Economic Evaluation of Family-Focused Programs When Parents Have a Mental Health Problem: Methodological Considerations.

OBJECTIVES: The nature of adverse effects of parental mental health problems and of the interventions to address them may require specific designs of economic evaluation studies. Nevertheless, methodological guidance is lacking. We aim to understand the broad spectrum of adverse effects from parental mental health problems in children and the economic consequences on an individual and societal level to navigate the design of economic evaluations in this field. METHODS: We conducted a systematic literature search of empirical studies on children's adverse effects from parental mental illness. We clustered types of impact, identified individual and public cost consequences, and illustrated the results in an impact inventory. RESULTS: We found a wide variety of short- and long-term (mental) health impacts, impacts on social functioning and socioeconomic implications for the children individually, and adverse effects on the societal level. Consequently, public costs can occur in various public sectors (eg, healthcare, education), and individuals may have to pay costs privately. CONCLUSIONS: Existing evaluations in this field mostly follow standard methodological approaches (eg, cost-utility analysis using quality-adjusted life-years) and apply a short-time horizon. Our findings suggest applying a long-term time horizon (at least up to early adulthood), considering cost-consequence analysis and alternatives to health-related quality of life and quality-adjusted life-years as outcome measures, and capturing the full range of possible public and private costs.

A theory-informed systematic review of clinicians' genetic testing practices.

This systematic literature review investigates factors impacting on clinicians' decisions to offer genetic tests in their practice, and maps them to a theoretical behaviour change framework. Better understanding of these factors will inform the design of effective interventions to integrate genomics tests into clinical care. We conducted a narrative synthesis of empirical research of medical specialists' perspectives on and experiences of offering genetic tests to their patients. This review was based upon the PRISMA statement and guidelines for reviewing qualitative research. Four electronic data sources were searched-MEDLINE, EMBASE, CINAHL, PubMed. Studies were independently assessed by two authors. Content analysis was applied to map the findings of included studies to a framework validated for behaviour and implementation research, the Theoretical Domains Framework (TDF). The TDF describes 14 factors known to influence behaviour and has been applied in diverse clinical settings to understand and/or modify health professional behaviour. Thirty-four studies published in 39 articles met inclusion and quality criteria. Most studies were published after 2011 (54%), Northern American (82%), quantitative in design (68%) and addressed familial cancer genetic tests (53%). Of the 14 TDF factors, 13 were identified. The three most common factors were: Environmental Context and Resources (n = 33), Beliefs about Consequences (n = 26), and Knowledge (n = 23). To support the adoption of genomic tests beyond specialist services, nuanced interventions targeting considerations beyond clinician education are needed. For instance, interventions addressing organisational constraints which may restrict clinicians' ability to offer genomic tests are required alongside those targeting factors intrinsic to the clinician.

Improving family communication after a new genetic diagnosis: a randomised controlled trial of a genetic counselling intervention.

BACKGROUND: Genetic information given to an individual newly diagnosed with a genetic condition is likely to have important health implications for other family members. The task of communicating with these relatives commonly falls to the newly diagnosed person. Talking to relatives about genetic information can be challenging and is influenced by many factors including family dynamics. Research shows that many relatives remain unaware of relevant genetic information and the possible impact on their own health. This study aims to evaluate whether a specific genetic counselling intervention for people newly diagnosed with a genetic condition, implemented over the telephone on a number of occasions, could increase the number of at-risk relatives who make contact with genetics services after a new genetic diagnosis within a family. METHODS: This is a prospective, multi-centre randomised controlled trial being conducted at genetics clinics at five public hospitals in Victoria, Australia. A complex genetic counselling intervention has been developed specifically for this trial. Probands (the first person in a family to present with a diagnosis of a genetic condition) are being recruited and randomised into one of two arms - the telephone genetic counselling intervention arm and the control arm receiving usual care. The number of at-risk relatives for each proband will be estimated from a family pedigree collected at the time of diagnosis. The primary outcome will be measured by comparing the proportion of at-risk relatives in each arm of the trial who make subsequent contact with genetics services. DISCUSSION: This study, the first randomised controlled trial of a complex genetic counselling intervention to enhance family communication, will provide evidence about how best to assist probands to communicate important new genetic information to their at-risk relatives. This will inform genetic counselling practice in the context of future genomic testing. TRIAL REGISTRATION: Australia and New Zealand Clinical Trials Register (ANZCTR): ANZCTRN12608000642381.