Marten J. Poley - Erasmus Universiteit Rotterdam

6Chapter 1disability, and death of productive persons, both paid and unpaid. 34 Whichmethod to use for measuring these costs (the so-called friction cost or the humancapital cost approach) has long been a subject of debate, 35-38 and is still notsettled. 39,40 Productivity costs can be substantial, especially when the humancapital approach is followed.Both these cost categories—i.e., indirect healthcare costs and indirect nonhealthcarecosts—can have a large impact on cost-effectiveness ratios. This isundeniably also true for cost-effectiveness ratios of neonatal surgery. Exactlythese often ignored cost categories are potentially major drivers of the results inneonatal surgery.Effects. In neonatal surgery, avoided mortality has been a widely acceptedmeasure of success since many years. Furthermore, the emphasis has been onthe immediate outcome of surgery. More often than not, the immediate outcomeis satisfactory in all respects, with alleviation of the symptoms, cure of theprimary pathology, and a rapid return to a normal pattern of life. As aconsequence, little thought has been given to the possibility that at a later stageproblems may develop. 41 Of course, on the face of it, certain surgical diseases ofthe neonate seem unlikely to require any form of long-term follow-up (such asumbilical hernia, acute appendicitis, or circumcision), whereas others seem to callfor ongoing follow-up (such as esophageal atresia, Hirschsprung's disease, etc.).Similarly, within a specific diagnosis there can be a group that does well and agroup that does badly. A good example is the difference in outcome between 'low'and 'high' congenital anorectal malformations (ARM). Yet, research to supportthese perceptions is highly needed. This type of research is not only necessarywhen performing cost-effectiveness analyses, but will also help to predict theoutcome and as such can be used to inform the parents about the prospects fortheir child.It should be emphasized here that it is essential to carefully distinguish betweendifferent types of outcome measures. 42,43 For example, the prevalence ofsymptoms does not of itself account for the relevance of these symptoms to thepatient. One study for example found that fecal incontinence and constipation hadalmost no effect on the generic HRQoL of patients with ARM or Hirschsprung'sdisease. 44 Another study showed that adolescents with Hirschsprung's diseasehad more severe levels of fecal incontinence, but no more psychopathology norpsychosocial dysfunction than healthy controls. 45 So, to gain a full understandingof the long-term outcomes of neonatal surgical diseases, it is important to includevarious outcome measures, such as measures of symptomatology and HRQoL.As said above, the time horizon should be sufficiently long to capture allsignificant effects. This is especially relevant for the case of neonatal surgery.After all, many of the most important outcomes, such as language or cognitiveabilities, educational attainment, and adult employment status, are