38Chapter 33.2 MATERIALS AND METHODSThere is a close relationship between this study on the cost-effectiveness oftreatment for CDH and our study in ARM patients. For the purpose ofcomparability, the methodologies of both studies were replicated as far as it waspossible. We, therefore, have restricted ourselves to a brief description of ourmethodology, except for discrepancies between the 2 studies. For a more detaileddescription, please refer to Chapter 2.The data were collected in 1997, 1998, and 1999. Patients included underwenttreatment for CDH in the Sophia Children's Hospital in <strong>Rotterdam</strong> after 1969. Inprinciple, all patients until 1986 were treated by emergency surgery to close thediaphragmatic defect, followed by preoperative stabilization and delayed repairfrom that time onward. 12 ECMO was applied in the hospital from January 1992.Treatment of pulmonary hypertension with inhaled nitric oxide was not availableduring the study period. Patients born after 1996 were excluded to ensure aminimal follow-up period. Two patients were excluded because their CDH was notdiagnosed during the neonatal period. To examine to what extent respiratorydifficulties, stomach aches, and other disease-specific symptoms really areconsequences of CDH, we recruited a control group of children who visited theday care department in 1998 for minor day-case surgery.As with our study on ARM, the current study was carried out from a societalperspective using the technique of cost-utility analysis. Accordingly, the outcomesare presented in costs per quality-adjusted life year (QALY). 13 Both costs andeffects basically were analyzed in a life-time setting.Treatment comparatorIn economic evaluations of health care programs, treatments or diagnoses arecompared with one or more alternatives. Ideally, the alternative is the most costeffectivealternative treatment. More often, the studied program is compared withthe most common treatment (i.e., the existing, local practice). For CDH, however,there is no realistic alternative. Therefore, the costs and effects had to becompared with 'no treatment'. In this scenario, all diagnoses or treatments arewithheld; hence, no costs are generated. Furthermore, we assumed that in thisscenario, the inability to obtain sufficient oxygen uptake because of theunderlying lung anomalies would bring about death shortly after birth.CostsIdentification, measurement, and valuation of the costs. Direct medical costsmake up the health care cost incurred between the child's birth and the end ofthe hospital admission during which the defect in the diaphragm was closed.These costs were calculated only for patients born between 1993 and 1996,because computerized data on previous patients were lacking. Consistent with themethods outlined in Chapter 2 we calculated real costs of hospital days,
Cost-Effectiveness of Treatment for Congenital Diaphragmatic Hernia 39laboratory tests, diagnostic radiology, surgeries, intercollegial consultations, andvisits to the outpatient department. There is, however, one difference with ourprevious study. Costs of ECMO were calculated as part of the direct medical costs.These costs, i.e., the additional costs over the costs of a 'standard' day in anintensive care unit, were taken from a large Dutch survey on ECMO in neonates. 14Direct nonmedical costs are costs incurred outside the health care sector thataccrue to patients and their families while receiving health care. These costs werelimited to transportation costs regarding visits to health care providers.Indirect medical costs are the health care costs made after the period that refersto the direct medical costs. Costs of hospital admissions, visits to the outpatientdepartment, consultations with a general practitioner, and consultations with aphysiotherapist are all part of these costs. No reliable data were available oncosts of visits to alternative healers, use of medication outside the hospital, anduse of medicinal oxygen at home. We excluded future health care costs ofunrelated diseases.Indirect nonmedical costs are costs associated with production loss andreplacement owing to illness, disability, and death of productive persons, bothpaid and unpaid. 15 As we adopted a societal perspective, both productivitychanges in patients and in their parents or caregivers were included.EffectsIdentification, measurement, and valuation of the effects. We repeated themethodology outlined in the previous chapter regarding the effects. 4 In summary,we calculated both the life expectancy and the health-related quality of life. Weassumed that patients who survived the first few years would have a normal lifeexpectancy (75.4 years at birth 16 ). The life span of the deceased patients alsowas determined. On the basis of these data, the mean life expectancy wascalculated. Using the EQ-5D questionnaire, 17 we were then able to calculate theQALY gains. Descriptive quality-of-life data were collected using a disease-specificquestionnaire that aimed to measure respiratory difficulties, stomach aches, andother disease-specific symptoms. A symptom score of the most relevant clinicalitems was constructed, ranging from 0 to 45 (maximum number of symptoms).Medical consumption also was considered an indication of the quality of life.Generally, patients younger than 16 years did not fill in the questionnairesthemselves. Their parents were used as proxies. The disease-specificquestionnaire was the only exception. Here, the age limit was put at 12 years.Cost-effectivenessIncremental costs per QALY of treatment for CDH compared with 'no treatment'were calculated. Both future costs and future effects were discounted (to the
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100Chapter 6This study was of impor
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102Chapter 618. Poley MJ, Stolk EA,
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106Chapter 7ABSTRACTMortality rates
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108Chapter 77.2 THE RELEVANCE OF CO
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Summary 149mortality. Finally, it i
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Summary 151neonatal surgery. It is
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Samenvatting 155zoals directe niet-
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162AcknowledgmentsAlthough I am ind
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About the AuthorBorn in De Meern (N