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Appendix C

Economic evaluation of alternative testing strategies in the

detection of hyperbilirubinaemia

C.1 Introduction

Jaundice (a yellow colouration of the skin) is caused by hyperbilirubinaemia and is common in
the newborn baby. Rarely, if bilirubin levels are sufficiently high, bilirubin can cross the blood–
brain barrier and cause a brain-damaging condition called kernicterus, a lifelong disabling
neurological problem with manifestations of cerebral palsy and deafness with high costs of care.
Hyperbilirubinaemia can also cause deafness without cerebral palsy, and other adverse
outcomes have been described. Levels of bilirubin can be controlled with phototherapy, but the
only way to reduce very high levels in an emergency is with an exchange transfusion. This is a
costly intensive care procedure that carries a mortality risk. Phototherapy is generally effective in
controlling bilirubin levels, preventing them from rising to a level at which kernicterus occurs,
and hence some clinicians have called for kernicterus to be classified as a ‘never event’. 231
There is some evidence to show that cases of kernicterus have risen recently, probably as a
result of earlier discharge following childbirth. 232;233
Current practice in England and Wales is varied but the GDG estimates that fewer than 10% of
babies undergo specific testing of their bilirubin levels following visual examination. At present,
babies who develop kernicterus often present late and with bilirubin levels already in the toxic
range. The key to prevention of kernicterus is early detection of cases at a time when
phototherapy can be effective. Any guideline recommendation that requires more widespread
testing will have important resource implications for the NHS as well as require a change in
practice in many places. Therefore the guideline recommendation regarding identification of
cases by testing for hyperbilirubinaemia was highlighted by the GDG as an important priority
for economic analysis. The NHS operates within resource constraints and a more intensive
testing and treatment strategy can only be justified if it represents a better use of scarce
resources than could be obtained in some alternative use of those resources.

C.2 Literature review

A literature search was undertaken to assess the economic evidence base for strategies to
prevent kernicterus in newborn babies. This initial search yielded 33 papers and the abstracts of
these papers were read to exclude papers that were clearly not relevant. As a result of this initial
screen, five papers were retrieved, of which only one was identified as a relevant economic
evaluation.

This was a US study that compared the cost-effectiveness of four strategies against current
practice to prevent kernicterus in newborn infants. 234 An incremental cost-effectiveness analysis
was undertaken using a decision-analytic framework. The strategies were modelled for a
population of healthy term infants who were being discharged within 48 hours of an
uncomplicated vaginal birth. The strategies were:
1. current practice – physicians and nurses assess the need for serum bilirubin testing after
delivery and prior to discharge based on a review of clinical history and physical
examination, including visual inspection of skin colour; clinical judgement and assessment of
risk are used to determine the timing of follow-up
2. universal follow-up 1–2 days after early discharge, but in other respects similar to the first
strategy

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