Developing a public health-tracking system for follow-up of newborn screening metabolic conditions: a four-state pilot project structure and initial findings.

PURPOSE: The aim of this study was to describe the methods, cases, and initial results of a pilot project using existing public health data collection programs (birth defect surveillance or newborn screening) to conduct long-term follow-up of children with metabolic disorders. METHODS: California, Iowa, New York, and Utah expanded birth defect surveillance or newborn screening programs to collect long-term follow-up data on 19 metabolic disorders. Data elements to monitor health status and services delivered were identified, and record abstraction and data linkages were conducted. Children were followed up through to the age of 3 years. RESULTS: A total of 261 metabolic cases were diagnosed in 1,343,696 live births (19.4 cases/100,000; 95% confidence interval = 17.1-21.8). Four deaths were identified. Children with fatty acid oxidation disorders had a higher percentage of health service encounters compared with children with other disorders of at least one health service encounter (hospitalization, emergency room, metabolic clinic, genetic service provider, or social worker) except for hospitalizations; children with organic acid disorders had a higher percentage of at least one hospitalization during their third year of life than children with other disorders. CONCLUSION: Existing public health data programs can be leveraged to conduct population-based newborn screening long-term follow-up. This approach is flexible according to state needs and resources. These data will enable the states in assessing health burden, assuring access to services, and supporting policy development.

Reproductive patterns among mothers of males diagnosed with Duchenne or Becker muscular dystrophy.

Diagnosis of a child with Duchenne or Becker muscular dystrophy (DBMD) may impact future maternal reproductive choice; however, little is known about the reproductive patterns of mothers with a male child diagnosed with DBMD. Using population-based surveillance data collected by the muscular dystrophy surveillance, tracking, and research network, the proportion of mothers who conceived and delivered a live birth following the diagnosis of DBMD in an affected male child and factors associated with such reproductive choice were identified. To accomplish this, maternal demographic data were linked to birth certificate data to construct the reproductive history for 239 mothers. Univariable and bivariable analyses were conducted to determine the proportion of mothers delivering a live birth and associated factors. By the time of the current study, 96 (40.2%) of the 239 mothers had at least one live birth following delivery of their oldest affected male child; 53 (22.2%) of these mothers had a live birth before and 43 (18.0%) had a live birth after DBMD diagnosis of a male child. Mothers with a live birth after diagnosis were significantly younger at diagnosis of the oldest affected male child (26.2 ± 4.2 years vs. 31.5 ± 5.5 years), and were less likely to be white non-Hispanic compared to those with no live birth after diagnosis. These results suggest that about one in five mothers deliver a live birth subsequent to DBMD diagnosis in a male child. Maternal age and race/ethnicity were associated with this reproductive choice.

Pregnancy spacing among women delaying initiation of childbearing.

INTRODUCTION: An increasing proportion of women in the US and other countries delay initiation of childbearing until their thirties. Little is known about their subsequent pregnancies, particularly with regard to pregnancy spacing. OBJECTIVES: To determine interpregnancy interval (IPI) patterns, factors associated with IPI among women delaying initiation of childbearing until their thirties, and ascertain if delay in initiation of childbearing is associated with increased likelihood for short interpregnancy interval of less than 6 months. METHODS: A retrospective cohort study was performed using the Missouri maternal linked file for 1978-1997, inclusive. Analysis was limited to mothers aged 20-50 years at first pregnancy, having a first and second pregnancy during the study period; the sample size included 242,559 mother-infant pairs. Analysis strategies included stratified analysis, and multivariable logistic regression. Interpregnancy interval was main outcome variable, and was grouped in seven categories: 0-5, 6-11, 12-17, 18-23, 24-59, 60-119, >or=120 months. RESULTS: The mean interpregnancy interval was significantly shorter for women delaying start of childbearing (>or=30 years) compared to 20-29 year olds. Observed intervals are 31 (+/-24) months for mothers aged 20-29 years, 25 (+/-17) months for mothers aged 30-34 years, 21 (+/- 14) for 35-39 year olds, and 19 (+/-16) for 40-50 year olds (P < 0.0001). A significant trend for shorter intervals was noted as maternal age at first pregnancy increased (P < 0.0001). Factors associated with interpregnancy interval for women delaying initiation of childbearing included adverse outcome in preceding pregnancy, and low educational status. Mothers aged 35 and above at first pregnancy had increased odds for a second pregnancy following short IPI <6 months; (35-39 years OR = 1.26 95% CI 1.11-1.44; 40-50 OR = 1.91 95% CI 1.13-3.24). Mothers aged 30-34 years have lower odds for short IPI (OR = 0.93 95% CI 0.87-0.99). CONCLUSION: First time mothers aged 35 and above have higher odds of having a second pregnancy shortly after their first pregnancy. Given the increasing number of first time mothers aged 35 and above, these findings are of relevance for preconception counseling for this unique population of women.

Prevalence of Duchenne and Becker muscular dystrophies in the United States.

OBJECTIVE: To estimate prevalence of childhood-onset Duchenne and Becker muscular dystrophies (DBMD) in 6 sites in the United States by race/ethnicity and phenotype (Duchenne muscular dystrophy [DMD] or Becker muscular dystrophy [BMD]). METHODS: In 2002, the Centers for Disease Control and Prevention established the Muscular Dystrophy Surveillance, Tracking, and Research Network (MD STARnet) to conduct longitudinal, population-based surveillance and research of DBMD in the United States. Six sites conducted active, multiple-source case finding and record abstraction to identify MD STARnet cases born January 1982 to December 2011. We used cross-sectional analyses to estimate prevalence of DBMD per 10 000 boys, ages 5 to 9 years, for 4 quinquennia (1991-1995, 1996-2000, 2001-2005, and 2006-2010) and prevalence per 10 000 male individuals, ages 5 to 24 years, in 2010. Prevalence was also estimated by race/ethnicity and phenotype. RESULTS: Overall, 649 cases resided in an MD STARnet site during ≥1 quinquennia. Prevalence estimates per 10 000 boys, ages 5 to 9 years, were 1.93, 2.05, 2.04, and 1.51, respectively, for 1991-1995, 1996-2000, 2001-2005, and 2006-2010. Prevalence tended to be higher for Hispanic individuals than non-Hispanic white or black individuals, and higher for DMD than BMD. In 2010, prevalence of DBMD was 1.38 per 10 000 male individuals, ages 5 to 24 years. CONCLUSIONS: We present population-based prevalence estimates for DBMD in 6 US sites. Prevalence differed by race/ethnicity, suggesting potential cultural and socioeconomic influences in the diagnosis of DBMD. Prevalence also was higher for DMD than BMD. Continued longitudinal surveillance will permit us to examine racial/ethnic and socioeconomic differences in treatment and outcomes for MD STARnet cases.

Use of complementary and alternative medicine by males with Duchenne or Becker muscular dystrophy.

Use of complementary and alternative medicine by males with Duchenne or Becker muscular dystrophy was examined using interview reports from caregivers enrolled in the population-based Muscular Dystrophy Surveillance, Tracking, and Research Network. Of the 200 caregivers interviewed, 160 (80%) reported "ever" using complementary and alternative medicine for their affected children. Mind-body medicine (61.5%) was most frequently used, followed by biologically based practices (48.0%), manipulative and body-based practices (29.0%), and whole medical systems (8.5%). Caregivers reporting use of whole medical systems had higher education and income levels compared with nonusers; affected males had shorter disease duration. Caregivers reporting use of mind-body medicine, excluding aquatherapy, had higher education level compared with nonusers. Overall, complementary and alternative medicine use was high; disease duration, education, and income levels influenced use. These findings have implications for developing clinical care protocols and monitoring possible interactions between complementary and alternative medicine and conventional medical therapies.

Younger maternal age (at initiation of childbearing) and recurrent perinatal mortality.

OBJECTIVE: To assess whether young maternal age at initiation of childbearing is associated with recurrence of perinatal mortality (PM), as well as its components: stillbirth and neonatal death. STUDY DESIGN: We conducted a population-based, retrospective cohort study on the Missouri maternally linked longitudinal data files comprising adolescent (10-19 years; n = 73,533) or mature (20-24 years; n = 78,618) mothers in their first pregnancy with follow-up in their second pregnancy to document the occurrence of PM or its components. The study covered the period 1989-2005. We used unconditional logistic regression modeling to generate odds ratios and to control for confounding. RESULTS: A history of perinatal mortality, stillbirth, or neonatal mortality increased the risk of a recurrence by 4-5 times. Among women with a history of PM or stillbirth in the first pregnancy, maternal age at initiation of pregnancy was not a risk factor for subsequent PM or its components. However, adolescent mothers with a history of neonatal mortality in the first pregnancy were about 5 times as likely to experience stillbirth in the second pregnancy, as compared to their mature counterparts. CONCLUSIONS: Young maternal age at the initiation of childbearing is not associated with an overall increased risk of recurrent perinatal loss. However, prior history of neonatal mortality among teen mothers is strongly predictive of subsequent stillbirth.

Racial disparities in perinatal outcomes and pregnancy spacing among women delaying initiation of childbearing.

INTRODUCTION: Reducing racial/ethnic disparities is a key objective of the Healthy People 2010 initiative. Unfortunately, racial disparities among women delaying initiation of childbearing have received limited attention. As more women in the US are delaying initiation of childbearing, it is important to examine racial disparities in reproductive health outcomes for this subgroup of women. OBJECTIVE: To examine racial disparities in perinatal outcomes, interpregnancy interval, and to assess the risk for adverse outcomes in subsequent pregnancy for women delaying initiation of childbearing until age 30 or older compared to those initiating childbearing at age 20-29. METHODS: We conducted a retrospective cohort study using the Missouri maternally linked cohort files 1978-1997. Final study sample included 239,930 singleton sibling pairs (Whites and African Americans). Outcome variables included first and second pregnancy outcomes (fetal death, low birth weight, preterm delivery and small-for-gestational age) and interpregnancy interval between first and second pregnancy. Independent variables included maternal age at first pregnancy and race. Analysis strategies used involved stratified analyses and multivariable unconditional logistic regression; interactions between maternal race, age and interpregnancy interval were examined in the regression models. RESULTS: Compared to Whites, African American mothers initiating childbearing at age 30 or older had significantly higher rates of adverse outcomes in the first and second pregnancy (P < 0.0001). Generally, African Americans had significantly higher rates of second pregnancy following intervals <6 months compared to Whites; however, no significant racial differences were noted in interpregnancy interval distribution pattern after controlling for maternal age at first pregnancy. African Americans delaying initiation of childbearing had significantly higher risk for adverse perinatal outcomes in the second pregnancy compared to Whites after controlling for potential confounders, however there were no significant interactions between maternal age at first pregnancy, race and short interpregnancy interval. CONCLUSION: Although African Americans were less likely to delay initiation of childbearing than were White women, their risk for adverse perinatal outcomes was much greater. As health care providers strive to address racial disparities in birth outcomes, there is need to pay attention to this unique group of women as their population continues to increase.

Interpregnancy interval and subsequent perinatal outcomes among women delaying initiation of childbearing.

AIM: While delayed initiation of childbearing is associated with adverse perinatal outcomes, whether or not risk persists and whether interpregnancy interval (IPI) affects the subsequent pregnancy remains unclear. OBJECTIVES: To examine second-pregnancy perinatal outcomes for women initiating childbearing age > or = 30 compared to those initiating childbearing aged 20-29, specifically examining the distribution of adverse perinatal outcomes, and their associations with the interpregnancy interval. METHODS: Retrospective cohort study using the Missouri maternally linked files 1978-1997. Perinatal outcomes included fetal death, low birthweight, preterm birth and small-for-gestational age. Predictor variables included maternal age at first pregnancy and IPI between the first and second pregnancy. RESULTS: With an increasing maternal age at first pregnancy, rates of very low birthweight (P = 0.0095), preterm delivery (P = 0.0126), moderately preterm (P = 0.0458), and extremely preterm (P = 0.0008) in the second pregnancy increased, while the rate of small-for-gestational age (P < 0.0001) declined. Interpregnancy intervals <6 and > or = 60 months were associated with a higher rate of adverse outcomes after controlling for maternal age at first pregnancy. Intervals of 12-17 months had the lowest rate of adverse outcomes for mothers 35+. Maternal age > or = 35 years at first pregnancy and IPI <6 months were independent risk factors for an adverse outcome in the second pregnancy, however no statistical interaction between these factors was observed. CONCLUSION: Delayed initiation of childbearing is associated with a persistent risk of adverse perinatal outcomes in the second pregnancy, with a short IPI contributing to this risk. As numbers of women delaying childbearing beyond age 30 increase, providers should consider these risks in counseling women about their reproductive plans.

Use of postpartum health services in rural Uganda: knowledge, attitudes, and barriers.

The purpose of this study was to explore the knowledge, attitudes and barriers to use of postpartum care service among rural communities in Uganda. Study was a part of a larger reproductive health evaluation project, and was cross-sectional in nature utilizing qualitative research methods using the narrative inquiry. Two matched rural communities were used in this study; Semuto in Luwero district, and Lwamaggwa in Rakai district. Fifty key informants who were purposefully selected from each study site were interviewed. They included community leaders, political leaders, health care providers, women leaders and community members. One-on-one interviews were conducted with key community informants using an interview guide. The purpose of the interview was explained to each participant, and written informed consent was obtained before the start of the interview. Respondents were allowed to express their views, opinions and observations on several health issues including postpartum health care services. There was a low level of knowledge about postpartum care services among the respondents of the two communities. There was lack of awareness about postpartum care and it's benefits. The main barriers to use of services were; misconceptions regarding the importance of postpartum care, distance to health facilities, poverty, and health system factors notably; poor facilities, lack of essential drugs, and poor attitudes of health workers. In the effort to improve reproductive health care services, there is an urgent need to improve postpartum services, and make them more accessible and user friendly. The training of providers at all levels is essential, in addition to educating families on the importance of postpartum care services.