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doi:10.1111/jog.13662 J. Obstet. Gynaecol. Res.

2018

Stomach herniation prior to nonreassuring fetal status in a


case of fetal gastroschisis

Yuki Kojima, Katsusuke Ozawa, Rika Sugibayashi, Seiji Wada and Haruhiko Sago
Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child Health and Development, Tokyo, Japan

Abstract
The neonatal prognosis in gastroschisis is generally good; however, intrauterine fetal death (IUFD) and non-
reassuring fetal status (NRFS) sometimes occur in fetal cases of gastroschisis. Previously, we reported stom-
ach herniation as a possible risk factor for IUFD or NRFS in fetuses with gastroschisis. We report a case of
fetal gastroschisis showing stomach herniation and increased velocity of the umbilical venous (UV) flow the
day before NRFS by electronic fetal monitoring (EFM). A 27-year-old pregnant woman was followed up
from 16 weeks’ gestation due to fetal gastroschisis. At 33 + 1 weeks’ gestation, EFM displayed a reassuring
fetal state, while the ultrasound examination revealed fetal stomach herniation and an increased flow veloc-
ity of the UV in the abdomen. One day later, EFM displayed NRFS, and the patient underwent emergency
caesarean section. This case showed stomach herniation preceding the occurrence of NRFS. Stomach hernia-
tion may be a predictor of NRFS in fetuses with gastroschisis.
Key words: fetal death, gastroschisis, nonreassuring fetal status, stomach herniation, velocity of umbilical
venous flow.

Introduction with gastroschisis.5 Here, we here report a case of


fetal gastroschisis that showed stomach herniation
Gastroschisis is an abdominal wall defect and a seri- and increased velocity of the umbilical venous
ous congenital anomaly requiring neonatal surgery as (UV) flow by electronic fetal monitoring (EFM) the
the fetal intestinal tract extending from the wall defect day before NRFS.
gradually expands in utero. The incidence of gastro-
schisis is reported to be 2–5 per 10 000 births. There
have been several reports on ultrasound findings, Case Report
such as bowel dilatation, associated with the postnatal
outcome in fetuses with gastroschisis.1–3 Although A 27-year-old woman (gravida 1, para 0) was referred
neonatal prognosis is generally good, intrauterine to our hospital due to fetal gastroschisis at 16 weeks’
fetal death (IUFD) and nonreassuring fetal status gestation. She did not smoke or drink alcohol. Our ini-
(NRFS) sometimes occur. South et al.4 reported that tial ultrasound scan at 16 weeks’ gestation revealed gas-
waiting until 37 weeks’ gestation for delivery troschisis with an echogenic mass (26 × 10 × 12 mm)
increased the risk of IUFD in fetuses with gastroschi- protruding from the anterior abdomen wall. The mass
sis. However, it is unclear which prenatal findings was on the right side of the annulus umbilical, and the
best predict IUFD and NRFS in fetuses with stomach and liver were inside of the abdomen (Fig. 1a).
gastroschisis. No other fetal morphological anomalies were found.
We previously reported that stomach herniation is Serial ultrasound examinations were carried out every
a possible risk factor for IUFD or NRFS in fetuses 2 weeks until 31 weeks’ gestation and every week

Received: August 4 2017.


Accepted: March 23 2018.
Correspondence: Dr Haruhiko Sago, Center for Maternal-Fetal, Neonatal and Reproductive Medicine, National Center for Child
Health and Development, 2-10-1 Okura, Setagaya, Tokyo 157-8535, Japan. Tel.: +81-3-3416-0181, Email: sagou-h@ncchd.go.jp

© 2018 Japan Society of Obstetrics and Gynecology 1


Y. Kojima et al.

Figure 1 Ultrasound images. (a) Extra-abdominal intestine (circle) and stomach in the abdomen (arrow) of the fetus at
20 weeks’ gestation. (b) Stomach in the fetal abdomen (arrow head) at 32 weeks’ gestation. (c) Stomach herniated from
the fetal abdomen at 33 + 1 weeks’ gestation (arrow).

thereafter (Table 1). A routine fetal ultrasound examina- per minute and reduced accelerations and baseline
tion showed a dilated extra-abdominal intestine at variability (Fig. 2b). After 5 h, EFM displayed a basal
25 weeks’ gestation, so we measured the inner diameter heart rate of 170 beats per minute (tachycardia) with
and wall thickness of the extra-abdominal intestine. We reduced baseline variability (Fig. 2c), which was a
also measured the blood flow pattern in the umbilical sign of NRFS, as defined by the Japanese intrapartum
artery (UA) and the velocity of the UV flow inside of management guidelines.7 We therefore chose prompt
the fetal abdomen using a Doppler examination from delivery and performed emergency caesarean section.
23 weeks’ gestation onward. EFM was performed from A female infant weighing 1630 g with Apgar scores
33 weeks’ gestation onward. of 8 and 9 after 1 and 5 min, respectively, was deliv-
The fetal stomach was in the abdomen at 32 weeks’ ered. We failed to collect a blood sample from the
gestation (Fig. 1b). However, an ultrasound examina- umbilical artery due to its collapse; the infant venous
tion showed fetal stomach herniation (Fig. 1c) and blood pH was 7.360, and the base excess was 0.3. The
increased velocity of the UV flow inside of the fetal amniotic fluid was turbid, with yellow–brown matter
abdomen (70.9 cm/s; the 5th and 95th percentiles of floating in it. The infant was admitted to the neonatal
the velocity of UV flow were 14.9 and 40.9 cm/s, intensive care unit. An abdominal wall defect was
respectively6) and oligohydramnios at 33 + 1 weeks’ confirmed, with the dilated intestine protruding
gestation. EFM displayed a reassuring fetal state through it. A physical examination revealed no other
(Fig. 2a). However, the next day (at 33 + 2 weeks’ anomalies. The dilated intestine was swollen and
gestation), EFM showed a basal heart rate of 160 beats inflamed by amniotic fluid, but there were no findings

Table 1 Fetal ultrasound findings


16w1d 18w1d 20w1d 23w1d 25w4d 27w4d 29w1d 31w1d 32w1d 33w1d
BPD (mm) 36.3 41.7 48.9 55.3 63.2 65.9 70.3 76.0 77.9 76.5
SD† 1.1 0.5 0.7 −0.3 0 −0.9 −0.8 −0.3 −0.6 −1.5
AC (cm) 9.2 11.8 13.0 15.2 16.5 18.5 19.9 20.7 20.7 19.9
SD † −1.5 −0.8 −1.4 −1.9 −2.5 −2.3 −2.3 −2.6 −3.2 −4.0
FL (mm) 19.2 27.4 31.4 37.1 43.6 45.7 47.7 54.0 55.5 54.3
SD † −0.5 0.6 0.2 −0.4 −0.1 −0.9 −0.6 −0.6 −0.8 −1.7
MVP (cm) 4.3 5.2 4.0 3.2 3.6 3.1 4.1 4.6 3.2 1.9
Dilated extra- ― ― ― ― 0.5 0.8 0.8 1.1 1.2 1.3
abdominal bowel
(cm)
Bowel wall thickness ― ― ― ― 1.7 ― 1.8 ― 1.5 2.0
(mm)
Velocity of umbilical ― ― ― 19.4 14.6 23.4 ― 23.8 ― 70.9
venous flow
(cm/s)
†SD was calculated using the formula of the Japanese Society of Ultrasound in Medicine. AC, abdominal circumference; BPD, biparietal
diameter; d, days; FL, femur length; MVP, maximum vertical pocket; SD, standard deviation; w, weeks.

2 © 2018 Japan Society of Obstetrics and Gynecology


Stomach herniation in gastroschisis

Figure 2 Electronic fetal


monitoring (EFM). (a)
EFM on admission. (b)
EFM in the morning at
33 + 2 weeks’ gestation.
(c) EFM in the afternoon at
33 + 2 weeks’ gestation.

of intestinal atresia or necrosis. The infant received a case showed that stomach herniation and increased
silo placement at 5 h after delivery. Primary closure of UV flow velocity preceded the NRFS in a fetus with
the defect was performed at 6 days of age. Oral feed- gastroschisis.
ing with mother’s milk started at 9 days of age, and We previously reported that ultrasound findings of
the infant left the hospital at 57 days of age. stomach herniation increased the risk of IUFD or
NRFS in fetal gastroschisis.5 Antonio et al.8 reported
that gastric dilatation was associated with neonatal
Discussion death in their meta-analysis. Sinkey et al.9 reported
that an ultrasound finding associated with perinatal
We detected stomach herniation and an increased loss was an abnormal stomach location, including
velocity of the UV flow the day before NRFS. This stomach herniation before delivery. These stomach

© 2018 Japan Society of Obstetrics and Gynecology 3


Y. Kojima et al.

findings, which suggest compression of the umbilical fetal stomach herniation and abnormal UV Doppler
cord, may be associated with deterioration of the fetal findings, and the outcomes of newborns in cases of
well-being. Our case suggested that stomach hernia- fetal gastroschisis.
tion might precede the occurrence of NRFS in fetuses
with gastroschisis.
Increased velocity of the UV flow inside the fetal Disclosure
abdomen was detected before fetal tachycardia in our
case. There have been some reports of a relationship None declared.
between the flow pattern of the umbilical artery and
fetal distress. In a previous report, a notch in the UA
flow was found on a Doppler examination with NRFS References
on the EFM.10 We observed no significant changes in 1. Santiago-Munoz PC, McIntire DD, Barber RG, Megison SM,
the UA flow over the course of the pregnancy in our Twickler DW, Dashe JS. Outcomes of pregnancies with fetal
case; instead, the velocity of the UV flow increased. This gastroschisis. Obstet Gynecol 2007; 110: 663–668.
may be because (i) the UV was more susceptible to the 2. Long AM, Court J, Morabito A, Gillham JC. Antenatal
diagnosis of bowel dilatation in gastroschisis is predictive
effect of pressure than the UA, (ii) UV flow velocity of poor postnatal outcome. J Pediatr Surg 2011; 46:
might have increased due to compression of the UV 1070–1075.
inside the abdomen by stomach herniation or (iii) fetal 3. Robertson JA, Kimble RM, Stockton K, Sekar R. Antenatal
tachycardia may have been caused by hypovolemia ultrasound features in fetuses with gastroschisis and its pre-
diction in neonatal outcome. Aust N Z J Obstet Gynecol 2017;
due to pressure on the UV by stomach herniation.
57: 52–56.
The dilatation and the wall thickness of the extra- 4. South AP, Stutey KM, Derr JM. Metaanalysis of the preva-
abdominal intestine gradually progressed but were lence of intrauterine fetal death in gastroschisis. Am J Obstet
not severe in our case, and the intestine of the new- Gynecol 2013; 209: 114.e1–114.e13.
born was in good condition. A previous study 5. Kanda E, Ogawa K, Sugibayashi R et al. Stomach hernia-
tion predict fetal death or non-reassuring fetal status in
showed a relationship between the dilated extra-
gastroschisis at late pregnancy. Prenat Diagn 2013; 33:
abdominal intestine in the fetus and bowel disorder 1302–1304.
in the newborn, such as intestinal disabilities and nec- 6. Acharya G, Wilsgaard T, Berntsen GKR, Maltau JM,
rotizing enterocolitis needing enterectomy.9 Brown Kiserud T. Reference ranges for umbilical vein blood flow in
et al.11 reported that there was no relationship the second half of pregnancy based on longitudinal data.
between NRFS and the severity of extra-abdominal Prenat Diagn 2005; 25: 99–111.
7. Okai T, Ikeda T, Kawarabayashi T et al. Intrapartum man-
bowel dilatation. Durfee et al.12 reported that bowel agement guidelines based on fetal heart rate pattern classifi-
dilatation developed in 72 of 84 cases (86%), and cation. J Obstet Gynecol Res 2010; 36: 925–928.
bowel wall thickness developed in 40 of 73 cases 8. Antonio FD, Virgone C, Rizzo G et al. Prenatal risk factors
(55%), although there was no relationship between and outcomes in gastroschisis: A meta-analysis. Pediatrics
2015; 136: e159–e169.
bowel dilatation or wall thickness in the fetus and the
9. Sinkey RG, Habli MA, South AP et al. Sonographic markers
survival of fetuses and newborns with gastroschisis. associated with adverse neonatal outcomes among fetuses
We therefore believe that there is still room for discus- with gastroschisis: An 11-year, single-center review.
sion about the relationship between extra-abdominal Am J Obstet Gynecol 2016; 214: 275.e1–275.e7.
intestinal findings and the survival of fetuses or new- 10. Kalache KD, Bierlich A, Hammer H, Bollmann R. Is unex-
plained third trimester intrauterine death of fetuses with
borns with gastroschisis.
gastroschisis caused by umbilical cord compression due to
Fetuses with gastroschisis are at risk of fetal distress acute extra-abdominal bowel dilatation? Prenat Diagn 2002;
and IUFD. Our case showed that fetal stomach herni- 22: 715–717.
ation is associated with an increased velocity of the 11. Brown N, Nardi M, Greer RM et al. Prenatal extra-
UV flow preceding NRFS. This suggests the impor- abdominal bowel dilatation is a risk factor for intrapartum
tance of management with a combination of ultra- fetal compromise for fetuses with gastroschisis. Prenat Diagn
2015; 35: 529–533.
sound and EFM to prevent fetal distress and IUFD in 12. Durfee SM, Benson CB, Adams SR et al. Postnatal outcome
cases of gastroschisis. Further studies should explore of fetuses with the prenatal diagnosis of gastroschisis. J
the relationship between ultrasound findings, such as Ultrasound Med 2013; 32: 407–412.

4 © 2018 Japan Society of Obstetrics and Gynecology

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