Violanda Grigorescu

Population-Based Analysis of Survival for Hypoplastic Left Heart Syndrome

OBJECTIVE To analyze survival patterns among infants with hypoplastic left heart syndrome (HLHS) in the State of Michigan. STUDY DESIGN Cases of HLHS prevalent at live birth were identified and confirmed within the Michigan Birth Defects Registry from 1992 to 2005 (n=406). Characteristics of infants with HLHS were compared with a 10:1 random control sample. RESULTS Compared with 4060 control subjects, the 406 cases of HLHS were more frequently male (62.6% vs 51.4%), born prematurely (<37 weeks gestation; 15.3% vs 8.7%), and born at low birth weight (LBW) (<2.5 kg; 16.0% vs 6.6%). HLHS 1-year survival rate improved over the study period (P=.041). Chromosomal abnormalities, LBW, premature birth, and living in a high poverty neighborhood were significantly associated with death. Controlling for neighborhood poverty, term infants versus preterm with HLHS or LBW were 3.2 times (95% CI: 1.9-5.3; P<.001) more likely to survive at least 1 year. Controlling for age and weight, infants from low-poverty versus high-poverty areas were 1.8 times (95% CI: 1.1-2.8; P=.015) more likely to survive at least 1 year. CONCLUSIONS Among infants with HLHS in Michigan, those who were premature, LBW, had chromosomal abnormalities, or lived in a high-poverty area were at increased risk for early death.

False-Positive Newborn Screening Result and Future Health Care Use in a State Medicaid Cohort

OBJECTIVE: To compare health care visit rates between infants with false-positive and those with normal newborn screening (NBS) results. PATIENTS AND METHODS: We analyzed administrative claims of Medicaid-enrolled infants born in Michigan in 2006 and calculated the average number of outpatient, emergency department, and hospital visits for infants aged 3 to 12 months according to NBS results. We calculated an adjusted incidence rate ratio for each visit category, adjusting for covariates and accounting for interaction effects. RESULTS: Of the 49 959 infants in the analysis, 818 had a false-positive NBS result. We noted a significant interaction between gestational age and NBS results. We found that preterm, but not term, infants with false-positive results had more acute outpatient visits than their counterparts with normal NBS results. We found no difference in adjusted rates of other visit types (emergency department, inpatient, outpatient well) between infants with false-positive and normal NBS results, regardless of gestational age. CONCLUSIONS: Increased rates of acute outpatient visits among preterm infants with false-positive NBS screening results may be attributable to underlying chronic illness or parental anxiety. The absence of increased health care utilization among term infants may be unique to this Medicaid population or a subgroup phenomenon that was not detectable in this analysis.

Transient hypothyroidism at 3-year follow-up among cases of congenital hypothyroidism detected by newborn screening.

OBJECTIVE To investigate the rate of transient thyroid deficiency and treatment compliance among cases with congenital hypothyroidism diagnosed and followed-up after age 3 years by newborn screening (NBS). STUDY DESIGN Cases detected by Michigan NBS between October 1, 2003, and December 31, 2007, and followed-up after age 3 years were included. The χ(2) and Fisher exact tests were used to test differences among followed and lost cases. Logistic regression models were used to investigate predictors of treatment cessation. RESULTS Roughly 45% of eligible cases were lost to follow-up, and disease state (transient or permanent congenital hypothyroidism) could not be determined for 12 cases (7.9%). Of the 72 followed cases, 34 (47%) were considered permanent congenital hypothyroidism based on thyroid imaging findings (n = 7) or an increase in medication dosage over time (n = 27). One-quarter of followed cases with congenital hypothyroidism were no longer being treated, and of these, just over 83% stopped treatment without medical supervision. Of 23 cases that underwent a medically supervised trial without thyroid hormone medication, treatment was reinstated in 20. Laboratory confirmation of euthyroidism was available for 6 of 18 cases clinically deemed transient. After adjustment, black race was the strongest predictor of treatment cessation (OR, 9.86; 95% CI, 1.82-53.31). Treatment cessation was also more common among low birth weight infants and those admitted to the neonatal intensive care unit at birth. CONCLUSION We recommend that NBS programs include long-term follow-up through at least age 3 years to determine treatment compliance and disease permanence. Further research is needed to determine ideal follow-up program operations and reassessment methods for congenital hypothyroidism disease permanence. Guidelines that provide evidence-based reassessment methods would be beneficial for the healthcare providers of children with congenital hypothyroidism.

Variation in immunoreactive trypsinogen concentrations among Michigan newborns and implications for cystic fibrosis newborn screening.

To investigate variation in immunoreactive trypsinogen (IRT) concentrations by race, sex, birth weight, and gestational age and their implications for the use of percentile‐based cutoffs for cystic fibrosis (CF) newborn screening (NBS) programs.

Predictors of insufficient sweat production during confirmatory testing for cystic fibrosis

Michigans Newborn Screening (NBS) Program began statewide screening for cystic fibrosis (CF) in October 2007. Confirmatory sweat testing is performed in infants having initial immunoreactive trypsinogen concentrations ≥99.8th percentile or ≥96th percentile and at least one CF mutation identified by DNA analysis. Some infants fail to produce a sufficient quantity of sweat (QNS—quantity not sufficient) to test for CF, meaning disease confirmation is delayed and sweat testing is later repeated. In this study, we evaluate predictors of QNS results. Information from the linked birth certificates and NBS diagnostic confirmation data were used. The study population was resident infants born in Michigan in 2008 who underwent a sweat test. Bivariate analyses revealed that preterm birth, low birth weight, CF care center, and race were significantly associated with QNS sweat testing results. Adjusted analyses indicated that preterm infants were 2.4 times more likely to have QNS results (95% CI 0.9, 6.4). When age at time of test, accounting for gestational age (gestational age at delivery plus postdelivery age of life = corrected age), was used in the multivariable model, infants <39 weeks were 7.4 times more likely to have QNS results (95% CI 2.5, 21.8). Waiting to sweat test until an infant is aged 39 weeks or more (corrected age) would likely reduce the rate of QNS results, thereby reducing the burden of repeat sweat testing on families and healthcare providers. Further research is necessary to understand the impact of potential delays in diagnosis/treatment relative to postponing sweat testing. Pediatr Pulmonol. 2011; 46:23–30.

Revival of a Core Public Health Function: State- and Urban-Based Maternal Death Review Processes

This article reviews some of the current challenges for maternal death review in the United States, describes key findings from an assessment of U.S. capacity for conducting maternal death reviews, and introduces a new Maternal Mortality Initiative that aims to develop standardized guidelines for state- or city-based maternal deaths review processes.

Obesity, Assisted Reproductive Technology, and Early Preterm Birth—Florida, 2004–2006

Florida resident birth certificates for 2004-2006 were linked to the Centers for Disease Control and Preventions National ART Surveillance System and were used to investigate 1) whether the association of assisted reproductive technology (ART) with preterm birth varies by prepregnancy body mass index and 2) whether the association varies by plurality. Preterm birth was defined as early preterm birth (gestation <34 weeks) and late preterm birth (gestation 34-36 weeks). Descriptive statistics and multinomial logistic regression were used to explore maternal and infant differences by ART status and plurality. Of 581,403 women included in the study, 24.0% were overweight, 18.6% were obese, 7.3% had late preterm birth, 2.6% had early preterm birth, and 0.67% conceived through ART. Among singleton births, ART was associated with increased early preterm birth risk among underweight (odds ratio (OR) = 2.94, 95% confidence interval (CI): 1.27, 6.81), overweight (OR = 1.75, 95% CI: 1.12, 2.72), and obese (OR = 2.37, 95% CI: 1.51, 3.71) women. Among twins, ART was significantly associated with increased risk among overweight (OR = 1.61, 95% CI: 1.12, 2.32) and obese (OR = 1.85, 95% CI: 1.18, 2.90) women. Differences in the associations between ART and early preterm birth by body mass index and plurality warrant further investigation.

Maternal characteristics and pregnancy outcomes after assisted reproductive technology by infertility diagnosis: ovulatory dysfunction versus tubal obstruction.

OBJECTIVE To examine differences in maternal characteristics and pregnancy outcomes between women with ovulatory dysfunction (OD) and women with tubal obstruction (TO) who underwent assisted reproductive technology (ART). DESIGN Retrospective cohort study. SETTING Centers for Disease Control and Prevention. PATIENT(S) Exposed and nonexposed groups were selected from the 2000-2006 National ART Surveillance System linked with live-birth certificates from three states: Florida, Massachusetts, and Michigan. INTERVENTION(S) None. MAIN OUTCOME MEASURE(S) Maternal characteristics and pregnancy outcomes, including newborns health status right after delivery (Apgar score, <7 vs. ≥ 7) as the study outcome of interest, were assessed among women with OD/polycystic ovary syndrome (PCOS) and TO who used ART. RESULT(S) A significantly higher prevalence of women with OD/PCOS were younger (<35 years of age; 65.7% vs. 48.9%), were white (85.4% vs. 74.4%), had higher education (29.4% vs. 15.6%), and experienced diabetes (8.8% vs. 5.3%) compared with those having TO. The odds of having a lower (<7) Apgar score at 5 minutes were almost twice as high among newborns of women with OD/PCOS compared with those with TO (crude odds ratio, 1.86; 95% confidence interval [CI], 1.31, 2.64; adjusted odds ratio, 1.90; 95% CI, 1.30, 2.77). CONCLUSION(S) Women with OD/PCOS who underwent ART have different characteristics and health issues (higher prevalence of diabetes) and infant outcomes (lower Apgar score) compared with women with TO.

Newborn screening follow-up within the lifespan context: Michigan's experience.

Experience in using various data sources for surveillance systems and studies complements the growing knowledge base regarding requirements for newborn screening follow-up, which include integration with services other than clinical subspecialties. A proposed model for utilizing state resources to develop sickle cell disease surveillance across the lifespan is presented. This surveillance process should help evaluate the burden of sickle cell disease across the lifespan, and it could be used as a model for other hemoglobinopathies as well as other newborn screening disorders. Through the continued assessment and monitoring of prevalence, comorbidities, service utilization, cost, and patient outcomes, the newborn screening follow-up program will be able to inform public health policy.

Cost of Racial Disparity in Preterm Birth: Evidence from Michigan

This study examined the economic costs associated with racial disparity in preterm birth and preterm fetal death in Michigan. Linked 2003 Michigan vital statistics and hospital discharge data were used for data analysis. Thirteen percent of the singleton births among non-Hispanic Blacks were before 37 completed weeks of gestation, compared with only 7.7% among non-Hispanic Whites (risk ratio = 1.66, 95% confidence interval: 1.59–1.72; p<.0001). One thousand one hundred and eighty four (1,184) non-Hispanic Black, singleton preterm births and preterm fetal deaths would have been avoided in 2003 had their preterm birth rate been the same as Michigan non-Hispanic Whites. Economic costs associated with these excess Black preterm births and preterm fetal deaths amounted to