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1.
PLoS One ; 14(7): e0219600, 2019.
Artigo em Inglês | MEDLINE | ID: mdl-31295316

RESUMO

CONTEXT: Permanent childhood hearing loss (PCHL) can affect speech, language, and wider outcomes. Adverse effects are mitigated through universal newborn hearing screening (UNHS) and early intervention. OBJECTIVE: We undertook a systematic review and meta-analysis to estimate prevalence of UNHS-detected PCHL (bilateral loss ≥26 dB HL) and its variation by admission to neonatal intensive care unit (NICU). A secondary objective was to report UNHS programme performance (PROSPERO: CRD42016051267). DATA SOURCES: Multiple electronic databases were interrogated in January 2017, with further reports identified from article citations and unpublished literature (November 2017). STUDY SELECTION: UNHS reports from very highly-developed (VHD) countries with relevant prevalence and performance data; no language or date restrictions. DATA EXTRACTION: Three reviewers independently extracted data and assessed quality. RESULTS: We identified 41 eligible reports from 32 study populations (1799863 screened infants) in 6195 non-duplicate references. Pooled UNHS-detected PCHL prevalence was 1.1 per 1000 screened children (95% confidence interval [CI]: 0.9, 1.3; I2 = 89.2%). This was 6.9 times (95% CI: 3.8, 12.5) higher among those admitted to NICU. Smaller studies were significantly associated with higher prevalences (Egger's test: p = 0.02). Sensitivity and specificity ranged from 89-100% and 92-100% respectively, positive predictive values from 2-84%, with all negative predictive values 100%. LIMITATIONS: Results are generalisable to VHD countries only. Estimates and inferences were limited by available data. CONCLUSIONS: In VHD countries, 1 per 1000 screened newborns require referral to clinical services for PCHL. Prevalence is higher in those admitted to NICU. Improved reporting would support further examination of screen performance and child demographics.


Assuntos
Análise Custo-Benefício , Transtornos da Audição/epidemiologia , Perda Auditiva/epidemiologia , Criança , Pré-Escolar , Feminino , Transtornos da Audição/diagnóstico , Transtornos da Audição/economia , Transtornos da Audição/patologia , Perda Auditiva/diagnóstico , Perda Auditiva/economia , Perda Auditiva/patologia , Testes Auditivos/economia , Humanos , Lactente , Recém-Nascido , Masculino , Triagem Neonatal/economia
2.
Pediatrics ; 143(5)2019 05.
Artigo em Inglês | MEDLINE | ID: mdl-30952779

RESUMO

BACKGROUND: Severe neonatal hyperbilirubinemia (>20 mg/dL) affects ∼1 million infants annually. Improved jaundice screening in low-income countries is needed to prevent bilirubin encephalopathy and mortality. METHODS: The Bili-ruler is an icterometer for the assessment of neonatal jaundice that was designed by using advanced digital color processing. A total of 790 newborns were enrolled in a validation study at Brigham and Women's Hospital (Boston) and Sylhet Osmani Medical College Hospital (Sylhet, Bangladesh). Independent Bili-ruler measurements were made and compared with reference standard transcutaneous bilirubin (TcB) and total serum bilirubin (TSB) concentrations. RESULTS: Bili-ruler scores on the nose were correlated with TcB and TSB levels (r = 0.76 and 0.78, respectively). The Bili-ruler distinguished different clinical thresholds of hyperbilirubinemia, defined by TcB, with high sensitivity and specificity (score ≥3.5: 90.1% [95% confidence interval (CI): 84.8%-95.4%] and 85.9% [95% CI: 83.2%-88.6%], respectively, for TcB ≥13 mg/dL). The Bili-ruler also performed reasonably well compared to TSB (score ≥3.5: sensitivity 84.5% [95% CI: 79.1%-90.3%] and specificity 83.2% [95% CI: 76.1%-90.3%] for TSB ≥11 mg/dL). Areas under the receiver operating characteristic curve for identifying TcB ≥11, ≥13, and ≥15 were 0.92, 0.93, and 0.94, respectively, and 0.90, 0.87, and 0.86 for identifying TSB ≥11, ≥13, and ≥15. Interrater reliability was high; 97% of scores by independent readers fell within 1 score of one another (N = 88). CONCLUSIONS: The Bili-ruler is a low-cost, noninvasive tool with high diagnostic accuracy for neonatal jaundice screening. This device may be used to improve referrals from community or peripheral health centers to higher-level facilities with capacity for bilirubin testing and/or phototherapy.


Assuntos
Recursos em Saúde/economia , Icterícia Neonatal/diagnóstico , Icterícia Neonatal/economia , Triagem Neonatal/economia , Triagem Neonatal/instrumentação , Adulto , Bangladesh/epidemiologia , Boston/epidemiologia , Cor , Feminino , Recursos em Saúde/tendências , Humanos , Hiperbilirrubinemia Neonatal/diagnóstico , Hiperbilirrubinemia Neonatal/economia , Hiperbilirrubinemia Neonatal/epidemiologia , Recém-Nascido , Icterícia/diagnóstico , Icterícia/economia , Icterícia/epidemiologia , Icterícia Neonatal/epidemiologia , Masculino , Triagem Neonatal/tendências , Adulto Jovem
3.
Public Health Nurs ; 36(4): 541-544, 2019 07.
Artigo em Inglês | MEDLINE | ID: mdl-30945355

RESUMO

Newborn screening (NBS) is a public health program that detects genetic conditions in neonates enabling treatment before clinical symptoms manifest. Severe combined immune deficiency (SCID) is a primary immune deficiency found in the absence of functioning T and B lymphocytes. Hematopoietic cell transplantation is a potentially curative treatment if received within the first 42 months of life; without treatment, this condition is fatal in the first 2 years of life due to severe opportunistic infections. SCID was added to the recommended uniform panel of conditions for inclusion in state NBS programs in 2010. This manuscript examines the societal costs and benefits of NBS for SCID in Arkansas and implications to health services and social welfare. Total cost per year of all NBS for SCID and resulting early treatment for one patient with SCID in Arkansas is estimated at $1,078,714. Cost of late treatment of one patient with SCID is estimated at $1.43 million. Based on an expected diagnosis of one patient per year in Arkansas, this results in an estimated net cost savings for NBS for SCID in Arkansas of $351,286 per year. Based on cost-effectiveness analysis, NBS for SCID in Arkansas is cost-effective, with higher societal benefit than cost.


Assuntos
Análise Custo-Benefício , Testes Genéticos/métodos , Triagem Neonatal/economia , Triagem Neonatal/métodos , Imunodeficiência Combinada Severa/diagnóstico , Arkansas , Linfócitos B/imunologia , Linfócitos B/transplante , Transplante de Células-Tronco Hematopoéticas/métodos , Humanos , Recém-Nascido , Imunodeficiência Combinada Severa/terapia , Linfócitos T/imunologia , Linfócitos T/transplante
4.
Clin Immunol ; 202: 33-39, 2019 05.
Artigo em Inglês | MEDLINE | ID: mdl-30946917

RESUMO

PURPOSE: Severe combined immunodeficiency (SCID) refers to a group of genetic disorders characterized by greatly compromised cellular and humoral immunity. Children with SCID are asymptomatic at birth, but they die from infections within the first months of life if not treated. Quantification of T-cell receptor excision circles is an extremely sensitive screening method for detecting newborns who may have SCID.The goal of the DEPISTREC study was to evaluate the feasibility of nationwide newborn screening for severe T-cell lymphopenia in France as well as its economic and clinical utility. METHODS: The test universally used for neonatal screening for SCID was the quantification of TRECs on Guthrie cards. We compared a group of 190,517 babies from 48 maternities across the country who underwent newborn SCID screening with a control group of 1.4 million babies out of whom 28 were diagnosed with SCID without such screening during the course of the study. RESULTS: Within the screening group, 62 babies were found to be lymphopenic, including three with SCID. The cost of screening ranged from 4.7€ to €8.15 per newborn. The average 18-month cost was €257,574 vs €204,697 in the control group. CONCLUSIONS: In this large-scale study, we demonstrate that routine SCID screening is feasible and effective. This screening offers the additional benefit of aiding in the diagnosis of non-SCID lymphopenia. Economic evaluation allowed us to calculate the cost per test. Newborn screening may also prevent death by SCID before any curative treatment can be administered. The difference in cost between screened and control children could not be ascertained because of the very low numbers and death of one of the children tested.


Assuntos
Linfopenia/diagnóstico , Triagem Neonatal/economia , Imunodeficiência Combinada Severa/diagnóstico , Custos e Análise de Custo , Teste em Amostras de Sangue Seco/economia , Feminino , França , Humanos , Lactente , Recém-Nascido , Contagem de Linfócitos , Linfopenia/economia , Masculino , Receptores de Antígenos de Linfócitos T/imunologia , Imunodeficiência Combinada Severa/economia , Linfócitos T/imunologia
5.
Eur J Pediatr ; 178(5): 721-729, 2019 May.
Artigo em Inglês | MEDLINE | ID: mdl-30805731

RESUMO

Severe combined immunodeficiency (SCID) is a condition that often results in severe infections and death at young age. Early detection shortly after birth, followed by treatment before infections occur, largely increases the chances of survival. As the incidence of SCID is low, assessing cost-effectiveness of adding screening for SCID to the newborn screening program is relevant for decision making. Lifetime costs and effects of newborn screening for SCID were compared to a situation without screening in the Netherlands in a decision analysis model. Model parameters were based on literature and expert opinions. Sensitivity analyses were performed. Due to earlier detection, the number of deaths due to SCID per 100,000 children was assessed to decrease from 0.57 to 0.23 and a number of 11.7 quality adjusted life-years (QALYs) gained was expected. Total yearly healthcare costs, including costs of screening, diagnostics, and treatment, were €390,800 higher in a situation with screening compared to a situation without screening, resulting in a cost-utility ratio of €33,400 per QALY gained.Conclusion: Newborn screening for SCID might be cost-effective. However, there is still a lot of uncertainty around the cost-effectiveness estimate. Pilot screening projects are warranted to obtain more accurate estimates for the European situation. What is Known: • Severe combined immunodeficiency (SCID) is a condition that often results in severe infections and death at a young age. • As the incidence of SCID is low, assessing cost-effectiveness of adding screening for SCID to the newborn screening program is needed. What is New: • Newborn screening for SCID is expected to reduce mortality from 0.57 to 0.23 per 100,000 children at additional healthcare costs of €390,800. The cost-utility ratio is €33,400 per QALY gained. • Due to large uncertainty around cost-effectiveness estimates, pilot screening projects are warranted for Europe.


Assuntos
Análise Custo-Benefício , Custos de Cuidados de Saúde/estatística & dados numéricos , Triagem Neonatal/economia , Imunodeficiência Combinada Severa/diagnóstico , Humanos , Recém-Nascido , Países Baixos , Anos de Vida Ajustados por Qualidade de Vida , Imunodeficiência Combinada Severa/economia
6.
Am J Hematol ; 94(1): 39-45, 2019 01.
Artigo em Inglês | MEDLINE | ID: mdl-30290004

RESUMO

Sickle cell disease (SCD) is a common, life-threatening genetic disorder that is best managed when diagnosed early by newborn screening. However, SCD is most prevalent in low-resource regions of the world where newborn screening is rare and diagnosis at the point-of-care is challenging. In many such regions, the majority of affected children die, undiagnosed, before the age of 5 years. A rapid and affordable point-of-care test for SCD is needed. The diagnostic accuracy of HemoTypeSC, a point-of-care immunoassay, for SCD was evaluated in individuals who had SCD, hemoglobin C disease, the related carrier (trait) states, or a normal hemoglobin phenotype. Children and adults participated in low-, medium- and high-resource environments (Ghana [n = 383], Martinique [n = 46], and USA [n = 158]). Paired blood specimens were obtained for HemoTypeSC and a reference diagnostic assay. HemoTypeSC testing was performed at the site of blood collection, and the reference test was performed in a laboratory at each site. In 587 participants, across all study sites, HemoTypeSC had an overall sensitivity of 99.5% and specificity of 99.9% across all hemoglobin phenotypes. The test had 100% sensitivity and specificity for sickle cell anemia. Sensitivity and specificity for detection of normal and trait states were >99%. HemoTypeSC is an inexpensive (<$2 per test), accurate, and rapid point-of-care test that can be used in resource-limited regions with a high prevalence of SCD to provide timely diagnosis and support newborn screening programs.


Assuntos
Anemia Falciforme/diagnóstico , Imunoensaio , Sistemas Automatizados de Assistência Junto ao Leito , Adulto , Anemia Falciforme/sangue , Anemia Falciforme/epidemiologia , Anticorpos Monoclonais/imunologia , Criança , Países em Desenvolvimento , Diagnóstico Precoce , Feminino , Gana/epidemiologia , Hemoglobina A/análise , Hemoglobina C/análise , Doença da Hemoglobina C/sangue , Doença da Hemoglobina C/diagnóstico , Doença da Hemoglobina C/epidemiologia , Hemoglobina Falciforme/análise , Humanos , Imunoensaio/economia , Recém-Nascido , Masculino , Martinica/epidemiologia , Triagem Neonatal/economia , Triagem Neonatal/métodos , Prevalência , Estudos Prospectivos , Sensibilidade e Especificidade , Traço Falciforme/sangue , Traço Falciforme/diagnóstico , Traço Falciforme/epidemiologia , Método Simples-Cego
7.
Eur J Pediatr ; 178(1): 97-103, 2019 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-30334077

RESUMO

Pulse oximetry (PO) screening is used to screen newborns for critical congenital heart defects (CCHD). Analyses performed in hospital settings suggest that PO screening is cost-effective. We assessed the costs and cost-effectiveness of PO screening in the Dutch perinatal care setting, with home births and early postnatal discharge, compared to a situation without PO screening. Data from a prospective accuracy study with 23,959 infants in the Netherlands were combined with a time and motion study and supplemented data. Costs and effects of the situations with and without PO screening were compared for a cohort of 100,000 newborns. Mean screening time per newborn was 4.9 min per measurement and 3.8 min for informing parents. The additional costs of screening were in total €14.71 per screened newborn (€11.00 personnel, €3.71 equipment costs). Total additional costs of screening and referral were €1,670,000 per 100,000 infants. This resulted in an incremental cost-effectiveness ratio of €139,000 per additional newborn with CCHD detected with PO, when compared to a situation without PO screening. A willingness-to-pay threshold of €20,000 per gained QALY for screening in the Netherlands makes the screening likely to be cost-effective.Conclusion: PO screening in the Dutch care setting is likely to be cost-effective. What is Known: • Pulse oximetry is increasingly implemented as a screening tool for critical congenital heart defects in newborns. • Previous studies suggest that the screening in cost-effective and in the USA a reduction in infant mortality from critical congenital heart defects was demonstrated. What is New: • This is the first cost-effectiveness analysis for pulse oximetry screening in a setting with screening after home births, with screening at two moments. • Costs of pulse oximetry screening in a setting with hospital and homebirth deliveries were €14.71 and is likely to be cost-effective accordint to Dutch standards.


Assuntos
Custos de Cuidados de Saúde/estatística & dados numéricos , Cardiopatias Congênitas/diagnóstico , Triagem Neonatal/economia , Oximetria/economia , Análise Custo-Benefício , Parto Domiciliar/estatística & dados numéricos , Humanos , Recém-Nascido , Triagem Neonatal/métodos , Países Baixos , Oximetria/métodos , Alta do Paciente/tendências , Estudos Prospectivos
8.
Orphanet J Rare Dis ; 13(1): 179, 2018 10 11.
Artigo em Inglês | MEDLINE | ID: mdl-30309370

RESUMO

BACKGROUND: A decision tree model was built to estimate the economic impact of introducing screening for X-linked adrenoleukodystrophy (X-ALD) into an existing tandem mass spectrometry based newborn screening programme. The model was based upon the UK National Health Service (NHS) Newborn Blood Spot Screening Programme and a public service perspective was used with a lifetime horizon. The model structure and parameterisation were based upon literature reviews and expert clinical judgment. Outcomes included health, social care and education costs and quality adjusted life years (QALYs). The model assessed screening of boys only and evaluated the impact of improved outcomes from hematopoietic stem cell transplantation in patients with cerebral childhood X-ALD (CCALD). Threshold analyses were used to examine the potential impact of utility decrements for non-CCALD patients identified by screening. RESULTS: It is estimated that screening 780,000 newborns annually will identify 18 (95%CI 12, 27) boys with X-ALD, of whom 10 (95% CI 6, 15) will develop CCALD. It is estimated that screening may detect 7 (95% CI 3, 12) children with other peroxisomal disorders who may also have arisen symptomatically. If results for girls are returned an additional 17 (95% CI 12, 25) cases of X-ALD will be identified. The programme is estimated to cost an additional £402,000 (95% CI £399-407,000) with savings in lifetime health, social care and education costs leading to an overall discounted cost saving of £3.04 (95% CI £5.69, £1.19) million per year. Patients with CCALD are estimated to gain 8.5 discounted QALYs each giving an overall programme benefit of 82 (95% CI 43, 139) QALYs. CONCLUSION: Including screening of boys for X-ALD into an existing tandem mass spectrometry based newborn screening programme is projected to reduce lifetime costs and improve outcomes for those with CCALD. The potential disbenefit to those identified with non-CCALD conditions would need to be substantial in order to outweigh the benefit to those with CCALD. Further evidence is required on the potential QALY impact of early diagnosis both for non-CCALD X-ALD and other peroxisomal disorders. The favourable economic results are driven by estimated reductions in the social care and education costs.


Assuntos
Adrenoleucodistrofia/sangue , Adrenoleucodistrofia/diagnóstico , Triagem Neonatal/economia , Triagem Neonatal/métodos , Humanos , Recém-Nascido
9.
J Clin Endocrinol Metab ; 103(11): 4043-4088, 2018 11 01.
Artigo em Inglês | MEDLINE | ID: mdl-30272171

RESUMO

Objective: To update the congenital adrenal hyperplasia due to steroid 21-hydroxylase deficiency clinical practice guideline published by the Endocrine Society in 2010. Conclusions: The writing committee presents updated best practice guidelines for the clinical management of congenital adrenal hyperplasia based on published evidence and expert opinion with added considerations for patient safety, quality of life, cost, and utilization.


Assuntos
Hiperplasia Suprarrenal Congênita/terapia , Endocrinologia/normas , Sociedades Médicas/normas , Hiperplasia Suprarrenal Congênita/diagnóstico , Hiperplasia Suprarrenal Congênita/economia , Hiperplasia Suprarrenal Congênita/genética , Análise Custo-Benefício , Feminino , Terapias Fetais/economia , Terapias Fetais/métodos , Terapias Fetais/normas , Aconselhamento Genético/economia , Aconselhamento Genético/métodos , Aconselhamento Genético/normas , Glucocorticoides/uso terapêutico , Humanos , Recém-Nascido , Assistência de Longa Duração/economia , Assistência de Longa Duração/métodos , Assistência de Longa Duração/normas , Triagem Neonatal/economia , Triagem Neonatal/normas , Segurança do Paciente/normas , Qualidade de Vida , Terapias em Estudo/economia , Terapias em Estudo/métodos , Terapias em Estudo/normas
10.
J Pediatr ; 203: 371-379.e7, 2018 12.
Artigo em Inglês | MEDLINE | ID: mdl-30268400

RESUMO

OBJECTIVE: To assess longitudinal estimates of inpatient costs through early childhood in patients with critical congenital heart defects (CCHDs), for whom reliable estimates are scarce, using a population-based cohort of clinically validated CCHD cases. STUDY DESIGN: Longitudinal retrospective cohort of infants with CCHDs live born from 1997 to 2012 in Utah. Cases identified from birth defect registry data were linked to inpatient discharge abstracts and vital records to track inpatient days and costs through age 10 years. Costs were adjusted for inflation and discounted by 3% per year to generate present value estimates. Multivariable models identified infant and maternal factors potentially associated with higher resource utilization and were used to calculate adjusted costs by defect type. RESULTS: The final statewide cohort included 1439 CCHD cases among 803 509 livebirths (1.8/1000). The average cost per affected child through age 10 years was $136 682 with a median of $74 924 because of a small number of extremely high cost children; costs were highest for pulmonary atresia with ventricular septal defect and hypoplastic left heart syndrome. Inpatient costs increased by 1.6% per year during the study period. A single birth year cohort (~50 000 births/year) had estimated expenditures of $11 902 899 through age 10 years. Extrapolating to the US population, inpatient costs for a single birth year cohort through age 10 years were ~$1 billion. CONCLUSIONS: Inpatient costs for CCHDs throughout childhood are high and rising. These revised estimates will contribute to comparative effectiveness research aimed at improving the value of care on a patient and population level.


Assuntos
Custos de Cuidados de Saúde , Cardiopatias Congênitas/economia , Cardiopatias Congênitas/epidemiologia , Triagem Neonatal/economia , Triagem Neonatal/métodos , Anormalidades Congênitas , Bases de Dados Factuais , Feminino , Comunicação Interventricular/economia , Comunicação Interventricular/epidemiologia , Hospitalização/economia , Humanos , Síndrome do Coração Esquerdo Hipoplásico/economia , Síndrome do Coração Esquerdo Hipoplásico/epidemiologia , Lactente , Recém-Nascido , Pacientes Internados , Estudos Longitudinais , Masculino , Análise Multivariada , Atresia Pulmonar/economia , Atresia Pulmonar/epidemiologia , Sistema de Registros , Estudos Retrospectivos , Utah/epidemiologia
11.
Hastings Cent Rep ; 48 Suppl 2: S43-S44, 2018 Jul.
Artigo em Inglês | MEDLINE | ID: mdl-30133724

RESUMO

While the NSIGHT program was driven by a desire to define and gather data about both the benefits and harms of introducing genomic sequencing into the care of newborns, it remains to be seen how much influence these data will have in shaping the use of this technology in newborns. Ultimately, three additional forces-commercial interests, the technological imperative, and advocates-may play a significant role in shaping the use of sequencing in newborns. Policy-makers and clinicians should be aware of the effects of these additional forces when considering the appropriate use of this technology in clinical practice and public health screening programs.


Assuntos
Testes Genéticos/economia , Triagem Neonatal/economia , Triagem Neonatal/métodos , Sequenciamento Completo do Genoma/economia , Difusão de Inovações , Testes Genéticos/ética , Humanos , Recém-Nascido , Triagem Neonatal/ética , Sequenciamento Completo do Genoma/ética
12.
BMC Pediatr ; 18(1): 225, 2018 07 09.
Artigo em Inglês | MEDLINE | ID: mdl-29986673

RESUMO

BACKGROUND: The greatest opportunity for lifelong impact of genomic sequencing is during the newborn period. The "BabySeq Project" is a randomized trial that explores the medical, behavioral, and economic impacts of integrating genomic sequencing into the care of healthy and sick newborns. METHODS: Families of newborns are enrolled from Boston Children's Hospital and Brigham and Women's Hospital nurseries, and half are randomized to receive genomic sequencing and a report that includes monogenic disease variants, recessive carrier variants for childhood onset or actionable disorders, and pharmacogenomic variants. All families participate in a disclosure session, which includes the return of results for those in the sequencing arm. Outcomes are collected through review of medical records and surveys of parents and health care providers and include the rationale for choice of genes and variants to report; what genomic data adds to the medical management of sick and healthy babies; and the medical, behavioral, and economic impacts of integrating genomic sequencing into the care of healthy and sick newborns. DISCUSSION: The BabySeq Project will provide empirical data about the risks, benefits and costs of newborn genomic sequencing and will inform policy decisions related to universal genomic screening of newborns. TRIAL REGISTRATION: The study is registered in ClinicalTrials.gov Identifier: NCT02422511 . Registration date: 10 April 2015.


Assuntos
Triagem Neonatal/métodos , Sequenciamento Completo do Exoma , Família/psicologia , Aconselhamento Genético , Predisposição Genética para Doença/psicologia , Custos de Cuidados de Saúde , Humanos , Recém-Nascido , Triagem Neonatal/economia , Triagem Neonatal/psicologia , Medição de Risco
13.
Neonatology ; 114(2): 155-162, 2018.
Artigo em Inglês | MEDLINE | ID: mdl-29895035

RESUMO

BACKGROUND: Babies at risk of neonatal hypoglycaemia are often screened using cot-side glucometers, but non-enzymatic glucometers are inaccurate, potentially resulting in over-treatment and under-treatment, and low values require laboratory confirmation. More accurate enzymatic glucometers are available but at apparently higher costs. OBJECTIVE: Our objective was to compare the cost of screening for neonatal hypoglycaemia using point-of-care enzymatic and non-enzymatic glucometers. METHODS: We used a decision tree to model costs, including consumables and staff time. Sensitivity analyses assessed the impact of staff time, staff costs, probability that low results are confirmed via laboratory testing, false-positive and false-negative rates of non-enzymatic glucometers, and the blood glucose concentration threshold. RESULTS: In the primary analysis, screening using an enzymatic glucometer cost NZD 86.94 (USD 63.47) while using a non-enzymatic glucometer cost NZD 97.08 (USD 70.87) per baby. Sensitivity analyses showed that using an enzymatic glucometer is cost saving with wide variations in staff time and costs, irrespective of the false-positive level of non-enzymatic glucometers, and where ≥78% of low values are laboratory confirmed. Where non-enzymatic glucometers may be less costly (e.g., false-negative rate exceeds 15%), instances of hypoglycaemia will be missed. Reducing the blood glucose concentration threshold to 1.94 mmol/L reduced the incidence of hypoglycaemia from 52 to 13%, and the cost of screening using a non-enzymatic glucometer to NZD 47.71 (USD 34.83). CONCLUSIONS: In view of their lower cost in most circumstances and greater accuracy, enzymatic glucometers should be routinely utilised for point-of-care screening for neonatal hypoglycaemia.


Assuntos
Análise Química do Sangue/instrumentação , Glicemia/análise , Hipoglicemia/diagnóstico , Triagem Neonatal/economia , Testes Imediatos/economia , Custos e Análise de Custo , Humanos , Hipoglicemia/sangue , Recém-Nascido , Nova Zelândia , Reprodutibilidade dos Testes
14.
Am J Trop Med Hyg ; 98(6): 1733-1742, 2018 06.
Artigo em Inglês | MEDLINE | ID: mdl-29714163

RESUMO

Chagas disease, caused by Trypanosoma cruzi, is transmitted by insect vectors through transfusions, transplants, insect feces in food, and from mother to child during gestation. Congenital infection could perpetuate Chagas disease indefinitely, even in countries without vector transmission. An estimated 30% of infected persons will develop lifelong, potentially fatal, cardiac or digestive complications. Treatment of infants with benznidazole is highly efficacious in eliminating infection. This work evaluates the costs of maternal screening and infant testing and treatment of Chagas disease in the United States. We constructed a decision-analytic model to find the lower cost option, comparing costs of testing and treatment, as needed, for mothers and infants with the lifetime societal costs without testing and the consequent morbidity and mortality due to lack of treatment or late treatment. We found that maternal screening, infant testing, and treatment of Chagas disease in the United States are cost saving for all rates of congenital transmission greater than 0.001% and all levels of maternal prevalence above 0.06% compared with no screening program. Newly approved diagnostics make universal screening cost saving with maternal prevalence as low as 0.008%. The present value of lifetime societal savings due to screening and treatment is about $634 million saved for every birth year cohort. The benefits of universal screening for T. cruzi as part of routine prenatal testing far outweigh the program costs for all U.S. births.


Assuntos
Doença de Chagas/epidemiologia , Transmissão Vertical de Doença Infecciosa/prevenção & controle , Complicações Parasitárias na Gravidez/epidemiologia , Trypanosoma cruzi/isolamento & purificação , Doença de Chagas/mortalidade , Doença de Chagas/parasitologia , Doença de Chagas/transmissão , Estudos de Coortes , Redução de Custos , Testes Diagnósticos de Rotina , Feminino , Humanos , Recém-Nascido , Programas de Rastreamento/economia , Morbidade , Mães , Triagem Neonatal/economia , Gravidez , Complicações Parasitárias na Gravidez/mortalidade , Complicações Parasitárias na Gravidez/parasitologia , Prevalência , Estados Unidos
16.
Int J Technol Assess Health Care ; 34(2): 189-195, 2018 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-29633672

RESUMO

OBJECTIVES: This paper aims to describe the added value of combining cost-effectiveness and ethical evaluations when the preferences of the decision maker toward cost-effectiveness evaluation outcomes are not known, with the French national neonatal screening of cystic fibrosis (CF) as a case-study. METHODS: A cost-effectiveness analysis comparing four CF neonatal screening strategies, with or without DNA testing, was performed. Ethical positions toward their outcomes were described. In addition, a post-hoc analysis of the ethical issues being considered relevant from the decision-makers' perspective was conducted. RESULTS: Two strategies were found equally cost-effective. Among them, choosing the non-DNA or a DNA-based strategy constrains the decision maker to render a judgement between different ethical issues or disagreements associated with the screening program. CONCLUSIONS: The analysis supports the relevance of combining cost-effectiveness and ethics evaluation in developing health policy, as a way to reveal or clarify the motives associated with health. The choice of the decision maker to favor the DNA-based strategy, which was not originally recommended, creates the opportunity to make explicit the role played by ethical issues in the decision.


Assuntos
Fibrose Cística/diagnóstico , Tomada de Decisões , Triagem Neonatal/economia , Triagem Neonatal/ética , Análise Custo-Benefício , Fibrose Cística/genética , Erros de Diagnóstico , França , Testes Genéticos , Humanos , Recém-Nascido , Proteínas Associadas a Pancreatite/sangue , Tripsinogênio/sangue , Incerteza
17.
Can J Ophthalmol ; 53(2): 162-167, 2018 04.
Artigo em Inglês | MEDLINE | ID: mdl-29631829

RESUMO

OBJECTIVE: To compare costs of 2 screening modalities for retinopathy of prematurity (ROP): telemedicine imaging with remote interpretation versus in-person binocular indirect ophthalmoscopy (BIO). DESIGN: Retrospective chart review. PARTICIPANTS: Infants from an existing telemedicine screening program at 2 cities in Ontario, Canada. METHODS: We conducted a cost analysis comparison from the perspective of the Ministry of Health. Patient level data was used for the telemedicine group. A hypothetical control group consisted of the minimum number of BIO and interhospital transfers if the existing patients were screened in person. Costs included in-person examinations, transfers, setting up, and ongoing costs of telemedicine screening. Costs were compared using the Mann-Whitney U test and are reported in 2014 Canadian dollars. RESULTS: A total of 102 and 72 infants were screened from Sudbury and Barrie, respectively; 3% and 2% of infants in the telemedicine group were transferred for BIO from Sudbury and Barrie, respectively. All infants in the control group would have required at least one transfer for BIO. The average total cost per eye examination was $4855 ± $5616 and $4540 ± $3129 for the telemedicine group and $19 834 ± $13 814 and $2429 ± $1664 for the control group from Sudbury and Barrie, respectively (p < 0.001). Interhospital transfer cost for the control group was $19 489 ± $13 605 and $2055 ± $1471 compared to $635 ± $3968 and $30 ± $197 for the telemedicine group (p < 0.001) in Sudbury and Barrie, respectively. CONCLUSIONS: Telemedicine appears to be an economically attractive option depending on the location and number of infants screened. This information is useful for planning similar ROP screening programs.


Assuntos
Programas de Rastreamento/economia , Triagem Neonatal/economia , Retinopatia da Prematuridade/diagnóstico , Telemedicina/métodos , Custos e Análise de Custo , Feminino , Idade Gestacional , Humanos , Incidência , Recém-Nascido , Masculino , Triagem Neonatal/métodos , Ontário/epidemiologia , Oftalmoscopia/economia , Oftalmoscopia/métodos , Retinopatia da Prematuridade/economia , Retinopatia da Prematuridade/epidemiologia , Estudos Retrospectivos
18.
J Pediatr Gastroenterol Nutr ; 66(6): 845-849, 2018 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-29570556

RESUMO

BACKGROUND AND OBJECTIVES: Biliary atresia (BA), a rare newborn liver disease, is the leading cause of liver-related death in children. Early disease recognition and timely surgical Kasai hepatoportoenterostomy (KP) offers long-term survival without liver transplant. Universal BA screening in Taiwan using infant stool color cards (ISCCs) has proven effectiveness. We report our experience with infant stool color card (ISCC) BA screening in a province-wide program in British Columbia (BC). The objective of this study is to assess program performance and cost from launch April 1, 2014 to March 31, 2016. METHODS: ISCCs distributed to families upon maternity ward discharge. Parents were instructed to monitor their infant's stool color for 1 month and contacted the screening center with concerns. The number of live births, ISCC distribution, BA cases, and costs were recorded. Cases with Program screen success had both acholic stool recognition (ISCC screen success) and timely referral for BA. RESULTS: All 126 maternity units received ISCCs. Of 87,583 live births there were 6 BA cases. Of the 5 cases with ISCC Screen Success 3 had Program Screen Success. The median KP age in the program screen success and failure groups was 49 (42-52) and 116 (49-184) days, respectively. Program sensitivity was 50%, specificity 99%, positive predictive value 4%, and negative predictive value 99%. A random sample of 1054 charts at BC Children's Hospital found an ISCC distribution rate of 94%. After a phase-in period, the annual program cost was $30,033.82, and the ISCC cost per birth was $0.68. CONCLUSIONS: The screening program has high specificity and distribution with low cost. Successful program case identification had earlier age at KP. Program modifications aim to improve sensitivity. Longer-term studies will determine program impact on health outcomes.


Assuntos
Atresia Biliar/diagnóstico , Triagem Neonatal/métodos , Atresia Biliar/economia , Atresia Biliar/cirurgia , Colúmbia Britânica , Análise Custo-Benefício , Fezes , Feminino , Custos de Cuidados de Saúde , Humanos , Recém-Nascido , Masculino , Triagem Neonatal/economia , Portoenterostomia Hepática , Avaliação de Programas e Projetos de Saúde , Sensibilidade e Especificidade
19.
J Cyst Fibros ; 17(3): 306-315, 2018 05.
Artigo em Inglês | MEDLINE | ID: mdl-29572018

RESUMO

BACKGROUND: Early detection of cystic fibrosis through newborn screening has significant clinical benefits. Cost-effectiveness plays an important role in selecting the optimal screening strategy from the many available options. OBJECTIVES: The objectives of this study are (1) to summarize study estimates of cost-effectiveness of cystic fibrosis newborn screening (CFNBS) strategies as compared to other strategies, (2) to assess the quality of the studies identified, and (3) to identify determinants of cost-effectiveness. METHODS: Electronic databases were searched from 2007 to June 2017. Health economic evaluations describing the cost-effectiveness of two or more CFNBS strategies were included. RESULTS: Six health economic evaluations were found. Where included in the comparison, IRT/PAP consistently was the most cost-effective strategy in terms of cost per case detected or life years gained. However, some heterogeneity with respect to cut-off values used and the number of DNA mutations included in the screening strategies was observed, and the methodological quality differed considerably between studies. CONCLUSIONS: The evidence suggested that (i) all screening strategies are cost-effective as compared to the no-screening option and (ii) IRT-PAP seems to be the most cost-effective screening strategy towards CFNBS. Methodological and contextual differences of the individual studies make it difficult to derive strong conclusions from this evidence. Nevertheless, from a health-economic perspective, IRT-PAP should be included as an alternative when deciding on the screening strategy in the implementation of CFNBS.


Assuntos
Fibrose Cística/diagnóstico , Triagem Neonatal , Análise Custo-Benefício , Humanos , Recém-Nascido , Triagem Neonatal/economia , Triagem Neonatal/métodos
20.
Clin Exp Ophthalmol ; 46(6): 645-651, 2018 08.
Artigo em Inglês | MEDLINE | ID: mdl-29369477

RESUMO

IMPORTANCE: Demand for retinopathy of prematurity (ROP) screening is increasing for infants born at rural and regional hospitals where the service is not generally available. The health system cost for screening regional/remote infants has not been reported. BACKGROUND: The objective of this study is to evaluate the cost of ROP screening at a large centralized tertiary neonatal service for infants from regional/rural hospitals. DESIGN: This is a retrospective study to establish the cost of transferring regional/rural infants to the Royal Brisbane and Women's Hospital for ROP screening over a 28-month period. PARTICIPANTS: A total of 131 infants were included in this study. METHODS: Individual infant costs were calculated from analysis of clinical and administrative records. MAIN OUTCOME MEASURES: Economic cost of ROP screening for all transfers from regional/rural hospitals to Royal Brisbane and Women's Hospital. RESULTS: The average economic cost of ROP screening for this cohort was AUD$5110 per infant screened and the total cost was AUD$669 413. The average cost per infant screened was highest for infants from a regional centre with a population of 75 000 (AUD$14 856 per child), which was also geographically furthest from Brisbane. No infant in this cohort transferred from a regional nursery reached criteria for intervention for ROP by standard guidelines. CONCLUSIONS AND RELEVANCE: Health system costs for ROP screening of remote infants at a centralized hospital are high. Alternative strategies using telemedicine can now be compared with centralized screening.


Assuntos
Custos de Cuidados de Saúde , Triagem Neonatal/economia , Retinopatia da Prematuridade/epidemiologia , População Rural , Telemedicina/métodos , População Urbana , Custos e Análise de Custo , Feminino , Seguimentos , Idade Gestacional , Humanos , Incidência , Recém-Nascido , Masculino , Oftalmoscopia , Queensland/epidemiologia , Reprodutibilidade dos Testes , Retinopatia da Prematuridade/diagnóstico , Retinopatia da Prematuridade/economia , Estudos Retrospectivos
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