110Chapter 7In the early 2000s, our research group performed economic evaluations oftreatment of congenital anorectal malformations (ARM) and congenitaldiaphragmatic hernia (CDH). 24-26 The results may briefly be summarized asfollows. The direct medical costs were found to be considerable, with long-termcosts after the initial treatment being only a fraction of these costs. HRQoL inpatients once treated for ARM and now between 1 and 4 years of age was lowerthan that of the general population. The older children showed better HRQoL.Although surgery for ARM was associated with substantial residualsymptomatology, patients aged 16 years and over showed hardly any HRQoLdifferences with population standards. We found CDH to be still associated withhigh mortality rates, despite the many advances in treatment over recentdecades. Although respiratory difficulties and stomach aches were reported,HRQoL in CDH survivors did not differ significantly from that of the generalpopulation. Costs per quality-adjusted life year (QALY) amounted to € 2,482 forARM and € 2,434 for CDH treatment.How to interpret cost-effectiveness ratios?Seeing that cost-effectiveness ratios like these are not very informative inthemselves, we need to bring in figures from other health care programs forcomparison to draw meaningful conclusions. Put in the perspective of the manytimes higher cost-effectiveness thresholds that seem to guide public authorities indecisions to accept or reject a technology, the costs per QALY we calculated forARM (€ 2,482) and CDH (€ 2,434) are clearly very modest. Examples of suchthresholds are those of the UK National Institute for Clinical Excellence (about£ 35,000 or £ 40,000 per QALY, which equals € 52,000 or € 60,000 at currentexchange rates) 27 or the Australian Pharmaceutical Benefit Advisory Committee(between AU$ 42,000 and AU$ 76,000 per life-year, which is approximately€ 27,000 and € 48,000). 28 In the Netherlands, a threshold of approximately€ 18,000 per life-year is sometimes seen as an acceptable cost-effectivenessratio. 29In the context of this chapter, we shall assume that our encouraging results alsoapply to surgical treatment of other congenital anomalies. This assumption seemsfairly plausible, since ARM and CDH were not selected randomly but purposefullychosen as being opposite extremes in terms of mortality and morbidity. ARM ischaracterized by relatively low mortality and relatively high morbidity intoadulthood, whereas for CDH the opposite applies. Notwithstanding the diversity ofthe conditions, treatments are remarkably close in terms of cost-effectiveness.So, treatments for other 'isolated' anomalies such as Hirschsprung's disease andesophageal atresia are likely to be cost-effective as well. Because most neonateshaving to undergo surgery present with just one major anomaly, the overallconclusions drawn from our research are favorable for neonatal surgery ingeneral.
Cost-Effectiveness of Neonatal Surgery: First Skepticized, Now Increasingly Accepted 111The 2000 review updatedWe updated the earlier review cited above 18 to see whether recent years havebrought more empirical results. We searched the literature from the year 1999onwards for complete economic evaluations of both diagnostic and therapeuticinterventions in neonatal surgery. In spite of the fact that we applied liberalcriteria for inclusion—several studies, for example, only evaluated postoperativecomplication rates rather than more advanced patient outcome measures—weidentified no more than 11 relevant studies, including our economic evaluations inARM and CDH referred to earlier. Even though our review covered just a fewyears and only included studies published in English, the conclusion can only bethat the number of published economic evaluations in the field of neonatalsurgery is still small. Table 7.1 details the interventions investigated in all thesestudies, differentiating between the six groups of congenital anomalies listed byRavitch and colleagues 30 and other conditions. The authors predominantly drawfavorable conclusions on cost-effectiveness. What attracts attention is that,leaving our studies on ARM and CDH out of consideration, all studies except one(dealing with circumcision) concentrated on particular novel aspects of treatmentor diagnosis. So, drawing more conclusions on the entire treatment process ofneonatal surgery, next to our studies on ARM and CDH, is not possible.7.4 IS THERE A ROLE FOR ARGUMENTS OTHER THAN COST-EFFECTIVENESS?Above, we argued that it is not necessarily unethical to consider costeffectivenessarguments. However, we feel it would be unethical indeed to basedecisions on how much priority to give to a particular health care procedure oneconomic grounds exclusively. In the case of neonatal surgery, crucial ethicalquestions arise for example when considering whether therapy should be offeredor perhaps withheld, which often involve life-and-death decisions. In the case ofchildren with serious birth defects, the 'best interest' of the child may justifywithholding or withdrawing life-prolonging surgical intervention. There may befundamental uncertainties as to the child's future development, or in somesituations, a life strongly burdened by disability and dependency on life-supportsystems may be the ultimate outcome. 31-33 Treatment modalities that mayinvolve such ethical dilemmas include ECMO, the care for extremely prematureinfants, and prenatal diagnosis and fetal surgery. 34-38 Apart from ethicalarguments, religious, cultural, aesthetic, or legal aspects need to be considered inneonatal surgery, such as a child's right to be free of intrusive unnecessarymedical and surgical procedures before having reached the age of full and legaldiscretion (e.g., neonatal circumcision 39 ).
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DOCTORAL COMMITTEEPromotors:Prof.dr
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PUBLICATIONSChapters 2 to 7 are bas
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6ChapterINFORMAL CARE FOR CHILDREN
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2Chapter 11.1 BACKGROUND AND MOTIVA
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4Chapter 1provides a good overview
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6Chapter 1disability, and death of
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Introduction 9particular equity pri
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Introduction 1320. Oostenbrink JB,
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Introduction 1554. Heyman MB, Harma
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THE COST-EFFECTIVENESS OFTREATMENT
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Cost-Effectiveness of Treatment for
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Cost-Effectiveness of Treatment for
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Cost-Effectiveness of Treatment for
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Cost-Effectiveness of Treatment for
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Cost-Effectiveness of Treatment for
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Cost-Effectiveness of Treatment for
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Cost-Effectiveness of Treatment for
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36Chapter 3ABSTRACTBackground/Purpo
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38Chapter 33.2 MATERIALS AND METHOD
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40Chapter 3child's date of birth) a
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42Chapter 3diaphragm was closed (Ta
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44Chapter 3Total costs of treatment
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46Chapter 3Regarding the treatment
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48Chapter 3REFERENCES1. Stolk EA, P
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50Chapter 332. Jaillard S, Pierrat
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52Chapter 4ABSTRACTAims:To examine
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54Chapter 4Outcome measuresThe pati
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56Chapter 4Clearly, the symptoms st
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58Chapter 4The respondents did not
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- Page 98 and 99: 86Chapter 568. Hui TT, Danielson PD
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162AcknowledgmentsAlthough I am ind
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About the AuthorBorn in De Meern (N