Reducing the Burden of Anemia and Neural Tube Defects in Low- and Middle-Income Countries: An Analysis to Identify Countries with an Immediate Potential to Benefit from Large-Scale Mandatory Fortification of Wheat Flour and Rice.

Using a predetermined set of criteria, including burden of anemia and neural tube defects (NTDs) and an enabling environment for large-scale fortification, this paper identifies 18 low- and middle-income countries with the highest and most immediate potential for large-scale wheat flour and/or rice fortification in terms of health impact and economic benefit. Adequately fortified staples, delivered at estimated coverage rates in these countries, have the potential to avert 72.1 million cases of anemia among non-pregnant women of reproductive age; 51,636 live births associated with folic acid-preventable NTDs (i.e., spina bifida, anencephaly); and 46,378 child deaths associated with NTDs annually. This equates to a 34% reduction in the number of cases of anemia and 38% reduction in the number of NTDs in the 18 countries identified. An estimated 5.4 million disability-adjusted life years (DALYs) could be averted annually, and an economic value of 31.8 billion United States dollars (USD) generated from 1 year of fortification at scale in women and children beneficiaries. This paper presents a missed opportunity and warrants an urgent call to action for the countries identified to potentially avert a significant number of preventable birth defects, anemia, and under-five child mortality and move closer to achieving health equity by 2030 for the Sustainable Development Goals.

Social and financial barriers may contribute to a "hidden mortality" in Uganda for children with congenital anomalies.

BACKGROUND: The true incidence of congenital anomalies in sub-Saharan Africa is unknown. Owing to complex challenges associated with congenital anomalies, many affected babies may never present to a health facility, resulting in an underestimation of disease burden. METHODS: Interviews were conducted with Ugandans between September 2018 and May 2019. Responses from community members versus families of children with congenital anomalies were compared. RESULTS: A total of 198 Ugandans were interviewed (91 family members, 80 community members). All participants (N = 198) believed that seeking surgical care would lead to poverty, 43% (n = 84) assumed fathers would abandon the child, and 26% (n = 45) thought a child with a congenital anomaly in their community had been left to die. Causes of anomalies were believed to be contraceptive methods (48%, n = 95), witchcraft (17%, n = 34), or drugs (10%, n = 19). Of family members, 25 (28%) were advised to allow the child to die. Families with affected children were more likely to have a lower income (P < .001), believe anomalies could be treated (P = .007), but thought that allowing the child to die was best for the family (32% vs 9%; P < .0001). Monthly household income <50,000 Uganda shillings ($13 United States dollars) was a significant predictor of the father leaving the family (P = .024), being advised to not pursue medical care (P = .046), and believing that God should decide the child's fate (P = .047). CONCLUSION: Families face significant financial and social pressures when deciding to seek surgical care for a child with a congenital anomaly. Many children with anomalies may die and never reach a health facility to be counted, thus contributing to a hidden mortality.

The disproportionate cost of operation and congenital anomalies in infancy.

BACKGROUND: Congenital anomalies are the leading cause of infant death and pediatric hospitalization, but existing estimates of the associated costs of health care are either cross-sectional surveys or economic projections. We sought to determine the percent of total hospital health care expenditures attributable to major anomalies requiring surgery within the first year of life. METHODS: Utilizing comprehensive California statewide data from 2008 to 2012, cohorts of infants undergoing major surgery, with birth defects and with surgical anomalies, were constructed alongside a referent group of newborns with no anomalies or operations. Cost-to-charge and physician fee ratios were used to estimate hospital and professional costs, respectively. For each cohort, costs were broken down according to admission, birth episode, and first year of life, with additional stratifications by birth weight, gestational age, and organ system. RESULTS: In total, 68,126 of 2,205,070 infants (3.1%) underwent major surgery (n = 32,614) or had a diagnosis of a severe congenital anomaly (n = 57,793). These accounted for $7.7 billion of the $18.9 billion (40.7%) of the total health care costs/expenditures of the first-year-of-life hospitalizations, $7.0 billion (48.6%) of the costs for infants with comparatively long birth episodes, and $5.2 billion (54.7%) of the total neonatal intensive care unit admission costs. Infants with surgical anomalies (n = 21,264) totaled $4.1 billion (21.7%) at $80,872 per infant. Cardiovascular and gastrointestinal diseases accounted for most admission costs secondary to major surgery or congenital anomalies. CONCLUSION: In a population-based cohort of infant births compared with other critically ill neonates, surgical congenital anomalies are disproportionately costly within the United States health care system. The care of these infants, half of whom are covered by Medi-Cal or Medicaid, stands as a particular focus in an age of reform of health care payments.

Survival and healthcare utilization of infants diagnosed with lethal congenital malformations.

OBJECTIVE: We assessed survival, hospital length of stay (LOS), and costs of medical care for infants with lethal congenital malformations, and also examined the relationship between medical and surgical therapies and survival. STUDY DESIGN: Retrospective cohort study including infants born 1998-2009 with lethal congenital malformations, identified using a longitudinally linked maternal/infant database. RESULTS: The cohort included 786 infants: trisomy 18 (T18, n = 350), trisomy 13 (T13, n = 206), anencephaly (n = 125), bilateral renal agenesis (n = 53), thanatophoric dysplasia/achondrogenesis/lethal osteogenesis imperfecta (n = 38), and infants > 1 of the birth defects (n = 14). Compared to infants without birth defects, infants with T18, T13, bilateral renal agenesis, and skeletal dysplasias had longer survival rates, higher inpatient medical costs, and longer LOS. CONCLUSION: Care practices and survival have changed over time for infants with T18, T13, bilateral renal agenesis, and skeletal dysplasias. This information will be useful for clinicians in counseling families and in shaping goals of care prenatally and postnatally.

Employer-Sponsored Plan Expenditures for Infants Born Preterm.

BACKGROUND: Care for infants born preterm or with major birth defects is costly. Specific estimates of financial burden for different payers are lacking, in part because use of administrative data to identify preterm infants and costs is challenging. METHODS: We used private health insurance claims data and billing codes to identify live births during 2013 and calculated first-year expenditures for employer-sponsored health plans for infants born preterm, both overall and stratified by major birth defects. RESULTS: We conservatively estimated that 7.7% of insured infants born preterm accounted for 37% of $2.0 billion spent by participating plans on the care of infants born during 2013. With a mean difference in plan expenditures of ∼$47 100 per infant, preterm births cost the included plans an extra $600 million during the first year of life. Extrapolating to the national level, we projected aggregate employer-sponsored plan expenditures of $6 billion for infants born preterm during 2013. Infants with major birth defects accounted for 5.8% of preterm births but 24.5% of expenditures during infancy. By using an alternative algorithm to identify preterm infants, it was revealed that incremental expenditures were higher: $78 000 per preterm infant and $14 billion nationally. CONCLUSION: Preterm births (especially in conjunction with major birth defects) represent a substantial burden on payers, and efforts to mitigate this burden are needed. In addition, researchers need to conduct studies using linked vital records, birth defects surveillance, and administrative data to accurately and longitudinally assess per-infant costs attributable to preterm birth and the interaction of preterm birth with major birth defects.

Time-driven activity-based costing to estimate cost of care at multidisciplinary aerodigestive centers.

OBJECTIVES/HYPOTHESIS: Providing high-value healthcare to patients is increasingly becoming an objective for providers including those at multidisciplinary aerodigestive centers. Measuring value has two components: 1) identify relevant health outcomes and 2) determine relevant treatment costs. Via their inherent structure, multidisciplinary care units consolidate care for complex patients. However, their potential impact on decreasing healthcare costs is less clear. The goal of this study was to estimate the potential cost savings of treating patients with laryngeal clefts at multidisciplinary aerodigestive centers. STUDY DESIGN: Retrospective chart review. METHODS: Time-driven activity-based costing was used to estimate the cost of care for patients with laryngeal cleft seen between 2008 and 2013 at the Massachusetts Eye and Ear Infirmary Pediatric Aerodigestive Center. Retrospective chart review was performed to identify clinic utilization by patients as well as patient diet outcomes after treatment. Patients were stratified into neurologically complex and neurologically noncomplex groups. RESULTS: The cost of care for patients requiring surgical intervention was five and three times as expensive of the cost of care for patients not requiring surgery for neurologically noncomplex and complex patients, respectively. Following treatment, 50% and 55% of complex and noncomplex patients returned to normal diet, whereas 83% and 87% of patients experienced improved diets, respectively. Additionally, multidisciplinary team-based care for children with laryngeal clefts potentially achieves 20% to 40% cost savings. CONCLUSIONS: These findings demonstrate how time-driven activity-based costing can be used to estimate and compare patient costs in multidisciplinary aerodigestive centers. LEVEL OF EVIDENCE: 2c. Laryngoscope, 127:2152-2158, 2017.

Inpatient Hospitalization Costs Associated with Birth Defects Among Persons of All Ages - United States, 2013.

In the United States, major structural or genetic birth defects affect approximately 3% of live births (1) and are responsible for 20% of infant deaths (2). Birth defects can affect persons across their lifespan and are the cause of significant lifelong disabilities. CDC used the Healthcare Cost and Utilization Project (HCUP) 2013 National Inpatient Sample (NIS), a 20% stratified sample of discharges from nonfederal community hospitals, to estimate the annual cost of birth defect-associated hospitalizations in the United States, both for persons of all ages and by age group. Birth defect-associated hospitalizations had disproportionately high costs, accounting for 3.0% of all hospitalizations and 5.2% of total hospital costs. The estimated annual cost of birth defect-associated hospitalizations in the United States in 2013 was $22.9 billion. Estimates of the cost of birth defect-associated hospitalizations offer important information about the impact of birth defects among persons of all ages on the overall health care system and can be used to prioritize prevention, early detection, and care.

Cost Analysis of Following Up Incomplete Low-Risk Fetal Anatomy Ultrasounds.

OBJECTIVES: To examine the clinical utility and cost of follow-up ultrasounds performed as a result of suboptimal views at the time of initial second-trimester ultrasound in a cohort of low-risk pregnant women. METHODS: We conducted a retrospective cohort study of women at low risk for fetal structural anomalies who had second-trimester ultrasounds at 16 to less than 24 weeks of gestation from 2011 to 2013. We determined the probability of women having follow-up ultrasounds as a result of suboptimal views at the time of the initial second-trimester ultrasound, and calculated the probability of detecting an anomaly on follow-up ultrasound. These probabilities were used to estimate the national cost of our current ultrasound practice, and the cost to identify one fetal anomaly on follow-up ultrasound. RESULTS: During the study period, 1,752 women met inclusion criteria. Four fetuses (0.23% [95% CI 0.06-0.58]) were found to have anomalies at the initial ultrasound. Because of suboptimal views, 205 women (11.7%) returned for a follow-up ultrasound, and one (0.49% [95% CI 0.01-2.7]) anomaly was detected. Two women (0.11%) still had suboptimal views and returned for an additional follow-up ultrasound, with no anomalies detected. When the incidence of incomplete ultrasounds was applied to a similar low-risk national cohort, the annual cost of these follow-up scans was estimated at $85,457,160. In our cohort, the cost to detect an anomaly on follow-up ultrasound was approximately $55,000. CONCLUSIONS: The clinical yield of performing follow-up ultrasounds because of suboptimal views on low-risk second-trimester ultrasounds is low. Since so few fetal abnormalities were identified on follow-up scans, this added cost and patient burden may not be warranted.

[Analysis of a Family-centred Care Programme with Follow-up Home-visits in Neonatology - In Times of the Directive from G-BA]. / Konzeptanalyse einer stationären und ambulanten psychosozialen familienzentrierten Betreuung in der Neonatologie in Zeiten des G-BA-Beschlusses.

BACKGROUND: Marked progress in neonatology changed care of very preterm infants (VLBW) over the last decades - but also the attitude towards family-centred care (FCC). With the directive of the German Federal Joined Committee (G-BA), politicians recognize the necessity of neonatal FCC. AIM: To evaluate time and personnel costs necessary at a centre of established FCC. METHODS: Elternberatung "Frühstart" is a FCC programme for VLBW and seriously ill neonates from preganancy at risk to follow-up home-visits delivered by one interdisciplinary team. Analysis (2011-2014): 1.) Number of cases /participation in programme, 2.) resources of time, 3) and personnel, 4.) funding, 5) economic impact. RESULTS: 1.1.2011-31.12.2014: 441 cases (total cases: 2 212) participated in the programme. Participation of VLBW: mean 92% (86.4-97,2%). Costs of time are highest in neonates with congenital malformations: median 13.8 h, VLBW: median 11,2 h. Transition to home is most time intensive: median 7,3 (0-42.5) h. In average of 3.1 full-time nurses (part-time workers) are able to counsel 48 families/quarter. In severe cases funding is partly provided by health care insurances for social medical aftercare: positive applications: mean 92.7% (79.6-97.7%). CONCLUSION: Participation in the FCC programme in neonatology is high and costs of time are manageable.

Predictors of outcomes and hospital charges following atlantoaxial fusion.

BACKGROUND CONTEXT: Atlantoaxial fusion is used to correct atlantoaxial instability that is often secondary to traumatic fractures, Down syndrome, or rheumatoid arthritis. The effect of age and comorbidities on outcomes following atlantoaxial fusion is unknown. PURPOSE: This study aimed to better understand trends and predictors of outcomes and charges following atlantoaxial fusion and to identify confounding variables that should be included in future prospective studies. STUDY DESIGN: A retrospective analysis of data from the Nationwide Inpatient Sample (NIS), a nationally representative, all-payer database of inpatient diagnoses and procedures in the United States. PATIENT SAMPLE: We included all patients who underwent atlantoaxial fusion (International Classification of Disease, Ninth Revision, Clinical Modification code 81.01) between 1998 and 2011 who were 18 years or older at the time of admission. OUTCOME MEASURES: Outcome measures included in-hospital charges, hospital length of stay (LOS), in-hospital mortality, and discharge disposition. METHODS: Predictors of outcome following atlantoaxial fusion were assessed using a series of univariable analyses. Those predictors with a p-value of less than .2 were included in the final multivariable models. Independent predictors of outcome were those that were significant at an alpha level of 0.05 following inclusion in the final multivariable models. Logistic regression was used to determine predictors of in-hospital mortality and discharge disposition whereas linear regression was used to determine predictors of hospital charges and LOS. Discharge weights were used to produce generalizable results. RESULTS: From 1998 to 2011, there were 8,914 hospitalizations recorded wherein atlantoaxial fusion was performed during the inpatient hospital stay. Of these hospitalizations, 8,189 (91.9%) met inclusion criteria. Of the study sample, 62% was white, and the majority of patients were either insured by Medicare (47.2%) or had private health insurance (35.6%). The most common comorbidity as defined by the NIS and the Elixhauser comorbidity index was hypertension (43.2%). The in-hospital mortality rate for the study population was 2.7%, and the median LOS was 6.0 days. The median total charge (inflation adjusted) per hospitalization was $73,561. Of the patients, 48.9% were discharged to home. Significant predictors of in-hospital mortality included increased age, emergent or urgent admissions, weekend admissions, congestive heart failure, coagulopathy, depression, electrolyte disorder, metastatic cancer, neurologic disorder, paralysis, and non-bleeding peptic ulcer. Many of these variables were also found to be predictors of LOS, hospital charges, and discharge disposition. CONCLUSION: This study found that older patients and those with greater comorbidity burden had greater odds of postoperative mortality and were being discharged to another care facility, had longer hospital LOS, and incurred greater hospital charges following atlantoaxial fusion.

Length of stay and cost analysis of neonates undergoing surgery at a tertiary neonatal unit in England.

Introduction There is a lack of knowledge on the average length of stay (LOS) in neonatal units after surgical repair of common congenital anomalies. There are few if any publications reporting the activity performed by units undertaking neonatal surgery. Such activity is important for contracting arrangements, commissioning specialist services and counselling parents. The aim of this study was to describe postnatal LOS for infants admitted to a single tertiary referral neonatal unit with congenital malformations requiring surgery. Methods Data on nine conditions were collected prospectively for babies on the neonatal unit over a five-year period (2006-2011). For those transferred back to their local unit following surgery, the local unit was contacted to determine the total LOS. Only those babies who had surgery during their first admission to our unit and who survived to discharge were included in the study. Cost estimates were based on the tariffs agreed for neonatal care between our trust and the London specialised commissioning group in 2011-2012. Results The median LOS for the conditions studied was: gastroschisis 35 days (range: 19-154 days), oesophageal atresia 33 days (range: 9-133 days), congenital diaphragmatic hernia 28 days (range: 7-99 days), intestinal atresia 24 days (range: 6-168 days), Hirschsprung's disease 21 days (range: 15-36 days), sacrococcygeal teratoma 17 days (range: 12-55 days), myelomeningocoele 15.5 days (range: 8-24 days), anorectal malformation 15 days (range: 6-90 days) and exomphalos 12 days (range: 3-228 days). The total neonatal bed day costs for the median LOS ranged from £8,701 (myelomeningocoele) to £23,874 (gastroschisis). The cost of surgery was not included. Conclusions There is wide variation in LOS for the same conditions in a single neonatal unit. This can be explained by different types and severity within the same congenital anomalies, different surgeons and other clinical confounders (eg sepsis, surgical complications, associated anomalies). These data will enable us to give more detailed information to families following prenatal or postnatal diagnosis. They also allow more detailed planning of resource allocation for neonatal admissions.

Hospitalization charges for children with birth defects in Texas, 2001 to 2010.

BACKGROUND: State-specific information about hospitalizations of children with birth defects can improve understanding of changes in occurrence, treatment practices, and health care financing policies. This study analyzed aggregated data on hospital charges and length of stay for a large, diverse population. METHODS: We extracted hospitalization data for children diagnosed with birth defects from the Texas Hospital Inpatient Discharge Public Use Data File (2001-2010). Analyses compared total charges and length of stay for children with and without a diagnosis code of any birth defect among 45 standard categories. We also examined trends for total charges by expected payer type. RESULTS: In Texas, 431,296 hospital stays were reported for children with birth defects, with total charges of $24.8 billion. Mean hospital stay for children with birth defects was more than twice that of those without, whereas mean of hospital total charges was approximately six times greater. Pyloric stenosis accounted for the largest number of hospitalizations, followed by certain cardiac defects. Pediatric hospitalizations for birth defects increased 273.7%, compared with a 214.7% increase overall. The percentage of charges with Medicaid as expected payer (2004-2010) ranged from 56.5 to 62.0%. CONCLUSION: Charges associated with these conditions are far greater than those associated with pediatric hospitalizations for other causes, whether in the newborn period or beyond. However, these charges vary depending on specific diagnoses, expected payer source, and year of treatment.

Trends in the incidence of diabetes, its clinical sequelae, and associated costs in pregnancy.

BACKGROUND: Increasing diabetes prevalence affects a substantial number of pregnant women in the United States. Our aims were to evaluate health outcomes, medical costs, risks and types of complications associated with diabetes in pregnancy for mothers and newborns. METHODS: In this retrospective claims analysis, patients were identified from the Truven Health MarketScan(®) database (2004-2011 inclusive). Participants were aged 18-45 years, with ascertainable diabetes status [Yes/No], date of birth event >2005 and continuous health plan enrolment ≥21 months before and 3 months after the birth. RESULTS: In total, 839 792 pregnancies were identified, and 66 041 (7.86%) were associated with diabetes mellitus [type 1 (T1DM), 0.13%; type 2 (T2DM), 1.21%; gestational (GDM), 6.29%; and GDM progressing to T2DM (patients without prior diabetes who had a T2DM diagnosis after the birth event), 0.23%]. Relative risk (RR) of stillbirth (2.51), miscarriage (1.28) and Caesarean section (C-section) (1.77) was significantly greater with T2DM versus non-diabetes. Risk of C-section was also significantly greater for other diabetes types [RR 1.92 (T1DM); 1.37 (GDM); 1.63 (GDM progressing to T2DM)]. Risk of overall major congenital (RR ≥ 1.17), major congenital circulatory (RR ≥ 1.19) or major congenital heart (RR ≥ 1.18) complications was greater in newborns of mothers with diabetes versus without. Mothers with T2DM had significantly higher risk (RR ≥ 1.36) of anaemia, depression, hypertension, infection, migraine, or cardiac, obstetrical or respiratory complications than non-diabetes patients. Mean medical costs were higher with all diabetes types, particularly T1DM ($27 531), than non-diabetes ($14 355). CONCLUSIONS: Complications and costs of healthcare were greater with diabetes, highlighting the need to optimize diabetes management in pregnancy.

Effect of multiple chronic diseases on health care expenditures in childhood.

OBJECTIVES: To examine multiple chronic conditions and related health care expenditures in children. METHODS: Retrospective cohort study of all dependents of Mayo Clinic employees aged 0-17 on Jan 1, 2004 with continuous health benefits coverage for 4 years (N=14,727). Chronic conditions, health care utilization, and associated expenditures were obtained from medical and pharmacy claims. RESULTS: The most prevalent chronic conditions were asthma/chronic obstructive pulmonary disease (12%), allergic rhinitis (11%), and behavior problems (9%). The most costly conditions were congenital anomalies, asthma/chronic obstructive pulmonary disease, and behavior problems ($9602, $4335, and $5378 annual cost per child, respectively). Annual health care expenditures increased substantially with the number of chronic conditions, and a small proportion of children with multiple chronic conditions accounted for a large proportion of health care costs. In addition, those with multiple chronic conditions were more likely to persist in the top 10th percentile spender group in year-to-year spending. CONCLUSION: Children with multiple chronic conditions accounted for a large proportion of health care expenditures. These children were also likely to persist as high spenders in the 4-year time frame. Further research into effective ways to manage the health care delivery for children with multiple chronic conditions is needed.

Birth defects data from population-based birth defects surveillance programs in the United States, 2007 to 2011: highlighting orofacial clefts.

© 2014 The Authors Birth Defects Research Part A: Clinical and Molecular Teratology Published by Wiley Periodicals, Inc.

Cost effectiveness of teratology counseling - the Motherisk experience.

BACKGROUND: While the benefits of evidence-based counseling to large numbers of women and physicians are intuitively evident, there is an urgent need to document that teratology counseling, in addition to improving the quality of life of women and families, also leads to cost saving. The objective of the present study was to calculate the cost effectiveness of the Motherisk Program, a large teratology information and counseling service at The Hospital for Sick Children and the University of Toronto. METHODS: We analyzed data from the Motherisk Program on its 2012 activities in two domains: 1) Calculation of cost-saving in preventing unjustified pregnancy terminations; and 2) prevention of major birth defects. Cost of pregnancy termination and lifelong cost of specific birth defects were identified from primary literature and prorated for cost of living for the year 2013. RESULTS: Prevention of 255 pregnancy terminations per year led to cost savings of $516,630. The total estimated number of major malformations prevented by Motherisk counseling in 2012 was 8.41 cases at a total estimated cost of $9,032,492. CONCLUSIONS: With an estimated minimum annual prevention of 8 major malformations, and numerous unnecessary terminations of otherwise- wanted pregnancies, a cost saving of $10 million can be calculated. In 2013 the operating budget of Motherisk counseling totaled $640,000. Even based on the narrow range of activities for which we calculated cost, this service is highly cost- effective. Because most teratology counseling services are operating in a very similar method to Motherisk, it is fair to assume that these results, although dependent on the size of the service, are generalizable to other countries.

[The causes of physical disability in municipalities of the northeast of Brazil and an estimate of costs of specialized services]. / As causas da deficiência física em municípios do nordeste brasileiro e estimativa de custos de serviços especializados.

The population of the northeast of Brazil is characterized by high rates of endogamy and disabilities. An epidemiological cross-sectional study using the informant method was conducted in eight communities in the hinterlands of Paraiba to describe genetic and acquired diseases that cause disabilities and to estimate the costs of specialized services such as physiotherapy and the acquisition of technological assistential equipment. From a population of 48,499 inhabitants, 338 individuals were screened and 123 (0.34%) were clinically, genetically and functionally assessed by a multidisciplinary team of specialists. Genetic factors were responsible for 58.5% of the disabilities, with some clusters of prevalent diseases being found within the sampled communities, namely progressive spinal muscular atrophy, spinocerebellar ataxia, muscular dystrophy and Spoan syndrome. The socioeconomic profile and the demand for rehabilitation services and technological assistance highlight the need to introduce and implement specific public health policies in these communities.