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Diagnosis of Hirschsprung's Disease by Immunostaining Rectal Suction Biopsies for Calretinin, S100 Protein and Protein Gene Product 9.5
Published on: April 26, 2019
Economic evaluation of Hirschsprung disease testing strategies for children with medically-refractory chronic
Prisca C Obidike1, Tanya N Wanchek2, Kristy L Rialon3
1Division of Pediatric Surgery, Department of Surgery, University of Virginia, Charlottesville, VA, USA.
Insights
For suspected Hirschsprung disease (HSCR) in children, contrast enema (CE) is the most cost-effective initial diagnostic strategy. This approach balances effectiveness and healthcare costs for early HSCR detection.
Area of Science:
- Pediatric Surgery
- Diagnostic Imaging
- Health Economics
Background:
- Early diagnosis of Hirschsprung disease (HSCR) is crucial for preventing severe complications and reducing healthcare costs.
- Common diagnostic tests include contrast enema (CE), anorectal manometry (AMAN), and rectal biopsy (RB), with RB considered the gold standard.
- The optimal, most cost-effective initial diagnostic strategy for HSCR remains undetermined.
Purpose of the Study:
- To determine the most cost-effective initial diagnostic strategy for evaluating suspected Hirschsprung disease (HSCR) in children aged 1-18 years.
- To compare the cost-effectiveness of CE-first, AMAN-first, and RB-first diagnostic pathways.
- To inform clinical decision-making regarding HSCR diagnostic testing.
Main Methods:
- A decision-analytic model evaluated CE-first, AMAN-first, and RB-first strategies over a three-year period.
- The model incorporated diagnostic test accuracy, HSCR prevalence, and surgical intervention timing.
- Health utilities (Child Health Utility 9D) and costs were analyzed using a willingness-to-pay threshold of $50,000 per QALY.
Main Results:
- The CE-first strategy was identified as the most cost-effective initial approach at the $50,000/QALY threshold.
- RB-first was more effective but costlier, becoming cost-effective only at high HSCR prevalence (>29.2%) or test sensitivity (>97.8%).
- The AMAN-first strategy was the least cost-effective, requiring high sensitivity (>90.5%) to be considered.
Conclusions:
- Contrast enema (CE) is the most cost-effective initial diagnostic strategy for evaluating suspected Hirschsprung disease (HSCR) in children over one year old presenting to specialty care.
- These economic findings support informed clinical decisions for HSCR diagnostic testing pathways.
- The study highlights the importance of cost-effectiveness analysis in pediatric diagnostic strategies.
Background:
Early and accurate diagnosis of Hirschsprung disease (HSCR) prevents life-threatening complications, improves patient outcomes, and reduces healthcare utilization. Contrast enema (CE), anorectal manometry (AMAN), and rectal biopsy (RB) are commonly used diagnostic tests, with RB being the gold standard. However, the most cost-effective initial diagnostic strategy remains unclear.
Methods:
A decision-analytic model was used to compare CE-first, AMAN-first, and RB-first strategies for the evaluation of HSCR in children between 1 and 18 years of age presenting to specialty care for chronic constipation. The model incorporated diagnostic test performance, disease prevalence, and timing of surgical intervention over a three-year time horizon. Health utilities were estimated by mapping published Pediatric Quality of Life Inventory (PedsQL) scores to Child Health Utility 9D (CHU9D) values. Costs were averaged from institutional data. Willingness-to-pay (WTP) threshold was set at $50,000 per quality-adjusted life year (QALY). Outcomes included costs, QALYs, net monetary benefit (NMB), and incremental cost-effectiveness ratios (ICERs). Deterministic and probabilistic sensitivity analyses were performed.
Results:
CE-first emerged as the most cost-effective diagnostic strategy at a WTP of $50,000/QALY. RB-first provided greater effectiveness but at higher cost and was not cost-effective at the base-case threshold. RB-first became preferred when HSCR prevalence exceeded 29.2% and when the sensitivity of the test exceeded 97.8%. AMAN-first was least cost-effective and became preferred only when its sensitivity exceeded 90.5%.
Conclusions:
In the evaluation of children over 1 year of age presenting to specialty care with suspected HSCR, CE was the most cost-effective initial strategy in most scenarios. These findings provide economic insight to guide clinical decision-making for the diagnostic testing of HSCR.
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