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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.
Published online: 16 Apr 2017.

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THE JOURNAL OF MATERNAL-FETAL & NEONATAL MEDICINE, 2017
http://dx.doi.org/10.1080/14767058.2017.1311859

REVIEW ARTICLE

A novel approach in the treatment of neonatal gastroschisis: a review of the


literature and a single-center experience
Vito Brigantia, Daniela Luverob, Caterina Guliac, Roberto Piergentilid, Simona Zaamie, Elsa Laura Buffonef,
Cristina Valloneg, Roberto Angiolib, Claudio Giorlandinoh and Fabrizio Signoreg
a
Department of Pediatric Surgery and Urology, Azienda Ospedaliera San Camillo-Forlanini, Rome, Italy; bDepartment of Medicine, Unit
of Gynaecology and Obstetrics, Universita Campus Bio-Medico di Roma, Rome, Italy; cDepartment of Urologic and Gynaecologic
Sciences, Policlinico Umberto I, Sapienza University of Rome, Italy; dInstitute of Molecular Biology and Pathology, National Research
Council, Department of Biology and Biotechnologies, Sapienza University of Rome, Italy; eDepartment of Anatomical, Histological
Forensic and Orthopaedic Sciences, Sapienza University of Rome, Italy; fDepartment of Neonatal Intensive Care, Azienda
Ospedaliera San Camillo-Forlanini, Rome, Italy; gDepartment of Gynaecology, Azienda Ospedaliera San Camillo-Forlanini, Rome, Italy;
h
Department of Obstetrics and Gynecology, Altamedica Main Center, Rome, Italy

ABSTRACT ARTICLE HISTORY


Gastroschisis is a congenital abdominal wall defect and its management remains an issue. We Received 22 December 2016
performed a review of the literature to summarize its evaluation, management and outcome Revised 12 March 2017
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and we describe a new type of surgical reduction performed in our center without anesthesia Accepted 23 March 2017
(GA), immediately after birth, in the delivery room. Between January 2002 and March 2013, we
enrolled all live born infants with gastroschisis referred to the third-level Division of Obstetrics KEYWORDS
and Gynecology San Camillo of Rome. Two groups of infants were identified: group 1 in which Gastroschisis defect; surgical
gastroschis reduction was performed by the traditional technique and group 2 in which reduc- reduction; abdominal wall
tion was immediately performed after birth in the delivery room without GA. Twelve infants defects; neonatal surgery
were enrolled in group 1, and seven infants in group 2. Statistical significance was observed
between the groups regarding the hospital stay, for the duration of parenteral nutrition and full
oral feeds (p .004). Survival was similar between two groups. The reduction without GA per-
formed immediately after birth in a delivery room encourages the relationship between the
mother and her child and appears to be a safe and feasible technique in a selected group of
patients with simple gastroschisis defect; for this reason, it could represent a valid alternative to
traditional approach.

Introduction reduction without GA, when applicable, has become


the preferred first option for children with this defect
Gastroschisis is a congenital defect in the abdominal
(ward reduction) [6]. Recently, these infants usually
wall usually to the right of a normally inserted umbil-
ical cord. It consists in a bowel and occasionally other managed with preformed silos followed by gradual
organs herniation outside the abdomen with no cover- reduction over 35 days to prevent important compli-
ing membrane or sac [1]. cations [7].
The incidence appears increasing worldwide with a Unfortunately, the outcome of both surgical
strong association with young maternal age [2], approaches appears controversial due to still high
although the pathophysiology seems to be unknown. complications rate and the lack of information about
The management remains an issue and there is a their feasibility. For these reasons, we reviewed the lit-
considerable debate over the best strategy of treat- erature underlined the evaluation, management and
ment: the traditional and the ward reductions. The outcome of gastroschisis according to the surgical
traditional approach is the midgut reduction and treatments choice. We also reported the data
abdominal wall closure after birth, in the operating obtained by the analysis of a particular type of surgical
theater, under general anesthesia (GA) (traditional reduction performed in our center, immediately after
reduction) [35]. Since 1998 the innovative experience birth, in the delivery room, without anesthesia (GA).
by Bianchi et al suggested that delayed midgut We compared retrospectively its outcomes with those

CONTACT Daniela Luvero d.luvero@unicampus.it Department of Medicine, Unit of Gynaecology and Obstetrics, Universita Campus Bio-Medico di

Roma, via Alvaro del Portillo 21, Rome 00128, Italy
2017 Informa UK Limited, trading as Taylor & Francis Group
2 V. BRIGANTI ET AL.

of the classical approach, in terms of mortality and cord due to the rapid growth of the bowel tract and
morbidity. returns to the abdominal cavity before the 12th week
and a normal abdominal wall is formed
[1,10,13,2426].
Definition, epidemiology, embriology
Some theories tries to define the etiology of gastro-
Gastroschisis is an increasingly congenital anterior schisis: a thrombosis of the right omphalomesenteric
abdominal wall defect with intraperitoneal abdominal vein with the necrosis of the abdominal wall [21,27]
contents protruding to the exterior [1,7] not covered due to the use of vasoconstrictive drugs such as ephe-
by a sac. This results in herniation of the organs adja- drine, cocaine, smoking during gestation [23]. Other
cent to the normally inserted umbilical cord, usually theories include the rapidly increasing volume of the
the bowel but may also include the stomach, liver, intestine with a failure of herniation resulting in an
spleen and bladder [8] (Figure 1). abdominal wall rupture, a failure in the fusion of folds
The association with chromosomal abnormalities is in the midline and a failure of the mesoderm to form
uncommon with only unusual familial case [9], but
the anterior abdominal wall [2831]. A recent theory is
gastroschisis may be associated with structural gastro-
that the determining defect in gastroschisis is failure
intestinal anomalies such as atresia, stenosis and mal-
of the yolk sac and related vitelline structures to be
rotation in 10% of the cases [1012]. Rarely, it is also
incorporated into the umbilical stalk [32].
associated with BeckwithWiedemann syndrome
[13,14].
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The current incidence is approximately 1 to 4, 9 per Prenatal diagnosis, management and


10,000 [2,15] and is usually early diagnosed with pre- outcomes
natal ultrasound [1618]. Gastroschisis was associated
Gastroschisis is often diagnosed before birth by ultra-
with a lower overall maternal age with an incidence
sound [15] with a specificity >95%. The gestational
five times higher in mothers less than 20 years old
age for detection is about 20 weeks [18] (Figures 2 and
[19,20].
3). This wall defect shows an alpha-fetoprotein (AFP)
Although the real pathogenesis is not clear, pos-
elevations in the amniotic fluid and consequently in
sible causes for this early onset seems to be environ-
mental teratogens, oral contraceptives, aspirin, illicit
drugs, smoking and vasoconstrictive agents [2123].
The pathogenesis of gastroschisis remains contro-
versial. The gastrointestinal tract develops from the
primitive digestive tube derived from the yolk sac. The
embryonic disk is folded into cranial, 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 brain. The caudal fold rises
cranially bringing with it the bladder. Each of them
converges around the sac forming the umbilical cord. Figure 2. Bowel herniation at 22 weeks of gestation.
By the 610th week of gestation, there is a physio-
logical herniation of the intestine into the umbilical

Figure 1. Bowel herniation at 13 weeks of gestation. Figure 3. 3D imaging of bowel herniation.


THE JOURNAL OF MATERNAL-FETAL & NEONATAL MEDICINE 3

the maternal serum. AFP is usually used to evaluate abdominal herniation and proceed to abdominal wall
chromosomal abnormalities and neural tube defects, closure as soon as possible after birth, in the operating
but it is also almost always markedly elevated in this theater and under general anesthesia [47,48]. The
kind of wall defects [33]. abdominal wall defect was closed by skin apposition
The prenatal diagnosis of gastroschisis allows to or prosthetic materials. Since 1998 Bianchi et al. sug-
talk with families about the condition, treatment and gested that delayed midgut reduction and umbilical
prognosis of the fetus. Moreover, an early identifica- port capping without anesthesia at bedside appeared
tion may help to identify high-risk patients in order to a safe technique and the preferred first option for this
choose a specialized center to optimize their outcome pathology. There was no additional morbidity or mor-
[1,34]. In addition, it permits to predict and prevent tality [3].
adverse events related to gastroschisis such as intra- Recent studies have showed that a delayed reduc-
uterine growth retardation (IUGR), oligohydramnios, tion can decrease the risk of developing important
premature delivery and fetal death. complications such as compartment syndrome (ACS)
In a fetus with gastroschisis, the exposed bowel is [7].
vulnerable to injuries (volvulus, atresia, inflammation Overall survival in gastroschisis condition has
or serositis) in 1015% of cases [1,12]. The most devas- improved considerably, from 50% to 60% in 1960 to
tating complication is the fetal death caused by mid- greater than 90% currently. Most of papers examine
gut volvulus or acute compromise of umbilical blood short-term outcomes associated with abdominal wall
flow by the eviscerated bowel [1]. The modality and closure. On the contrary, long-term outcomes regard-
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timing of delivery remain controversial. No advantages


ing umbilical hernia, surgical intervention for bowel
in terms of survival have been demonstrated between
dismotility or adhesions are rare in the literature. Risk
vaginal and cesarean deliveries [3538], although the
stratification at birth may help in choosing the optimal
preferred modality for delivery remains the vaginal.
treatment for this congenital defect. Based on recent
Moreover, no benefit to preterm delivery has been
evidences, the ward reduction is feasible and safety
found from a large randomized English trial [39] and
when performed in all children with simple gastroschi-
other studies [40,41].
sis, avoiding general anesthesia and ventilation and
On the other hand, other prospective but not
their associated complications.
randomized trials report a reduction of total parental
nutrition duration, decreased hospitalization and
higher rates of primary repair [4244]. After the first Materials and methods
evaluation of the baby (airway, breathing and circula-
Between January 2002 and March 2013, we recorded
tion) and stabilization of vital parameters, the herni-
all data regarding liveborn infants with gastroschisis
ated viscera must to be covered by a transparent
referred to the third-level Division of Obstetrics and
plastic bag (bowel bag). Vascular access is obtained to
Gynecology San Camillo of Rome and sent from the
inject intravenous fluids, nasogastric tube is main-
Department of Fetal and Maternal Medicine of
tained to permit a gastric decompression in order to
prevent the distention of the gastrointestinal tract and Altamedica. Cases were identified from the fetal and
minimize the risk of aspiration. neonatal hospital database. The study was approved
A controversial issue regards the timing of abdom- by the local ethics committee. We only selected simple
inal wall defect closure: no significant difference cases of gastroschis (only bowel herniation) with no
between an immediate closure and a delayed closure bowel perforation, no ischemic gut and the absence of
after a silo has been demonstrated in literature [45,46]. intestinal atresia, at gestational age between 31 and
Otherwise in 1998, Bianchi et al showed that a delayed 40 weeks, an APGAR index >7, no cardiac anomalies
reduction and closure leads to a more stable cardio- prenatally diagnosed. Written informed consent for the
vascular, respiratory and renal parameters without procedure was obtained by the parents of neonates.
additional risk of infections [3]. The priority of surgical The following data were collected retrospectively from
management is to prevent further bowel injuries, and the databases: APGAR, gestational age in weeks, birth
for this reason, different surgical techniques have been weight in grams, sex, methods of gastroschisis reduc-
described. The first case of gastroschisis was reported tion, hospital stays (days), time to full oral feeds (day),
in 1733, the first successful closure was performed in time (min) from delivery to starting the closure of
1943 by Watkins. defect, operative times for gastroschisis reduction
Up to 1998, the unquestioned conventional practice (min), duration of total parental nutrition (TPN) (days),
for children with gastroschisis has been to reduce the incidence of bowel complications such as obstruction,
4 V. BRIGANTI ET AL.

necrosis, atresia, infections. Delivery was vaginal or MannWhitney and Fisher tests. Statistical significance
cesarean section. was set at p < .05.
We have therefore retrospectively identified two
groups of infants: neonates in which gastroschis reduc-
Results
tion was performed by the traditional technique
(group 1: elective delayed reduction under anesthesia Twenty cases of gastroschisis were treated in the
in operating room) and a group in which we per- third-level Division of Obstetrics and Gynecology San
formed this reduction immediately after delivery in the Camillo of Rome between January 2002 and March
delivery room without analgesia or sedation (group 2). 2013. Twelve infants were treated with the traditional
Concerning group 1, in the immediate period after technique, eight infants with the other approach.
delivery, gastroschisis was managed by protecting the Based on eligibility criteria only 19 patients were
eviscerated bowel in a plastic bag, decompressing the enrolled for the final analysis (12 in group 1 and 7 in
stomach by a nasogastric tube and keeping the baby group 2).
in an incubator. The child, the status of the bowel and Table 1 summarizes baseline characteristics
mesentery and the diameter of umbilical port were between two groups. Both groups were comparable
assessed. In both cases, neonates were strictly moni- and homogeneous regarding gestational age, birth
tored (respiration, circulation). In the group 2, the weight, APGAR index and type of delivery (vaginal or
umbilical port was closed by capping with the umbil- cesarean section). Statistical significance was observed
between the two groups (Table 2) regarding the hos-
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ical cord, suturing the cord to the muscle fascia with


interrupted sutures, while in the traditional surgical pital stay: in group 2, there was a median of 21 days
closure group, the umbilical port was closed by a pros- (range 1328), in group 1 was a median of 25 days
thesis. Parental feeding, through a central venous (range 1432). The age of reduction (minutes) in the
catheter, was started if enteral nutrition was not estab- group 2 was a median of 15 min from the birth (range
lished by the seventh day. Nasogastric tube aspiration 824), while in the group 1 was a median of 220 min
and intravenous fluids were maintained until bowel (range 120300). No statistical significance was
function became normal. An antibiotics profilaxis was observed for reduction time (p .35). Otherwise,
continued for 57 days.
Table 2. Comparisons of characteristics between the two
Long-term complications were also collected: umbil-
groups.
ical hernia, developmental delay, bowel obstruction,
Characteristics Group 1 Group 2 p value
medical comorbidities, mortality in the neonatal Female 4 4 .50
period, abdominal reoperations. Survival curves were Hospital stay (days) 25 (1432) 21 (1328) .003
Age at reduction (min) 220 (120300) 15 (824) .001
constructed according to the KaplanMeier estimator, Time of reduction (min) 61 (4875) 56 (4569) .35
and differences were compared by the log-rank test. Parental nutrition (days) 14 (722) 10 (718) .004
Full oral feed (day) 13 (916) 9 (512) .004
Changes from baseline were analyzed using the

Table 1. Baseline characteristics.


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
11 F 2006 2750 35 7 C None TR Yes Yes 19 None None
12 M 2006 2940 40 8 V None TR Yes No 14 None None
13 M 2002 2600 37 8 V None ER Yes No 13 None None
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
16 M 2008 2650 38 9 V None ER Yes no 17 None None
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
BD: Birth date; BW: birth weight; C: cesarean section; GA: gestational age; H: hospitalization; RT: reduction technique; TD: type of delivery; TPN: total par-
ental nutrition; V: vaginal delivery.
THE JOURNAL OF MATERNAL-FETAL & NEONATAL MEDICINE 5

Table 3. Outcomes. for group 2, allowing a reduction in the hospital stay


Outcomes (%) Group 1 Group 2 comparable with literature data. Our study also shows
Survival at 2 years 92% 88% that the outcome is not altered by the early gastro-
Mortality at 2 years 0% (0/12) 0% (0/8)
Morbidity 33% (4/12) 12% (1/8) schisis closure. Once the vital parameters of neonate
Return to the theater within 7 days 25% (3/12) 12% (1/8) were stable, reduction should be undertaken in the
of primary treatment (No)
Umbilical hernia 25% (3/12) 37% (3/8)
delivery room without further delay, preventing the
Herniotomy 66% (2/3) 0% (3/3) fluids loss through the eviscerated bowel and edema
derived from their prolonged exposure to the external.
statistical significance was observed for the duration of In literature, it was agreed that it would be preferably
parenteral nutrition and full oral feeds (p .004). to manage these infants with silos followed by gradual
Related morbidity and survival are listed in Table 3. reduction over 35 days [7] to prevent abdominal com-
Two-year survival was similar between two groups. No partment syndrome.
cases of death were recorded in the two groups. The most common short-term complication
Complications observed in group 1 were 4 (2 bowel observed in group 2 was an umbilical hernia (37%),
occlusions, 1 infection, 1 multiple cause), in group 2 but all cases were resolved spontaneously and not
was 1 (1 infection). Three infants in group 1 and only required umbilical herniotomy. The same trend has
2 infants in group 2 were returned to theater within been showed, in most cases, by patients without gas-
7 days from primary treatment. troschisis with spontaneous resolution of umbilical
hernia.
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Interestingly, we excluded a single case of an infant


Discussion
who has had a disastrous of compartment syndrome
The optimal management of gastroschisis is controver- due to a severe multiorgans herniation (liver, stomach,
sial. Up to 1998, patients born with gastroschisis were bowel) and an increased dilatation of bowel during
treated in the operating theater under general the reduction of defect.
anesthesia. Regarding long-term outcomes, the survival and
Since 1998, Bianchi et al suggested that delayed mortality rates were similar. The group 2 morbidity
midgut reduction and umbilical port capping without rate did not show any significant difference compared
anesthesia at bedside appeared safe and the preferred to the standard approach. No deaths were recorded in
first option for this pathology [3]. Unfortunately, there the two groups.
is a lack of information about the feasibility and out- Nevertheless, our analysis has several limitations
comes regarding the short- and long-term complica- due to its retrospective nature, the limited follow-up
tions for children treated for this abdominal wall defect. (2 years) and important data and cases lost for
On this scenario, we proposed a retrospective data inaccessibility to the folder. In conclusion, this tech-
analysis about a surgical technique similar to the ward nique appears to be a safe and feasible approach in a
reduction but performed immediately after birth in the selected group of patients with simple and not com-
delivery room, that seems to be a feasible and safety plicated gastroschisis defect in order to prevent the
alternative to traditional approach. disastrous outcome of ACS. It represents a valid alter-
Although our study population is not randomized, native to traditional reduction and seems to promote
the two groups appear comparable and homogeneous the relationship between the mother and her baby
with respect to gestational age, birth weight and sex since the initial minutes of life.
of infants. Randomized multicenter casecontrol studies are
This analysis showed that the reduction of gastro- needed to validate its retrospective nature.
schisis defects can be performed immediately after the
birth in the delivery room encouraging the direct rela-
Acknowledgements
tionship between the mother and her child, in order
to prevent unnecessary transport to the surgical the- Authors thank Obstetrics and Gynecology Department of
ater with a consequent increase of costs and possible San Camillo and Campus Bio Medico of Rome. They also
thank Altamedica Main Center for the access to the ultra-
complications related to the external exposure of evis- sound archives.
cerated bowel for a long time. In addition, this proced-
ure not requests GA, avoiding the possible adverse
effects of GA on the developing brain [49]. Disclosure statement
Interestingly, we showed a reduction of parental The authors report no conflicts of interest. The authors alone
nutrition period with a consequent full oral feed faster are responsible for the content and writing of this article.
6 V. BRIGANTI ET AL.

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