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Considering the influence of body’s growth and development on thyroid volume (TVOL), whether five existed corrected methods could be applied to correct TVOL remains unclear, in terms of Chinese children’s increased growth and development trends. This study aimed to compare the applicability of five correction methods: Body Surface Area corrected Volume (BSAV), Body Mass Indicator corrected Volume (BMIV), Weight and Height corrected Volume Indicator (WHVI), Height corrected Volume Indicator 1 (HVI1) and Height corrected Volume Indicator 2 (HVI2) and to establish the reference values for correction methods. The data of Iodine Nutrition and Thyroid Function Survey were used to analyse the differences in TVOL between normal and abnormal thyroid function children. Data of National Iodine Deficiency Disorders Survey were used to compare five correction methods and to establish their reference values. The median urinary iodine concentrations of children surveyed were 256·1 μg/l in 2009 and 192·6 μg/l in 2019. No significant difference was found in TVOL and thyroid goitre rate between children with normal and abnormal thyroid function. In the determination of goitre, HVI1, HVI2, BSAV and BMIV all showed high agreement with TVOL, while the area under the receiver operating characteristic curve (AUC) of WHVI was relatively low for children aged 8 (AUC = 0·8993) and 9 (AUC = 0·8866) years. Most differences of TVOL between light and heavy weight, short and tall height children can be eliminated by BSAV. BSAV was the best corrected method in this research. Reference values were established for corrected TVOL in Chinese children aged 8–10 years by sex.
This article explores how the Chinese government aims to maintain social stability by encouraging citizens to become volunteers. We propose that a new type of governance, namely, “state-enlisted voluntarism,” is being deployed in which public security volunteers are mobilized and monitored by the state. Analysis based on ten-year nationwide empirical data gathered from local areas in China suggests that the government intentionally enlists citizens into its hierarchical system to strengthen its administrative capacity and maintain a stable society without the risk of domestic threats. We find that direct enlistment approaches empower citizens as state proxies, and that indirect enlistment approaches ensure that various social stakeholders are comprehensively controlled. In general, the Chinese government has four reasons to institutionalize the state enlistment of voluntarism: to increase human resources at the grassroots; transform social organizations into subordinates; frame policy innovations as political credits; and to avoid blame. Our findings also suggest that China's party-state system mobilizes citizens into implementation-oriented activities rather than engages them in policymaking to maintain social stability at the grassroots.
We examined whether physical activity (PA) explains the association between dietary inflammatory potential and osteoarthritis (OA) in the elderly. A total of 1249 elderly people (≥65 years) were eligible for this study from the National Health and Nutrition Examination Survey (NHANES) from 2011 to 2016. The semi-quantitative Food Frequency Questionnaire (FFQ) and Global PA Questionnaire (GPAQ) were used to evaluate the diet and PA of the elderly, respectively. The multivariable logistic regression model estimated the odds ratio (OR) and 95% confidence interval (CI) between Energy-adjusted Dietary Inflammatory Index (E-DII) and OA. The interaction of E-DII and PA on depressive events was tested, and the mediation analysis of PA was performed. The average E-DII in this study was +0.68 (SE 0.08), and the score ranges from -5.32 (most anti-inflammatory) to +4.26 (most pro-inflammatory). In comparison with the first quartile, the elderly from the second quartile (OR: 1.16 [95% CI: 1.06, 1.68]) to the fourth quartile (OR: 1.64 [95% CI: 1.13, 2.37]) had a higher risk of OA before adjustment for PA. An interaction was observed between E-DII and PA in terms of the risk of OA (PInteraction < 0.001). The whole related part was mediated by PA (20.08%). Our findings indicated that the higher pro-inflammatory potential of diet was associated with a higher risk of OA, and low PA was an important part of the mediating factor in the relationship between systemic low-grade dietary inflammation and the risk of OA.
Many studies have substantiated the perceptual symbol system, which assumes a routine generation of perceptual information during language comprehension, but little is known about the processing format in which the perceptual information of different dimensions is conveyed simultaneously during sentence comprehension. The current study provides the first experimental evidence of how multidimensional perceptual information (color and shape) was processed during online sentence comprehension in Mandarin. We designed three consecutive sentence–picture verification tasks that only differed in the delay of the display of pictures preceded by declarative sentences. The processing was analyzed in three stages based on time intervals (i.e., 0ms, +750ms, +1500ms). The response accuracy and response time data were reported. The initial stage (i.e., ISI=0ms) attested the match effect of color and shape, but the simulated representation of color and shape did not interact. In the intermediate stage (i.e., ISI=750ms), the routinely simulated color and shape interacted, but the match facilitation was found only in cases where one perceptual information was in mismatch while the other was not. In the final stage (i.e., ISI=1500ms), the match facilitation of one particular perceptual property was influenced by a mismatch with the other perceptual property. These results suggested that multiple perceptual information presented simultaneously was processed in an additive manner to a large extent before entering into the final stage, where the simulated perceptual information was integrated in a multiplicative manner. The results also suggested that color and shape were comparable to object recognition when conjointly conveyed. In relation to other evidence from behavioral and event-related potential studies on sentence reading in the discussion, we subscribed to the idea that the full semantic integration became available over time.
Dongxiang wild rice (Oryza rufipogon Griff.) (DXWR) is the northernmost distributed wild rice found in the world. Similar to other populations of O. rufipogon, DXWR contains a large number of agronomically valuable genes, which makes it a natural gene pool for rice breeding. Molecular markers, especially simple repeat sequence (SSR) markers, play important roles in plant breeding. Although a large number of SSR markers have been developed, most of them are derived from the genome coding sequences, rarely from non-coding sequences. Meanwhile, long non-coding RNAs (lncRNAs), which are derived from the transcription of non-coding sequences, play vital roles in plant growth, development and stress responses. In our previous study, we obtained 1655 lncRNA transcripts from DXWR using strand-specific RNA sequencing. In this study, 1878 SSR loci were detected from the lncRNA sequences of DXWR, and 1258 lncRNA-derived-SSR markers were developed on the genome-wide scale. To verify the validity and applicability of these markers, 72 pairs of primers were randomly selected to test 44 rice accessions. The results showed that 42 (58.33%) pairs of primers have abundant polymorphism among these rice materials; the polymorphism information content values ranged from 0.04 to 0.87 with an average of 0.50; the genetic diversity index of SSR loci varied from 0.04 to 0.88 with an average of 0.56; and the number of alleles per marker ranged from 2 to 11 with an average of 4.36. Thus, we concluded that these lncRNA-derived-SSR markers are a very useful source for future basic and applied research.
Excessive iodine can lead to goiters. However, the relationship between the water iodine concentration (WIC) and goiter rate (GR) is unclear. This study aims to explore the factors that influence children’s GR in areas with high WIC and analyse the threshold value of the GR increase associated with the WIC. According to the monitoring of the areas with high WIC in China in 2018–2020, a total of 54 050 children in eight high water iodine provinces were chosen. Drinking water, urine and edible salt samples of children were collected. The thyroid volume (Tvol) was measured. A generalised additive model (GAM) was used to analyse the relationship between the WIC and GR in children. Among the 54 050 children in areas with high WIC, the overall GR was 3·34 %, the median of water iodine concentration was 127·0 µg/l, the median of urinary iodine concentration was 318 µg/l and the non-iodised salt coverage rate (NISCR) was 63·51 %. According to the GAM analysis results, water iodine and urinary iodine are factors that influence the Tvol and GR, while the NISCR affects only the GR. When the WIC was more than 420 µg/l or the urinary iodine concentration was more than 800 µg/l, the GR increased rapidly. When the NISCR reached more than 85 %, the GR was the lowest. Thus, in areas with high WIC, WIC more than 420 µg/l may increase the risk of goiter, and the NISCR should be increased to over 85 % to avoid goiters in children.
Epidemiological studies have shown that higher intake of flavonoid is inversely associated with CHD risk. However, which flavonoid subclass could reduce CHD risk has remained controversial. The present meta-analysis of prospective cohort studies aimed to quantitatively assess the associations between flavonoid subclasses and CHD risk. A systematic literature search was implemented from PubMed and Web of Science databases up to March 2021, and eligible studies were identified. Multivariate-adjust relative risks (RR) with corresponding 95 % CI were pooled by using a random-effects model. A restricted cubic spline regression model was performed for non-linear dose–response analysis. A total of 19 independent prospective cohort studies with 894 471 participants and 34 707 events were included. The results showed that dietary intakes of anthocyanins (RR = 0·90; 95 % CI: 0·83, 0·98), proanthocyanidins (RR = 0·78; 95 % CI: 0·65, 0·94), flavonols (RR = 0·88; 95 % CI: 0·79, 0·98), flavones (RR = 0·94; 95 % CI: 0·89, 0·99) and isoflavones (RR = 0·90; 95 % CI: 0·83, 0·98) were negatively associated with CHD risk. Dose–response analysis showed that increment of 50 mg/d anthocyanins, 100 mg/d proanthocyanidins, 25 mg/d flavonols, 5 mg/d flavones and 0·5 mg/d isoflavones were associated with 5 % reduction in CHD risk, respectively. Sensitivity and subgroup analyses were used to further support these associations. The present results indicate that dietary intakes of fruits and vegetables abundant five flavonoid subclasses, namely anthocyanins, proanthocyanidins, flavonols, flavones and isoflavones, are associated with a lower risk of CHD.
The policy of Universal Salt Iodisation (USI) could reduce population’s thyroid volume (TVOL) in iodine deficiency areas. Conversely, the improved growth and developmental status of children might increase the TVOL accordingly. Whether the decreased TVOL by USI conceals the increase effect of height and weight on TVOL is unclear. The aim of this study was to analyse the association between height, weight, iodine supplementation and TVOL. Five national Iodine Deficiency Disorder surveys were matched into four pairs according to the purpose of analysis. County-level data of both detected by paired surveys were incorporated; 1:1 random pairing method was used to match counties or individuals. The difference of TVOL between different height, weight, different iodine supplementation measures groups and the association between TVOL and them were studied. The mean height and weight of children aged 8–10 years increased from 129·9 cm and 26·9 kg in 2002 to 136·2 cm and 32·1 kg in 2019, while the median TVOL decreased from 3·10 ml to 2·61 ml. Iodine supplementation measures can affect TVOL; after excluding iodine effects, the median TVOL was increased with the height and weight. On the other side, after excluding the influence of height and weight, the median TVOL remained decreased. Only age, weight and salt iodine were significantly associated with TVOL in multiple linear models. Development of height and weight in children is the evidence of improved nutrition. The decreased TVOL caused by iodised salt measures conceals the increase effect of height and weight on TVOL. Age, weight and salt iodine affect TVOL significantly.
The present study evaluated whether fat mass assessment using the triceps skinfold (TSF) thickness provides additional prognostic value to the Global Leadership Initiative on Malnutrition (GLIM) framework in patients with lung cancer (LC). We performed an observational cohort study including 2672 LC patients in China. Comprehensive demographic, disease and nutritional characteristics were collected. Malnutrition was retrospectively defined using the GLIM criteria, and optimal stratification was used to determine the best thresholds for the TSF. The associations of malnutrition and TSF categories with survival were estimated independently and jointly by calculating multivariable-adjusted hazard ratios (HR). Malnutrition was identified in 808 (30·2 %) patients, and the best TSF thresholds were 9·5 mm in men and 12 mm in women. Accordingly, 496 (18·6 %) patients were identified as having a low TSF. Patients with concurrent malnutrition and a low TSF had a 54 % (HR = 1·54, 95 % CI = 1·25, 1·88) greater death hazard compared with well-nourished individuals, which was also greater compared with malnourished patients with a normal TSF (HR = 1·23, 95 % CI = 1·06, 1·43) or malnourished patients without TSF assessment (HR = 1·31, 95 % CI = 1·14, 1·50). These associations were concentrated among those patients with adequate muscle mass (as indicated by the calf circumference). Additional fat mass assessment using the TSF enhances the prognostic value of the GLIM criteria. Using the population-derived thresholds for the TSF may provide significant prognostic value when used in combination with the GLIM criteria to guide strategies to optimise the long-term outcomes in patients with LC.
Catastrophe insurance markets fail to provide sufficient protections against natural catastrophes, whereas they have the capacity to absorb the losses. In this paper, we assume the catastrophic risks are dependent and extremely heavy-tailed, and insurers have limited liability to cover losses up to a certain amount. We provide a comprehensive study to show that the diversification in the catastrophe insurance markets can be transited from suboptimal to preferred by increasing the number of insurers in the market. This highlights the importance of coordination among insurers and the government intervention in encouraging insurers to participate in the catastrophe insurance market to exploit risk sharing. Simulation studies are provided to illuminate the key findings of our results.
A deep ice core was drilled at Dome A, Antarctic Plateau, East Antarctica, which started with the installation of a casing in January 2012 and reached 800.8 m in January 2017. To date, a total of 337 successful ice-core drilling runs have been conducted, including 118 runs to drill the pilot hole. The total drilling time was 52 days, of which eight days were required for drilling down and reaming the pilot hole, and 44 days for deep ice coring. The average penetration depths of individual runs were 1 and 3.1 m for the pilot hole drilling and deep ice coring, respectively. The quality of the ice cores was imperfect in the brittle zone (650−800 m). Some of the troubles encountered are discussed for reference, such as armoured cable knotting, screws falling into the hole bottom, and damaged parts, among others.
Upper Ordovician strata exposed from the Baiyanhuashan section is the most representative Late Ordovician unit in the northwestern margin of the North China Craton (NCC). In total, 1,215 conodont specimens were obtained from 24 samples through the Wulanhudong and Baiyanhuashan formations at the Baiyanhuashan section. Thirty-six species belonging to 17 genera, including Tasmanognathus coronatus new species, are present. Based on this material, three conodont biozones—the Belodina confluens Biozone, the Yaoxianognathus neimengguensis Biozone, and the Yaoxianognathus yaoxianensis Biozone—have been documented, suggesting that the Baiyanhuashan conodont fauna has a stratigraphic range spanning the early to middle Katian. The Baiyanhuashan conodont fauna includes species both endemic to North China and widespread in tropical zones, allowing a reassessment of the previous correlations of the Katian conodont zonal successions proposed for North China with those established for shallow-water carbonate platforms at low latitudes.
ABSTRACT IMPACT: The knowledge acquired from my research can inform the development of early diagnostic methods for HIV-associated neurocognitive disorders. OBJECTIVES/GOALS: In the era of combination antiretroviral therapy (cART), the prevalence of HIV-associated neurocognitive disorders (HAND) remains high but the neural mechanisms are unclear. We examined whether older people with HIV (PWH) with minimal cognitive impairment have reduced functional connectivity in frontostriatal circuits compared to controls. METHODS/STUDY POPULATION: 99 PWH (mean age 56.6 years, 75% male, 62% Black, mean duration of HIV-infection 26.2 years ±9.3, 90% viral load <50 copies, 98% on stable cART) and 38 demographically-comparable controls (mean age 54.5 years, 71% male, 58% Black) participated in a cross-sectional study. A 7-domain neuropsychological battery and an Activities of Daily Living index were used to determine HAND diagnoses: 32 PWH met criteria for asymptomatic to mild HAND. Motor skill was assessed using the Grooved Pegboard Test by measuring performance speed. Structural MRI and resting-state functional MRI were collected. Seed-to-voxel analyses were conducted using 4 distinct regions in the striatum as seed regions. We used a voxel threshold of p<0.001 and cluster threshold of p<0.05 (FDR-corrected) after controlling for demographic variables. RESULTS/ANTICIPATED RESULTS: Compared to controls, PWH had lower resting state functional connectivity between the default mode region of the striatum (i.e., medial caudate) and bilateral superior frontal gyrus, supplementary motor cortex and paracingulate gyrus (p<0.05; cluster size: 567 voxels). Also, compared to controls, PWH had reduced resting state functional connectivity between the motor division of the striatum (i.e., posterior putamen) and anterior cingulate cortex and left supplementary motor cortex (p<0.05, cluster size: 405 voxels). Performance speed on the Grooved Pegboard motor test negatively correlated with functional connectivity between the motor region of the striatum and supplementary motor frontal regions in all participants (Spearman’s rho=-0.18, p=0.04). DISCUSSION/SIGNIFICANCE OF FINDINGS: Our results support the hypothesis that frontostriatal abnormalities are widely present in PWH and might play a key role in HAND development. Our data suggest that dysfunction within the frontostriatal circuits may be involved in motor impairment in PWH, and ongoing inflammation may contribute to motor impairment and frontostriatal injury.
The tobacco cutworm Spodoptera litura (Lepidoptera: Noctuidae) is a polyphagous pest with a highly selective and sensitive chemosensory system involved in complex physiological behaviors such as searching for food sources, feeding, courtship, and oviposition. However, effective management strategies for controlling the insect pest populations under threshold levels are lacking. Therefore, there is an urgent need to formulate eco-friendly pest control strategies based on the disruption of the insect chemosensory system. In this study, we identified 158 putative chemosensory genes based on transcriptomic and genomic data for S. litura, including 45 odorant-binding proteins (OBPs, nine were new), 23 chemosensory proteins (CSPs), 60 odorant receptors (ORs, three were new), and 30 gustatory receptors (GRs, three were new), a number higher than those reported by previous transcriptome studies. Subsequently, we constructed phylogenetic trees based on these genes in moths and analyzed the dynamic expression of various genes in head capsules across larval instars using quantitative real-time polymerase chain reaction. Nine genes–SlitOBP8, SlitOBP9, SlitOBP25, SlitCSP1, SlitCSP7, SlitCSP18, SlitOR34, SlitGR240, and SlitGR242–were highly expressed in the heads of 3- to 5-day-old S. litura larvae. The genes differentially expressed in olfactory organs during larval development might play crucial roles in the chemosensory system of S. litura larvae. Our findings substantially expand the gene inventory for S. litura and present potential target genes for further studies on larval feeding in S. litura.
Previously, we reported the myelin regulatory factor (MYRF) as a candidate gene for nanophthalmos. We have also produced Myrf knockdown (Myrf+/−) mouse strain to investigate the cellular and molecular phenotypes of reduced MYRF expression in the retina. Myrf+/− mouse strain was generated using the CRISPR/Cas9 system. Optomotor response system, electroretinogram (ERG), spectral-domain optical coherence tomography (SD-OCT), histology, and immunohistochemistry were performed to evaluate retinal spatial vision, electrophysiological function, retinal thickness, and pathological changes in cone or rod photoreceptors, respectively. RNA sequencing (RNA-seq) was performed to investigate the underlying molecular mechanism linking Myrf deficiency with photoreceptor defects. The genotype and phenotype of CRISPR/Cas9-induced Myrf+/− mice and their offspring were comprehensively investigated. Photoreceptor defects were detected in the retinas of Myrf+/− mice. Visual acuity and ERG responses were decreased in Myrf+/− mice compared with the control mice (Myrf+/+). The loss of cone and rod neurons was proportional to the decreased outer nuclear layer (ONL) thickness. Moreover, RNA-seq revealed that phototransduction and estrogen signaling pathways played important roles in the Kyoto Encyclopedia of Genes and Genomes (KEGG) analysis. Myrf+/− mouse strain provides a good model to investigate the function of the MYRF gene. Photoreceptor defects with impaired functions of spatial vision and retinal electrophysiology indicate an important role played by MYRF in retinal development. Alterations in phototransduction and estrogen signaling pathways play important roles in linking Myrf deficiency with retinal photoreceptor defects.
According to the World Health Organization, there were approximately 0.5 million new cases of rifampicin-resistant tuberculosis in 2018, of which 78 percent were multidrug-resistant tuberculosis (MDR-TB), and China has one of the largest shares of the global burden (14%). In recent years, the Chinese government has made progress in TB control and prevention, but for MDR-TB, treatment options are still limited and expensive, and novel drugs are not always available. This research aims to evaluate the cost-effectiveness of adding bedaquiline to a background regimen (BR) of drugs for MDR-TB treatment in China, and to provide evidence for government to improve public health policies.
A cohort-based Markov model was developed to evaluate the incremental cost-effectiveness ratio (ICER) of bedaquiline plus BR (BBR) versus BR alone in MDR-TB treatment, over a 10-year time horizon. Data were sourced from a phase II clinical trial, real-world data in China, published literature, and expert opinion. Outcomes were evaluated in quality-adjusted life years (QALYs) and life-years gained (LYG). The discount rate was 3.5%. Probabilistic and deterministic sensitivity analyses were conducted.
The discounted costs per person for BBR was CNY 135,706 [USD 19,172], compared with CNY 92,465 [USD 13,063] for BR. The discounted utility per person for BBR was also higher than that for BR (3.943 QALYs versus 3.193 QALYs). The ICER of BBR was CNY 58,096 [USD 8,208]/QALY, which was lower than the willingness-to-pay threshold of CNY 212,676 [USD 30,046] (three-times the gross domestic product per capita). Therefore, BBR was considered to be cost-effective. The sensitivity analysis confirmed the robustness of the results. BBR remained cost-effective in the sensitivity analysis, with a 77.2 percent probability of being cost-effective versus BR.
In China, bedaquiline is not included in the National Reimbursement Medicine List, which results in a heavy financial burden for MDR-TB patients. From this study, BBR was cost-effective by significantly reducing time to sputum culture conversion and increasing QALYs and LYGs, which offset the higher drug costs.
In this paper, the generation of relativistic electron mirrors (REMs) and the reflection of an ultra-short laser off this mirrors are discussed, applying two-dimensional particle-in-cell (2D-PIC) simulations. REMs with ultra-high acceleration and expanding velocity can be produced from a solid nanofoil illuminated normally by an ultra-intense femtosecond laser pulse with a sharp rising edge. Chirped attosecond pulse can be produced through the reflection of a counter-propagating probe laser off the accelerating REM. In the electron moving frame, the plasma frequency of the REM keeps decreasing due to its rapidly expanding. The laser frequency, on the contrary, keeps increasing due to the acceleration of REM and the relativistic Doppler shift from the lab frame to the electron moving frame. Within an ultra-short time interval, the two frequencies will be equal in the electron moving frame, which leads the resonance between laser and REM. The reflected radiation near this interval and the corresponding spectra will be amplified due to the resonance. Through adjusting the arriving time of the probe laser, certain part of the reflected field could be selectively amplified or depressed, leading to the selectively adjusting of the corresponding spectra.
Birth defects seriously affect children's survival and quality of life and bring great suffering and financial burden to children and their families. Down's syndrome is one of the most common birth defects. Compared with traditional serological screening methods, non-invasive prenatal testing (NIPT) has higher sensitivity and specificity in the screening of Down's syndrome. In April 2017, the People's Government of Fuyang City, Anhui Province launched a NIPT free screening program. From the perspective of the beneficiary, this research investigated the awareness, willingness to pay and satisfaction of pregnant women in Fuyang City, Anhui Province, to better improve the use of NIPT.
A questionnaire survey was conducted on 1,221 pregnant women who experienced this program in Fuyang City, Anhui Province. Multivariate ordered logistic regression models were established to analyze the factors affecting the satisfaction of NIPT.
A total of 1,217 valid questionnaires were collected. Research indicated 82.5 percent knew about NIPT and 81.9 percent were willing to pay personally when its price was CNY 800 (USD 113.88) per test among pregnant women. The satisfaction of pregnant women with NIPT showed that the waiting time for test results was relatively low (4.5 out of 5 points) compared with other aspects of satisfaction. The higher the education level of the pregnant women, the lower their satisfaction with NIPT.
It is necessary to pay attention to the characteristics of education and to improve the awareness and satisfaction of NIPT among pregnant women. Meanwhile, if it is affordable enough for NIPT services to be provided by the government, this mode should be promoted. In conjunction with the willingness to pay of pregnant women, NIPT payment methods should be developed appropriately.
According to the ‘farming/dispersal’ hypothesis, the Early and Mid-Holocene spread of Neolithic material culture in East Asia would have arisen from dispersals of established farming populations. The authors test this hypothesis by considering the Beixin Culture that appeared in the south-west Haidai region of northern China c. 5000 BC, before spreading north and east to the coast over the subsequent millennium. While this culture had architecture, elaborate pottery and other forms of Neolithic material culture, analysis of archaeobotanical evidence from Guanqiaocunnan (4340–3970 BC) suggests an economic base of hunting, gathering and cultivating, rather than a reliance on farming.
Primary immunodeficiency diseases (PIDs) are a heterogeneous group of over 200 disorders with defects in the function and/or development of the immune system. Although early screening is imperative for improving therapeutic efficiency and preventing disease-associated morbidity, its widespread use has been limited, owing to the low incidence of PIDs. It is particularly important to evaluate the cost-effectiveness of PIDs screening for newborns. The aim of this study was to provide an overview of the existing cost-effectiveness evidence on newborn screening of PIDs and to provide reference for decision-makers in China and other developing countries.
We conducted a systematic review using three electronic databases (PubMed, CNKI, and CSPD) of cost and cost-effectiveness studies of PIDs screening published during 2000–2019. Two reviewers independently searched databases and screened titles, abstracts and full texts; a third reviewer resolved disputes when necessary. The initial search returned 124 references, of which 10 full articles were included in the review. Five of the studies conducted analyses using model-based techniques.
Severe combined immunodeficiency (SCID) was the predominantly studied condition (80%). Most studies (70%) examined the T-cell receptor excision circle (TREC) assay. A healthcare system's perspective was commonly used (50%) for cost calculations, and most studies (50%) were US-based. The majority (67%) of the studies found the TREC assay an effective screening tool for SCID, but the incremental cost-effectiveness ratio (ICER) varied across screening test specificity and disease incidence.
Evidence from the published literature demonstrated that newborn screening for PIDs generally appeared to be cost-effective, and most importantly, it is lifesaving and allows children with PIDs an opportunity to live a healthier life. However, the type of PIDs included in this study were limited and most studies were done in developed countries whose health systems are different from low-/middle-income countries (LMIC). Further research is required to identify the cost-effectiveness of PIDs screening both in developed and developing countries.