RESUMEN
OBJECTIVE: To collect information for the purpose of establishing starting points and possibilities for a cost-effectiveness analysis of screening for adolescent idiopathic scoliosis (AIS). DESIGN: Interviews, literature review, questionnaires, an estimation of costs and discussions with experts and involved parties. METHOD: Following an initial interview with 16 orthopaedic surgeons and school doctors a literature study into the efficacy of treatment was carried out regarding the years 1989-1999. The variation in current practice was delineated by means of a questionnaire sent to all 51 municipal health services in the Netherlands. The costs of screening and treatment were estimated on the basis of health insurance premiums and a municipal health service cost model. All of the results were presented to five methodological experts and finally the study results and the recommendations of the five methodological experts were evaluated during a meeting of persons especially invited for this purpose. RESULTS: Screening for AIS was established to realise early diagnosis and treatment with a brace, so as to reduce unsatisfactory cosmetic outcomes and the need for surgery. Screening was performed using the bending test and was performed in 40/48 (83%) of the participating municipal health services. The overall costs of screening and treatment amount to 6 million euros per year. There was no convincing evidence that the screening programme was sufficiently sensitive and bracing sufficiently effective. Neither was there proof of the opposite. The following was recommended: obtain reliable data by carrying out a randomised controlled trial on the effectiveness of treating AIS with bracing in an early stage; carry out a case-control study combined with a retrospective patient follow-up study to evaluate the current screening practice; draw up a national standard for the screening of postural disorders in youth healthcare to ensure effective practice.
Asunto(s)
Tirantes/economía , Tamizaje Masivo/economía , Escoliosis/diagnóstico , Escoliosis/terapia , Adolescente , Análisis Costo-Beneficio , Humanos , Entrevistas como Asunto , Países Bajos , Estudios Retrospectivos , Escoliosis/economía , Índice de Severidad de la Enfermedad , Encuestas y Cuestionarios , Resultado del TratamientoRESUMEN
BACKGROUND: Reports on increasing hypospadias trends are based on birth defect registries, which are prone to inaccuracy. We assessed the prevalence of hypospadias precisely, by prospective examination of all newborns in Rotterdam over a 2-year period. METHODS: A total of 7292 consecutive male births were examined for the presence of hypospadias, classified by severity. RESULTS: The frequency of hypospadias in newborn boys was 0.73% (53/7292). The rate among live births was 38 per 10 000, which is 6 times the previously reported rate for the Southwestern Netherlands (6.2) (P < 0.0001). This registry excludes glandular hypospadias. Without glandular cases, our rate is 26 per 10 000, which is still 4-fold higher (P < 0.0001). The ratio of minor to major hypospadias was 0.3. In 79% of cases, surgery was indicated. CONCLUSIONS: We found a 4-fold higher than expected hypospadias rate, which may be explained by case ascertainment differences. The proportion of major cases was higher than generally assumed. This study provides evidence for substantial geographical differences. Explanations for temporal and geographical differences need to be explored. To monitor hypospadias rates and trends accurately, complete case ascertainment, including standardized classification of severity, is warranted.
Asunto(s)
Hipospadias/epidemiología , Estudios Transversales , Humanos , Recién Nacido , Masculino , Países Bajos/epidemiología , Prevalencia , Índice de Severidad de la EnfermedadRESUMEN
OBJECTIVE: To discuss the merits of the patient follow up study design for the evaluation of some specific mass screening programmes. DESIGN: Theoretical evaluation illustrated by two examples. SETTING: Department of Public Health Erasmus University Rotterdam. MAIN RESULTS: The gold standard for evaluation of favourable effects of screening is the randomised controlled trial (RCT). Application of an RCT, however, is often not feasible, in which cases observational studies will have to be relied on. The case-control study design is generally considered to be second best. In some situations, however, a patient follow up study design may be applicable and may have some major advantages. The use of the patient follow up design for screening evaluation will often be very problematic or even unacceptable, particularly as far as screening for cancer is concerned. The most important objections are resulting from lead time bias, length bias, selection bias and over-treatment bias. For the evaluation of screening for congenital heart disease and congenital hip dislocation in Dutch child health care, however, these objections may relatively simply be overcome. Lead time bias will be of little importance, as the ages of onset of these disorders are fixed, namely at birth, and their ultimate outcomes may be expected within relatively short time. Length bias may largely be avoided by correction for severity of the disorder, which can be adequately assessed by modern diagnostic procedures. Selection bias is generally hard to rule out, but in these cases it probably plays a minor part. Over-treatment can be avoided by the policy of "watchful waiting", which in these disorders can be applied with little risk for fatal outcomes. In principle bias might be avoided more successfully in a case-control screening evaluation than in a patient follow up study. However, the patient follow up study is for both screening programmes discussed here the more feasible design and can provide more supplementary information. The results of two example studies suggest that both screenings probably yield considerable benefits CONCLUSION: Under a number of specific conditions a patient follow up study is an efficient alternative to more customary designs for screening evaluation.
Asunto(s)
Cardiopatías Congénitas/diagnóstico , Luxación Congénita de la Cadera/diagnóstico , Tamizaje Masivo/métodos , Niño , Protección a la Infancia , Estudios de Seguimiento , Mal Uso de los Servicios de Salud , Humanos , Países Bajos , Evaluación de Programas y Proyectos de Salud , Sesgo de SelecciónRESUMEN
BACKGROUND: The actual yield from current screening for clinically significant congenital heart malformations in Dutch child health care is far from optimal. In this study factors that determine the effectiveness of this screening are identified and recommendations for the optimization of the screening programme are formulated. METHODS: Eighty-two patients with a clinically significant congenital heart malformation were consecutively included in this study. Parents and child health centre physicians were interviewed in order to establish the screening, detection and referral history. Paediatric cardiologists established whether these patients were diagnosed 'in time' or 'too late'. RESULTS: Incomplete performance of the screening examination has more influence on the occurrence of delayed diagnoses than failure by parents to adhere to the complete visit schedule. Adequate screening advances detection of congenital heart malformations. Severity, however, is the most predominant determinant of the age at referral and diagnosis, as well as of the risk of complications. In only 7 out of 39 patients diagnosed 'too late', could no avoidable cause for an adverse outcome be found. In 10 cases (25%) there was a prolonged interval between first referral and diagnosis. CONCLUSION: To optimize the yield of the screening programme, improvement in the performance of the child health centre physicians and the co-operation of other physicians involved in reducing the interval between referral and diagnosis are required. Thus a considerable improvement in the prevention of complications of congenital heart malformations can be obtained.
Asunto(s)
Servicios de Salud del Niño , Cardiopatías Congénitas/prevención & control , Tamizaje Masivo , Calidad de la Atención de Salud , Preescolar , Humanos , Lactante , Modelos Logísticos , Países Bajos , Oportunidad Relativa , Derivación y Consulta , Índice de Severidad de la Enfermedad , Factores de TiempoRESUMEN
BACKGROUND: Although screening for congenital heart malformations is part of the child health care programme in several countries, there are very few published evaluations of these activities. This report is concerned with the evaluation of this screening at the Dutch Child Health Centres (CHC). METHODS: All consecutive patients, aged between 32 days and 4 years, presented at the Sophia Children's Hospital Rotterdam throughout a period of 2 years, with a congenital heart malformation were included in this study. Paediatric cardiologists established whether or not these patients were diagnosed after haemodynamic complications had already developed (diagnosed 'too late'). Parents and CHC-physicians were interviewed in order to establish the screening and detection history. Test properties were established for all patients with a congenital heart malformation (n = 290), intended effects of screening were established in patients with clinically significant malformations (n = 82). RESULTS: The sensitivity of the actual screening programme was 0.57 (95% CI : 0.51-0.62), the specificity 0.985 (95% CI : 0.981-0.990) and the predictive value of a positive test result 0.13 (95% CI: 0.10-0.19). Sensitivity in a subpopulation of patients adequately screened was 0.89 (95% CI: 0.74-0.96). Adequately screened patients were less likely to be diagnosed 'too late' than inadequately screened patients (odds ratio [OR] = 0.20, 95% CI: 0.04-1.05). The actual risk of being diagnosed 'too late' in the study-population (48%) was only slightly less than the estimated risk for patients not exposed to CHC-screening (58%, 95% CI: 43%-72%). Adequately screened patients however were at considerably less risk (17%, 95% CI: 4%-48%). CONCLUSION: Screening for congenital heart malformations in CHC contributes to the timely detection of these disorders. The actual yield, however, is far from optimal, and the screening programme should be improved.
Asunto(s)
Servicios de Salud del Niño/estadística & datos numéricos , Cardiopatías Congénitas/diagnóstico , Hospitales Pediátricos/estadística & datos numéricos , Preescolar , Estudios de Seguimiento , Cardiopatías Congénitas/epidemiología , Humanos , Incidencia , Lactante , Países Bajos/epidemiología , Pronóstico , Estudios Retrospectivos , Sensibilidad y EspecificidadRESUMEN
BACKGROUND: Second trimester routine ultrasound evaluation of the fetal heart by means of the four-chamber view has been proposed for prenatal detection of cardiac anomalies. The aim of this study was to evaluate the efficacy of this procedure. METHODS AND RESULTS: A prospective follow-up study on 6922 scanned fetuses was performed. Pregnant women without known risk factors who were scheduled for a routine fetal ultrasound examination between 16 and 24 weeks gestation were invited to participate. Follow-up until 6 months postpartum was available for 5660 subjects (81.8%), of whom 5319 fulfilled all eligibility criteria. by comparing the prenatal diagnosis to the postnatal diagnosis, we obtained sensitivity, specificity, and predictive value (positive and negative). A total of 80 cases of congenital malformations were diagnosed during the study: 44 cases of congenital heart disease, 40 cases of noncardiac malformations, and a combination of the two in 4 cases. The fetal four chamber-view examination was considered abnormal in 7 women who were subsequently referred for extensive fetal ultrasound examination. Two proved to be carrying an affected fetus. Similarly, prenatal referral of 14 women because of suspected noncardiac malformations yielded 12 such cases. The fetal four chamber-view examination had a sensitivity of 4.5% (95% CI, 0.6% to 15%). Sensitivity for noncardiac anomalies was 30% (95% CI, 16.6% to 46.5%). Overall sensitivity of ultrasound examination was 16.3% (95% CI, 2.09% to 48.8%). Specificity and negative predictive value were high (>98%). The positive predictive value was low with wide CIs. CONCLUSIONS: These results suggest that the current mode of routine prenatal ultrasound screening for congenital malformations is inefficient, particularly for cardiac anomalies.
Asunto(s)
Ecocardiografía , Cardiopatías Congénitas/prevención & control , Tamizaje Masivo , Ultrasonografía Prenatal , Adolescente , Adulto , Estudios de Evaluación como Asunto , Femenino , Humanos , Persona de Mediana Edad , Embarazo , Resultado del Embarazo , Valores de ReferenciaRESUMEN
OBJECTIVE: A summary of research data on the evaluation of eight screening activities in Child Health Care in the Netherlands. DESIGN: Literature review. SETTING: Department of Public Health, Erasmus University Rotterdam, the Netherlands. METHOD: The literature was reviewed regarding screening effects on congenital hip pathology, congenital heart disease, retained testis, hydrocephalus, hearing loss, psychomotor retardation, visual disabilities, and phenylketonuria (PKU) and congenital hypothyroidism (CHT). RESULTS: For 8 types of screenings 17 targets could be established. Targets were defined in terms of a decrease of the adverse outcome of the diseases as a result of the screening. The efficacy of the screening for 3 of these targets (decrease of the adverse outcomes of PKU and CHT and needless orchidopexy) was convincingly established. For the other targets the available data were insufficient. Data on undesirable effects of screening were scarce. For 6 of the 8 screening activities some data on test properties were available. CONCLUSION: Research data on the effects of screening in Child Health Care in the Netherlands are incomplete. It is important to improve the scientific basis of these prevention activities.
Asunto(s)
Anomalías Congénitas/diagnóstico , Adulto , Discapacidades del Desarrollo/diagnóstico , Femenino , Trastornos de la Audición/diagnóstico , Cardiopatías Congénitas/diagnóstico , Luxación Congénita de la Cadera/diagnóstico , Humanos , Hidrocefalia/diagnóstico , Lactante , Recién Nacido , Centros de Salud Materno-Infantil , Errores Innatos del Metabolismo/diagnóstico , Países Bajos , Trastornos de la Visión/diagnósticoRESUMEN
Data on body composition in conjunction with reference centiles are helpful in identifying the severity of growth and nutritional disorders in infancy and for evaluating the adequacy of treatment given during this important period of rapid growth. Total body fat (TBF) and fat-free mass (FFM) were estimated from total body electrical conductivity (TBEC) measurements in 423 healthy term Caucasian infants, aged 14-379 days. Cross sectional age, weight, and length related centile standards are presented for TBF and FFM. Centiles were calculated using Altman's method, based on polynomial regression and modelling of the residual variation. The TBF percentage steeply increased during the first half year of life, and slowly declined beyond this age. Various simple TBEC derived anthropometric prediction equations for TBF and FFM are available to be used in conjunction with these standards. Regression equations for the P50 and the residual SD, depending on age, weight, or length, are provided for constructing centile charts and calculating standard deviation scores.
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Tejido Adiposo/crecimiento & desarrollo , Composición Corporal , Envejecimiento/fisiología , Estatura/fisiología , Peso Corporal/fisiología , Conductividad Eléctrica , Femenino , Crecimiento , Humanos , Lactante , Masculino , Valores de Referencia , Caracteres SexualesRESUMEN
Anthropometry is frequently used for nutritional assessment. Little is known in infants about the validity of anthropometric measurements in relation to whole-body fat (TBF) and fat-free mass (FFM) composition. We compared TBF and FFM estimations by total-body electrical conductivity (TOBEC) with anthropometry in 435 healthy infants ages 21-365 d. TBF was best correlated with weight-for-length and calf circumference (r2 = 0.84, r2 = 0.83). FFM was best correlated with body weight (r2 = 0.93). Upper-arm anthropometry, skinfold thickness, and Quetelet's and Ponderal indexes were poorly correlated with TBF and FFM (r2 < 0.65). New anthropometry-based prediction equations were calculated (r2 = 0.90 for TBF and r2 = 0.95 for FFM). New simple indexes (analogous to Quetelet's index) were calculated for TBF (weight x calf circumference/length; r2 = 0.87) and for FFM (square root of weight x length; r2 = 0.95). Prediction equations and indexes were cross-validated in a second population by a second observer. Interobserver variation was largest for equations with skinfold thicknesses included. We conclude that anthropometry can be used for rough estimations of infant body composition, although indexes different than those used in children and adults are preferred.
Asunto(s)
Antropometría , Composición Corporal , Tejido Adiposo , Conductividad Eléctrica , Femenino , Humanos , Lactante , Recién Nacido , Masculino , Estado Nutricional , Valor Predictivo de las Pruebas , Análisis de RegresiónRESUMEN
Measurement of total-body electrical conductivity (TOBEC) has emerged as a rapid, safe, and reproducible method for estimation of infant total body fat (TBF). Agreement of two anthropometric methods [by Dauncey et al (1977) and Weststrate et al (1989)] with TOBEC-TBF was assessed in 435 healthy infants aged 21-365 d. Dauncey-TBF correlated with TOBEC-TBF by r2 = 0.61 and exceeded TOBEC-TBF by 0.14 +/- 0.25 kg in infants < 4 mo of age. Thereafter, TOBEC-TBF exceeded Dauncey-TBF by 0.20 +/- 0.47 kg. We modified Dauncey's method, which significantly improved the correlation to r2 = 0.75. Weststrate-TBF correlated with TOBEC-TBF by r2 = 0.87, but exceeded TOBEC-TBF by 0.5 kg. Both methods showed poor agreement with TOBEC-TBF. We conclude that both methods, although suitable for comparison of TBF between groups, cannot be used to accurately assess TBF in an individual infant.
Asunto(s)
Tejido Adiposo , Composición Corporal , Antropometría/métodos , Conductividad Eléctrica , Femenino , Humanos , Lactante , Masculino , Factores Sexuales , Grosor de los Pliegues CutáneosRESUMEN
In 79 children treated in the Sophia Children's Hospital in Rotterdam because of imminent limping due to congenital dislocation of the hip, the contribution of screening tests by physicians of Infant Health Centres to the timely discovery of this disease was examined. When both limitation of abduction of the hip and difference in length of the legs are examined 6 times in the period of 1-8 months of age, the following results in relation to children not or less well screened may be expected: The risk of discovery of the disease at an age when surgery is not excluded, is reduced to one-third. As a result of the shorter delay between discovery and treatment in practice the risk of having to undergo surgery is halved. The risk of discovery at an age when osteotomy according to Salter is required, is nil. In practice the risk of having to undergo osteotomy is reduced to one quarter. Lowering the frequency of the screening tests reduces the contribution. Examination of just limitation of abduction of the hip also appears to reduce the contribution.