Impact evaluation of a cash-plus programme for children with disabilities in the Xiengkhouang Province in Lao PDR: study protocol for a non-randomised controlled trial.

INTRODUCTION: More than 170 countries have implemented disability-targeted social protection programmes, although few have been rigorously evaluated. Consequently, a non-randomised controlled trial is being conducted of a pilot 'cash-plus' programme implemented by UNICEF Laos and the Laos government for children with disabilities in the Xiengkhouang Province in Laos. The intervention combines a regular cash transfer with provision of assistive devices and access for caregivers to a family support programme. METHODS AND ANALYSIS: The non-randomised controlled trial will involve 350 children with disabilities across 3 districts identified by programme implementers as eligible for the programme (intervention arm). Implementers have also identified approximately 180 children with disabilities in neighbouring districts, who would otherwise meet eligibility criteria but do not live in the project areas (control arm). The trial will assess the impact of the programme on child well-being (primary outcome), as well as household poverty, caregiver quality of life and time use (secondary outcomes). Baseline data are being collected May-October 2023, with endline 24 months later. Analysis will be intention to treat. A complementary process evaluation will explore the implementation, acceptability of the programme, challenges and enablers to its delivery and mechanisms of impact. ETHICS AND DISSEMINATION: The study has received ethical approval from the London School of Hygiene and Tropical Medicine and the National Ethics Committee for Health Research in Laos. Informed consent and assent will be taken by trained data collectors. Data will be collected and stored on a secure, encrypted server and its use will follow a detailed data management plan. Findings will be disseminated in academic journals and in short briefs for policy and programmatic actors, and in online and in-person events. TRIAL REGISTRATION NUMBER: ISRCTN80603476

Development of a Discrete Choice Experiment (DCE) questionnaire to elicit values by pregnant women and decision-makers for the expansion of a NIPS-based prenatal screening program

Abstract BackgroundIn an accountable world, being able to take into account the value given by relevant stakeholders to an intervention that could be offered to the population is considered as desirable. DCE is an approach particularly suited for the measurement of such values in the field of prenatal care. Yet, DCE studies in the field of prenatal screening have focused mainly on pregnant women and their care providers but have neglected another key actor, the decision-makers. The objective of the study was to develop a DCE instrument applicable to pregnant women and decision-makers, for the evaluation of new conditions to be added to a screening program for fetal chromosomal anomalies.MethodsAn instrument development study was undertaken. Methods employed included a literature review, a qualitative study performed on pregnant women and decision-makers, and a pilot project to validate the developed instrument and test the feasibility of its administration through an online survey platform. ResultsAn initial list of ten attributes and levels were built from the information provided by the literature review and the qualitative research component of the study. Seven attributes were built based on responses provided by participants from both groups. Two attributes were built from what was said by women only and one from what was said by decision-makers only. Search for consensus through consultations and a focus group discussion led to the retention of eight attributes. A pilot project was then performed with 33 pregnant women. This led to the exclusion of one attribute that showed poor influence on the choice making. The final version of the instrument contains seven attributes.ConclusionThis paper presents the construction of a DCE instrument that can be administered to pregnant women on the demand side, and decision-makers on the supply side. Such an instrument to measure the social desirability of an intervention could be an added value to the decision-making process of Health Technology Assessment agencies.</jats:p

Expansion of non-invasive prenatal screening to the screening of 10 types of chromosomal anomalies: a cost-effectiveness analysis

Objectives To determine the cost-effectiveness of the addition of chromosomal anomalies detectable by non-invasive prenatal screening (NIPS), in a prenatal screening programme targeting common aneuploidies.Design, setting and participants A simulation study was conducted to study the addition of chromosomal anomalies detectable by NIPS (sex chromosome aneuploidies, 22q11.2 deletion syndrome, large deletion/duplication &gt;7 Mb and rare autosomal trisomies) to five basic strategies currently aiming the common trisomies: three strategies currently offered by the public healthcare systems in Canada, whose first-tier test is performed with biochemical markers, and two programmes whose first-tier test consists of NIPS-based methods.Outcome measures The total number of cases of chromosomal anomalies detected and the costs related to the consumption of medical services.Results The most effective and the most cost-effective option in almost all prenatal screening strategies is the option that includes all targeted additional conditions. In the strategies where NIPS is used as first-tier testing, the cost per additional case detected by adding all possible additional anomalies to a programme that currently targets only common trisomies is $C25 710 (95$C25 489 to $C25 934) for massively parallel shotgun sequencing and$C57 711 (95% CI $C57 141 to$C58 292) for targeted massively parallel sequencing, respectively. The acceptability curves show that at a willingness-to-pay of $C50 000 per one additional case detected, the expansion of NIPS-based methods for the detection of all possible additional conditions has a 90% probability of being cost-effective.Conclusion From an economic perspective, in strategies that use NIPS as a first-tier screening test, expanding the programmes to detect any considered chromosomal anomalies other than the three common trisomies would be cost-effective. However, the potential expansion of prenatal screening programmes also requires consideration of societal issues, including ethical ones

DCE pilot study design.

ObjectiveAn instrument for measuring intervention preferences applicable to both patients and policymakers would make it possible to better confront the needs of the supply and demand sides of the health care system. This study aimed to develop a discrete choice experiments (DCE) questionnaire to elicit the preferences of patients and policymakers. The instrument was specifically developed to estimate preferences for new conditions to be added to a screening program for fetal chromosomal anomalies.MethodsA DCE development study was conducted. The methods employed included a literature review, a qualitative study (based on individual semi-structured interviews, consultations, and a focus group discussion) with pregnant women and policymakers, and a pilot project with 33 pregnant women to validate the first version of the instrument and test the feasibility of its administration.ResultsAn initial list of 10 attributes was built based on a literature review and the qualitative research components of the study. Five attributes were built based on the responses provided by the participants from both groups. Eight attributes were consensually retained. A pilot project performed on 33 pregnant women led to a final instrument containing seven attributes: ‘conditions to be screened’, ‘test performance’, ‘moment at gestational age to obtain the test result’, ‘degree of test result certainty to the severity of the disability’, ‘test sufficiency’, ‘information provided from test result’, and ‘cost related to the test’.ConclusionIt is possible to reach a consensus on the construction of a DCE instrument intended to be administered to pregnant women and policymakers. However, complete validation of the consensual instrument is limited because there are too few voting members of health technology assessment agencies committees to statistically ascertain the relevance of the attributes and their levels.</div

Study protocol for a descriptive analysis of non-invasive prenatal testing uptake and performance in singleton and twin pregnancies using Ontario birth registry data

Introduction Despite the availability of funded first-tier non-invasive prenatal testing (NIPT) for twin pregnancies in Ontario, Canada, research gaps persist regarding the feasibility and effectiveness of NIPT in this demographic. This protocol documents our planned comprehensive overview of twin data from the large Ontario provincial registry and evaluates the performance of NIPT among singleton and twin pregnancies.Methods and analysis We will conduct a descriptive study using routinely collected data housed in the Better Outcomes Registry &amp; Network Ontario. The study population will include all singleton and twin pregnancies with an estimated date of delivery between 1 September 2016 and 31 March 2023. We will compare patient characteristics, NIPT uptake and test performance metrics (including sensitivity, specificity, positive predictive value and negative predictive value) between singleton and twin pregnancies. Subgroup analyses will be conducted, including assessment by the mode of conception, trimester of initial screening, age of the pregnant individual and eligibility for publicly funded first-tier NIPT.Ethics and dissemination This study has received approval from the Research Ethics Boards of the Children’s Hospital of Eastern Ontario (24/01PE) and the University of Ottawa (H-04-24-10309). Results will be disseminated through scientific conferences and publication in a peer-reviewed journal. By making our protocol and findings publicly available, we aim to establish a foundational reference for future investigations in Ontario. Additionally, we seek to support the design and implementation of further studies on NIPT in twin pregnancies in Canada and elsewhere

Attributes/levels identification.

ObjectiveAn instrument for measuring intervention preferences applicable to both patients and policymakers would make it possible to better confront the needs of the supply and demand sides of the health care system. This study aimed to develop a discrete choice experiments (DCE) questionnaire to elicit the preferences of patients and policymakers. The instrument was specifically developed to estimate preferences for new conditions to be added to a screening program for fetal chromosomal anomalies.MethodsA DCE development study was conducted. The methods employed included a literature review, a qualitative study (based on individual semi-structured interviews, consultations, and a focus group discussion) with pregnant women and policymakers, and a pilot project with 33 pregnant women to validate the first version of the instrument and test the feasibility of its administration.ResultsAn initial list of 10 attributes was built based on a literature review and the qualitative research components of the study. Five attributes were built based on the responses provided by the participants from both groups. Eight attributes were consensually retained. A pilot project performed on 33 pregnant women led to a final instrument containing seven attributes: ‘conditions to be screened’, ‘test performance’, ‘moment at gestational age to obtain the test result’, ‘degree of test result certainty to the severity of the disability’, ‘test sufficiency’, ‘information provided from test result’, and ‘cost related to the test’.ConclusionIt is possible to reach a consensus on the construction of a DCE instrument intended to be administered to pregnant women and policymakers. However, complete validation of the consensual instrument is limited because there are too few voting members of health technology assessment agencies committees to statistically ascertain the relevance of the attributes and their levels.</div

Final list of attributes and levels for full-scale DCE.

Final list of attributes and levels for full-scale DCE.</p

Interview guide.

ObjectiveAn instrument for measuring intervention preferences applicable to both patients and policymakers would make it possible to better confront the needs of the supply and demand sides of the health care system. This study aimed to develop a discrete choice experiments (DCE) questionnaire to elicit the preferences of patients and policymakers. The instrument was specifically developed to estimate preferences for new conditions to be added to a screening program for fetal chromosomal anomalies.MethodsA DCE development study was conducted. The methods employed included a literature review, a qualitative study (based on individual semi-structured interviews, consultations, and a focus group discussion) with pregnant women and policymakers, and a pilot project with 33 pregnant women to validate the first version of the instrument and test the feasibility of its administration.ResultsAn initial list of 10 attributes was built based on a literature review and the qualitative research components of the study. Five attributes were built based on the responses provided by the participants from both groups. Eight attributes were consensually retained. A pilot project performed on 33 pregnant women led to a final instrument containing seven attributes: ‘conditions to be screened’, ‘test performance’, ‘moment at gestational age to obtain the test result’, ‘degree of test result certainty to the severity of the disability’, ‘test sufficiency’, ‘information provided from test result’, and ‘cost related to the test’.ConclusionIt is possible to reach a consensus on the construction of a DCE instrument intended to be administered to pregnant women and policymakers. However, complete validation of the consensual instrument is limited because there are too few voting members of health technology assessment agencies committees to statistically ascertain the relevance of the attributes and their levels.</div

List of attribute and attribute levels for the pilot project.

List of attribute and attribute levels for the pilot project.</p

List of potential attributes and levels for consultations and focused group discussion.

List of potential attributes and levels for consultations and focused group discussion.</p