977e1980.
45. McAteer JP, Lariviere CA, Drugas GT, Abdullah F, Oldham KT,
Goldin AB. Influence of surgeon experience, hospital volume,
and specialty designation on outcomes in pediatric surgery.
JAMA PediATR. 2013;167:468.
46. Dylkowski D, Dave S, Andrew Mcclure J, Welk B, Winick-Ng J, Jones S.
Repair of congenital esophageal atresia with tracheoesophageal
fistula repair in Ontario over the last 20 years: volume and
outcomes. J PEDIATR Surg. 2018;53:925e928.
47. Lawrence AE, Minneci PC, Deans KJ, Kelley-Quon LI,
Cooper JN. Relationships between hospital and
surgeon operative volumes and outcomes of
esophageal atresia/
tracheoesophageal fistula repair. J PEDIATR Surg. 2019;54:44e49.
48. Hackam DJ, Filler RM, Pearl RH. Enterocolitis after the surgical
treatment of Hirschsprung’s disease: risk factors and
financial impact. J PEDIATR Surg. 1998;33:830e833.
49. Miyake H, Hock A, Koike Y, et al. Duhamel and transanal
endorectal pull-throughs for hirschsprung’ disease: a
systematic review and meta-analysis. Eur J PEDIATR Surg.
2018;28:081e088.
50. Prato AP, Gentilino V, Giunta C, et al. Hirschsprung disease:
do risk factors of poor surgical outcome exist? 2008;.
2008;43(4):612e619.
51. Sulkowski JP, Deans KJ, Asti L, Mattei P, Minneci PC. Using the
Pediatric Health Information System to study rare congenital
pediatric surgical diseases: development of a cohort of
esophageal atresia patients. J PEDIATR Surg.
2013;48:1850e1855.
52. Sulkowski JP, Cooper JN, Lopez JJ, et al. Morbidity and
mortality in patients with esophageal atresia. Surgery.
2014;156:483e491.
53. Huo YR, Phan K, Morris DL, Liauw W. Systematic review and a meta-
analysis of hospital and surgeon volume/outcome relationships in
colorectal cancer surgery. J GASTROINTEST Oncol.
2017;8:534e546.
54. Buurma M, Kroon HM, Reimers MS, Neijenhuis PA. Influence
of individual surgeon volume on oncological outcome of
colorectal cancer surgery. Int J Surg Oncol. 2015;2015:1e10.
55. Gani F, Cerullo M, Zhang X, et al. Effect of surgeon
“experience” with laparoscopy on postoperative
outcomes after colorectal surgery. Surgery.
2017;162:880e890.
56. Liu C-J, Chou Y-J, Teng C-J, et al. Association of surgeon
volume and hospital volume with the outcome of patients
receiving definitive surgery for colorectal cancer: a
nationwide population-based study. CANCER.
2015;121:2782e2790.
57. Balentine CJ, Naik AD, Robinson CN, et al. Association of high-
volume hospitals with greater likelihood of discharge to
home following colorectal surgery. JAMA Surg.
2014;149:244.
58. Apfeld JC, Kastenberg ZJ, Sylvester KG, Lee HC. The effect of level of
care on gastroschisis outcomes. J PEDIATR.
2017;190:79e84.e1.
59. Morche J, Mathes T, Jacobs A, et al. Relationship between
volume and outcome for congenital diaphragmatic hernia: a
systematic review protocol. Syst Rev. 2018;7.
60. Lal DR, Gadepalli SK, Downard CD, et al. Challenging surgical
dogma in the management of proximal esophageal atresia
with distal tracheoesophageal fistula: outcomes from the Cheng S, Wang J, Pan W, et al. Pathologically assessed grade of
Midwest Pediatric Surgery Consortium. J PediATR Surg. Hirschsprung-associated enterocolitis in resected colon in children with
2018;53:1267e1272. Hirschsprung’s disease predicts postoperative bowel function. J PEDIATR
61. Apfeld JC, Kastenberg ZJ, Gibbons AT, Carmichael SL, Lee HC, Sylvester Surg. 2017;52:1776e1781.
KG. Treating center volume and congenital diaphragmatic hernia 67. Halleran DR, Lane VA, Leonhart KL, et al. Development of a patient-
outcomes in California. J PEDIATR. 2020. reported experience and outcome measures in pediatric
62. Pruitt LCC, Skarda DE, Rollins MD, Bucher BT. Hirschsprung- patients undergoing bowel management for CONSTIPATION
associated enterocolitis in children treated at US children’s AND FECAL INCONTINENCE. J PEDIATR GASTROENTEROL Nutr.

hospitals. J PEDIATR Surg. 2019;55:535e540. 2019;69:e34ee38.


63. Lariviere CA, McAteer JP, Huaco JA, et al. Outcomes in 68. Reeder RW, Wood RJ, Avansino JR, et al. The pediatric
pediatric surgery by hospital volume: a population-based colorectal and pelvic learning consortium (PCPLC): rationale,
comparison. PEDIATR Surg Int. 2013;29:561e570. infrastructure, and initial steps. Tech Coloproctol.
64. Bezner SK, Bernstein IH, Oldham KT, Goldin AB, Fischer AC, 2018;22:395e399.
Chen LE. Pediatric surgeons’ attitudes toward regionalization 69. Heinrich M, Ha¨ berle B, Von Schweinitz D, Stehr
of neonatal surgical care 2014;. 2014;49(10):1475e1479. M. Re-operations for Hirschsprung’s disease:
65. Anupama B, Zheng S, Xiao X. Ten-year experience in the long-term complications. Eur J PediAtr Surg.
management of total colonic aganglionosis. J PEDIATR Surg. 2011;21:325e330.
2007;42:1671e1676.

66.

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