Pediatric short-bowel syndrome: the cost of comprehensive care1,2
Ariel U Spencer, Debra Kovacevich, Michelle McKinney-Barnett, Deanna Hair, Julie Canham, Christopher Maksym,
and Daniel H Teitelbaum
ABSTRACT
Background: Little information is available about the financial
charges incurred by patients with short-bowel syndrome (SBS). This
is particularly true for pediatric SBS patients who receive some of the
most complex medical care.
Objectives: The aims of this study were to determine the total cost
of care for these patients and to analyze their utilization of home and
hospital-based health care services.
Design: This was a retrospective review of the total charges incurred
by 41 children with SBS over the past decade, encompassing both
inpatient and home-care charges.
Results: The mean (앐 SD) total cost of care for pediatric SBS was
US$505 250 앐 US$248 398 (corrected for inflation to the year
2005) for the first year of care alone. Inpatient hospitalization ac-
counted for most of these expenses (US$416 818 앐 US$242 689, or
82% of the total), and this was attributable to prolonged requirements
for intensive care resources, numerous surgical procedures, and mul-
tiple readmissions during the first year of diagnosis. Hospital-based
costs steadily declined in subsequent years, but home-care services,
in stark contrast, unexpectedly increased every year for the first 5 y
of diagnosis—a trend that was highly significant (P 쏝 0.005), reach-
ing US$184 520 앐 US$111 075 for the fifth year of home care. This
increasing cost was attributable to increasing complications of par-
enteral nutrition, especially infectious complications. Although per-
patient charges varied widely, the mean total cost of care per child
over a 5-y period was US$1 619 851 앐 US$1 028 985. A strong
correlation was found between higher charges and infants with
쏝10% of predicted small-bowel length.
Conclusions: This study was the first to calculate the total costs for
pediatric SBS patients and to provide an in-depth analysis of these
patients’ actual utilization of health care services. This information
may help guide health care providers and families who have children
with SBS. The comprehensive care of pediatric SBS patients costs
significantly more than has previously been estimated. Contrary to
previous views, home care significantly increases each year after
diagnosis. Am J Clin Nutr 2008;88:1552–9.
INTRODUCTION
Short-bowel syndrome (SBS) is a devastating disease process
in infants and children with mortality rates persistently reported
in the 20 –30% range despite marked improvements in the care of
these children (1– 4). Care for SBS patients is complex and
resource-intensive, often beginning at the time of birth or shortly
thereafter, and requires prolonged admission to an intensive care
unit, multiple surgical procedures (5) within the first year of life,
and specialized nutritional support (6 –10). Treatment of such
1552 Am J Clin Nutr 2008;88:1552–9. Printed in USA. © 2008 American Society for Nutrition
patients may be required for several years and generally involves
a multidisciplinary approach, including inpatient care, home in-
fusion services, home care, nursing visits, and clinical appoint-
ments (10 –15). Approximately 40 000 patients receive home
parenteral nutrition (PN) in the United States (16). Although
children account for a small number of SBS patients, the mone-
tary cost associated with the care of this group of patients can be
sizeable.
No comprehensive study to date has examined the actual costs
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of caring for pediatric SBS patients over a defined period of time.
The cost of caring for pediatric patients receiving home PN has
been estimated from adult data (17); however, these adult pa-
tients were not exclusively SBS patients. Furthermore, the low
number of hospitalizations in that particular report suggests that
the adult patients required far less care than many pediatric pa-
tients require. In fact, the actual total cost of care (COC) for
pediatric SBS remains unknown (18, 19). Although estimates
have been made, little to no substantive information is actually
available (20), and many of these monetary estimates date back
to almost 3 decades ago (21, 22), which makes the extrapolation
to current clinical practice difficult. Additionally, the etiologies
and comorbidities of pediatric SBS differ significantly from
those for adult SBS patients (23–25). These patients often require
not only intensive hospital treatment, but also a sustained pro-
gram of home-based care for several years, especially PN, until
intestinal function is restored. Knowing the total COC may sig-
nificantly affect the welfare of patients and their families and
potentially influence decisions regarding therapeutic options;
this is particularly important because many third party payors
have strict capitations on life expenditures for medical insurance.
Finally, it is important for government services to better under-
stand the economics of SBS. Current limitations on reimburse-
ment for such patients may grossly underestimate the actual
financial costs required to provide adequate care for such chil-
dren (26).
1
From the Department of Surgery (AUS and DHT) and the College of
Pharmacy (CM), University of Michigan, Ann Arbor, MI; the CS Mott
Children’s Hospital (AUS, DK, and DHT), the University of Michigan
Health System Financial Planning (DH), and HomeMed Service (DK and
MM-B), Ann Arbor, MI; and the University of Michigan Visiting Nurse
Corporation, Ann Arbor, MI (JC).
2
Address reprint requests and correspondence to DH Teitelbaum, Sec-
tion of Pediatric Surgery, University of Michigan Hospitals, F3970 Mott
Children’s Hospital, Box 0245, Ann Arbor, MI 48109. E-mail: dttlbm@
umich.edu.
Received February 15, 2008. Accepted for publication July 11, 2008.
doi: 10.3945/ajcn.2008.26007.
 COSTS OF SHORT-BOWEL SYNDROME
The lack of comprehensive data on the total COC stems from
the fact that pediatric SBS requires health care services in both
the home and in the hospital setting over a protracted period of
time. Few investigators have had access to this type of economic
data. Our health system has the very unique ability to track a large
number of pediatric patients over the past decade who received
the vast majority of their care from our University of Michigan
Health Service. This allowed us to calculate a comprehensive
estimate of the total charges incurred for these patients, both
during their inpatient hospitalizations and over the duration of all
home care. This study gives the most comprehensive picture of
these charges to date and should provide very useful information
for health care providers who care for pediatric SBS patients.
SUBJECTS AND METHODS
Patient population
Pediatric SBS is defined as the loss of 욷70% of small intestinal
length from surgical resection or congenital defect or 욷2 mo of
PN dependence due to complete intestinal dysfunction. Stan-
dard, commercially available PN formulas were used. The onset
of the diagnosis of SBS was always considered the reference
point (time ҃ zero). Between March 1992 and January 2005, 117
pediatric SBS patients were cared for at the CS Mott Children’s
Hospital of the University of Michigan. Of this group, 41 families
elected to receive their postdischarge home care through
HomeMed at the University of Michigan Health System—a
home infusion pharmacy service operated by our hospital sys-
tem. These patients also utilized our University home health
agency and had their follow-up clinic visits at our Intestinal
Failure Clinic. For any 1 y of care, the total charges for these 41
patients (referred to as COC in this report) for each of these
patients could be calculated for their care.
This study was a retrospective review of all in-hospital and
home-care charges during the study period and was conducted in
accordance with the ethical standards of the University of Mich-
igan Institutional Review Board (IRB) and was approved by this
Board (approval 2000-0254).
Calculation of charges
All charges were converted to 2005 US dollars using inflation
correction factors published by the US Department of Labor’s
Bureau of Labor Statistics. Charges were rounded to the nearest
dollar after the calculations were made. In-hospital charges were
defined as all inpatient hospitalizations (including procedural
charges, diagnostic testing, and hospital room charges), as clas-
sified by DRG code and procedural code. The COC for this work
represents the allowable billable charges as set by Medicaid.
Additionally, because of the nature of our billing system, all
out-patient clinic visits and outpatient diagnostic test charges that
were drawn in our clinic were included in the in-hospital portion
of the charges. Home-care charges were defined as all care pro-
vided in the home, including both home infusion charges and
nursing visits, as classified according to an internal system.
Home infusion charges included all therapies performed in the
home with the exclusion of nursing visits. Briefly, home-care
charges were placed into 1 of 5 categories: antimicrobial medi-
cations, other parenteral medications, intravenous fluid therapy
(nonnutrition), enteral nutrition therapy and accompanying
products, and PN. Costs associated with supplies, pumps, tubing,
and accessories were placed into the corresponding category.
1553
Prescribed medications filled by the patient’s family pharmacy
were not included in these home infusion figures. In general,
nursing visit charges were low and accounted for 울2% of net
charges. Most nursing visit charges were incurred within the first
year of care. Because of this, nursing visit charges were incor-
porated in the total COC under the subheading of home care. The
duration of each type of therapy in days, as well as in the year after
the diagnosis of SBS, was recorded for each charge. All analyses
were conducted from the purchaser’s perspective; that is, the
charges calculated were the price that a third-party payor would
pay for the services rendered. Charges in each case follow those
prescribed by Medicare. Nonreimbursed charges were not in-
cluded in the analysis, so that results describe only the actual
charges associated with pediatric SBS.
We looked at the first 5 y of SBS, calculating the total COC per
year for the first 5 y. We chose this time frame because most
patients who survive SBS achieve independence from PN within
the first 5 y, although some continue to remain PN-dependent.
Definitions
SBS was defined as a loss of 욷70% of small intestinal length
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from surgical resection or congenital defect or 욷2 mo of PN
dependence because of complete intestinal dysfunction. The on-
set of the diagnosis of SBS was always considered the reference
point (time ҃ zero). Thus, patients treated at different times
throughout the study period could be directly compared at equiv-
alent points in time subsequent to the development of SBS (on-
set). In addition, not all patients elected to continue their home
care through the University of Michigan for the entire duration of
the study. Therefore, data were included only for those years
when a patient was fully serviced by our institution. Finally, once
a patient was completely weaned off of PN and had their intra-
venous line removed, we considered the patient to be terminated
from the study. Because this group of patients was not a constant
number, the various shifts in patient numbers throughout the
study are conveniently provided in Table 1.
Statistical methods
Charges for all patients during the first through the fifth year
after onset of SBS were aggregated for each year, and means
were compared with analysis of variance (ANOVA). The least-
squares method was used for post hoc analysis to detect statistical
TABLE 1
Breakdown of the outcomes of all 41 patients reported in this study1
No. of No. of No. of patients
Year after patients at patients No. of who continued
onset of start of year weaned from patients PN by end of
SBS on PN PN who died year
1 41 12 6 23
2 23 5 1 17
3 17 2 1 14
4 122 4 0 8
5 8 0 1 73
1
Note that the results for each year represent the status of patients at the
end of each year. SBS, short-bowel syndrome; PN, parenteral nutrition.
2
Two patients were 쏝4 y of age and therefore were not included in the
data for the following year.
3
Follow-up of these 7 patients showed that 4 were successfully weaned
from PN and 3 died.
1554 SPENCER ET AL
differences between groups. P 쏝 0.05 was considered signifi-
cant. Data are expressed as means 앐 SDs; medians are provided
in some cases.
Covariates that could potentially influence the amount of total
charges were examined by using linear multivariate regression
analysis. Because many patients did not survive past the first
year, and the greatest costs were incurred during this time period,
this multivariate analysis was confined to the first year after the
diagnosis of SBS. In the initial linear regression model, 3 covari-
ates were considered (survival, cholestasis, and the estimated
percentage of the remaining small intestine). Because the per-
centage of the remaining small-bowel length varied depending
on the gestational age of the child when initially measured, we
used a conversion method to estimate the percentage of predicted
small bowel remaining based on gestational age, as opposed to a
set number of centimeters (4, 27). Covariates that were found not
to be significant were subsequently removed by backward elim-
ination (28, 29). Statistical analysis was performed with SPSS
version 13.0 (SPSS Inc, Chicago, IL).
RESULTS
Demographics
Of the 41 pediatric SBS patients, 16 (39%) were boys. The
onset of SBS ranged from 0 d to 17.7 y. Onset of SBS occurred
at the time of birth in 21 infants (congenital defects, including
gastroschisis and severe intestinal atresia), within 2 mo of birth
in 12 infants (predominantly due to necrotizing enterocolitis),
and between 5 mo and 17.7 y of age in the remaining 8 patients.
SBS was due to volvulus, trauma, and miscellaneous etiologies
in these older patients. None of the patients had a known malig-
nancy. The study population was representative of the spectrum
of etiologies of pediatric SBS (Table 2). With the onset of SBS,
all 41 patients required total PN support. In comparison with a
larger group of 117 infants from a previous study (4), no signif-
icant differences were noted between the distribution of the di-
agnoses, gestational age (35.6 앐 0.9 wk in this series compared
with 33 앐 4.7 wk in the larger group), and overall survival (30%
for the current study and 27.5% for the larger group of SBS
patients).
Duration of follow-up
All data were stratified according to the year after onset of
SBS. Data were analyzed for each patient for the years when the
patient received both home care and hospital care at our institu-
tion. The number of patients available for analysis during each
year declined with patient deaths and attrition due to weaning off
of PN (Table 1). Some patients were referred to our institution
TABLE 2
Etiologies of short-bowel syndrome
Diagnosis No. of patients Percentage of total
% wide SD is noted, which reflects the diversity of this patient
Gastroschisis 11 26.8
Intestinal atresia 6 14.6
Volvulus 7 17.1
Necrotizing enterocolitis 9 22.0
Miscellaneous 8 19.5
FIGURE 1. Quarterly rate of readmission to the hospital for short-bowel
syndrome (SBS). The rate of readmission to the hospital was very high in the
first year, but declined steadily thereafter (R2 ҃ 0.762, P 쏝 0.0001). For
example, a readmission rate of 50% in the graph would indicate that, at any
given time, 50% of the patients who were potentially at risk of readmission
(ie, surviving and not already hospitalized at that point in time) would be
readmitted to the hospital during the time period shown. Data were calculated
quarterly; error bars indicate the mean 앐 SEM of each 3-mo period.
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after having received care elsewhere, and their data were ana-
lyzed only from the years when all their medical care was pro-
vided at our institution. As stated above, once patients were
receiving 100% enteral supplementation, they were considered
to have completed contributing data for this study. Although
home care may still have been provided, the level of intensity
declined dramatically, and many of these patients continued with
long-term enteral supplementation. Finally, because of the size-
able attrition of patients over the first 5 y after the onset of SBS,
only data for the first 5 y after the onset of SBS were included in
the analysis, even though some of the patients received PN for
longer periods of time.
Frequency and duration of admission to hospital
During the first year after the onset of SBS, frequent readmis-
sions were observed for most patients. On average, patients re-
quired 5.8 앐 3.4 readmissions after the initial hospitalization
during the first 12 mo alone (median: 6 readmissions; range:
1–12 readmissions). This resulted in a mean (앐 SD) of 119 앐 61
in-hospital days in the first year after diagnosis of SBS (per
patient). The duration of the initial hospitalization (at onset of
SBS) averaged 67.1 앐 50.3 d (median: 44 d; range: 19 –161 d).
However, the duration of hospitalizations for subsequent read-
missions during the first year were much shorter (10.0 앐 14.7 d;
median: 5 d) per admission (P 쏝 0.0001). The frequency of
readmission, although initially very high, also steadily decreased
with time (Figure 1). This trend in the decline in readmission
rates was highly significant (P 쏝 0.0001, R2 ҃ 0.762).
Total cost of care
A summary of the total COC for the 5 y of the study is shown
in Figure 2. The COC is summarized as mean dollar values. A
population. COC was greatest in the first year, amounting to
쏜US$500 000. Also interesting was that the COC for subsequent
years was fairly stable, between 앒$250 000 and $300 00/y (me-
dian: $296 808/y). We next broke down these charges between
in-hospital and home care.
 COSTS OF SHORT-BOWEL SYNDROME 1555
TOTAL COST OF CARE

$800,000

$700,000

$600,000

US Dollars
USD $500,000

$400,000

$300,000

$200,000

$100,000

$0
1 2 3 4 5
Year
FIGURE 2. Mean (앐 SD) annual total cost of care for pediatric short-bowel syndrome patients for each of the first 5 y after diagnosis. Although costs declined
after the first year, they remained persistently at 앒$250 000 to $300 000/y thereafter. Not shown are data from the few patients with short-bowel syndrome who survived

Downloaded from ajcn.nutrition.org by guest on April 7, 2015

beyond the fifth year. For these long-term survivors, costs remained persistently elevated. Costs are in US dollars (USD) corrected for inflation to the year 2005.

In-hospital admission charges
Overall, for the entire 5-y period of study, the average pediatric
SBS patient spent a mean (앐 SD) total of 146 앐 188 d in the
hospital (median: 82 d; range: 14 –1064 d) and incurred a total
cost of $1 619 851 앐 $1 028 985, inclusive of both hospital and
home-care costs. However, hospital costs varied widely between
patients, and therefore a more detailed analysis was performed.
Data from each patient were stratified according to the year
post-onset of SBS.
The total in-hospital COC values per year are shown in Table
3. Not surprisingly, these costs diminished each year, as the total
number of hospitalizations and the duration of hospitalizations
declined annually (Figure 1); mean in-hospital charges declined
to 쏝$50 000 by year 5. Apart from minor fluctuations, there was
no significant change in the daily COC of inpatient hospitaliza-
tions over the course of time.
Home-care charges
A summary of all home-care patient charges is given in
Table 3, including the contrast between inpatient charges and
home-care charges during these same years. What is particu-
larly striking about the data is the fact that home-care costs
continued to rise each year. Although the inpatient COC
trended steadily downward during these 5 y, as mentioned
above, the total home-care COC continued to increase every
year after the onset of SBS. Home-care COC started at
$87 932 앐 70 079 (median charge: $81 160) for the first year
and rose to $184 520 앐 111 075 by year 5 (median charge:
$202 980). Note that data reflect an annual, not a cumulative,
average. Thus, for patients who continue to utilize home-care
services for SBS care (including enteral supplements), annual
costs continued to rise. A more detailed breakdown of home-
care charges is given in Table 4. This breakdown shows the
causes of this persistent increase in home-care charges. Spe-
cifically, the persistently high COC for PN, along with in-
creasingly high charges for other therapies (particularly anti-
microbials), helped to explain this rise in home-care charges.
This rise in charges occurred despite the fact that many infants
were actually being weaned off of PN during this time period.
Outcome of patients and relation of survival and length
of the small bowel to COC
The duration of PN dependence ranged from 6 mo to 11.6 y,
and all patients who were eventually weaned off PN (defined as
maintaining adequate growth on enteral nutrition alone) survived
throughout the duration of the study. In contrast, all patients who
died remained dependent on PN up to the time of death. Of the
patients who died, the mean survival was 4.3 앐 5.7 y (range: 6 mo

TABLE 3
Annual hospital length of stay, in-hospital charges, and home care charges1
Years after onset of SBS Time as an inpatient Annual hospital charges Annual home-care charges

d US$ US$
1 119 앐 61 (8–219) 416 818 앐 242 689 87 932 앐 70 079
2 30.7 앐 44.3 (0–193) 117 794 앐 134 277 116 988 앐 89 802
3 15.5 앐 32.6 (0–112) 81 795 앐 142 921 132 882 앐 113 243
4 15.6 앐 35.7 (0–138) 54 065 앐 92 285 180 163 앐 91 466
5 15.6 앐 45.7 (0–172) 48 829 앐 110 656 184 520 앐 111 075
1
All values are x៮ 앐 SD; range in parentheses. SBS short-bowel syndrome.
1556 SPENCER ET AL
TABLE 4
Breakdown of the most common home-care charges by therapy type for 20051
Therapy Year 1 Year 2
Antimicrobials 11 887 앐 11 766 19 141 앐 19 161
Injectables 4242 앐 5359 5983 앐 6622
Intravenous hydration 6149 앐 5827 7187 앐 6053
Enteral nutrition 5827 앐 5538 11 248 앐 9384
Parenteral nutrition 83 262 앐 59 085 106 154 앐 64 118
1
All values are x៮ 앐 SD. Costs are in US dollars corrected for inflation to the year 2005.
2
No SD is given because the amount reflects a small number of patients.
to 19 y; median: 1.3 y). Two patients who remain PN-dependent
were still alive at the last follow-up at age 쏝5 y. The time
required to wean off of PN (enteral independence) in the group
that ultimately did was 2.6 앐 3.1 y (range: 2.4 mo to 12.6 y of PN
dependence; median: 1.5 y). The data provided in Table 1 better
clarifies the patients analyzed in this study.
On the basis of our finding of an almost 30% mortality rate, we
next stratified our patients as survivors and nonsurvivors. The
annual COC for patients in the group who ultimately died, com-
pared with those who survived, is shown in Figure 3. The strik-
ing difference between patients who ultimately survived and
those who did not is that the annual COC was always greater in
the nonsurviving group. The mean annual cost of in-hospital care
for patients in the group that survived, compared with the group
that did not survive, are shown in Figure 3B. Aside from the first
year, hospital costs were consistently greater in the group that
ultimately died, and the difference became more pronounced in
subsequent years. This was thought to be due to the fact that the
survivors had a dramatic decrease in their annual hospital costs
with time. As can be seen, hospitalization cost data after the
second year appeared to be a strong independent predictor of the
ultimate outcome. Home-care costs are shown in Figure 3C.
Remarkably, even though patients who died spent a great deal of
time in the hospital, their home-care costs were also consistently
higher than those for the patients who ultimately survived. The
data shown included only those children who continued to utilize
home care.
Similar to the observations of others (30), our group has pre-
viously shown that patient survival could be predicted by bowel
length. Our own previous work also showed a correlation between
those with 쏝10% of their predicted small-bowel length and an
inability to wean off PN (4). The total COC per bowel length is
shown in Figure 4. Those patients with 쏝10% of predicted small-
bowel length had a consistently greater COC for all of the years
studied. These higher costs were predominately due to the fact that
these patients had a greater frequency of inpatient hospitalization
and a greater utilization of hospital resources. As was previously
shown, patients with 쏝10% of the estimated normal small-bowel
length remaining were significantly more likely to remain perma-
nently PN-dependent and are at a much greater risk of morbidity and
mortality. For the entire 5-y analysis, these patients incurred an
almost doubling of charges: $2 029 958 앐 1 044 620 compared
with $1 301 026 앐 862 123
Regression analysis of charges during the first year of
SBS
To better define covariates that may predict a greater COC, a
regression analysis was done for patients in the first year after
Year 3 Year 4 Year 5
US$
23 996 앐 32 431 27 792 앐 25 371 34 784 앐 33 746
17 716 앐 23 992 9631 앐 7020 16 9512
13 473 앐 26 876 16 205 앐 26 031 7777 앐 5344
13 917 앐 10 742 16 319 앐 11 706 21 288 앐 10 637
152 411 앐 51 522 147 035 앐 61 274 126 579 앐 82 124
SBS. Interestingly, nonsurvival (nonstandardized ␤ coefficient:
73 807; standardized coefficient: 0.113; P ҃ 0.719) and the de-
velopment of cholestasis (defined as conjugated bilirubin 쏜2.5
mg/dL; nonstandardized ␤ coefficient: Ҁ84 756; standardized
coefficient: Ҁ0.124; P ҃ 0.606) were not found to be significant
predictors of greater COC. This was interesting because these 2
factors are often thought to incur the greatest complexity of
patient care. However, a strongly positive correlation was found
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between those children with small-bowel lengths 쏝10% of that
predicted for gestational age and higher patient charges (P 쏝
0.003). This significance also persisted despite the removal of
nonsurvivors (nonstandardized ␤ coefficient: 507 403 앐
225 194; standardized coefficient: 0.623; 95% CIs: 28 177,
1 047 208; P ҃ 0.003). Univariate analysis of other factors that
could potentially influence survival, including the specific eti-
ology of SBS, the presence or absence of an ileocecal valve, and
the total number of septic episodes, showed no significant cor-
relation.
DISCUSSION
The care of patients with SBS is complex, is expensive, and
requires a long-term commitment by trained individuals (3, 31–
33). Predicting which patients will survive and their overall out-
comes was addressed well over 3 decades ago (34). Recently,
several evaluations of large pediatric intestinal failure centers
have identified survival rates of 앒70 – 80% (4, 13, 35). Risk
factors for survival in these series include the overall length of
retained small bowel, the presence or absence of an ileocecal
valve, and whether or not the child develops PN-associated liver
disease. Unfortunately, many of these patients may require long-
term PN for years. The care of a child with SBS is particularly
challenging, and, unlike in adults, is associated with a higher rate
of liver injury and difficulty in maintaining vascular access (10,
11, 13–15, 31, 36 –39).
Our study addressed a critical issue regarding the care of these
children—the overall financial charges that pediatric SBS pa-
tients incur during their care. Although previous reports have
estimated the COC to be between $125 000 and $250 000/y (18,
19, 40), most of these are estimates are based primarily on data
for adult patients. Furthermore, most of these values are not
based on complete data sets, but rather on estimations based on
idealized patients. Our findings show that the cost over the first
5 y after the onset of SBS is higher than some of these previously
estimated values— exceeding $1.6 million for the first 5 y of
treatment and ranges from 1.3 to 2.0 million dollars if PN is
required for all 5 y. The study also showed that charges were
predominately inpatient during the first year, and these charges
 COSTS OF SHORT-BOWEL SYNDROME 1557
Annual Total Cost Of Care accounted for a large portion (앒60%) of all charges incurred.
A However, we also unexpectedly found that the cost of home care
900,000
Nonsurvivors for these children continued to rise over time. This rise in home
Survivors
800,000 charges occurred despite predicted improvement for many of
700,000
these children during the same time period. This suggests that,
although there are numerous benefits to patients receiving home
600,000 care, costs for such care remain a substantial financial burden to
US Dollars

500,000 the patients’ families and insurance providers.

The cost of delivery of PN in the United States is a difficult
400,000 number to determine because of the large number of medical
300,000 providers and insurance payors. Additionally, costs of PN in the
United States appear to be disproportionately higher (4-fold)
200,000
than those in Canada and the United Kingdom, which makes an
100,000 extrapolation of costs from other countries difficult (41). An
estimate based on Medicare expenditures from 1989 to 1992
0
showed that yearly costs rose to 137 million dollars by the end of
1 2 3 4 5
Year
the analysis period (16). Extrapolation of comprehensive costs of
pediatric SBS from these numbers is not possible because these
B Annual Hospital-associated Costs costs were only for home PN, were primarily based on adult data,
and did not include the hospital-associated costs of pediatric
450,000
Nonsurvivors SBS. Furthermore, children with SBS (based on the diagnosis

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Survivors
400,000 “congenital disorders”) made up 쏝2.5% of the total population
350,000 analyzed. A recent comprehensive study of adult home PN pa-
tients indicated costs that were markedly less than those found in
300,000
our current study. In that study, Reddy and Malone (17) found
US Dollars

250,000 that costs (also based on charges) varied considerably between

200,000 patients. The mean PN charges were only $70 000/y, and mean
annual charges for enteral nutrition were merely $18 000. Im-
150,000
portantly, that study only dealt with patients older than 18 y,
100,000 again emphasizing the fact that pediatric SBS care and costs may
50,000 substantially differ from those of adult SBS. Additionally, the
indications for receiving home PN and enteral nutrition in that
0
study were not necessarily for SBS; thus, the charges probably do
1 2 3 4 5
not fully reflect the complex medical conditions associated with
Year
pediatric SBS patients. For example, the average number of
hospitalizations in Reddy and Malone’s study was between 1 and
C Annual Total Home Care Costs

2/y—far fewer than in our series of pediatric patients. Another

350,000
Nonsurvivors
Survivors
300,000
Total annual cost of care, per bowel length

250,000
$1,400,000
Less than 10% length
US Dollars

200,000 > 10% bowel length

$1,200,000
150,000
$1,000,000
100,000
US Dollars

$800,000
50,000

0 $600,000
1 2 3 4 5
Year $400,000

FIGURE 3. Mean (앐 SD) annual total cost of care for pediatric short-
bowel syndrome patients stratified as survivors and nonsurvivors. The annual $200,000
total cost of care was always greater in nonsurvivors, including the first year
of care for total costs (A), in-hospital costs (3B), and home-care costs (C). $0
Remarkably, even though patients who died spent a great deal of time in the 1 2 3 4 5

hospital, their home-care costs were consistently higher than those of patients Year

who ultimately survived. Costs are in US dollars (USD) corrected for infla-
tion to the year 2005.
FIGURE 4. Mean (앐 SD) annual total cost of care for pediatric short-
bowel syndrome patients stratified by an intestinal length 쏝10% or 욷10% of
that predicted by gestational age. Patients with an intestinal length 쏝10%
consistently had a higher cost of care for both in-hospital and home care.
Costs are in US dollars (USD) corrected for inflation to the year 2005.
1558 SPENCER ET AL
limitation of this study was that charges were estimates based on
1996 Medicare allowable charges, whereas the results in our
study represented actual charges submitted to third-party payors.
Although these costs were probably similar (aside from the 9-y
difference in inflationary correction), we believe our study al-
lowed for a more accurate assessment of patient charges for
pediatric SBS.
In-hospital charges accounted for more than one-half of all
charges. In-hospital charges directly correlated with the duration
of hospital days. Thus, in-hospital charges were greatest in the
first year after SBS and progressively decreased with time.
Clearly, the charges incurred by these patients accounted for the
bulk of their initial hospitalization, which was usually in an
intensive care unit setting and was quite lengthy. It is well-
established that home PN administration is economically much
less expensive than is in-hospital PN (42, 43). Therefore, it was
interesting that home-care charges continued to rise through year
5 of the study. A major component of this rise was the fact that
care shifted from the predominant inpatient side to home care
over the course of care. Nevertheless, if this was the only cause,
then it would seem that charges would plateau after year 3. We
suspect that despite the charges being corrected for inflationary
factors, the cost of medical care continued to rise in a greater
proportion throughout the study, and this would be reflected in
this progressive rise in charges over time. The causes of these
increased charges are not completely understood. It may also be
that as patients grow and age they may transition to a higher
requirement of PN (ie, 1- to 2-L volumes of infusion). Addition-
ally, more complex antimicrobials (ie, liposomal-based antifun-
gal therapy) may also contribute to these higher charges. The fact
that the combined charges of in-hospital and home care equaled
or exceeded $250 000/y, even after the fifth year after onset of
SBS, emphasizes the fact that previous estimates of the COC for
SBS patients have underestimated the ongoing costs of care for
pediatric SBS when compared with the charges actually in-
curred.
One potential, albeit small, source of error may arise from the
fact that we included outpatient clinic charges and the costs of
blood draws in the total hospital charges instead of in the home-
care charges. This was done simply because these charges are
billed through the university hospital accounts. Because of the
nature of our billing system, all outpatient clinic visits and out-
patient diagnostic test charges that were drawn in our clinic were
included in the in-hospital portion of the charges. Although doing
this may detract from total home-care charges, in fact, these visits
accounted for 쏝1% of all yearly COC. For example, 6 yearly
clinic encounters (level 3 or 4 charges of care ҂ 앒$150) would
amount to $900/y. Therefore, we simply kept the original hos-
pital data intact, because this small factor would not significantly
change the overall result of the study. Likewise, no significant
impact was made on home-care charges.
Costs in this study were estimated for a cohort of 41 infants and
children. Although this was a small study, we believe that our
patient population accurately reflected a broad spectrum of SBS
patients. The overall survival in our current report mirrored a
recent large series of 쏜100 SBS infants and children also re-
ported by our Intestinal Failure Center and other recent series (2,
4, 7, 10). Additionally, similar to other reports of children with
SBS, the ability to gain enteral independence was noted in more
than two-thirds of our patients (4, 10, 30). Of note was the fact
that many of our children gained such independence after several
years of home PN. This is noteworthy because several other
authors have expressed doubt that any patient with SBS is likely
to be weaned off of PN after 3 y of PN dependence (44, 45).
Importantly, these reports are in adults. The data strongly suggest
that children have a much greater capacity for intestinal adapta-
tion and regeneration after SBS than do adults. In our cohort of
patients, we observed 8 patients (out of 41) with a diagnosis of
SBS who were PN-dependent for a minimum of 3 y, who ulti-
mately were successfully weaned from PN 쏜3 y after the onset
of SBS; 3 were weaned off PN even after having received it for
5 y. Intensive nutritional care of infants and children with SBS
has been shown to be effective in weaning such patients from PN
who otherwise would have been maintained on this therapy (9).
It was interesting to note that those patients with a small-bowel
length 쏝10% of predicted for their gestational age incurred the
highest charges. The mean charges exceeded 2 million US dol-
lars—a level that typically exceeds the lifetime maximum ben-
efits of many third-party payors. Thus, families, and medical
providers may incur a large portion of this financial responsibil-
ity. This high financial cost may also place a significant burden
on many hospitals because 앒15% of patients younger than 18 y
Downloaded from ajcn.nutrition.org by guest on April 7, 2015
will lack insurance coverage for at least a portion of the time (46).
Thus, our findings may require a rethinking about the current
provider capitations on medical and surgical care. In a recent
analysis of survival in pediatric SBS patients, we found that those
patients with a small-bowel length 쏝10% of predicted had a
significantly lower survival than did those infants with longer
bowel lengths (4) (47). Conceivably, this high-risk group of
patients may possibly need to be identified early on for alterna-
tive therapies such as intestinal transplant assessment, because
determination of which patients may be candidates for transplan-
tation has been a challenge (48). It was also noteworthy that other
covariates in our study did not predict higher patient charges,
including the development of cholestasis and whether or not the
patient eventually died of SBS. It is, however, possible that the
early death of some of these patients resulted in a low total
amount of charges, which offset higher charges from these gen-
erally more complex patients. It is conceivable that as the care of
SBS patients becomes further coordinated (14, 15), as surgical
techniques become further advanced (49), as additional medical
therapies are used (50, 51), and as mortality rates continue to
decline (13), the overall COC for such patients may well decline
in future assessments of such patients.
In conclusion, this study was one of the first to comprehen-
sively examine the true cost of caring for a child with SBS. The
yearly costs of care are high, and home care continues to increase
with each year the patient remains on PN, despite the fact that this
care is predominately in a home setting after the first year after
the onset of SBS. The data also provide further support for the
financial benefit of providing this care at home rather than on an
inpatient basis. These numbers may prove useful when attempt-
ing to evaluate the financial burden a family faces with a child
with such a diagnosis. The data may also suggest a rethinking of
financial capitations that may be placed on such patients.
We thank Andreea Neaga, Jared Safran, Robert Drongowski, and Dana
Spivak for the initial preparation of the patient database.
The authors’ responsibilities were as follows—AUS: study design, data
collection, data analysis, interpretation of the data, and writing and editing of
the manuscript; DK: home-care data collection, home-care data analysis, and
interpretation of the home-care data; MM-B: data collection, data analysis,
interpretation of the data, and critical analysis of the financial data; DH: data
 COSTS OF SHORT-BOWEL SYNDROME
collection, data analysis, interpretation of the data, and critical analysis of the
financial data; JC: visiting nursing data collection, visiting nursing data
analysis, and interpretation of the visiting nursing data; CM: home-care data
collection, home-care data analysis, interpretation of the home-care data, and
writing and editing of the manuscript; and DHT (lead author): study concept,
study design, interpretation of data, and writing of the manuscript. None of
the authors had any financial conflict of interest with the company or orga-
nization sponsoring the research at the time the research was done.
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