Childhood disabilities and the cost of developmental therapies: the serviceprovider perspective.

PURPOSE: For children with neurodevelopmental disabilities (CWNDs), early diagnosis that leads to early intervention with regular targeted therapies is critical. In Qatar, private therapy centres that address this demand often have highly exclusive prices restricting families from availing them. This paper examines the challenges faced by families with CWNDs, as well as various financial and systemic obstacles, from the vantage point of these centres, all of which culminate in an extraordinarily high disability price tag for disability families in Qatar. METHODS: This study is based on qualitative, semi-structured, and in-depth interviews with private therapy centres and developmental paediatricians. RESULTS: Therapy centre representatives expressed common struggles in lengthy and cumbersome administration and licencing procedures, difficulty in hiring and retaining high quality staff, and expenses that need to be paid to the state. From their experience, families largely struggle with delayed diagnoses that significantly slow down intervention plans and therapies as well as staggeringly high financial costs with a dearth of funding options. CONCLUSIONS: We recommend sincere engagement, dialogue, and cooperation between multiple stakeholders; a supportive ecosystem to balance and distribute the demand that includes schools and parents; as well more efficient administrative procedures and recruitment strategies.

A single center experience with publicly funded clinical exome sequencing for neurodevelopmental disorders or multiple congenital anomalies.

Exome sequencing (ES) is an important diagnostic tool for individuals with neurodevelopmental disorders (NDD) and/or multiple congenital anomalies (MCA). However, the cost of ES limits the test's accessibility for many patients. We evaluated the yield of publicly funded clinical ES, performed at a tertiary center in Israel, over a 3-year period (2018-2020). Probands presented with (1) moderate-to-profound global developmental delay (GDD)/intellectual disability (ID); or (2) mild GDD/ID with epilepsy or congenital anomaly; and/or (3) MCA. Subjects with normal chromosomal microarray analysis who met inclusion criteria were included, totaling 280 consecutive cases. Trio ES (proband and parents) was the default option. In 252 cases (90.0%), indication of NDD was noted. Most probands were males (62.9%), and their mean age at ES submission was 9.3 years (range 1 month to 51 years). Molecular diagnosis was reached in 109 probands (38.9%), mainly due to de novo variants (91/109, 83.5%). Disease-causing variants were identified in 92 genes, 15 of which were implicated in more than a single case. Male sex, families with multiple-affected members and premature birth were significantly associated with lower ES yield (p < 0.05). Other factors, including MCA and coexistence of epilepsy, autism spectrum disorder, microcephaly or abnormal brain magnetic resonance imaging findings, were not associated with the yield. To conclude, our findings support the utility of clinical ES in a real-world setting, as part of a publicly funded genetic workup for individuals with GDD/ID and/or MCA.

Association between maternal hypertensive disorders, fetal growth and childhood learning outcomes.

OBJECTIVE: Both small for gestational age (SGA) birthweight and pregnancies complicated by maternal hypertension (HTN) are independently associated with poorer childhood learning outcomes, however the relative contribution of each remains unknown. STUDY DESIGN: A retrospective cohort was created in which 2014-2017 third grade Rhode Island Department of Education data were linked to Rhode Island Department of Health birth certificate data. The study population was composed of non-anomalous, singleton births between 22- and 42-weeks' gestation. Reading and math proficiency were compared among four groups: 1) appropriate for gestational age (AGA) and no maternal HTN (referent), 2) AGA with HTN, 3) SGA without HTN and 4) SGA with HTN. MAIN OUTCOME MEASURES: Bivariable and multivariable log-binomial regression were used to examine the association between subject proficiency and pregnancy complication, adjusting for potential confounders. RESULTS: Of the 23,097 who met inclusion criteria, 1004 (4%) were AGA with HTN, 1575 (7%) were SGA without HTN and 176 (1%) were SGA with HTN. Overall, when adjusted for maternal age, gestational age, sex and socioeconomic factors, only children born SGA without HTN had reduced reading proficiency (relative risk (RR) 0.86 95% confidence interval (CI) 0.78, 0.92) and math proficiency (RR 0.88 95% CI 0.82, 0.94) compared to children born AGA without HTN. CONCLUSION: In a diverse, statewide cohort, only SGA without HTN was associated with lower reading and math proficiency compared to uncomplicated pregnancies. This suggests that only decreased fetal growth from causes other than HTN is associated with risk of poorer school-age outcomes, and has implications for early resource allocation.

Examining the Association of Socioeconomic Position with Microcephaly and Delayed Childhood Neurodevelopment among Children with Prenatal Zika Virus Exposure.

Increased rates of Zika virus have been identified in economically deprived areas in Brazil at the population level; yet, the implications of the interaction between socioeconomic position and prenatal Zika virus exposure on adverse neurodevelopmental outcomes remains insufficiently evaluated at the individual level. Using data collected between September 2015 and September 2019 from 163 children with qRT-PCR and/or IgM-confirmed prenatal exposure to Zika virus participating in a prospective cohort study in Rio de Janeiro, Brazil (NCT03255369), this study evaluated the relationships of socioeconomic indicators with microcephaly at birth and Bayley-III neurodevelopmental scores during the early life course. Adjusted logistic regression models indicated increased odds of microcephaly in children born to families with lower household income (OR, 95% CI: 3.85, 1.43 to 10.37) and higher household crowding (OR, 95% CI: 1.83, 1.16 to 2.91), while maternal secondary and higher education appeared to have a protective effect for microcephaly compared to primary education (OR, 95% CI: 0.33, 0.11 to 0.98 and 0.10, 0.03 to 0.36, respectively). Consistent with these findings, adjusted linear regression models indicated lower composite language (-10.78, 95% CI: -19.87 to -1.69), motor (-10.45, 95% CI: -19.22 to -1.69), and cognitive (-17.20, 95% CI: -26.13 to -8.28) scores in children whose families participated in the Bolsa Família social protection programme. As such, the results from this investigation further emphasise the detrimental effects of childhood disadvantage on human health and development by providing novel evidence on the link between individual level socioeconomic indicators and microcephaly and delayed early life neurodevelopment following prenatal Zika virus exposure.

Effective/cost effective interventions of child mental health problems in low- and middle-income countries (LAMIC): Systematic review.

BACKGROUND: This systematic review protocol aims to examine the evidence of effectiveness and cost-effectiveness of interventions for children and adolescents with, or at risk of developing mental disorders in low- and middle-income countries (LAMICs). METHODS: We will search Medline Ovid, EMBASE Ovid, PsycINFO Ovid, CINAHL, LILACS, BDENF and IBECS. We will include randomised and non-randomised controlled trials, economic modelling studies and economic evaluations. Participants are 6 to 18 year-old children and adolescents who live in a LAMIC and who present with, or are at high risk of developing, one or more of the conditions: depression, anxiety, behavioural disorders, eating disorders, psychosis, substance abuse, autism and intellectual disabilities as defined by the DSM-V. Interventions which address suicide, self-harm will also be included, if identified during the extraction process. We will include in person or e-health interventions which have some evidence of effectiveness (in relation to clinical and/or functional outcomes) and which have been delivered to young people in LAMICs. We will consider a wide range of delivery channels (e.g., in person, web-based or virtual, phone), different practitioners (healthcare practitioners, teachers, lay health care providers) and sectors (i.e., primary, secondary and tertiary health care, education, guardianship councils). In the pilot of screening procedures, 5% of all references will be screened by two reviewers. Divergences will be resolved by one expert in mental health research. Reviewers will be retrained afterwards to ensure reliability. The remaining 95% will be screened by one reviewer. Covidence web-based tool will be used to perform screening of references and full text paper, and data extraction. RESULTS: The protocol of this systematic review will be disseminated in a peer-reviewed journal and presented at relevant conferences. The results will be presented descriptively and, if possible, meta-analysis will be conducted. Ethical approval is not needed for anonymised secondary data. CONCLUSION: the systematic review could help health specialists and other professionals to identify evidence-based strategies to deal with child and adolescents with mental health conditions.

Impact of maternal obesity on fetal cardiac screening: which follow-up strategy is cost-effective?

OBJECTIVE: To perform a cost-effectiveness analysis of different follow-up strategies for non-obese and obese women who had incomplete fetal cardiac screening for major congenital heart disease (CHD). METHODS: Three decision-analytic models, one each for non-obese, obese and Class-III-obese women, were developed to compare five follow-up strategies for initial suboptimal fetal cardiac screening. The five strategies were: (1) no follow-up ultrasound (US) examination but direct referral to fetal echocardiography (FE); (2) one follow-up US, then FE if fetal cardiac views were still suboptimal; (3) up to two follow-up US, then FE if fetal cardiac views were still suboptimal; (4) one follow-up US and no FE; and (5) up to two follow-up US and no FE. The models were designed to identify fetuses with major CHD in a theoretical cohort of 4 000 000 births in the USA. Outcomes related to neonatal mortality and neurodevelopmental disability were evaluated. A cost-effectiveness willingness-to-pay threshold was set at US$100 000 per quality-adjusted life year (QALY). Base-case and sensitivity analysis and Monte-Carlo simulation were performed. RESULTS: In our base-case models for all body mass index (BMI) groups, no follow-up US, but direct referral to FE led to the best outcomes, detecting 7%, 25% and 82% more fetuses with CHD in non-obese, obese and Class-III-obese women, respectively, compared with the baseline strategy of one follow-up US and no FE. However, no follow-up US, but direct referral to FE was above the US$100 000/QALY threshold and therefore not cost-effective. The cost-effective strategy for all BMI groups was one follow-up US and no FE. Both up to two follow-up US with no FE and up to two follow-up US with FE were dominated (being more costly and less effective), while one follow-up US with FE was over the cost-effectiveness threshold. One follow-up US and no FE was the optimal strategy in 97%, 93% and 86% of trials in Monte-Carlo simulation for non-obese, obese and Class-III-obese models, respectively. CONCLUSION: For both non-obese and obese women with incomplete fetal cardiac screening, the optimal CHD follow-up screening strategy is no further US and immediate referral to FE; however, this strategy is not cost-effective. Considering costs, one follow-up US and no FE is the preferred strategy. For both obese and non-obese women, Monte-Carlo simulations showed clearly that one follow-up US and no FE was the optimal strategy. Both non-obese and obese women with initial incomplete cardiac screening examination should therefore be offered one follow-up US. Copyright © 2019 ISUOG. Published by John Wiley & Sons Ltd.

Association between cumulating substances use and cumulating several school, violence and mental health difficulties in early adolescents.

Multiple substances (alcohol, tobacco, cannabis and other illicit drugs (OID)) have been frequently used in early adolescents maybe due to school, violence and mental-health difficulties. We investigated the associations between substance-use patterns and related difficulties among 1559 middle-school adolescents from north-eastern France (mean age 13.5 ±â€¯1.3). They completed a questionnaire including socioeconomic features, school, violence and mental-health difficulties (school grade repetition, sustained physical/verbal violence, sexual abuse, perpetrated violence, poor social support, depressive symptoms and suicide attempt; cumulated number noted SVMDscore) and the time of their first occurrence during the life course. Data were analyzed using logistic and negative binomial regression models. Alcohol, tobacco, cannabis and OID use affected 35.2, 11.2, 5.6 and 2.8% of the subjects respectively. The risk of using tobacco only, alcohol and tobacco, alcohol plus tobacco and cannabis, or all alcohol, tobacco, cannabis and OID strongly increased with the SVMDscore (socioeconomic features-adjusted odds ratio reaching 85). The risk began in early years in middle schools and then steadily increased, more markedly for elevated SVMDscore. Exposure to several SVMDs may be a transmission vector towards the substance use, starting mostly with alcohol/tobacco, and then shifting to cannabis/OID. These findings help to understand substance-use risk patterns and identify at-risk adolescents.

Trajectories of poverty and economic hardship among American families supporting a child with a neurodisability.

BACKGROUND: Caring for a child with a neurodisability (ND) impacts the financial decisions, relationships and well-being of family members, but evidence on the economic trajectories of families throughout the life course is missing. METHODS: Using data from the Panel Study of Income Dynamics, we tracked the families of 3317 children starting 5 years before childbirth until the child reached 20 years of age. We used regression and latent growth curve modelling to estimate trajectories of poverty and economic hardship over time. RESULTS: Families with a child with an ND had higher rates of poverty and economic hardship prior to childbirth and persistently over time. Analysis uncovered five latent trajectories for each indicator. After controlling for family and caregiver characteristics that preceded the birth of the child, raising a child with an ND was not associated with a unique trajectory of poverty. Families raising a child with an ND were however more likely to experience persistent economic hardship. CONCLUSIONS: The study establishes descriptive evidence for how having a child with an ND relates to changes in family economic conditions. The social and economic conditions that precede the child's birth seem to be driving the economic inequalities observed later throughout the life course.

Assessing social recovery of vulnerable youth in global mental health settings: a pilot study of clinical research tools in Malaysia.

BACKGROUND: A social recovery approach to youth mental health focuses on increasing the time spent in valuable and meaningful structured activities, with a view to preventing enduring mental health problems and social disability. In Malaysia, access to mental health care is particularly limited and little research has focused on identifying young people at risk of serious socially disabling mental health problems such as psychosis. We provide preliminary evidence for the feasibility and acceptability of core social recovery assessment tools in a Malaysian context, comparing the experiential process of engaging young Malaysian participants in social recovery assessments with prior accounts from a UK sample. METHODS: Nine vulnerable young people from low-income backgrounds were recruited from a non-government social enterprise and partner organisations in Peninsular Malaysia. Participants completed a battery of social recovery assessment tools (including time use, unusual experiences, self-schematic beliefs and values). Time for completion and completion rates were used as indices of feasibility. Acceptability was examined using qualitative interviews in which participants were asked to reflect on the experience of completing the assessment tools. Following a deductive approach, the themes were examined for fit with previous UK qualitative accounts of social recovery assessments. RESULTS: Feasibility was indicated by relatively efficient completion time and high completion rates. Qualitative interviews highlighted the perceived benefits of social recovery assessments, such as providing psychoeducation, aiding in self-reflection and stimulating goal setting, in line with findings from UK youth samples. CONCLUSIONS: We provide preliminary evidence for the feasibility and acceptability of social recovery assessment tools in a low-resource context, comparing the experiential process of engaging young Malaysian participants in social recovery assessments with prior accounts from a UK sample. We also suggest that respondents may derive some personal and psychoeducational benefits from participating in assessments (e.g. of their time use and mental health) within a social recovery framework.

Value-Driven Care in Developmental-Behavioral Pediatrics, Part 1: The Value Proposition of Developmental-Behavioral Pediatrics.

The drive to improve quality and reduce cost of health care is leading to a value-driven transformation of the US health care landscape. This is the first of a 2-part series on value-driven care and its implications for developmental-behavioral pediatrics (DBP). Tools derived from business/manufacturing models have been applied with some success to health care to better understand and enhance value. Value can be defined most simply as health outcomes achieved per dollar spent, but there are challenges in accounting for the full cost of a cycle or episode of care. When taking into account the "five Ds"-Development, Dependency, Differential Epidemiology, Demographics, and Dollars-the potential value of pediatric care is brought into sharper focus. The field of DBP in particular has the potential to add value through prevention, integration, efficiency, standardization, and innovation. The value proposition of DBP is illustrated by focusing on particular conditions or cycles of care commonly encountered in DBP practice: (1) children 0 to 3 years of age identified as having global developmental delay, (2) recent diagnosis of complex attention-deficit hyperactivity disorder (ADHD) (ADHD plus comorbid conditions), and (3) children with autism spectrum disorder and disruptive behavior. The second part of the series focuses on quality of care, emphasizing the important challenges that lie ahead for the field of DBP in measuring outcomes of care. With a discussion of national trends and a local example of a DBP program's response to these trends, the series is intended to provoke discussion and action in the field, contribute to the demonstration of value of a DBP approach to care, and help to chart a course toward growth and sustainability of DBP in an era of value-based care.

Value-Driven Care in Developmental-Behavioral Pediatrics, Part 2: Measuring Quality and Meeting the Challenge.

The drive to improve quality and reduce cost of health care is leading to a value-driven transformation of the US health care landscape. This is the second of a 2-part series on value-driven care and its implications for developmental-behavioral pediatrics (DBP). Part 1 addressed costs of care and enhancing value of care, with a particular focus on the value proposition of the field of DBP. This study begins with a discussion of the Donabedian's model of quality, including structure, process, patient satisfaction, and outcomes. The challenges of measuring process and outcomes in DBP are discussed, with a focus on (1) children 0 to 3 years of age identified as having global developmental delay, (2) recent diagnosis of complex attention-deficit hyperactivity disorder (ADHD) (ADHD plus comorbid conditions), and (3) children with autism spectrum disorder (ASD) and disruptive behavior. The study concludes with some of the important next steps for DBP providers, researchers, health care systems, professional societies, and families. With a discussion of national trends and a local example of a DBP program's response to these trends, the series is intended to provoke discussion and action in the field, contribute to the demonstration of value of a DBP approach to care, and help to chart a course toward growth and sustainability of DBP in an era of value-based care.

Private payer coverage policies for exome sequencing (ES) in pediatric patients: trends over time and analysis of evidence cited.

PURPOSE: Exome sequencing (ES) is being adopted for neurodevelopmental disorders in pediatric patients. However, little is known about current coverage policies or the evidence cited supporting these policies. Our study is the first in-depth review of private payer ES coverage policies for pediatric patients with neurodevelopmental disorders. METHODS: We reviewed private payer coverage policies and examined evidence cited in the policies of the 15 largest payers in 2017, and trends in coverage policies and evidence cited (2015-2017) for the five largest payers. RESULTS: There were four relevant policies (N = 5 payers) in 2015 and 13 policies (N = 15 payers) in 2017. In 2015, no payer covered ES, but by 2017, three payers from the original registry payers did. In 2017, 8 of the 15 payers covered ES. We found variations in the number and types of evidence cited. Positive coverage policies tended to include a larger number and range of citations. CONCLUSION: We conclude that more systematic assessment of evidence cited in coverage policies can provide a greater understanding of coverage policies and how evidence is used. Such assessments could facilitate the ability of researchers to provide the needed evidence, and the ability of clinicians to provide the most appropriate testing for patients.

Hidden Costs in Paediatric Psychiatry Consultation Liaison Services.

AIM: Children with acute mental health (MH) concerns increasingly present to Emergency Departments, and in the absence of an accessible MH bed, are admitted. This study estimated the hidden associated costs. METHODS: All Emergency MH admissions over a 12-month period (2016) were identified (N=105). Costs associated with length of stay (LOS) and one-to-one nursing were calculated. RESULTS: The average Length of Stay for acute MH presentations was 6 days, there were 615 bed day associated with this cohort, totalling costs of €1,216,470, with an average cost/patient of €12,684. Sixty-eight patients (65%) required an average of 5 days of one-to-one nursing, totalling 335 days. Estimating that 40% of this was provided by agency staff, the hospital cost was €115,374. Taken together, the costs associated with primary Emergency Mental Health presentations is €1,331,844. Costs associated with patients who were previously known to MH services were €843,417. DISCUSSION: Despite an increasing number of dedicated MH beds, demand outweighs availability, and immediate access remains problematic, the default often being a paediatric admission. Adequate funding and appropriate use of these scare and costly resources must be part of national MH policy planning, especially with ongoing planning for the National Children's Hospital.

Public Health Implications of Very Preterm Birth.

With advanced perinatal care and technology, survival among infants born very preterm (<32 weeks gestation) has improved dramatically over the last several decades. However, adverse medical and neurodevelopmental outcomes for those born very preterm remains high, particularly at the lowest gestational ages. Public health plays a critical role in providing data to assess population-based risks associated with very preterm birth, addressing disparities, and identifying opportunities for prevention, including improving the health of reproductive-age women, before, during, and after pregnancy.

Economic Evaluation of Interventions for Children with Neurodevelopmental Disorders: Opportunities and Challenges.

Economic evaluation is a tool used to inform decision makers on the efficiency of comparative healthcare interventions and inform resource allocation decisions. There is a growing need for the use of economic evaluations to assess the value of interventions for children with neurodevelopmental disorders (NDDs), a population that has increasing demands for healthcare services. Unfortunately, few evaluations have been conducted to date, perhaps stemming from challenges in applying existing economic evaluation methodologies in this heterogeneous population. Opportunities exist to innovate methods to address key challenges in conducting economic evaluations of interventions for children with NDDs. In this paper, we discuss important considerations and highlight areas for future work. This includes the paucity of appropriate instruments for measuring outcomes meaningful to children with NDDs and their families, difficulties in the measurement of costs due to service utilization in a wide variety of sectors, complexities in the measurement of caregiver and family effects and considerations in estimating long-term productivity costs. Innovation and application of evaluation approaches in these areas will help inform decisions around whether the resources currently spent on interventions for children with NDDs represent good value for money, or whether greater benefits for children could be generated by spending money in other ways.

Impact of functional somatic symptoms on 5-7-year-olds' healthcare use and costs.

OBJECTIVE: Knowledge about childhood functional somatic symptoms (FSS) and healthcare costs is scarce. This study aims to assess whether FSS in children aged 5-7 years are associated with increased future primary healthcare. DESIGN: At baseline of the observational cohort study, between years 2005 and 2007, 1327 children from the Copenhagen Child Cohort were assessed at ages 5-7 years for FSS and chronic physical diseases using the Soma Assessment Interview. Information on primary healthcare use was obtained from the National Health Insurance Service Register, and measured as the price of all medical services outside the hospital during a 4.5-year follow-up period from the day of assessment. Regression with bootstrap bias-corrected and accelerated CIs were performed. RESULTS: 1018 (76.8%) children had no FSS with primary healthcare use adjusted for other child health problems, maternal education and family changes of €448.2, 388.2-523.8 and number of face-to-face contacts: 11.90, 10.71-13.25; 250 (18.9%) had FSS with healthcare use €441.0, 355.0-550.3 and face-to-face contacts: 11.22, 9.60-12.91, and 58 (4.4%) had impairing FSS with healthcare use: €625.9, 447.9-867.8 and face-to-face contacts: 14.65, 11.20-19.00. In unadjusted regression analysis, impairing FSS were associated with increased healthcare use (increased costs: €246.0, 67.6-494.3). The adjusted association was slightly attenuated (increased costs: €177.8, 1.3-417.0). CONCLUSIONS: Impairing FSS in children aged 5-7 years is a predictor for the child's future primary healthcare use. More research on complex predictive models is needed to further explore the clinical significance of these results, and to contribute to the underpinning of early interventions towards impairing FSS in children.

[Child psychiatry: limited research, evidence for cost-effectiveness of treatment]. / Kinderpsychiatrie: beperkt onderzoek, aanwijzingen voor substantiële baten.

BACKGROUND: Research into cost-effectiveness of treatment of child psychiatric disorders is extremely limited. There are two main reasons for this: it's a new field and the type of research required is intrinsically complicated.
AIM: To review selected articles that reveal the prevalence of child psychiatric disorders, demonstrate the complexity of cost-efficiency research in child psychiatry and point to the possible benefits of appropriate treatment.
METHOD: We provide an overview of a selected number of articles dealing with the prevalence of child psychiatric disorders and the costs involved, we stress the diffulty of assessing whether current treatment is cost-effective and we describe the possible benefits of treatment.
RESULTS: However, the limited number of articles that we located do indicate that the treatment of children with psychiatric disorders is cost-effective. Not only does it benefit the child, it also eases the burden on the parents and on society as a whole. Findings need to be interpreted in the light of the limited scope and shortcomings of the research done so far.
CONCLUSION: Although current research seems to be cost-effective, we stress the need for further investigations, particularly in the form of longer-term studies.