Cost-effectiveness analysis of different screening modes for thalassemia in Hunan Province
10.3760/cma.j.cn113903-20220802-00750
- VernacularTitle:湖南省地中海贫血不同筛查模式的成本效果分析
- Author:
Hui XI
1
;
Qin LIU
;
Donghua XIE
;
Xu ZHOU
;
Wanglan TANG
;
Deguo TANG
;
Chunyan ZENG
;
Qiong WANG
;
Xinghui NIE
;
Jinping PENG
;
Xiaoya GAO
;
Hongliang WU
;
Haoqing ZHANG
;
Li QIU
;
Zonghui FENG
;
Shuyuan WANG
;
Shuxiang ZHOU
;
Jun HE
;
Shihao ZHOU
;
Faqun ZHOU
;
Junqing ZHENG
;
Hua WANG
;
Junqun FANG
;
Changbiao LIANG
Author Information
1. 湖南省妇幼保健院医学遗传科，国家卫生健康委员会出生缺陷研究与预防重点实验室，长沙 410028
- Keywords:
Thalassemia;
Prenatal diagnosis;
Practice patterns;
Cost-benefit analysis
- From:
Chinese Journal of Perinatal Medicine
2023;26(6):468-475
- CountryChina
- Language:Chinese
-
Abstract:
Objective:To analyze the costs and effectiveness of five common screening modes and genetic screening for thalassemia in China in order to find the optimal way and provide evidence for the implementation of thalassemia prevention and control projects in Hunan Province.Methods:From June 2020 to April 2021, 12 971 couples from 14 cities and autonomous prefectures in Hunan Province were selected as the study population. The diagnosis of thalassemia was based on the results of genetic testing. Results of routine blood test and hemoglobin electrophoresis were collected and analyzed. The efficacy of five screening modes, at the cut-off value of <80 fl or 82 fl for the mean corpuscular volume (MCV), was analyzed by positive predictive value, negative predictive value, Jorden index and cost-effectiveness ratio. Sensitivity analysis was used to assess the feasibility of genetic screening at different costs after fixing the costs of routine blood and hemoglobin electrophoresis. The five thalassemia screening models are as follows: Mode 1: The woman had a blood routine test first. If the result was positive, the spouse required a blood routine test. If both results were positive, a thalassemia gene test should be offered to the couple. Mode 2: Both husband and wife were screened by blood routine and hemoglobin electrophoresis. If one or both of them were positive, both would be tested for thalassemia gene. Mode 3: The couple received blood routine tests initially. If either was positive, both should receive hemoglobin electrophoresis testing. If either was positive, both parties will conduct thalassemia gene testing. Mode 4: The woman was screened by blood routine and hemoglobin electrophoresis. If any one of them was positive, the woman would be tested for thalassemia gene. If the gene test result was positive, the spouse should receive thalassemia gene. Mode 5: Both spouses conducted a blood routine test. If either was positive, both would conduct hemoglobin electrophoresis test. If both were positive, both spouses should receive thalassemia gene testing. Gene testing mode: The woman would be tested for thalassemia, and her spouse would have thalassemia test too if her result was positive.Results:When using MCV<80 fl as the cut-off for diagnosing thalassemia, the Youden indices of the five prenatal screening modes in Hunan Province were 0.551, 0.639, 0.898, 0.555 and 0.356, while when using MCV<82 fl as the cut-off, the Youden indices were 0.549, 0.629, 0.851, 0.548 and 0.356. When the MCV cut-off value was <80 fl, the missed diagnosis rates of the five screening modes were 44.44%, 0.00, 0.00, 18.52% and 62.96%, and the cost-effectiveness ratios were 21 709, 250 939, 76 870, 138 463 and 92 860 yuan (RMB)/couple, respectively. When the price of genetic testing was lower than 55 yuan (RMB), the cost-effectiveness ratio of genetic screening was lower than that of Mode 3.Conclusions:MCV<80 fl can be considered as the positive criteria in blood routine screening for thalassemia in Hunan Province, and the cost-effectiveness ratio of Mode 3 (the couple received blood routine tests initially. If either was positive, both should receive hemoglobin electrophoresis testing. If either was positive, both parties will conduct thalassemia gene testing) is the best. Genetic screening has certain advantages with the decreasing price.
