Slepov, Gordienko, Soroka, Gladyshko, and Ponomarenko: Impact of the Mode of Delivery on Gastroschisis Anatomical Features in the Newborn Children



Problem statement and analysis of the recent research

Gastroschisis (GS) is severe malformation characterized by penetration defect of the anterior abdominal wall through which in-utero the abdominal organs eventerate in amniotic fluid. Eventerated organs (small and large intestine, stomach, pancreas, uterus with appendages in girls and testes in boys, etc.) are not covered by embryonic membranes or their remnants. The defect is adjacent to normal umbilical cord usually on the right side of umbilicus. Umbilical ring is clefted [7, 22, 13, 4]. Cases of different localization of the defect of anterior abdominal wall (AAW) and different anatomy are very rare [10, 15, 16].

The term “Gastroschisis” comes from the Greek term for the definition of continuous anterior abdominal wall cleft, as more proper than “laparoschisis” [25]. This term was first adopted in English literature in 1973 by Calder, and for the first time GS was described in 1557 by Lycosthenes [8].

The frequency of GS in recent years has increased more than 10 times and is about 4-5 cases per 10,000 neonates [9, 18, 14].

There are simple GS with isolated defect and complicated GS in cases of associated malformations in utero or acquired pathology [11, 12]. Frequency of GS association with other malformations is 8.3-31% [12, 14, 23]. Intrauterine pathology in case of complicated GS forms include: midgut volvulus, stenosis, necrosis, perforation [20].

GS mortality rate still remains high and, ranges from 4 to 100% depending on world region [24, 13, 3, 2, 4, 22].

GS treatment outcome is affected by many factors. Mode of delivery of pregnant women with fetal with GS is prominent among them [4]. In the late twentieth and early twenty-first century, a growing number of researchers come to univocal conclusion that elective and early cesarean section has positive impact on elimination (prevention) of GS complications compared to previous experience in natural deliveries in term [17, 20, 19, 6, 13, 4].

In the course of multi-institutional trial (4 hospitals) American researchers have shown that early prenatal planned C-section reduces frequency of fibrinous layers on the walls of the eventerated intestine and a total number of complications of GS by 6.4 times (relative to natural childbirth) [20].

Thus, many foreign clinicians prove that the mode of delivery influence the condition of eventerated organs of GS which certainly affects the outcome. However, modern literature does not provide reports of research on the impact of the delivery mode on gastroschisis anatomical features. Therefore, this issue needs further investigation.

The objective of the research was to determine the effect of the delivery mode on anatomical features in newborn children.

Materials and methods of the research

Retrospective analysis of case histories of 100 children with GS born naturally or via cesarean section during the period from 1987 to 2015 was conducted.

All infants with GS were divided into 3 clinical groups depending on the prenatal diagnosis, mode of delivery, transportation, place and term of surgical correction of the defects.

The first (I) group (main) included 43 newborn children with GS. In all cases the lesion was diagnosed prenatally. These patients were born in controlled conditions of “IPOG NAMS” (“transport in the womb”). Almost all children (93.0%) were born by cesarean section. Surgical correction of defects was conducted in the first minutes of life (16.6±0.9 min) – “First minutes surgery”, under Institute conditions during 2006-2015. Gestational age of children at birth was 33-41 weeks, an average of 36.6±0.2 weeks. Among them there were 21 (48.8%) girls and 22 (51.2%) boys. Full-term children constituted 37.2% (n=16), preterm children comprised 62.8% (n=27). Body weight ranged from 1830 grams to 4020 grams, 2528.8±81.7 g on average. Intrauterine growth retardation (IUGR) was detected in 53.5% (n=23) of cases. Simple (isolated) GS was observed in 25 (58.1%) children, complicated one was found in 18 (41.9%) patients. Moreover, 10 (23.2%) cases were associated with multiple congenital malformations (MCM).

Mothers’ age in the first (main) group ranged from 16 to 36 years, an average of 22.8±0.6 years. Most infants with GS were from first pregnancy constituting 65.1% (n=28). The course of pregnancy in 74.4% of cases (n=32) was complicated: the threat of miscarriage in 12 (37.5%) women, extra genital diseases in 5 (15.6%) mothers, placental insufficiency in 7 (21.9%) cases, infection in 10 (31.2%) patients, anemia in 2 (6.2%) women. Cesarean section was performed on an emergency basis in 22.5% (n=9) of cases, electively in 76.8% of women (n=31). Moreover, the planned early cesarean section (36-37 weeks) was in 45.5% of cases (n=18) and the planned term of 32.5% (n=13).

The second (II) group (comparison) had 27 infants with GS treated also under the conditions of institution for the period 1987-2005. Prenatally diagnosis of the defect was in small number of children, namely in 29.6% (n=8). Some of these children were born in terms of control “IPOG NAMS”, namely 51.9% (n=14). Others, 48.1% (n=13) were transported to the institute from the maternity homes of Kyiv, Zhytomyr, Chernihiv and Rivne regions. The vast majority of women (n=25, 92.6%) had natural mode of delivery. A delayed surgical correction of the defects was conducted after 1-64 hours, 9.73±2.53 hours after birth on average.

Gestational age of operated children in this group was 34 - 40 weeks, an average of 37±0.2 weeks. There were 15 (55.5%) girls and 12 (44.5%) boys. There were 15 (55.5%) full-term infants and12 (45.5%) preterm ones. Body weight was 1480 - 3400 g, 2568.5±91.2 g on average. IUGR was diagnosed in 7 (25.9%) children. Simple GS was detected in 24 (88.9%) cases, complicated one was observed in 3 (11.1%) children, with MCM in 3.7% (n=1) of cases.

Maternal age in group II (comparison) averaged 19.7±0.5 years. Most women were primi-gravidas constituting 77.8% (n=21). Pathological pregnancy was detected in 17 (63.0%) women and the threat of termination in 6 (22.2%) cases, infection in 4 (14.8%) mothers, extra genital diseases in 3 (11.1%) cases, anemia in 4 (14.8%) women, feto-placental failure in 2 (7.4%) cases.

Third (III) group (comparison) included 30 infants with GS treated under the conditions of Mykolaiv Regional Children’s Hospital for the period of 1987-2005. The vast majority of cases (n=27, 90.0%) did not have prenatal diagnosis. All infants with GS were transported to Children’s Hospital from maternity homes of Mykolaiv region. In all cases, delivery took place entirely naturally. Delayed surgical correction of defects was conducted during of 1-64 hours, 9.73±2.53 hours after birth on average.

Gestational age of infants in this group ranges 32-40 weeks, 36.6±0.3 weeks on average. There were 13 (43.3%) girls and 17 (56.7%) boys. There were 8 (26.7%) full-term infants and 22 (73.3%) preterm ones. Body weight ranged from 1700 to 3400 grams, 2434.3±77.9 g on average. IUGR was detected in 36.7% (n=11) of cases. Simple GS was diagnosed in 21 (70.0%) patients, complicated one was observed in 9 (30.0%) children. Among them 1 (3.3%) child had MCM.

Maternal age in group III (comparison) was 16-27 years, 20.7±0.5 years on average. Children with GS in this group were mostly born after first pregnancy constituting 70.0% (n=21). Complicated pregnancy was noted in 23 (76.7%) mothers, namely infection was detected in 8 (26.7%) women, feto placental failure – in 5 (16.7%) cases, the threat of termination – in 3 (10.0%) women, anemia was detected in 3 (10.0%) mothers.

The location and size of defects of the anterior abdominal wall and the frequency and nature of eventerated organs were studied in children with GS in three clinical groups born via cesarean section or naturally. The following methods of study were used: prenatal and postnatal ultrasound, general clinical, radiological, results of intraoperative revision, morphological studies (in case of the dead children autopsy), statistical method.

Results of the research and their discussion

Gastroschisis anatomical variations depending on the mode of delivery are presented in the following Table 1.

Table 1

Gastroschisis anatomical variations depending on the mode of delivery

GS anatomical variations I group
C-section n=40
II group
Vaginal delivery n=25
III group
Vaginal delivery n=30
Localization of AAW defect, typical, % 100.0 100.0 100.0
Size of AAW defect, cm 3.15±0.09* 4.17±0.3* 4.7±0.29**
Characteristics and rate of eventerated organs:
Small and large intestine,% 100.0 96.0 100.0
Stomach, % 62.5 60.0 70.0
Pancreas, % 25.0* 56.0* 63.3**
Duodenum,% 25.0* 56.0* 63.3**
Uterus with appendages (in girls), and testicle (in boys), % 27.5 12.0 -
Liver and/or gall bladder, % 7.5 20.0 13.3
Urinary bladder, % 2.5 8.0 -
Greater omentum, % - 4.0 -
Shortened intestine with common mesentery, % 100.0 96.0 100.0
Isolated GS, % 57.5* 88.0* 70.0
Complicated GS, % 42.5* 12.0* 30.0
Associated congenital malformations, % 40.0* 12.0* 20.0

AAW defect localization was typical all 100% of cases (n=40) in children with GS born by cesarean section (I group), namely on the umbilical ring and unchanged umbilical cord with eventerated organs not covered by embryonic memrbane. The defect sizes varied from 2 cm to 5 cm, 3.15±0.09 cm on average and characteristics of eventerated organs were the following: small and large intestines - 100% (n=40), stomach - 62.5% (n=25 ), uterus with appendages (girls) and testicle (boys) – 27.5% (n=15), pancreas – 25.0% (n=10), liver or gall bladder – 7.5% (n=3), bladder – 2.5% (n=1).

Isolated GS was found in 57.5% (n=23) of patients; complicated one was observed in 42.5% (n=17) of cases. Associated malformations were diagnosed in 42.0% (n=16) of children with GS. Among them small bowel atresia (n=5) or large bowel atresia (n=1) was detected in 15.0% of cases; Meckel’s diverticulum was noted in 7.5% of patients (n=3), mesenteric penetration defect was observed in 12.5% of cases (n=5); angiodysplasia of bowel wall was noted in 7.5% of patients (n=3), intestinal dysplasia in 10.0% (n=4); cryptorchidism in 12.5% (n=5); inguinal hernia in 2.5% (n=1); hydronephrosis in 5.0% (n=2); artrogriposis was detected in 2.5% of cases (n=1). Multiple malformations were found in 22.5% (n=9) of cases. In all cases, n=40 (100%) intestine was shortened and had the common mesentery.

All 25 children of Group II (comparison) were born naturally; defect localization of AAW was typical: paraumbilically with split umbilical ring. AAW defect sizes were within 0.3 – 8.0 cm, 4.17±0.3 cm on average. Eventerated organs included the following: small and large intestine n=24 (96.0%), stomach n=15 (60.0%), duodenum n=14 (56.0%), pancreas n=14 (56.0%), liver (n=4), or gall bladder (n=1) - n=5 (20.0%), uterus with appendages in girls - n=3 (12.0%), urinary bladder - n=2 (8.0%), big omentum n=1 (4.0%). The intestine was shortened with common mesentery in 96.0% of cases (n=24). Isolated GS was found in 88.0% (n=22) of patients, complicated GS was noted in12.0% (n=3) of cases. Associated malformations were diagnosed in 12.0% (n=3) of cases. Among them colon atresia was observed in 4.0% of patients (n=1), cryptorchidism – was found in 4.0% of cases (n=1), hydronephrosis was noted in 4.0% of children (n=1), megaureter was detected in 4.0% of cases (n=1 ). One child had multiple defects constituting 4.0% (n=1).

In the third comparison group all 30 (100%) children with GS were born naturally, the location of AAW penetration defect was typical, namely periomphalic, on the right of split umbilical ring. Defect sizes were between 2 and 10 cm, 4.7±0.29 cm on average. Eventerated organs outside AAW included the following: small and large intestine n=30 (100.0%), stomach n=21 (70.0%), duodenum n=19 (63.3%), pancreas n=19 (63.3%), liver (n=3), or gall bladder (n=1) -n=5 (13.3 %). Isolated GS was found in 70.0% (n=21) of patients; complicated GS was detected in 9.0% (n=9) of cases. Associated malformations were diagnosed in 20.0% (n=6) of cases. Among them atresia of the small intestine (n=1) or large (n=2) intestine constituted 10.0% (n=3), enteric cyst comprised 3.3% (n=1), cryptorchidism was observed in 3.3% of cases (n=1), hydronephrosis constituted 3.3% (n=1), angiodysplasia of liver comprised 3.3% (n=1). MCM was found in 3.3% of (n=1) patients. Shortened intestine and common mesentery for small and large intestine was observed in all 30 cases.

After statistical analysis of anatomical features of GS, depending on the mode of delivery we got the following results: AAW penetration defect localization remained constant and typical for GS in all three clinical groups of patients meaning that the mode of delivery did not affect the location of the defect of AAW due to GS.

The size of the AAW penetration defect was substantially and significantly lower in children with GS born by cesarean section than in those born by vaginal delivery. Thus, the size of the defect amounted to 3.15±0.09 cm in the I group of children born by cesarean section, while in the second and third groups after vaginal birth, the defects were 4.17±0.3 cm, p<0.01 and 4.7±0.29 cm, p<0.01, respectively. The latter fact is investigated and described in literature for the first time. We consider AAW defect in children with GSH born by vaginal to be caused by the following: during labor, uterus involution, additional organs are displaced outside abdominal cavity due to AAW defect. In such cases, the retroperitoneal organs (pancreas, duodenum) and total mesentery of the small and large intestine get into the defect. This leads to tension and increase in diameter of AAW defect. It is confirmed by the fact that in children with GS born naturally (II and III clinical group), the incidence eventeration of retroperitoneal organs (pancreas, duodenum) was significantly higher than in infants born by cesarean section (I clinical group), 56.0% and 63.3% to 25.0% at p<0.01, respectively. Significant difference in the incidence of other eventerated contents in case of GS in all three clinical groups was not noted. Most often eventerated organs were small and large intestine, and the stomach and, rarely, the uterus with appendages (girls) and testicle (boys), liver and / or gallbladder, urinary bladder and greater omentum.

Conclusions

The delivery method (C-section vs. vaginal birth) affects the anatomical features of GS in newborns.

The size of AAW defect is significantly larger in children born with GS through vaginal delivery than in infants born via cesarean section, constituting, 4.17±0.3 cm (Group II) and 4.7±0.29 cm (Group III) and 3.15±0.09 cm, p<0.01 respectively. The incidence rate of eventerated retroperitoneal organs (pancreas, duodenum) is higher (56.0% and 63.3% - the second and third group and 25.0 % - I group, p<0.01).

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Copyright (c) 2017 O K Slepov, I Y Gordienko, V P Soroka, O P Gladyshko, O P Ponomarenko

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