Journal of Pediatric Surgery 52 (2017) 1132–1134
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Improved outcomes for inborn babies with uncomplicated gastroschisis B.G. Dalton, K.W. Gonzalez, S.R. Reddy, R.J. Hendrickson, C.W. Iqbal ⁎ Children's Mercy Hospital Kansas City, MO
a r t i c l e
i n f o
Article history: Received 14 July 2016 Received in revised form 16 November 2016 Accepted 9 December 2016 Key words: Gastroschisis Uncomplicated Inborn Outborn
a b s t r a c t Introduction: Gastroschisis (GS) is a common abdominal wall defect necessitating neonatal surgery and intensive care. We hypothesized that inborn patients had improved outcomes compared to patients born at an outside hospital (outborn) and transferred for definitive treatment. Methods: A single center, retrospective chart review at a pediatric tertiary care center was performed from 2010 to 2015. All patients whose primary surgical treatment of GS was performed at this center were included. We compared patients delivered within our center (inborn) to patients delivered outside of our center and transferred for surgical care (outborn). Babies with complicated gastroschisis were excluded. Results: During the study period 79 patients with GS were identified. Of these, 53 were inborn and 26 were outborn. Sixteen patients were excluded for complicated GS. The rate of complicated GS was higher in the outborn group (32%) compared to the inborn population (11%) (p = 0.03). Duration of stay, readmission rate and time on TPN were all significantly decreased for inborn patients, while time to definitive closure was similar. Mortality was 0% for both inborn and outborn patients. Conclusion: Patients with uncomplicated GS seem to benefit from delivery with immediate pediatric surgical care available eliminating the need for transfer. Level of evidence: III © 2017 Elsevier Inc. All rights reserved.
Gastroschisis (GS) is a common abdominal wall defect effecting an estimated 1 to 5 in 10,000 live births worldwide [1–3]. More recently, the incidence is increasing [2]. GS is a surgical emergency that requires immediate protection of the bowel to prevent injury, hypothermia, and hypovolemia. This can be done by placement of a silo or attempted primary closure. Even after closure, patients can experience prolonged ileus, feeding difficulties, and necrotizing enterocolitis which can lengthen the duration of hospitalization and result in costs as high as $80,000 per initial hospital admission [4,5]. If injury has occurred to the bowel prenatally by volvulus or the presence of an atresia, the duration of hospitalization can be months long requiring extended periods of total parenteral nutrition (TPN). For these reasons, optimizing outcomes for patients with GS is imperative. Outcomes for GS babies have recently been linked to birth hospital in recent studies [3,6]. Given the wide variability in surgical outcomes expected for babies with complicated GS, we sought to compare outcomes for babies with uncomplicated GS that were inborn versus outborn. We hypothesized that inborn patients with uncomplicated GS would have improved outcomes compared to outborn patients that were subsequently transferred to our center after delivery.
⁎ Corresponding author at: Overland Park Pediatric Specialty Group, 10550 Quivira Rd, Overland Park, KS 66215. E-mail address:
[email protected] (C.W. Iqbal). http://dx.doi.org/10.1016/j.jpedsurg.2016.12.003 0022-3468/© 2017 Elsevier Inc. All rights reserved.
1. Methods After obtaining institutional review board approval, we identified all patients diagnosed with GS from January 2010 through January 2015 by diagnosis code. This timeframe was selected to coincide with the opening of delivery services at our center and capture all patients born within our center. Only patients who had all stages of the abdominal closure performed at our center were included. Patients with complicated GS were defined as those having an atresia or those babies requiring immediate intestinal resection owing to volvulus or perforation. Inborn patients were defined as those who were delivered within our center; outborn patients were defined as those patients delivered elsewhere who were subsequently transferred for pediatric surgical care. Outcomes were compared between the inborn and outborn group and included time to complete abdominal closure, total duration of stay, duration of TPN, time of mechanical ventilation, complication rates, readmission rates, and mortality. Complications were defined as deviation from normal postoperative course attributable to either the disease process or interventions. Time to abdominal closure was defined as the time from birth to definitive closure. All continuous variables are reported as the mean ± standard deviation unless otherwise indicated. Comparative analysis was performed using student t test for continuous variables and Fisher’s exact for binary variables. A chi squared goodness of fit test was performed to ensure normal distribution of data. Significance was defined as a P ≤ 0.05.
B.G. Dalton et al. / Journal of Pediatric Surgery 52 (2017) 1132–1134
2. Results
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Table 2 Uncomplicated gastroschisis inborn vs outborn.
We identified 79 patients treated at our center for GS. Of these, 53 were inborn and 26 were outborn. We excluded 16 patients who met criteria for complicated GS resulting in 45 inborn patients and 18 outborn patients with uncomplicated GS that were included for study. Interestingly, the rate of complicated GS was higher in the outborn group (32%) compared to the inborn population (11%) (p = 0.03). Table 1 shows demographic data for patients with uncomplicated GS. Nearly half of all babies with GS were delivered by Cesarean delivery however there was no difference in the rate of Cesarean delivery between the two groups (p = 1.0). The two groups were similar in terms of sex, gestational age, and weight. All inborn babies had a prenatal diagnosis—which would have been required for delivery in the center. Two babies in the outborn group had not been diagnosed prenatally, however the other 14 were known to have gastroschisis. Fifteen of 45 (33%) patients in the inborn group underwent successful primary closure on the first day of life compared to 9 of 18 (50%) patients in the outborn group (p = .26). The remaining 9 outborn patients were all successfully closed primarily after initial silo placement. Primary fascial closure after initial silo placement occurred in 23 of 45 patients in the inborn group. There were 4 patients in the inborn group that were closed with bioprosthetic mesh. Two of these patients were noted to have tight defects at the time of initial evaluation, necessitating opening of these defects. Three patients underwent bedside sutureless closure. The duration of hospitalization was shorter in inborn patients by a mean of 10 days (30 ± 15 versus 40 ± 26 days, p = 0.03) as was the duration of TPN (23 ± 13 versus 32 ± 23 days, p = 0.04). Babies that were outborn also had a higher readmission rate (11 versus 22%, p = 0.02). There was no difference in time to definitive abdominal closure, ventilator days, or time to feeds (Table 2). Complications are outlined in Table 3. The overall complication rate was 17.4% and was higher in the outborn group (38.9 versus 8.8%, p = 0.02). Three of the patients in the outborn group developed medically managed necrotizing enterocolitis (NEC). Two other patients in the outborn group also experienced complications including tracheitis requiring steroids and evisceration requiring operative closure. There were no episodes of NEC in the inborn group, however 3 patients experienced a complication including wound breakdown in 2 patients that resolved with local wound care and gastric perforation in 1 patient requiring reoperation. Two patients in the inborn group experienced the late complication of small bowel obstruction requiring laparotomy with small bowel resection 2 months and 1.5 years after initial discharge, respectively. Two patients in the outborn group required reoperation for small bowel obstruction secondary to midgut volvulus. Both patients underwent laparotomy with lysis of adhesions and Ladd's procedures without small bowel resection. These operations took place 4 and 6 months, respectively, after initial discharge. There were no deaths in this series. 3. Discussion Several previous studies have examined the influence of birthplace on outcomes for children with GS [3,7–11]. These studies have yielded variable results on the efficacy of patients with GS being born at the treating facility. However, the 2 largest studies have shown a benefit
Value
Inborn (n = 45)
Outborn (n = 18)
P
Length of stay (d) Hours to closurea Ventilator days Days to first feed Days to full feed TPN days 90d readmission % Total hospital days
30 ± 15 73 ± 65 7.6 ± 8.5 13 ± 8.5 16 ± 54 23 ± 12 11 31 ± 15
40 ± 26 79 ± 88 7.7 ± 4.1 15 ± 8.4 36 ± 23 32 ± 23 22 43 ± 27
0.03 0.39 0.30 0.17 0.07 0.03 0.02 0.01
a All patients included, patients with sutureless closure considered closed at time closure dressing placed.
in patients with inborn status in shorter length of stay, time to full enteral feeds, time to primary closure, and complication rate. A recent multicenter retrospective study including more than 500 patients with GS showed significant advantages of inborn patients in shorter length of stay (median 34 days vs 41.7 days). In the same study time from birth to definitive closure (median 3.7 vs 5.4 days) and time to full enteral feeds (median 27 vs 31 days) were both significantly shorter in favor of inborn patients [3]. A database study of prenatally diagnosed GS with almost 400 patients over a 3 year period showed that location of delivery was an independent predictor (odds ratio 1.6) of overall complication rate with inborn patients experiencing a lower rate. The complications examined included abdominal compartment syndrome, bowel obstruction, line sepsis, wound dehiscence, and wound infection [7]. The present study is mostly in agreement with these conclusions revealing a shorter length of stay, lower readmission rate, fewer total hospital days and a lower complication rate for the inborn population. We did not see a difference in time to feeds, however our data trended towards a more favorable time to feeds in the inborn group. This conclusion is supported by the significant difference in time on TPN. There is a concern for non-normality of data in our study contributing to the findings. However, employing a chi squared goodness of fit for both length of stay and TPN days found only 1 patient in the outborn group and 2 patients in the outborn group outside the limit of normal distribution (N2 standard deviations from mean). These differences in our study cannot be attributed to earlier closure as inborn and outborn babies had a similar time to closure. Moreover, the methods of closure did not vary between the groups either. Other factors such as peripartum hemodynamic stability and differing institutional perinatal resuscitation strategies could contribute to this difference but were unable to be analyzed in this series. While our management is protocolized, there is too much variability in how the outborn babies were managed prior to transfer. Nonetheless, this suggestion underscores the critical role that neonatal resuscitation and expedient surgical care likely play in the outcomes for babies with uncomplicated GS. The rate of NEC was higher in the outborn group. In fact, there were no episodes of NEC in the inborn group. Given the role that intestinal flora play in the development of NEC, this finding raises the question of whether or not outborn babies with GS are placed at higher risk for NEC owing to exposure to different types of microorganisms [12–15]. We did see a higher rate of complicated GS in the outborn group. While we excluded these patients in our comparative analysis for outcomes, this finding warrants discussion. There are facilities in our region Table 3 Complications of inborn and outborn patients with gastroschisis.
Table 1 Demographic data uncomplicated inborn vs outborn patients. Value
Inborn (n = 45)
Outborn (n = 18)
P
% male Birth weight (kg) Gestational age (wk) Vaginal delivery (%) Prenatal Diagnosis (%)
67 2.6 ± 0.5 36.4 ± 1.4 56 100
64 2.6 ± 0.4 36 ± 1.5 56 89
1.00 1.00 0.13 1.00 0.08
Complication
Inborn (n = 45)
Outborn (n = 18)
P
Overall (%) Wound dehiscence (%) Gastric perforation (%) Tracheitis (%) Evisceration (%) Reoperation for SBO (%)
8.8 4.4 2.2 0 0 4.4
38.9 0 0 5.5 5.5 11.1
0.02 0.5 0.7 0.29 0.29 0.32
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that also manage GS, however it is likely that a lower threshold exists to transfer babies with complicated GS to our tertiary referral center. While our findings seem to suggest that babies with uncomplicated GS benefit from delivery in the same center as the GS will be definitively managed surgically, there are limitations to the study. First, this is a retrospective study and is subject to bias, although most patients were managed under strict protocols owing to a concurrent prospective randomized trial being conducted at our center comparing silo versus primary closure for GS. Second, prenatal data on the outborn patients are limited. It is possible that outborn patients were born owing to precipitous onset of labor that prevented evaluation and delivery at a center with neonatal and pediatric surgical services. In which case, those babies may have already experienced distress that contributed to the difference in outcomes. Additionally, there may have been bias in which patients were transferred and which patients were managed at centers with pediatric surgical coverage. Given that most babies with GS are diagnosed prenatally, we recommend delivery in a center capable of providing complete care for the mother but that also has neonatal and pediatric surgical services readily available for the baby. References [1] Gamba P, Midrio P. Abdominal wall defects: prenatal diagnosis, newborn management, and long-term outcomes. Semin Pediatr Surg 2014;23(5):283–90. http://dx. doi.org/10.1053/j.sempedsurg.2014.09.009 [Epub 2014 Sep 4. Review].
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