An integrated modelling methodology for estimating global incidence and prevalence of hereditary spastic paraplegia subtypes SPG4, SPG7, SPG11, and SPG15.

BACKGROUND: Hereditary spastic paraplegias (HSPs) are progressively debilitating neurodegenerative disorders that follow heterogenous patterns of Mendelian inheritance. Available epidemiological evidence provides limited incidence and prevalence data, especially at the genetic subtype level, preventing a realistic estimation of the true social burden of the disease. The objectives of this study were to (1) review the literature on epidemiology of HSPs; and (2) develop an epidemiological model of the prevalence of HSP, focusing on four common HSP genetic subtypes at the country and region-level. METHODS: A model was constructed estimating the incidence at birth, survival, and prevalence of four genetic subtypes of HSP based on the most appropriate published literature. The key model parameters were assessed by HSP clinical experts, who provided feedback on the validity of assumptions. A model was then finalized and validated through comparison of outputs against available evidence. The global, regional, and national prevalence and patient pool were calculated per geographic region and per genetic subtype. RESULTS: The HSP global prevalence was estimated to be 3.6 per 100,000 for all HSP forms, whilst the estimated global prevalence per genetic subtype was 0.90 (SPG4), 0.22 (SPG7), 0.34 (SPG11), and 0.13 (SPG15), respectively. This equates to an estimated 3365 (SPG4) and 872 (SPG11) symptomatic patients, respectively, in the USA. CONCLUSIONS: This is the first epidemiological model of HSP prevalence at the genetic subtype-level reported at multiple geographic levels. This study offers additional data to better capture the burden of illness due to mutations in common genes causing HSP, that can inform public health policy and healthcare service planning, especially in regions with higher estimated prevalence of HSP.

Correction: Developing Theory-Driven, Evidence-Based Serious Games for Health: Framework Based on Research Community Insights.

[This corrects the article DOI: 10.2196/11565.].

Improving Patient Preference Elicitation by Applying Concepts From the Consumer Research Field: Narrative Literature Review.

BACKGROUND: Although preference research finds its origins in consumer research, preference elicitation methods have increasingly attracted attention in different decision-making contexts in health care. Simulating real-life decision making is believed to be important during consumer preference elicitation. OBJECTIVE: The aims of this study were to compare the process of decision making between patients and consumers and to identify methods from the consumer research field that could be applied in patient preference elicitation. METHODS: A narrative literature review was performed to identify preference elicitation concepts from a consumer context that could offer improvements in health care. RESULTS: The process of decision making between patients and consumers was highly comparable. The following five concepts from the consumer research field that could effectively simulate a real-life decision-making process for applications in health care were identified: simulating alternatives, self-reflection, feedback-driven exploration, separated (adaptive) dual response, and arranging profiles in blocks. CONCLUSIONS: Owing to similarities in the decision-making process, patients could be considered as a subgroup of consumers, suggesting that preference elicitation concepts from the consumer field may be relevant in health care. Five concepts that help to simulate real-life decision making have the potential to improve patient preference elicitation. However, the extent to which real decision-making contexts can be mimicked in health care remains unknown.

A Picture is Worth a Thousand Words: The Role of Survey Training Materials in Stated-Preference Studies.

BACKGROUND: Online survey-based methods are increasingly used to elicit preferences for healthcare. This digitization creates an opportunity for interactive survey elements, potentially improving respondents' understanding and/or engagement. OBJECTIVE: Our objective was to understand whether, and how, training materials in a survey influenced stated preferences. METHODS: An online discrete-choice experiment (DCE) was designed to elicit public preferences for a new targeted approach to prescribing biologics ("biologic calculator") for rheumatoid arthritis (RA) compared with conventional prescribing. The DCE presented three alternatives, two biologic calculators and a conventional approach (opt out), described by five attributes: delay to treatment, positive predictive value, negative predictive value, infection risk, and cost saving to the national health service. Respondents were randomized to receive training materials as plain text or an animated storyline. Training materials contained information about RA and approaches to treatment and described the biologic calculator. Background questions included sociodemographics and self-reported measures of task difficulty and attribute non-attendance. DCE data were analyzed using conditional and heteroskedastic conditional logit (HCL) models. RESULTS: In total, 300 respondents completed the DCE, receiving either plain text (n = 158) or the animated storyline (n = 142). The HCL showed the estimated coefficients for all attributes aligned with a priori expectations and were statistically significant. The scale term was statistically significant, indicating that respondents who received plain-text materials had more random choices. Further tests suggested preference homogeneity after accounting for differences in scale. CONCLUSIONS: Using animated training materials did not change the preferences of respondents, but they appeared to improve choice consistency, potentially allowing researchers to include more complex designs with increased numbers of attributes, levels, alternatives or choice sets.

A pilot study of the effectiveness of a serious game CliniPup® on perioperative anxiety and pain in children.

INTRODUCTION/AIM: Children experience important anxiety before surgery. Anxiety and pain are positively correlated. Serious gaming is a non-pharmacological intervention to prepare children and parents for an operation. We aimed to evaluate the effectiveness of the serious game CliniPup® on anxiety and pain in children undergoing ambulatory surgery. METHOD: A prospective randomized controlled pilot trial in 72 children aged 5 to 11 years old scheduled for day-care surgery (general surgery, dentistry, otorhinolaryngology, urology) was performed. Participants were randomly assigned into 3 groups: A (CliniPup®), B ("Empty game" without educational information), or C (no game, oral information at the outpatient clinic, current standard of care). Anxiety, pain, and behaviour were evaluated by validated instruments at six time-points: T0: baseline, T1: 1 week preoperatively, T2: at hospital admission, T3: before discharge, T4: 1 week postoperatively, T5: 1 month postoperatively. RESULTS: After playing the game (T1), the estimated mean anxiety score (VASa) was lower in Group A (1.9 units) versus Group B (2.7 units). The estimated mean VASa at T1 for Group A was 2.6 units lower compared to Group C (p = 0.003). For Group B, VASa levels were 1.8 units lower than in Group C (p = 0.045). After correction for "surgery type", Group A continued to show a significantly lower VASa compared to Group C (p = 0.044). On the other time points, no difference in anxiety and pain were observed, nor in post-hospitalization behaviour. CONCLUSION: Children that played the CliniPup® game one week before surgery had a significant reduction in preoperative anxiety after playing the game, but not on the other time points. No differences on peri-operative pain were observed during the different time points. TYPE OF STUDY: Randomized Trial. LEVEL OF EVIDENCE: Level II.

Developing Theory-Driven, Evidence-Based Serious Games for Health: Framework Based on Research Community Insights.

BACKGROUND: The idea of using serious games to effectuate better outcomes in health care has gained significant traction among a growing community of researchers, developers, and health care professionals. Many now recognize the importance of creating evidence-based games that are purposefully designed to address physical and mental health challenges faced by end users. To date, no regulatory resources have been established to guide the development of serious games for health (SGH). Developers must therefore look elsewhere for guidance. Although a more robust level of evidence exists in the research literature, it is neither structured nor is there any clear consensus. Developers currently use a variety of approaches and methodologies. The establishment of a well-defined framework that represents the consensus views of the SGH research community would help developers improve the efficiency of internal development processes, as well as chances of success. A consensus framework would also enhance the credibility of SGH and help provide quality evidence of their effectiveness. OBJECTIVE: This research aimed to (1) identify and evaluate the requirements, recommendations, and guidelines proposed by the SGH community in the research literature, and; (2) develop a consensus framework to guide developers, designers, researchers, and health care professionals in the development of evidence-based SGH. METHODS: A critical review of the literature was performed in October to November 2018. A 3-step search strategy and a predefined set of inclusion criteria were used to identify relevant articles in PubMed, ScienceDirect, Institute of Electrical and Electronics Engineers Xplore, CiteSeerX, and Google Scholar. A supplemental search of publications from regulatory authorities was conducted to capture their specific requirements. Three researchers independently evaluated the identified articles. The evidence was coded and categorized for analysis. RESULTS: This review identified 5 categories of high-level requirements and 20 low-level requirements suggested by the SGH community. These advocate a methodological approach that is multidisciplinary, iterative, and participatory. On the basis of the requirements identified, we propose a framework for developing theory-driven, evidence-based SGH. It comprises 5 stages that are informed by various stakeholders. It focuses on building strong scientific and design foundations that guide the creative and technical development. It includes quantitative trials to evaluate whether the SGH achieve the intended outcomes, as well as efforts to disseminate trial findings and follow-up monitoring after the SGH are rolled out for use. CONCLUSIONS: This review resulted in the formulation of a framework for developing theory-driven, evidence-based SGH that represents many of the requirements set out by SGH stakeholders in the literature. It covers all aspects of the development process (scientific, technological, and design) and is transparently described in sufficient detail to allow SGH stakeholders to implement it in a wide variety of projects, irrespective of discipline, health care segments, or focus.

Development of CliniPup, a Serious Game Aimed at Reducing Perioperative Anxiety and Pain in Children: Mixed Methods Study.

BACKGROUND: An increasing number of children undergo ambulatory surgery each year, and a significant proportion experience substantial preoperative anxiety and postoperative pain. The management of perioperative anxiety and pain remains challenging in children and is inadequate, which negatively impacts the physical, psychosocial, and economic outcomes. Existing nonpharmacological interventions are costly, time consuming, vary in availability, and lack benefits. Therefore, there is a need for an evidence-based, accessible, nonpharmacological intervention as an adjunct to existing pharmacological alternatives to reduce perioperative anxiety and pain in children undergoing ambulatory surgery. Technology-enabled interventions have been proposed as a method to address the unmet need in this setting. In particular, serious games hold a unique potential to change health beliefs and behaviors in children. OBJECTIVE: The objective of this research was to describe the rationale, scientific evidence, design aspects, and features of CliniPup, a serious game aimed at reducing perioperative anxiety and pain in children undergoing ambulatory surgery. METHODS: The SERES Framework for serious game development was used to create the serious game, CliniPup. In particular, we used a mixed methods approach that consisted of a structured literature review supplemented with ethnographic research, such as expert interviews and a time-motion exercise. The resulting scientific evidence base was leveraged to ensure that the resulting serious game was relevant, realistic, and theory driven. A participatory design approach was applied, wherein clinical experts qualitatively reviewed several versions of the serious game, and an iterative creative process was used to integrate the applicable feedback. RESULTS: CliniPup, a serious game, was developed to incorporate a scientific evidence base from a structured literature review, realistic content collected during ethnographic research such as expert interviews, explicit pedagogical objectives from scientific literature, and game mechanics and user interface design that address key aspects of the evidence. CONCLUSIONS: This report details the systematic development of CliniPup, a serious game aimed at reducing perioperative anxiety and pain in children undergoing ambulatory surgery. Clinical experts validated CliniPup's underlying scientific evidence base and design foundations, suggesting that it was well designed for preliminary evaluation in the target population. An evaluation plan is proposed and briefly described.

A Web-Based Serious Game for Health to Reduce Perioperative Anxiety and Pain in Children (CliniPup): Pilot Randomized Controlled Trial.

BACKGROUND: As pediatric ambulatory surgeries are rising and existing methods to reduce perioperative anxiety and pain are lacking in this population, a serious game for health (SGH), CliniPup, was developed to address this unmet need. CliniPup was generated using the SERES framework for serious game development. OBJECTIVE: The goal of the research was to clinically evaluate CliniPup as an adjunct therapy to existing pharmacological interventions aimed at reducing perioperative anxiety and pain in children undergoing ambulatory surgery. METHODS: CliniPup was evaluated in a prospective randomized controlled pilot trial in 20 children aged 6 to 10 years who underwent elective surgery and their parents. Study participants were randomly assigned to the test (n=12) or control group (n=8). Children in the test group played CliniPup 2 days prior to surgery, and children in the control group received standard of care. On the day of surgery, pediatric anxiety was measured with the modified Yale Preoperative Anxiety Scale and parental anxiety was assessed with the State-Trait Anxiety Inventory. Pediatric postoperative pain was assessed by the Wong-Baker Faces Pain Rating Scale. Child and parent user experience and satisfaction were also evaluated in the test group using structured questionnaires. RESULTS: Despite the small sample, preoperative anxiety scores were significantly lower (P=.01) in children who played CliniPup prior to surgery compared to controls. Parental preoperative anxiety scores were also lower in the test group (P=.10) but did not reach significance. No significant differences were observed in postoperative pain scores between groups (P=.54). The evaluation of user experience and satisfaction revealed that both children and parents were satisfied with CliniPup and would recommend the game to peers. CONCLUSIONS: Results of the pilot trial introduce CliniPup as a potentially effective and attractive adjunct therapy to reduce preoperative anxiety in children undergoing ambulatory surgery with a trend toward positive impact on parental preoperative anxiety. These results support the use of the SERES framework to generate an evidence-based SGH that results in positive health outcomes for patients. Based on these preliminary findings, we propose a research agenda to further develop and investigate this tool. TRIAL REGISTRATION: ClinicalTrials.gov NCT03874442; https://clinicaltrials.gov/ct2/show/NCT03874442 (Archived by WebCite at http://www.webcitation.org/78KZab8qc).