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Influence of Maternal Factors on Health Outcomes in Gastroschisis:A Canadian Population-Based Study

Imam Adli (030.10.132)Dhika Claresta (030.09.068)

INTRODUCTION• Canada has seen an increased incidence of gastroschisis over

the last 2 decades to approximately 1 per 2,000 live births • The increasing incidence of gastroschisis worldwide has

encouraged researchers to reexamine the epidemiology of this condition

• The etiology of gastroschisis is unclear, but there are maternal factors strongly associated with its development which may also be associated with worse neonatal outcome

• Using a disease specific national database, our goal is to describe maternal characteristics associated with gas- troschisis incidence and outcome.

MATERIALS AND METHODS• This study includes all cases of gastroschisis in the Canadian

Pediatric Surgery Network (CAPSNet) database from May 2005 through May 2010

• The CAPSNet is a national database representing 16 Canadian centers, each with a functionally linked high risk obstetrical center, level III neonatal intensive care unit, and availability of pediatric subspecialty surgery and anesthesia

• Maternal characteristics included: young age (<20 years), illicit drug use (specifically cocaine), aboriginal ethnicity, smoking during pregnancy, maternal hometown population, population density and geographic isolation (hometown 11,000 km from a tertiary pediatric surgical center or inaccessible by road or ferry)

• Several neonatal outcome variables were collected. The gastroschisis prognosis score (GPS) reflects the bowel condition at birth

• Features making up the score include the presence and severity of matting, the presence of atresia, necrosis or perforation.

• Other neonatal outcome variables included duration of parenteral nutrition (PN), and duration of hospitalization

Statistical Analysis :

• Analyses were performed using SPSS version 18

• Categorical variables were compared using Fisher’s exact test. Ordinal data were compared with Mantel-Haenszel test for trend

• Initial univariate and then multivariable analyses were per-formed with maternal variables chosen a priori (age <20 years, single parent status, alcohol and/or cigarette use during pregnancy, illicit drug use, cocaine use, geographic isolation, population density and median income)

• Dependent variables included perinatal outcomes (prenatal care, caesarian section delivery, delivery at planned center and gestational age of newborn), neonatal nutritional outcomes (PN duration 130 and 160 days, and total days of PN) and neonatal clinical outcomes (GPS moderate or high score, duration of hospitalization, days of mechanical ventilation and all-cause mortality)

• A p value 0.05 was considered statistically significant

RESULTS

Maternal Features Associated with Gastroschisis• Mothers in this database have a mean age of 22.7 more or less

5.9 years with 110/473 (23%) less than 20 years old

• Mothers frequently come from sparsely populated communities, and 24/501 (4.8%) are geographically isolated

• 267/535 mothers for whom ethnicity was recorded, white Caucasian (70%) was most commonly reported followed by native/aboriginal (18.7%)

• 161/535 (30%) of mothers reported smoking during pregnancy (38% of mothers <20 years and 32.3% of mothers >20 years). This population also reports high illicit drug usage during pregnancy 115/535 (21.5%)

Pregnancy and Delivery Outcomes• Delivery of prenatal care is excellent for most infants in this

database with 95% receiving some prenatal care and only 3.6% reporting no prenatal care

• In univariate analysis, illicit drug use and single parent status were associated with absence of prenatal care

• Multivariable analysis revealed that only single parent status was independently associated with absence of prenatal care

• Geographic isolation was independently associated with caesarian section delivery

• Maternal cocaine use was independently associated with failure to deliver at the planned institution

Neonatal Outcomes• The overall mortality of all patients included in this

database was 3.2%, with death occurring after a mean duration of hospitalization of 77 more or less 90 days

• Although maternal geographic isolation, cigarette and cocaine use all had weak association with nutritional out-comes on univariate analysis, none of these features were independently predictive of worse nutritional outcomes

• Cocaine use is independently associated with a high GPS score

• While geographic isolation is independently associated with prolonged hospitalization

• No features were independently associated with mortality or days of ventilation

• Maternal aboriginal status forced into the regression analyses was not an independent predictor of neonatal nutritional or clinical outcomes

DISCUSSION• In this report, mothers of infants with gastroschisis have a

mean age of 22.7 years, considerably younger than the average maternal age of 29 reported by Statistics Canada

• Young maternal age has been identified as a risk factor for the development of gastroschisis, Other factors include smoking, drug use, alcohol consumption, ibuprofen and vasoactive medication

• Caucasian race has an increased association with gastroschisis when compared with Hispanics and African-Americans

• Aboriginal mothers in this group have a high rate of geographic isolation (18%), young age (48% are less than 20 years of age), smoking (47%) and illicit drug use (33%)

• There are reports of case clustering and association with poor socioeconomic status

• Cigarette smoking has been linked to an increased incidence of gastroschisis. The results of our study concur with 31% of the mothers reporting cigarette smoking during pregnancy

• The Canadian Perinatal Health Report identified that 37.2% of mothers <20 years smoked during pregnancy compared with an overall maternal smoking rate of 13.4%

• The proportion of gastroschisis mothers >20 years old in this database who reported smoking during pregnancy was 37%, no different from that of other young mothers

• However, the proportion of mothers >20 years old that smoked was 34.6%, much higher than expected

• This observation suggests that cigarette smoking may have an even more significant role in the older maternal population

• The proportion of mothers that reported illicit drug use was 21.5% (marijuana in the majority of cases)

• CAPSNet did not collect information on the use of vasoactive medications or ibuprofen

• It appears that the Canadian female population at risk for the development of gastroschisis is young, frequently single and often aboriginal

• The prevalence of these risk factors in the population of mothers with gastroschisis in Canada and the relationship between these factors and poor perinatal health care outcomes suggest that targeting at risk groups may diminish the incidence of gastroschisis and may improve some of the perinatal health outcomes

• In this larger study, univariate analysis shows a mild association between cigarette smoking and prolonged PN, while multivariable analysis no longer shows this relationship

• The relation between cocaine use and bowel injury remains apparent, with cocaine use independently predicting severely injured bowel (higher GPS score)

• Aboriginals are overrepresented in mothers with gas-troschisis, yet aboriginal status did not independently predict adverse outcomes. This provides further evidence that it is the social and geographic determinants of health that influence outcome in infants with gastroschisis

• Most infants receive prenatal care in this database. These results are encouraging when viewed as a reflection of primary health care provision to mothers in Canada. There remain, however, mothers who do not receive adequate prenatal care who tend to be single, from remote communities and those with substance abuse issues

• There is a tremendous financial and social burden incurred on the health care system, the community and families when an infant with gastroschisis is born to a mother from a remote town

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