The jury is still out: changes in gastroschisis management over the last decade are associated with both benefits and shortcomings

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1 Journal of Pediatric Surgery (2012) 47, The jury is still out: changes in gastroschisis management over the last decade are associated with both benefits and shortcomings Brent R. Weil a, Charles M. Leys a,b, Frederick J. Rescorla a,b, a Department of Surgery, Indiana University School of Medicine, Indianapolis, IN 46202, USA b Section of Pediatric Surgery, Indiana University School of Medicine, Indianapolis, IN 46202, USA Received 28 September 2011; accepted 6 October 2011 Key words: Gastroschisis; Preformed silo; Abdominal wall defects; Congenital anomalies Abstract Purpose: Management of gastroschisis has shifted from early primary closure to preformed silo placement and delayed closure. We aimed to identify how closure techniques have changed and how outcomes have been affected. Methods: Records of patients undergoing gastroschisis closure at a single institution from 2000 to 2009 were reviewed. Patient characteristics and outcomes were collected and compared among those undergoing primary closure vs preformed silo placement. Outcomes were also compared in an era when primary closure predominated ( ) vs one when primary silo predominated ( ). Results: From 2000 to 2009, 203 patients underwent gastroschisis closure. Primary closure was performed in 50% of patients from 2000 to 2002 vs 12.3% from 2003 to Preformed silos were placed in 34.7% of patients from 2000 to 2002 vs 84.4% from 2003 to Patients treated from 2000 to 2002 experienced shorter hospital stays and shorter time to achievement of full enteral nutrition. Patients treated from 2003 to 2009 developed fewer ventral hernias and wound infections and required less ventilator days. Patients undergoing early primary closure developed ventral hernias at higher rates compared with those treated with preformed silos. Intensive care unit stay was longer for patients receiving preformed silos. Conclusion: Change in our management strategy has resulted in prolonged intensive care unit stay and time to full feeds but reduced postoperative hernias and wound infections Elsevier Inc. All rights reserved. The surgical management of gastroschisis has changed. An attempt at immediate primary closure, long the standard practice, has now been largely supplanted with initial Corresponding author. Section of Pediatric Surgery, Riley Hospital for Children, Department of Surgery, Indiana University, Indianapolis, IN Tel.: ; fax: address: frescorl@iupui.edu (F.J. Rescorla). placement of a preformed, spring-loaded Silastic silo, followed by gradual reduction of the abdominal viscera and delayed closure of the abdominal wall defect. Studies examining the effectiveness of planned delayed primary closure have sighted several advantages including reduced ventilator times, fewer instances of abdominal compartment syndrome, less time to achievement of full enteral feeding, and improved convenience for surgeons and staff [1-6] /$ see front matter 2012 Elsevier Inc. All rights reserved. doi: /j.jpedsurg

2 120 B.R. Weil et al. Gastroschisis is a rare congenital abdominal wall defect occurring in an estimated 5 of 10,000 total births [7,8]. As such, studies aimed at comparing outcomes between 2 different treatment strategies are often unavoidably handicapped by low numbers, lack of randomization, use of historical controls, and/or their retrospective nature. This leads to inadequately powered studies and limit the ability to draw conclusions on the merits of either treatment strategy [8]. Herein, we chose to compare outcomes between not only preformed silo placement and primary closure but also between 2 periods one in which immediate primary closure was most commonly performed and one in which placement of a preformed silo was the most commonly used strategy. By providing a comparison of outcomes over time in addition to a comparison of closure techniques, it was our intent to provide a more complete view of how changes in practice have affected outcomes in the treatment of gastroschisis at a single institution. We hypothesized that our practice has shifted over time to placement of a preformed silo as the preferred initial approach and that this change would be associated with improvement in several clinical outcomes including reduced intensive care unit (ICU) stay, reduced ventilator days, and a reduction in the time to achievement of full enteral feeds. 1. Methods Following approval by the Institutional Review Board of Indiana University, records of patients undergoing gastroschisis closure at Riley Hospital for Children from 2000 to 2009 were reviewed. Patients were identified using an institutional search for all individuals assigned the diagnostic code for other congenital anomalies of the abdominal wall (International Classification of Diseases, Ninth Revision, Clinical Modification, code ). Billing records of the Division for Pediatric Surgery for this same diagnostic code were also searched and cross-checked with the institutional list to assure that a comprehensive list was generated. Both hard and electronic copies of records were searched, and only patients identified as having gastroschisis were included. Patients with omphaloceles were excluded. Basic demographic and historical data were collected. Because they have been shown to influence outcomes, data regarding the presence of associated intestinal complications (includes intestinal atresia(s), perinatal intestinal ischemia, and/or necrotizing enterocolitis) were also collected [9]. In the first comparison, patients were grouped according to whether they underwent an attempt at immediate primary closure or placement of a preformed silo and planned delayed primary closure as the initial treatment strategy. Patients were also grouped according to those receiving treatment from the years 2000 to 2002 (when early primary closure was the dominant technique) and those receiving treatment from 2003 to 2009 (when placement of a preformed silo was the predominant management strategy). Several outcomes were then assessed including development of a ventral hernia, development of a surgical site infection, length of hospital stay, length of neonatal ICU (NICU) stay, total ventilator days, time from birth to achievement of full enteral feeds, and mortality. Data were analyzed using GraphPad Prism Software (La Jolla, CA). Categorical data were compared using the χ 2 method. Continuous data were analyzed for normalcy via the D'Agostino (Pearson omnibus) method. Nonparametric were compared using the Mann-Whitney U test. Parametric data were compared using the Student t test, and P b.05 was considered significant. 204 patients diagnosed with gastroschisis 1 death prior to surgical treatment 203 patients undergoing surgical closure of gastroschisis 13 patients treated with placment of a hands-sewn silo 190 patients included in analysis by closure strategy 203 patients included in analysis by time period 43 patients treated with immediate primary closure 147 patients treated with preformed silo 49 patients treated from patients treated from Fig. 1 Flow chart representing the breakdown of patients in this study.

3 Changes in gastroschisis management Fig. 2 Placement of a preformed, spring-loaded Silastic silo by years from 2000 to Values for each year represent percentages of total gastroschisis cases treated in which a preformed silo was placed as initial management. White bars represent the years encompassed in the earlier period in which immediate primary closure was the dominant strategy, whereas gray bars represent the later period in which initial placement of a preformed silo was the most commonly used strategy. 2. Results A total of 204 patients treated for gastroschisis from 2000 to 2009 were identified (Fig. 1). Of these, 13 underwent placement of a hand-sewn silo for a perceived need to monitor a concurrent intra-abdominal condition or because the defect itself was not amenable to primary repair or placement of a preformed silo as judged by the treating surgeon. These patients were excluded from further analysis in the comparison of closure techniques but were included in the analysis of the 2 periods, so a fuller picture of the outcomes could be presented. In addition, 1 patient died before undergoing surgical evaluation and was excluded from both sets of analyses. This left a total of 190 patients for the closure technique analysis and 203 patients in the period 121 analysis. Survey of surgeons involved in the care of these patients as well as review of the records themselves revealed that placement of a preformed silo with planned delayed closure was used throughout the entire period analyzed but did not become the preferred strategy until approximately 2003 (Fig. 2). Based on these findings, outcomes between patients treated from 2000 to 2002 and from 2003 to 2009 were compared in addition to a comparison of primary silo placement with immediate primary closure. Analysis of the 2 treatment strategies revealed that Apgar score at 5 minutes was slightly less in the group that underwent placement of a preformed silo as initial management. Patient characteristics were otherwise similar (Table 1). A nonstatistically significant trend toward an increased frequency of intestinal complications was observed in the group treated with attempted primary closure vs the group treated with placement of a preformed silo (27.9% vs 15.6%; P =.07).Patients undergoing early primary closure developed ventral hernias at higher rates compared with those treated with a preformed silo (P =.0006;Table 2). Neonatal ICU stay was longer for patients receiving preformed silos (P =.002). Outcomes were otherwise similar between the 2 groups. When comparing the 2 periods, a higher proportion of vaginal births (as opposed to cesarean delivery) were observed intheperiodfrom2000to2002(79.6%vs53.2%;p =.001; Table 3). Patient characteristics, including frequency of intestinal complications, were otherwise similar between the 2 groups. Patients treated from 2000 to 2002 experienced shorter ICU stay and shorter time to achievement of full enteral nutrition (P b.05; Table 4). Patients treated from 2003 to 2009 experienced reduced ventilator days, fewer postoperative ventral hernias, and fewer wound infections (P b.05). A total of 12 deaths were observed. The 2 patient deaths in the primary closure group (4.7%) were each attributable to complications related to multiple intestinal atresias and intestinal necrosis. The 9 deaths in the silo group (6.1%) were attributable to multiple intestinal atresias and/or necrosis in 5 patients, necrotizing enterocolitis in 1 patient, neonatal respiratory distress syndrome in 1 patient, congenital cardiac Table 1 Patient characteristics in closure strategy comparison Primary closure (n = 43) Preformed silo (n = 147) P 95% CI OR Male 22 (51.2%) 78 (53.1%) Female 21 (48.8%) 69 (46.9%) Premature 23 (53.5%) 90 (61.2%) Gestational age (wk) Median, 36 (range, 31-40) Median, 36 (range, 26-39).14 Birth weight (kg) Median, 2.5 (range, ) Median, 2.4 (range, ).31 Apgar 1 Median, 7 (range, 2-9) Median, 7 (range, 0-9).66 Apgar 5 Median, 9 (range, 7-9) Median, 8 (range, 3-9).01 Maternal age (y) Median, 20 (range, 15-31) Median, 21 (range, 13-36).45 Vaginal birth 30 (69.8%) 84 (57.1%) Cesarean delivery 13 (30.2%) 63 (42.9%) Intestinal complications a 12 (27.9%) 23 (15.6%) Values represent means unless otherwise stated. CI indicates confidence interval; OR, odds ratio. a Includes perinatal ischemia, atresia, and necrotizing enterocolitis.

4 122 B.R. Weil et al. Table 2 Comparison of outcomes between closure strategies Primary closure (n = 43) Preformed silo (n = 147) P 95% CI OR Development of ventral hernia 18 (41.9%) 25 (17%) Wound infection 3 (7%) 25 (17%) Hospital stay (d) Median, 25 (range, ) Median, 34 (range, ).06 ICU stay (d) Median, 9 (range, 2-128) Median, 14 (range, 3-200).002 Total ventilator days Median, 4.5 (range, 0-128) Median, 3 (range, 0-100).06 Days from birth until full enteral nutrition Median, 29 (range, ) Median, 22 (range, ).07 goals achieved Mortality 2 (4.7%) 9 (6.1%) Values represent means unless otherwise stated. anomalies in 1 patient, and total parented nutrition associated liver disease in 1 patient. Subdivided by period, the 1 death occurring in the earlier period was attributable to multiple intestinal atresias and intestinal necrosis. Of the 11 mortalities observed in the later period, 6 were attributable to intestinal atresias and intestinal necrosis, 1 to necrotizing enterocolitis, 1 to congenital cardiac anomalies, 1 to total parented nutrition associated liver disease, and 2 to neonatal respiratory distress syndrome (1 child died before surgery). A slight trend toward increase mortality in the later period was observed (2% vs 7.1%; P =.19). 3. Discussion Goals in the treatment of infants with gastroschisis include correction of associated intra-abdominal pathologies and full-thickness closure of the abdominal wall while avoiding the physiologic consequences of intra-abdominal hypertension. The ideal management strategy accomplishes these goals as expeditiously and in as few steps as possible. This will serve to minimize the complications associated with multiple operations and prolonged NICU and hospital stays while allowing the infant to take in full caloric feeds as soon as possible. Placement of a preformed silo was initially described in the 1970s and gained popularity as a closure strategy in the 1990s [10,11]. Based on their experience with the planned placement of preformed, spring-loaded Silastic silos in the 1990s and early 2000s, Schlatter et al [6] espoused the benefits of this technique for the management of gastroschisis. Compared with a matched retrospective cohort of infants treated with immediate primary closure, the group cited improved closure rates, reduced ventilator days, and shorter time to full enteral feeding. Despite its reported benefits and the rapid adoption of the strategy of preformed silo placement by many groups, including our own, definitive evidence for its superiority over immediate primary closure is lacking and practice patterns continue to vary widely [12,13]. Studies comparing closure techniques have been mostly retrospective, have used historical cohorts for the primary closure group, and/or include relatively small numbers of patients. Although they Table 3 Patient characteristics in period comparison (n = 49) (n = 154) P 95% CI OR Male 23 (46.9%) 84 (54.5%) Female 26 (53.1%) 70 (45.5%) Premature 29 (59.2%) 95 (61.7%) Gestational age (wk) Median, 36 (range, 31-39) Median, 36 (range, 26-40).09 Birth weight (kg) Median, 2.6 (range, ) Median, 2.4 (range, ).42 Apgar 1 Median, 8 (range, 0-9) Median, 7 (range, 0-9).58 Apgar 5 Median, 8 (range, 4-9) Median, 8 (range, 3-9).29 Maternal age (y) Median, 20 (range, 15-36) Median, 21 (range, 13-34).23 Vaginal birth 39 (79.6%) 82 (53.2%).001 Cesarean delivery 10 (20.4%) 72 (46.8%) Intestinal complications a 6 (12.2%) 33 (21.4%).16 Method of closure Primary closure 24 (50%) 19 (12.3%) b.0001 Preformed Silo 17 (34.7%) 130 (84.4%) Hand-sewn silo 8 (16.3%) 5 (3.3%) Values represent means unless otherwise stated. a Includes perinatal ischemia, atresia, and necrotizing enterocolitis.

5 Changes in gastroschisis management 123 Table 4 Comparison of outcomes between closure periods (n = 49) (n = 154) P 95% CI OR Development of ventral hernia 18 (36.7%) 30 (19.5%) Wound infection 14 (28.6%) 11 (11.7%) Hospital stay (d) Median, 31 (range, ) Median, 35 (range, ).09 ICU stay (d) Median, 12 (range, 2-92) Median, 14 (range, 3-200).005 Total ventilator days Median, 6 (range, 0-22) Median, 3 (range, 0-128).02 Days from birth until full enteral nutrition Median, 25 (range, ) Median, 31 (range, ).04 goals achieved Mortality 1 (2%) 11 (7.1%) Values represent means unless otherwise stated. represent the preponderance of evidence available, these studies must be interpreted with caution. To date, only 1 prospective, randomized trial has been completed [1]. Although a trend toward reduced ventilator days was noted in this study, there were otherwise no differences in outcomes between the 2 closure strategies. The authors do appropriately suggest that the ability to turn an urgent operation into a more elective operation with the placement of a preformed silo is also a potential advantage of this strategy. In addition, at least 1 retrospective study suggests that immediate primary closure may be associated with an improved course compared with silo placement [14]. The present study supports mixed outcomes associated with the 2 strategies. Although not statistically significant, our retrospective comparison of the 2 treatment strategies did suggest that placement of a preformed silo may be associated with reduced ventilator days and a shorter time to full enteral nutrition as has been previously reported. On the other hand, placement of a preformed silo as primary therapy was associated with a prolonged NICU stay and a trend toward longer hospital stay. This study also suggests that development of a ventral hernia occurs at a significantly higher rate after attempt at primary repair as compared with placement of a preformed silo. It should be noted that during the period when primary closure was nearly always attempted immediately, closure of the skin only (without fascial closure) was occasionally performed when full thickness abdominal closure was not possible, leaving a ventral hernia requiring attention at a future operation. We chose to include these cases in our analysis because, from a practical standpoint, a second operation is needed for definitive repair, potentially exposing the patient to additional risk. Thus, our data indicate that a reduced chance for development of a ventral hernia requiring an additional operation may be another possible advantage to a strategy using preformed silo placement as primary therapy. The finding of a slight trend toward increased mortality in the later period was unexpected. However, given the relatively low mortality rate overall and the diverse causes of death, we were unable to attribute any increase in mortality to the change that occurred in our surgical management. Nevertheless, the influence of closure technique on mortality, particular in patients with complex gastroschisis and the presence of intestinal atresias, should be considered in future investigations. Limitations to the interpretation of these data are chiefly related to the retrospective nature and, in particular, to the issues of selection biases and confounding variables that may exist. As a specific example, Apgar scores were slightly lower among patients undergoing silo placement. Certainly, the lower median Apgar score could serve as a surrogate to suggest that patients undergoing silo placement were generally sicker and could be a confounding variable in terms of explaining these children's longer overall ICU stay. In an opposite sense, children in the primary closure group exhibited a higher rate of intestinal complications, many of which directly contributed to often significantly prolonged ICU stays. In this regard, it appears that the immediate primary closure group may have been the sicker cohort. In any case, we cannot fully account for the existence of these confounding variables, and so, attributing changes in outcomes solely to differences in closure technique should not be done based on the closure strategy comparison alone. The comparison of the earlier and later periods is important because it may lessen some of the problems of selection bias and, perhaps, provide a more accurate perspective of how change in practice patterns have influenced outcomes. Practice patterns clearly changed between the 2 periods, with placement of a preformed silo occurring more frequently in the later period. If a comparison of closure techniques alone was valid, it would stand to reason that outcomes of the earlier and later periods would mirror those of the strategy or immediate primary closure and placement of a preformed silo, respectively. This was, for the most part, what was observed. The comparison of periods revealed that outcomes in the earlier period were improved in terms of reduced ICU stay and quicker time until achievement of full enteral feeds. Conversely, outcomes in the later period were improved with regard to reduced ventilator days, reduced incidence of ventral hernias, and less wound infections. These results, with the exception of time to achievement of full enteral feeds which was reduced in the earlier period and yet also appear to be reduced in patients treated with preformed silo placement correlate

6 124 B.R. Weil et al. with the results from the comparison of closure techniques. Although we limited this analysis to the past decade to minimize the impact of overall improvement in the care of critically ill neonates on our outcomes, we cannot fully negate the impact that this may have had on the outcomes for the period analysis. An additional limitation of the present study is that the data in this series were not adequate to collect information on the size of the gastroschisis defect or the degree of visceral herniation. Subjectively, however, it did appear that an attempt at immediate primary closure, particularly in the period from 2003 to 2009, was often made when gastroschisis defects were small with only a limited amount of visceral herniation that could be easily reduced and accommodated within the abdomen. These patients, as logic might dictate, did generally quite well, requiring minimal ventilator times and short stays in the NICU, and were quickly advanced to goal enteral feeds. As such, the potential benefits of early primary closure that are implied in portions of this study may not be generalizable. Nevertheless, these data do suggest that immediate primary closure may remain a reasonable choice for the treatment of patients with smaller defects. In summary, ICU and hospital stay have slightly increased over time, whereas ventral hernia rates and ventilator days have decreased with time. These changes correlate with a change in practice from a predominance of immediate primary closure to the preference of placing a preformed silo. How changes in time to achievement of full enteral nutrition may relate to changes in practice patterns with regard to closure strategy is unclear in this study. We conclude that placement of a preformed silo is a reasonable option for the management of gastroschisis. Outcomes, when compared with primary closure, however, are mixed. Therefore, use of a strategy involving immediate primary closure should still be considered, particularly for smaller defects, and may, in these cases, hasten the time to NICU and hospital discharge. References [1] Pastor AC, Phillips JD, Fenton SJ, et al. Routine use of a SILASTIC spring-loaded silo for infants with gastroschisis: a multicenter randomized controlled trial. J Pediatr Surg 2008;43: [2] Banyard D, Ramones T, Phillips SE, et al. Method to our madness: an 18-year retrospective analysis on gastroschisis closure. J Pediatr Surg 2010;45: [3] Jensen AR, Waldhausen JH, Kim SS. The use of a spring-loaded silo for gastroschisis: impact on practice patterns and outcomes. Arch Surg 2009;144: [4] Wu Y, Vogel AM, Sailhamer EA, et al. Primary insertion of a Silastic spring-loaded silo for gastroschisis. Am Surg 2003;69: [5] Allotey J, Davenport M, Njere I, et al. Benefit of preformed silos in the management of gastroschisis. Pediatr Surg Int 2007;23: [6] Schlatter M, Norris K, Uitvlugt N, et al. Improved outcomes in the treatment of gastroschisis using a preformed silo and delayed repair approach. J Pediatr Surg 2003;38: [7] Kilby MD. The incidence of gastroschisis. BMJ 2006;332: [8] Holland AJ, Walker K, Badawi N. Gastroschisis: an update. Pediatr Surg Int 2010;26: [9] Synder CW, Biggio JR, Brinson P, et al. Effects of multidisciplinary prenatal care and delivery mode on gastroschisis outcomes. J Pediatr Surg 2011;46:86-9. [10] Fischer JD, Chun K, Moores DC, et al. Gastroschisis: a simple technique for staged silo closure. J Pediatr Surg 1995;30: [11] Shermeta DW, Haller JA. A new preformed transparent silo for the management of gastroschisis. J Pediatr Surg 1975;10: [12] Owen A, Marven S, Johnson P, et al. Gastroschisis: a national cohort study to describe contemporary surgical strategies and outcomes. J Pediatr Surg 2010;45: [13] Lansdale N, Hill R, Gull-Zamir S, et al. Staged reduction of gastroschisis using preformed silos: practicalities and problems. J Pediatr Surg 2009;44: [14] McNamara WF, Hartin CW, Escobar MA, et al. Outcome differences between gastroschisis repair methods. J Surg Res 2011;165:19-24.