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.

Submit your article to this journal

Article views: 86

View related articles

View Crossmark data

Full Terms & Conditions of access and use can be found at

http://www.tandfonline.com/action/journalInformation?journalCode=ijmf20

Download by: [HINARI] Date: 24 October 2017, At: 20:01

 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, Universit
a 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
Downloaded by [HINARI] at 20:01 24 October 2017

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

Gastroschisis is a congenital defect in the abdominal
wall usually to the right of a normally inserted umbil-
ical cord. It consists in a bowel and occasionally other
organs herniation outside the abdomen with no cover-
ing 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 treat-
ment: the traditional and the ward reductions. The
traditional approach is the midgut reduction and
abdominal wall closure after birth, in the operating
theater, under general anesthesia (GA) (traditional
reduction) [35]. Since 1998 the innovative experience
by Bianchi et al suggested that delayed midgut
the preferred first option for children with this defect
(ward reduction) [6]. Recently, these infants usually
managed with preformed silos followed by gradual
reduction over 35 days to prevent important compli-
cations [7].
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 reviewed the lit-
erature underlined the evaluation, management and
outcome of gastroschisis according to the surgical
treatments choice. We also reported the data
obtained by the analysis of a 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

CONTACT Daniela Luvero d.luvero@unicampus.it Department of Medicine, Unit of Gynaecology and Obstetrics, Universit
a 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
morbidity.
Definition, epidemiology, embriology
Gastroschisis is an increasingly congenital anterior
abdominal wall defect with intraperitoneal abdominal
contents protruding to the exterior [1,7] not covered
by a sac. This results in herniation of the organs adja-
cent to the normally inserted umbilical cord, usually
the bowel but may also include the stomach, liver,
spleen and bladder [8] (Figure 1).
The association with chromosomal abnormalities is
uncommon with only unusual familial case [9], but
gastroschisis may be associated with structural gastro-
intestinal anomalies such as atresia, stenosis and mal-
rotation in 10% of the cases [1012]. Rarely, it is also
associated with BeckwithWiedemann syndrome
[13,14].
Downloaded by [HINARI] at 20:01 24 October 2017
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
[1,10,13,2426].
Some theories tries to define the etiology of gastro-
schisis: 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 ephe-
drine, 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 [2831]. 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].

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 maternal serum. AFP is usually used to evaluate
chromosomal abnormalities and neural tube defects,
but it is also almost always markedly elevated in this
kind of wall defects [33].
The prenatal diagnosis of gastroschisis allows to
talk with families about the condition, treatment and
prognosis of the fetus. Moreover, an early identifica-
tion may help to identify high-risk patients in order to
choose a specialized center to optimize their outcome
[1,34]. In addition, it permits to predict and prevent
adverse events related to gastroschisis such as intra-
uterine growth retardation (IUGR), oligohydramnios,
premature delivery and fetal death.
In a fetus with gastroschisis, the exposed bowel is
vulnerable to injuries (volvulus, atresia, inflammation
or serositis) in 1015% of cases [1,12]. The most devas-
tating complication is the fetal death caused by mid-
gut volvulus or acute compromise of umbilical blood
flow by the eviscerated bowel [1]. The modality and
Downloaded by [HINARI] at 20:01 24 October 2017
timing of delivery remain controversial. No advantages
in terms of survival have been demonstrated between
vaginal and cesarean deliveries [3538], although the
preferred modality for delivery remains the vaginal.
Moreover, no benefit to preterm delivery has been
found from a large randomized English trial [39] and
other studies [40,41].
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 [4244]. After the first
evaluation of the baby (airway, breathing and circula-
tion) and stabilization of vital parameters, the herni-
ated viscera must to be covered by a transparent
plastic bag (bowel bag). Vascular access is obtained to
inject intravenous fluids, nasogastric tube is main-
tained 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 abdom-
inal wall defect closure: no significant difference
between an immediate closure and a delayed closure
after a silo has been demonstrated in literature [45,46].
Otherwise in 1998, Bianchi et al showed that a delayed
reduction and closure leads to a more stable cardio-
vascular, 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, 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.
Up to 1998, the unquestioned conventional practice
for children with gastroschisis has been to reduce the
THE JOURNAL OF MATERNAL-FETAL & NEONATAL MEDICINE 3
abdominal herniation and proceed to abdominal wall
closure as soon as possible after birth, in the operating
theater and under general anesthesia [47,48]. The
abdominal wall defect was closed by skin apposition
or prosthetic materials. Since 1998 Bianchi et al. sug-
gested that delayed midgut reduction and umbilical
port capping without anesthesia at bedside appeared
a safe technique and the preferred first option for this
pathology. There was no additional morbidity or mor-
tality [3].
Recent studies have showed that a delayed reduc-
tion 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 regard-
ing 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 gastroschi-
sis, avoiding general anesthesia and ventilation and
their associated complications.
Materials and methods
Between January 2002 and March 2013, we recorded
all data regarding liveborn infants with gastroschisis
referred to the third-level Division of Obstetrics and
Gynecology San Camillo of 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 committee. We only selected simple
cases of gastroschis (only bowel herniation) with no
bowel perforation, no ischemic gut and the absence of
intestinal atresia, at gestational age between 31 and
40 weeks, an APGAR index >7, no cardiac anomalies
prenatally diagnosed. Written informed consent 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, methods of gastroschisis reduc-
tion, 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), duration of total parental nutrition (TPN) (days),
incidence of bowel complications such as obstruction,
 4 V. BRIGANTI ET AL.

necrosis, atresia, infections. Delivery was vaginal or
cesarean section.
We have therefore retrospectively identified two
groups of infants: neonates in which gastroschis reduc-
tion was performed by the traditional technique
(group 1: elective delayed reduction under anesthesia
in operating room) and a group in which we per-
formed 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 moni-
tored (respiration, circulation). In the group 2, the
umbilical port was closed by capping with the umbil-
Downloaded by [HINARI] at 20:01 24 October 2017
MannWhitney and Fisher tests. Statistical significance
was set at p < .05.
Results
Twenty cases of gastroschisis were treated in the
third-level Division of Obstetrics and Gynecology San
Camillo of Rome between January 2002 and March
2013. Twelve infants were treated with the traditional
technique, eight infants with the other approach.
Based on eligibility criteria only 19 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 hos-

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.

Table 3. Outcomes.
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 theater within 7 days 25% (3/12) 12% (1/8)
of primary treatment (No)
Umbilical hernia 25% (3/12) 37% (3/8)
Herniotomy 66% (2/3) 0% (3/3)
statistical significance was observed for the duration of
parenteral nutrition and full oral feeds (p .004).
Related morbidity and survival are listed in Table 3.
Two-year survival was similar between two groups. No
cases of death were recorded 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 theater within
7 days from primary treatment.
Downloaded by [HINARI] at 20:01 24 October 2017
Discussion
The optimal management of gastroschisis is controver-
sial. Up to 1998, patients born with gastroschisis were
treated in the operating theater under general
anesthesia.
Since 1998, Bianchi et al suggested that delayed
midgut reduction and umbilical port capping 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 out-
comes regarding the short- and long-term complica-
tions for children treated for this abdominal wall defect.
On this scenario, we proposed a retrospective data
analysis about a surgical technique similar to the 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 gastro-
schisis defects can be performed immediately after the
birth in the delivery room encouraging the direct rela-
tionship between the mother and her child, in order
to prevent unnecessary transport to the surgical the-
ater with a consequent increase of costs and possible
complications related to the external exposure of evis-
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].
Interestingly, we showed a reduction of parental
nutrition period with a consequent full oral feed faster
THE JOURNAL OF MATERNAL-FETAL & NEONATAL MEDICINE 5
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 gastro-
schisis closure. Once the vital parameters of neonate
were stable, reduction should be undertaken in the
delivery room without 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 these infants with silos followed by gradual
reduction over 35 days [7] to prevent abdominal com-
partment syndrome.
The most common short-term complication
observed in group 2 was an umbilical hernia (37%),
but all cases were resolved spontaneously and not
required umbilical herniotomy. The same trend has
been showed, in most cases, by patients without gas-
troschisis with spontaneous resolution of umbilical
hernia.
Interestingly, we excluded a single case of an infant
who has had a disastrous of compartment syndrome
due to a severe multiorgans 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 recorded 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 tech-
nique appears to be a safe and feasible approach in a
selected group of patients with simple and not com-
plicated gastroschisis defect in order to prevent the
disastrous outcome of ACS. It represents a valid alter-
native to traditional reduction and seems to promote
the relationship between the mother and her baby
since the initial minutes of life.
Randomized multicenter casecontrol studies are
needed to validate its retrospective nature.
Acknowledgements
Authors thank Obstetrics and Gynecology Department of
San Camillo and Campus Bio Medico of Rome. They also
thank Altamedica Main Center for the access to the ultra-
sound archives.
Disclosure statement
The authors report no conflicts of interest. The authors alone
are responsible for the content and writing of this article.
 6 V. BRIGANTI ET AL.
References
1. Daniel J. Ledbetter. Congenital abdominal wall defects
and reconstruction in pediatric surgery gastroschisis
and omphalocele. Surg Clin N Am 2012;92:71327.
2. Loane M, Dolk H, Bradbury I. Increasing prevalence of
gastroschisis in Europe 19802002: a phenomenon
restricted to younger mothers? Paediatr Perinat
Epidemiol 2007;21:3639.
3. Bianchi A, Dickson AP. Elective delayed reduction and
no anesthesia: minimal intervention management for
gastrochisis. J Pediatr Surg 1998;33:133840.
4. Bowen J, Wilcox D, Bianchi A, et al. The umbilicus in
gastroschisis: aesthetic considerations. Pediatr Surg Int
1996;11:2379.
5. Filston HC. Gastroschisis primary fascial closure. Ann
Surg 1983;194:260.
6. Bianchi AP. Dickson Elective delayed reduction and no
anesthesia: minimal intervention management for gas-
troschisis. J Pediatr Surg 1998;33:133840.
7. Rao SC, Pirie S, Minutillo C, et al. Ward reduction of
gastroschisis in a single stage without general anaes-
Downloaded by [HINARI] at 20:01 24 October 2017
thesia may increase the risk of short-term morbidities:
results of a retrospective audit. J Paediatr Child Health
2009;45:3848.
8. Abdullah F, Arnold MA, Nabaweesi R, et al.
Gastroschisis in the United States 1988-2003: analysis
and risk categorization of 4344 patients. J Perinatol
2007;27:505.
9. Kohl M, Wiesel A, Schier F. Familial recurrence of gas-
troschisis: literature review and data from the popula-
tion-based birth registry Mainz Model. J Pediatr Surg
2010;45:190712.
10. Chabra, Gleason CA. Gastroschisis: embryology, patho-
genesis, epidemiology. Neoreviews 2005;6:e4939.
11. Frolov P, Alali J, Klein MD. Clinical risk factors for gas-
troschisis and omphalocele in humans: a review of the
literature. Pediatr Surg Int 2010;26:113548.
12. Owen A, Marven S, Johnson P, et al. Gastroschisis: a
national cohort study to describe contemporary surgi-
cal strategies and outcomes. J Pediatr Surg 2010;45:
180816.
13. Grosfeld JL, ONeill JA, Fonkalsrud EW, Coran AG.
Pediatric surgery. 6th ed. Philadelphia: Mosby Elsevier;
2006: 11571171.
14. Beckwith JB, Wang LI, Donnell GN, et al. Hyperplastic
fetal visceromegaly with macroglossia, omphalocele,
cytomegaly of adrenal fetal cortex, postnatal somatic
gigantism and other abnormalities: newly recognized
syndrome. Proc Am Pediatr Soc 1964;1064:1617.
15. Fillingham A, Rankin J. Prevalence, prenatal diagnosis
and survival of gastroschisis. Prenat Diagn 2008;28:
12327.
16. Reid KP, Dickinson JE, Doherty DA. The epidemiologic
incidence of congenital gastroschisis in Western
Australia. Am J Obstet Gynecol 2003;189:7648.
17. Fratelli N, Papageorghiou AT, Bhide A, et al. Outcome
of antenatally diagnosed abdominal wall defects.
Ultrasound Obstet Gynecol 2007;30:26670.
18. Barisic I, Clementi M, Hausler M, Euroscan Study
Group, et al. Evaluation of prenatal ultrasound
diagnosis of fetal abdominal wall defects by 19
European registries. Ultrasound Obstet Gynecol
2001;18:30916.
19. Forrester MB, Merz RD. Epidemiology of abdominal
wall defects, Hawaii, 19861997. Teratology 1999;3:
11723.
20. Kilby MD. The incidence of gastroschisis: Is increasing
in the UK, particularly among babies of young moth-
ers. BMJ 2006;332:2501.
21. Hoyme HE, Higginbottom MC, Jones KL. The vascular
pathogenesis of gastroschisis: intrauterine interruption
of the omphalomesenteric artery. J Pediatr
1981;98:22831.
22. Werler MM, Sheehan JE, Mitchell AA. Association of
vasoconstrictive exposures with risks of gastroschisis
and small intestinal atresia. Epidemiology 2003;14:
34954.
23. Werler MM, Sheehan JE, Mitchell AA. Maternal medica-
tion use and risks of gastroschisis and small intestinal
atresia. Am J Epidemiol 2002;155:2631.
24. Sadler TW. The embryologic origin of ventral body wall
defects. Semin Pediatr Surg 2010;19:20914.
25. Vincent E, Mortellaro SD, St. Peter FB, et al. Review of
the evidence on the closure of abdominal wall defects.
Pediatr Surg Int 2011;27:3917.
26. Holcomb GW, Murphy JP. Ashcrafts pediatric surgery,
5th ed. Amsterdam: Saunders Elsevier; 2010: 625636.
27. Feldkamp ML, Carey JC, Sadler TW. Development of
gastroschisis: review of hypotheses, a novel hypothesis,
and implications for research. Am J Med Genet 2007;
143:63952.
28. deVries PA. The pathogenesis of gastroschisis and
omphalocele. J Pediatr Surg 1980;15:24551.
29. Klein MD. Robot-enhanced fetoscopic surgery. J
Pediatr Surg 2004;39:14635.
30. Shanske AL, Pande S, Aref K, et al. Omphalocele-exstro-
phy-imperforate anus-spinal defects (OEIS) in triplet
pregnancy after IVF and CVS. Birth Defects Res Part A
Clin Mol Teratol 2003;67:46771.
31. Izant RJ, Jr, Brown F, Rothmann BF. Current embry-
ology and treatment of gastroschisis and omphalocele.
Arch Surg 1966;93:4953.
32. Stevenson RE, Rogers RC, Chandler JC, et al. Escape of
the yolk sac: a hypothesis to explain the embryogen-
esis of gastroschisis. Clin Genet 2009;75:32633.
33. Saller DN, Jr, Canick JA, Palomaki GE, et al. Second-tri-
mester maternal serum alpha-fetoprotein, unconju-
gated estriol, and hCG levels in pregnancies with
ventral wall defects. Obstet Gynecol 1994;84:8525.
34. Badillo AT, Hedrick HL, Wilson RD, et al. Prenatal ultra-
sonographic gastrointestinal abnormalities in fetuses
with gastroschisis do not correlate with postnatal out-
comes. J Pediatr Surg 2008;43:64753.
35. Boutros J, Regier M, Skarsgard ED. Is timing every-
thing? The influence of gestational age, birth weight,
route, and intent of delivery on outcome in gastroschi-
sis. J Pediatr Surg 2009;44:91217.
36. Segel SY, Marder SJ, Parry S, et al. Fetal abdominal
wall defects and mode of delivery: a systematic review.
Obstet Gynecol 2001;98:86773.
37. Abdel-Latif ME, Bolisetty S, Abeywardana S, et al. Mode
of delivery and neonatal survival of infants with

gastroschisis in Australia and New Zealand. J Pediatr
Surg 2008;43:168590.
38. Puligandla PS, Janvier A, Flageole H, et al. Routine
cesarean delivery does not improve the outcome of
infants with gastroschisis. J Pediatr Surg 2004;39:
7425.
39. Logghe HL, Mason GC, Thornton JG, et al. A randomized
controlled trial of elective preterm delivery of fetuses
with gastroschisis. J Pediatr Surg 2005;40:172631.
40. Soares H, Silva A, Rocha G, et al. Gastroschisis: preterm
or term delivery? Clinics (Sao Paulo, Brazil) 2010;65:
13942.
41. Ergun O, Barksdale E, Erg un FS, et al. The timing of
delivery of infants with gastroschisis influences out-
come. J Pediatr Surg 2005;40:4248.
42. Moir CR, Ramsey PS, Ogburn PL, et al. A prospective
trial of elective preterm delivery for fetal gastroschisis.
Am J Perinatol 2004;21:28994.
43. Hadidi A, Subotic U, Goeppl M, et al. Early elective
cesarean delivery before 36 weeks vs late spontaneous
delivery in infants with gastroschisis. J Pediatr Surg
2008;43:13426.
Downloaded by [HINARI] at 20:01 24 October 2017
THE JOURNAL OF MATERNAL-FETAL & NEONATAL MEDICINE 7
44. Vegunta RK, Wallace LJ, Leonardi MR, et al. Perinatal
management of gastroschisis: analysis of a newly
established clinical pathway. J Pediatr Surg 2005;40:
52834.
45. Pastor AC, Phillips JD, Fenton SJ, et al. Routine use of
a SILASTIC spring-loaded silo for infants with gastro-
schisis: a multicenter randomized controlled trial.
J Pediatr Surg 2008;43:180712.
46. Skarsgard ED, Claydon J, Bouchard S, et al. Canadian
Pediatric Surgical Network: a populationbased pediatric
surgery network and database for analyzing surgical
birth defects. The first 100 cases of gastroschisis.
J Pediatr Surg 2008;43:304.
47. Davies MRQ, Beale PG. The pivotal role of the surgeon
in the results achieved in gastroschisis. Pediatr Surg Int
1996;11:82.
48. Clausner A, Lukowitz A, Rump K, et al. Treatment of
congenital abdominal wall defects -a 25-year review of
132 patients. Pediatr Surg Int 1996;11:7681.
49. Loepke AW1, Soriano SG. An assessment of the effects
of general anesthetics on developing brain structure
and neurocognitive function. Anesth Analg 2008;106:1.