Materials and Methods
We conducted a retrospective cohort study of all patients who received a diagnosis of gastroschisis prenatally who delivered at 1 of the medical centers that comprise the Cincinnati Fetal Center (TriHealth Hospital or University of Cincinnati Medical Center). Subjects included mothers who were evaluated in the Cincinnati Fetal Center between January 2003 and May 2014. Exclusion criteria included delivery at <24 weeks gestation, other associated anomalies, lethal karyotype, or lost to follow-up evaluation. The study was approved by the institutional review boards at Cincinnati Children’s Hospital Medical Center, the University of Cincinnati Medical Center, and TriHealth Hospital.
Subjects were identified by querying a database that is maintained by the Cincinnati Fetal Center. The database includes prenatal and postnatal diagnoses for all mothers who are seen in consultation. Data regarding maternal demographics, obstetric and medical complications, delivery details, and maternal medical record were collected. In addition, in-utero sonograms were reviewed at 2 time points: the initial ultrasound scan at 1 of the 2 tertiary referral perinatal centers (University of Cincinnati Medical Center or TriHealth Hospital) and the ultrasound scans that were performed closest to delivery. All sonograms were performed by the Registered Diagnostic Medical Sonographers in an American Institute of Ultrasound in Medicine–accredited perinatal ultrasound unit.
Although markers have not been defined uniformly in previous studies, for the purposes of our evaluation, sonographic markers that were evaluated included (1) oligohydramnios defined as an amniotic fluid index <5 cm, (2) the presence of intrauterine growth restriction defined as an estimated fetal weight at <10th percentile defined by the Hadlock Curve, (3) abdominal circumference <10th percentile and <5th percentile, (4) the presence of intraabdominal , extraabdominal , or any bowel dilation defined as dilation >1 cm ( Figure 1 , A), and (5) abnormal gastric bubble, which included either an abnormally dilated stomach or an abnormally positioned stomach ( Figure 1 , B and C). An abnormal stomach bubble was determined by the interpreting maternal-fetal medicine specialist as previously reported in the literature that included subjective stomach dilation and/or malposition. We assessed the presence of bowel dilation using an objective cut-off of 1 cm, as previously reported as a cut-off for normality acknowledging that there is not a consensus in the literature. Subjects were categorized according to the diagnosis that was made at the time of imaging when possible and when not possible by annotated review of the archived imaging. Four perinatal outcomes were assessed: perinatal death, necrotizing enterocolitis, need for bowel resection within 48 hours of delivery, and a composite of significant bowel injury that included a diagnosis of bowel atresia or necrosis at the time of initial surgical exploration. Atresia was defined as an anatomic narrowing through which no stool could be passed; necrosis was defined as nonviable tissue found at time of surgical exploration.
Patients who were referred to the Cincinnati Fetal Center were followed with ultrasound scans for fetal growth every 3-4 weeks and twice weekly antenatal testing in the third trimester. Cesarean delivery was recommended for obstetric indications and not solely for the finding of gastroschisis, unless there was liver herniation. Timing of delivery was based on recommendations from the managing maternal-fetal medicine physician. A subanalysis that compared trial of labor vs elective cesarean and the primary perinatal outcomes was performed.
Chi-square analysis was used to analyze categoric variables. Logistic regression was performed to calculate odds ratios (OR) and 95% confidence intervals (CIs) for each sonographic marker for the adverse outcomes: perinatal loss, need for bowel resection, and presence of our composite for significant bowel injury. ORs were considered significant if the 95% CI did not include the null value of 1.0. All data analysis was performed with IBM SPSS statistical software (version 22; IBM Corporation, Armonk, NY).
Results
We identified 177 patients who delivered a fetus with gastroschisis at a study center during the study period. After consideration of the inclusion/exclusion criteria, 154 were eligible for analysis ( Figure 2 ). The demographic characteristics of our population are outlined in Table 1 . The cohort had a mean maternal age of 21.9 ± 4.1 years, median gravidity of 1, and a mean body mass index of 28.8 ± 6.6 kg/m 2 . Body mass index range extended from 17.3-57.1 kg/m 2 . Tobacco use complicated 29.9% of cases. Median gestational age at delivery was 36 weeks with 7.8% (12/154) delivered at <34 weeks gestation, 68.6% (106/154) delivered at 34-37 weeks gestation, and 23.4% (36/154) delivered at ≥37 weeks gestation. The perinatal mortality rate was 5.8%, which included 4 cases of intrauterine fetal death (2.6%) at 33, 34, 36, and 38 weeks gestational age and 5 cases of neonatal death (3.2%).
| Variable | Measure |
|---|---|
| Demographic factor | |
| Age, mean ± SD | 21.9 ± 4.1 |
| Age, range | 13-39 |
| Race, n (%) | |
| African American | 28 (18.2) |
| Asian | 2 (1.3) |
| White | 86 (55.8) |
| Hispanic | 2 (1.3) |
| Unknown | 35 (22.7) |
| Gravidity, median (interquartile range) | 1 (1–2) |
| Parity, median (interquartile range) | 0 (0–1) |
| Body mass index, kg/m 2 | |
| Mean ± SD | 28.8 ± 6.6 |
| Range | 17.3–57.1 |
| Tobacco use, n/N (%) | 44/147 (29.9) |
| Delivery information | |
| Gestational age at delivery, median (interquartile range) | 36 (35, 37) |
| Delivery, n (%) | |
| <34 Weeks gestation | 12 (7.8) |
| 34-37 Weeks gestation | 106 (68.8) |
| ≥37 Weeks gestation | 36 (23.4) |
| Cesarean delivery, n (%) | 64/150 (42.7) |
| Perinatal death, n (%) | |
| Intrauterine growth restriction | 4 (2.6) |
| Neonatal mortality | 5 (3.2) |
a Because of retrospective nature of review, some patient information and outcomes are missing. Denominator is listed for variables where N is less than the total number of participants
Delivery outcomes are summarized in Table 1 . The cesarean delivery rate was 42.7% (64/150), 20.3% (13/64) of whom were delivered electively via cesarean, with gastroschisis as the sole indication for their cesarean delivery. Among the women who attempted a vaginal delivery, the vaginal delivery rate was 79%. Mode of delivery was not associated with increased perinatal deaths ( P > .999), need for bowel resection ( P > .999), our bowel injury composite ( P = .8), or necrotizing enterocolitis ( P = .4).
The ultrasound scan at diagnosis was performed at a median gestational age of 21 weeks (interquartile range, 19–24 weeks; Table 2 ). The ultrasound scan before delivery was performed a median of 1 week (interquartile range, 1–2 weeks) before delivery ( Table 3 ). Perinatal loss was the first adverse perinatal outcome examined (n = 9; 5.8%). Markers that were associated with perinatal loss at the diagnostic ultrasound scan were abdominal circumference <5th percentile (OR, 5.56; 95% CI, 1.25–24.76), an abnormal gastric bubble (OR, 11.20; 95% CI, 2.1 –58.33), and abnormal stomach location (OR, 17.1; 95% CI, 2.99–97.85). One sonographic marker before delivery that was associated with perinatal death was an abnormal stomach bubble (OR, 5.53; 95% CI, 1.03–29.72). The second adverse perinatal outcome examined was necrotizing enterocolitis (n = 8; 5.2%). Oligohydramnios (n = 2) that was noted on the diagnostic ultrasound scan was associated with necrotizing enterocolitis of the neonate (OR, 26.25; 95% CI, 1.38–499.61). The third adverse perinatal outcome that was examined was the need for bowel resection (n = 16; 10.4%). No sonographic markers at the time of diagnosis were associated with this outcome. Gastric dilation (n = 16) that was noted on the ultrasound scan before delivery was associated with the need for bowel resection (OR, 4.36; 95% CI, 1.10–17.34). The fourth adverse perinatal outcome that was examined was a composite of significant bowel injury (n = 18; 11.7%) that included atresia and necrosis. Bowel dilation that was noted on the diagnostic ultrasound scan (OR, 9.28; 95% CI, 1.76–48.88) and on the sonogram before delivery (OR, 3.83; 95% CI, 1.1–12.39), specifically extraabdominal dilation (OR, 12.14; 95% CI, 2.82–52.27), was associated with the bowel composite.
| Indicator | Perinatal loss | Composite of significant bowel injury | Necrotizing enterocolitis | Need for bowel resection |
|---|---|---|---|---|
| Sonographic marker, n (%) | 9 (5.8) | 18 (11.7) | 8 (5.2) | 16 (10.4) |
| Intrauterine growth restriction b (n = 12) | 1.51 (0.17–13.21) | — c | 1.91 (0.21–17.23) | 0.80 (0.10–6.70) |
| Abdominal circumference <10th (n = 47) | 2.98 (0.68–13.08) | 0.83 (0.24–2.95) | 0.43 (0.05–4.03) | 1.58 (0.49–5.03) |
| Abdominal circumference <5th (n = 32) | 5.56 (1.25–24.76) | 0.60 (0.12–2.92) | 2.49 (0.39–15.80) | 0.96 (0.25–3.76) |
| Oligohydramnios (n = 2) | — c | — c | 26.25 (1.38–499.61) | — c |
| Bowel dilation (n = 7) | — c | 9.28 (1.76–48.88) | 4.04 (0.39–42.01) | 1.35 (0.15–12.16) |
| Intraabdominal dilation (n = 2) | — c | — c | — c | — c |
| Extraabdominal dilation (n =5 ) | — c | 3.59 (0.34–38.20) | — c | 2.94 (0.28–30.74) |
| Abnormal gastric bubble (n = 9) | 11.20 (2.15–58.33) | 1.41 (0.16–12.69) | — c | 3.03 (0.54–16.88) |
| Abnormal stomach location (n = 7) | 17.1 (2.99–97.85) | 2.02 (0.21–19.04) | — c | 1.68 (0.18–15.64) |
| Gastric dilation (n = 3) | 8.29 (0.67–102.85) | — c | — c | 4.43 (0.67–51.42) |
a The ultrasound scan at diagnosis was performed at a median (25th–75th interquartile range) of 21 (range, 19-24) weeks gestation
b Defined as estimated fetal weight <10th percentile
| Indicator | Perinatal loss | Composite of significant bowel injury | Necrotizing enterocolitis | Need for bowel resection |
|---|---|---|---|---|
| Intrauterine growth restriction (n = 39) | 1.89 (0.36 – 9.84) | 0.15 (0.01 – 1.18) | 0.64 (0.06 – 6.36) | 0.61 (0.15-2.40) |
| Abdominal circumference | ||||
| <10th (n = 62) | 1.46 (0.33–6.43) | 0.90 (0.28–2.89) | 0.90 (0.12–6.67) | 1.99 (0.57–6.89) |
| <5th (n = 46) | 0.89 (0.20–3.93) | 0.60 (0.17–2.09) | 0.52 (0.05–5.16) | 0.91 (0.28–3.00) |
| Oligohydramnios (n = 8) | 2.16 (0.23–20.13) | — b | — b | 1.33 (0.15–12.04) |
| Bowel dilation (n = 43) | 3.69 (0.65–21.07) | 3.83 (1.19–12.39) | 5.58 (0.56–55.64) | 1.66 (0.52–5.34) |
| Intraabdominal dilation (n = 11) | 1.84 (0.20–17.35) | 1.53 (0.46–5.09) | 3.11 (0.29–33.12) | 2.10 (0.39–11.30) |
| Extraabdominal dilation (n = 34) | 1.56 (0.25–9.80) | 12.14 (2.82–52.27) | 2.35 (0.32–17.46) | 1.21 (0.34–4.36) |
| Abnormal gastric bubble (n = 28) | 4.80 (0.86–30.37) | 2.86 (0.79–10.30) | 1.09 (0.11–11.02) | 2.94 (0.82–10.58) |
| Abnormal stomach location (n = 20) | 5.53 (1.03–29.72) | 2.46 (0.67–9.02) | 1.77 (0.17–18.12) | 1.58 (0.39–6.37) |
| Gastric dilation (n = 16) | 1.50 (0.16–14.35) | 2.53 (0.59–10.89) | — b | 4.36 (1.10–17.34) |
| Birth | ||||
| Birthweight | 0.999 (0.997–1.00) | 1.000 (0.999–1.00) | 0.999 (0.997–1.00) | 0.999 (0.998–1.00) |
| Birthweight <10% (n = 32) | 2.26 (0.51–10.00) | 1.54 (0.50–4.76) | 2.35 (0.53–10.51) | 3.03 (0.96–9.53) |
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