Skip to Main Content

RTI uses cookies to offer you the best experience online. By and clicking “accept” on this website, you opt in and you agree to the use of cookies. If you would like to know more about how RTI uses cookies and how to manage them please view our Privacy Policy here. You can “opt out” or change your mind by visiting: http://optout.aboutads.info/. Click “accept” to agree.

Accept
RTI International
  • About
    • Office Locations
    • Executive Leadership
    • Corporate Governance
    • Partner with Us
      • U.S. Government
      • Clients and Funding Agencies
      • Industry and Commercial Clients
      • Foundations and Associations
      • Bilateral Agencies and Multilateral Banks
      • Universities and Academic Research Institutions
      • Suppliers and Small Businesses
    • Commitment to Quality
      • RTI's Client Listening Program
    • Ethics and Human Research Protection
    • Living Our Mission
    • Veteran Opportunities at RTI

    About

  • Practice Areas
    • Health
      • Public Health and Well-Being
      • Health Care Transformation
      • Behavioral Health
      • Health Behavior Change
      • Precision Medicine
      • RTI Health Solutions (RTI-HS)
      • RTI Center for Community Health Evaluation and Economics Research
      • Health Equity
      • RTI Health Advance
    • Transformative Research Unit for Equity​
      • Equity Capacity Building Hub
      • Social and Economic Justice Research Collaborative
      • Narrative Research and Community Engagement Lab
    • Education and Workforce Development
      • Early Childhood
      • K-12 Education
      • Postsecondary Education
      • Career and Adult Education and Workforce Development
      • Education Policy, Systems, and Governance
      • Education Research Methodologies
      • Education Technologies
    • International Development
      • Energy for Development
      • Environment
      • Global Food Security, Agriculture, and Nutrition
      • Global Health
      • International Education
      • Monitoring, Evaluation, Research, Learning, and Adapting (MERLA)
      • Youth and Economic Opportunity
      • Building Resilience Against COVID-19 in Developing Countries
      • Water, Sanitation, and Hygiene (WASH)
      • RTI Center for Governance
    • Climate Change
      • Clean Energy Technology and Renewables
      • Climate Finance
      • Climate Justice and Equity
      • Climate Planning, Preparedness and Resilience
      • Climate Policy
      • Climate Vulnerability, Adaptation, and Mitigation
      • Economic Impacts of Climate Change
    • Water
      • Food-Energy-Water Nexus
      • Water Quality
      • WASH (Water, Sanitation, Hygiene)
      • Water Resources Management
    • Energy Research
      • Carbon Capture and Utilization
      • Biomass Conversion
      • Natural Gas
      • Energy Efficiency
      • Industrial Water
      • Syngas Processing
    • Environmental Sciences
      • Air Quality
      • RTI Center for Water Resources
      • Urban Sustainability
      • Toxics
      • Climate Change
      • Building Resiliency in the FEW Nexus
      • Climate Change Sciences and Analysis
      • Environmental Policy
      • Environmental Justice
      • Sustainable Materials & Waste Management Solutions
    • Justice Research and Policy
      • RTI Center for Community Safety and Crime Prevention
      • RTI Center for Policing Research and Investigative Science
      • Child Well-Being and Family Strengthening
      • RTI Center for Forensic Sciences
    • Food Security and Agriculture
      • Market Systems Strengthening
      • Food Safety
      • Food and Nutrition
      • Global Food Security, Agriculture, and Nutrition
      • Climate-Smart Agriculture
      • Youth in Agriculture
      • Agricultural Innovation
      • Obesity Prevention
    • Innovation Ecosystems
      • Innovation Advising
      • Innovation for Economic Growth
      • Innovation for Emerging and Developing Economies
      • Innovation for Organizations
      • Research, Technology, and Innovation Policy
      • Technology Acceleration
    • Military Support
      • Military Behavioral Health
      • Military Health and Human Performance
      • Military Sexual Assault, Harassment, and Domestic Violence Prevention
      • Wearable Sensor Technologies
      • Military Health System Transformation

    Practice Areas

  • Services + Capabilities
    • Surveys and Data Collection
      • Survey Design
      • Instrument Development
      • Survey Methodologies
      • Data Collection
      • Establishment Surveys
      • Health Registries
      • Data Analysis and Reporting
      • Research Operations Center
    • Statistics and Data Science
      • Survey Statistics
      • Environmental Statistics
      • Coordinating Centers for Multisite Studies
      • Analysis and Design of Complex Data
      • Biostatistics
      • RTI Center for Data Science
    • Evaluation, Assessment and Analysis
      • Evaluation Design and Execution
      • Advanced Qualitative, Quantitative, and Mixed Methods
      • Evaluation, Monitoring, and Assessment
      • Economic Analysis
      • Evaluating Communication Interventions and Campaigns
      • Evidence Synthesis for Policy and Practice
      • Risk Assessment and Prediction
    • Program Design and Implementation
      • Systems Strengthening and Scaling
      • Capacity Assessment and Building
      • Policy Reform Support
      • Curriculum and Teacher Professional Development
      • Interventions and Prevention Programs
      • Implementation Science
    • Digital Solutions for Social Impact
      • Human-Centered Design of Digital Solutions
      • Digital Product Development
      • Digital Communication Campaigns
      • Digital Data Analytics
    • Research Technologies
      • Survey Technologies
      • Data Management and Decision Support Systems
      • Geospatial Science, Technology, and Visualization
      • ICT for Limited-Resource Settings
      • Mobile Applications
      • Web Applications
      • Bioinformatics
      • Interactive Computing
    • Drug Discovery and Development
      • Medicinal Chemistry
      • Molecular Design and Cheminformatics
      • Behavioral Pharmacology
      • Drug Metabolism and Pharmacokinetics (DMPK)
      • In Vitro Pharmacology, Bioassay Development, and High-Throughput Screening (HTS)
      • Isotope Labeling
      • Regulatory Consulting and Support for Medical Products
    • Analytical Laboratory Sciences
      • Bioanalytical and Toxicology Research
      • Forensic Sciences
      • Physicochemical Characterizations
      • Metabolomics
      • Proficiency Testing and Reference Materials
      • Quality Assurance and Regulatory Compliance
      • Microbiology
      • Analytical Chemistry and Pharmaceutics
    • Engineering & Technology R&D
      • Biomedical Technologies
      • Decarbonization Sciences
      • Environmental Exposure & Protection
      • Materials & Environment
      • Sustainable Energy Solutions

    Services + Capabilities

  • Centers
    • RTI Center for Advanced Methods Development
    • RTI Center for Communication Science
      • Communication Research
      • Communication Design
      • Communication Delivery
    • RTI Center for Data Science
    • RTI Center for Education Services
      • Teaching and Learning
      • Education Leadership
      • Peer Learning Networks
      • Strategic Consulting
    • RTI Center for Forensic Sciences
    • RTI Center for Global Noncommunicable Diseases
      • Program Financing & Economics for NCDs
      • Health Systems Strengthening for NCDs
      • Communication Science and Behavior Change for NCDs
      • Implementation Science for NCDs
    • RTI GenOmics, Bioinformatics, and Translational Research Center
      • Disability Studies
      • Ethics
      • Newborn Screening
    • RTI Center for Water Resources
      • Water Resources Sectors
      • Water Resources Services
      • Water Resources Tools
    • RTI Center for Governance
    • RTI Global Gender Center
    • North Carolina Center for Optimizing Military Performance
    • NCCU-RTI Center for Applied Research in Environmental Sciences
    • RTI Center for Climate Solutions

    Centers

  • Impact
    • Newsroom
    • Insights Blog
    • Events
    • Publications
    • RTI Press
      • About the RTI Press
      • Instructions for Authors
      • RTI Press Collections
    • Projects
    • Global Reach
      • Asia
      • Eastern Europe and Central Asia
      • RTI International India
      • Africa
      • Middle East and North Africa (MENA)
      • Latin America and the Caribbean (LAC)

    Impact

  • Experts
    • Our Experts
    • In-Depth With Our Experts
    • Related News
    • Experts In the Media
    • RTI Fellow Program

    Experts

  • Emerging Issues
    • COVID-19 Research
    • Artificial Intelligence
    • Global Health Security
    • Cannabis Research
    • Opioid Research
      • Interventions for Opioid Use Disorders
      • Preventing Opioid Misuse and Overdose
      • Treating Opioid Use Disorders
    • Policing Research and Investigative Science
    • Drone Research and Application
    • E-cigarette Research
    • Zika Virus Research
    • Integrated Governance

    Emerging Issues

  • COVID-19 Research + Response
  • Global Reach
  • Insights Blog
  • Newsroom
  • RTI Press
  • Publications
  • Partner With Us
  • Careers
  • Facebook IconTwitter IconInstagram IconYouTube IconLinkedin Icon
  • Home
  • Impact
  • Publications
  • A literature review of the economic burden of fragile X syndrome

A literature review of the economic burden of fragile X syndrome

Sacco, P. N., Raspa, M., Leahy, L., & Cabo, R. (2018). A literature review of the economic burden of fragile X syndrome. Neurology, 90(15 Supplement), [P3.319].

Copy citation

Abstract

Objective: Published data were evaluated to understand the economic burden of Fragile X syndrome (FXS).

Background: Individuals with FXS experience intellectual disability and reduced functional abilities, including problems with attention and anxiety. Evidence is beginning to converge on the use of medical specialists, allied health professionals, and prescription medication among individuals with FXS. Resource utilization and costs are critical to populate economic models to demonstrate the value of new health technologies for the treatment of FXS.

Design/Methods: A targeted review searched PubMed (2006–2016) using search terms on the following topics: resource utilization (e.g., medical services, medical tests, procedures, therapy, hospitalization) direct and indirect costs (e.g., economic burden, productivity, absenteeism, long term care).

Results: The original search yielded 730 unique abstracts. Sixteen articles were retained; full text evaluation identified 13 studies for this review. Study geographies included: 10 United States (US), 2 European Union (EU), 1 Canada. US administrative claims data found FXS patients had median all-cause direct costs ranging $2222 to $2955 (commercial/Medicare), $4548 to $9702 (Medicaid) and annualized total all-cause mean direct costs of $14,677 (privately insured). Healthcare cost drivers in the US were outpatient visits, home-care and long-term care services, medical procedures, hospitalizations, medications, other out of pocket expenses. In a survey in 5 EU countries, mean total annual direct cost per patient varied widely (€4951 to €58,862); mean annual direct healthcare costs per patient ranged €110 to €2,675. Healthcare cost drivers in the EU were medical visits, hospitalizations, medications. We will report on studies which investigated the association of functional limitations on economic burden.

Conclusions: This review suggests FXS poses substantial economic burden. The cost of care varies across payer types, geographies, symptom severity. Further study is warranted to better characterize symptom-related cost drivers and impact of economic burden for all stakeholders.

Study Supported by: Ovid Therapeutics

Disclosure: Dr. Sacco has received research support from Ovid Therapeutics. Dr. Raspa has received research support from Ovid Therapeutics. Dr. Leahy has received personal compensation for consulting, serving on a scientific advisory board, speaking, or other activities with Ovid Therapeutics. Dr. Cabo has received personal compensation for consulting, serving on a scientific advisory board, speaking, or other activities with Ovid Therapeutics. Dr. Cabo holds stock and/or stock options in Ovid Therapeutics.

Download/Explore
Share
  • Share on Facebook
  • Share on Twitter
  • Share on Linkedin
  • Email

Publications Info

To contact an RTI author, request a report, or for additional information about publications by our experts, send us your request.

  • +1 919 541 8787
  • publications@rti.org

Meet the Experts

View All Experts
Melissa Raspa

Melissa Raspa

Recent Publications

Article

Protection of forest ecosystems in the eastern United States from elevated atmospheric deposition of sulfur and nitrogen

Article

The use of patient experience feedback in rehabilitation quality improvement and codesign activities

Article

SPTSSA variants alter sphingolipid synthesis and cause a complex hereditary spastic paraplegia

OCCASIONAL PAPER

Bringing an equity-centered framework to research

RTI Logo
Partner With Us
  • US Government
  • Commercial
  • Foundations & Associations
  • Multilateral Donors
  • Universities
  • Suppliers
Site
  • Privacy Policy
  • Security Policy
  • Site Map
  • Terms of Use
  • Accessibility
  • Contact Us
Contact Us
Facebook Icon Twitter Icon Instagram Icon YouTube Icon Linkedin Icon
delivering the promise of science
for global good
RTI Health Solutions RTI Innovation Advisors RTI Health Advance

© 2023 RTI International. RTI International is a trade name of Research Triangle Institute. RTI and the RTI logo are U.S. registered trademarks of Research Triangle Institute.