Marten J. Poley - Erasmus Universiteit Rotterdam

Introduction 7developmentally programmed to occur years if not decades after theintervention. 46 To take just one example, it requires a follow-up of more than twodecades to see whether early surgery for undescended testes has a favorableeffect on fertility.The position of the parents. A consequence of taking a societal perspective isthat the position of the parents should not be ignored in cost-effectivenessstudies of neonatal surgery. Major health problems in infants affect the wholefamily. In a small-scale study from Norway for example, 59% of the parents ofchildren with 'low' ARM and 23% of the parents of children with Hirschsprung'sdisease reported that their child's malformation had a negative influence on theirmarital relationship and family life. Fifty-three percent of the parents of childrenwith a 'low' ARM recalled that they became isolated from their family, friends, andsocial activities. 47Again it is important to precisely distinguish between different outcomemeasures. To assess the effects of providing so-called 'informal care' to childrensuffering from congenital anomalies, there are various options, such as measuringgeneral effects on the caregivers' wellbeing, marital and life satisfaction, broadlydefinedquality of life, burden of informal care, or HRQoL. Earlier studies inchildren with a chronic illness or disability mainly focused on the burden thatinformal caregiving may cause. Hassink et al. for example investigated stress inparents of children with ARM and found that especially the parents of older boyswho were incontinent for feces experienced stress when caring for their child. 48Such studies also demonstrated that impacts on the family differ according to thetype of condition. 49,50 Yet, present understanding of the effects of caregiving onparents' multidimensional HRQoL is still rudimentary in neonatal surgery. It isexactly this outcome measure however that seems to have great value to informcost-effectiveness analysis. HRQoL represents an overall outcome measure,comprises a valuation of some type, may be measured by using instrumentssimilar to the ones used to assess patients' HRQoL, and closely matches the maingoal of health care (i.e., to preserve or restore health). Despite these appealingfeatures, the full potential and limitations of the HRQoL outcome measure inparents are not yet fully understood.Like the effects on the parents, the costs of providing informal care are presentlyoften ignored in economic evaluations. 33,51 Yet, it is increasingly beingacknowledged that a child with a chronic health condition requires additionalcaregiver time. 52 Studies in parents of children with atopic dermatitis, childrenwith cystic fibrosis, and gastrostomy-dependent children showed that caregivingtakes up significant amounts of time, reducing the time available for otheractivities such as work or recreation, and that it may be associated with high outof-pocketexpenses. 53-55 However, evidence in neonatal surgery is largely lacking.Obviously, the costs that fall to the caregivers, such as transportation costs