Table 1

Different possible options, their costs, benefits and challenges

Screening optionMarginal costs associated/1000Incremental yieldEfficiencyResponsiveness
0 No first year screening (responsive service only)NoneYield estimate uncertain - maybe < 0.2 per 1000 in first year (<20%)

Very poor: Cost per case indeterminatePoor (with possible exception of profound PCHI)
H0 Universal IDT£29.2k Total = £29.2kPresent average yield is 0.25 which might be increased to 0.4 with good quality controlPoor: Cost per case £90-£110kFair
H1 Hall report recommendations: Targeted neonatal screening: introduce targeted screening where not already implemented and make more systematic where very limited at present
Universal IDT
£5.7k £29.2k Total = £34.9kYield estimates if both NICU and family history groups can get high coverage and better HVDT quality control 0.5 per 1000 by six months, 0.75 per 1000 by 1 yearPoor: Cost per case about £45k (incremental cost per case for IDT about £110k)Fair
H2 As for H1, but following R&D implement technologically advanced IDT, with increased levels of stimuliAs for H1, with IDT reducing to about £23k
Total = £28.1
Yield as per H1, possibly increasing to 0.8 per 1000 by 1 yearFair: Cost per case about £35k (incremental cost per case for IDT about £90k)Fair
H3 As for H1, but replace Universal IDT with 6-8 m universal HV surveillance by questionnaireAs for H1, with HV costs reducing to £23.4k?
Total = £29.1k
Yield as per H1 or slightly less, particularly the moderatesPoor: Cost per case about £50k (incremental cost per case for IDT about £95k)Fair
T1 Targeted neonatal screening: introduce targeted screening where not already implemented and make more systematic where limited at presentAS for H1, but no HVDT Total = £5.7kYield estimates, given high coverage of both NICU and family history children, 0.5 per 1000 by six months, but probably poor thereafterFair to good: Cost per case about £11.4kFair
T2 As for H1, but replace IDT with targeted infant distraction test

As for H1, but IDT possibly reducing to about £9k
Total = £14.7k
Yield likely to be more than T1 but less than H1Good: Cost per case about £19.3kGood
U1 Introduce universal neonatal screening




£15.8k Total = £15.8kYield 0.9 per 1000 by six  monthsVery good: Cost per case about  £17kGood
U2 Introduce universal neonatal screening
Modify infant distraction test to be targeted on those not tested and high risk of progressive PCHI
£15.8k £3.5k Total = £19.3kYield 0.9 per 1000 by six months and possibly 1.0 per 1000 by one yearVery good: Cost per case about £19.3k (incremental cost per case for IDT £34k, very approximate)Very good
EquityBenefitsChallenges
Very lowReleases time/money to invest in responsive system, improvement of habilitation facilities for severe/profoundModerate and severe PCHI not identified until >2 y, possible identification if language screen about 2 y
LowWould consolidate present services and remove uncertainty for HVsNeed better training, facilities and quality standards for IDT
ID age suboptimal
MediumLittle change to system, would help build up targeted screeningNeed better training, facilities and quality standards for IDT, for targeted screening and for very early habitation in all districts Implementation of family history difficult Overall sensitivity of system poor ID age suboptimal


MediumLimited change to system, and better test possible for IDT if acceptedAs H1, but sensitivity possibly better New equipment needs to be developed, evaluated and bought

Medium
Low sensitivity for moderate losses
Limited change to system, well trialed already. More in line with HV mission

As per H1
LowBetter quality control  possibleAs per H1, tuning the responsive system to find the remaining 0.6 per 1000, possibly needing to spend more on HVS


Medium
Likely to miss ethnic minority and low SEGs
Better quality control possible, if less HVs involved, or specialist referral systemAs per H1 plus definition of HV target population, crucial to combat inverse care law, needs research to define
MediumAge ID very good Greater potential for habilitation and education to give benefitTraining, coordination and follow-up pose significant implementation challenges. What to do for those not tested and for late onset/progressive cases National support needed
HighAge of ID best that can be achieved for all PCHI groups Greater potential for habilitation and education to give benefitTraining, coordination and follow-up pose significant implementation challenges, including how to target progressive cases National support needed
  • It is assumed (i) that there will be Health Visitor Surveillance for all children age 0-5 years (as per Health For All Children, cost unknown) and (ii) that the School Entry Screen will be retained for all options (at a cost of about £3.5-4.5k per live births). All costs standardised to per 1000 live births (not 1000 children tested). The 1000 cost per case identified is a broad estimate based on the programme cost and yield.