- Email: [email protected]
Cost modeling for management strategies of uncomplicated gastroschisis
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Available online at www.sciencedirect.com
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Cost modeling for management strategies of uncomplicated gastroschisis James X. Wu, MD,a,b Steven L. Lee, MD,a,b,c,d and Daniel A. DeUgarte, MDa,b,c,d,* a
David Geffen School of Medicine at UCLA Department of Surgery, UCLA c Department of Surgery, Harbor-UCLA Medical Center d LA Biomed b
article info
abstract
Article history:
Introduction: Compared to operative fascial closure, nonoperative flap and/or skin-closure
Received 4 February 2016
repair for gastroschisis has several potential advantages: avoidance of anesthesia,
Received in revised form
decreased pain, and improved cosmesis. Disadvantages include a higher risk of hernia. We
14 April 2016
hypothesized that routine nonoperative closure results in cost savings versus conventional
Accepted 10 June 2016
management in uncomplicated gastroschisis.
Available online xxx
Methods: A decision tree was constructed to compare three different strategies for the management of uncomplicated gastroschisis: nonoperative closure, primary closure, and
Keywords:
routine silo. Model variables were abstracted from a literature review and the Medicare
Gastroschisis
Physician Fee schedule. Uncertainty surrounding model parameters was assessed via
Cost
one-way and probabilistic sensitivity analyses.
Nonoperative
Results: According to our model, the nonoperative strategy for uncomplicated gastroschisis
Sutureless
was the least costly, with an expected cost of $198,085 per patient. Primary closure
Flap
cost $208,763 per patient. Routine silo placement was the most costly, $239,038 per
Ward reduction
patient. One-way sensitivity analysis suggested the cost of primary closure would be
Plastic
less costly than nonoperative management if the initial success rate of nonoperative management was less than 35.4% or if the initial success rate of primary operative closure
was
greater
than
87.8%.
Probabilistic
sensitivity
analysis
found
that
nonoperative management was the least costly strategy among 97.4% of 10,000 Monte Carlo simulations. Conclusions: A nonoperative strategy for uncomplicated gastroschisis with routine attempted flap and/or skin closure repair is less costly than strategies using routine primary closure and routine silo placement. Given the expected cost savings and other potential advantages of the nonoperative strategy (including avoidance of general anesthesia), more studies examining outcomes of the flap and/or skin closure are indicated. ª 2016 Elsevier Inc. All rights reserved.
* Corresponding author. Department of Surgery, UCLA and Harbor-UCLA, David Geffen School of Medicine UCLA, Box 709818, 10833 Le Conte Avenue, Los Angeles, CA 90095-7098. Tel.: þ1 310 206 2429; fax: þ1 310 206 1120. E-mail address: [email protected] (D.A. DeUgarte). 0022-4804/$ e see front matter ª 2016 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jss.2016.06.039
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j o u r n a l o f s u r g i c a l r e s e a r c h 2 0 1 6 ( - ) 1 e6
Introduction
Decision model
Gastroschisis is the most common congenital abdominal wall defect, characterized by the herniation of intestines through an opening in the abdominal wall. Gastroschisis continues to increase in prevalence, affecting an estimated 4.9 per 10,000 births.1 Treatment is prolonged and costly. The length of neonatal stay for gastroschisis averages more than 1 mo, and hospital costs can exceed $100,000.2-5 Significant debate exists over the optimal surgical management strategy of gastroschisis.6 Originally, patients underwent attempted primary closure, and silo placement was only performed when initial closure failed. Later, the development of a preformed silo resulted in many institutions adopting a strategy of routine silo placement with the purported benefit of decreasing potential complications of high intra-abdominal pressures associated with attempts at primary closure.7,8 The results for routine silo placement have been mixed with some concerns that this strategy may lead to longer lengths of stay and higher hospital costs.7,9,10 Recently, many centers have initiated a nonoperative strategy involving umbilical flap or skin closure at the bedside (reserving an operating room team and general anesthesia only for complex cases).11,12 The benefits of nonoperative strategies (using “umbilical flap,” “sutureless,” “ward reduction,” and “plastic” techniques) include avoidance of general anesthesia, theoretically reduced intra-abdominal pressures and pain (because the fascia is not closed), and reported excellent cosmetic results.12-23 The main disadvantage of the nonoperative strategy is that most patients are left with a fascial defect, a proportion of which will require delayed repair.14-17,20,21 Decision analysis techniques offer a useful framework for comparing treatment strategies with complex tradeoffs.24-26 Using computer-generated decision tree software, we aimed to compare the expected treatment costs of different management strategies for uncomplicated gastroschisis. We hypothesized a nonoperative strategy with attempted flap and/or skin closure would be the least costly management of uncomplicated gastroschisis.
We constructed a decision tree using decision analysis software (TreeAge Pro, Williamstown, MA). Our tree compared three management strategies: (1) nonoperative, (2) primary closure, and (3) and routine silo. Figure 1 schematically illustrates the treatment strategies and potential outcomes.
Methods Reference case We began by defining the reference case: a hypothetical patient with uncomplicated gastroschisis. We elected to exclude complicated gastroschisis because surgical management strategies for these patients may be influenced by the patient’s condition (e.g., intestinal atresia and/or necrosis, liver herniation). Furthermore, complicated gastroschisis occurs in only a minority of patients (<15%).27 Finally, patients with complicated gastroschisis are often outliers with respect to length of stay and hospital costs that would be difficult to account for in a decision model.27
Nonoperative strategy The probability of initial success of the nonoperative strategy (P1) was defined as the likelihood of achieving bedside umbilical flap or skin closure without the need for general anesthesia or an operating room team within the first day of life. For the purposes of the decision tree model, patients who have initial failure of the nonoperative strategy undergo silo placement and have a probability of either delayed operative repair with general anesthesia and an operating room team (P2) or nonoperative flap and/or skin closure.
Primary closure strategy The probability of initial success of the primary closure strategy (P3) was defined as the probability of fascial closure with an attempt at primary operative closure. Patients who have initial failure of the primary closure strategy undergo silo placement and delayed operative repair.
Routine silo strategy In this strategy, we assume that all patients (P4 ¼ 100%) in this strategy undergo routine silo placement with delayed operative repair. A mean number of silo days for each scenario requiring delayed closure were extrapolated from the literature. The length of stay for each scenario was extrapolated from a combination of literature review and estimated increases in length of stay for each day of delay in closure (estimated 2 d longer length of neonatal stay for each additional day of silo utilization).9,28 The probability of a hernia (fascial defect) being present, and the probability of the hernia requiring repair were extrapolated from the literature for both nonoperative skin and/or flap closure (P5) and operative fascial closure (P6). We assumed that patients would require two-surgeon follow-up consultations if a hernia was present at the time of discharge and an additional follow-up consultation if the hernia did not resolve and required surgical repair. The primary closure strategy was used as the reference group for all comparison of costs.
Literature review A PubMed search using the keyword “gastroschisis” was performed. All abstracts that contained information about outcomes, cost, and length of stay were reviewed for relevant model variables.
Model variables: clinical events Probabilities of initial management success and/or failure, need for operative closure, mean silo days for delayed closure,
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NonOperave
p1
1 - p1
Successful NonOp Closure
p5 p2
Failure -> Silo Placement
1 – p2
Delayed NonOp Closure
Hernia Aer Non-Op Closure
p5
Delayed OR Closure p6
Primary Closure
p3
Successful OR Closure p6
1 – p3
Roune Silo
Failure -> Silo Placement
p4
Delayed OR Closure
Delayed OR Closure
Hernia Aer OR Closure
p6
p6
Figure 1 e Schematic diagram of decision tree model. The probabilities used for each branching point in the decision tree are labeled to correspond with Table 1. In this model, the cost of follow-up and operative repair of hernia are included.
and mean length of stay were abstracted from a review of the literature. Index values were primarily based on results from randomized controlled trial and prospective studies. Ranges were obtained by including all results reported. Efforts were made to extrapolate data only for uncomplicated gastroschisis patients. Model variables for clinical events are listed in Table 1.
Model variables: costs In our model, we accounted only for direct medical costs, comprised of hospital costs and surgeon fees. Hospital costs included costs of the neonatal intensive care unit stay and operative and/or facility fees were abstracted from a review of the literature and data from the Healthcare Cost and Utilization Project.4 Surgeon fees were obtained from the Medicare Physician Fee schedule.31 We did not consider indirect costs to the patient or healthcare provider in any treatment strategy. To simplify the model, we assumed that all hospital days were spent in the neonatal intensive care unit. Model costs are listed in Table 2.29,30,32,33
Sensitivity analysis One-way sensitivity analysis was performed on each outcome probability using routine primary closure as the reference group. For clinical probabilities, sensitivity analysis estimates the most cost-effective treatment strategy when individual values are allowed to vary between 0 and 1, the maximum values compatible with the model. For cost estimates, we
allowed values vary from $0 to an upper limit of twice the index value. For length of hospital stay, we allow values to vary from 0 to twice the index length of stay. For probabilistic sensitivity analysis, all model variables (probabilities and costs) were set as static with triangular frequency distributions. We performed 10,000 separate Monte Carlo simulations during which each model variable was assigned a different value from its triangular distribution. The parameters for each probability’s triangular distribution reflect the range of values reported in existing literature or a maximum variance of 50% of the index case value.
Results According to our model, nonoperative management of uncomplicated gastroschisis was the least costly strategy, which cost $198,085 per patient. Primary closure cost $208,763 per patient. Routine silo placement was the most costly, $239,038 per patient. The model variables that could independently impact the model outcome are summarized in Table 3. One-way sensitivity analysis suggested the cost of primary closure would be less costly than nonoperative management if the initial success rate of nonoperative management was less than 35.4% or if the initial success rate of primary operative closure was greater than 87.8%. Since the cost of a single day in the neonatal intensive care unit was high relative to the other costs in our model, many of the cost variables that did not affect length of stay did not have a significant effect on
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j o u r n a l o f s u r g i c a l r e s e a r c h 2 0 1 6 ( - ) 1 e6
Table 1 e Model variables: clinical events.* Nonoperative
Primary closure
Routine silo
12-22
7-10,12,13,15,16,20,21,28-30
7-10,12,13,28,30
References Probability of closure P1: probability of initial nonoperative closure
60% (47%-90%)
d
d
P2: probability of operative closurey
30% (0%-39%)
d
d
P3: probability of initial operative closure
d
75% (39%-90%)
d
P4: probability of delayed operative closure
d
d
100%
If delayed closure, mean number of silo days Mean silo days
d
Mean silo days if failure
d
10 (6-13.5)
10 (6-13.5)
Initial success
35 (26-36)
40 (31-50)
Initial failure treated nonoperative
40 (30-50)
Delayed operative closure
50 (40-60)
6 (3-9) d
Mean length of stay (d)
d
45 (35-46) d
50 (40-60)
d
40% (0%-42%)
40% (0%-42%)
12.5% (5%-25%)
12.5% (5%-25%)
Probability of hernia requiring follow-up Nonoperative closure
100% (22%-100%)
Operative closure
40% (0%-42%)
Probability of hernia not resolving Nonoperative closure
10% (7%-40%)
Operative closure
12.5% (5%-25%)
Cumulate probability of having hernia that requires follow-up and repair P5: nonoperative closure
10% (15% to 40%)
P6: operative closure
5% (0% to 11%)
5% (0% to 11%)
5% (0% to 11%)
Probabilities used for the cost analysis (as illustrated in Figure 1) are labeled accordingly (P1, P2, P3, P4, P5, and P6). * Results are reported as index values (range for Monte Carlo simulation). y Probably of requiring operative closure if there is initial failure with nonoperative management strategy.
model outcomes. Conversely, all length of stay variables could affect model outcome. Assuming expected length of stay for successful primary closure is 35 d, primary closure would be less costly than nonoperative closure if patients that underwent nonoperative repair had a length of stay >38.6 d.
Monte Carlo probabilistic sensitivity analysis demonstrated that the nonoperative strategy was the least costly compared with primary closure and routine silo placement in 97.4% of 10,000 simulated cases.
Discussion Table 2 e Model variables: medical costs.* Costs
Index (range)
References
Neonatal intensive care unit (NICU) $5000 ($2600-$15,000)
2-4,9,32,33
Operative/facility costs
$7500 ($5000-$15,000)
2,15
Surgeon fee (initial closure)
$5000 ($3333-$10,000)
2,31
Surgeon fee (delayed closure)
$1200 ($1100-$3300)
2,31
NICU cost per day Gastroschisis closure
Hernia closure Surgeon follow-up visit
$200 ($150-$450)
Operative/facility costs
$3000 ($2000-$6000)
Surgeon fee (hernia repair)
$350 ($300-$900)
31 4 31
* Results are reported as index values (range for Monte Carlo simulation).
In our model, a nonoperative management strategy for uncomplicated gastroschisis was less costly than primary-closure and routine-silo strategies. Our findings are consistent with those reported by other studies.15,23 The cost reduction that we observed in our model is primarily because of reported reductions in length of hospital stay with the nonoperative technique. The reported reductions in length of hospital day are likely due to avoidance of intubation, muscle relaxation, and general anesthesia. Furthermore, avoiding fascial closure likely decreases intra-abdominal pressure and reduces the need for opioids, which may prolong ileus. Although fascial defects are frequently observed with a nonoperative approach, they appear to behave like umbilical hernias and require operative repair less frequently than expected. In addition, costs of outpatient hernia repair are minimal in comparison to the added costs of operative gastroschisis repair during the neonatal period. The nonoperative technique also has excellent, often scarless cosmetic results. Because of these
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Table 3 e One-way sensitivity analysis. Model variable
Threshold when primary closure is less costly than nonoperative closure
Success of initial primary closure
>87.8%
Success of nonoperative closure
<35.4%
% Requiring operative closure after failed nonoperative closure
>76.8%
Length of stay for successful primary closure
<32.1 d (index 35)
Length of stay for failed initial primary closure
<41.5 d (index 50)
Length of stay for successful nonoperative closure
>38.6 d (index 35)
Length of stay for failed nonoperative closure requiring operative repair
>67.8 d (index 50)
Length of stay for failed nonoperative closure with subsequent successful nonoperative repair
>47.6 d (index 40)
5
high costs of gastroschisis, we provide additional support for the routine implementation of a nonoperative strategy for uncomplicated gastroschisis. Not surprisingly, many centers are now adopting this approach. We have already observed the practice pendulum swing from a preference for primary closure to routine silo and back.6-8 Additional time and evaluation will determine if the nonoperative management strategy takes root and is here to stay.
Acknowledgment Authors’ contributions: D.A.D. contributed to study conception, study design, data acquisition, data analysis, drafting the article, and final approval. J.X.W. contributed to study conception, study design, data acquisition, data analysis, drafting the article, and final approval. S.L.L. contributed to study conception, study design, data interpretation, article revision, and final approval.
Disclosure
*All other model variables could not independently change model outcome.
reported benefits, nonoperative techniques are now being adopted by many centers.13 One of the compelling reasons to use nonoperative closure techniques is the potential to avoid general anesthesia. In particular, there have been many recent concerns about the impact of general anesthesia on the neonatal brain.34,35 Avoiding fascial closure and the need for general anesthesia or delaying it to an elective hernia repair when the patient is older is appealing. The concerns about general anesthesia during the neonatal period have not been completely elucidated, and an additional study is required.35 There are several potential limitations to this study. First, our data were derived from the literature, and the favorable results for the nonoperative technique may have been influenced by publication bias. Every effort was made to obtain index values from randomized controlled trials and prospective studies; however, few studies had these designs. Second, the rate of hernias requiring operative repair is likely underreported because of short follow-up or loss to follow-up in both the nonoperative and operative strategies. Third, our study did not evaluate the impact of the management strategies on quality of life or well-being of the family with respect to the impact of an “operation,” an unrepaired hernia, or delay in discharge. Finally, our model was simplified to address uncomplicated gastroschisis, and the results are not necessarily generalizable to all cases of gastroschisis. Interestingly, the Canadian Pediatric Surgery network prospective registry results demonstrate similar outcomes between “flap” closure and fascial closure techniques even in high-risk patients suggesting that the nonoperative strategy is suitable for all gastroschisis patients.13 In summary, a nonoperative strategy for the management of gastroschisis appears to have many benefits including potential cost savings. Given the increasing prevalence and
The authors report no proprietary or commercial interest in any product mentioned or concept discussed in this article.
references
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24. Cronenwett JL, Birkmeyer JD, Nackman GB, et al. Costeffectiveness of carotid endarterectomy in asymptomatic patients. J Vasc Surg. 1997;25:298e311. 25. Stroupe KT, Morrison DA, Hlatky MA, et al. Cost-effectiveness of coronary artery bypass grafts versus percutaneous coronary intervention for revascularization of high-risk patients. Circulation. 2006;114:1251e1257. 26. Wu JX, Dawes AJ, Sacks GD, Brunicardi FC, Keeler EB. Cost effectiveness of nonoperative management versus laparoscopic appendectomy for acute uncomplicated appendicitis. Surgery. 2015;158:712e721. 27. Overcash RT, DeUgarte DA, Stephenson ML, et al. Factors associated with gastroschisis outcomes. Obstet Gynecol. 2014;124:551e557. 28. Lusk LA, Brown EG, Overcash RT, et al. Multi-institutional practice patterns and outcomes in uncomplicated gastroschisis: a report from the University of California Fetal Consortium (UCfC). J Pediatr Surg. 2014;49:1782e1786. 29. Vegunta RK, Wallace LJ, Leonardi MR, et al. Perinatal management of gastroschisis: analysis of a newly established clinical pathway. J Pediatr Surg. 2005;40:528e534. 30. Owen A, Marven S, Jackson L, et al. Experience of bedside preformed silo staged reduction and closure for gastroschisis. J Pediatr Surg. 2006;41:1830e1835. 31. Services CfMM. Physician fee schedule search Tool; 2014. Available at: http://www.cms.gov/apps/physician-feeschedule/search/search-criteria.aspx. Accessed August 2015. 32. Centers for Disease C, Prevention. Hospital stays, hospital charges, and in-hospital deaths among infants with selected birth defectseUnited States, 2003. MMWR Morb Mortal Wkly Rep. 2007;56:25e29. 33. Cain MA, Salemi JL, Paul Tanner J, et al. Perinatal outcomes and hospital costs in gastroschisis based on gestational age at delivery. Obstet Gynecol. 2014;124:543e550. 34. Davidson AJ, Disma N, de Graaff JC, et al. Neurodevelopmental outcome at 2 years of age after general anaesthesia and awake-regional anaesthesia in infancy (GAS): an international multicentre, randomised controlled trial. Lancet. 2016;387:239e250. 35. Flick RP, Katusic SK, Colligan RC, et al. Cognitive and behavioral outcomes after early exposure to anesthesia and surgery. Pediatrics. 2011;128:e1053ee1061. Q2
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Available online at www.sciencedirect.com
ScienceDirect journal homepage: www.JournalofSurgicalResearch.com
Cost modeling for management strategies of uncomplicated gastroschisis James X. Wu, MD,a,b Steven L. Lee, MD,a,b,c,d and Daniel A. DeUgarte, MDa,b,c,d,* a
David Geffen School of Medicine at UCLA Department of Surgery, UCLA c Department of Surgery, Harbor-UCLA Medical Center d LA Biomed b
article info
abstract
Article history:
Introduction: Compared to operative fascial closure, nonoperative flap and/or skin-closure
Received 4 February 2016
repair for gastroschisis has several potential advantages: avoidance of anesthesia,
Received in revised form
decreased pain, and improved cosmesis. Disadvantages include a higher risk of hernia. We
14 April 2016
hypothesized that routine nonoperative closure results in cost savings versus conventional
Accepted 10 June 2016
management in uncomplicated gastroschisis.
Available online xxx
Methods: A decision tree was constructed to compare three different strategies for the management of uncomplicated gastroschisis: nonoperative closure, primary closure, and
Keywords:
routine silo. Model variables were abstracted from a literature review and the Medicare
Gastroschisis
Physician Fee schedule. Uncertainty surrounding model parameters was assessed via
Cost
one-way and probabilistic sensitivity analyses.
Nonoperative
Results: According to our model, the nonoperative strategy for uncomplicated gastroschisis
Sutureless
was the least costly, with an expected cost of $198,085 per patient. Primary closure
Flap
cost $208,763 per patient. Routine silo placement was the most costly, $239,038 per
Ward reduction
patient. One-way sensitivity analysis suggested the cost of primary closure would be
Plastic
less costly than nonoperative management if the initial success rate of nonoperative management was less than 35.4% or if the initial success rate of primary operative closure
was
greater
than
87.8%.
Probabilistic
sensitivity
analysis
found
that
nonoperative management was the least costly strategy among 97.4% of 10,000 Monte Carlo simulations. Conclusions: A nonoperative strategy for uncomplicated gastroschisis with routine attempted flap and/or skin closure repair is less costly than strategies using routine primary closure and routine silo placement. Given the expected cost savings and other potential advantages of the nonoperative strategy (including avoidance of general anesthesia), more studies examining outcomes of the flap and/or skin closure are indicated. ª 2016 Elsevier Inc. All rights reserved.
* Corresponding author. Department of Surgery, UCLA and Harbor-UCLA, David Geffen School of Medicine UCLA, Box 709818, 10833 Le Conte Avenue, Los Angeles, CA 90095-7098. Tel.: þ1 310 206 2429; fax: þ1 310 206 1120. E-mail address: [email protected] (D.A. DeUgarte). 0022-4804/$ e see front matter ª 2016 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jss.2016.06.039
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j o u r n a l o f s u r g i c a l r e s e a r c h 2 0 1 6 ( - ) 1 e6
Introduction
Decision model
Gastroschisis is the most common congenital abdominal wall defect, characterized by the herniation of intestines through an opening in the abdominal wall. Gastroschisis continues to increase in prevalence, affecting an estimated 4.9 per 10,000 births.1 Treatment is prolonged and costly. The length of neonatal stay for gastroschisis averages more than 1 mo, and hospital costs can exceed $100,000.2-5 Significant debate exists over the optimal surgical management strategy of gastroschisis.6 Originally, patients underwent attempted primary closure, and silo placement was only performed when initial closure failed. Later, the development of a preformed silo resulted in many institutions adopting a strategy of routine silo placement with the purported benefit of decreasing potential complications of high intra-abdominal pressures associated with attempts at primary closure.7,8 The results for routine silo placement have been mixed with some concerns that this strategy may lead to longer lengths of stay and higher hospital costs.7,9,10 Recently, many centers have initiated a nonoperative strategy involving umbilical flap or skin closure at the bedside (reserving an operating room team and general anesthesia only for complex cases).11,12 The benefits of nonoperative strategies (using “umbilical flap,” “sutureless,” “ward reduction,” and “plastic” techniques) include avoidance of general anesthesia, theoretically reduced intra-abdominal pressures and pain (because the fascia is not closed), and reported excellent cosmetic results.12-23 The main disadvantage of the nonoperative strategy is that most patients are left with a fascial defect, a proportion of which will require delayed repair.14-17,20,21 Decision analysis techniques offer a useful framework for comparing treatment strategies with complex tradeoffs.24-26 Using computer-generated decision tree software, we aimed to compare the expected treatment costs of different management strategies for uncomplicated gastroschisis. We hypothesized a nonoperative strategy with attempted flap and/or skin closure would be the least costly management of uncomplicated gastroschisis.
We constructed a decision tree using decision analysis software (TreeAge Pro, Williamstown, MA). Our tree compared three management strategies: (1) nonoperative, (2) primary closure, and (3) and routine silo. Figure 1 schematically illustrates the treatment strategies and potential outcomes.
Methods Reference case We began by defining the reference case: a hypothetical patient with uncomplicated gastroschisis. We elected to exclude complicated gastroschisis because surgical management strategies for these patients may be influenced by the patient’s condition (e.g., intestinal atresia and/or necrosis, liver herniation). Furthermore, complicated gastroschisis occurs in only a minority of patients (<15%).27 Finally, patients with complicated gastroschisis are often outliers with respect to length of stay and hospital costs that would be difficult to account for in a decision model.27
Nonoperative strategy The probability of initial success of the nonoperative strategy (P1) was defined as the likelihood of achieving bedside umbilical flap or skin closure without the need for general anesthesia or an operating room team within the first day of life. For the purposes of the decision tree model, patients who have initial failure of the nonoperative strategy undergo silo placement and have a probability of either delayed operative repair with general anesthesia and an operating room team (P2) or nonoperative flap and/or skin closure.
Primary closure strategy The probability of initial success of the primary closure strategy (P3) was defined as the probability of fascial closure with an attempt at primary operative closure. Patients who have initial failure of the primary closure strategy undergo silo placement and delayed operative repair.
Routine silo strategy In this strategy, we assume that all patients (P4 ¼ 100%) in this strategy undergo routine silo placement with delayed operative repair. A mean number of silo days for each scenario requiring delayed closure were extrapolated from the literature. The length of stay for each scenario was extrapolated from a combination of literature review and estimated increases in length of stay for each day of delay in closure (estimated 2 d longer length of neonatal stay for each additional day of silo utilization).9,28 The probability of a hernia (fascial defect) being present, and the probability of the hernia requiring repair were extrapolated from the literature for both nonoperative skin and/or flap closure (P5) and operative fascial closure (P6). We assumed that patients would require two-surgeon follow-up consultations if a hernia was present at the time of discharge and an additional follow-up consultation if the hernia did not resolve and required surgical repair. The primary closure strategy was used as the reference group for all comparison of costs.
Literature review A PubMed search using the keyword “gastroschisis” was performed. All abstracts that contained information about outcomes, cost, and length of stay were reviewed for relevant model variables.
Model variables: clinical events Probabilities of initial management success and/or failure, need for operative closure, mean silo days for delayed closure,
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NonOperave
p1
1 - p1
Successful NonOp Closure
p5 p2
Failure -> Silo Placement
1 – p2
Delayed NonOp Closure
Hernia Aer Non-Op Closure
p5
Delayed OR Closure p6
Primary Closure
p3
Successful OR Closure p6
1 – p3
Roune Silo
Failure -> Silo Placement
p4
Delayed OR Closure
Delayed OR Closure
Hernia Aer OR Closure
p6
p6
Figure 1 e Schematic diagram of decision tree model. The probabilities used for each branching point in the decision tree are labeled to correspond with Table 1. In this model, the cost of follow-up and operative repair of hernia are included.
and mean length of stay were abstracted from a review of the literature. Index values were primarily based on results from randomized controlled trial and prospective studies. Ranges were obtained by including all results reported. Efforts were made to extrapolate data only for uncomplicated gastroschisis patients. Model variables for clinical events are listed in Table 1.
Model variables: costs In our model, we accounted only for direct medical costs, comprised of hospital costs and surgeon fees. Hospital costs included costs of the neonatal intensive care unit stay and operative and/or facility fees were abstracted from a review of the literature and data from the Healthcare Cost and Utilization Project.4 Surgeon fees were obtained from the Medicare Physician Fee schedule.31 We did not consider indirect costs to the patient or healthcare provider in any treatment strategy. To simplify the model, we assumed that all hospital days were spent in the neonatal intensive care unit. Model costs are listed in Table 2.29,30,32,33
Sensitivity analysis One-way sensitivity analysis was performed on each outcome probability using routine primary closure as the reference group. For clinical probabilities, sensitivity analysis estimates the most cost-effective treatment strategy when individual values are allowed to vary between 0 and 1, the maximum values compatible with the model. For cost estimates, we
allowed values vary from $0 to an upper limit of twice the index value. For length of hospital stay, we allow values to vary from 0 to twice the index length of stay. For probabilistic sensitivity analysis, all model variables (probabilities and costs) were set as static with triangular frequency distributions. We performed 10,000 separate Monte Carlo simulations during which each model variable was assigned a different value from its triangular distribution. The parameters for each probability’s triangular distribution reflect the range of values reported in existing literature or a maximum variance of 50% of the index case value.
Results According to our model, nonoperative management of uncomplicated gastroschisis was the least costly strategy, which cost $198,085 per patient. Primary closure cost $208,763 per patient. Routine silo placement was the most costly, $239,038 per patient. The model variables that could independently impact the model outcome are summarized in Table 3. One-way sensitivity analysis suggested the cost of primary closure would be less costly than nonoperative management if the initial success rate of nonoperative management was less than 35.4% or if the initial success rate of primary operative closure was greater than 87.8%. Since the cost of a single day in the neonatal intensive care unit was high relative to the other costs in our model, many of the cost variables that did not affect length of stay did not have a significant effect on
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j o u r n a l o f s u r g i c a l r e s e a r c h 2 0 1 6 ( - ) 1 e6
Table 1 e Model variables: clinical events.* Nonoperative
Primary closure
Routine silo
12-22
7-10,12,13,15,16,20,21,28-30
7-10,12,13,28,30
References Probability of closure P1: probability of initial nonoperative closure
60% (47%-90%)
d
d
P2: probability of operative closurey
30% (0%-39%)
d
d
P3: probability of initial operative closure
d
75% (39%-90%)
d
P4: probability of delayed operative closure
d
d
100%
If delayed closure, mean number of silo days Mean silo days
d
Mean silo days if failure
d
10 (6-13.5)
10 (6-13.5)
Initial success
35 (26-36)
40 (31-50)
Initial failure treated nonoperative
40 (30-50)
Delayed operative closure
50 (40-60)
6 (3-9) d
Mean length of stay (d)
d
45 (35-46) d
50 (40-60)
d
40% (0%-42%)
40% (0%-42%)
12.5% (5%-25%)
12.5% (5%-25%)
Probability of hernia requiring follow-up Nonoperative closure
100% (22%-100%)
Operative closure
40% (0%-42%)
Probability of hernia not resolving Nonoperative closure
10% (7%-40%)
Operative closure
12.5% (5%-25%)
Cumulate probability of having hernia that requires follow-up and repair P5: nonoperative closure
10% (15% to 40%)
P6: operative closure
5% (0% to 11%)
5% (0% to 11%)
5% (0% to 11%)
Probabilities used for the cost analysis (as illustrated in Figure 1) are labeled accordingly (P1, P2, P3, P4, P5, and P6). * Results are reported as index values (range for Monte Carlo simulation). y Probably of requiring operative closure if there is initial failure with nonoperative management strategy.
model outcomes. Conversely, all length of stay variables could affect model outcome. Assuming expected length of stay for successful primary closure is 35 d, primary closure would be less costly than nonoperative closure if patients that underwent nonoperative repair had a length of stay >38.6 d.
Monte Carlo probabilistic sensitivity analysis demonstrated that the nonoperative strategy was the least costly compared with primary closure and routine silo placement in 97.4% of 10,000 simulated cases.
Discussion Table 2 e Model variables: medical costs.* Costs
Index (range)
References
Neonatal intensive care unit (NICU) $5000 ($2600-$15,000)
2-4,9,32,33
Operative/facility costs
$7500 ($5000-$15,000)
2,15
Surgeon fee (initial closure)
$5000 ($3333-$10,000)
2,31
Surgeon fee (delayed closure)
$1200 ($1100-$3300)
2,31
NICU cost per day Gastroschisis closure
Hernia closure Surgeon follow-up visit
$200 ($150-$450)
Operative/facility costs
$3000 ($2000-$6000)
Surgeon fee (hernia repair)
$350 ($300-$900)
31 4 31
* Results are reported as index values (range for Monte Carlo simulation).
In our model, a nonoperative management strategy for uncomplicated gastroschisis was less costly than primary-closure and routine-silo strategies. Our findings are consistent with those reported by other studies.15,23 The cost reduction that we observed in our model is primarily because of reported reductions in length of hospital stay with the nonoperative technique. The reported reductions in length of hospital day are likely due to avoidance of intubation, muscle relaxation, and general anesthesia. Furthermore, avoiding fascial closure likely decreases intra-abdominal pressure and reduces the need for opioids, which may prolong ileus. Although fascial defects are frequently observed with a nonoperative approach, they appear to behave like umbilical hernias and require operative repair less frequently than expected. In addition, costs of outpatient hernia repair are minimal in comparison to the added costs of operative gastroschisis repair during the neonatal period. The nonoperative technique also has excellent, often scarless cosmetic results. Because of these
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Table 3 e One-way sensitivity analysis. Model variable
Threshold when primary closure is less costly than nonoperative closure
Success of initial primary closure
>87.8%
Success of nonoperative closure
<35.4%
% Requiring operative closure after failed nonoperative closure
>76.8%
Length of stay for successful primary closure
<32.1 d (index 35)
Length of stay for failed initial primary closure
<41.5 d (index 50)
Length of stay for successful nonoperative closure
>38.6 d (index 35)
Length of stay for failed nonoperative closure requiring operative repair
>67.8 d (index 50)
Length of stay for failed nonoperative closure with subsequent successful nonoperative repair
>47.6 d (index 40)
5
high costs of gastroschisis, we provide additional support for the routine implementation of a nonoperative strategy for uncomplicated gastroschisis. Not surprisingly, many centers are now adopting this approach. We have already observed the practice pendulum swing from a preference for primary closure to routine silo and back.6-8 Additional time and evaluation will determine if the nonoperative management strategy takes root and is here to stay.
Acknowledgment Authors’ contributions: D.A.D. contributed to study conception, study design, data acquisition, data analysis, drafting the article, and final approval. J.X.W. contributed to study conception, study design, data acquisition, data analysis, drafting the article, and final approval. S.L.L. contributed to study conception, study design, data interpretation, article revision, and final approval.
Disclosure
*All other model variables could not independently change model outcome.
reported benefits, nonoperative techniques are now being adopted by many centers.13 One of the compelling reasons to use nonoperative closure techniques is the potential to avoid general anesthesia. In particular, there have been many recent concerns about the impact of general anesthesia on the neonatal brain.34,35 Avoiding fascial closure and the need for general anesthesia or delaying it to an elective hernia repair when the patient is older is appealing. The concerns about general anesthesia during the neonatal period have not been completely elucidated, and an additional study is required.35 There are several potential limitations to this study. First, our data were derived from the literature, and the favorable results for the nonoperative technique may have been influenced by publication bias. Every effort was made to obtain index values from randomized controlled trials and prospective studies; however, few studies had these designs. Second, the rate of hernias requiring operative repair is likely underreported because of short follow-up or loss to follow-up in both the nonoperative and operative strategies. Third, our study did not evaluate the impact of the management strategies on quality of life or well-being of the family with respect to the impact of an “operation,” an unrepaired hernia, or delay in discharge. Finally, our model was simplified to address uncomplicated gastroschisis, and the results are not necessarily generalizable to all cases of gastroschisis. Interestingly, the Canadian Pediatric Surgery network prospective registry results demonstrate similar outcomes between “flap” closure and fascial closure techniques even in high-risk patients suggesting that the nonoperative strategy is suitable for all gastroschisis patients.13 In summary, a nonoperative strategy for the management of gastroschisis appears to have many benefits including potential cost savings. Given the increasing prevalence and
The authors report no proprietary or commercial interest in any product mentioned or concept discussed in this article.
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