The 'gentle touch' technique in the treatment of gastroschisis

Purpose

The aim of this study was to assess the effectiveness of gravity reduction of the intestines in newborns with gastroschisis.

Methods

Throughout the calendar year 2001, all newborns with gastroschisis were prospectively treated by means of an established clinical pathway protocol. Silon insertion and all subsequent therapies were performed in the neonatal intensive care unit (NICU) No physical compression of the bowel nor stretching of the abdominal wall was done, and bowel reduction was entirely passive, accomplished by the force of gravity. Demographic data along with time to abdominal wall closure, ileus resolution, and cessation of total parenteral nutrition (TPN) all were recorded.

Results

Sixteen babies were treated using the established pathway without prenatal selection or postnatal rejection. There were 10 boys and 6 girls. The mean maternal age was 18.5 years. Seventy-five percent of the mothers were Hispanics. Mean time to Silon removal was 5.5 days, initiation of enteric feeding was 16 days and TPN cessation 23 days. There were no significant complications in this group nor was there any mortality.

Conclusions

Gravity reduction of the intestines in babies with gastroschisis is most gentle and effective. Blood flow compromise, mechanical injuries of the exposed bowel, or abdominal compartment syndrome thus were averted. The outcome analysis favorably compares this method with reported series.     

Impact of maternal substance abuse and smoking on children with gastroschisis

Background/Purpose

Conflicting information exists regarding the effects of maternal substance abuse on gastroschisis. The objectives of this study are to determine if maternal smoking is associated with an increased risk of gastroschisis and whether substance abuse is associated with the severity of gastroschisis.

Methods

The Canadian Pediatric Surgery Network (CAPSNET) database was evaluated for associations between maternal substance abuse and the severity of the gastroschisis. We also compared smoking rates from this group to overall Canadian maternal smoking rates.

Results

One hundred fourteen cases of gastroschisis acquired over 18 months were evaluated. After adjusting for covariates, illicit drug use was associated with bowel necrosis (OR, 9.4; 95% CI,1.3-70) and marijuana use with matting of the intestines (OR, 4.0; 95% CI, 1.0-16). Functional outcomes assessment revealed that slower initiation of enteral feeds was associated with maternal smoking (OR, 3.8; 95% CI, 1.4-10). The overall maternal smoking rate in this cohort (30.7%) was significantly higher than the known Canadian rate (13.4%). This may be accounted for by the considerably higher smoking rate of mothers 20 to 24 years of age in our cohort (48.9%).

Conclusions

Substance abuse and smoking are associated with a greater severity of gastroschisis in terms of both the degree of intestinal injury and functional outcomes. High smoking rates among young mothers may be putting children with gastroschisis at risk for poor outcomes.     

Effects of intrauterine treatment on interstitial cells of Cajal in gastroschisis

Aim

An experimental study was performed to investigate the effects of amnio-allantoic fluid exchange and intrauterine bicarbonate treatment on intestinal damage and interstitial cells of Cajal (ICC) in gastroschisis.

Materials and Methods

Thirteen-day-old fertilized chick eggs were randomly allocated into 4 groups as control, gastroschisis, gastroschisis + amnio-allantoic fluid exchange, and gastroschisis + bicarbonate treatment groups. In the treatment groups, amnio-allantoic exchange and bicarbonate treatments were performed for 3 days, after creating gastroschisis. Specimens were processed for hematoxylin-eosin and c-kit immunohistochemistry on the 18th day of incubation, after macroscopic examination. The intestines were evaluated with light microscopy for the presence of mucosal congestion and muscular and serosal edema. Mean muscular thickness and density of ICC were measured.

Results

Mean muscular thickness significantly increased in the gastroschisis group when compared with control and treatment groups. Labeling intensity, morphology, and localization of the ICC were similar in all groups. Mean ICC density significantly decreased in the gastroschisis group when compared with the control group (P < .01), and it significantly increased after amnio-allantoic fluid exchange treatment (P < .01).

Conclusions

The decrease in ICC density encountered in damaged intestinal loops in gastroschisis was prevented with intrauterine treatment. The beneficial effects of amniotic exchange on intestinal motility may depend on both prevention of intestinal damage and preservation of ICC density and function. The density of ICC might be a reliable numeric parameter both to predict intestinal motility disorders in gastroschisis and to compare the effectiveness of intrauterine treatment methods.     

Preservation of extracorporeal tissue in closing gastroschisis augments intestinal length

Background

Prenatal closure of the umbilical ring in gastroschisis may result in an amorphous, nonviable appearing extracorporeal tissue that is resected during the repair. However, it is unclear whether such remnant intestine is truly nonviable.

Methods and Results

We examined the outcomes of patients when this tissue is preserved. We identified 8 patients who presented with a closing gastroschisis and a mass of tissue connected by a vascular pedicle. Four patients underwent abdominal exploration with resection of the mass and gastroschisis closure. Histologic examination revealed normal intestinal wall architecture. All patients in this group developed short bowel syndrome, requiring long-term parenteral nutrition. Conversely, 4 patients underwent abdominal exploration with internalization of the remnant tissue, a maneuver referred to as “parking,” along with either silo placement, or primary closure of the gastroschisis. At re-exploration, 3 of 4 patients were found to have viable intestine, and bowel continuity was reestablished. The mean parenteral nutrition requirement for this group was significantly shorter than the resected group.

Conclusion

In this series, we show that this amorphous tissue, when preserved, may exhibit normal intestinal architecture and absorptive function. Therefore, such remnant tissue should be preserved as it may significantly increase bowel length and minimize parenteral nutrition requirement.     

Familial recurrence of gastroschisis: Literature review and data from the population-based birth registry “Mainz Model”

Background

Familial forms of gastroschisis are considered rare. A search for these forms in a population-based birth registry in 1993 found a recurrence risk of 3.5% among first-degree relatives. Since then, similar investigations in population-based registries have led to contradictory results.

Methods

A search of the population-based birth registry “Mainz Model” for familial cases of gastroschisis and a systematic review of the literature were performed.

Results

The Mainz Model database yielded 1 familial recurrence out of 27 gastroschisis cases. From the literature, 37 affected families could be retrieved. Among 412 gastroschises from population-based registries, 10 familial recurrences have been found. These translate into a recurrence risk of 2.4%, with a strong tendency toward underestimation.

Conclusion

The existing data support the hypothesis that familial recurrence of gastroschisis is much more likely than previously thought.     

Early elective cesarean delivery before 36 weeks vs late spontaneous delivery in infants with gastroschisis

Purpose

The aim of this study is to assess the value of early elective cesarean delivery for patients with gastroschisis in comparison with late spontaneous delivery.

Methods

Analysis of infants with gastroschisis admitted between 1986 and 2006 at a tertiary care center was performed. The findings were analyzed statistically.

Results

Eighty-six patients were involved in the study. This included 15 patients who underwent emergency cesarean delivery (EM CD group) because of fetal distress and/or bowel ischemia. The remaining 71 patients born electively were stratified into 4 groups. The early elective cesarean delivery (ECD) group included 23 patients born by ECD before 36 weeks; late vaginal delivery (LVD) group included 23 patients who had LVD after 36 weeks; 24 patients had LCD after 36 weeks because of delayed diagnosis that resulted in late referral; and 1 patient had early spontaneous vaginal delivery (EVD group) before 36 weeks. The mean time to start oral feeding, incidence of complications, and primary closure were significantly better in the ECD group than in the LVD group. The duration of ventilation and the length of stay were shorter in ECD group, but the difference was not statistically significant.

Conclusion

Elective cesarean delivery before 36 weeks allows earlier enteral feeding and is associated with less complications and higher incidence of primary closure (statistically significant).     

Gastroschisis: a national cohort study to describe contemporary surgical strategies and outcomes

Background

Information on adoption of newer surgical strategies for gastroschisis and their outcomes is largely limited to hospital-based studies. The aim of this study was to use a new UK national surveillance system to identify cases and thus to describe the contemporary surgical management and outcomes of gastroschisis.

Methods

We conducted a national cohort study using the British Association of Paediatric Surgeons Congenital Anomalies Surveillance System to identify cases between October 2006 and March 2008.

Results

All 28 surgical units in the United Kingdom and Ireland participated (100%). Data were received for 95% of notified cases of gastroschisis (n = 393). Three hundred thirty-six infants (85.5%) had simple gastroschisis; 45 infants (11.5%) had complex gastroschisis. For 12 infants (3.0%), the type of gastroschisis could not be categorized. Operative primary closure (n = 170, or 51%) and staged closure after a preformed silo (n = 120, or 36%) were the most commonly used intended techniques for simple gastroschisis. Outcomes for infants with complex gastroschisis were significantly poorer than for simple cases, although all deaths occurred in the simple group.

Conclusions

This study provides a comprehensive picture of current UK practice in the surgical management of gastroschisis. Further follow-up data will help to elucidate additional prognostic factors and guide future research.     

Routine use of a SILASTIC spring-loaded silo for infants with gastroschisis: a multicenter randomized controlled trial

Background

Retrospective studies have suggested that routine use of a preformed silo for infants with gastroschisis may be associated with improved outcomes. We performed a prospective multicenter randomized controlled trial to test this hypothesis.

Methods

Eligible infants were randomized to (1) routine bedside placement of a preformed Silastic spring-loaded silo, with gradual reduction and elective abdominal wall closure, or (2) primary closure.

Results

There were 27 infants in each group. There was no significant difference between groups with respect to age, weight, sex, Apgar scores, prenatal diagnosis, or mode of delivery. The total number of days on the ventilator was lower in the spring-loaded silo group, although it did not reach statistical significance (3.2 vs 5.3, P = .07). There was no significant difference between groups with respect to length of time on total parenteral nutrition, length of stay, or incidence of sepsis and necrotizing enterocolitis.

Conclusion

Routine use of a preformed silo was associated with similar outcomes to primary closure for infants with gastroschisis but with a strong trend toward fewer days on the ventilator. Use of a preformed silo has the advantage of permitting definitive abdominal wall closure in a more elective setting.     

Development and validation of a risk stratification index to predict death in gastroschisis

Background

Gastroschisis is a rare congenital anomaly, the improved surgical management of which has contributed to a survival rate greater than 90%. Development of an accurate risk stratification system to help identify the subset of patients at greatest risk for death may lead to further improvements in outcome.

Methods

Infants with gastroschisis were identified from 16 years of the National Inpatient Sample database and the Kids' Inpatient Database using the International Classification of Diseases, Ninth Revision, Clinical Modification procedure code 54.71 (repair of gastroschisis) and an age of less than 8 days. Logistic regression analysis determined which coexisting diagnoses were significantly associated with death. Odds ratios from the logistic regression model were simplified and used as weighting factors to create an additive index. The index was validated using the 2003 Kids' Inpatient Database data set.

Results

Intestinal atresia, necrotizing enterocolitis, rare cardiac anomalies, and lung hypoplasia were strongly associated with death and used to create a scoring system with a potential range of 0 to 10. Every point increase on the scale of gastroschisis risk stratification index is associated with a 95% relative increase in the likelihood of death.

Conclusion

We have developed a novel index, which is superior to previous classification systems in identifying patients with gastroschisis who are at highest risk for death.     

Impact of corticosteroid on intestinal injury in a gastroschisis rat model: morphometric analysis

Background/Purpose

The goal in the treatment of gastroschisis is to prevent intestinal injury. Corticosteroids are known by their effects at the inflammatory response and by the improvement on the intestinal maturity. The authors evaluated the effects of maternal corticosteroid administration on the intestines of rats that underwent fetal gastroschisis.

Methods

A Correia-Pinto-modified gastroschisis rat model was used. Two groups were assessed: the control group (group 1) and the dexamethasone group (group 2). Each group was composed of control and sham fetuses, and fetuses with gastroschisis. Fetal body weight, intestinal weight, intestinal length, and protein were assessed. Histologic analysis involved measures of intestinal loop diameter, total intestinal wall, mucosa and submucosa, both circular and longitudinal muscle layers, and serosal thicknesses. Differences between groups and subgroups were tested by the analysis of variance method with a significant P value less than .05.

Results

Dexamethasone decreased in all the morphometric data except in the intestinal length. Dexamethasone increased the intestinal protein content in fetuses with gastroschisis, and control and sham fetuses. In both groups, all histologic parameters were increased in fetuses with gastroschisis (P < .0001).

Conclusions

Dexamethasone caused a substantial decrease in intestinal weight in GFs, increased the intestinal protein content, and it may be useful in decreasing the intestinal damage of gastroschisis.     

A multicenter, randomized, double-blind, placebo-controlled trial of the prokinetic agent erythromycin in the postoperative recovery of infants with gastroschisis

Background/purpose

The recovery of gut function after repair of gastroschisis is frequently prolonged, and these infants are prone to complications associated with parenteral nutrition. This trial was designed to investigate the effect of the prokinetic agent, erythromycin, on the attainment of full enteral feeding in infants after primary repair of uncomplicated gastroschisis.

Methods

A multicenter, randomized, double-blind, placebo-controlled trial was used to investigate the effect of enteral erythromycin (3 mg/kg/dose 4 times daily) compared with placebo on the attainment of full enteral feeding tolerance after primary repair of uncomplicated gastroschisis. Eleven neonatal surgical units in the United Kingdom participated in the study. The primary end-point was the time taken to achieve continuous enteral feeding at 150 mL/kg/24 hours sustained for 48 hours.

Results

Of 70 eligible infants, 62 were recruited and randomly divided. There were 30 patients in group I (placebo) and 32 in group II (erythromycin). The groups were comparable in terms of mean gestational age, mean birth weight, extent of evisceration, and degree of intestinal peel. There was no statistically significant difference between the 2 groups in the time taken to achieve full enteral feeding (27.2 v 28.7 days; P = .75). Similarly, no significant differences were found in the incidence of catheter-related sepsis, duration of parenteral nutrition, or time to discharge between the 2 groups.

Conclusions

Enterally administered erythromycin at a dose of 3 mg/kg 4 times daily conferred no advantage in the time taken to achieve full enteral feeding after primary repair of uncomplicated gastroschisis.     

The effect of gastroschisis on experimental diaphragmatic hernia in fetal rabbits

Purpose

In this study, the authors analyzed the effect of experimentally induced gastroschisis on pulmonary hypoplasia in fetal rabbits with congenital diaphragmatic hernia (CDH).

Methods

Twenty-three pregnant rabbits underwent fetal surgery on gestational day 24 through 27. A left diaphragmatic hernia was created in 1 fetus (DH group) from each rabbit, and a left diaphragmatic hernia with gastroschisis was created in another fetus (GS group). The fetuses were delivered on gestational day 27 through 33. Histologic and morphometric examination of the lungs in each group was done.

Results

In the DH group, the lungs were hypoplastic with a decrease in lung weight to body weight ratio and an increase pulmonary arterial medial wall thickness. The alveolar septae were markedly thickened with increased interstitial tissue and diminished alveolar air spaces. In the GS group, the alveolar septae were thickened but narrower than those of DH group, and air spaces were increased. The pulmonary arterial wall was markedly thickened in the DH group but only slightly thickened in the GS group.

Conclusions

Pulmonary hypoplasia seen in newborn rabbits after experimentally induced diaphragmatic hernia is less severe in those rabbits with both gastroschisis and DH.     

Intestinal transplantation for short bowel syndrome secondary to gastroschisis

Background/Purpose

Gastroschisis is the most frequent cause of pediatric intestinal transplantation. This study reviews our experience of intestinal transplantation secondary to gastroschisis to elucidate those factors affecting the outcome of children with short bowel syndrome.

Methods

A retrospective review was performed for children who underwent intestinal transplantation for gastroschisis at the University of Miami between June 2003 and August 1994.

Results

Thirty-two transplants were performed in 28 children with gastroschisis during the study period. Associated intestinal anomalies were present in 22 infants (atresia [n = 14], volvulus [n = 3], and/or ischemia [n = 16]). Spontaneous prenatal closure of gastroschisis, a rare anomaly associated with bowel atresia and ischemia because of a very small abdominal defect, was seen in 9 patients. Most of the patients had a complicated course and required multiple abdominal surgeries before transplant. Fifteen (53.6%) patients are currently alive at a median follow-up of 23.5 months. Short-term survival rate has significantly improved in recent years.

Conclusions

Patients with complex gastroschisis and intestinal anomalies have a significant risk for progression to short bowel syndrome. Intestinal transplantation can be a lifesaving option and provides a satisfactory outcome for children with short bowel syndrome secondary to gastroschisis.     

Evaluation of a bioabsorable polylactide film in a large animal model for the reduction of retrosternal adhesions

Objectives

An adult pig model of retrosternal adhesion formation via an inferior hemisternotomy was used to evaluate the formation and development of pericardial and retrosternal adhesions, as well as adhesion reduction using two thicknesses of a bioabsorbable polylactide film.

Materials and methods

Twenty-five adult female pigs (70 kg) were allocated to either a control group or four different treatments using two thicknesses (0.02 or 0.05 mm) of a polylactide film. In each animal, the film was placed either inside the pericardium or inside and outside the pericardium.

Results

All animals demonstrated adhesions between the posterior and lateral surfaces of the heart and pericardium. Thick fibrous retrosternal adhesions and pericardial adhesions were noted in the control animals with complete obliteration of the anatomical plane. The polylactide films preserved the anatomical planes and reduced the adhesion response.

Conclusions

A reproducible animal model was used to examine the formation and reduction of retrosternal and pericardial adhesions. A polylactide film placed inside the pericardium or between the heart and sternum was able to limit adhesion formation and maintain the anatomical planes, which would facilitate reentry.     

Noise exposure and convertible cars

Objective

To investigate whether drivers of convertible cars are exposed to excessive noise levels.

Study Design

Prospective.

Setting

Data were collected along a main United Kingdom highway.

Subjects and Methods

Seven cars were included within the study, encompassing a range in cost, power, and comfort. A calibrated, integrating noise meter was used to measure average noise levels (Leq dB) and peak levels (Lmax dB) encountered in the region of the driver's roadside ear with the convertible roof lowered. Readings were recorded at speeds of 50, 60, and 70 mph with the windows lowered and also at 70 mph with the windows raised.

Results

Noise levels for all testing conditions had a range for Leq of 82 dB to 92 dB, whereas the maximum Lmax level noted for articulated lorries was 99 dB.

Conclusion

A minimal trend toward increasing noise levels with speed was noted for the speeds tested. A statistically significant difference in noise reduction was found by raising the car windows. Although average levels were noted to be above the 85 dB criterion level legislated by some organizations, the length and frequency of most car journeys with the convertible roof lowered is unlikely to significantly increase the noise exposure risk of most individuals. Future studies may be able to evaluate whether a temporary threshold shift phenomenon may occur.     

Urinary trypsin inhibitor present in fetal urine prevents intraamniotic meconium-induced intestinal damage in gastroschisis

Background/Purpose

Contact with amniotic fluid causes intestinal damage in gastroschisis, and intraamniotic meconium has been shown to be responsible. Meconium has been shown to contain a significant amount of IL-8, which may be the responsible cytokine for harmful effects of meconium.Neonatal urine contains high amount of urinary trypsin inhibitor (UTI) compared with adult human urine. Urinary trypsin inhibitor has been shown to exert inhibitory effects on IL-8. Therefore, far from being destructive, presence of fetal urine in the amniotic fluid might be beneficial because human urine contains UTI.An experimental study has been performed to investigate whether presence of intraamniotic human urine (consequently UTI) besides meconium is beneficial on intestines of chick embryo with gastroschisis.

Methods

Five-day-old fertilized chick eggs were used. Gastroschisis was created through amniotic cavity without opening the allantoic cavity. Sterile urine and meconium were obtained from newborn humans. Study was conducted in 2 stages. In the first stage, gastroschisis was created, and meconium suspensions at minimal harmful meconium concentration were prepared using natural and denatured human neonatal urine and instilled into the amniotic cavity. In the second stage of study, various concentrations of UTI plus meconium suspension at minimal harmful meconium concentration was instilled into the amniotic cavity.

Results

Serosal thickening, inflammation, and focal fibrin deposits were observed in intestines of the groups with meconium and meconium in denatured urine. Histopathologic features of intestines of the group with meconium in natural urine did not differ from the intestines of the control group.Histopathologic examination of intestines of groups with meconium and meconium plus 50 U/mL UTI showed serosal thickening, inflammation, focal fibrin, and collagen deposits. Histopathologic features of intestines of the groups with 1:400 intraamniotic meconium plus 100 and 200 U/mL UTI did not differ from the intestines of control group.

Conclusion

Urinary trypsin inhibitor 100 U/mL prevented the intestinal damage via inhibiting IL-8, which is contained by 1:400 concentration of meconium. Therefore, besides the existence of threshold level of meconium, the existence of UTI, which is capable of inhibiting IL-8 contained by threshold level of meconium, may be a factor in the occurrence of intestinal damage in gastroschisis.     

Intravenous landiolol, a novel β(1)-adrenergic blocker, reduces the minimum alveolar concentration of sevoflurane in women

Study Objective

To investigate the effect of intravenous (IV) landiolol, a novel β₁-adrenergic blocker, on the minimum alveolar concentration (MAC) of sevoflurane in adult women.

Design

Prospective, randomized study.

Setting

University hospital.

Patients

42 ASA physical status 1 and 2 women, aged 24-57 years, who were scheduled to undergo elective abdominal surgery.

Interventions

Anesthesia was induced in all patients by vital capacity rapid inhalation induction of sevoflurane. In the landiolol group, administration of landiolol began when patients took a vital-capacity breath: 0.125 mg/kg/min for one minute and then 0.04 mg/kg/min. Normal saline was administered in the control group.

Measurements

MAC was determined by a technique adapted from the conventional up-down method.

Main Results

The MAC of sevoflurane was 2.2% ± 0.2% in the control group and 1.7% ± 0.2% in the landiolol group, a statistically significant difference (P = 0.0005).

Conclusions

IV landiolol reduces the MAC of sevoflurane in women by approximately 20%.     

Insertion/deletion polymorphism of the angiotensin-converting enzyme predicts left ventricular hypertrophy after renal transplantation

Background

Kidney transplant recipients show a higher risk for cardiovascular complications, such as left ventricular hypertrophy and heart failure, leading to the premature death in many cases.

Methods

We investigated the contribution of angiotensin-converting enzyme (ACE) insertion/deletion (I/D) polymorphism to the development of left ventricular hypertrophy (LVH), an indicator of heart disease progression among kidney transplant recipients.

Results

We observed a significant correlation between graft function and left ventricular mass index. The occurrence of LVH or severe LVH was significantly greater among patients with at least one D-allele (ID or DD).

Conclusion

The use of ACE inhibitors or angiotensin receptor blockers seemed to be advantageous for patients with the ID and especially, the DD genotype.     

Increasing prevalence of gastroschisis repairs in the United States: 1996-2003

Purpose

Local and state registries have shown recent increases in the prevalence of gastroschisis in the United States and abroad. The purpose of this study was to use a nationally representative database to identify national trends in the prevalence of gastroschisis repairs.

Methods

Records of infants undergoing gastroschisis repair were identified in the Nationwide Inpatient Sample from 1996 to 2003. Birth data were obtained from the National Center for Health Statistics and used to calculate the rate of procedures/live births stratified by US census region. Survey statistics were used to account for the sampling design of the Nationwide Inpatient Sample database.

Results

Between 1996 and 2003, a total of 9459 gastroschisis repairs were performed in the United States (3 procedures for every 10,000 births). A significant increase in the population-based rate of these procedures was observed in each census regions and nationwide. A twofold higher procedure rate was observed in 2003 than in 1996 (rate ratio, 2.0; 95% confidence interval, 1.1-2.9; P < .001). No significant change in unadjusted hospital mortality was observed regionally or nationally.

Conclusions

The population-based rate of gastroschisis repairs significantly increased regionally and nationwide between 1996 and 2003, paralleling recent trends described at the local and state level.     

Peroperative Gastrograffin bowel lavage in gastroschisis

Aims

The aim of the study was to evaluate potential benefits in the use of peroperative bowel lavage with Gastrograffin in neonates with gastroschisis.

Methods

A retrospective analysis of newborns with gastroschisis was performed over a 10-year period in 2 centers in the United Kingdom. Two groups were studied wherein one had peroperative bowel lavage with Gastrograffin and the other did not.

Results

Data were collected on 116 patients of whom 93 were suitable for analysis. There were no statistically significant differences in primary closure rate, duration of ventilation, parenteral nutrition, or hospital stay. Intestinal obstruction occurred more frequently in the nonlavage group.

Conclusion

Gastrograffin lavage peroperatively in gastroschisis offers no potential advantage in reducing ventilatory requirements, parenteral nutrition, and hospital stay. It also does not achieve greater primary closure rates, but may reduce the incidence of intestinal obstruction.