Economic Evaluation of the Thai Diagnostic Autism Scale for Autism Spectrum Disorder Diagnosis in Children Aged 1-5 Years Old.

The Thai Diagnostic Autism Scale (TDAS) was developed for autism spectrum disorder (ASD) diagnosis in Thai children aged 1-5 years. Previous studies have indicated its good performance; however, additional health resources and healthcare providers are necessary for evaluation. Therefore, this study aimed to assess the cost-effectiveness of TDAS compared to clinical diagnosis (ClinDx) for ASD diagnosis in Thai children aged 1-5 years from a societal perspective. The analysis employed a hybrid model consisting of a decision tree model for a diagnostic phase with a state transition model for a follow-up phase. A literature review was conducted to determine TDAS performance and the relative risk of death in patients with ASD. Direct medical costs were assessed through a retrospective medical records review, and a cross-sectional survey was conducted to determine direct nonmedical costs, ASD severities, and utility values. The cost of TDAS was derived from a healthcare provider interview (n = 10). The incremental cost-effectiveness ratio (ICER) compared the total lifetime cost and quality-adjusted life years (QALY) between TDAS and ClinDx. We found that TDAS could improve QALY by 1.96 but increased total lifetime cost by 5577 USD, resulting in an ICER of 2852 USD/QALY. Sensitivity analysis indicated an 81.16% chance that TDAS is cost-effective. The probabilities of different ASD severities were key influencing factors of the findings. In conclusion, TDAS is the cost-effective option for ASD diagnosis in Thai children aged 1-5 years compared to ClinDx, despite some uncertainties around inputs. Further monitoring and evaluation are warranted if TDAS is to be implemented nationwide.

A 9-year retrospective cohort study of the monitoring and screening of childhood developmental delay in Thailand.

BACKGROUND: Developmental delay in early childhood can have negative long-term cognitive and psychiatric sequelae, along with poor academic achievement, so early screening and surveillance are paramount. The aim of this study is to evaluate the impact of screening and surveillance on child developmental delay using the Developmental Surveillance and Promotion Manual (DSPM) and the Thai Early Developmental Assessment for Intervention (TEDA4I) for Thai children aged 0-5 years old. METHODS: Data were obtained from the routine developmental screening for specific disorders at ages 9, 18, 30, 42 and 60 months conducted using DSPM and TEDA4I from 2013 to 2021. Descriptive statistics were used to analyse the data, and the results are visualised graphically herein. RESULTS: Only 56% of the children were screened for child developmental delay using DSPM. The proportion of children screened increased from <1% in 2013 to 90% in 2021. Suspected developmental delay prevalence increased significantly from 3.91% in 2013-2015 to 10.00% in 2016-2018 and 26.48% in 2019-2021. Moreover, of the children with suspected developmental delay who received developmental stimulation within a month, only 87.9% returned for follow-up visits when they were evaluated again using TEDA4I to ascertain any abnormalities and specific areas of deficit. The overall proportion of children diagnosed with developmental delay was 1.29%. During the pandemic, the proportion of screening tests for child developmental delay at routine vaccination visits and follow-ups decreased but was still at least 80% in each region. CONCLUSIONS: Since 1%-3% of children have suspected developmental delay, early detection is key to treating it as soon as possible. We anticipate that our findings will raise awareness in parents and caregivers about childhood developmental delay and lead to the implementation of early intervention and follow-up at the rural level in Thailand.

Factors Associated with Suspected Developmental Delay in Thai Children Born with Low Birth Weight or Asphyxia.

OBJECTIVES: The aim of the study was to identify factors associated with a risk of suspected developmental delay (SDD) in high-risk children in Thailand. METHODS: We used data on children enrolled for developmental delay (DD) screening across Thailand collected by the Rajanagarindra Institute of Child Development, Department of Mental Health, Ministry of Public Health, Thailand. Children who were under 5 years of age with a birth weight of fewer than 2500 g and/or birth asphyxia in Thailand with high risk of DD were assessed using the Developmental Assessment for Intervention Manual (DAIM) between August 2013 and November 2019 (N = 14,314). RESULTS: The high-risk children who had a gestational age at birth of < 37 weeks (adjusted odds ratio = 1.54; 95% confidence interval = 1.39-1.70) and/or had a birth weight < 2500 g (1.22; 1.02-1.45), or had mothers who were not government officers (1.46; 1.11-1.93), had a low education level (1.36; 1.19-1.55), had a poor nutritional status (1.34; 1.09-1.65), and/or who were living in a high-altitude area (1.59; 1.32-1.91) were at a higher risk of SDD. CONCLUSIONS FOR PRACTICE: Children with a low birth weight and/or asphyxia during birth had a high risk of DD. SDD monitoring of children by community health workers and/or by developing outreach strategies, especially in underserved regions, should be considered. In addition, developing policies and guidelines, and intervention for high-risk children ought to be conducted to reduce the subsequent problems caused by the late detection of DD.

The Optimal Cut-Off Point for Thai Diagnostic Autism Scale and Probability Prediction of Autism Spectrum Disorder Diagnosis in Suspected Children.

The Thai Diagnostic Autism Scale (TDAS) was developed to diagnose autism spectrum disorder (ASD) under the context and characteristics of the Thai population. Although the tool has an excellent agreement, the interpretation of diagnostic results needs to rely on the optimal cut-off point to maximize efficiency and clarity. This study aims to find an optimal cut-off point for TDAS in the diagnosis of ASD and to compare its agreement with the DSM-5 ASD criteria. This study was conducted on 156 children aged 12-48 months old who were suspected of having ASD and had enrolled from hospitals in the four regions of Thailand in 2017-2018. The optimal cut-off point for TDAS was considered by using receiver operating characteristic (ROC) curves according to the DSM-5 ASD criteria. The areas under the curve (AUCs) for TDAS and ADOS-2 were also compared. Multivariable logistic regression was performed to create a predictive model for the probability of ASD. The AUC of TDAS was significantly higher than that of ADOS-2 (0.8748 vs. 0.7993; p = 0.033). The optimal cut-off point for TDAS was ≥20 points (accuracy = 82.05%, sensitivity = 82.86%, and specificity = 80.93%). Our findings show that TDAS with a cut-off point can yield higher diagnostic accuracy than ADOS-2 and TDAS domain. Diagnosis by using this cut-off point could be useful in practical assessments.

Detection of Electroencephalographic Abnormalities and Its Associated Factors among Children with Autism Spectrum Disorder in Thailand.

Epilepsy often causes more severe behavioral problems in children with autism spectrum disorder (ASD) and is strongly associated with poor cognitive functioning. Interestingly, individuals with ASD without a history of epilepsy can have abnormal electroencephalographic (EEG) activity. The aim of this study was to examine associations between EEG abnormalities and the ASD severity in children. The children with ASD who enrolled at the Rajanagarindra Institute of Child Development, Thailand were included in this study. The severity of ASD was measured by interviewing their parents with the Thai autism treatment evaluation checklist. The short sensory profile checklist was used for screening the abnormality of children in each domain. Ordinal logistic regression analysis was used to examine associations between factors potentially linked to EEG abnormalities. Most of the study participants were boys (87.5%) and the median age was 5 years. Among the 128 children, 69.5% showed EEG abnormalities (41.4% slow-wave and 28.1% epileptiform-discharge). The results show that a larger number of symptoms and increased severity of ASD were independently associated with a higher risk of EEG abnormalities. Our results emphasize the need for guidelines on the presence of EEG abnormalities in children with ASD for the early detection of epilepsy and improving treatment outcomes.

Development and psychometric evaluation of a Thai Diagnostic Autism Scale for the early diagnosis of Autism Spectrum Disorder.

The Thai Diagnostic Autism Scale (TDAS) was developed for use as a diagnostic tool for the early diagnosis of Autism Spectrum Disorder (ASD) in Thai children aged 12-48 months old. TDAS consists of 23 items (13 and 17 items in the observational and interview sections, respectively) classified into seven domains (A1-A3 and B1-B4) according to the criteria in the Diagnostic and Statistical Manual of Mental Disorder, fifth edition (DSM-5). Children with a single score in the A1-A3 domains and at least two of the B1-B4 domains were classified with ASD. The item-objective congruence (IOC) index, confirmatory factor analysis, and Kappa coefficient were used to evaluate the content, constructs, and inter-rater validity levels between the evaluators and concurrent validity between TDAS and physicians' diagnoses, respectively. TDAS showed good overall content validity (IOC range 0.71-1.00), suitable construct validity (root-mean-squared errors of approximation of 0.076 and 0.067, comparative fit indexes of 0.902 and 0.858, and Tucker-Lewis indexes of 0.882 and 0.837 for the observation and interview sections, respectively), and excellent diagnostic agreement between TDAS and the evaluators (Kappa = 1.000) as well as between TDAS and the physicians' diagnoses (Kappa = 0.871). The sensitivity and specificity of TDAS were 100% and 82.4%, respectively. In conclusion, TDAS yielded a high level of content validity, concurrent validity, and inter-rater reliability for the early diagnosis of ASD in Thai children. A large-scale study using TDAS is needed to determine an appropriate cut-off point as well as its efficacy. LAY SUMMARY: The Thai Diagnostic Autism Scale was developed for use as a diagnostic tool for the early diagnosis of Autism Spectrum Disorder (ASD) among Thai children. It contains 23 items in seven domains for the screening via observations and interviews. The psychometric properties of this diagnostic tool provide its reliability and suitability for the early diagnosis of ASD. A large-scale study using it is needed to determine an appropriate cut-off point as well as its efficacy.

Whole-Exome Sequencing Identifies One De Novo Variant in the FGD6 Gene in a Thai Family with Autism Spectrum Disorder.

Autism spectrum disorder (ASD) has a strong genetic basis, although the genetics of autism is complex and it is unclear. Genetic testing such as microarray or sequencing was widely used to identify autism markers, but they are unsuccessful in several cases. The objective of this study is to identify causative variants of autism in two Thai families by using whole-exome sequencing technique. Whole-exome sequencing was performed with autism-affected children from two unrelated families. Each sample was sequenced on SOLiD 5500xl Genetic Analyzer system followed by combined bioinformatics pipeline including annotation and filtering process to identify candidate variants. Candidate variants were validated, and the segregation study with other family members was performed using Sanger sequencing. This study identified a possible causative variant for ASD, c.2951G>A, in the FGD6 gene. We demonstrated the potential for ASD genetic variants associated with ASD using whole-exome sequencing and a bioinformatics filtering procedure. These techniques could be useful in identifying possible causative ASD variants, especially in cases in which variants cannot be identified by other techniques.

Exome Sequencing Identifies Compound Heterozygous Mutations in SCN5A Associated with Congenital Complete Heart Block in the Thai Population.

Background. Congenital heart block is characterized by blockage of electrical impulses from the atrioventricular node (AV node) to the ventricles. This blockage can be caused by ion channel impairment that is the result of genetic variation. This study aimed to investigate the possible causative variants in a Thai family with complete heart block by using whole exome sequencing. Methods. Genomic DNA was collected from a family consisting of five family members in three generations in which one of three children in generation III had complete heart block. Whole exome sequencing was performed on one complete heart block affected child and one unaffected sibling. Bioinformatics was used to identify annotated and filtered variants. Candidate variants were validated and the segregation analysis of other family members was performed. Results. This study identified compound heterozygous variants, c.101G>A and c.3832G>A, in the SCN5A gene and c.28730C>T in the TTN gene. Conclusions. Compound heterozygous variants in the SCN5A gene were found in the complete heart block affected child but these two variants were found only in the this affected sibling and were not found in other unaffected family members. Hence, these variants in the SCN5A gene were the most possible disease-causing variants in this family.