The Journal of Maternal-Fetal & Neonatal Medicine

ISSN: 1476-7058 (Print) 1476-4954 (Online) Journal homepage: http://www.tandfonline.com/loi/ijmf20

A Novel Approach in the Treatment of Neonatal

Gastroschisis: A review of the Literature and a
Single Center Experience

Vito Briganti, Daniela Luvero, Caterina Gulia, Roberto Piergentili, Simona

Zaami, Elsa Laura Buffone, Cristina Vallone, Roberto Angioli, Claudio
Giorlandino & Fabrizio Signore

To cite this article: Vito Briganti, Daniela Luvero, Caterina Gulia, Roberto Piergentili, Simona
Zaami, Elsa Laura Buffone, Cristina Vallone, Roberto Angioli, Claudio Giorlandino & Fabrizio
Signore (2017): A Novel Approach in the Treatment of Neonatal Gastroschisis: A review of the
Literature and a Single Center Experience, The Journal of Maternal-Fetal & Neonatal Medicine,
DOI: 10.1080/14767058.2017.1311859

To link to this article: http://dx.doi.org/10.1080/14767058.2017.1311859

Accepted author version posted online: 24

Mar 2017.

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 A Novel Approach in the Treatment of Neonatal Gastroschisis:
A review of the Literature and a Single Center Experience

Briganti Vito a, Luvero Daniela b, Gulia Caterina c, Piergentili Roberto d, Zaami Simona e, Buffone

Elsa Laura f, Vallone Cristina g, Angioli Roberto b, Giorlandino Claudio h, Signore Fabrizio g

Affiliations:

a
Unit of Pediatric Surgery and Urology, Azienda Ospedaliera San Camillo-Forlanini, Rome (Italy)

b
Unit of Gynaecology and Obstetrics, Department of Medicine, Università Campus Bio-Medico di

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Roma, via Álvaro del Portillo 21, 00128 Rome, Italy

c TE
Department of Urologic and Gynaecologic Sciences, Policlinico Umberto I, Sapienza – University
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of Rome (Italy)
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d
Institute of Molecular Biology and Pathology, National Research Council, and Department of
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Biology and Biotechnologies, Sapienza – University of Rome (Italy)

e
Department of Anatomical, Histological Forensic and Orthopaedic Sciences, Sapienza –
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University of Rome (Italy)

f
Unit of Neonatal Intensive Care, Azienda Ospedaliera San Camillo-Forlanini, Rome (Italy)
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g
Unit of Gynaecology, Azienda Ospedaliera San Camillo-Forlanini, Rome (Italy)

h
Altamedica Main Center, Viale Liegi, 45, Rome, (Italy)
Introduction

Gastroschisis is a congenital defect in the abdominal wall usually to the right of a normally inserted

umbilical cord. It consists in a bowel and occasionally other organs herniation outside the abdomen

with no covering membrane or sac [1].

The incidence appears increasing worldwide with a strong association with young maternal age, [2]

although the pathophysiology seems to be unknown.

The management remains an issue and there is a considerable debate over the best strategy of

treatment: the traditional and the ward reductions. The traditional approach is the midgut reduction

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and abdominal wall closure after birth, in the operating theatre, under general anesthesia (GA)

(Traditional reduction) [3,4,5]. Since 1998 the innovative experience by Bianchi et al suggested that

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delayed midgut reduction without GA, when applicable, has become the preferred first option for
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children with this defect (Ward reduction) [6]. Recently, these infants usually managed with

preformed silos followed by gradual reduction over 3-5 days to prevent important complications
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[7].
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Unfortunately, the outcome of both surgical approaches appears controversial due to still high

complications’ rate and the lack of information about their feasibility. For these reasons we
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reviewed the literature underlined the evaluation, management and outcome of gastroschisis

according to the surgical treatment’s choice. We also reported the data obtained by the analysis of a
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particular type of surgical reduction performed in our center, immediately after birth, in the delivery

room, without anesthesia (GA). We compared retrospectively its outcomes with those of the

classical approach, in terms of mortality and morbidity.

Definition, Epidemiology, Embriology

Gastroschisis is an increasingly congenital anterior abdominal wall defect with intraperitoneal

abdominal contents protruding to the exterior [7, 1] not covered by a sac. This results in herniation
of the organs adjacent to the normally inserted umbilical cord, usually the bowel, but may also

include the stomach, liver, spleen and bladder. [8] (Fig 1).

The association with chromosomal abnormalities is uncommon with only unusual familial case [9],

but gastroschisis may be associated with structural gastrointestinal anomalies such as atresia,

stenosis and malrotation in 10% of the cases [10, 11,12]. Rarely it is also associated to Beckwith-

Wiedemann syndrome [13,14].

The current incidence is approximately 1 to 4,9 per 10,000 [2,15] and is usually early diagnosed

with prenatal ultrasound [16-18]. Gastroschisis was associated with a lower overall maternal age

with an incidence five times higher in mothers less than 20 years old [19, 20].

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Although the real pathogenesis is not clear, possible causes for this early onset seems to be

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environmental teratogens, oral contraceptives, aspirin, illicit drugs, smoking and vasoconstrictive
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agents [21-23].

The pathogenesis of gastroschisis remains controversial. The gastrointestinal tract develops from
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the primitive digestive tube derived from the yolk sac. The embryonic disk is folded into cranial,
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caudal and two lateral folds. In the early gestation lateral folds meet in the anterior midline creating

the pleuroperitoneal space, the cranial fold descends bringing the heart into the midline down the
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brain. The caudal fold rises cranially bringing with it the bladder. Each of them converges around

the sac forming the umbelical cord. By the 6-10th week of gestation there is a physiological
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herniation of the intestine into the umbelical cord due to the rapid growth of the bowel tract, and

returns to the abdominal cavity before the 12th week and a normal abdominal wall is formed. [1, 10,

13, 24-26].

Some theories tryies to define the etiology of gastroschisis: a thrombosis of the right

omphalomesenteric vein with the necrosis of the abdominal wall [21, 27] due to the use of

vasoconstrictive drugs such as ephedrine, cocaine, smoking during gestation [23]. Other theories
include: the rapidly increasing volume of the intestine with a failure of herniation resulting in an

abdominal wall rupture, a failure in the fusion of folds in the midline and a failure of the mesoderm

to form the anterior abdominal wall [28-31]. A recent theory is that the determining defect in

gastroschisis is failure of the yolk sac and related vitelline structures to be incorporated into the

umbilical stalk [32].

Prenatal Diagnosis, Management and Outcomes

Gastroschisis is often diagnosed before birth by ultrasound [33] with a specificity > 95%. The

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gestational age for detection is about 20 weeks [18] (Fig 2-3). This wall defect shows an alpha-

fetoprotein (AFP) elevations in the amniotic fluid and consequently in the maternal serum. AFP is

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usually used to evaluate chromosomal abnormalities and neural tube defects, but it is also almost
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always markedly elevated in this kind of wall defects [34].

The prenatal diagnosis of gastroschisis allows to talk with families about the condition, treatment
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and prognosis of the fetus. Moreover, an early identification may help to identify high risk patients
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in order to choose a specialized center to optimize their outcome [1, 35]. In addition, it permits to

predict and prevent adverse events related to gastroschisis such as intrauterine growth retardation
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(IUGR), oligohydramnios, premature delivery and fetal death.

In a fetus with gastroschisis, the exposed bowel is vulnerable to injuries (volvulus, atresia,
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inflammation or serositis) in 10% to 15% of cases [1, 12]. The most devastating complication is the

fetal death caused by midgut volvulus or acute compromise of umbilical blood flow by the

eviscerated bowel [1]. The modality and timing of delivery remain controversial. No advantages in

terms of survival have been demonstrated between vaginal and caesarean deliveries [36-39],

although the preferred modality for delivery remains the vaginal. Moreover, no benefit to preterm

delivery has been found from a large randomized English trial [40] and other studies [41,42].
On the other hand, other prospective but not randomized trials report a reduction of total parental

nutrition duration, decreased hospitalization, and higher rates of primary repair. [43-45]. After the

first evaluation of the baby (airway, breathing and circulation) and stabilization of vital parameters,

the herniated viscera must to be covered by a transparent plastic bag (Bowel bag). Vascular access

is obtained to inject intravenous fluids, nasogastric tube is maintened to permit a gastric

decompression in order to prevent the distention of the gastrointestinal tract and minimize the risk

of aspiration.

A controversial issue regards the timing of abdominal wall defect closure: no significant difference

between an immediate closure and a delayed closure after a silo has been demonstrated in literature

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[46,47]. Otherwise in 1998, Bianchi et al showed that a delayed reduction and closure leads to a

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more stable cardiovascular, respiratory and renal parameters without additional risk of infections

[3]. The priority of surgical management is to prevent further bowel injuries and for this reason
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different surgical techniques have been described. The first case of gastroschisis was reported in

1733, the first successful closure was performed in 1943 by Watkins.

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Up to 1998 the unquestioned conventional practice for children with gastroschisis has been to
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reduce the abdominal herniation and proceed to abdominal wall closure as soon as possible after

birth, in the operating theatre and under general anesthesia [48,49]. The abdominal wall defect was
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closed by skin apposition or prothesic materials. Since 1998 Bianchi et al suggested that delayed

midgut reduction and umbilical port capping without anesthesia at bedside appeared a safe
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technique. There was no additional morbidity or mortality and when applicable , has become the

preferred first option [3].

Recent studies have showed that a delayed reduction can decrease the risk of developing important

complications such as compartment syndrome (ACS) [7].

Overall survival in gastroschisis condition has improved considerably, from 50% to 60% in 1960 to

greater than 90% currently. Most of papers examine short term outcomes associated with abdominal

wall closure. On the contrary, long term outcomes regarding umbilical hernia, surgical intervention

for bowel dismotility or adhesions are rare in the literature. Risk stratification at birth may help in

choosing the optimal treatment for this congenital defect. Based on recent evidences the ward

reduction is feasible and safety when performed in all children with simple gastroschisis, avoiding

general anesthesia and ventilation and their associated complications.

Materials and Methods

Between January 2002 and March 2013, we recordered all data regarding liveborn infants with

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gastroschisis referred to the third level Division of Obstetrics and Gynecology "San Camillo "of

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Rome and sent from the Department of Fetal and Maternal Medicine of Altamedica. Cases were

identified from the fetal and neonatal hospital database. The study was approved by the local Ethics
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Committee. We only selected simple cases of gastroschis (only bowel herniation) with no bowel

perforation, no ischemic gut and absence of intestinal atresia, at gestational age between 31 and 40
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weeks, an APGAR index > 7, no cardiac anomalies prenatally diagnosed. Written informed consent
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for the procedure was obtained by the parents of neonates. The following data were collected

retrospectively from the databases: APGAR, gestational age in weeks, birth weight in grams, sex,
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methods of gastroschisis reduction, hospital stays (days), time to full oral feeds (day), time (min)

from delivery to starting the closure of defect, operative times for gastroschisis reduction (min),
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duration of total parental nutrition (TPN) (days), incidence of bowel complications such as

obstruction, necrosis, atresia, infections. Delivery was vaginal or caesarean section.

We have therefore retrospectively identified two groups of infants: neonates in which gastroschis

reduction was performed by the traditional technique (group 1: elective delayed reduction under

anesthesia in operating room) and a group in which we performed this reduction immediately after

delivery in the delivery room without analgesia or sedation (group 2).

Concerning group 1, in the immediate period after delivery, gastroschisis was managed by

protecting the eviscerated bowel in a plastic bag, decompressing the stomach by a nasogastric tube

and keeping the baby in an incubator. The child, the status of the bowel and mesentery and the

diameter of umbilical port were assessed. In both cases neonates were strictly monitored

(respiration, circulation). In the group 2 the umbilical port was closed by capping with the umbilical

cord, suturing the cord to the muscle fascia with interrupted sutures, while in the traditional surgical

closure group the umbilical port was closed by a prothesis. Parental feeding, through a central

venous catheter, was started if enteral nutrition was not established by the seventh day. Nasogastric

tube aspiration and intravenous fluids were maintained until bowel function became normal. An

antibiotics profilaxis was continued for 5-7 days.

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Long term complications were also collected: umbilical hernia, developmental delay, bowel

obstruction, medical comorbidities, mortality in the neonatal period, abdominal reoperations.

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Survival curves were constructed according to the Kaplan–Meier estimator, and differences were

compared by the log-rank test. Changes from baseline were analyzed using the Mann–Whitney and
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Fisher tests. Statistical significance was set at p < 0.05

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Results

Twenty cases of gastroschisis were treated in the third level Division of Obstetrics and Gynecology
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“San Camillo “of Rome between January 2002 and March 2013. Twelve infants were treated with

the traditional technique, 8 infants with the other approach. Based on elegibility criteria only 19
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patients were enrolled for the final analysis (12 in group 1 and 7 in group 2).

Table 1 summarizes baseline characteristics between two groups. Both groups were comparable and

homogeneous regarding gestational age, birth weight, APGAR index and type of delivery (vaginal

or cesarean section). Statistical significance was observed between the two groups (table 2)

regarding the hospital stay: in group 2 there was a median of 21 days (range 13-28), in group 1 was
a median of 25 days (range 14-32). The age of reduction (minutes) in the group 2 was a median of

15 minutes from the birth (range 8-24), while in the group 1 was a median of 220 minutes (range

120-300). No statistical significance was observed for reduction time (p=0.35). Otherwise statistical

significance was observed for duration of parenteral nutrition and full oral feeds (p=.004).

Related morbidity and survival are listed in table 3. Two-years survival was similar between two

groups. No cases of death were recordered in the two groups. Complications observed in group 1

were 4 (2 bowel occlusions, 1 infection, 1 multiple cause,), in group 2 was 1 (1 infection). Three

infants in group 1 and only 2 infants in group 2 were returned to theatre within 7 days from primary

treatment.

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Discussion TE
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The optimal management of gastroschisis is controversial. Up to 1998 patients borned with

gastroschisis were treated in the operating theatre under general anesthesia.

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Since 1998, Bianchi et al suggested that delayed midgut reduction and umbilical port capping
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without anesthesia at bedside appeared safe and the preferred first option for this pathology [3].

Unfortunately, there is a lack of information about the feasibility and outcomes regarding the short
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and long-term complications for children treated for this abdominal wall defect.

On this scenario, we proposed a retrospective data analysis about a surgical technique similar to the
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ward reduction, but performed immediately after birth in the delivery room, that seems to be a

feasible and safety alternative to traditional approach.

Although our study population is not randomized, the two groups appear comparable and

homogeneous with respect to gestational age, birth weight and sex of infants.
This analysis showed that the reduction of gastroschisis defects can be performed immediately after

the birth in the delivery room encouraging the direct relationship between the mother and her child,

in order to prevent unnecessary transport to the surgical theatre with a consequent increase of costs

and possible complications related to the external exposure of eviscerated bowel for a long time. In

addition this procedure not requests GA, avoiding the possible adverse effects of GA on the

developing brain [50].

Interestingly, we showed a reduction of parental nutrition period with a consequent full oral feed

faster for group 2, allowing a reduction in the hospital stay comparable with literature data. Our

study also shows that the outcome is not altered by the early gastroschisis closure. Once the vital

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parameters of neonate were stable, reduction should be undertaken in the delivery room without

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further delay, preventing the fluids loss through the eviscerated bowel and edema derived from their

prolonged exposure to the external. In literature, it was agreed that it would be preferably to manage
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these infants with silos followed by gradual reduction over 3-5 days [7] to prevent abdominal

compartment syndrome.
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The most common short-term complication observed in group 2 was an umbilical hernia (37%), but
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all cases were resolved spontaneously and not required umbilical herniotomy. The same trend has

been showed, in most cases, by patients without gastroschisis with spontaneous resolution of
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umbilical hernia.

Interestingly, we excluded a single case of an infant who has had a disastrous of compartment
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syndrome due to a severe multi organs herniation (liver, stomach, bowel) and an increased

dilatation of bowel during the reduction of defect.

Regarding long-term outcomes the survival and mortality rates were similar. The group 2 morbidity

rate did not show any significant difference compared to the standard approach. No deaths were

recordered in the two groups.

Nevertheless, our analysis has several limitations due to its retrospective nature, the limited follow

up (2 years) and important data and cases lost for inaccessibility to the folder. In conclusion, this

technique appears to be a safe and feasible approach in a selected group of patients with simple and

not complicated gastroschisis defect in order to prevent the disastrous outcome of ACS. It

represents a valid alternative to traditional reduction, and seems to promote the relationship between

the mother and her baby since the initial minutes of life.

Randomized multicenter case control studies are needed to validate its retrospective nature.

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Acknowledgments

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We thanks to Obstetrics and Gynecology Department of San Camillo and Campus Bio Medico of

Rome. We also thanks to Altamedica Main Center for the access to the ultrasound archives.
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Conflict of Interest
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No potential conflict of interest was reported by the authors

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CASES SEX BD BW GA APGAR TD MALFORMATIONS RT CVC TPN H COMPLICATIONS REOPERATION

1 M 2002 2400 35 7 V None TR Yes Yes 30 None None

2 M 2002 2300 34 7 V None TR Yes Yes 29 Occlusion resection

3 F 2002 2400 35 7 V None TR Yes Yes 29 Infection Removed patch

4 M 2003 2100 34 8 C None TR Yes Yes 26 None None

5 M 2003 2070 33 8 C None TR Yes Yes 32 Multiple Removed patch and

resection

6 M 2004 940 31 7 C None TR Yes Yes 31 Occlusion None

7 M 2005 1770 33 7 C None TR Yes Yes 27 None None

8 F 2005 2500 39 9 V None TR Yes No 21 None None

9 F 2005 2600 37 8 V None TR Yes No 19 None None

10 M 2005 2680 34 8 C None TR Yes No 27 None None

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11 F 2006 2750 35 7 C None TR Yes Yes 19 None None

12

13
M

M
2006

2002
2940

2600
40

37
8

8
V

V
None

None
TR

ER
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Yes

Yes
No

No
14

13
None

None
None

None
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14 F 2005 2680 34 7 C None ER Yes Yes 19 None None

15 F 2008 2050 34 7 C None ER Yes Yes 23 None None

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16 M 2008 2650 38 9 V None ER Yes no 17 None None

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17 M 2008 2700 39 9 V None ER Yes no 16 None None

18 F 2011 2075 34 9 C None ER Yes Yes 28 Infection Revision

19 F 2013 2680 36 7 C None ER Yes yes 20 None None

ST

Legend: BD: Birth date; BW: birth weight; GA: gestational age; TD: type of delivery; V: vaginal delivery; C:
cesarean section; RT: reduction technique; TPN: total parental nutrition; H: hospitalitation
JU
Characteristics Group 1 Group 2 P value
Female 4 4 .50
Hospital stay (days) 25 (14-32) 21 (13-28) .003
Age at reduction (min) 220 (120-300) 15 (8-24) .001
Time of reduction (min) 61 (48-75) 56 (45-69) .35
Parental nutrition (days) 14 (7-22) 10 (7-18) .004
Full oral feed (day) 13 (9-16) 9 (5-12) .004

Outcomes (%) Group 1 Group 2

Survival at 2 years 92% 88%
Mortality at 2 years 0% (0/12) 0% (0/8)
Morbidity 33% (4/12) 12% (1/8)
Return to the theatre within 7 days of 25% (3/12) 12% (1/8)
primary treatment (No)
Umbilical hernia 25%(3/12) 37% (3/8)
Herniotomy 66% (2/3) 0% (3/3)

D
TE
EP
C
AC
ST
JU