Multidisciplinary Development of a Low-Cost Gastroschisis Silo for Use in Sub-Saharan Africa.

BACKGROUND: Gastroschisis silos are often unavailable in sub-Saharan Africa (SSA), contributing to high mortality. We describe a collaboration between engineers and surgeons in the United States and Uganda to develop a silo from locally available materials. METHODS: Design criteria included the following: < $5 cost, 5 ± 0.25 cm opening diameter, deformability of the opening construct, ≥ 500 mL volume, ≥ 30 N tensile strength, no statistical difference in the leakage rate between the low-cost silo and preformed silo, ease of manufacturing, and reusability. Pugh scoring matrices were used to assess designs. Materials considered included the following: urine collection bags, intravenous bags, or zipper storage bags for the silo and female condom rings or O-rings for the silo opening construct. Silos were assembled with clothing irons and sewn with thread. Colleagues in Uganda, Malawi, Tanzania, and Kenya investigated material cost and availability. RESULTS: Urine collection bags and female condom rings were chosen as the most accessible materials. Silos were estimated to cost < $1 in SSA. Silos yielded a diameter of 5.01 ± 0.11 cm and a volume of 675 ± 7 mL. The iron + sewn seal, sewn seal, and ironed seal on the silos yielded tensile strengths of 31.1 ± 5.3 N, 30.1 ± 2.9 N, and 14.7 ± 2.4 N, respectively, compared with the seal of the current standard-of-care silo of 41.8 ± 6.1 N. The low-cost silos had comparable leakage rates along the opening and along the seal with the spring-loaded preformed silo. The silos were easily constructed by biomedical engineering students within 15 min. All silos were able to be sterilized by submersion. CONCLUSIONS: A low-cost gastroschisis silo was constructed from materials locally available in SSA. Further in vivo and clinical studies are needed to determine if mortality can be improved with this design.

Major abdominal wall defects in the low- and middle-income setting: current status and priorities.

Major congenital abdominal wall defects (gastroschisis and omphalocele) may account for up to 21% of emergency neonatal interventions in low- and middle-income countries. In many low- and middle-income countries, the reported mortality of these malformations is 30-100%, while in high-income countries, mortality in infants with major abdominal wall reaches less than 5%. This review highlights the challenges faced in the management of newborns with major congenital abdominal wall defects in the resource-limited setting. Current high-income country best practice is assessed and opportunities for appropriate priority setting and collaborations to improve outcomes are discussed.

Neonatal surgery in low- vs. high-volume institutions: a KID inpatient database outcomes and cost study after repair of congenital diaphragmatic hernia, esophageal atresia, and gastroschisis.

BACKGROUND/PURPOSE: The volume-outcome relationship and optimal surgical volumes for repair of congenital anomalies in neonates is unknown. METHODS: A retrospective study of infants who underwent diaphragmatic hernia (CDH), gastroschisis (GS), and esophageal atresia/tracheoesophageal fistula (EA/TEF) repair at US hospitals using the Kids' Inpatient Database 2009-2012. Distribution of institutional volumes was calculated. Multi-level logistic/linear regressions were used to determine the association between volume and mortality, length of stay, and costs. RESULTS: Total surgical volumes were 1186 for CDH, 1280 for EA/TEF, and 3372 for GS. Median case volume per institution was three for CDH and EA/TEF, and four for GS. Hospitals with annual case volumes ≥ 75th percentile were considered high volume. Approximately, half of all surgeries were performed at low-volume hospitals. No clinically meaningful association between volume and outcomes was found for any procedure. Median cost was greater at high- vs. low-volume hospitals [CDH: $165,964 (p < 0.0001) vs. $104,107, EA/TEF: $85,791 vs. $67,487 (p < 0.006), GS: $83,156 vs. $72,710 (p < 0.0009)]. CONCLUSIONS: An association between volume and outcome was not identified in this study using robust outcome measures. The cost of care was higher in high-volume institutions compared to low-volume institutions. LEVEL OF EVIDENCE: III.

The economic burden of gastroschisis: costs of a birth defect.

BACKGROUND: Intestinal dysfunction and feeding intolerance are comorbidities associated with the abdominal wall birth defect of gastroschisis (GS). These factors contribute to prolonged hospitalization in this population of patients. The purpose of this study was to evaluate the economic burden on a state and national level. METHODS: From 2007-2011, the Healthcare Cost and Utilization Project database was queried for the following national and state of Texas data: number of discharges, length of stay (LOS), costs, and charges for all pediatric hospital stays ± CPT code 54.71 denoting GS repair for infants aged <1 y. The effect of GS on LOS, cost, and charges was calculated by the weighted average of the differences and is represented by the combined estimated difference (CED). RESULTS: Infants <1 y represent 74% of all pediatric discharges nationally and only 0.04% of these discharges are accounted for by GS patients. Nationally, GS patients had significantly longer LOS (CED 38.5 ± 0.9 d, P < 0.0001); increased costs (CED $79,733 ± $2119, P < 0.0001); and charges (CED $249,999 ± $9562, P < 0.0001). The Texas state data mirrored our findings for the national data. There was no significant difference in the LOS, costs, and charges between the national and state level. CONCLUSIONS: Our study shows that GS patients represent an extremely small minority of national and Texas pediatric discharges; however, these patients LOS and costs greatly exceed non-GS patients. Further investigation into factors influencing the development of intestinal dysfunction in these patients is needed to significantly impact the economic burden of the abdominal wall birth defect of GS.

Birth Weight, Compromised Bowel and Sepsis are the Main Variables Significantly Influencing Outcome in Gastroschisis.

BACKGROUND: The survival rate for gastroschisis has improved to more than 90% in the developed countries, but increased mortality, morbidity, consequent long hospitalisation and high costs are the rule in Romania. METHODS: Analytic retrospective study of all patients with gastroschisis treated at our department between 1990 and 2012. The study protocol included: demographic data, antenatal diagnosis, prematurity, mode of delivery, birth weight,associated anomalies, time to surgery, presence of compromised bowel, type of repair, post-operative complications, time to full enteral feeding, length of hospitalisation, mortality. RESULTS: 115 newborns with gastroschisis were treated during 23 years. Antenatal diagnosis was made only in 13 cases ata mean gestational age of 25 weeks. Delivery was vaginal in 80.8%. Associated malformations were present in 47 patients. Twenty-four patients had complex gastroschisis.Primary repair was done in 90 cases (79%) and in 24 patients a silo was used. Overall survival was only 29.8%, the main cause of death being severe sepsis with multiple organ failure(61.4%) and bronchopneumonia (52.6%). The rate of complications associated with closure, needing reintervention was 19.3%. CONCLUSIONS: Analysis of risk factors by logistic regression showed that low birth weight increased the risk of postoperative complications 17.4 times, sepsis increased the risk of complicated postoperative course 12.2 times, and the presence of compromised intestinal loops (complex gastroschisis) 5.5 times.

Variation in hospital costs for gastroschisis closure techniques.

BACKGROUND: In newborns with gastroschisis, both primary repair and delayed fascial closure with initial silo placement are considered safe with similar outcomes although cost differences have not been explored. METHODS: A retrospective review was performed of newborns admitted with gastroschisis at a single center from 2011 to 2016. Demographic, clinical, and cost data during the initial hospitalization were collected. Differences between procedure costs and clinical endpoints were analyzed using multivariable linear regression adjusting for prematurity, complicated gastroschisis, and performance of additional operations. RESULTS: 80 patients with gastroschisis met inclusion criteria. Rates of primary fascial, primary umbilical cord closure, and delayed closure were 14%, 65%, and 21%, respectively. Delayed closure was associated with an increase in total hospital costs by 57% compared to primary repair (p < 0.001). In addition, delayed closure was associated with increased total and NICU LOS (p < 0.05), parenteral nutrition duration (p = 0.02), ventilator days (p < 0.001), time to goal enteral feeds (p = 0.01), and all cost sub-categories except ward room costs (p < 0.01). CONCLUSION: Delayed fascial closure was associated with significantly greater hospital costs during the index admission.

Impact of Multidisciplinary Standardization of Care for Gastroschisis: Treatment, Outcomes, and Cost.

BACKGROUND/PURPOSE: Elimination of unnecessary practice variation through standardization creates opportunities for improved outcomes and cost-effectiveness. A quality improvement (QI) initiative at our institution used evidence and consensus to standardize management of gastroschisis (GS) from birth to discharge. METHODS: An interdisciplinary team utilized best practice evidence and expert opinion to standardize GS care. Following stakeholder engagement and education, care standardization was implemented in September 2014. A comparative cohort study was conducted on consecutive patients treated before (n=33) and after (n=24) standardization. Demographic, treatment, and outcome measures were collected from a prospective GS registry. Direct costs were estimated, and protocol compliance was audited. RESULTS: BW, GA, and bowel injury severity were comparable between groups. Key practice changes were: closure technique (pre-88% primary fascial, post-83% umbilical cord flap; p<0.001), closure location (pre-97% OR, post-67% NICU; p<0.001), and GA avoidance (pre-0%, post-48%; p<0.001). Median post-closure ventilation days were shorter (pre-4, post-1; p<0.001), and SSI rates trended lower (pre-21%, post-8%; p=0.3) in the post-implementation group with no differences in TPN days or LOS. No significant difference was seen in average per-patient costs: pre-$85,725 ($29,974-221,061), post-$76,329 ($14,205-176,856). CONCLUSION: Care standardization for GS enables practice transformation, cost-effective outcome improvement, and supports an organizational culture dedicated to continuous improvement. LEVEL OF EVIDENCE: III.

Impact of societal factors and health care delivery systems on gastroschisis outcomes.

Care of infants with gastroschisis is associated with a significant burden on health care delivery systems. Mortality rates in patients with gastroschisis have significantly improved over the past few decades. However, the condition is still associated with significant short-term and potentially long-term morbidity. Significant variations in clinical outcomes and resource utilization may be explained by several factors including provider and hospital experience, level of neonatal intensive care, variations in hospital regionalization of care, and differences in healthcare delivery systems. Reviewing and assessing these hospital and healthcare system related factors are paramount in addressing variations in gastroschisis care and improving outcomes for these vulnerable infants.

Perinatal outcomes and hospital costs in gastroschisis based on gestational age at delivery.

OBJECTIVE: To investigate the association between gestational age at delivery and perinatal outcomes among gastroschisis-affected pregnancies that result in live birth. METHODS: We conducted a retrospective cohort study using a linked maternal-infant database for more than 2.3 million liveborn neonates in Florida from 1998 to 2009. Cases were identified using a combination of International Classification of Diseases, 9th Edition, Clinical Modification, diagnosis and procedure codes indicative of gastroschisis. We restricted our analyses to singleton cases without another major birth defect or medical conditions that would justify early elective delivery. We categorized cases based on gestational age in weeks and compared perinatal outcomes. RESULTS: Among 1,005 neonates with gastroschisis, 324 (32.3%) were isolated, singleton cases without an additional indication for early delivery. We observed decreased rates of adverse pregnancy outcomes among those neonates delivered in the early term period (37-38 weeks of gestation) compared with preterm (less than 34 weeks of gestation); specifically, jaundice (18.5% compared with 42.3%, P=.01) and respiratory distress syndrome (5.9% compared with 23.1%, P≤.01). As the gestational age at birth increased, we observed fewer mean number of days spent in the hospital (less than 34 weeks of gestation: 55.9, P<.01; 34-36 weeks of gestation: 51.9, P=.02; 37-38 weeks of gestation: 36.9 [reference]) and lower direct inpatient medical costs (in thousands, U.S. dollars; less than 34 weeks of gestation: 79, P=.01; 34-36 weeks of gestation: 71, P=.04; 37-38 weeks of gestation: 51 [reference]) per infant in the first year of life. CONCLUSION: In pregnancies complicated by gastroschisis, and with no other known major indications, birth at early term or later term gestation, when compared with delivery before 37 weeks of gestation, is associated with improved perinatal outcomes and lower medical costs. LEVEL OF EVIDENCE: II.

Method to our madness: an 18-year retrospective analysis on gastroschisis closure.

BACKGROUND: The advent of preformed silos has facilitated routine bedside placement often without any attempt of intestinal reduction. It is unclear whether a strategy of routine silo (RS) placement with delayed fascial repair is beneficial over attempted primary repair (aPR) and silo placement only for those patients who cannot be reduced. We retrospectively compared clinical outcomes of neonates having aPR to those having RS placement to determine the impact of routine silo use and silo duration on gastroschisis care. METHODS: Neonatal records from patients with gastroschisis at a single children's hospital between 1990 and 2008 were reviewed. Demographic and outcome data were recorded and subjected to statistical analyses. Documentation of attempted intestinal reduction was used as a surrogate marker for aPR. The remaining patients were placed in the RS group. RESULTS: Two hundred forty-eight neonates with gastroschisis were identified. Thirteen were excluded for congenital or clinical issues which precluded aPR. Of the remaining 235 patients, neonates with RS had significantly more ventilator days (6.2 vs 4.4; P = .0011), more time of total parenteral nutrition (36.5 vs. 28.5; P = .0018), longer length of stay (LOS, 46.5 vs. 40.5; P = .0011), and greater hospital charges ($216,000 vs $172,000; P < .0001) than patients who had aPR. There was no significant difference observed in complications or survival. Linear regression modeling demonstrated that time to closure was significantly related to LOS as an independent variable. Each day to closure was associated with 2.2 extra days of hospitalization and approximately $9557 in hospital charges. CONCLUSION: Although limited by retrospective biases, this study demonstrates that time to closure is the most significant variable related to LOS in gastroschisis. This relationship is intuitive since longer time to closure is probably determined by the severity of gastroschisis. The method of closure, by primary repair or silo, is of secondary importance. Conversely, unnecessarily increasing the time to closure may increase the LOS. The speed of reduction, whether through primary repair or by silo, should be guided by physiologic principles.

Gastroschisis: small hole, big cost.

BACKGROUND/PURPOSE: This study was designed to assess the outcome and financial costs incurred for the treatment of gastroschisis. METHODS: A retrospective analysis was conducted of all patients with gastroschisis at a single institution over the past decade (n = 69). Hospital costs were determined and standardized to December 2001 dollars. RESULTS: Of the 69 patients, average gestational age at delivery was 35.9 weeks. Thirty-six patients had a primary fascial closure; 33 had a silo placed. The mean time to first feeding was 22 days and full feeding, 33 days. Average length of stay was 47 days. There were 3 deaths (2 shortly after birth, and one 131 days later owing to sepsis). The average cost of hospitalization and physician fees for patients with gastroschisis was $123,200. Using multivariate regression analysis, significant variables (P <.05) associated with cost of hospitalization were number of operative procedures, ventilatory days, male gender, and length of stay. Room expenses (43%), physician fees (15%), respiratory and pulmonary care (10%), and supply and devices (10%) made up the majority of costs. CONCLUSIONS: Cost of care associated with treatment for gastroschisis is high. Strategies designed to reduce cost must limit gastrointestinal, respiratory, and operative complications and reduce length of stay.