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Objectives: To measure the rates of thyroid gland imaging and levothyroxine (L-T4) discontinuation and to assess whether discontinuation was monitored with thyroid-stimulating hormone testing in subjects with congenital hypothyroidism.Study Design: This is a retrospective analysis of claims data from the IBM MarketScan Databases for children born between 2010 and 2016 and continuously enrolled in a noncapitated employer-sponsored private health insurance plan or in Medicaid for ≥36 months from the date of the first filled L-T4 prescription.Results: There were 263 privately insured and 241 Medicaid-enrolled children who met the inclusion criteria. More privately insured than Medicaid-enrolled children had imaging between the first filled prescription and 180 days after the last filled prescription (24.3% vs 12.9%; P = .001). By 36 months, 35.7% discontinued L-T4, with no difference by insurance status (P = .48). Among those who discontinued, 29.1% of privately insured children and 47.7% of Medicaid-enrolled children had no claims for thyroid-stimulating hormone testing within the next 180 days (P = .01).Conclusions: Nearly one-third of children with suspected congenital hypothyroidism discontinued L-T4 by 3 years and fewer Medicaid-enrolled than privately insured children received timely follow-up thyroid-stimulating hormone testing. Future studies are indicated to understand the quality of care and developmental outcomes for children with congenital hypothyroidism and barriers to guideline adherence in evaluating for transient congenital hypothyroidism. [ABSTRACT FROM AUTHOR]

Objective: To evaluate the expected cost-effectiveness and net benefit of the recent implementation of newborn screening (NBS) for severe combined immunodeficiency (SCID) in Washington State.Study Design: We constructed a decision analysis model to estimate the costs and benefits of NBS in an annual birth cohort of 86 600 infants based on projections of avoided infant deaths. Point estimates and ranges for input variables, including the birth prevalence of SCID, proportion detected asymptomatically without screening through family history, screening test characteristics, survival rates, and costs of screening, diagnosis, and treatment were derived from published estimates, expert opinion, and the Washington NBS program. We estimated treatment costs stratified by age of identification and SCID type (with or without adenosine deaminase deficiency). Economic benefit was estimated using values of $4.2 and $9.0 million per death averted. We performed sensitivity analyses to evaluate the influence of key variables on the incremental cost-effectiveness ratio (ICER) of net direct cost per life-year saved.Results: Our model predicts an additional 1.19 newborn infants with SCID detected preclinically through screening, in addition to those who would have been detected early through family history, and 0.40 deaths averted annually. Our base-case model suggests an ICER of $35 311 per life-year saved, and a benefit-cost ratio of either 5.31 or 2.71. Sensitivity analyses found ICER values <$100 000 and positive net benefit for plausible assumptions on all variables.Conclusions: Our model suggests that NBS for SCID in Washington is likely to be cost-effective and to show positive net economic benefit. [ABSTRACT FROM AUTHOR]

Venous thromboembolism (VTE), which includes deep vein thrombosis and pulmonary embolism, is an important cause of preventable mortality and morbidity. In this study, we summarize estimates of per-patient and aggregate medical costs or expenditures attributable to incident VTE in the United States. Per-patient estimates of incremental costs can be calculated as the difference in costs between patients with and without an event after controlling for differences in underlying health status. We identified estimates of the incremental per-patient costs of acute VTEs and VTE-related complications, including recurrent VTE, post-thrombotic syndrome, chronic thromboembolic pulmonary hypertension, and anticoagulation-related adverse drug events. Based on the studies identified, treatment of an acute VTE on average appears to be associated with incremental direct medical costs of $12,000 to $15,000 (2014 US dollars) among first-year survivors, controlling for risk factors. Subsequent complications are conservatively estimated to increase cumulative costs to $18,000-23,000 per incident case. Annual incident VTE events conservatively cost the US healthcare system $7-10 billion each year for 375,000 to 425,000 newly diagnosed, medically treated incident VTE cases. Future studies should track long-term costs for cohorts of people with incident VTE, control for comorbid conditions that have been shown to be associated with VTE, and estimate incremental medical costs for people with VTE who do not survive. The costs associated with treating VTE can be used to assess the potential economic benefit and cost-savings from prevention efforts, although costs will vary among different patient groups. [ABSTRACT FROM AUTHOR]

Objective: To assess the cost-effectiveness of a pilot newborn screening (NBS) and treatment program for sickle cell anemia (SCA) in Luanda, Angola.Study Design: In July 2011, a pilot NBS and treatment program was implemented in Luanda, Angola. Infants identified with SCA were enrolled in a specialized SCA clinic in which they received preventive care and sickle cell education. In this analysis, the World Health Organization (WHO) and generalized cost-effectiveness analysis methods were used to estimate gross intervention costs of the NBS and treatment program. To determine healthy life-years (HLYs) gained by screening and treatment, we assumed NBS reduced mortality to that of the Angolan population during the first 5 years based upon WHO and Global Burden of Diseases Study 2010 estimates, but provided no significant survival benefit for children who survive through age 5 years. A secondary sensitivity analysis with more conservative estimates of mortality benefits also was performed. The costs of downstream medical costs, including acute care, were not included.Results: Based upon the costs of screening 36,453 infants and treating the 236 infants with SCA followed after NBS in the pilot project, NBS and treatment program is projected to result in the gain of 452-1105 HLYs, depending upon the discounting rate and survival assumptions used. The corresponding estimated cost per HLY gained is $1380-$3565, less than the gross domestic product per capita in Angola.Conclusions: These data demonstrate that NBS and treatment for SCA appear to be highly cost-effective across all scenarios for Angola by the WHO criteria. [ABSTRACT FROM AUTHOR]

Background There are limitations to using administrative data to identify postoperative venous thromboembolism (VTE). We used a novel approach to quantify postoperative VTE events among Department of Veterans Affairs (VA) surgical patients during 2005-2010. Methods We used VA administrative data to exclude patients with VTE during 12 months prior to surgery. We identified probable postoperative VTE events within 30 and 90 days post-surgery in three settings: 1) pre-discharge inpatient, using a VTE diagnosis code and a pharmacy record for anticoagulation; 2) post-discharge inpatient, using a VTE diagnosis code followed by a pharmacy record for anticoagulation within 7 days; and 3) outpatient, using a VTE diagnosis code and either anticoagulation or a therapeutic procedure code with natural language processing (NLP) to confirm acute VTE in clinical notes. Results Among 468,515 surgeries without prior VTE, probable VTEs were documented within 30 and 90 days in 3,931 (0.8%) and 5,904 (1.3%), respectively. Of probable VTEs within 30 or 90 days post-surgery, 47.8% and 62.9%, respectively, were diagnosed post-discharge. Among post-discharge VTE diagnoses, 86% resulted in a VA hospital readmission. Fewer than 25% of outpatient records with both VTE diagnoses and anticoagulation prescriptions were confirmed by NLP as acute VTE events. Conclusion More than half of postoperative VTE events were diagnosed post-discharge; analyses of surgical discharge records are inadequate to identify postoperative VTE. The NLP results demonstrate that the combination of VTE diagnoses and anticoagulation prescriptions in outpatient administrative records cannot be used to validly identify postoperative VTE events. Highlights • ~ 40% of possible pre-discharge VTEs based on ICD-9 codes classified as not probable. • NLP was used on outpatient clinical notes to assess VTEs with no VA readmission. • Just 21-23% of possible outpatient VTE diagnosed classified as probable based on NLP. • Probable VTEs < 90 days of surgery in 1.3% of VA patients, majority after discharge. [ABSTRACT FROM AUTHOR]

From 2009-2015 to 2016-2019, the proportion of infants in the US with congenital cytomegalovirus treated with valganciclovir roughly doubled for infants enrolled with employer-sponsored insurance (from 16% to 29%) and Medicaid (from 16% to 36%). The proportion treated with valganciclovir increased for all congenital cytomegalovirus disease severity groups. [ABSTRACT FROM AUTHOR]

Highlights: [•] A sample of children with FXS were matched with children with autism spectrum disorder (ASD) only, or intellectual disabilities (ID) only, or ASD and ID. [•] Higher percentage of caregivers of FXS reported negative financial and employment impacts than caregivers of ASD or ID only. [•] Caregivers of FXS reported similar financial and employment impacts as caregivers of both ASD and ID. [•] Co-occurring anxiety or seizures, limits in overall ability, and more irritability were associated with negative caregiver impacts. [Copyright &y& Elsevier]

Abstract: Background: The preventive health care needs of people with disabilities often go unmet, resulting in medical complications that may require hospitalization. Such complications could be due, in part, to difficulty accessing care or the quality of ambulatory care services received. Objective: To use hospitalizations for urinary tract infections (UTIs) as a marker of the potential quality of ambulatory care services received by people affected by spina bifida. Methods: MarketScan inpatient and outpatient medical claims data for 2000 through 2003 were used to identify hospitalizations for UTI, which is an ambulatory care sensitive condition, for people affected by spina bifida and to calculate inpatient discharge rates, average lengths of stay, and average medical care expenditures for such hospitalizations. Results: People affected by spina bifida averaged 0.5 hospitalizations per year, and there were 22.8 inpatient admissions with UTI per 1000 persons with spina bifida during the period 2000–2003, in comparison to an average of 0.44 admission with UTI per 1000 persons for those without spina bifida. If the number of UTI hospitalizations among people affected by spina bifida were reduced by 50%, expenditures could be reduced by $4.4 million per 1000 patients. Conclusions: Consensus on the evaluation and management of bacteriuria could enhance clinical care and reduce the disparity in UTI discharge rates among people affected by spina bifida compared to those without spina bifida. National evidence-based guidelines are needed. [Copyright &y& Elsevier]

Objective: To analyze trends in sickle cell disease (SCD)-related mortality among black children during 1983-2002. Study design: Using the multiple-cause mortality files compiled by the Centers for Disease Control and Prevention''s National Center for Health Statistics, we analyzed deaths among children classified as black who were age 14 years and younger and had SCD identified on their death certificates. Results: Relative to the rate for 1983-1986, the SCD mortality rate for 1999-2002 decreased by 68% (95% confidence interval [CI] = 58% to 75%) at age 0 to 3 years, by 39% (95% CI = 16% to 56%) at age 4 to 9 years, and by 24% (95% CI = −9% to 47%) at age 10 to 14 years. For the most recent period studied, a significant (42%) reduction in mortality at age 0 to 3 years was seen between 1995-1998 and 1999-2002, with essentially no reduction in SCD mortality at older ages. Conclusions: Recent decreases in SCD mortality in black children under age 4 years coincided with the introduction of the 7-valent pneumococcal conjugate vaccine in 2000, although temporal association is not evidence of causation. The lack of significant recent reduction in SCD mortality in older children indicates the need for new treatment approaches. [Copyright &y& Elsevier]

Background: Women with a pregnancy affected by a neural tube defect (NTD) are encouraged to take folic acid prior to a subsequent pregnancy, but it is unknown whether organized attempts to identify and counsel such women to prevent recurrent NTDs are cost effective. Methods: Data from the South Carolina recurrence–prevention program for October 2001–September 2002 were analyzed between October 2002 and December 2003 to calculate costs. Cost-effectiveness modeling of the program during 1992–2006 was conducted during 2007. Results were calculated for three scenarios based on recurrence risk, supplement use, and the effectiveness of folic acid in preventing recurrences. For each scenario, quality-adjusted life years (QALYs) were calculated separately using prevented NTD-affected live births; prevented NTD-affected births (including fetal deaths); and all prevented NTD-affected pregnancies. Results: The prevention program cost approximately $155,000 per year in 2003 dollars to protect 35 pregnancies and prevent approximately one NTD. The direct costs associated with an NTD depend on type and outcome, but are approximately $560,000 in 2003 dollars for a live birth with spina bifida. The base-case cost-effectiveness ratio was $39,600 per QALY gained from avoided NTD-affected live births and stillbirths, and $14,700 per QALY gained from the avoidance of all NTD-affected pregnancies. The baseline NTD recurrence risk and the use of folic acid supplements by women who are at high risk for an NTD-affected pregnancy were influential parameters. Conclusions: The South Carolina NTD recurrence–prevention program appears comparable in cost effectiveness to other preventive services. Other states might consider including NTD recurrence prevention in birth defect–prevention programs. [Copyright &y& Elsevier]

Objective: To examine whether symptoms of attention-deficit/hyperactivity disorder (ADHD) during childhood are associated with child maltreatment. Study design: The study sample consisted of 14 322 participants in the National Longitudinal Study of Adolescent Health. We used logistic regression and propensity score matching to examine the relationship, adjusting for demographic, socioeconomic, and familial risk factors for child maltreatment. Results: Inattentive type, by criteria of Diagnostic and Statistical Manual of Mental Disorders Fourth Edition, was significantly associated with the likelihood of supervision neglect (odds ratio [OR]: 1.6; 95% confidence interval [CI]: 1.2-2.2), physical neglect (OR: 2.1; 95% CI: 1.4-3.1), physical abuse (OR: 1.6; 95% CI: 1.1-2.3), and contact sexual abuse (OR: 2.6; 95% CI: 1.5-4.5). To a lesser extent, hyperactive type was associated with the likelihood of supervision neglect (OR: 1.5; 95% CI: 1.1-2.0) and physical abuse (OR: 1.3; 95% CI: 1.0-1.6). The association between hyperactive type and physical neglect or contact sexual abuse was not significant. The results from the propensity score matching were similar. Conclusions: Childhood ADHD symptoms were associated with self-reported child maltreatment. Health care providers should be alert to the potential for child maltreatment among children with ADHD symptoms, especially those with inattentive symptoms. [Copyright &y& Elsevier]

Objective: To use health care insurance claims data from a privately insured population to estimate health care use and expenditures for infants and children aged 0 to 4 years with Down syndrome. Study design: Data from the 2004 Medstat MarketScan database were used to estimate medical care use and expenditures related to inpatient admissions, outpatient services, and prescription drug claims for children with and those without Down syndrome. Costs were further stratified by the presence or absence of a congenital heart defect (CHD). Results: The mean medical costs for infants and children with Down syndrome were $36384 during 2004; median medical costs were $11164. Mean and median medical costs for children 0 to 4 years of age with Down syndrome were 12 to 13 times higher than for children without Down syndrome. For infants with Down syndrome and CHDs, mean and median costs were 5 to 7 times higher than for infants with Down syndrome who did not have CHDs. Conclusions: These findings may facilitate future assessments of the effect of the Down syndrome on the health care system. [Copyright &y& Elsevier]

Objective: To estimate the population impact of child mortality as a result of cystic fibrosis (CF) potentially preventable by newborn screening. Study design: A systematic literature review of mortality in children with classic CF without meconium ileus (MI) in screened and unscreened cohorts was extended by contacting investigators for unpublished data. In addition, survival in US states with and without newborn screening (NBS) programs for CF was compared using data from the Cystic Fibrosis Foundation Patient Registry (CFFPR). Results: Among non-US studies, CF-related mortality risk to approximately 10 years of age was lower by 5 to 10 per 100 in screened cohorts. Unpublished US data from a trial of NBS for CF indicate no CF-related deaths to 10 years of age in either cohort. CFFPR data suggest improved survival among children with CF born in US states with NBS, with a CF-related mortality difference to 10 years of age between the screened and unscreened groups between 1.5 and 2 per 100 children with CF without MI. Conclusion: In addition to improving nutritional outcomes, newborn screening for CF may result in improved child survival. The absolute differential in mortality risk, although modest in size, appears comparable to NBS for certain other genetic disorders. [Copyright &y& Elsevier]

Objective: This is a critical review of published economic analyses on congenital cytomegalovirus infection and strategies for its detection and prevention. Findings: The review identified four cost-of-illness studies and nine cost-effectiveness analyses: three of vaccination of young women, two of prenatal screening, and four of newborn screening. All reported either large economic costs or favorable cost-effectiveness of interventions. However, sensitivity analyses did not address some of the most critical assumptions. Conclusions: Reviewed economic analyses overattributed certain adverse long-term outcomes to congenital cytomegalovirus infection, while other long-term costs were not included. Overall, limited conceptual frameworks, unrepresentative data sources, and unsupported or inadequately documented assumptions regarding outcomes and costs hinder the ability of policymakers to draw conclusions. A major challenge is the limited information on long-term outcomes and costs for representative cohorts of individuals with congenital cytomegalovirus, which further research could helpfully address. [ABSTRACT FROM AUTHOR]

The societal cost of preterm birth indicates potential economic gains from interventions that reduce the incidence of preterm birth. Changes in the epidemiology of preterm birth and healthcare costs require periodic updates to cost estimates. Previously reported incremental cost estimates for the United States in 2004 were updated. The discounted present value of the excess cost associated with prematurity for the 2016 US birth cohort was estimated to be $25.2 billion: $17.1 billion for medical care of persons born preterm, $2.0 billion for delivery care, $1.3 billion for early intervention and special education, and $4.8 billion in lost productivity due to associated disabilities in adults. The nominal and inflation-adjusted incremental costs per preterm birth increased by 26% and 4%, respectively, during 2004–2016. The aggregate cost decreased by 4%, associated with declines in overall births and the preterm birth rate and changes in the distribution by gestational age. [ABSTRACT FROM AUTHOR]

Abstract: This paper calculates the medical expenditures for pediatric Medicaid enrollees with fetal alcohol syndrome (FAS), those with and those without reported intellectual disability (ID). The pediatric portion of the MarketScan® Medicaid Multi-State databases for the years 2003–2005 was used. Children with FAS were identified based on International Classification of Diseases, Ninth Revision, Clinical Modification codes. Children without FAS formed the comparison group. Annual mean, median, and 95th percentile total expenditures were calculated for those continuously enrolled during 2005. Children with FAS incurred annual mean medical expenditures that were nine times as high as those of children without FAS during 2005 ($16,782 vs. $1,859). ID more commonly was listed as a medical diagnosis among children with FAS than among children in the comparison group (12% vs. 0.5%), and mean expenditures of children with FAS and ID were 2.8 times those of children with FAS but without reported ID. Children with FAS incurred higher medical expenditures compared with children without FAS. A subset of children with FAS who had ID sufficiently serious to be recorded in medical records increased those expenditures still further. Our estimate of mean expenditures for children with FAS was several times higher than previous estimates in the United States. [Copyright &y& Elsevier]

Cytomegalovirus (CMV) is the most common infectious cause of congenital malformation and a leading cause of developmental disabilities such as sensorineural hearing loss (SNHL), motor and cognitive deficits. The significant disease burden from congenital CMV infection (cCMV) led the US National Institute of Medicine to rank CMV vaccine development as the highest priority. An average of 6.7/1000 live births are affected by cCMV, but the prevalence varies across and within countries. In contrast to other congenital infections such as rubella and toxoplasmosis, the prevalence of cCMV increases with CMV seroprevalence rates in the population. The true global burden of cCMV disease is likely underestimated because most infected infants (85–90 %) have asymptomatic infection and are not identified. However, about 7–11 % of those with asymptomatic infection will develop SNHL throughout early childhood. Although no licensed CMV vaccine exists, several candidate vaccines are in development, including one currently in phase 3 trials. Licensure of one or more vaccine candidates is feasible within the next five years. Various models of CMV vaccine strategies employing different target populations have shown to provide substantial benefit in reducing cCMV. Although CMV can cause end-organ disease with significant morbidity and mortality in immunocompromised individuals, the focus of this vaccine value profile (VVP) is on preventing or reducing the cCMV disease burden. This CMV VVP provides a high-level, comprehensive assessment of the currently available data to inform the potential public health, economic, and societal value of CMV vaccines. The CMV VVP was developed by a working group of subject matter experts from academia, public health groups, policy organizations, and non-profit organizations. All contributors have extensive expertise on various elements of the CMV VVP and have described the state of knowledge and identified the current gaps. The VVP was developed using only existing and publicly available information. [ABSTRACT FROM AUTHOR]

Introduction: Although fortification of food with folic acid has been calculated to be cost saving in the U.S., updated estimates are needed. This analysis calculates new estimates from the societal perspective of net cost savings per year associated with mandatory folic acid fortification of enriched cereal grain products in the U.S. that was implemented during 1997-1998.Methods: Estimates of annual numbers of live-born spina bifida cases in 1995-1996 relative to 1999-2011 based on birth defects surveillance data were combined during 2015 with published estimates of the present value of lifetime direct costs updated in 2014 U.S. dollars for a live-born infant with spina bifida to estimate avoided direct costs and net cost savings.Results: The fortification mandate is estimated to have reduced the annual number of U.S. live-born spina bifida cases by 767, with a lower-bound estimate of 614. The present value of mean direct lifetime cost per infant with spina bifida is estimated to be $791,900, or $577,000 excluding caregiving costs. Using a best estimate of numbers of avoided live-born spina bifida cases, fortification is estimated to reduce the present value of total direct costs for each year's birth cohort by $603 million more than the cost of fortification. A lower-bound estimate of cost savings using conservative assumptions, including the upper-bound estimate of fortification cost, is $299 million.Conclusions: The estimates of cost savings are larger than previously reported, even using conservative assumptions. The analysis can also inform assessments of folic acid fortification in other countries. [ABSTRACT FROM AUTHOR]

Objective Preconception care for women with diabetes can reduce the occurrence of adverse birth outcomes. We aimed to estimate the preconception care (PCC)–preventable health and cost burden of adverse birth outcomes associated with diagnosed and undiagnosed pregestational diabetes mellitus (PGDM) in the United States. Study Design Among women of reproductive age (15-44 years), we estimated age- and race/ethnicity-specific prevalence of diagnosed and undiagnosed diabetes. We applied age and race/ethnicity-specific pregnancy rates, estimates of the risk reduction from PCC for 3 adverse birth outcomes (preterm birth, major birth defects, and perinatal mortality), and lifetime medical and lost productivity costs for children with those outcomes. Using a probabilistic model, we estimated the reduction in adverse birth outcomes and costs associated with universal PCC compared with no PCC among women with PGDM. We did not assess maternal outcomes and associated costs. Results We estimated 2.2% of US births are to women with PGDM. Among women with diagnosed diabetes, universal PCC might avert 8397 (90% prediction interval [PI], 5252-11,449) preterm deliveries, 3725 (90% PI, 3259-4126) birth defects, and 1872 (90% PI, 1239-2415) perinatal deaths annually. Associated discounted lifetime costs averted for the affected cohort of children could be as high as $4.3 billion (90% PI, 3.4-5.1 billion) (2012 US dollars). PCC among women with undiagnosed diabetes could yield an additional $1.2 billion (90% PI, 951 million-1.4 billion) in averted cost. Conclusion Results suggest a substantial health and cost burden associated with PGDM that could be prevented by universal PCC, which might offset the cost of providing such care. [ABSTRACT FROM AUTHOR]

Context: Young people with blood disorders face challenges in maintaining their physical health as they age. Sickle cell disease has well-documented complications in various organ systems. Increasingly, professionals, consumers, and advocates involved in blood disorders are concerned about the cumulative and ongoing effect of organ-specific complications on function and participation. Evidence acquisition: Publications were identified that looked at the relationship between sickle cell disease and associated impairments and restrictions in participation as defined by the International Classification of Function, Disability, and Health (ICF). Evidence synthesis: This article organizes a literature review in PubMed using ICF terms that define functional limitations and participation restrictions in sickle cell disease. Conclusions: Individuals with sickle cell disease experience complications in multiple organ systems that affect related functions and, consequently, participation in community living. The effects begin early in childhood and accumulate across the life course into adulthood. Intervention research is needed to understand how contextual factors can promote optimal function and participation in the face of mounting impairments. [ABSTRACT FROM AUTHOR]

Abstract: Deep vein thrombosis (DVT) and pulmonary embolism (PE), collectively known as venous thromboembolism (VTE), are an important and growing public health issue, associated with considerable morbidity and mortality. Presently, there is no national surveillance for DVT and PE. This article provides a summary of an expert workgroup meeting convened January 12, 2010, by the CDC. The purpose of the meeting was to inform CDC on the development of U.S. population-based public health surveillance activities for DVT/PE. Topics discussed included: (1) stakeholders, needs, gaps, and target populations; (2) requirements of surveillance systems; (3) challenges, limitations, and potential barriers to implementation of surveillance activities; and (4) integration of research and education with surveillance activities. [Copyright &y& Elsevier]

Abstract: Sickle cell disease (SCD) is common throughout much of sub-Saharan Africa, affecting up to 3% of births in some parts of the continent. Nevertheless, it remains a low priority for many health ministries. The most common form of SCD is caused by homozygosity for the β-globin S gene mutation (SS disease). It is widely believed that this condition is associated with very high child mortality, but reliable contemporary data are lacking. We have reviewed available African data on mortality associated with SS disease from published and unpublished sources, with an emphasis on two types of studies: cross-sectional population surveys and cohort studies. We have concluded that, although current data are inadequate to support definitive statements, they are consistent with an early-life mortality of 50%–90% among children born in Africa with SS disease. Inclusion of SCD interventions in child survival policies and programs in Africa could benefit from more precise estimates of numbers of deaths among children with SCD. A simple, representative, and affordable approach to estimate SCD child mortality is to test blood specimens already collected through large population surveys targeting conditions such as HIV, malaria, and malnutrition, and covering children of varying ages. Thus, although there is enough evidence to justify investments in screening, prophylaxis, and treatment for African children with SCD, better data are needed to estimate the numbers of child deaths preventable by such interventions and their cost effectiveness. [Copyright &y& Elsevier]

Context: Large administrative healthcare data sets are an important source of data for health services research on sickle cell disease (SCD) and thalassemia. This paper identifies and describes major U.S. healthcare administrative databases and their use in published health services research on hemoglobinopathies. Evidence acquisition: Publications that used U.S. administrative healthcare data sets to assess healthcare use or expenditures were identified through PubMed searches using key words for SCD and either costs, expenditures, or hospital discharges; no additional articles were identified by using thalassemia as a key word. Additional articles were identified through manual searches of related articles or reference lists. Evidence synthesis: A total of 26 original health services research articles were identified. The types of administrative data used for health services research on hemoglobinopathies included federal- and state-specific hospital discharge data sets and public and private health insurance claims databases. Gaps in recent health services research on hemoglobin disorders included a paucity of research related to thalassemia, few studies of adults with hemoglobinopathies, and few studies focusing on emergency department or outpatient clinic use. Conclusions: Administrative data sets provide a unique means to study healthcare use among people with SCD or thalassemia because of the ability to examine large sample sizes at fairly low cost, resulting in greater generalizability than is the case with clinic-based data. Limitations of administrative data in general include potential misclassification, under-reporting, and lack of sociodemographic information. [Copyright &y& Elsevier]

Abstract: Background: Congenital cytomegalovirus (CMV) infection is a leading cause of sensorineural hearing loss (SNHL) and developmental disability in children. Early identification of infected children through screening could allow for early intervention and improvement in functional outcomes among the subset who develop sequelae. Objectives: To outline potential options and strategies for screening newborns for congenital CMV infection and to discuss barriers to screening and data needs to inform future policy decisions. Study design: Commentary based on the literature and expert opinion on newborn dried blood spot screening, newborn hearing screening/Early Hearing Detection and Intervention (EHDI) programs, and congenital CMV. Results: Although no population-based screening for congenital CMV is underway, pilot newborn screening studies using a variety of assays with urine or dried blood spot specimens are underway. Challenges to screening are both practical—uncertain sensitivity of blood spot assays suitable for large-scale screening and lack of infrastructure for collection of urine specimens; and evidentiary—the need to demonstrate improved outcomes and value of screening to offset the expense and potential adverse psychosocial consequences for children and families whose children require periodic monitoring but never develop sequelae. Conclusions: Screening for congenital CMV infection is a potentially important intervention that merits additional research, including the logistical feasibility of different screening options and psychosocial consequences for families. [Copyright &y& Elsevier]

Objective This study assessed the risk of venous thromboembolism (VTE) among privately insured adults in the U.S. with one or more of the following autoimmune diseases: autoimmune hemolytic anemia (AIHA), immune thrombocytopenic purpura (ITP), rheumatoid arthritis (RA), and systemic lupus erythematosus (SLE). Materials and Methods Using the Truven Health MarketScan® Databases, patients 18-64 years of age with a diagnosis of AIHA, ITP, RA, or SLE in 2007 and a sex and age-group matched comparison group of enrollees were followed up through 2010 to identify VTE events. Survival curve and Cox proportional hazards analyses were conducted to assess differences between groups. Results Among patients with AIHA, ITP, RA, or SLE, or >1 of these diseases, the risk of at least one VTE event was 19.74, 7.72, 4.90, 9.89, and 13.35 per 1,000 person-years, respectively; among the comparison group, the risk was 1.91 per 1,000 person-years. The adjusted hazard ratios (aHRs) for VTE among patients with AIHA, ITP, RA, or SLE, or >1 of these diseases (when compared with the comparison group) tended to decline over follow-up time; at 1 year, the aHRs were 6.30 (95% confidence interval [CI]: 4.44-8.94), 2.95 (95% CI: 2.18-4.00), 2.13 (95% CI: 1.89-2.40), 4.68 (95% CI: 4.10-5.33), and 5.11 (95% CI: 4.26-6.14), respectively. Conclusion Having AIHA, ITP, RA, or SLE, or >1 of these diseases was associated with an increased likelihood of a VTE event. More research is necessary to develop better understanding of VTE occurrence among people with autoimmune diseases. [ABSTRACT FROM AUTHOR]

Objective: During the 2016-2017 Zika virus (ZIKV) outbreak, the prevention of unintended pregnancies was identified as a primary strategy to prevent birth defects. This study estimated the cost-effectiveness of the Zika Contraception Access Network (Z-CAN), an emergency response intervention that provided women in Puerto Rico with access to the full range of reversible contraception at no cost and compared results with a preimplementation hypothetical cost-effectiveness analysis (CEA).Study Design: We evaluated costs and outcomes of Z-CAN from a health sector perspective compared to no intervention using a decision tree model. Number of people served, contraception methods mix, and costs under Z-CAN were from actual program data and other input parameters were from the literature. Health outcome measures included the number of Zika-associated microcephaly (ZAM) cases and unintended pregnancies. The economic benefits of the Z-CAN intervention were ZIKV-associated direct costs avoided, including lifetime medical and supportive costs associated with ZAM cases, costs of monitoring ZIKV-exposed pregnancies and infants born from Zika-virus infected mothers, and the costs of unintended pregnancies prevented during the outbreak as a result of increased contraception use through the Z-CAN intervention.Results: The Z-CAN intervention cost a total of $26.1 million, including costs for the full range of reversible contraceptive methods, contraception related services, and programmatic activities. The program is estimated to have prevented 85% of cases of estimated ZAM cases and unintended pregnancies in the absence of Z-CAN. The intervention cost was projected to have been more than offset by $79.9 million in ZIKV-associated costs avoided, 96% of which were lifetime ZAM-associated costs, as well as $137.0 million from avoided unintended pregnancies, with total net savings in one year of $216.9 million. The results were consistent with the previous CEA study.Conclusion: Z-CAN was likely cost-saving in the context of a public health emergency response setting. [ABSTRACT FROM AUTHOR]

Abstract: Introduction: We used health insurance claims data from large samples of Medicaid-enrolled and privately insured children to identify children with venous thromboembolism (VTE) and to assess their use of health services and associated expenditures during 2009. Materials and Methods: Data from the 2009 Thomson Reuters MarketScan® Commercial Database and Multi-State Medicaid database were used to estimate annual expenditures for children 1–17years of age with VTE. Generalized linear models were used to calculate adjusted annual expenditures for Medicaid-enrolled and privately insured children with VTE, controlling for age, sex, type of health plan, VTE classification (deep vein thrombosis and/or pulmonary embolism), and type of VTE event (idiopathic or secondary) and race (Medicaid only) or region (Commercial only). Results: During 2009, Medicaid-enrolled and privately insured children with VTE had an average of 1–2 inpatient admissions and 8–10 non-emergency department visits. Unadjusted mean total expenditures were similar for Medicaid-enrolled and privately insured children with VTE, $105,359 and $87,767, respectively. Adjusted mean expenditures for children with secondary VTE were five times higher than for children with idiopathic VTE. Conclusions: Given the high frequency of secondary VTE in children, most of the associated expenditures may be due to other health conditions. However, children who develop a VTE incur substantial costs of care, even in the absence of related conditions. Additional research is needed to evaluate the long term outcomes for children with VTE including rates of readmission, complications, and the impact of co-morbid conditions. [Copyright &y& Elsevier]

This study estimated medical expenditures attributable to cerebral palsy (CP) among children enrolled in Medicaid, stratified by the presence of co-occurring intellectual disability (ID), relative to children without CP or ID. The MarketScan® Medicaid Multi-State database was used to identify children with CP for 2003–2005 by using the International Classification of Diseases, Ninth Revision; Clinical Modification (ICD-9-CM) code 343.xx. Children with ID were identified for 2005 by using ICD-9-CM code 317.xx–319.xx. Children without CP or ID during the same period served as control subjects. Medical expenditures were estimated for case and control children for 2005. The difference between the average expenditures for children with and without CP was used as a proxy for attributable expenditures for the condition. The attributable expenditures of co-occurring ID were calculated similarly as the difference in average expenditures among children with CP with and without ID. A total of 9927 children with CP were identified. Among them, 2022 (20.3%) children had co-occurring ID recorded in medical claims. Children with CP but without ID incurred medical expenditures that were $15,047 higher than those of control children without CP or ID. By contrast, children with CP and co-occurring ID incurred costs that were $41,664 higher, compared with control children, and $26,617 more than children with CP but without ID. Administrative data from a large, multistate database demonstrated high medical expenditures for publicly insured children with CP. Expenditures approximately tripled for children with CP and co-occurring ID. [Copyright &y& Elsevier]

Purpose: To review the published evidence of the impact of alcohol screening and brief intervention (SBI) delivered in emergency departments (EDs) on healthcare utilization and costs.Principal Results: This scoping review used existing literature reviews supplemented with an electronic database. We included studies if they assessed SBIs for alcohol delivered in an ED setting and reported healthcare utilization and/or costs. We abstracted methodological approaches and healthcare utilization outcomes from each study and categorized them based on substance of focus (alcohol only vs. alcohol and other substances). We updated cost estimates from each study to 2018 U.S. dollars. We identified seven studies published between 2010 and 2019 that met study inclusion criteria. Two of the seven studies evaluated SBI that targeted both alcohol and other substances. Six studies found a reduction in healthcare utilization or costs, and one found an increase in healthcare utilization.Major Conclusions: This literature review suggests that SBI delivered in ED settings can be a cost-reducing approach to treating excessive alcohol consumption, a factor that policy-makers and payers might consider in prioritizing interventions. [ABSTRACT FROM AUTHOR]

Purpose The aim of this study was to assess the clinical characteristics and trends in valganciclovir use among infants diagnosed with congenital cytomegalovirus (CMV) disease in the United States. Methods We analyzed data from medical claims dated 2009–2015 from the Truven Health MarketScan ® Commercial Claims and Encounters and Medicaid databases. We identified infants with a live birth code in the first claim who were continuously enrolled for at least 45 days. Among infants diagnosed with congenital CMV disease, identified by an ICD-9-CM or ICD-10-CM code for congenital CMV infection or CMV disease within 45 days of birth, we assessed data from claims containing codes for any CMV-associated clinical condition within the same period, and data from claims for hearing loss and/or valganciclovir within the first 180 days of life. Findings In the commercial and Medicaid databases, we identified 257 (2.5/10,000) and 445 (3.3/10,000) infants, respectively, diagnosed with congenital CMV disease, among whom 135 (53%) and 282 (63%) had ≥1 CMV-associated condition, 30 (12%) and 32 (7%) had hearing loss, and 41 (16%) and 78 (18%) had a claim for valganciclovir. Among infants with congenital CMV disease who had a claim for valganciclovir, 37 (90%) among commercially insured infants and 68 (87%) among Medicaid-insured infants had ≥1 CMV-associated condition and/or hearing loss. From 2009 to 2015, the percentages with a claim for valganciclovir increased from 0% to 29% among commercially insured infants and from 4% to 37% among Medicaid-insured infants ( P < 0.0001). Implications During 2009–2015, there was a strong upward trend in valganciclovir claims among insured infants who were diagnosed with congenital CMV disease, the majority of whom had CMV-associated conditions and/or hearing loss. [ABSTRACT FROM AUTHOR]

Summary: Background: Congenital cytomegalovirus (CMV) infection is the leading infectious cause of congenital hearing loss and neurodevelopmental disability in developed countries. Information on congenital CMV infection in developing countries appears to be lacking. Methods: We conducted a systematic literature review to identify studies from developing countries with population-based samples of at least 300 infants that used laboratory methods established as reliable for the diagnosis of congenital CMV infection. Results: Most studies were excluded due to biased samples or inadequate diagnostic methods; consequently the search identified just 11 studies that were from Africa, Asia, and Latin America. The number of newborns tested ranged from 317 to 12 195. Maternal CMV seroprevalence ranged from 84% to 100%. CMV birth prevalence varied from 0.6% to 6.1%. CMV-associated impairments were not documented in most studies. Conclusions: Birth prevalence ranges were higher than for Europe and North America, as expected based on the higher maternal CMV seroprevalence. With very limited data available on sequelae, the disease burden of congenital CMV in developing countries remains largely unknown at this time. [Copyright &y& Elsevier]

Highlights: [•] Down syndrome (DS) is associated with greater hospital use to 3 years of age. [•] Hispanic children with DS have greater hospital use than Non-Hispanic Whites. [•] Non-Hispanic Blacks with DS and heart defects also have greater hospital use. [•] Hospitalization costs to age 3 attributable to DS totaled almost $18 million. [•] Costs were higher for Hispanics and Non-Hispanic Blacks vs. Non-Hispanic Whites. [Copyright &y& Elsevier]

Abstract: Hereditary hemochromatosis type 1, also known as hereditary hemochromatosis classical (HHC), is an iron overload disorder associated, in most cases, with mutations of the hemochromatosis (HFE) gene. Although suggested algorithms for diagnosing iron overload are available, there are still questions about options for genetic and biochemical screening for hemochromatosis and duration of treatment. This article provides a summary of an expert workgroup meeting convened on September 24–25, 2009, entitled “Iron Overload: What is the Role of Public Health?” The purpose of the meeting was to enable subject matter experts to share their most recent clinical and scientific iron overload information and to facilitate the discussion of future endeavors, with special emphasis on the role of public health in this field. The two main topics were the research priorities of the field, including clinical, genetic, and public health issues, and the concerns about the validity of current screening recommendations for the condition. [Copyright &y& Elsevier]

Background: Patients with sickle cell disease (SCD) often use emergency department services to obtain medical care. Limited information is available about emergency department use among patients with SCD. Purpose: This study assessed characteristics of emergency department visits made nationally by patients with SCD. Methods: Data from the National Hospital Ambulatory Medical Care Survey (NHAMCS) for the years 1999–2007 were analyzed. The NHAMCS is a survey of hospital emergency department and outpatient visits. Emergency department visits by patients with SCD were identified using ICD-9-CM codes, and nationally weighted estimates were calculated. Results: On average, approximately 197,333 emergency department visits were estimated to have occurred each year between 1999 and 2007 with SCD as one of the diagnoses listed. The expected source of payment was private insurance for 14%, Medicaid/State Children''s Health Insurance Program for 58%, Medicare for 14%, and other/unknown for 15%. Approximately 29% of visits resulted in hospital admission; this was 37% among patients aged 0–19 years, and 26% among patients aged ≥20 years. The episode of care was indicated as a follow-up visit for 23% of the visits. Patient-cited reasons for the emergency department visit included chest pain (11%); other pain or unspecified pain (67%); fever/infection (6%); and shortness of breath/breathing problem/cough (5%), among other reasons. Conclusions: Substantial numbers of emergency department visits occur among people with SCD. The most common reason for the emergency department visits is pain symptoms. The findings of this study can help to improve health services delivery and utilization among patients with SCD. [Copyright &y& Elsevier]

Abstract: Background: Use of the intrauterine device (IUD) in the United States has recently increased. New evidence for women with employer-sponsored health insurance permits analysis of variation and trends in such use. Study Design: A retrospective analysis of annual IUD insertion rates between 2002 and 2008 was conducted by evaluating claims from the MarketScan® Commercial Research Databases for US women insured by plans that covered IUD insertions. Estimates were weighted to be nationally representative. Results: IUD insertion rates increased from 1.6/1000 women of reproductive age to 9.8/1000 over 2002–2008 and varied substantially by state. Insertion rates of the levonorgestrel-releasing intrauterine system (LNG-IUS) increased from 0.4/1000 to 7.7/1000, whereas the insertion rates of copper T380A IUD (copper IUD) increased from 0.6/1000 to 1.5/1000. IUD insertions, which are most common among women aged 25–34 years, increased at roughly the same rate across all age groups. Conclusions: The sixfold increase in IUD insertion rates between 2002 and 2008 was accompanied by an increase in the share IUD use with the LNG-IUS from 40% to 85%. Substantial geographic and age variations existed. [Copyright &y& Elsevier]

Objective: To evaluate the determinants of intrauterine device (IUD) use and reasons for choosing IUDs over other reversible contraceptive methods. Design: Descriptive statistics and multinomial logistic regression were used to assess multiple factors associated with IUD use and the use of other reversible methods in the United States. Setting: Not applicable. Patient(s): Women at risk of pregnancy from the 2006 to 2008 National Survey of Family Growth and a 2004 Guttmacher Institute survey. Intervention(s): None. Main Outcome Measure(s): Sociodemographic and reproductive characteristics, family background, and health insurance coverage. Result(s): IUD use was positively associated with women’s parity and the highest education level of respondent’s mother; it was less common among women who had ≥4 sexual partners in the last 12 months and those who were widowed, divorced, or separated. IUD users reported pregnancy prevention, provider recommendation, and no interruption of sex as the most important reasons for choosing the method and reported a high level of satisfaction. Conclusion(s): IUD users differed substantially from users of other reversible contraceptives. IUD use was especially uncommon among nulliparae. Most current IUD users were satisfied with their choice. [ABSTRACT FROM AUTHOR]

Background: Although it is known that people with sickle cell disease (SCD) have relatively high utilization of medical care, most previous estimates of SCD-attributable expenditures have been limited to either inpatient care or single-state data. Purpose: To extend known findings by measuring the attributable or incremental expenditures per child with SCD compared to children without this illness and to thereby estimate SCD-attributable expenditures among children in the U.S. Methods: MarketScan Medicaid and Commercial Claims databases for 2005 were used to estimate total medical expenditures of children with and without SCD. Expenditures attributable to SCD were calculated as the difference in age-adjusted mean expenditures during 2005 for children with SCD relative to children without SCD in the two databases. Results: Children with SCD incurred medical expenditures that were $9369 and $13,469 higher than those of children without SCD enrolled in Medicaid and private insurance, respectively. In other words, expenditures of children with SCD were 6 and 11 times those of children without SCD enrolled in Medicaid and private insurance, respectively. Conclusions: Using a large, multistate, multipayer patient sample, SCD-attributable medical expenditures in children were conservatively and approximately estimated at $335 million in 2005. [Copyright &y& Elsevier]