Lisa Marengo - Academia.edu (original) (raw)

Papers by Lisa Marengo

Research paper thumbnail of The Association Between Major Birth Defects and Preterm Birth

Maternal and Child Health Journal, Mar 1, 2009

Objective To evaluate the association between preterm birth and major birth defects by maternal a... more Objective To evaluate the association between preterm birth and major birth defects by maternal and infant characteristics and specific types of birth defects. Study Design We pooled data for 1995-2000 from 13 states with population-based birth defects surveillance systems, representing about 30% of all U.S. births. Analyses were limited to singleton, live births from 24-44 weeks gestational age. Results Overall, birth defects were more than twice as common among preterm births (24-36 weeks) compared with term births (37-41 weeks gestation) (prevalence ratio [PR] = 2.65, 95% confidence interval [CI] 2.62-2.68), and approximately 8% of preterm births had a birth defect. Birth defects were over five times more likely among very preterm births (24-31 weeks gestation) compared with term births (PR = 5.25,, with about 16% of very preterm births having a birth defect. Defects most strongly associated with very preterm birth included central nervous system defects (PR = 16.23, 95% CI 15.49-17.00) and cardiovascular defects (PR = 9.29, 95% CI 9.03-9.56).

Research paper thumbnail of The Utility of the National Death Index as a Supplemental Data Source in Ascertaining 5-Year Mortality Among Texas Heterotaxy Cases

The Texas Birth Defects Registry (TBDR) is an active surveillance system which covers all pregnan... more The Texas Birth Defects Registry (TBDR) is an active surveillance system which covers all pregnancy outcomes and routinely links birth defects cases to in-state vital records. This study describes the value of using the National Death Index (NDI) data to supplement Texas state death certificates from vital records for a birth defects survival analysis. The cohort for this study were live-born cases with heterotaxy, a complex birth defect, delivered to Texas residents between 1999 and 2006, with a 5-year follow-up period for survival determination. Cases were linked to their Texas birth and death certificates, if present. Any live-born case that did not link to a Texas death certificate was sent to the NDI to search for any deaths that occurred. We identified 366 heterotaxy cases that were live-born in delivery years 1999-2006, 134 of which were linked to a Texas death certificate. The 232 remaining cases were sent to the NDI to search for a death certificate not found previously. This resulted in only 2 additional out-of-state deaths. Future quantification of NDI yields for birth defects survival studies would assist with further assessing the efficacy of utilizing the NDI for capturing early childhood mortality in states that routinely link to in-state death certificates.

Research paper thumbnail of Should aggregate US Census data be used as a proxy for individual household income in a birth defects registry?

Journal of Registry Management, 2011

BACKGROUND: Birth Defects Registries do not have access to income data and low household income i... more BACKGROUND: Birth Defects Registries do not have access to income data and low household income is associated with adverse pregnancy outcomes of stillbirth, preterm birth, and birth defects. We compared 1999 income data from the National Birth Defects Prevention Study (NBDPS) with 2000 Census block group income data for the residence location of these same mothers.METHODS: We geocoded 339 case mothers and 121 control mothers and assessed household income among case and control mothers by using NBDPS and census block group data. Correlation and concordance were assessed between the 2 data sources' household income data.RESULTS: The household income distribution was similar between case and control mothers within each data source. Both case and control mothers in the NBDPS's lowest household income category (<$20,000/year) reported lower income than was documented in their census block group's median household income (p-value<0.0001). A weak correlation was identified between NBDPS interview and census block group income data (control mothers, rs=0.53; case mothers, rs=0.32). There was also poor to fair concordance between the 2 data sources (control mothers, kw=0.28; 95% CI=0.19-0.37 and case mothers, kw=0.18; 95% CI=0.13-0.24).CONCLUSION: These findings demonstrate dissimilar household incomes between NBDPS and census block group data. Caution should be used if block-level data is used as a proxy for individual-level household incomes in population-based birth defects surveillance and research.

Research paper thumbnail of The Association Between Race/Ethnicity and Major Birth Defects in the United States, 1999–2007

Http Dx Doi Org 10 2105 Ajph 2014 302098, Jul 17, 2014

We investigated the relationship between race/ethnicity and 27 major birth defects. We pooled dat... more We investigated the relationship between race/ethnicity and 27 major birth defects. We pooled data from 12 population-based birth defects surveillance systems in the United States that included 13.5 million live births (1 of 3 of US births) from 1999 to 2007. Using Poisson regression, we calculated prevalence estimates for each birth defect and 13 racial/ethnic groupings, along with crude and adjusted prevalence ratios (aPRs). Non-Hispanic Whites served as the referent group. American Indians/Alaska Natives had a significantly higher and 50% or greater prevalence for 7 conditions (aPR = 3.97; 95% confidence interval [CI] = 2.89, 5.44 for anotia or microtia); aPRs of 1.5 to 2.1 for cleft lip, trisomy 18, and encephalocele, and lower, upper, and any limb deficiency). Cubans and Asians, especially Chinese and Asian Indians, had either significantly lower or similar prevalences of these defects compared with non-Hispanic Whites, with the exception of anotia or microtia among Chinese (aPR = 2.08; 95% CI = 1.30, 3.33) and Filipinos (aPR = 1.90; 95% CI = 1.10, 3.30) and tetralogy of Fallot among Vietnamese (aPR = 1.60; 95% CI = 1.11, 2.32). This is the largest population-based study to our knowledge to systematically examine the prevalence of a range of major birth defects across many racial/ethnic groups, including Asian and Hispanic subgroups. The relatively high prevalence of birth defects in American Indians/Alaska Natives warrants further attention.

Research paper thumbnail of Incidence of and Risk Factors for Delayed Diagnosis of Critical Congenital Heart Defects: A Population Based Study

Research paper thumbnail of Prevalence, Correlates, and Outcomes of Omphalocele in the United States, 1995–2005

Obstetrics & Gynecology, 2015

To examine the trends in the prevalence, epidemiologic correlates, and 1-year survival of omphalo... more To examine the trends in the prevalence, epidemiologic correlates, and 1-year survival of omphalocele using 1995-2005 data from the National Birth Defects Prevention Network in the United States. We examined 2,308 cases of omphalocele over 11 years from 12 state population-based birth defects registries. We used Poisson regression to estimate prevalence and risk factors for omphalocele and Kaplan-Meier survival curves and Cox proportional hazards regression to estimate survival patterns and hazard ratios, respectively, to examine isolated compared with nonisolated cases. Birth prevalence of omphalocele was 1.92 per 10,000 live births with no consistent trend over time. Neonates with omphalocele were more likely to be male (prevalence ratio 1.22, 95% confidence interval [CI] 1.12-1.34), born to mothers 35 years of age or older (prevalence ratio 1.77, 95% CI 1.54-2.04) and younger than 20 years (prevalence ratio 1.34, 95% CI 1.14-1.56), and of multiple births (prevalence ratio 2.22, 95% CI 1.85-2.66). The highest proportion of neonates with omphalocele had congenital heart defects (32%). The infant mortality rate was 28.7%, with 75% of those occurring in the first 28 days. The best survival was for isolated cases and the worst for neonates with chromosomal defects (hazard ratio 7.75, 95% CI 5.40-11.10) and low-birth-weight neonates (hazard ratio 7.51, 95% CI 5.86-9.63). Prevalence of omphalocele has remained constant from 1995 to 2005. Maternal age (younger than 20 years and 35 years or older), multiple gestation, and male sex are important correlates of omphalocele, whereas co-occurrence with chromosomal defects and very low birth weight are consistent determinants of 1-year survival among these neonates. II.

Research paper thumbnail of Should aggregate US Census data be used as a proxy for individual household income in a birth defects registry?

Journal of registry management, 2011

Birth Defects Registries do not have access to income data and low household income is associated... more Birth Defects Registries do not have access to income data and low household income is associated with adverse pregnancy outcomes of stillbirth, preterm birth, and birth defects. We compared 1999 income data from the National Birth Defects Prevention Study (NBDPS) with 2000 Census block group income data for the residence location of these same mothers. We geocoded 339 case mothers and 121 control mothers and assessed household income among case and control mothers by using NBDPS and census block group data. Correlation and concordance were assessed between the 2 data sources' household income data. The household income distribution was similar between case and control mothers within each data source. Both case and control mothers in the NBDPS's lowest household income category (<$20,000/year) reported lower income than was documented in their census block group's median household income (p-value<0.0001). A weak correlation was identified between NBDPS interview an...

Research paper thumbnail of Prevalence and Correlates of Gastroschisis in 15 States, 1995 to 2005

Obstetrics & Gynecology, 2013

To identify trends in the prevalence and epidemiologic correlates of gastroschisis using a large ... more To identify trends in the prevalence and epidemiologic correlates of gastroschisis using a large population-based sample with cases identified by the National Birth Defects Prevention Network over the course of an 11-year period. This study examined 4,713 cases of gastroschisis occurring in 15 states during 1995-2005, using public use natality data sets for denominators. Multivariable Poisson regression was used to identify statistically significant risk factors, and Joinpoint regression analyses were conducted to assess temporal trends in gastroschisis prevalence by maternal age and race and ethnicity. Results show an increasing temporal trend for gastroschisis (from 2.32 per 10,000 to 4.42 per 10,000 live births). Increasing prevalence of gastroschisis has occurred primarily among younger mothers (11.45 per 10,000 live births among mothers younger than age 20 years compared with 5.35 per 10,000 among women aged 20 to 24 years). In the multivariable analysis, using non-Hispanic whites as the referent group, non-Hispanic black women had the lowest risk of having a gastroschisis-affected pregnancy (prevalence ratio 0.42, 95% confidence interval [CI] 0.37-0.48), followed by Hispanics (prevalence ratio 0.86, 95% CI 0.81-0.92). Gastroschisis prevalence did not differ by newborn sex. Our findings demonstrate that the prevalence of gastroschisis has been increasing since 1995 among 15 states in the United States, and that higher rates of gastroschisis are associated with non-Hispanic white maternal race and ethnicity, and maternal age younger than 25 years (particularly younger than 20 years of age). III.

Research paper thumbnail of Descriptive epidemiology of non-syndromic complete atrioventricular canal defects

Paediatric and perinatal epidemiology, 2012

Complete atrioventricular canal defects (CAVC) are a common heart defect, but few epidemiologic s... more Complete atrioventricular canal defects (CAVC) are a common heart defect, but few epidemiologic studies have evaluated non-syndromic CAVC. Risk factors for non-syndromic CAVC have not been well established. To assess the relationship between risk for non-syndromic CAVC in offspring and several sociodemographic and reproductive parental factors, including maternal diabetes and obesity, we conducted Poisson regression analyses, using data ascertained through the Texas Birth Defects Registry, a large, population-based birth defects registry. Data were evaluated for 563 non-syndromic cases with CAVC. Significant associations were observed between non-syndromic CAVC in offspring and maternal pregestational diabetes (adjusted prevalence ratio (aPR) 6.74; 95% confidence interval (CI) 3.67, 12.37), gestational diabetes (aPR 1.69; 95% CI 1.03, 2.79) and obesity (aPR 1.69; 95% CI 1.24, 2.30). Our findings add non-syndromic CAVC to the growing list of birth defects that appear to be associate...

Research paper thumbnail of The prevalence and predictors of anencephaly and spina bifida in Texas

Paediatric and Perinatal Epidemiology, 2009

The prevalence and predictors of anencephaly and spina bifida in Texas. Paediatric and Perinatal ... more The prevalence and predictors of anencephaly and spina bifida in Texas. Paediatric and Perinatal Epidemiology 2009; 23: 41-50.

Research paper thumbnail of Body Mass Index and Birth Defects: Texas, 2005–2008

Maternal and Child Health Journal, 2013

Texas ranks 12th nationally in the proportion of adult residents who are obese; approximately 67 ... more Texas ranks 12th nationally in the proportion of adult residents who are obese; approximately 67 % of Texans are overweight or obese. Studies indicate that obesity is related to an increased risk for birth defects; however, small sample sizes have limited the scope of birth defects investigated, and only four levels of body mass index (BMI) are typically explored. Using six BMI levels, we evaluated the association between maternal BMI and birth defects in a population-based registry covering ~1.6 million births. Texas birth defect cases were linked to 2005-2008 vital records. Maternal BMI was calculated using self-reported prepregnancy weight and height from the vital record and categorized as follows: underweight (BMI &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;18.5), normal weight (BMI 18.5-24.9), overweight (BMI 25-29.9), class I obese (BMI 30-34.9), class II obese (BMI 35-39.9) and class III obese (BMI ≥40). Prevalence ratios for specific birth defects for maternal BMI categories were estimated by using normal weight as the referent, adjusted for maternal age and race/ethnicity, and stratified by maternal diabetes status. Risk for certain birth defects increased with increasing BMI (i.e., atrial and ventricular septal defects, pulmonary valve atresia, patent ductus arteriosus, and clubfoot). Risk for birth defects was substantially increased among some obese mothers (BMI ≥30) (e.g., spina bifida, tetralogy of Fallot, cleft lip with or without cleft palate, hypospadias, and epispadias). Conversely, mothers with higher BMI had a lower risk for having an infant or fetus with gastroschisis (aPR = 0.35; 95 % CI = 0.12, 0.80). Given the increased risk for birth defects associated with obesity, preconception counseling should emphasize the importance of maintaining normal weight.

Research paper thumbnail of The Association Between Major Birth Defects and Preterm Birth

Maternal and Child Health Journal, 2009

Objective To evaluate the association between preterm birth and major birth defects by maternal a... more Objective To evaluate the association between preterm birth and major birth defects by maternal and infant characteristics and specific types of birth defects. Study Design We pooled data for 1995-2000 from 13 states with population-based birth defects surveillance systems, representing about 30% of all U.S. births. Analyses were limited to singleton, live births from 24-44 weeks gestational age. Results Overall, birth defects were more than twice as common among preterm births (24-36 weeks) compared with term births (37-41 weeks gestation) (prevalence ratio [PR] = 2.65, 95% confidence interval [CI] 2.62-2.68), and approximately 8% of preterm births had a birth defect. Birth defects were over five times more likely among very preterm births (24-31 weeks gestation) compared with term births (PR = 5.25,, with about 16% of very preterm births having a birth defect. Defects most strongly associated with very preterm birth included central nervous system defects (PR = 16.23, 95% CI 15.49-17.00) and cardiovascular defects (PR = 9.29, 95% CI 9.03-9.56).

Research paper thumbnail of Time trends in the prevalence of birth defects in Texas 1999-2007: Real or artifactual?

Birth Defects Research Part A: Clinical and Molecular Teratology, 2011

BACKGROUND: Few studies have reported time trends for total birth defects or for a comprehensive ... more BACKGROUND: Few studies have reported time trends for total birth defects or for a comprehensive range of phenotypes. METHODS: We examined data from the Texas Birth Defects Registry (TBDR) from 1999 through 2007. Poisson regression was used to fit trend lines to birth prevalence over time for total birth defects (each infant/fetus counted once), for every birth defect collected by the TBDR, and for subsets of cases or defects grouped various ways. RESULTS: From 1999 through 2007, birth prevalence of total birth defects in Texas increased 3.6% per year. Increases were observed in all population groups, persisted after adjustment for demographic characteristics, and were strongest in regions of Texas that were more urban. There was a wide variety of different defects showing significant increases. The trends of several defects were driven by their mild cases. Perhaps the most compelling finding was that larger upward trends were observed in defects that had been rated as more susceptible to diagnostic variation. One notable exception to that was gastroschisis, which showed an average increase of over 5% per year, the total birth defects rate in TBDR increased at 3.6% per year, similar to 3.7% per year in birth certificate check boxes. CONCLUSIONS: In our opinion, the weight of evidence in our study suggests that the observed increase over time in total birth defects and in many specific birth defects is artifactual. This likely reflects increased awareness, referral, detection, or documentation in health care facilities visited by TBDR staff, resulting in more complete ascertainment by the registry, rather than a true change over time in the occurrence of most birth defects. Birth Defects Research (Part A) 91:902-917,

Research paper thumbnail of Descriptive epidemiology of nonsyndromic omphalocele in Texas, 1999-2004

American Journal of Medical Genetics Part A, 2009

Omphalocele is a congenital malformation that involves protrusion of abdominal contents into the ... more Omphalocele is a congenital malformation that involves protrusion of abdominal contents into the umbilicus. Though omphalocele may present as a manifestation of several chromosomal and Mendelian syndromes, the etiology for nonsyndromic omphalocele is unknown. The present study sought to estimate the birth prevalence of nonsyndromic omphalocele in offspring of women residing in Texas from 1999 to 2004, and to describe prevalence as a function of parental and infant demographic characteristics. Data on 325 cases with nonsyndromic omphalocele and 2,208,758 live births delivered during 1999-2004 were obtained from the Texas Birth Defects Registry. These data were used to estimate omphalocele birth prevalence and obtain both crude and adjusted prevalence ratios for the association of nonsyndromic omphalocele with parental and infant demographic characteristics. Nonsyndromic omphaloceles were significantly more common among the offspring of women without previous live births (adjusted prevalence ratio: 1.80, 95% CI: 1.41-2.30), compared to the offspring of women with previous live births. The prevalence of nonsyndromic omphalocele was also increased among women aged 25-29 (adjusted prevalence ratio: 1.68, 95% CI: 1.12-2.50) and women aged 40 and older (adjusted prevalence ratio: 4.83, 95% CI: 2.63-8.86) compared to the offspring of women age &lt;20, and in infants of multiple gestation pregnancies compared to singleton infants (adjusted prevalence ratio: 2.03, 95% CI: 1.22-3.37). In addition, among Hispanic women, the prevalence of nonsyndromic omphalocele was higher in the offspring of those born in the U.S. as compared to those born elsewhere (adjusted prevalence ratio: 1.50, 95% CI: 1.12-2.00). These findings augment the existing omphalocele literature.

Research paper thumbnail of Lee etal 2012

BACKGROUND: The causes of choanal atresia or stenosis (CA) are largely unknown. Infant thyroxine ... more BACKGROUND: The causes of choanal atresia or stenosis (CA) are largely unknown. Infant thyroxine (T 4 ) levels collected during newborn screening may be proxy measures for a risk factor present during the critical period of development. Therefore, we conducted a case-control study to examine the association between newborn T 4 levels and CA. METHODS: Data for cases with CA and controls were obtained from the Texas Birth Defects Registry for the period of 2004 to 2007. Information on infant T 4 levels at birth was obtained from the Texas Newborn Screening Program. Controls (n 5 3570) were drawn from unaffected births in Texas for the same period and frequency matched to cases (n 5 69) on year of birth, then linked to the newborn screening database. Logistic regression was used to evaluate the association between continuous and categorical infant T 4 levels and nonsyndromic CA. RESULTS: After adjustment for gestational age and year of birth, infant T 4 levels were inversely associated with CA (adjusted odds ratio [AOR], 0.85; 95% confidence interval [CI], 0.80-0.90). We observed a linear trend (p < 0.001) across quartiles of T 4 ; compared to infants with low levels, AORs for CA were 0.50 (95% CI, 0.28-0.91), 0.39 (95% CI, 0.20-0.75), and 0.15 (95% CI, 0.06-0.40) for infants with medium-to-low, medium, and high levels, respectively. CONCLUSIONS: Our findings suggest a role of low thyroid hormone levels in the development of CA, or that low newborn T 4 levels are potential proxy measures of a risk factor present during the critical period. Birth Defects Research (Part A) 94:951-954, 2012. Ó

Research paper thumbnail of Trends in Survival Among Children With Down Syndrome in 10 Regions of the United States

Research paper thumbnail of Ethnic Predilection of Heterotaxy Syndrome: A Population Based Study of Three Million Births

Journal of the American College of Cardiology, 2011

... Coll. Cardiol. and Heather Dickerson Keila N. Lopez, John Belmont, Mark Canfield, LisaMarengo... more ... Coll. Cardiol. and Heather Dickerson Keila N. Lopez, John Belmont, Mark Canfield, LisaMarengo, David L. Morales, POPULATION BASED STUDY OF THREE MILLION BIRTHS ETHNIC PREDILECTION OF HETEROTAXY SYNDROME: A ...

Research paper thumbnail of Escherichia coli O157 Cluster Evaluation

Emerging Infectious Diseases, 2004

We investigated a multistate cluster of Escherichia coli O157:H7 isolates; pulsed-field gel elect... more We investigated a multistate cluster of Escherichia coli O157:H7 isolates; pulsed-field gel electrophoresis subtyping, using a single enzyme, suggested an epidemiologic association. An investigation and additional subtyping, however, did not support the association. Confirming E. coli O157 clusters with two or more restriction endonucleases is necessary before public health resources are allocated to follow-up investigations.

Research paper thumbnail of Outbreak of Shiga Toxin-Producing Escherichia coli O111:H8 Infection

Clinical Infectious Diseases, 2004

Escherichia coli (STEC) other than E. coli O157. An outbreak of STEC O111:H8 infections indisting... more Escherichia coli (STEC) other than E. coli O157. An outbreak of STEC O111:H8 infections indistinguishable from E. coli O157:H7 at a youth camp highlights the need to improve non-O157 STEC surveillance. Interviews of 521 (80%) of 650 attendees revealed 55 (11%) were ill; 2 developed hemolytic-uremic syndrome. Illness was associated with consuming salad during the camp's first lunch meal (hazard ratio [HR], 4.68; ), consuming ice provided in barrels P ! .01 on the camp's final day (HR, 3.41; ), eating cob corn (HR, 3.22; ), and eating a dinner roll (HR, P ! .01 P ! .01 2.82; ). Cultures of 2 of 11 stools yielded E. coli O111:H8. Results of serologic testing and additional P ! .01 stool cultures demonstrated no evidence of infection with other bacterial pathogens, including E. coli O157, and supported infection with E. coli O111. Clinical laboratories should routinely screen suspect specimens for non-O157 STEC and should serotype and report Shiga-positive isolates.

Research paper thumbnail of Multistate study of the epidemiology of clubfoot

Birth Defects Research Part A: Clinical and Molecular Teratology, 2009

BACKGROUND: Although clubfoot is a common birth defect, with a prevalence of approximately 1 per ... more BACKGROUND: Although clubfoot is a common birth defect, with a prevalence of approximately 1 per 1000 livebirths, the etiology of clubfoot remains largely unknown. Studies of the prevalence and risk factors for clubfoot in the United States have previously been limited to specific states. The purpose of this study was to pool data from several birth defects surveillance programs to better estimate the prevalence of clubfoot and investigate its risk factors. METHODS: The 10 population-based birth defects surveillance programs that participated in this study ascertained 6139 cases of clubfoot from 2001 through 2005. A random sample of 10 controls per case, matched on year and state of birth, was selected from birth certificates. Data on infant and maternal risk factors were collected from birth certificates. Prevalence was calculated by pooling the state-specific data. Conditional logistic regression was used to investigate the association between risk factors and clubfoot. RESULTS: The overall prevalence of clubfoot was 1.29 per 1000 livebirths; 1.38 among non-Hispanic whites, 1.30 among Hispanics, and 1.14 among non-Hispanic blacks or African Americans. Maternal age, parity, education, and marital status were significantly associated with clubfoot. Maternal smoking and diabetes also showed significant associations. Several of these observed associations were consistent between surveillance programs. CONCLUSIONS: We estimated the prevalence of clubfoot using data from several birth defects programs, representing one-quarter of all births in the United States. Our findings underline the importance of birth defects surveillance programs and their utility in monitoring population-based prevalence and investigating risk factors. Birth Defects Research (Part A) 85: 897-904, 2009. Ó

Research paper thumbnail of The Association Between Major Birth Defects and Preterm Birth

Maternal and Child Health Journal, Mar 1, 2009

Objective To evaluate the association between preterm birth and major birth defects by maternal a... more Objective To evaluate the association between preterm birth and major birth defects by maternal and infant characteristics and specific types of birth defects. Study Design We pooled data for 1995-2000 from 13 states with population-based birth defects surveillance systems, representing about 30% of all U.S. births. Analyses were limited to singleton, live births from 24-44 weeks gestational age. Results Overall, birth defects were more than twice as common among preterm births (24-36 weeks) compared with term births (37-41 weeks gestation) (prevalence ratio [PR] = 2.65, 95% confidence interval [CI] 2.62-2.68), and approximately 8% of preterm births had a birth defect. Birth defects were over five times more likely among very preterm births (24-31 weeks gestation) compared with term births (PR = 5.25,, with about 16% of very preterm births having a birth defect. Defects most strongly associated with very preterm birth included central nervous system defects (PR = 16.23, 95% CI 15.49-17.00) and cardiovascular defects (PR = 9.29, 95% CI 9.03-9.56).

Research paper thumbnail of The Utility of the National Death Index as a Supplemental Data Source in Ascertaining 5-Year Mortality Among Texas Heterotaxy Cases

The Texas Birth Defects Registry (TBDR) is an active surveillance system which covers all pregnan... more The Texas Birth Defects Registry (TBDR) is an active surveillance system which covers all pregnancy outcomes and routinely links birth defects cases to in-state vital records. This study describes the value of using the National Death Index (NDI) data to supplement Texas state death certificates from vital records for a birth defects survival analysis. The cohort for this study were live-born cases with heterotaxy, a complex birth defect, delivered to Texas residents between 1999 and 2006, with a 5-year follow-up period for survival determination. Cases were linked to their Texas birth and death certificates, if present. Any live-born case that did not link to a Texas death certificate was sent to the NDI to search for any deaths that occurred. We identified 366 heterotaxy cases that were live-born in delivery years 1999-2006, 134 of which were linked to a Texas death certificate. The 232 remaining cases were sent to the NDI to search for a death certificate not found previously. This resulted in only 2 additional out-of-state deaths. Future quantification of NDI yields for birth defects survival studies would assist with further assessing the efficacy of utilizing the NDI for capturing early childhood mortality in states that routinely link to in-state death certificates.

Research paper thumbnail of Should aggregate US Census data be used as a proxy for individual household income in a birth defects registry?

Journal of Registry Management, 2011

BACKGROUND: Birth Defects Registries do not have access to income data and low household income i... more BACKGROUND: Birth Defects Registries do not have access to income data and low household income is associated with adverse pregnancy outcomes of stillbirth, preterm birth, and birth defects. We compared 1999 income data from the National Birth Defects Prevention Study (NBDPS) with 2000 Census block group income data for the residence location of these same mothers.METHODS: We geocoded 339 case mothers and 121 control mothers and assessed household income among case and control mothers by using NBDPS and census block group data. Correlation and concordance were assessed between the 2 data sources' household income data.RESULTS: The household income distribution was similar between case and control mothers within each data source. Both case and control mothers in the NBDPS's lowest household income category (<$20,000/year) reported lower income than was documented in their census block group's median household income (p-value<0.0001). A weak correlation was identified between NBDPS interview and census block group income data (control mothers, rs=0.53; case mothers, rs=0.32). There was also poor to fair concordance between the 2 data sources (control mothers, kw=0.28; 95% CI=0.19-0.37 and case mothers, kw=0.18; 95% CI=0.13-0.24).CONCLUSION: These findings demonstrate dissimilar household incomes between NBDPS and census block group data. Caution should be used if block-level data is used as a proxy for individual-level household incomes in population-based birth defects surveillance and research.

Research paper thumbnail of The Association Between Race/Ethnicity and Major Birth Defects in the United States, 1999–2007

Http Dx Doi Org 10 2105 Ajph 2014 302098, Jul 17, 2014

We investigated the relationship between race/ethnicity and 27 major birth defects. We pooled dat... more We investigated the relationship between race/ethnicity and 27 major birth defects. We pooled data from 12 population-based birth defects surveillance systems in the United States that included 13.5 million live births (1 of 3 of US births) from 1999 to 2007. Using Poisson regression, we calculated prevalence estimates for each birth defect and 13 racial/ethnic groupings, along with crude and adjusted prevalence ratios (aPRs). Non-Hispanic Whites served as the referent group. American Indians/Alaska Natives had a significantly higher and 50% or greater prevalence for 7 conditions (aPR = 3.97; 95% confidence interval [CI] = 2.89, 5.44 for anotia or microtia); aPRs of 1.5 to 2.1 for cleft lip, trisomy 18, and encephalocele, and lower, upper, and any limb deficiency). Cubans and Asians, especially Chinese and Asian Indians, had either significantly lower or similar prevalences of these defects compared with non-Hispanic Whites, with the exception of anotia or microtia among Chinese (aPR = 2.08; 95% CI = 1.30, 3.33) and Filipinos (aPR = 1.90; 95% CI = 1.10, 3.30) and tetralogy of Fallot among Vietnamese (aPR = 1.60; 95% CI = 1.11, 2.32). This is the largest population-based study to our knowledge to systematically examine the prevalence of a range of major birth defects across many racial/ethnic groups, including Asian and Hispanic subgroups. The relatively high prevalence of birth defects in American Indians/Alaska Natives warrants further attention.

Research paper thumbnail of Incidence of and Risk Factors for Delayed Diagnosis of Critical Congenital Heart Defects: A Population Based Study

Research paper thumbnail of Prevalence, Correlates, and Outcomes of Omphalocele in the United States, 1995–2005

Obstetrics & Gynecology, 2015

To examine the trends in the prevalence, epidemiologic correlates, and 1-year survival of omphalo... more To examine the trends in the prevalence, epidemiologic correlates, and 1-year survival of omphalocele using 1995-2005 data from the National Birth Defects Prevention Network in the United States. We examined 2,308 cases of omphalocele over 11 years from 12 state population-based birth defects registries. We used Poisson regression to estimate prevalence and risk factors for omphalocele and Kaplan-Meier survival curves and Cox proportional hazards regression to estimate survival patterns and hazard ratios, respectively, to examine isolated compared with nonisolated cases. Birth prevalence of omphalocele was 1.92 per 10,000 live births with no consistent trend over time. Neonates with omphalocele were more likely to be male (prevalence ratio 1.22, 95% confidence interval [CI] 1.12-1.34), born to mothers 35 years of age or older (prevalence ratio 1.77, 95% CI 1.54-2.04) and younger than 20 years (prevalence ratio 1.34, 95% CI 1.14-1.56), and of multiple births (prevalence ratio 2.22, 95% CI 1.85-2.66). The highest proportion of neonates with omphalocele had congenital heart defects (32%). The infant mortality rate was 28.7%, with 75% of those occurring in the first 28 days. The best survival was for isolated cases and the worst for neonates with chromosomal defects (hazard ratio 7.75, 95% CI 5.40-11.10) and low-birth-weight neonates (hazard ratio 7.51, 95% CI 5.86-9.63). Prevalence of omphalocele has remained constant from 1995 to 2005. Maternal age (younger than 20 years and 35 years or older), multiple gestation, and male sex are important correlates of omphalocele, whereas co-occurrence with chromosomal defects and very low birth weight are consistent determinants of 1-year survival among these neonates. II.

Research paper thumbnail of Should aggregate US Census data be used as a proxy for individual household income in a birth defects registry?

Journal of registry management, 2011

Birth Defects Registries do not have access to income data and low household income is associated... more Birth Defects Registries do not have access to income data and low household income is associated with adverse pregnancy outcomes of stillbirth, preterm birth, and birth defects. We compared 1999 income data from the National Birth Defects Prevention Study (NBDPS) with 2000 Census block group income data for the residence location of these same mothers. We geocoded 339 case mothers and 121 control mothers and assessed household income among case and control mothers by using NBDPS and census block group data. Correlation and concordance were assessed between the 2 data sources' household income data. The household income distribution was similar between case and control mothers within each data source. Both case and control mothers in the NBDPS's lowest household income category (<$20,000/year) reported lower income than was documented in their census block group's median household income (p-value<0.0001). A weak correlation was identified between NBDPS interview an...

Research paper thumbnail of Prevalence and Correlates of Gastroschisis in 15 States, 1995 to 2005

Obstetrics & Gynecology, 2013

To identify trends in the prevalence and epidemiologic correlates of gastroschisis using a large ... more To identify trends in the prevalence and epidemiologic correlates of gastroschisis using a large population-based sample with cases identified by the National Birth Defects Prevention Network over the course of an 11-year period. This study examined 4,713 cases of gastroschisis occurring in 15 states during 1995-2005, using public use natality data sets for denominators. Multivariable Poisson regression was used to identify statistically significant risk factors, and Joinpoint regression analyses were conducted to assess temporal trends in gastroschisis prevalence by maternal age and race and ethnicity. Results show an increasing temporal trend for gastroschisis (from 2.32 per 10,000 to 4.42 per 10,000 live births). Increasing prevalence of gastroschisis has occurred primarily among younger mothers (11.45 per 10,000 live births among mothers younger than age 20 years compared with 5.35 per 10,000 among women aged 20 to 24 years). In the multivariable analysis, using non-Hispanic whites as the referent group, non-Hispanic black women had the lowest risk of having a gastroschisis-affected pregnancy (prevalence ratio 0.42, 95% confidence interval [CI] 0.37-0.48), followed by Hispanics (prevalence ratio 0.86, 95% CI 0.81-0.92). Gastroschisis prevalence did not differ by newborn sex. Our findings demonstrate that the prevalence of gastroschisis has been increasing since 1995 among 15 states in the United States, and that higher rates of gastroschisis are associated with non-Hispanic white maternal race and ethnicity, and maternal age younger than 25 years (particularly younger than 20 years of age). III.

Research paper thumbnail of Descriptive epidemiology of non-syndromic complete atrioventricular canal defects

Paediatric and perinatal epidemiology, 2012

Complete atrioventricular canal defects (CAVC) are a common heart defect, but few epidemiologic s... more Complete atrioventricular canal defects (CAVC) are a common heart defect, but few epidemiologic studies have evaluated non-syndromic CAVC. Risk factors for non-syndromic CAVC have not been well established. To assess the relationship between risk for non-syndromic CAVC in offspring and several sociodemographic and reproductive parental factors, including maternal diabetes and obesity, we conducted Poisson regression analyses, using data ascertained through the Texas Birth Defects Registry, a large, population-based birth defects registry. Data were evaluated for 563 non-syndromic cases with CAVC. Significant associations were observed between non-syndromic CAVC in offspring and maternal pregestational diabetes (adjusted prevalence ratio (aPR) 6.74; 95% confidence interval (CI) 3.67, 12.37), gestational diabetes (aPR 1.69; 95% CI 1.03, 2.79) and obesity (aPR 1.69; 95% CI 1.24, 2.30). Our findings add non-syndromic CAVC to the growing list of birth defects that appear to be associate...

Research paper thumbnail of The prevalence and predictors of anencephaly and spina bifida in Texas

Paediatric and Perinatal Epidemiology, 2009

The prevalence and predictors of anencephaly and spina bifida in Texas. Paediatric and Perinatal ... more The prevalence and predictors of anencephaly and spina bifida in Texas. Paediatric and Perinatal Epidemiology 2009; 23: 41-50.

Research paper thumbnail of Body Mass Index and Birth Defects: Texas, 2005–2008

Maternal and Child Health Journal, 2013

Texas ranks 12th nationally in the proportion of adult residents who are obese; approximately 67 ... more Texas ranks 12th nationally in the proportion of adult residents who are obese; approximately 67 % of Texans are overweight or obese. Studies indicate that obesity is related to an increased risk for birth defects; however, small sample sizes have limited the scope of birth defects investigated, and only four levels of body mass index (BMI) are typically explored. Using six BMI levels, we evaluated the association between maternal BMI and birth defects in a population-based registry covering ~1.6 million births. Texas birth defect cases were linked to 2005-2008 vital records. Maternal BMI was calculated using self-reported prepregnancy weight and height from the vital record and categorized as follows: underweight (BMI &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;18.5), normal weight (BMI 18.5-24.9), overweight (BMI 25-29.9), class I obese (BMI 30-34.9), class II obese (BMI 35-39.9) and class III obese (BMI ≥40). Prevalence ratios for specific birth defects for maternal BMI categories were estimated by using normal weight as the referent, adjusted for maternal age and race/ethnicity, and stratified by maternal diabetes status. Risk for certain birth defects increased with increasing BMI (i.e., atrial and ventricular septal defects, pulmonary valve atresia, patent ductus arteriosus, and clubfoot). Risk for birth defects was substantially increased among some obese mothers (BMI ≥30) (e.g., spina bifida, tetralogy of Fallot, cleft lip with or without cleft palate, hypospadias, and epispadias). Conversely, mothers with higher BMI had a lower risk for having an infant or fetus with gastroschisis (aPR = 0.35; 95 % CI = 0.12, 0.80). Given the increased risk for birth defects associated with obesity, preconception counseling should emphasize the importance of maintaining normal weight.

Research paper thumbnail of The Association Between Major Birth Defects and Preterm Birth

Maternal and Child Health Journal, 2009

Objective To evaluate the association between preterm birth and major birth defects by maternal a... more Objective To evaluate the association between preterm birth and major birth defects by maternal and infant characteristics and specific types of birth defects. Study Design We pooled data for 1995-2000 from 13 states with population-based birth defects surveillance systems, representing about 30% of all U.S. births. Analyses were limited to singleton, live births from 24-44 weeks gestational age. Results Overall, birth defects were more than twice as common among preterm births (24-36 weeks) compared with term births (37-41 weeks gestation) (prevalence ratio [PR] = 2.65, 95% confidence interval [CI] 2.62-2.68), and approximately 8% of preterm births had a birth defect. Birth defects were over five times more likely among very preterm births (24-31 weeks gestation) compared with term births (PR = 5.25,, with about 16% of very preterm births having a birth defect. Defects most strongly associated with very preterm birth included central nervous system defects (PR = 16.23, 95% CI 15.49-17.00) and cardiovascular defects (PR = 9.29, 95% CI 9.03-9.56).

Research paper thumbnail of Time trends in the prevalence of birth defects in Texas 1999-2007: Real or artifactual?

Birth Defects Research Part A: Clinical and Molecular Teratology, 2011

BACKGROUND: Few studies have reported time trends for total birth defects or for a comprehensive ... more BACKGROUND: Few studies have reported time trends for total birth defects or for a comprehensive range of phenotypes. METHODS: We examined data from the Texas Birth Defects Registry (TBDR) from 1999 through 2007. Poisson regression was used to fit trend lines to birth prevalence over time for total birth defects (each infant/fetus counted once), for every birth defect collected by the TBDR, and for subsets of cases or defects grouped various ways. RESULTS: From 1999 through 2007, birth prevalence of total birth defects in Texas increased 3.6% per year. Increases were observed in all population groups, persisted after adjustment for demographic characteristics, and were strongest in regions of Texas that were more urban. There was a wide variety of different defects showing significant increases. The trends of several defects were driven by their mild cases. Perhaps the most compelling finding was that larger upward trends were observed in defects that had been rated as more susceptible to diagnostic variation. One notable exception to that was gastroschisis, which showed an average increase of over 5% per year, the total birth defects rate in TBDR increased at 3.6% per year, similar to 3.7% per year in birth certificate check boxes. CONCLUSIONS: In our opinion, the weight of evidence in our study suggests that the observed increase over time in total birth defects and in many specific birth defects is artifactual. This likely reflects increased awareness, referral, detection, or documentation in health care facilities visited by TBDR staff, resulting in more complete ascertainment by the registry, rather than a true change over time in the occurrence of most birth defects. Birth Defects Research (Part A) 91:902-917,

Research paper thumbnail of Descriptive epidemiology of nonsyndromic omphalocele in Texas, 1999-2004

American Journal of Medical Genetics Part A, 2009

Omphalocele is a congenital malformation that involves protrusion of abdominal contents into the ... more Omphalocele is a congenital malformation that involves protrusion of abdominal contents into the umbilicus. Though omphalocele may present as a manifestation of several chromosomal and Mendelian syndromes, the etiology for nonsyndromic omphalocele is unknown. The present study sought to estimate the birth prevalence of nonsyndromic omphalocele in offspring of women residing in Texas from 1999 to 2004, and to describe prevalence as a function of parental and infant demographic characteristics. Data on 325 cases with nonsyndromic omphalocele and 2,208,758 live births delivered during 1999-2004 were obtained from the Texas Birth Defects Registry. These data were used to estimate omphalocele birth prevalence and obtain both crude and adjusted prevalence ratios for the association of nonsyndromic omphalocele with parental and infant demographic characteristics. Nonsyndromic omphaloceles were significantly more common among the offspring of women without previous live births (adjusted prevalence ratio: 1.80, 95% CI: 1.41-2.30), compared to the offspring of women with previous live births. The prevalence of nonsyndromic omphalocele was also increased among women aged 25-29 (adjusted prevalence ratio: 1.68, 95% CI: 1.12-2.50) and women aged 40 and older (adjusted prevalence ratio: 4.83, 95% CI: 2.63-8.86) compared to the offspring of women age &lt;20, and in infants of multiple gestation pregnancies compared to singleton infants (adjusted prevalence ratio: 2.03, 95% CI: 1.22-3.37). In addition, among Hispanic women, the prevalence of nonsyndromic omphalocele was higher in the offspring of those born in the U.S. as compared to those born elsewhere (adjusted prevalence ratio: 1.50, 95% CI: 1.12-2.00). These findings augment the existing omphalocele literature.

Research paper thumbnail of Lee etal 2012

BACKGROUND: The causes of choanal atresia or stenosis (CA) are largely unknown. Infant thyroxine ... more BACKGROUND: The causes of choanal atresia or stenosis (CA) are largely unknown. Infant thyroxine (T 4 ) levels collected during newborn screening may be proxy measures for a risk factor present during the critical period of development. Therefore, we conducted a case-control study to examine the association between newborn T 4 levels and CA. METHODS: Data for cases with CA and controls were obtained from the Texas Birth Defects Registry for the period of 2004 to 2007. Information on infant T 4 levels at birth was obtained from the Texas Newborn Screening Program. Controls (n 5 3570) were drawn from unaffected births in Texas for the same period and frequency matched to cases (n 5 69) on year of birth, then linked to the newborn screening database. Logistic regression was used to evaluate the association between continuous and categorical infant T 4 levels and nonsyndromic CA. RESULTS: After adjustment for gestational age and year of birth, infant T 4 levels were inversely associated with CA (adjusted odds ratio [AOR], 0.85; 95% confidence interval [CI], 0.80-0.90). We observed a linear trend (p < 0.001) across quartiles of T 4 ; compared to infants with low levels, AORs for CA were 0.50 (95% CI, 0.28-0.91), 0.39 (95% CI, 0.20-0.75), and 0.15 (95% CI, 0.06-0.40) for infants with medium-to-low, medium, and high levels, respectively. CONCLUSIONS: Our findings suggest a role of low thyroid hormone levels in the development of CA, or that low newborn T 4 levels are potential proxy measures of a risk factor present during the critical period. Birth Defects Research (Part A) 94:951-954, 2012. Ó

Research paper thumbnail of Trends in Survival Among Children With Down Syndrome in 10 Regions of the United States

Research paper thumbnail of Ethnic Predilection of Heterotaxy Syndrome: A Population Based Study of Three Million Births

Journal of the American College of Cardiology, 2011

... Coll. Cardiol. and Heather Dickerson Keila N. Lopez, John Belmont, Mark Canfield, LisaMarengo... more ... Coll. Cardiol. and Heather Dickerson Keila N. Lopez, John Belmont, Mark Canfield, LisaMarengo, David L. Morales, POPULATION BASED STUDY OF THREE MILLION BIRTHS ETHNIC PREDILECTION OF HETEROTAXY SYNDROME: A ...

Research paper thumbnail of Escherichia coli O157 Cluster Evaluation

Emerging Infectious Diseases, 2004

We investigated a multistate cluster of Escherichia coli O157:H7 isolates; pulsed-field gel elect... more We investigated a multistate cluster of Escherichia coli O157:H7 isolates; pulsed-field gel electrophoresis subtyping, using a single enzyme, suggested an epidemiologic association. An investigation and additional subtyping, however, did not support the association. Confirming E. coli O157 clusters with two or more restriction endonucleases is necessary before public health resources are allocated to follow-up investigations.

Research paper thumbnail of Outbreak of Shiga Toxin-Producing Escherichia coli O111:H8 Infection

Clinical Infectious Diseases, 2004

Escherichia coli (STEC) other than E. coli O157. An outbreak of STEC O111:H8 infections indisting... more Escherichia coli (STEC) other than E. coli O157. An outbreak of STEC O111:H8 infections indistinguishable from E. coli O157:H7 at a youth camp highlights the need to improve non-O157 STEC surveillance. Interviews of 521 (80%) of 650 attendees revealed 55 (11%) were ill; 2 developed hemolytic-uremic syndrome. Illness was associated with consuming salad during the camp's first lunch meal (hazard ratio [HR], 4.68; ), consuming ice provided in barrels P ! .01 on the camp's final day (HR, 3.41; ), eating cob corn (HR, 3.22; ), and eating a dinner roll (HR, P ! .01 P ! .01 2.82; ). Cultures of 2 of 11 stools yielded E. coli O111:H8. Results of serologic testing and additional P ! .01 stool cultures demonstrated no evidence of infection with other bacterial pathogens, including E. coli O157, and supported infection with E. coli O111. Clinical laboratories should routinely screen suspect specimens for non-O157 STEC and should serotype and report Shiga-positive isolates.

Research paper thumbnail of Multistate study of the epidemiology of clubfoot

Birth Defects Research Part A: Clinical and Molecular Teratology, 2009

BACKGROUND: Although clubfoot is a common birth defect, with a prevalence of approximately 1 per ... more BACKGROUND: Although clubfoot is a common birth defect, with a prevalence of approximately 1 per 1000 livebirths, the etiology of clubfoot remains largely unknown. Studies of the prevalence and risk factors for clubfoot in the United States have previously been limited to specific states. The purpose of this study was to pool data from several birth defects surveillance programs to better estimate the prevalence of clubfoot and investigate its risk factors. METHODS: The 10 population-based birth defects surveillance programs that participated in this study ascertained 6139 cases of clubfoot from 2001 through 2005. A random sample of 10 controls per case, matched on year and state of birth, was selected from birth certificates. Data on infant and maternal risk factors were collected from birth certificates. Prevalence was calculated by pooling the state-specific data. Conditional logistic regression was used to investigate the association between risk factors and clubfoot. RESULTS: The overall prevalence of clubfoot was 1.29 per 1000 livebirths; 1.38 among non-Hispanic whites, 1.30 among Hispanics, and 1.14 among non-Hispanic blacks or African Americans. Maternal age, parity, education, and marital status were significantly associated with clubfoot. Maternal smoking and diabetes also showed significant associations. Several of these observed associations were consistent between surveillance programs. CONCLUSIONS: We estimated the prevalence of clubfoot using data from several birth defects programs, representing one-quarter of all births in the United States. Our findings underline the importance of birth defects surveillance programs and their utility in monitoring population-based prevalence and investigating risk factors. Birth Defects Research (Part A) 85: 897-904, 2009. Ó