research articles on disability

Articles about Disability and Health

Key findings, feature articles, recent scientific articles.

Many Adults with Disabilities Report Frequent Mental Distress A study from the CDC found that adults with disabilities report experiencing more mental distress than those without disabilities.

Prevalence of Disability and Disability Types by Urban-Rural County Classification – United States, 2016 The  American Journal of Preventive Medicine  published a new report comparing the percentage of adults with disabilities living in urban versus rural U.S counties.

Prevalence of Disabilities and Health Care Access by Disability Status and Type Among Adults — United States, 2016 Read the published report describing adults with disabilities in the US, as well as the differences in healthcare access by disability type.

Using Medicaid Data to Characterize Persons with Intellectual and Developmental Disabilities in Five U.S. States An article in the American Journal on Intellectual and Developmental Disabilities used Medicaid data from 2008-2013 to identify and describe people with intellectual or developmental disability (IDD) in five states (Delaware, Iowa, Massachusetts, New York, and South Carolina).

Article Highlights: The Guide to Community Preventive Services and Disability Inclusion In a recent report in the American Journal of Preventive Medicine (AJPM), CDC scientists summarize how public health recommendations from the Guide to Community Preventive Services (Community Guide) can be adapted to better benefit people with disabilities.

Primary Care Providers’ Level of Preparedness for Recommending Physical Activity to Adults with Disabilities The Centers for Disease Control and Prevention’s (CDC’s) Preventing Chronic Disease journal published a study that looked at how prepared primary care providers (PCPs) feel to recommend physical activity to adults with disabilities.

Adults with One or More Functional Disabilities The Morbidity and Mortality Weekly Report (MMWR) has published a new report that describes the demographic and socioeconomic characteristics of working-age (18-64 years) adults with disabilities living in U.S. communities.

Socioeconomic Factors at the Intersection of Race and Ethnicity Influencing Health Risks for People with Disabilities The Journal of Racial and Ethnic Health Disparities has published a new study looking at the relationship between income and education, and two health behaviors—smoking and obesity—among people with disabilities from different racial and ethnic groups.

CDC’s Commitment to Disability Inclusion We celebrate the 33 rd  anniversary of the Americans with Disabilities Act (ADA), a civil rights law that promotes the inclusion of people with disabilities in every aspect of life.

Special Olympics Athletes Team Up to Roll-Out Fun Online Workouts Special Olympics fitness programming has demonstrated increases in positive health behaviors and health outcomes for athletes.

COVID-19 and People with Disabilities Learn how to build a disability-inclusive, accessible, and sustainable post COVID-19 world.

Disability and Health Data Learn about the health of people with disabilities in your state.

CDC and Special Olympics: Inclusive Health Learn how the Centers for Disease Control and Prevention (CDC) is working with Special Olympics to improve the health of people with ID.

Mental Health for All Learn some tips that people with disabilities can use to cope with stress.

Disability and Diabetes Prevention If you have a disability, learn what you can do to prevent or manage type 2 diabetes.

Physical Activity for People with Disabilities Do you have a disability? Find your own path to physical activity.

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COVID-19 Cases and Hospitalizations Among Medicare Beneficiaries With and Without Disabilities — United States, January 1, 2020–November 20, 2021. MMWR Morb Mortal Wkly Rep 2022;71:791–796. Yuan Y, Thierry JM, Bull-Otterson L, et al. Read Article |  Easy Read Summary

Physical activity types among US adults with mobility disability, Behavioral Risk Factor Surveillance System, 2017. Disabil Health J. 2020. Feb 3 Hollis ND, Zhang QC, Cyrus AC, Courtney-Long E, Watson K, Carroll D. Read article

Differences in perceived neighborhood environmental supports and barriers for walking between US adults with and without a disability. Prev Med. 2020; 134:106065. Omura JD, Hyde ET, Whitfield G.P, Hollis ND, Fulton JE, Carlson SA. Read article

Comparisons of Estimates from the Behavioral Health Risk Factor Surveillance System and Other National Health Surveys, 2011-2016 Am. J. Prev. Med. 2020 Jun;58(6):e181-e190. Jason Hsia, Guixiang Zhao, Machell Town, Junling Ren, Catherine A Okoro, Carol Pierannunzi, William Garvin. Read article

Prevalence of Disability and Disability Types by Urban-Rural County Classification – United States, 2016 Zhao, Guixiang et al. Prevalence of Disability and Disability Types by Urban–Rural County Classification—U.S., 2016 American Journal of Preventive Medicine, Volume 57, Issue 6, 749 – 756 Read article

Prevalence of disabilities and health care access by disability status and type among adults – United States, 2016 MMWR Morb Mortal Wkly Rep 2018; 64: 777-783. 67: 882–887 Okoro CA, Hollis ND, Cyrus AC, Griffin-Blake S. Read article

Using Medicaid data to characterize persons with IDD in five US states Am J Intellect Dev Disabil. 2018;123:371-381 McDermott S, Royer J, Cope T, et al. Read article

Factors associated with ambulatory care sensitive emergency department visits for South Carolina Medicaid members with ID J Intellect Disabil Res. 2018; 62:165-178 McDermott S, Royer J, Mann JR, Armour BS. Read article

Enhancing individual and community disaster preparedness: Individuals with disabilities and others with access and functional needs Disabil Health J. 2018;11:170-173 Kruger J, Hinton CF, Sinclair LB, Silverman B. Read article

Considering disability and health: Reflections on the Healthy People 2020 Midcourse Review Disabil Health J. 2018;11:333-338 Sinclair LB, Fox MH, Jonas BS, et al. Read article

The Guide to Community Preventive Services and Disability Inclusion Am J Prev Med. 2017; 53:898-903 Hinton CF, Kraus LE, Richards TA, et al. Read article

Primary Care Providers’ Level of Preparedness for Recommending Physical Activity to Adults With Disabilities Prev Chronic Dis 2017; 14:170328 Courtney-Long EA, Stevens AC, Carroll DD, et al. Read article

A longitudinal assessment of adherence to breast and cervical cancer screening recommendations among women with and without intellectual disability. Prev Med. 2017 Jul;100:167-172. Xu X, McDermott SW, Mann JR, Hardin JW, Deroche CB, Carroll DD, Courtney-Long EA. Read article

Socioeconomic Factors at the Intersection of Race and Ethnicity Influencing Health Risks for People with Disabilities Courtney-Long, E.A., Romano, S.D., Carroll, D.D. et al. J. Racial and Ethnic Health Disparities (2017) 4: 213. doi:10.1007/s40615-016-0220-5 Read summary

Comparison of 2 Disability Measures, Behavioral Risk Factor Surveillance System, 2013 Prev Chronic Dis 2016;13:160080 Stevens AC, Courtney-Long EA, Okoro CA, Carroll DD. Read article

Prevalence and Causes of Paralysis—United States, 2013 Am J Public Health. 2016 Oct;106(10):1855-7. Armour BS, Courtney-Long EA, Fox MH, Fredine H, Cahill A. Read article

Adults with One or More Functional Disabilities — United States, 2011–2014 MMWR Morb Mortal Wkly Rep 2016 / 65(38);1021–1025 Stevens AC, Carroll DD, Courtney-Long EA, et al. Read article

Additional Selected Articles

Prevalence of Disability and Disability Type among Adults, United States – 2013 MMWR Morb Mortal Wkly Rep 2015; 64: 777-783. Courtney-Long EA, Carroll DD, Zhang Q, et al. [ Read Article ]

Vital Signs: Disability and Physical Activity – United States, 2009-2012. MMWR; May 2014; 63. Carroll D, Courtney-Long E, Stevens A, Sloan M, Lullo C, Visser S, Fox M, Armour B, Campbell V, Brown D, and Dorn, J. [ Read Article ]

Disability Prevalence Among Healthy Weight, Overweight, and Obese Adults. Obesity (Silver Spring). 2013 Apr;21(4):852-5. doi: 10.1002/oby.20312. Armour BS, Courtney-Long EA, Campbell VA and Wethington HR. [ Read summary ]

Health Disparities of Adults with Intellectual Disabilities: What Do We Know? What Do We Do? Journal of Applied Research in Intellectual Disabilities.  2013 Jul 31. doi: 10.1111/jar.12067. Krahn G, Fox MH. [ Read summary ]

Using Population-Based Data to Examine Preventive Services by Disability Type among Dually Eligible (Medicare/Medicaid) Adults. The Disability and Health Journal. April, 2013; 6(2); 75-86. Reichard A, Fox MH. [ Read summary ]

Estimating disability prevalence among adults by body mass index: 2003-2009 National Health Interview Survey. Prev Chronic Dis. 2012; 9:120-136. Armour BS, Courtney-Long E, Campbell VA, Wethington HR. [ Read summary ]

CDC Grand Rounds: Public Health Practices to Include Persons with Disabilities. Centers for Disease Control and Prevention.  MMWR 2013;62:697-701. [ Read article ]

Factors associated with self-reported mammography use for women with and women without a disability . J Women’s Health 2011; 20:1279-86. Courtney-Long E, Armour B, Frammartino B, Miller J. [ Read summary ]

State-level differences in breast and cervical cancer screening by disability status: United States, 2008 Women’s Health Issues; Nov-Dec 2009; 19(6): 406-14. Armour BS, Thierry JM, Wolf LA. [ Read summary ]

Economic costs associated with mental retardation, cerebral palsy, hearing loss, and vision impairment — United States, 2003 Morbidity and Mortality Weekly Report; January 2004; 53(3): 57-59. CDC [ Read article ]

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• Published: 15 September 2022

How we can make academia more disability inclusive

• Jonathan M. Levitt 1  

Nature Human Behaviour volume  6 ,  pages 1324–1326 ( 2022 ) Cite this article

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The low representation of academics with disabilities is a longstanding problem on which progress has been slow. Drawing on my research on disability-related barriers and my experiences of disability, I make six practical suggestions for how academic staff and people with disabilities can help make academia more disability inclusive.

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Office for National Statistics. Employment by disability. Disability and employment, UK https://www.ons.gov.uk/peoplepopulationandcommunity/healthandsocialcare/disability/bulletins/disabilityandemploymentuk/2019#employment-by-disability (2019).

Office for National Statistics. Disability and education, UK https://www.ons.gov.uk/peoplepopulationandcommunity/healthandsocialcare/disability/bulletins/disabilityandeducationuk/2019 (2019).

Brock, J. “Textbook case” of disability discrimination in grant applications. Nature Index https://www.nature.com/nature-index/news-blog/textbook-case-of-disability-discrimination-in-research-grant-applications (2021).

Bhopal, K., Brown, H. & Jackson, J. in Dismantling Race in Higher Education: Racism, Whiteness and Decolonising the Academy (eds. Arday, J. & Mirza, H. S.) 125–139 (Palgrave Macmillan, 2018).

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I thank M. Levitt, C. Oppenheim and M. Thelwall for their helpful feedback, and F. Moreira for his helpful research support with the visually intensive parts of this paper.

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Disability Studies: Foundations & Key Concepts

This non-exhaustive reading list highlights some of the key debates and conceptual shifts in disability studies.

Disability studies emerged out of the disability civil rights movement in the late twentieth century. Early scholarship distinguishes the medical model of disability, which locates physical and mental impairments in individual bodies, from the social model, which understands the world as disabling people. The social model names both architectural and attitudinal barriers as the cause of disablement. Over the last few decades, the field has expanded to include individuals with a wide range of disabilities—not just physical conditions, but also mental and chronic ones.

This list, far from exhaustive, highlights some of the key debates and conceptual shifts in the field.  In addition to showcasing disability studies’ interdisciplinary focus, the list traces the relationship between D.S. and other minority fields of study. At its broadest, disability studies encourages scholars to value disability as a form of cultural difference. As the sources below reveal, ability should not be the default when it comes to human worth.

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Asch, Adrienne. “Recognizing Death while Affirming Life: Can End of Life Reform Uphold a Disabled Person’s Interest in Continued Life?” The Hastings Center Report , 2005

Bioethics scholar Adrienne Asch is one of the first scholars to bring a disability studies approach to bioethics. This essay addresses U.S. policy regarding decisions for end-of-life treatment and, more broadly, it critiques discourse surrounding “quality of life.” Asch attacks the slogan “better off dead than disabled” by showing how disability does not necessarily diminish one’s life. Instead, she argues, healthcare practitioners should focus on forms of care that give disabled people independence. She also offers pragmatic suggestions for how caretakers can affirm the humanity of patients receiving end-of-life treatment.

Baynton, Douglas. “Slaves, Immigrants, and Suffragists: The Uses of Disability in Citizenship Debates.” PMLA , 2005

Douglas Baynton’s groundbreaking essay foregrounds disability in accounts of American history. He assesses three U.S. debates regarding citizenship: the civil rights movement, women’s suffrage, and immigration legislation. This essay considers how disability has been used as a justification for the oppression of marginalized populations. For example, slaves were said to become “crazy” if they were granted freedom. Women were often described as mentally incapable of receiving an education. And immigrants have been cast as disabled due to racial difference. Baynton explores how attending to disability in its own right (rather than as a symptom of misogyny or racism) enables an intersectional analysis.

Brueggemann, Brenda Jo, Rosemarie Garland-Thomson, Georgina Kleege. “What Her Body Taught (Or, Teaching about and with a Disability): A Conversation.” Feminist Studies , 2005

Written from the perspective of three female scholars with disabilities, this essay is one of the first pieces of scholarship to address the presence of disabled faculty in the classroom. Brueggemann, Garland-Thomson, and Kleege explore the tension between wanting their disabilities to be normalized, but also wanting them to be present in students’ minds. In addition to discussing the difficulty of disclosure in the classroom, they explore how educators might adopt different approaches to teaching to accommodate instructors’ disabilities.

Davis, Lennard. “Crips Strike Back: The Rise of Disability Studies.” American Literary History , 1999

This essay reviews three publications that address disability studies from a humanities-based perspective. While D.S. first emerged in the social sciences, Davis makes a case for the centrality of disability studies scholarship in literary studies. More broadly, he proposes that disability studies should no longer be considered a narrow or specialized field. It’s applicable to us all.

Donaldson, Elizabeth J. “The Corpus of the Madwoman: Toward a Feminist Disability Studies Theory of Embodiment and Mental Illness.” NWSA Journal , 2002

This essay takes up the figure of the madwoman in literature. Adopting a feminist disability studies approach, Donaldson critiques the way feminist scholars read madwomen as merely symptomatic of patriarchal oppression, which discounts the reality of mental disability. She is resistant to framing disability as a metaphor and advocates for readings that consider disability alongside questions of gender.

Erevelles, Nirmala. “Race.” Keywords for Disability Studies (2015)

This short essay gives an overview of the relationship between disability studies and critical race studies. Erevelles shows how disability has been aligned with race. However, analogizing race to disability (or saying, “disability is like race”) eliminates the specificity of both identity categories. She uses special education as an example because it is a site where racial segregation takes place on the premise of disability. By attending to disabled people of color, she argues that we can achieve a more nuanced analysis, which accounts for how social forces like poverty and involuntary institutionalization exacerbate different forms of social marginalization.

Garland-Thomson, Rosemarie. “Feminist Disability Studies.” Signs , 2005

In placing feminist studies and disability studies in conversation, Garland-Thomson argues that both fields work to de-naturalize assumptions about embodiment and social roles. Her essay explores a range of pressing social issues, including selective abortion, caretaking, and reproductive rights.

Ginsburg, Faye and Rayna Rapp. “Disability Worlds.” Annual Review of Anthropology , 2013

Ginsburg and Rapp call for a critical approach to disability in the field of anthropology. Bridging the gap between the medical and anthropological fields, they shift toward understanding impairment as both environmental and cultural. They also consider what ethnography can bring to questions of disability within anthropological study.

Hershey, Laura. “Disabled Women Organize Worldwide.” o ff our backs , 2003

Recounting events from the NGO Forum on Women in China (1995) and the International Leadership Forum for Women with Disabilities in Maryland (1996), disability activist Laura Hershey moves beyond Western definitions of disability to offer a global perspective. In addition to showing how disabled women are doubly discriminated against, Hershey outlines how gender can influence the international movement for disability rights. She also considers how issues like poverty and illiteracy speak to the feminist and disability movements.

James, Jennifer C. and Cynthia Wu. “Editors’ Introduction: Race, Ethnicity, Disability, and Literature: Intersections and Interventions.” MELUS , 2006

This essay brings ethnic studies into conversation with debates regarding disability representation. From the nineteenth-century freakshow to forced sterilization, people of color have been disproportionately disabled, and James and Wu call for an intersectional approach to these complex subjectivities.

Kleege, Georgina. “Blind Rage: An Open Letter to Helen Keller.” Southwest Review , 1998

In this more personal essay, Kleege interrogates Helen Keller’s status as a disability icon. Kleege critiques the way disability has been individualized, refuting both tragic and triumphant representations of impairment. In describing her experience navigating everyday life as a blind woman, Kleege attends to the realities of living in a world not made for disabled people.

Kudlick, Catherine. “Disability History: Why We Need Another ‘Other.’” The American Historical Review , 2003

In this groundbreaking essay, Kudlick moves to situate disability studies in historical scholarship. Reframing disability as valuable, she argues that a renewed attention to bodily and mental impairments can revise our accounts of nearly all events in history—from women’s rights to labor movements. Her essay offers a comprehensive overview of books and articles pertaining to disability history, and, more specifically, deaf history.

Linker, Beth. “On the Borderland of Medical and Disability History: A Survey of Both Fields.” Bulletin of the History of Medicine, 2013

This essay addresses why the history of medicine and disability studies fail to interact. Linker begins by critiquing disability studies’ resistance to medical discourse. She argues that certain disabled people, especially those who are living with chronic conditions, often rely heavily on medical care, which is why the “medical model” should not be so readily dismissed. In turn, while disability history is typically understood as different from medical history, especially in the U.S., Linker argues for a greater need for cross-disciplinary collaboration.

Linton, Simi. “Reassigning Meaning.” Claiming Disability: Knowledge and Identity ,1988

One of the most foundational essays in the field of disability studies, Linton outlines how language has been important to naming disability as a political rather than medical category. She also addresses the problem of “overcoming rhetoric,” which fails to address disabled people’s need for access.

McRuer, Robert. “Crip Eye for the Normate Guy: Queer Theory and the Discipling of Disability Studies.” PMLA , 2005

McRuer is one of the first scholars to assess the relationship between disability and queerness. In this essay, he analyzes how the popular show The Queer Eye for the Straight Guy normalizes the disabled body. While representations of queer life often resist disability, he argues that queer and disability studies share a resistance to normalization, which should be embraced in future scholarship and activism.

Price, Margaret. Mad at School: Rhetorics of Mental Disability and Academic Life . University of Michigan, 2011

Price offers one of the first substantive accounts of mental disability, which has come belatedly to studies of physical disability. She focuses on higher education as a site that stigmatizes mental disability in its focus on rationality, cohesion, and cognitive agility. Her book offers a range of suggestions, many of which are pedagogical, for how mental disability might revitalize academic life.

Siebers, Tobin. “Disability in Theory: From Social Constructionism to the New Realism of the Body.” American Literary History , 2001

Siebers critiques the social model of disability, arguing that it fails to account for the experience of individual bodies. His term “the new realism of the body” calls for an assessment of the bodily effects of disability, which often cannot be altered through environmental transformations alone.

Wendell, Susan. “Unhealthy Disabled: Treating Chronic Illness as Disabilities.” Hypatia , 2001 

This article broadens the definition of disability to include individuals with chronic illnesses.  While people in the disability community often distinguish themselves from people who are ill, not all disabled people, she observes, are healthy. Wendell questions some of the main assumptions in disability activism and scholarship regarding social justice and reform. Dismantling the environmental effects of disablement will not always eliminate a body’s suffering, she argues.

Williamson, Bess. “Access.” Keywords for Disability Studies (2015)

This short essay gives a comprehensive account of the history of access and why it is a key term in D.S. Williamson argues that paying attention to access turns our focus away from the individual, highlighting instead the disabling makeup of the social world. Although access is easy to define, Williamson notes that it is hard to implement in practice because disabled people have conflicting needs.

Editor’s Note: This list has been updated to include publication dates.

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All differences by level of difficulty functioning and number of barriers had P  < .001 according to weighted χ 2 analyses. ADLs indicates activities of daily living.

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Biggs MA , Schroeder R , Casebolt MT, et al. Access to Reproductive Health Services Among People With Disabilities. JAMA Netw Open. 2023;6(11):e2344877. doi:10.1001/jamanetworkopen.2023.44877

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Access to Reproductive Health Services Among People With Disabilities

• 1 Advancing New Standards in Reproductive Health (ANSIRH), Bixby Center for Global Reproductive Health, Department of Obstetrics, Gynecology, and Reproductive Sciences, University of California San Francisco, San Francisco
• 2 Morrissey College of Arts and Sciences, Boston College, Chestnut Hill, Massachusetts
• 3 Independent scholars
• 4 Innovating Education in Reproductive Health, Bixby Center for Global Reproductive Health, Department of Obstetrics, Gynecology, and Reproductive Sciences, University of California San Francisco, San Francisco

Question   What is the prevalence of barriers to reproductive health (RH) access in the US among people with disabilities by disability type and status?

Findings   In this cross-sectional study of 6956 people assigned female at birth, people with disabilities across disability domains (hearing, vision, mobility, activities of daily living, and communication) and status experienced considerably more barriers in trying to access RH care in the past 3 years compared with people without disabilities.

Meaning   Findings of this study suggest a need to improve the transportation infrastructure and reinforce patient-centered approaches to engender inclusivity in health care settings.

Importance   People with disabilities face inequitable access to reproductive health (RH) services, yet the national prevalence of barriers to access experienced across disability types and statuses is unknown.

Objective   To assess the national prevalence of barriers to RH access experienced by people with disabilities.

Design, Setting, and Participants   This cross-sectional study analyzed results of an online probability-based national survey of RH experiences that was conducted from December 2021 to January 2022. The national sample consisted of English- and Spanish-speaking panel members of a market research firm. Panelists were invited to participate in a survey on their RH experiences and opinions. These participants were aged 15 to 49 years and assigned female at birth (AFAB). Weighted proportions were estimated, and bivariable and multivariable regression analyses were performed to assess associations between disability status and barriers to accessing RH services.

Exposure   Using 5 of the 6 Washington Group Short Set on Functioning items, 8 dichotomous disability indicators were created: (1) vision, (2) hearing, (3) mobility, (4) activities of daily living, (5) communication, (6) overall disability status (a lot or more difficulty functioning in ≥1 domain), (7) some difficulty functioning (below the disability threshold; some or more difficulty functioning in ≥1 domain), and (8) multiple disabilities (a lot or more difficulty functioning in ≥2 domains).

Main Outcomes and Measures   Number and types of barriers (logistical, access, cost, privacy, and interpersonal relationship) to accessing RH services in the past 3 years.

Results   After exclusion, the final sample included 6956 people AFAB, with a mean (SD) age of 36.0 (8.3) years. Of these participants, 8.5% (95% CI, 7.6%-9.5%) met the disability threshold. Participants with disabilities compared with those without disabilities were disproportionately more likely to be non-Hispanic Black (18.8% [95% CI, 14.4%-24.1%] vs 13.2% [95% CI, 11.9%-14.5%]) or Hispanic or Latinx (completed survey in English: 18.1% [95% CI, 14.0%-23.0%] vs 14.6% [95% CI, 13.3%-16.0%]; completed survey in Spanish: 8.9% [95% CI, 6.2%-12.8%] vs 6.2% [95% CI, 5.4%-7.1%]) individuals, to identify as LGBTQAI (lesbian, gay, bisexual, transgender, queer [or questioning], asexual [or allied], intersex; 16.4% [95% CI, 12.3%-21.6%] vs 11.8% [95% CI, 10.6%-13.1%]), to live below the federal poverty level (27.3% [95% CI, 22.3%-32.8%] vs 10.7% [95% CI, 9.7%-11.9%]), and to ever experienced medical mistreatment (49.6% [95% CI, 43.7%-55.5%] vs 36.5% [95% CI, 34.8%-38.2%]). Among those who had ever tried to access RH services (n = 6027), people with disabilities vs without disabilities were more likely to experience barriers (69.0% [95% CI, 62.9%-74.5%] vs 43.0% [95% CI, 41.2%-44.9%]), which were most often logistical (50.7%; 95% CI, 44.2%-57.2%) and access (49.9%; 95% CI, 43.4%-56.4%) barriers. The disability domains with the highest proportion of people who experienced 3 or more barriers in the past 3 years included activities of daily living (75.3%; 95% CI, 61.1%-85.6%), communication (65.1%; 95% CI, 49.5%-78.1%), and multiple (59.9%; 95% CI, 45.6%-72.7%) disabilities.

Conclusions and Relevance   This cross-sectional study found large disparities in access to RH services among people AFAB with disabilities. Findings indicated a need to alleviate barriers to RH care, including improving the transportation infrastructure and reinforcing patient-centered approaches that engender inclusivity in health care settings.

The Americans with Disability Act (ADA) defines disability as a “physical or mental impairment that substantially limits 1 or more major life activity.” 1 Levels of difficulty functioning can range in severity and span across multiple domains, each requiring unique accommodations to ensure adequate access and provision of high-quality health care, including sexual and reproductive health (RH) services. People with disabilities are disproportionately more likely than those without disabilities to have poor health, more health care needs, and gynecological cancers yet are less likely to receive gynecological cancer screenings and timely and consistent prenatal care, although they are similarly likely to get pregnant. 2 - 11 Although the ADA requires equal access to health care for people with disabilities, inequities persist. 12 Understanding the barriers that prevent people with disabilities from accessing RH services is critical to identifying inequities and informing patient-centered approaches to services.

Ableism, the practice of giving privilege to able-bodied people, adversely affects people’s access to and experiences with RH services as well as their reproductive well-being. 13 Examples of ableism in RH settings include lack of health care practitioner training on caring for people with disabilities, disability stigma, inaccessible health care facilities, nonexistent adaptive equipment and skilled language interpreters, and overall substandard quality of care. 13 - 22 Patients have described receiving poor-quality contraceptive care due to practitioners’ biased assumptions that people with disabilities are not sexually active and a lack of knowledge and insensitivity regarding management of contraceptive methods and their adverse effects alongside their disability. 20 - 22 Experiences of discrimination, exclusion, and stigma, paired with a health care infrastructure that does not adequately address the needs of people with disabilities, have likely contributed to substantial disparities in RH outcomes. People assigned female at birth (AFAB) with disabilities experience higher rates of intimate partner violence, reproductive coercion, unintended pregnancy, cervical and breast cancers, and poor birth outcomes than their counterparts. 2 , 20 , 23 - 26

Small qualitative studies have described the challenges that people with disabilities experience when trying to access RH services. These studies have found that people with disabilities often lack insurance for preferred contraceptive methods, lack privacy when discussing sensitive topics (eg, contraception) due to the presence of caregivers, and lack resources and information about RH screenings, which affect their adherence to screening protocols. 20 - 22 However, the extent to which people with disabilities experience insurance, privacy-related, and other barriers is unknown.

Limitations of studies that have examined the barriers experienced by people with disabilities include small sample sizes, no assessment of the barrier types experienced, a focus on a sample of pregnant people rather than preventive RH care, and minimal recent evidence. Given the increase in barriers to RH access since the COVID-19 pandemic, particularly among historically structurally marginalized populations, 27 an examination of the barriers experienced by people with disabilities is warranted. The current study aimed to assess the national prevalence of barriers to RH access experienced by people with disabilities, thereby filling an important research gap. We also examined the same barriers documented among the general public between 2017 and 2022. 27 Using these previous data allowed us to compare the prevalence of access barriers among people with disabilities vs the general public. For this study, the 2 primary research questions were (1) what is the prevalence of barriers in trying to access RH services among people with disabilities? and (2) do the barriers vary by disability status and disability type? By surveying people from December 2021 to January 2022 regarding barriers to RH access in the past 3 years, we captured their experiences during the COVID-19 pandemic. We hypothesized that people with disabilities, particularly those with multiple disabilities, experience more barriers to care.

This cross-sectional study analyzed results of an online, probability-based national survey, which was designed to assess the prevalence of attempts to self-manage an abortion, attitudes toward self-managed abortion, and interest in alternative models of medication abortion provision. The University of California San Francisco Institutional Review Board approved this study. Participants provided electronic informed consent before taking the survey. We followed the Strengthening the Reporting of Observational Studies in Epidemiology ( STROBE ) reporting guideline.

From December 2021 to January 2022, we fielded a large national survey to English- and Spanish-speaking, reproductive-aged (ages 15-49 years) people assigned female or male at birth; this analysis included only those AFAB. A market research firm administered the survey to their panel members (KnowledgePanel; Ipsos), using probability-based sampling techniques so that panelist recruitment was representative of the noninstitutionalized, English- and Spanish-speaking population living in the US when survey weights were applied. 28 Panelists were invited to participate in a survey on their RH experiences and opinions. The survey collected data on barriers to accessing RH services, health care experiences, abortion attitudes, and sociodemographic characteristics. Automatic reminders were sent to nonresponders 3 and 8 days after the initial survey invitation. Participants were reimbursed through a points program, whereby they received cash-equivalent checks in amounts reflecting their level of panel participation, which commonly amounted to $4 to $6 per month.

The primary outcomes were the number and types of barriers when trying to access RH services in the past 3 years, using previously published items. 27 , 29 Reproductive health services were defined as “a Pap smear, which is a test to check for cervical cancer, or family planning, like birth control methods.” Those who had ever tried to access RH services were asked to select the barriers they had experienced in the past 3 years from a predefined list of 10 barriers: finding transportation to an office or clinic, getting time off work or school to go to the appointment, finding childcare so I could go to the office or clinic, finding a place that offers RH services, finding a place where I felt comfortable, finding services with people who speak my language, paying for services, finding a place that accepts my insurance, getting services without telling people you did not want to tell, and going to the clinic because my partner or family member did not want me to go. We created a 4-part categorical variable for the number of barriers experienced (none, 1, 2, and ≥3) and grouped barriers into 5 conceptual themes (logistical, access, cost, privacy, and interpersonal relationship), consistent with a previous study. 27

We created 8 disability indicators as the primary exposures, using 5 of the 6 Washington Group Short Set (WG-SS) on Functioning items 30 : vision: do you have difficulty seeing?; hearing: do you have difficulty hearing, even when using hearing aids?; mobility: do you have difficulty walking or climbing stairs?; activities of daily living (ADLs): do you have difficulty with self-care, such as dressing or bathing?; and communication: using your usual language, do you have difficulty communicating, such as understanding or being understood? We excluded the cognition item, which asked how much difficulty do you have remembering or concentrating?, due to survey length constraints, overlap with the attention-deficit/hyperactivity disorder measure, and changes to the analytic plan. We changed the analytic plan to focus on WG-SS items, which were based on self-reported activity limitations, instead of including formal mental health diagnoses from a health care practitioner. For each item, the Likert-type answer options included no difficulty, some difficulty, a lot of difficulty, and cannot do at all. Following WG-SS criteria, participants who reported a lot of difficulty or cannot do at all in 1 or more domain were considered to have a disability (overall disability status) or a domain-specific disability (vision, hearing, mobility, ADLs, and communication). 31 We also lowered the cutoff to measure some difficulty functioning (defined as some or more difficulty functioning in ≥1 domain) and included a measure of multiple disabilities (defined as a lot or more difficulty functioning in ≥2 domains), following WG-SS criteria.

For all analyses, we used sampling weights to produce estimates that were representative of the noninstitutionalized US population AFAB, based on US Census data. Design weights accounted for any differential nonresponse. We estimated weighted proportions and conducted χ 2 analyses by overall disability status and participant characteristics ( Table 1 ), by attempts to access RH services and domain-specific disability ( Table 2 ), and by number ( Figure ) and types of barriers when trying to access RH services and disability indicators ( Table 3 ). We conducted unadjusted and adjusted log-binomial regression analyses to assess associations between disability indicators and types of barriers experienced when trying to access RH services ( Table 4 ). Covariates were selected a priori. We adjusted for age group (15-17, 18 and 19, 20-24, 25-29, 30-39, and 40-49 years), self-reported race and ethnicity and survey language (Hispanic or Latinx, who completed the survey in English; Hispanic or Latinx, who completed the survey in Spanish; non-Hispanic Asian, Native Hawaiian, or Pacific Islander [hereafter Asian]; non-Hispanic Black [hereafter Black]; non-Hispanic White [hereafter White]; and >1 non-Hispanic race or Other [American Indian or Alaska Native, Middle Eastern, North African, and people who selected Other], all of whom completed the survey in English), US nativity status, educational level, marital status (married; divorced, widowed, or separated; or never married), federal poverty level (<100%, 100%-199%, or ≥200%), metropolitan statistical area, geographic region of residence, LGBTQAI (lesbian, gay, bisexual, transgender, queer [or questioning], asexual [or allied], intersex and/or gender nonconforming) identity, medical mistreatment (when seeking health care, physicians, nurses, or other health care professionals made you feel your symptoms were not real, not severe, or not important, or made you feel ridiculed or humiliated), quality of medical care in the past year, and difficulty trusting health care physicians, nurses, or other health care professionals (ranging from not difficult to extremely difficult). Race and ethnicity were included in the analysis to ascertain whether structurally marginalized populations were disproportionately more likely to live with a disability and experience barriers when trying to access RH services.

Two-sided P  < .05 indicated statistical significance. We conducted all analyses in Stata 17 (StataCorp LLC). We used casewise deletion methods since the rate of missing outcome and covariate data was low at less than 1%.

A total of 44.6% of adults (6841 of 15 345) and 48.9% of adolescents aged 15 to 17 years (175 of 358) initiated the survey. We excluded 57 people with missing data on all disability or RH access barrier items, leaving a final analytical sample of 6956 people AFAB. Participants had a mean (SD) age of 36.0 (8.3) years and identified as being of Asian, Native Hawaiian, or Pacific Islander (5.7%; 95% CI, 4.9%-6.7%); Black (13.6%; 95% CI, 12.4%-15.0%); Hispanic or Latinx (completed survey in English: 14.9% [95% CI, 13.6%-16.2%]; completed survey in Spanish: 6.5% [95% CI, 5.7%-7.3%]); White (54.7%; 95% CI, 52.9%-56.4%); or other (4.6%; 95% CI, 3.9%-5.5%) race and ethnicity. A total of 46.4% (95% CI, 44.6%-48.1%) of participants had never been pregnant, and 8.5% (95% CI, 7.6%-9.5%) met the WG-SS threshold for disability, of whom 73.2% (95% CI, 67.1%-78.4%) had ever tried to access RH services, with no significant differences by overall disability status ( Table 1 ).

Compared with those without disabilities, participants with disabilities were disproportionately Black individuals (18.8% [95% CI, 14.4%-24.1%] vs 13.2% [95% CI, 11.9%-14.5%]), Latinx individuals (completed survey in English: 18.1% [95% CI, 14.0%-23.0%] vs 14.6% [95% CI, 13.3%-16.0%]; completed survey in Spanish: 8.9% [95% CI, 6.2%-12.8%] vs 6.2% [95% CI, 5.4%-7.1%]), with non–US nativity (19.1% [95% CI, 15.0%-24.0%] vs 14.3% [95% CI, 13.1%-15.6%]), living below the federal poverty level (27.3% [95% CI, 22.3%-32.8%] vs 10.7% [95% CI, 9.7%-11.9%]), members of the LGBTQAI community (16.4% [95% CI, 12.3%-21.6%] vs 11.8% [95% CI, 10.6%-13.1%]), to have ever considered self-managed abortion (4.5% [95% CI, 2.7%-7.4%] vs 2.6% [95% 2.1%-3.3%]), and to have ever experienced medical mistreatment (49.6% [95% CI, 43.7%-55.5%] vs 36.5% [95% CI, 34.8%-38.2%]) ( Table 1 ). Only 2 (communication and multiple disabilities) of the 8 disability indicators differed significantly in ever trying to access RH services ( Table 2 ). Among those who had ever tried to access RH services (n = 6027), across all 8 disability indicators, participants with disabilities experienced more barriers when trying to access RH services than those without disabilities ( Figure ). People with disabilities vs without disabilities were more likely to experience barriers (69.0% [95% CI, 62.9%-74.5%] vs 43.0% [95% CI, 41.2%-44.9%]), most often logistical (50.7%; 95% CI, 44.2%-57.2%) and access (49.9%; 95% CI, 43.4%-56.4%) barriers. The highest proportions of participants who experienced 3 or more barriers in the past 3 years included those with ADL (75.3%; 95% CI, 61.1%-85.6%), communication (65.1%; 95% CI, 49.5%-78.1%), and multiple (59.9%; 95% CI, 45.6%-72.7%) disabilities and those who met the overall disability status threshold (42.5%; 95% CI, 36.1%-49.1%).

Participants with overall disability status (eg, logistical barriers: 50.7% [95% CI, 44.2%-57.2%] vs 29.7% [95% CI, 28.0%-31.4%]), some difficulty functioning (eg, logistical barriers: 39.1% [95% CI, 36.2%-41.9%] vs 26.7% [95% CI, 24.7%-28.8%]), and multiple disabilities (eg, logistical barriers: 66.2% [95% CI, 53.1%-77.2%] vs 30.7% [95% CI, 29.0%-32.4%]) all experienced significantly more of each type of barrier in trying to access RH services in the past 3 years compared with their counterparts ( Table 3 ). In adjusted and unadjusted regression analyses, participants with overall disability status (eg, logistical barriers: risk ratio [RR], 1.32; 95% CI, 1.14-1.52) and those with multiple disabilities (eg, logistical barriers: RR, 1.61; 95% CI, 1.33-1.93) were significantly more likely to have experienced all types of barriers in trying to access RH services ( Table 4 ). Participants with some difficulty functioning (eg, logistical barriers: RR, 1.19; 95% CI, 1.07-1.32) were significantly more likely to experience each barrier, except for access barriers, which were not statistically significant in adjusted analyses.

In this large national representative survey conducted around the time of the COVID-19 pandemic, 8.5% of participants AFAB of reproductive age met the disability status threshold, of whom over two-thirds experienced barriers to RH access in the past 3 years. 30 While attempts to access RH services were largely similar by disability status, people with disabilities, particularly those with multiple disabilities, experienced more barriers in trying to access care. Participants with ADL or communication disabilities experienced the greatest number of barriers, with as many as three-quarters experiencing 3 or more barriers in the past 3 years, suggesting that these groups may require the most support. The preponderance of access barriers experienced was consistent with that in other studies that found disparities in general health care access for people with disabilities. 32 - 34 Thus, disparities in gynecological cancer screenings and contraceptive use may be due to structural barriers to care. Future research needs to examine access to other RH services, such as screening for sexually transmitted infections (STIs) and abortion care, which were not examined in this study, although we did find that people with disabilities are more likely to consider SMA than people without disabilities.

Previous work found that these same barriers to RH access increased from before (2017) to during the COVID-19 pandemic, particularly among people living in poverty and with less formal education. 27 Thus, the structural barriers to RH care observed in this study may have been compounded during the pandemic, disproportionately affecting people with disabilities. Many factors may explain the barriers experienced during this time, including health risks from COVID-19, lost wages, in-person care restrictions, reduced number of Title X family planning clinics, and other RH-related policy changes. 35 By assessing the attempts to access RH care, we captured the experiences of people who needed but may have been unable to receive care.

Similar to other work, 36 , 37 we found variation in demographic characteristics by disability status. Participants with disabilities were significantly more likely to be Black or Latinx individuals, live below the federal poverty level, and identify as LGBTQAI, and they were much more likely to report experiencing medical mistreatment (eg, being ridiculed, humiliated, or ignored by health care practitioners), receiving fair or poor-quality medical care from their regular physician, and to consider self-managed abortion. These participants may be further marginalized due to ableism, discrimination, limited resources, and intersectional racism and homophobia and thus may encounter additional barriers to care and may experience poor quality and potentially discriminatory health care.

We used multiple categorizations of disability to examine variability in the magnitude and types of barriers experienced by people with a range of disability statuses. We found significant inequities among participants across disability indicators, including those above and below the disability threshold, and all of these participants experienced more of any barrier to RH care compared with those without disabilities. This finding is especially important, as people with some difficulty functioning are often not categorized as having a disability in RH studies. 38 Efforts to increase the accessibility of RH care must move beyond complying with the ADA standards toward accommodating varying experiences of people with different conditions and levels of functioning.

Participants with disabilities most commonly experienced logistical (ie, finding transportation, getting time off work or school, and finding childcare) and access barriers (eg, finding a place that offers RH services; finding a place where I felt comfortable). A transportation infrastructure that does not meet the needs of people with disabilities likely contributes to inequitable access to care. 39 Telehealth models of care may help to streamline access and better serve the needs of some people. 40 , 41 The high proportion of people who reported difficulty finding a place where they felt comfortable may be associated with previous medical mistreatment and poor-quality health care, and may explain the higher proportion of people with disabilities vs those without disabilities who considered self-managed abortion. Additionally, participants with disabilities also reported difficulty with going to the clinic because their partner or family member did not want them to go. This finding may be explained in part by the greater interdependence between people with disabilities and others for transportation and support with accessing care as well as by the likelihood of people with disabilities to experience intimate partner violence, reproductive coercion, and abuse. 20 , 26 , 42 , 43 Special attention is needed to address privacy concerns and to identify potential coercion and abuse that might prevent people with disabilities from accessing care.

Findings should be interpreted according to study limitations. First, the measures of access and barriers to care were not comprehensive of all RH services, such as STI screening and abortion care, and did not include all access-related or disability-specific barriers. For example, we did not include barriers related to accessible equipment (eg, adjustable beds), methods of providing information (eg, compatibility of written materials with screen readers), or availability of sign language interpreters. Future research should examine these disability-specific barriers and the implications of abortion bans. Second, we used items from the WG-SS to measure disability, which is known to undercount individuals with chronic or psychiatric conditions, 44 and we included only 5 of the 6 WG-SS items, limiting the generalizability of the composite measures of disability status and multiple disabilities to other studies. Thus, the disability status prevalence rate of 8.5% was expectedly lower and not directly comparable with other studies using the WG-SS items, which reported an 11% prevalence rate among adult male and female populations, although the domain-specific disability indicators in this study are comparable with those in other studies. 31 , 45 Third, given the data collection methods, the study population excluded institutionalized people, which likely left out people who were institutionalized due to their disability. Thus, the estimates of rates of disability and barriers to RH services are not generalizable to institutionalized individuals. Finally, given that we lacked sociodemographic data on those who did not respond to the survey, we were unable to determine the extent to which the sample was biased due to nonresponse. However, the representativeness of the sample was assessed in a previous work, which compared the sociodemographic profile of the current weighted sample to that of the National Survey of Family Growth and found that distributions by age and race and ethnicity were largely similar. 27 Furthermore, the use of sampling weights reduced some of the bias introduced from unequal selection and nonresponse.

The findings of this cross-sectional study pointed to large disparities in access to RH care among people AFAB living with disabilities, most of whom experienced multiple barriers to RH care across disability types. While this study did not specifically examine barriers to abortion care, given that people with disabilities were more likely to consider self-managed abortion, restrictions on RH services, including abortion care, are likely to disproportionately affect people with disabilities, further widening health care disparities. The findings highlighted the need to alleviate barriers to RH care, including improving the transportation infrastructure, ensuring the availability of foreign language and sign language interpreters, training practitioners to better serve their patients with disabilities, and reinforcing patient-centered approaches that engender inclusivity in health care settings. There is a continued need for more research to elucidate the unmet RH needs and experiences among people with disabilities.

Accepted for Publication: October 14, 2023.

Published: November 29, 2023. doi:10.1001/jamanetworkopen.2023.44877

Open Access: This is an open access article distributed under the terms of the CC-BY License . © 2023 Biggs MA et al. JAMA Network Open .

Corresponding Author: M. Antonia Biggs, PhD, Advancing New Standards in Reproductive Health, Bixby Center for Global Reproductive Health, Department of Obstetrics, Gynecology and Reproductive Sciences, University of California San Francisco, 1330 Broadway, Ste 1100, Oakland, CA 94612 ( [email protected] ).

Author Contributions: Dr Biggs had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.

Concept and design: Biggs, Wilson-Beattie, Ralph, Kaller.

Acquisition, analysis, or interpretation of data: All authors.

Drafting of the manuscript: Biggs, Schroeder, Gichane.

Critical review of the manuscript for important intellectual content: All authors.

Statistical analysis: Biggs, Ralph, Adler.

Obtained funding: Ralph, Gichane.

Administrative, technical, or material support: Biggs, Schroeder, Casebolt, Laureano, Wilson-Beattie, Ralph, Kaller.

Supervision: Biggs, Ralph.

Conflict of Interest Disclosures: Dr Laureano reported receiving personal fees from University of California San Francisco (UCSF) during the conduct of the study. Dr Ralph reported receiving grants from Private Foundation during the conduct of the study. Dr Gichane reported receiving grants from UCSF during the conduct of the study. No other disclosures were reported.

Funding/Support: This research was supported by the Resource Allocation Program Award 2017924 from the UCSF National Center of Excellence in Women’s Health (Dr Gichane) and a core grant from Advancing New Standards in Reproductive Health (Drs Biggs and Ralph).

Role of the Funder/Sponsor: The funders had no role in the design and conduct of the study; collection, management, analysis, and interpretation of the data; preparation, review, or approval of the manuscript; and decision to submit the manuscript for publication.

Data Sharing Statement: See the Supplement .

Additional Contributions: Aura Orozco-Fuentes, MSc, and Molly Battistelli, MPH, UCSF, provided support for the successful implementation of the study. Daniel Grossman, MD, UCSF, provided overall project mentorship. These individuals received no additional compensation, outside of their usual salary, for their contributions.

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Relationships Between Affiliate Stigma, Special Education Plans, and Family-school Partnerships Among Latino Parents of Children with Disabilities

• ORIGINAL ARTICLE
• Published: 18 April 2024

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• Alysse Loomis   ORCID: orcid.org/0000-0001-9909-8092 1 ,
• Cristina Mogro-Wilson 2 ,
• Devon Musson Rose 1 &
• Emily Longo 2  

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Legislative mandates dictate that students with disabilities are provided instruction, services, and supports needed to progress in school (Individuals with Disabilities Education Act [IDEA] Part B, 2004 ) which can be in the form of Individualized Education Plans (IEPs), Individualized Family Service Plans (IFSP) for children three and under, and 504 plans. A number of studies have highlighted the ways in which these formal educational processes may increase stress among parents (Rios et al., Research in Autism Spectrum Disorders , 73 (March), 101534, 2020 ). However, formal educational processes may also be protective in the context of parents’ experiences of affiliate stigma and their perceptions of family-school partnerships, which have not been widely examined, particularly among Latino parents of children with disabilities. The current study examined the relationship between affiliate stigma, or the parent’s internalized stigma related to the child’s disability, and family-school partnerships in a sample of 141 Latino parents. In the current study we also examined whether the presence of a formal education plan (e.g., IEP, IFSP, 504 plan) moderated the relationship between affiliate stigma and family-school partnerships. We found that higher parent engagement but not affiliate stigma was related to significantly stronger family-school partnerships. In the sample 60% of parents reported that their child had an education plan, and presence of such a plan moderated the relationship between affiliate stigma and family-school partnerships. Recommendations are made for future research and practice related to supporting Latino parents within the special education system.

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Complex and alternate consent pathways in clinical trials: methodological and ethical challenges encountered by underserved groups and a call to action

• Amy M. Russell 1   na1 ,
• Victoria Shepherd   ORCID: orcid.org/0000-0002-7687-0817 2   na1 ,
• Kerry Woolfall 3 ,
• Bridget Young 3 ,
• Katie Gillies 4 ,
• Anna Volkmer 5 ,
• Mark Jayes 6 ,
• Richard Huxtable 7 ,
• Alexander Perkins 8 ,
• Nurulamin M. Noor 9 ,
• Beverley Nickolls 10 &
• Julia Wade 11  

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Informed consent is considered a fundamental requirement for participation in trials, yet obtaining consent is challenging in a number of populations and settings. This may be due to participants having communication or other disabilities, their capacity to consent fluctuates or they lack capacity, or in emergency situations where their medical condition or the urgent nature of the treatment precludes seeking consent from either the participant or a representative. These challenges, and the subsequent complexity of designing and conducting trials where alternative consent pathways are required, contribute to these populations being underserved in research. Recognising and addressing these challenges is essential to support trials involving these populations and ensure that they have an equitable opportunity to participate in, and benefit from, research. Given the complex nature of these challenges, which are encountered by both adults and children, a cross-disciplinary approach is required.

A UK-wide collaboration, a sub-group of the Trial Conduct Working Group in the MRC-NIHR Trial Methodology Research Partnership, was formed to collectively address these challenges. Members are drawn from disciplines including bioethics, qualitative research, trials methodology, healthcare professions, and social sciences. This commentary draws on our collective expertise to identify key populations where particular methodological and ethical challenges around consent are encountered, articulate the specific issues arising in each population, summarise ongoing and completed research, and identify targets for future research. Key populations include people with communication or other disabilities, people whose capacity to consent fluctuates, adults who lack the capacity to consent, and adults and children in emergency and urgent care settings. Work is ongoing by the sub-group to create a database of resources, to update NIHR guidance, and to develop proposals to address identified research gaps.

Collaboration across disciplines, sectors, organisations, and countries is essential if the ethical and methodological challenges surrounding trials involving complex and alternate consent pathways are to be addressed. Explicating these challenges, sharing resources, and identifying gaps for future research is an essential first step. We hope that doing so will serve as a call to action for others seeking ways to address the current consent-based exclusion of underserved populations from trials.

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Informed consent is seen as a cornerstone in the ethical conduct of clinical trials. However, in populations or settings where there are challenges to seeking or providing consent, alternative consent arrangements may be required. These challenges may arise due to communication barriers, where a participant’s capacity to provide consent fluctuates over time, where capacity is lost during a trial, or they are deemed to lack the capacity to consent at the outset. These challenges may be particularly pronounced in emergency settings where the urgent nature of the condition and the need for immediate action preclude the ability to seek prior consent for either adults or children. Populations where consent may pose a challenge have historically been excluded from trials and are recognised as being underserved by research as a result [ 1 ]. For example, one in three patients with hip fractures have a concomitant cognitive impairment, yet eight out of ten hip fracture trials exclude this population despite evidence that those with cognitive impairment are likely to experience different outcomes [ 2 ]. Even trials in conditions associated with cognitive impairment frequently exclude people with impaired capacity to consent [ 3 ]. This exclusion of relevant subgroups of patients risks presenting biased estimates of treatment effects [ 4 , 5 ] and limits the ability to provide evidence-based care for these groups.

For many of these populations, research inequity contributes to the health disparities that they already encounter [ 6 ]. For example, adults with intellectual disabilities die on average 10–15 years earlier than those without intellectual disabilities in the UK and the USA [ 7 , 8 ], yet 90% of clinical trials are designed in a way that automatically excludes them from participating [ 9 ]. The importance of widening opportunities for the participation of underserved populations in research has received recognition both in the UK and beyond, resulting in national and international initiatives to improve inclusivity and diversity in the design, conduct, and reporting of clinical trials [ 1 , 10 , 11 , 12 ]. Research funders increasingly require researchers to address issues around inclusivity and representativeness in their funding applications [ 13 ]. However, the challenges of conducting trials where consent is complex, and where consent-based exclusion denies populations the opportunity to participate in and benefit from research, have received less attention [ 14 ].

The ethical and methodological issues surrounding trials involving complex and alternative consent pathways have led to the formation of a new UK multi-institutional collaboration to collectively address some of these challenges. This collaboration forms a sub-group of the Trial Conduct Working Group in the MRC-NIHR Trial Methodology Research Partnership, consisting of members from disciplines including trials methodology, qualitative research, healthcare, bioethics, and social sciences. This paper summarises and discusses contexts where researchers may encounter particular methodological and ethical challenges around consent. The focus is on trials where the process of consent is challenging and alternative consent pathways are required, rather than where the informational content required for consent to be valid is complex [ 15 ], or where the trial design is complex such as a multistage randomised controlled trial [ 16 ].

Drawing on our experiences as an interdisciplinary group of researchers with an interest in complex and alternate consent pathways in trials, we will focus on key populations where consent-based challenges contribute to their exclusion: adults with communication or other disabilities [ 17 ], adults who lack the capacity to consent [ 18 ], adults whose capacity to consent fluctuates or is lost during a trial [ 19 ], and adults and children requiring emergency and urgent care [ 20 ]. The question of alternative consent pathways for children in non-emergency research will not be addressed in this article as it requires specific attention [ 21 ]. For each population, we articulate the challenges around inclusion in trials, summarise current evidence and ongoing work, and identify areas for future research. We hope that this will serve as a cri de cœur for others seeking ways to address the consent-based exclusion of underserved populations from trials.

Trials involving adults with communication, hearing, and sight disabilities

Despite the fact that the majority of legislation delineating consent processes urges professionals to make adjustments for people with communication, hearing, and visual impairments, they may be excluded from research simply due to the fact that obtaining informed consent is more challenging [ 22 ]. Communication disabilities can comprise a range of difficulties that impact a person’s ability to understand spoken or written information (sounds, words, or sentences) and express themselves verbally or non-verbally (articulate sounds/letters, select words, or use relevant grammar and sentence forms) in spoken, written, or picture form. Difficulties in accessing and comprehension of information are one of the most common barriers in consent scenarios across several diagnoses including dementia [ 23 ], stroke [ 24 ], and brain injury [ 25 ], as well as developmental disorders such as autism and learning/intellectual disabilities [ 26 ]. Other difficulties that can impede a person’s ability to access spoken or written information include hearing or visual impairments, which may or may not be associated with an underlying condition. The use of British Sign Language interpreters or translation of written materials to other languages including Braille is extremely important for those with hearing or visual impairment [ 27 ]. Beyond this, the heterogeneity amongst people with communication disabilities requires adaptations to be tailored to individual needs based on knowledge of the person’s communication strengths and difficulties. People with stroke-related language impairments (aphasia), for example, may benefit from the information being presented using active language, shorter sentences, or written keywords [ 28 ].

The challenges

Making changes to support communication needs is complex. Some researchers find current guidance such as the Mental Capacity Act Code of Practice [ 27 ] and Health Research Authority guidance [ 29 ] difficult to interpret and implement [ 30 , 31 ]. Researchers acknowledge a lack of skills, knowledge, and confidence in being able to adapt their language and communication to meet the needs of people with communication disabilities [ 31 ]. Other barriers identified include the lack of specific training, tools, time and access to ethically approved materials [ 31 , 32 , 33 ].

There is limited evidence relating to the inclusion of people with communication disabilities in the informed consent process. This is in part because people with communication disabilities often have been excluded from study recruitment processes [ 17 , 30 , 31 , 33 , 34 , 35 ], and because studies that have included them have tended not to report the recruitment and consent methods used [ 32 ].

Current research and guidance

People with communication disabilities may not be included in the informed consent process for different reasons: this group is frequently defined as ineligible for inclusion in studies per se, solely due to their communication disabilities [ 31 ]; even where included, researchers may consult proxies (e.g. family members) because they assume that people with communication disabilities lack the mental capacity to provide informed consent [ 17 , 31 , 33 ]; researchers may find the consent process for this group too challenging and time-consuming [ 31 ]. Reluctance to include people with communication disabilities in the consent process may follow challenges involving people with significant communication disabilities in patient and public involvement and engagement activity, and current involvement guidance does not provide specific information about how to include this group [ 36 ]. Recent UK studies have helped to contextualise these findings, by examining the legal, policy, and governance frameworks that apply to the recruitment of people with communication disabilities [ 30 , 37 , 38 ]. Whilst not specific to trials, these frameworks provide guidance for facilitating the inclusion of this group in the informed consent process. This includes recommendations to co-produce information materials with people with communication disabilities and to adapt communication environments and processes to improve their accessibility. These recommendations are supported by research that has developed and tested communication methods to support decision-making during the informed consent process for people with post-stroke aphasia [ 22 , 32 , 39 ] and intellectual disability [ 33 , 40 ].

In recent examples, researchers have been able to create and use accessible consent materials and implement these within stroke trials [ 41 , 42 , 43 ] using practical, evidence-based resources [ 22 , 44 , 45 ]. These have been co-produced to ensure the language is accessible, readable, and accompanied by transparent visual representations and alternative mediums (video for example). Furthermore, the recent ASSENT [ 46 ] and CONSULT [ 47 ] projects have developed inclusive consent guidance and resources to aid researchers.

Future research

More research is required to explore the inclusion of people with communication disabilities in the informed consent process in trials, in terms of current practice and professional and participant experience. Most existing research appears to have focused on two main groups: people with post-stroke aphasia and people with intellectual disabilities. Future research should explore the experiences and needs of people with different types of communication disabilities, for example, people living with dementia or with other progressive neurological conditions.

Further research is required to develop and evaluate additional tools, resources, and training interventions to support researchers to work with people with communication disabilities more easily and effectively during the informed consent process [ 37 ]. Evaluation should include the exploration of usability, acceptability to professionals and participants, and cost-effectiveness. In addition, studies should explore how researchers can form successful and equitable collaborations with people with communication disabilities as part of trial public involvement and engagement activity in order to co-produce inclusive consent processes and materials [ 48 ].

Trials involving adults whose capacity fluctuates or is lost during a trial

Informed consent can only be obtained from individuals who have the capacity to give consent. Fluctuating capacity can refer to situations where a person’s condition is cyclical (moving from an acute phase to a recovery phase) [ 49 ] or where their capacity is influenced by other factors including but not limited to health or environment [ 50 , 51 ]. It can also relate to capacity that is task-specific, where an individual may have the capacity to consent to certain aspects of a trial but may struggle to give informed consent to all aspects or understand long-term follow-up processes.

Fluctuating capacity raises three main challenges: (1) the potential exclusion of those believed to have fluctuating capacity where no clear assessment process is in place, (2) the need for a process of consent-taking at each data collection time point, and (3) the need to incorporate planning for a loss of capacity, temporary or otherwise, when creating trial processes, patient information and consent materials. Without forward planning, unanticipated lost capacity during data collection may lead to withdrawal and/or missing data and the unnecessary exclusion of participants [ 52 ].

Capacity is often framed (and commonly understood and implemented by recruiting staff) in binary terms as something a person has or does not have [ 53 , 54 ], which has been critiqued in certain populations and cultural contexts [ 55 ]. In England and Wales, the Mental Capacity Act 2005 makes it clear that capacity is task-specific. Once assessed, capacity is not an end point but an ongoing process of engagement with a participant.

An intention to carry out capacity assessments is often alluded to in trial protocols without further detail being given on why certain individuals will be assessed, who will conduct assessments, and what criteria they will use [ 9 ]. The Mental Capacity Act 2005 and the Code of Practice (2007) exist to protect individuals, but not to impede their right to participate in research, something researchers should acknowledge. However, there is a lack of practical guidance in these documents which results in uncertainty about how researchers should best assess capacity. This can lead to inconsistent approaches to assessment. Capacity should be assumed in individuals, and capacity assessments should also only take place after the individual has been given clear information, appropriate to their needs, and there is a question raised about their ability to provide informed consent. This again raises challenges for trials where standard information is required that can be complex, lengthy, and difficult to adapt to the needs of different groups (for example, people with communication disabilities) [ 56 , 57 ].

Suggestions for alternative forms of consent that may support those whose capacity fluctuates have been developed by researchers working with specific populations [ 58 ], including process consent in dementia research [ 59 ]. These distinguish between time and task-specific capacity and the capacity to take a longitudinal view, implying an understanding of future risks and benefits [ 49 ]. However, research to date often focuses on distinct populations, e.g. people receiving palliative care [ 60 ], people living with dementia, and stroke survivors. Attention to managing fluctuations in capacity is less often seen in population-wide trials. To reduce blanket exclusions for certain populations, and misuse of lack of capacity being used as an exclusion criterion, further research resulting in clear guidance is required.

Standardised tools for capacity assessment have been developed, but there is no gold standard for the assessment of capacity in clinic or in research, nor is there an agreement that any one tool can sufficiently capture the complexity of capacity assessment [ 61 ]. Current Mental Capacity Act-compliant tools remain difficult to adapt to the heterogeneity of the populations for whom capacity fluctuates [ 62 , 63 , 64 ]. Capacity assessment processes are also often only employed in certain trials which anticipate that their target population will require them.

Consent needs to be understood as task- and time-specific and requiring accessible information. Research is needed to generate guidance on what to do if capacity is lost during follow up and it must be based on a defined process of establishing the wishes of participants at the initial consent stages. More evidence is required on the best methods for capacity assessment and how to support researchers to assess capacity. Trials need to build protocols for how to prevent exclusion of those who may fluctuate in capacity to consent and on how to manage data collection from those whose capacity does fluctuate.

Trials involving adults who lack the capacity to consent

Even with support, some people will be unable to provide their own consent to take part in a trial. The exclusion of adults who lack the capacity to consent has been widely documented [ 18 , 65 , 66 ] and is due to a range of intersecting methodological and systemic barriers to their inclusion [ 34 ]. Specific consent-based challenges include the complexity of the patchwork of legal frameworks that govern trials involving adults lacking capacity both within the UK [ 67 ] and internationally [ 68 ], and the uncertainties of applying them in practice [ 69 ]. In the UK, clinical trials of an investigational medicinal product involving adults lacking capacity are governed by the Medicines for Human Use (Clinical Trials) Regulations [ 70 ], with other types of trials covered by mental capacity legislation such as the Mental Capacity Act in England and Wales [ 71 ]. In both cases, there are provisions for an alternative decision-maker to be involved in enrolment decisions, usually a family member or close friend, or someone acting in a professional capacity who is not involved in the research if no one is able or willing to act in a personal capacity [ 70 , 71 ]. For clinical trials, the alternative decision-maker is termed a legal representative and provides consent based on the person’s presumed will [ 70 ], and for other types of research, they act as a consultee and are asked to provide advice about participation based on the person’s wishes and preferences [ 29 ]. However, little guidance is available to families and health and social care professionals about their role in making decisions about trial participation, nor the legal basis for their decision [ 72 ].

Due in part to this legal complexity, a lack of knowledge about research involving adults who lack capacity, and paternalistic attitudes generally, may result in gatekeeping practices by researchers and health and social care professionals towards this population [ 73 , 74 ]. Involving health and social care professionals as a consultee or legal representative relies on them having the time and willingness to be involved. Some may be concerned about being unable to determine or represent the wishes and preferences that a person may hold and so may decline to become involved [ 38 ]. Other challenges arise due to the difficulties in identifying and contacting consultees and legal representatives [ 75 ]. Even when they have been identified, family members are less likely to agree to research participation on the person’s behalf than patients themselves [ 76 ]. This may be due to families’ difficulties in knowing what that person’s wishes and preferences would be about participation [ 77 ]. People rarely discuss their research preferences in the event that they might lose capacity, and there is no current mechanism in the UK for prospectively appointing a consultee or legal representative to make decisions about research [ 78 ].

Procedures for identifying and approaching consultees and legal representatives are one of the issues that research ethics committees (RECs) consider when reviewing applications for trials involving adults who lack capacity, alongside arrangements for assessing capacity to consent where required [ 29 ]. However, RECs’ resistance to the inclusion of adults who lack capacity in a trial, and whether there is sufficient justification to do so, is cited as one of the greatest barriers to their inclusion [ 79 , 80 ]. RECs do not interpret the legal frameworks consistently or, at times, correctly, with inaccurate terminology and requirements being cited [ 37 , 72 , 81 ]. There have been calls for greater explicitness and accuracy when applications for ethical review of these studies are both submitted and reviewed [ 72 , 81 ] and for incorporating more adaptations and accommodations into the recruitment process such as ensuring information is cognitively accessible [ 37 ].

Recent research has identified a number of barriers and facilitators to involving adults lacking consent in trials [ 19 , 34 ] leading to the creation of guidance, for example, for recruiting adults with impaired mental capacity at the end of life in research [ 19 ]. Recent initiatives to address the inclusion of underserved groups in research more broadly, such as the NIHR INCLUDE project [ 1 ], have led to the development of the INCLUDE Impaired Capacity to Consent Framework which is a tool to help researchers to design and conduct trials that are more inclusive of people with impaired capacity to consent [ 82 ].

Other studies have focused on the role of personal consultees and legal representatives. This includes a study that found that making ethically complex decisions about research on behalf of someone else can be challenging for many family members, with some experiencing a decisional and emotional burden as a result [ 83 ]. Current work includes the development of the first decision aid for families making decisions about research on behalf of someone who lacks the capacity to consent [ 84 ] which is currently being evaluated as a ‘Study Within a Trial’ (or ‘SWAT’) (CONSULT) [ 85 ] and the development of resources to help researchers [ 47 ].

Despite the ongoing work, there is a need for a more sustained effort to ensure that these groups have an equitable opportunity to participate in trials. More research is needed into how researchers can design more inclusive trials, and the involvement of health and social care professionals as nominated consultees, and the use of professional legal representatives when necessary. Unlike questions about why other underserved groups have been excluded from research, the legal position regarding people who lack capacity is that their inclusion requires justification [ 29 ]. Clearer guidance is required on how this justification is understood and interpreted.

A number of recommendations for further research at a policy and legislation level have been previously made, including proposals by the Nuffield Council on Bioethics [ 86 ] that consideration be given to extend the role of the welfare attorney in England and Wales to include decisions about research, both within the Mental Capacity Act [ 71 ] and the Clinical Trials Regulations [ 70 ]. There is also uncertainty about the role of Lasting Power of Attorney in decisions about research participation [ 78 ], with families wanting greater support and guidance when making decisions [ 83 ].

Adult and paediatric emergency and urgent care trials

Trials involving adults in emergency situations may encounter additional complexities. The challenges of obtaining consent from patients who are suddenly unable to communicate or convey their own wishes are encountered in trial contexts ranging from intrapartum [ 87 ] and acute coronary syndrome [ 88 ] to acute stroke where it has been described as the rate-limiting step in treatment RCTs [ 89 ]. Emergency and urgent care trials are conducted in a range of settings including prehospital [ 90 ] and critical care [ 91 ].

Historically, children have not received evidence-based healthcare in emergency and critical care settings due to their exclusion from trials arising from similar practical and ethical issues to those encountered in adult trials in these time-critical settings [ 92 ]. In order to increase the chances of saving a child’s life, treatments need to be given without delay, so there is no time to seek informed consent from parents or legal representatives. Even if there is a brief window of opportunity for recruitment discussions, parents may not be present or may be highly distressed and lack the capacity to make an informed decision about the use of their child’s information and potential ongoing involvement [ 93 ].

Emergency research is when treatment needs to be given urgently [ 94 ] and recruitment cannot be delayed until the patient either regains capacity or a consultee or legal representative can be found [ 95 ]. In such circumstances, research without prior consent (RWPC, also referred to as ‘deferred consent’) is permissible in many jurisdictions including the USA, Canada, parts of Australasia, and the UK through both the Mental Capacity Act [ 71 ] and the 2006 Amendment to the 2004 EU Clinical Trials Regulations [ 96 ]. However, there are variations in the provisions for RWPC in emergency research, both between and within countries [ 97 ]. Within the UK, for example, the law in Scotland does not provide any ‘exemptions’ or alternatives for the involvement of adults not able to consent for themselves in clinical trials in emergency situations [ 94 ]. This meant that trials such as RECOVERY-RS [ 98 ], which compared respiratory strategies for patients with COVID-19 respiratory failure, could not recruit Scottish patients. Similarly, the UK-REBOA trial in life-threatening torso haemorrhage was unable to recruit in Scotland despite being coordinated from there [ 99 ].

In recognition of the need to conduct these vital trials with children, various legal frameworks for paediatric trials have also been amended nationally and internationally, enabling research to be conducted without prior consent. In 2008, UK legislation was amended to allow research without prior consent in such circumstances [ 100 ], yet there was a lack of knowledge about how and when research teams should broach these research discussions with parents in a way that avoided further burdening families. There was also a need for guidance to inform what should happen when a child dies after trial enrolment without parents’ prior knowledge or consent. Despite the 2008 legislation that enabled much-needed research on emergency treatments for children, there was hesitancy amongst clinical and research communities about conducting trials involving critically ill children [ 101 ].

The use of RWPC in both adult and child populations is ethically complex, with diverse views about the acceptability of enrolling acutely ill patients without consent [ 102 ]. There are particular challenges around gaining ethical approval for the use of RWPC in borderline or ‘middle ground’ cases where a patient may be conscious or coherent, yet their condition or the lack of time limits the possibility of informed consent [ 103 ]. These, and other challenges [ 34 ], can lead to consent-based recruitment bias which means that patients enrolled in RCTs may not necessarily be representative of critically ill patients in clinical practice [ 20 , 104 ]. This has the potential to cause harm by obscuring any treatment effect [ 105 ].

A recent study in the UK (Perspectives Study) explored consent and recruitment in adult critical care research [ 106 ] and identified strategies to enhance consent and recruitment processes. This led to the development of good practice guidance and other resources including an accessible animation for members of the public [ 107 ]. An animation aimed at adults enrolled in emergency care research which describes RWPC was developed by another research team (CoMMiTED Study) [ 108 ]. Systematic reviews have explored stakeholders’ views about the acceptability of RWPC [ 109 ], including ethnic minority populations’ views [ 110 ]. Such studies have found that RWPC is generally acceptable to patients, families, and practitioners but highlighted the importance of contextual factors.

The CATheter infections in Children Trial (CATCH) was the first UK trial to include research without prior consent when comparing the effectiveness of different types of central venous catheters to prevent bloodstream infections in children. An embedded study (called CONNECT [ 111 ]) explored parent and practitioner views and experiences of recruitment and consent and found that parents were momentarily shocked or surprised when they were informed that their child had already been entered into CATCH without their consent [ 101 ]. However, initial concerns were often quickly addressed by practitioner explanations about why it had not been possible to seek consent before enrolment and how the trial interventions were already used in clinical care. To prevent burden and assist decision-making, parents stated it was important for the research staff to assess the appropriate timing of research discussions after a child’s enrolment in a trial. They suggested that the researcher should consult with the bedside nurse about appropriate timing and only approach parents after the initial emergency situation has passed, when a child’s condition has stabilised [ 101 ]. The CONNECT study used these findings alongside wider research, involving practitioners, families [ 112 ], and children [ 113 ] with experience in emergency care, to develop guidance for future paediatric and neonatal trials [ 114 ]. Since its publication in 2015, CONNECT guidance has informed the successful conduct of five studies. This includes the first clinical trial of a drug for long-lasting seizures (EcLiPSE trial), which successfully recruited to time and target with a 93% consent rate and led to changes in clinical guidelines for children in status epilepticus [ 115 ].

Research into consent in emergency settings is high on the trials methodological research agenda and was identified as a research priority by Clinical Trials Units in a UK survey [ 116 ]. Areas for future research involving adults identified by the Perspectives Study included the need for evidence-based guidance on the procedures for professionals acting as a consultee or legal representative and identifying strategies to communicate with relatives of critically ill patients about research, including where a participant enrolled without prior consent subsequently dies [ 106 ]. The NIHR RfPB-funded study ‘ENHANCE’ will begin in 2023 and aims to address this gap in knowledge through the involvement of bereaved families and other key stakeholders.

Ongoing work in paediatric populations aims to assess and refine CONNECT guidance in low- and middle-income countries. Further work is needed to explore views on research without prior consent in underserved populations, such as parents who do not speak English and who are often excluded from qualitative studies and guidance development.

The need for more guidance for RECs who are reviewing emergency and urgent care trials and support for consent processes for patients and members of the public who join research teams and advise on studies, has also been highlighted [ 106 , 109 , 117 ].

Conclusions

The need for alternative consent processes that address the inadvertent exclusion of certain populations has been detailed in this article. Drives for trial efficiency, lack of funding, or time for adaptation often result in the exclusion of certain populations. However, inequities in health outcomes will continue to be exacerbated by health research until trials become more inclusive of underserved populations. Alongside methodological innovation, further research is required to establish good practice, develop evidence-based guidance, and support skill acquisition in the global research workforce. Our key recommendations for future research are summarised in Table 1 . Importantly, this should be done in collaboration with people with lived experience and those who care for them.

The populations detailed above are not the only areas where consent is complex or alternative pathways are required. Some trials have complex consent processes, not because of their recruited population, but due to an innovative treatment or trial design, such as cluster RCTs and Trials within Cohorts (TwiCs) [ 118 ]. As we progress with the innovation of trial design, we must progress methodological innovation in consent at the same pace or risk leaving certain populations behind. Many of the methodological lessons learnt and proposed adjustments, such as the routine provision of accessible information, could also benefit other underserved groups including those with lower literacy levels and English language proficiency, as well as the wider population of potential research participants.

The TMRP Complex and Alternate Consent Pathways group is driving forward this research agenda in the UK and is open to new members to share methodological learning. We have updated the NIHR Clinical Trials Toolkit [ 119 ] to reflect the most up-to-date research in this area. However, as this commentary has shown, current guidance remains limited in its utility and requires greater clarity and practical applicability for researchers, participants, family members, and ethical review committees. We are keen to use the momentum of the group to identify others with an interest in this area in order to collaboratively develop the research agenda and address the consent-based ethical and methodological challenges in trials. Many of these issues are not restricted to the UK but are encountered internationally, which raises additional challenges when conducting multi-national trials [ 58 , 97 , 120 ]. We encourage researchers from other regions and jurisdictions to share their experiences and ongoing research programmes and to contribute to developing an international research agenda to address these global challenges.

Availability of data and materials

Not applicable as no dataset was generated.

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Acknowledgements

We would like to thank the wider contributors to the Complex and Alternate Consent Pathways group and the MRC-NIHR Trials Methodology Research Partnership who have participated in the discussions at various stages of this work. JW would like to acknowledge the support of the QuinteT research group, University of Bristol.

No funding was received for this work. VS is supported by a National Institute of Health Research Advanced Fellowship (CONSULT) funded by the Welsh government through Health and Care Research Wales (NIHR-FS(A)-2021). AMR is supported by a Wellcome Trust Fellowship (Capacity, Consent and Autonomy https://capacityconsent.leeds.ac.uk/ ) (219754/Z/19/Z). AV is supported by a National Institute for Health Research Advanced Fellowship (NIHR302240). KG is supported by funding from the Chief Scientist Office of the Scottish Government’s Health and Social Care Directorate (CZU/3/3). This work was supported by the MRC-NIHR Trials Methodology Research Partnership (MR/S014357/1). RH is supported in part by the Wellcome Trust (209841/Z/17/Z and 223290/Z/21/Z), EPSRC (EP/T020792/1), and the NIHR Biomedical Research Centre at University Hospitals Bristol and Weston NHS Foundation Trust and the University of Bristol. RH also serves on various local, regional, and national ethics committees and related groups. None of the organisations played a role in the drafting of this article, and the opinions stated are those of the authors.

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Amy M. Russell and Victoria Shepherd are joint first authors.

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Leeds Institute of Health Sciences, University of Leeds, Leeds, UK

Amy M. Russell

Centre for Trials Research, Cardiff University, 4th floor Neuadd Meirionnydd, Heath Park, Cardiff, CF14 4YS, UK

Victoria Shepherd

Department of Public Health, Policy and Systems, Institute of Population Health, University of Liverpool, Liverpool, UK

Kerry Woolfall & Bridget Young

Health Services Research Unit, University of Aberdeen, Aberdeen, UK

Katie Gillies

Department of Psychology and Language Sciences, University College London, London, UK

Anna Volkmer

Department of Health Professions, Manchester Metropolitan University, Manchester, UK

Centre for Ethics in Medicine, Population Health Science, Bristol Medical School, University of Bristol, Bristol, UK

Richard Huxtable

Department of Non-communicable Disease Epidemiology, London School of Hygiene and Tropical Medicine, London, UK

Alexander Perkins

Medical Research Council Clinical Trials Unit at University College London (MRC CTU at UCL), Institute of Clinical Trials and Methodology, University College London, London, UK

Nurulamin M. Noor

Centre for Evaluation and Methods, Wolfson Institute of Population Health, Queen Mary University London, London, UK

Beverley Nickolls

Population Health Science, Bristol Medical School, University of Bristol, Bristol, UK

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The original idea for this Complex and Alternate Consent Pathways group (C&ACP) arose from the discussions in the Trial Methodology Research Partnership (TMRP) Qualitative Research group and the Inclusivity subgroup of the Trial Conduct Working Group and was led by JW. All authors are members of the C&ACP group and contributed to the iterative discussion of the content and structure of the manuscript. AR and VS wrote the first draft of the paper, and all authors contributed to the revision of it. The authors read and approved the final manuscript.

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Russell, A.M., Shepherd, V., Woolfall, K. et al. Complex and alternate consent pathways in clinical trials: methodological and ethical challenges encountered by underserved groups and a call to action. Trials 24 , 151 (2023). https://doi.org/10.1186/s13063-023-07159-6

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Geographic disparities in telemedicine mental health use by applying three way ANOVA on Medicaid claims population data

• Benjamin Ukert 1 ,
• Mark Lawley 2 &
• Hye-Chung Kum 3  

BMC Health Services Research volume  24 , Article number:  494 ( 2024 ) Cite this article

Metrics details

Utilization of telemedicine care for vulnerable and low income populations, especially individuals with mental health conditions, is not well understood. The goal is to describe the utilization and regional disparities of telehealth care by mental health status in Texas. Texas Medicaid claims data were analyzed from September 1, 2012, to August 31, 2018 for Medicaid patients enrolled due to a disability.

We analyzed the growth in telemedicine care based on urban, suburban, and rural, and mental health status. We used t-tests to test for differences in sociodemographic characteristics across patients and performed a three-way Analyses of Variance (ANOVA) to evaluate whether the growth rates from 2013 to 2018 were different based on geography and patient type. We then estimated patient level multivariable ordinary least square regression models to estimate the relationship between the use of telemedicine and patient characteristics in 2013 and separately in 2018. Outcome was a binary variable of telemedicine use or not. Independent variables of interest include geography, age, gender, race, ethnicity, plan type, Medicare eligibility, diagnosed mental health condition, and ECI score.

Overall, Medicaid patients with a telemedicine visit grew at 81%, with rural patients growing the fastest (181%). Patients with a telemedicine visit for a mental health condition grew by 77%. Telemedicine patients with mental health diagnoses tended to have 2 to 3 more visits per year compared to non-telemedicine patients with mental health diagnoses. In 2013, multivariable regressions display that urban and suburban patients, those that had a mental health diagnosis were more likely to use telemedicine, while patients that were younger, women, Hispanics, and those dual eligible were less likely to use telemedicine. By 2018, urban and suburban patients were less likely to use telemedicine.

Conclusions

Growth in telemedicine care was strong in urban and rural areas between 2013 and 2018 even before the COVID-19 pandemic. Those with a mental health condition who received telemedicine care had a higher number of total mental health visits compared to those without telemedicine care. These findings hold across all geographic groups and suggest that mental health telemedicine visits did not substitute for face-to-face mental health visits.

Peer Review reports

Healthcare services delivered through remote technology, known as telemedicine, have been growing across the United States even before the COVID-19 pandemic [ 1 , 2 , 3 , 4 ]. Telemedicine presents itself to be a cost-effective alternative to the traditional face-to-face consultations between providers and patients [ 5 , 6 , 7 , 8 , 9 ]. Benefits may include greater access to care, more timely diagnoses of conditions, and the potential to close gaps in care. Telemedicine can be especially beneficial for individuals facing systemic barriers to access, such as those living in provider shortage areas or those with limited transportation options [ 10 , 11 ].

The sudden universal reliance on telemedicine for medical care due to the COVID-19 pandemic has made it even more important to understand the conditions under which telemedicine is best used relying on available population data on telemedicine utilization. A detailed understanding of telemedicine utilization patterns before the COVID-19 pandemic will be critical to develop a good baseline. Innovative solutions to expand telemedicine delivery exists at the state level for those enrolled in Medicaid plans. States control Medicaid benefit packages and they have the option to include telemedicine as a mode of service delivery [ 12 ].

In Texas, Medicaid began covering telemedicine services in 1997 and telemedicine can be an especially promising tool of care delivery in Texas due to its size, extensive rural areas, and spatial disparities in access to care. Similarly, Texas has a large Medicaid population currently hovering close to 5 million patients, and covers a variety of residents with high disease burdens that may be especially suitable for telemedicine [ 13 , 14 ]. Presently, over 400,000 disability-related individuals are covered by Texas Medicaid, representing mostly individuals with limitations that can onset at younger ages and people who are limited in walking or moving and taking care of personal need. Texas eligibility for disability coverage through Medicaid is determined based on the diagnosis of a disability by medical professionals that suggest the inability to engage in gainful activity (causes include blindness, accidents and other developed diseases) or the presence of mental impairment that lasts for more than a year, as described in 42 U.S.C. § 1382c(a)(3) [ 15 ]. Mental illness can be one factor that impairs functioning and independent living. It is common in the Medicaid population, with Medicaid being the largest payor for mental health services [ 16 ]. However, few people with mental illness receive mental health care treatment and little is known about the level of treatment in the Medicaid population and through telemedicine delivery specifically [ 1 , 17 , 18 ].

Understanding how Medicaid patients may be able to benefit from telemedicine, especially those with disabilities and mental illness, is important to reduce gaps in care that can otherwise lead to unnecessary acute care needs [ 19 ]. Further, little is known about the extent of mental health care and telemedicine visit growth in urban, suburban, and rural areas. Understanding growth patterns of telemedicine and mental health care delivery by geography is important for Texas and beyond [ 1 , 17 ]. Additionally, there is a dearth of evidence on whether those with a mental illness are accessing mental telemedicine services in Texas.

To fill this knowledge gap, we describe the growth in telemedicine across rural and urban counties in Texas among Medicaid patients from 2013 to 2018. We focus on Medicaid patients with the highest potential need in Texas, those who are covered by Medicaid due to a disability, and then evaluate the use of mental health care and telemedicine-delivered mental health care for those diagnosed with a mental health diagnosis. Finally, we are also interested in evaluating whether the growth of telemedicine has led to more telemedicine mental health care visits that could have substituted for face-to-face mental healthcare visits. We do so by comparing trends in mental health use for disabled Medicaid patients diagnosed with a mental illness who engaged in telemedicine mental health compared to those that did not.

Medicaid claims data for Medicaid Fee for Service (FFS) and Managed Care Organization (MCO) patients were obtained from Texas Health and Human Services Commission (HHSC). FFS Medicaid was the most common approach where the state pays providers based on the number and type of services rendered. Following national trends, Texas transitioned individuals from FFS to MCO health plans in the last 20 years and most individuals are enrolled in a MCO health plan today. In MCO plans the state pays a monthly fixed capitated payment for each covered individual to an insurance company who is then responsible for the management, delivery, and payment of healthcare services [ 20 ].

The data include all inpatient and outpatient care interactions and patient demographics, plan enrollment, and eligibility information. The claims included claims data from September 1, 2012 to August 31, 2018 (reflecting the collection of data for six state fiscal years) for Medicaid patients who had at least one healthcare interaction with a provider who billed for at least one telemedicine service procedure in a given state fiscal year. This adjusts for the fact that the number of providers providing telemedicine in these earlier years changed each year. Thus, the general study population are Medicaid patients who had access to a telemedicine provider in any given state fiscal year. We then restricted the dataset to those who were enrolled in a Medicaid plan under the disabled category because this group often faces barriers to in-person care and generally has behavioral health needs for which telemedicine may be especially suitable [ 21 ].

We define “patient” to refer to a disabled Medicaid patient whose provider engaged in some form of telemedicine; the term “tele patient” refers to a disabled Medicaid patient who used telemedicine; the term “mental health patient” refers to a disabled Medicaid patient who had a primary diagnosis of schizophrenia, bipolar disorder or depression (see Appendix Table 1 for diagnoses codes used) during a provider visit; and the term “mental health tele patient” refers to a disabled mental health patient who had a primary diagnosis for mental health during the telemedicine visit. We limit our analysis to those disabled Medicaid patients diagnosed with of schizophrenia, bipolar disorder or depression because we aim to focus on the disabled population who (1) have a diagnosed and long-term need for mental healthcare, and (2) where telemedicine represents an opportunity for continuous care delivery [ 2 ].

We examined growth in total patients, tele patients, mental health tele patients, volume of mental health visits, and mental health telemedicine visits for state fiscal years 2013–2018. Patients were identified based on whether they had a real-time interactive audio and video telemedicine visit or saw a provider who delivered telemedicine (but did not have a telemedicine visit). In some instances, it is possible that telemedicine was only rendered through audio, though, specific CPT and HCPCS codes for audio-only telemedicine visits became available for reimbursement purposes generally after 2017. Telemedicine visits were identified based on HCPCS procedure codes that directly identify teleservices (e.g. G0406-G0408) and CPT codes (e.g. 90,791–90,792) that included modifier codes GT or 95. A full list of codes can be found in Appendix Table 1. We identified mental health visits based on whether the primary diagnosis code included a diagnosis of schizophrenia, bipolar disorder or depression [ 22 ]. Diagnoses were identified from ICD9 and ICD10 codes.

Enrollment characteristics included age in years, gender, race (non-Hispanic white, non-Hispanic black, Hispanic, other), dual eligibility with Medicare, and FFS or MCO plan enrollment at the first time the patient was observed in the data in a state fiscal year. Patients were not required to be enrolled for the entire study period. Patients clinical profile was also described with the Elixhauser Comorbidity Score measured in the six months prior to the first of the telemedicine visit or the six months prior to the first eligibility month in a state fiscal year for non-telemedicine patients.

We combined patient’s county of residence with the 2013 nine county level urban-rural continuum information collected by the United States Department of Agriculture [ 23 ]. We defined urban counties as those with a metro area population of at least 250,000; suburban counties as those with an urban population of at least 2,500 and metro area population of less than 250,000; and rural counties as counties completely rural or having less than a 2,500 urban population.

The goal was to examine the growth across urban, suburban, and rural counties from 2013 to 2018 in the number of (1) Medicaid patients receiving any care from providers who deliver telemedicine, (2) tele patients, (3) mental health patients, and (4) mental health tele patients.

Statistical analysis

We describe our sample by first presenting demographic characteristics for all patients and then separately for tele and non-tele patients. We used t-tests to test for differences in sociodemographic characteristics across patients. We then estimated patient level multivariable ordinary least square regression models to estimate the relationship between the use of telemedicine and patient characteristics in 2013 and separately in 2018. Variables of interest include geography, age, gender, race, ethnicity, plan type, Medicare eligibility, diagnosed mental health condition, and ECI score. Marginal effects are presented. We also estimate ordinary least square regression results with the dependent variable being the annual growth of patients, and the growth in the annual number of visits for telemedicine patients.

To describe changes in overall growth in patients, we used three-way Analyses of Variance (ANOVA) to evaluate whether the growth rates in patients from 2013 to 2018 were different based on geography and patient type. Specifically, we tested whether the growth rate was different by geography (urban vs. suburban vs. rural), tele vs. non-tele patients, and mental health vs. non-mental health patients. We also examine the interactions between these groups and settings and report the p-values relative to the rural counties when multiple comparisons were possible. The growth rate for each combination was computed as (total number of 2018 patients – total number of 2013 patients) / total number of 2013 patients. We then took the log transformation of these rates and applied the standard three-way ANOVA, using the 3-way interaction as an estimate of the mean squared error.

Finally, we also estimated the 2013 to 2018 relationship of annual growth rates and patient characteristics for the growth rate for patients with a mental health diagnosis vs. without a mental health diagnosis, the growth rate of patients who used telemedicine vs. non-telemedicine patients, and the growth rate of telemedicine use among patients with a mental health diagnosis vs. those patients with a mental health diagnosis without telemedicine use.

There were 519,454 patients in our disabled sample with 9.2% (48,024 patients) receiving some care through telemedicine (Table  1 ). The average age of the sample was 37.06 years (36.8 among tele patients, p-value = 0.002), 50.02% were female (47.0% among tele patients, p  < 0.001), and 27.7% were dual eligible (23.8% among tele patients, p  < 0.001). Among racial groups, 24.9% were non-Hispanic White (27.9% among tele patients, p  < 0.001), 15.2% were non-Hispanic Black (14.7% among tele patients, p  = 0.008), and 28% were Hispanic (27.1% among tele patients, p  < 0.001). Finally, 45.2% of patients were enrolled in FFS Medicaid (35.6% among tele patients, p  < 0.001), and 89.8% of patients lived in urban and suburban counties (86.9% among tele patients).

Regression results in Table  2 display that in 2013, urban and suburban patients were more likely than patients in rural counties to use telemedicine (Table  2 , 2.7% points, p  < 0.001; 5.8% points; p  < 0.001, respectively). Patients that had a mental health diagnosis were more likely to use telemedicine (15.9% points, p  < 0.001) while patients that were younger, women, Hispanics, and those dual eligible were less likely to use telemedicine. By 2018, urban and suburban patients were less likely than patients in rural counties to use telemedicine (4.9% points, p  < 0.001; 3.5% points, p  < 0.001). Patients that were younger, women, and those dual eligible were less likely to use telemedicine.

Of the 519,454 patients in the sample, 207,921 (40.0%) had a mental health diagnosis at a visit. Of the 48,024 tele patients, 39,544 (82.3%) had a mental health diagnosis, whereas among the 471,430 non-tele patients, only 168,377 (35.7%) had a mental health diagnosis. The 168,377 mental health non-tele patients generated an average of 399,203 mental health visits per year for an average of 2.37 visits per patient per year. While the 39,544 mental health tele patients generated an average of 109,281, mental health visits per year for an average of 2.76 visits per patient per year. On average, for mental health tele patients, 12.5% of mental health visits occurred via telemedicine across the 6-year period.

Figure  1 displays disabled Medicaid patient types and patient growth rate from year 2013 to 2018 for urban, suburban, and rural counties. The number of patients receiving care from a provider who engaged in some form of telemedicine grew from 54,455 to 126,580 (132%), with the largest growth rate being in urban patients (255%, p  = 0.049). Over the same period, mental health patients grew by 90% from 25,177 (46% of total patients) to 47,719 (38% of total, p  = 0.059), with urban mental health growth being the highest compared to the rural growth rate (152%, p  = 0.124). See Appendix Table 2 for all counts.

All patients, mental health patients, tele patients, and mental health tele patients. Notes: Figure describes the growth in the unique number of Medicaid patients seen by telemedicine providers in Texas

The number of tele patients grew from 5,596 in 2013 to 10,145 in 2018 (81%, p  = 0.132), with the largest growth rate being in rural patients (181%) followed closely by urban (152%, p  = 0.779). Over the same period, the number of mental health tele patients grew from 4,582 (82% of all tele patients) to 8,138 (80% of tele patients, p  = 0.190) with the largest growth rate being in rural counties (160%) followed closely by urban (155%). See also ordinary least squares multivariable regressions in Appendix Table 3. The percentage of tele patients represented 10% of all patients in 2013 to 8% in 2018. This trend was similar for urban (10–7%) and suburban (11–8%) but much different for rural areas, where the percentage of patients using telemedicine grew from 6% in 2013 to 13% in 2018. Similar results are seen for mental health patients, with urban tele mental health patients hovering around 15% of total mental health patients from 2013 to 2018, suburban tele mental health patients dropping from 22 to 18%, and rural tele mental health patients growing from 11 to 22%.

Figure  2 provides information on mental health visits. The average number of mental health visit per patient was about 14.5 per year for patients with a primary diagnosis of mental health during the visit. The average number of visits increased slightly from 2013 with 14.3 visits per patient per year to 15.8 visits per patient per year in 2018. Urban and suburban mental health visit rates were lower compared to rural mental health visit rates, with 14.3 and 13.9 visits per year in 2013 for urban and suburban patients and 15.4 visits per year in 2013 for rural patients. By 2018, all areas experienced growth in average visits compared to 2013, and rural mental health patients averaged 19.1 visits per year, while urban and suburban enrollees averaged 15 and 15.8 visits per year ( p  = 0.323 and p  = 0.345, respectively). See counts in Appendix Table 4 and multivariable regressions in Appendix Table 5.

Average annual mental health visits per patient among mental health patients. Notes: Figure describes the average annual number of visits for different Medicaid patients

Stratifying mental health visits by telemedicine mental health and non-telemedicine mental health patients yields additional insights. Telemedicine mental health enrollees had, on average, 16.6 mental health visits compared to 14.0 mental health visits for non-telemedicine mental health patients ( p  < 0.001). Of note is that roughly two mental health visits out of the 16.6 mental health visits for telemedicine mental health patients were delivered through telemedicine. Thus, excluding telemedicine visits suggests that telemedicine mental health patients and non-telemedicine mental health patients had a similar level of mental health visits.

During the last 20 years, use in telemedicine accelerated, however, use varied substantially across geography and visit type. We observed an 81% growth rate of disabled Medicaid patients with a telemedicine visit between 2013 and 2018. However, patient panels grew at 132%, thus telemedicine usage did not keep pace with overall patient growth at telemedicine providers, with suburban patients showing the slowest increase. In contrast, for rural patients, telemedicine usage increased by 181% while the total number of rural patients increased by only 32%. Thus, relative to patient population size, the rural disabled Medicaid patients exhibited a much greater uptake of telemedicine than did the urban and suburban populations. Still, by 2018, 86% of telemedicine patients were still located in urban and suburban counties, which is similar to other published work, and large potential growth in telemedicine use persists as only 7% of all urban and suburban disabled Medicaid patients had a telemedicine visit [ 1 , 24 ].

At the same time, the number of tele mental health patients increased by 77% from 2013 to 2018. As before, overall telemedicine usage among disabled Medicaid mental health patients did not keep pace with overall growth in mental health patients of 90%. This was due to the suburban population, which exhibited a 37% growth in tele mental health patients as opposed to a 77% growth in total mental health patients. The trend was just the opposite for rural counties, which exhibited a 75% growth rate in mental health patients and a 160% growth rate in tele mental health patients. Still, as of 2018, 87% of tele mental health patients were located in urban and suburban counties and only 16% of all urban and suburban disabled Medicaid patients with a mental health visit had a telemedicine mental health visit. Relative to earlier work that suggests relatively little mental healthcare delivery through telemedicine among the Medicare population; our findings paint a different and much more positive picture [ 2 ].

One of the most important observations on mental health visits is that telemedicine patients tended to have between two and three additional visits per year than non-telemedicine mental health patients. Given that the health risk profile looked similar across telemedicine groups, these findings hold across all geographic groups and suggest that mental health telemedicine visits may not substitute face-to-face visits, but that they could have led to additional face-to face visits. These findings are similar to work that has displayed that telemedicine care leads to additional care [ 9 ]. Whether additional visits may provide a positive return on investment for the disabled Medicaid population remains an open question.

Between 2013 and 2018, we also observed a shift in geography of disabled Medicaid patients seeing a telemedicine provider. The percentage living in urban, suburban, and rural counties shifted from 25%, 59%, 15% in 2013 to 38%, 53%, and 8% in 2018, respectively, reflecting a growing prevalence of disability in urban areas over this time, though we observe similar growth in telemedicine providers in urban and rural counties between 2013 and 2018. At the same time, we observed an 18% drop in the share of disabled patients with a mental health diagnosis. This was due to decreased prevalence of mental health diagnoses in suburban and rural populations, which fell by 26% and 46%, respectively. In contrast, the prevalence of mental health diagnoses in urban counties increased by 8%. This is in line with evidence that suggests that urban areas have greater access to mental health providers than rural areas [ 25 ]. Future growth in mental health telemedicine visits will depend on the availability and participation of providers. Still, by 2018, the majority of mental health patients (53%) were located in suburban counties.

The future policy direction of telemedicine care is evolving. The Center for Medicare and Medicaid Services (CMS) has been at the forefront of telemedicine delivery by establishing payments for physicians treating Medicare beneficiaries in 1999 [ 26 ]. However, Medicare provides reimbursement only to those living in rural areas and has only extended coverage temporarily during the COVID outbreak to all Medicare beneficiaries [ 12 , 27 ]. At the onset of the COVID-19 pandemic, many providers shifted to provide telemedicine visits, and states Medicaid programs have expanded access to telemedicine use [ 13 , 28 ]. State regulatory environment has pivoted back to pre COVID-19 policies, by for example, not allowing licensure waivers for telemedicine use across state borders [ 29 ].

Our study has limitations. First, while we believe we capture most video telemedicine visits, it is possible that some phone visits are included as nost audio-only CPT codes became available during the COVID-19 pandemic in 2020 and later. Second, our findings are limited to a subset of disabled patients who saw a telemedicine provider. Third, our findings for disabled Medicaid patients may not be generalizable to other populations, such as the disabled Medicaid population in other states and other non-disabled Medicaid patients. Fourth, telemedicine access requires that patients have IT equipment and broadband internet access. The large number of rural areas in Texas with potentially limited broadband access during this time may understate the growth in telemedicine delivery relative to other states. Finally, our data relies on data in a state with a long history of telemedicine reimbursement policies, thus one could expect that a larger share of providers is willing and able to deliver care through non-face to face means compared to other states.

Our results have far-reaching implications for the future of telemedicine delivery in Texas and across the United States. The COVID-19 pandemic has led to substantial increases in short-term telemedicine use. Additionally, telemedicine has been championed to replace face-to-face visits. However, our study suggests that telemedicine care does not seem to substitute existing care among mental health visits, implying that additional care was delivered. Similar findings have previously been displayed among a sample of commercially insured employees [ 9 ]. However, telemedicine visits may have broader benefits, it could lead to improved continuity of care, early detection of condition on setting, and improve medication adherence. Most primary care physicians and specialists can agree that additional care seeking behavior may just display unmet needs, this is supported by the fact that the US has one of the lowest doctor visit ratios among OECD countries [ 30 ]. It remains to be seen whether telemedicine will remain a complement to care seeking rather than substituting care.

Data availability

The data that support the findings of this study are available from TX-HHSC but restrictions apply to the availability of these data due to confidentiality requirements. Data were used for evaluation purposes in the current study and so are not publicly available. Contact the corresponding author for more information on how to request the data.

Abbreviations

Analyses of Variance

Center for Medicare and Medicaid Services

Medicaid Fee for Service

Managed Care Organization

Texas Health and Human Services Commission

Organization for Economic Cooperation and Development

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Acknowledgements

We thank Morgan Goldstein and Marc Montrose at HHSC for assistance with data acquisition, interpretation, and constructive feedback on the findings.

This study was conducted with support in part by the Population Informatics Lab, the Texas Virtual Data Library (ViDaL) at Texas A&M University and Texas Health and Human Services Commission (TX-HHSC). The statements presented in this article are solely the responsibility of the authors and do not necessarily represent the views of the funders.

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Ukert, B., Lawley, M. & Kum, HC. Geographic disparities in telemedicine mental health use by applying three way ANOVA on Medicaid claims population data. BMC Health Serv Res 24 , 494 (2024). https://doi.org/10.1186/s12913-024-10898-0

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Students with disabilities match with physician residency programs at lower rates

by Yale University

Medical students who report having a disability are less likely to match into a residency program, according to a new Yale study involving residency programs across the United States.

The findings, researchers say, suggest students with disabilities may experience barriers to training and career opportunities, particularly in certain medical specialties.

"Physicians with disabilities are underrepresented in the medical field," said Mytien Nguyen, an M.D.-Ph.D. student at Yale School of Medicine and lead author of the study. "Lower match rates could contribute to that disparity, which ultimately has negative impacts on the field and patient care."

For the study, published n the journal JAMA , the researchers used data from the National Resident Matching Program, which matches applicants to residency programs in the United States. They analyzed match outcomes for individuals applying to residency programs in 2022 and 2023.

Of the nearly 70,000 people who applied for residencies during that time period, 5.9% reported having a disability, answering "yes" to the application question "Are you a person with disability (e.g., ADHD, learning, psychological, chronic health, mobility, hearing, vision, etc.)?"

After evaluating match rates for those reporting and not reporting a disability, the researchers found a small but significant difference in match rates, with 83.2% of those not reporting disabilities successfully matching to residency programs compared with 81.8% of applicants who reported a disability.

When researchers then looked at rates across medical specialties, they found several where match rates were not significantly different between the two groups. In others, however, including otolaryngology, neurology, physical medicine and rehabilitation, dermatology, pediatrics, internal medicine , and emergency medicine , match rates were higher for applicants with a disability.

Driving the overall lower match rates for students with disabilities were the rates for general surgery —65.5% compared with 76.2% for students not reporting disabilities—and orthopedic surgery—58.6% versus 73.3%.

During the matching process, students apply to specific programs, interview, and then rank programs in order of preference. Programs rank their applicants as well and the National Resident Matching Program uses an algorithm to determine matches based on the two sets of rankings.

The match rate variability across specialties, researchers say, may reflect differences in specialty culture or their applicant assessment practices, with biases--whether conscious or unconscious--affecting match rates.

"With this gap, I think it's important for each specialty, not just surgical specialty, to think about their processes and incorporate disability as a metric to examine in addition to race, ethnicity, and sex when they're evaluating their program's diversity," said Nguyen.

The differences across specialties may also point to positive approaches that others can adopt or build from, said senior author Dowin Boatright, who is now vice chair of research in the Department of Emergency Medicine at New York University Grossman School of Medicine and began this line of research while at Yale School of Medicine.

"Looking at specialties with higher match rates for applicants with disabilities may uncover successful strategies for equitable evaluation," he said.

Organizations like the Accreditation Council for Graduate Medical Education, which evaluates medical residency and internship programs, play a role as well, said Nguyen, noting the work the body is already doing to promote awareness and support for learners with disabilities.

"And of course the number of applicants with disabilities in our study was very low, which speaks to disparities that may exist earlier in the training pathway and need to be addressed as well," she said.

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• v.9(Suppl 1); 2020 Feb

A clinical primer on intellectual disability

Dilip r. patel.

1 Western Michigan University Homer Stryker MD School of Medicine, Kalamazoo, Michigan, USA;

Maria Demma Cabral

Joav merrick.

2 Hadassah Hebrew University Medical Center, Jarusalem, Israel;

3 University of Kentucky, Lexington, USA;

4 School of Public Health, Georgia State University, Atlanta, USA

Between 1% and 3% of persons in general population are estimated to have some degree of intellectual disability. A diagnosis of intellectual disability is based on clinical history, level of intellectual ability and level of adaptive function. Both, the intellectual and adaptive functioning are measured using individually administered standardized tests. More than 75% of persons who have intellectual disability have mild intellectual disability and an underlying specific etiology is less likely to be identified; whereas, in a small percentage of persons with severe intellectual disability, an underlying specific biologic cause is highly likely to be identified. Genetic abnormalities, inborn errors of metabolism and brain malformations are major categories of causes identified in severe to profound intellectual disability. The initial clinical presentation and recognition depends on the severity and underlying cause of intellectual disability. The etiology, severity, cognitive abilities, and adaptive function, vary among persons with intellectual disability and need consideration in developing a treatment plan. The physician plays an essential role in the evaluation, treatment of associated medical conditions and preventive care, and in facilitating and coordinating consultative services and community based care.

Introduction

Deficits in intellectual and adaptive functioning are the main characteristics of intellectual disability with a reported prevalence between 1% and 3% of the population with some regional variations ( 1 , 2 ). By definition, the onset of intellectual and adaptive deficits has their onset during the developmental period from infancy thorough adolescence. For simplicity, we have used the term persons in this review to include infants, children, adolescents and adults. Also, we recognize that different definitional criteria use the equally valid term intellectual developmental disorder instead of the term intellectual disability to describe the range of disorders of deficits in intellectual and adaptive functioning.

Most persons with intellectual disability have mild intellectual disability, for which an underlying biologic cause is less likely to be identified; whereas, a small percentage of persons who have severe to profound intellectual deficit, an underlying biologic cause is highly likely to be identified ( 1 - 4 ). In addition to clinical evaluation, both the cognitive ability and adaptive functioning are ascertained based on individually administered standardized tests. The effective delivery of healthcare for persons with intellectual disability requires participation and coordination between many different medical as well as other related disciplines and agencies that provide support services for persons with intellectual disability.

In most countries various laws, acts or regulations provide the framework for the legal rights of persons with disabilities, organization of community based support and intervention services, and variable levels of governmental and non-governmental funding for various support services—intervention, educational, vocational, social and other services—for persons who have intellectual and other developmental disabilities ( 1 - 4 ). In the clinical practice setting, the medical practitioner takes a lead role in medical evaluation and treatment as well as providing ongoing preventive healthcare for persons with intellectual disability. Additionally, the medical practitioner also plays an essential role in coordinating appropriate consultations with other specialists, as well as in facilitating access and care through community-based agencies and person.

Intellectual disability is best considered within the broader framework of disability in general. The conceptual framework and definition of disability has evolved over the years from a predominantly medical model to a broader model that encompasses and functionally merges the medical, social and functional aspects of disability ( 5 - 7 ). This has significant individual, societal, economic, political, healthcare delivery and policy implications, making it challenging to construct a unifying definition of disability ( 5 - 8 ).

The medical model of disability considers a person to have a disability based on a disease or health condition that limits their ability to carry out expected individual and societal obligations ( 5 - 8 ). This is accompanied by a range of disability based on severity within the context of the many conditions that can affect an individual’s functioning. The medical model attributes functional limitations associated with disability to the person, which implies that if their condition is cured, then the disability could be remediated ( 5 - 8 ). There is minimal consideration of contribution of the environmental or societal factors in the medical model, which generally views the person with a disability in a sick role ( 5 - 8 ).

The social model, on the other hand, takes into consideration the role of the environmental and societal factors in its construct of disability ( 5 - 8 ). Disability within the context of social model is considered based on the understanding that by itself, functional impairment at an individual level by itself may or may not result in disability; rather, sociocultural expectations and built environment together limit a person’s ability to engage in expected individual and societal obligations ( 5 - 8 ).

It is important to understand the frequently used terms impairment, handicap, and disability ( Table 1 ) ( 4 - 8 ). The World Health Organization (WHO) has developed the International Classification of Functioning, Disability and Health, which comprises three key components: (I) body function and structure; (II) activity; and (III) participation ( Table 2 ) ( 9 - 12 ). The general WHO classification provides a framework for delineating specific disabilities. The three key components of the WHO classification are interrelated and may interact with a given health condition and individual and environmental factors.

The term intellectual disability or intellectual developmental disorder is used to describe significantly sub-average intellectual and adaptive functioning based on clinical assessment and as measured by individually administered, appropriately normed, standardized and validated tests of intellectual functioning and adaptive behavior, with onset during the developmental period from infancy through adolescence ( 10 , 13 - 15 ). According to the WHO classification, “Disorders of intellectual development are a group of etiologically diverse conditions originating during the developmental period characterized by significantly below average intellectual functioning and adaptive behavior that are approximately two or more standard deviations below the mean (approximately less than the 2nd/3rd percentile) ( 10 ).” A diagnosis of intellectual disability should not be based solely on the results of tests of intellectual and adaptive functioning; rather, it is based on a combination of clinical assessment and judgment in conjunction with the results of standardized tests of intellectual and adaptive functioning ( 1 , 8 , 10 , 13 , 15 ). The assessment of intellectual functioning and adaptive behavior may be influenced by a number of factors ( Table 3 ) that should be considered in the administration of the tests and interpretation of their results ( 8 , 10 , 13 , 15 , 16 ).

According to the Diagnostic and Statistical Manual of Mental Disorders (DSM-V), the deficits in intellectual function include reasoning, problem solving, planning, abstract thinking, judgment, academic learning, and learning from experience ( 15 ). The critical components of intellectual functioning included in the DSM-V criteria are verbal comprehension, working memory, perceptual reasoning, quantitative reasoning, abstract thought, and cognitive efficacy ( 15 ).

Widely accepted definition and classification systems for intellectual disability consider adaptive functioning in terms of conceptual, social, and practical domains ( 8 - 10 , 13 - 15 ) ( Table 4 ). Deficits in adaptive functioning result in failure to meet developmental and sociocultural standards for personal independence and social responsibility. A number of factors ( Table 5 ) may either facilitate or hinder adaptive functioning in one or more activities of daily life, such as communications, social participation, and independent living, across multiple environments such as home, school, work, and community ( 4 , 7 , 8 , 13 , 15 ).

Epidemiology

The reported prevalence of intellectual disability is 1% globally and vary from 1% to 3% by country, with a male to female ratio of 2:1 ( 8 , 12 , 13 , 15 - 27 ). Although the prevalence of mild intellectual disability has declined somewhat in recent years, the prevalence of severe intellectual disability has remained same during that time at approximately 0.3–0.5% of population ( 12 , 13 , 16 , 27 ). Statistically, the prevalence of intellectual disability in the general population is expected to be 2.5% ( 13 , 15 ). This is based on the understanding that intelligence [measured as intelligence quotient (IQ), by individually administered standardized tests of intelligence] has a typical distribution in the population and applying 2 standard deviations below the mean as a cutoff point ( 13 , 15 ). When one child is diagnosed having severe intellectual disability, the recurrence risk for intellectual disability in subsequent child is estimated to range from 3% to 9% ( 13 , 15 , 16 ).

Measured IQ is considered an approximation of intellectual functioning, which may or may not necessarily correlate with the level of adaptive functioning ( 1 , 8 , 13 , 15 , 16 ). However, based on a number of epidemiological studies, persons with an IQ score of 50 or less are considered to have severe intellectual disability; whereas, persons with an IQ above 50 are considered to have mild intellectual disability ( 13 - 16 ). Almost all persons with a measured IQ of ≤50 manifest some degree of adaptive deficits ( 13 - 16 ). Major risk factors for mild intellectual disability are environmental and psychosocial, and include low socioeconomic status, residence in low-income countries, low maternal education, malnutrition, and inadequate access to healthcare ( 8 , 11 - 16 , 28 ). On the other hand, in persons with severe intellectual disability, a specific underlying genetic, biological or neurological can be identified in more than 75% of cases ( 8 , 11 - 13 , 28 ). The most common identified conditions in persons with severe intellectual disability include chromosomal abnormalities, genetic syndromes ( Table 6 ), congenital brain malformations, neurodegenerative diseases, congenital central nervous system infections, inborn errors of metabolism ( Table 7 ), maternal disease during pregnancy, in utero exposure to toxins, and birth injury ( 13 , 15 , 16 , 27 - 30 ).

Clinical features

The clinical symptoms and signs of intellectual disability are generally first recognized during infancy and childhood. The predominant clinical symptoms and signs suggestive of intellectual disability vary depending up on the age at presentation, specific cause and the severity of cognitive deficit ( 1 , 4 , 8 , 13 , 15 , 16 ). Infants and young children with severe intellectual deficit are highly likely to have an underlying cause and present early with clinical features associated with the underlying condition ( 13 , 16 , 28 , 30 ). On the other hand, a large percentage of children with mild range of intellectual deficits are highly unlikely to have an identifiable underlying specific cause or clinical findings, and may not be recognized to have intellectual disability until later in childhood ( 13 , 16 , 28 , 30 ). Some children with mild ID may not be recognized until 5–9 years of age ( 16 ). Abnormal findings on physical examination such as macrocephaly or microcephaly, dysmorphic features, multiple congenital anomalies, or difficulty with feeding in a newborn should prompt consideration of associated intellectual disability ( 8 , 13 , 16 ). Gross motor delay is a common presentation ID during infancy; whereas, during pre-school and early school years language, learning and academic difficulty as well as behavioral concerns is common presentation in children with ID ( 16 ).

Mild intellectual disability

Eighty-five percent of persons with intellectual disability have mild severity and are less likely to be recognized to have ID before 5–6 years of age ( 8 , 13 , 16 ). Mild intellectual disability is characterized by measured intellectual and adaptive functioning that is approximately two to three standard deviations below the mean (approximately 0.1–2.3 percentile) ( 10 ). The measured IQ for persons with mild ID is between 50–55 and 70 (based on population mean of 100; 1 standard deviation of 15, and margin of measurement error of ±5) ( 8 , 10 , 13 - 16 ). Persons with mild ID have difficulty in the acquisition and comprehension of complex language and academic skills ( 10 , 13 , 15 , 16 ). They can reach academic skills equivalent to 4 th –5 th grade level ( 10 , 16 ). In addition to language difficulty, persons with mild ID also have difficulty in arithmetic and writing skills ( 10 , 15 , 16 ). With appropriate support, they are able to develop basic skills in reading, writing and mathematics. Most are able to write simple letters and complete a simple job application ( 10 , 15 , 16 ).

Adolescents and adults with mild ID continue to show deficits and limitations in executive functioning such as planning, organizing, priority setting and abstract thinking ( 15 ). They also show limitations in tasks that require short-term recall. Persons with mild ID are at an increased risk for being manipulated by others ( 15 ). They show limited ability to accurately judge or comprehend the norms of social discourse and interactions, and, often, their behavior in social situations may be considered immature or inappropriate by others ( 15 ).

With appropriate support, persons with mild intellectual disability are able to acquire most skills needed for daily household, practical and self-care activities ( 10 , 13 , 15 ). Persons with mild intellectual disability can be sufficiently skilled as adults to be gainfully employed and able to maintain independence in daily life activities ( 10 , 13 , 15 ). They can learn simple employment related skills such as report to work at expected time, stay at task, and engage in appropriate social interactions with coworkers ( 10 , 13 , 15 ). They can also learn to drive and use public transportation. Overall, most persons with mild ID function at mental age of 9–11 years as adults ( 10 , 13 , 15 ). Persons with mild ID need intermittent support in daily living activities such as self-care, shopping, food preparation and money management ( 10 ). They need assistance in making healthcare and legal decisions. Persons with mild ID can acquire basic vocational skills as adults, and may be employed in jobs that do not require application of academic or conceptual skills ( 10 , 13 , 15 , 16 ).

Moderate intellectual disability

About 10% of persons with ID have moderate severity and likely to be recognized to have an ID by 3–5 years of age ( 8 , 13 , 16 ). A moderate ID is characterized by intellectual and adaptive functioning that are approximately three to four standard deviations below the mean (approximately 0.003–0.1 percentile) ( 10 ). The measured IQ of persons with moderate ID falls between 35–49 and 50–55 ( 8 , 10 , 13 - 16 ).

The rate of acquisition of basic language skills is slow in children with moderate ID ( 8 , 10 , 16 ). The acquisition and development of language as well as the ability to learn and attain proficiency in academic skills are limited in persons with moderate intellectual disability person ( 10 , 15 ). Persons with moderate ID show significant limitations in reading, writing, mathematics and other skills requiring understanding basic concepts ( 10 , 13 , 15 ). Persons with moderate ID may develop some basic skills, for example, ability for sight-word reading, copy information, and match number to number of items ( 10 , 13 , 15 ). They can acquire academic skills equivalent to early elementary school grade level ( 10 , 15 ). Persons with moderate ID experience difficulty in social communication, interpersonal interactions and understanding behavioral norms expected for age and social context ( 15 ). With ongoing support, persons with moderate ID can develop meaningful family and personal relationships. They have limitations in social decision-making and social judgment ( 15 ). Development of maladaptive behaviors is a significant concern in persons with moderate ID and this can further contribute to difficulty in social interactions and relationships ( 15 ).

Persons with moderate ID function at mental age of about 6-8 years as adults ( 8 , 10 , 13 , 15 , 16 ). Persons with moderate intellectual disability need substantial ongoing support as adults to maintain independence in daily living activities and to remain gainfully employed ( 13 ). With ongoing support and teaching, persons with moderate ID may be able to acquire basic daily living and job skills. They need continued supervision and guidance in daily activities as well as performing job related tasks. A person with moderate ID can attain relative independence in self-care and daily living tasks such as eating, dressing, elimination and personal hygiene ( 10 , 13 , 15 ).

Severe intellectual disability

About 4% of persons with ID have severe ID, and are highly likely to be recognized as having severe ID by 3 years of age or earlier ( 8 , 13 , 16 ). Severe ID is characterized by intellectual and adaptive functioning that are approximately four or more standard deviations below the mean (less than approximately the 0.003rd percentile) ( 10 ). The measured IQ of persons with severe ID falls between 20–25 and 35–40 ( 10 , 13 , 15 ). In addition to severe deficit in intellectual functioning, persons with severe ID may also have motor impairments and other associated conditions that further limit intellectual and adaptive functioning ( 8 , 15 ). Persons with severe ID function at mental age between 3 and 5 years as adults ( 8 , 10 , 15 , 16 ). Persons with severe ID need extensive, regular, consistent, and lifetime support in daily living activities, and are care dependent ( 13 , 15 ).

Persons with severe ID have significantly limited language and communication ability. They have significant limitations in spoken language; communication may be limited to use of single words or phrases ( 10 , 15 ). Their communication ability may be improved with use of augmentative communication methods. They often use gestures to communicate basic needs. Persons with severe ID have significant limitations in understanding concepts of numbers, quantity, time, management of money and problem solving ( 10 , 13 , 15 , 16 ). The social interactions and relationships for persons with severe ID are largely limited to immediate family members and care takers.

A person with severe ID requires intensive support in all activities of daily living including self-care and personal hygiene ( 10 , 15 ). They are not able to make sound judgments or decisions that may affect the wellbeing of self or others and require constant supervision ( 15 ). Maladaptive behavior, including self-harm, is a significant concern in persons with severe ID ( 15 ).

Profound intellectual disability

Approximately 1% of persons with ID have profound ID and recognized by 2 years of age ( 8 , 13 , 15 , 16 ). A profound ID is characterized by intellectual and adaptive functioning that are approximately four of more standard deviations below the mean (approximately less than the 0.003 rd percentile) ( 10 ). Persons with profound ID have IQ less than 20–25 ( 8 , 13 , 15 , 16 ). Persons with profound ID function at about 3 years of mental age and need high intensity, pervasive support across all environments ( 8 , 10 , 15 , 16 ). They have significant limitations in self-care, continence, communication and mobility. They may need complete custodial or nursing care and are total care dependent ( 13 , 15 ). Severe ID is also associated with other impairments such as motor and sensory disorders that further limit acquisition and use of motor, adaptive or social skills. Severe and profound ID are differentiated primarily based on differences in limitations of adaptive behavior because the validity and reliability of standardized tests of intelligence are not clearly established when used in persons with intellectual functioning that is below the 0.003 rd percentile person ( 10 ).

Persons with profound ID have substantial deficits in academic or conceptual skills. With intensive and persistent teaching and assistance, it is possible for a person with profound ID who do not have associated motor or sensory impairments, to develop some capacity for self-care ( 13 , 15 ). Some may be able to develop some capacity for nonverbal communication with use of augmentative communication methods. A person with profound ID may respond to familiar persons or caretakers with gestures and facial emotional cues ( 15 ). A person with profound ID needs constant supervision and is total care dependent in all aspects of daily living.

Undetermined severity

The standardized tests for assessment of intellectual and adaptive functioning are generally difficult to administer in very young children and the results may not be reliable or valid ( 10 , 15 ). This may also be the case in older children with associated other conditions that may pose barriers to effective testing, such as motor or sensory impairments and co-morbid medical or mental health disorders. When intellectual or adaptive functioning cannot be reliably assessed, based on standardized tests, a diagnosis of intellectual disability and its severity is made based on findings of clinical evaluation ( 8 , 10 , 15 , 16 ).

Etiological testing

The value of identifying a specific cause of intellectual disability, especially in case of mild intellectual disability, is subject to debate ( 30 - 33 ) ( Table 8 ). Because a biologic cause is more likely to be identified in severe intellectual disability, an etiological diagnosis should be pursued in all such cases. In many cases, certain disease specific clinical findings may indicate additional tests to identify a cause.

The initial diagnosis of intellectual disability is considered mainly based on clinical presentation. Key components of diagnostic evaluation are a complete medical history ( Table 9 ), general physical examination, dysmorphology examination and neurological examination ( 2 , 13 , 28 , 30 , 32 ). A diagnosis of intellectual disability requires an assessment of the person’s cognitive and adaptive abilities using individually administered standardized instruments by qualified professionals. Standardized test of intellectual and adaptive functioning should be appropriate for the population being tested. Standardized testing should be age appropriate, take into account the mental age of the child, be culturally sensitive, and accommodate for any motor, behavioral, or language variations ( 1 , 13 , 15 , 16 ). Hearing and vision should be assessed in all children as part of their intellectual disability evaluation ( 16 , 30 ).

Many conditions associated with severe to profound intellectual disability can be suspected based on their characteristic clinical features. On the other hand, in a large percentage of cases, especially in mild intellectual disability, no clinical characteristics suggestive of a specific condition are found. Laboratory tests for genetic disorders or abnormalities, laboratory tests for inborn errors of metabolism and neuroimaging studies are the main evaluation tools used to confirm or identify any specific etiology of intellectual disability. Such an extensive evaluation should be considered in consultation with appropriate professionals with expertise and experience in specific disciplines ( 8 , 16 , 30 - 37 ). The likelihood of identifying the etiology of intellectual disability by various laboratory tests and neuroimaging studies vary based on the severity of intellectual disability and the presence or absence of abnormal findings on clinical evaluation ( 1 , 16 , 29 - 34 ).

Newborn screening programs, where available and instituted, identify a number of inborn errors of metabolism. Early identification of treatable inborn errors of metabolism is important to improve long-term outcomes ( 34 , 35 ). Beyond the newborn period, in the absence of large scale implementation of newborn screening for inborn errors of metabolism, an evaluation for metabolic disorder is considered based on findings on clinical evaluation. Some of the finding that may suggest an inborn error of metabolism include a family history of IEM or unexplained neonatal death, consanguinity, failure to thrive, recurrent vomiting, seizures, lethargy, developmental regression, abnormal movements, hypotonia, facial dysmorphism, and enlarged spleen or liver ( 30 , 31 , 34 , 35 ). Specific tests of inborn errors of metabolism ( Table 10 ) should be considered in consultation with metabolic geneticist ( 30 , 31 , 34 , 35 ).

If a specific genetic diagnosis is suspected, work-up with appropriate genetic tests is considered in consultation with a geneticist. A chromosomal microarray test is recommended as a standard initial test in the evaluation of intellectual disability ( 30 ). If chromosomal microarray testing is not diagnostic, additional testing with exome or whole genome tests may be indicated ( 29 - 31 , 37 , 38 ). Fragile X testing should be included in all work-up as it is one of the most common genetic causes of intellectual disability ( 30 ). In females with moderate to severe intellectual disability, testing for Rett syndrome (methyl CpG-binding protein 2) is recommended ( 30 , 31 ). The overall yield of microarray analysis in the evaluation of intellectual disability is estimated to be 15%; whole exome sequencing is reported to add additional 30% and whole genome sequencing an additional 15% to overall yield ( 16 , 30 - 32 ).

Brain magnetic resonance imaging (MRI) scan is indicated in the presence of abnormal neurological history and examination findings. With abnormal neurological findings, 30–40% of MRI scans show abnormalities ( 16 , 30 , 32 ). In the absence of specific abnormal neurological findings, about 10% of scans show abnormal findings ( 16 , 30 , 32 ). However, in most cases it is difficulty to establish the significance of abnormal findings on neuroimaging as a cause of intellectual disability.

General comments on management

Improved prenatal care, improved access to maternal-child health, newborn screening for inborn errors of metabolism, and improved immunization rates, contribute to reduced rates of intellectual disability ( 16 , 27 , 28 ). The underlying cause, clinical manifestations, and cognitive and adaptive abilities vary widely between persons with intellectual disability. Such heterogeneity requires individualized considerations for treatment depending on individual needs; it is not within the scope of this review to describe in detail comprehensive management of all primary and associated conditions in persons who have intellectual disability. Notwithstanding the heterogeneity and need for individualized considerations, certain general principles of treatment for persons with ID provide a framework for their overall healthcare, which include the following ( 1 , 16 , 38 - 54 ):

• ❖ Treatment of medical complications;
• ❖ General preventive medical care;
• ❖ Treatment of co-morbid medical and mental health conditions;
• ❖ Treatment of challenging behaviors;
• ❖ Rehabilitation services;
• ❖ Educational support;
• ❖ Vocational training;
• ❖ Social support;
• ❖ Support for community based living;
• ❖ Support for appropriate level of employment;
• ❖ Transition to services from children focused to those more appropriate for adults;
• ❖ Guardianship, financial and legal considerations.

The optimum model of care for persons who have intellectual disability is an interdisciplinary care model in the setting of a clinical service or medical center. Primary care medical practitioner provides preventive and general medical care as well as facilitates and coordinates specialty consultative care and access to needed support services.

Like most countries in the world, in the United States, several federal and state laws, regulations and acts provide the regulatory basis for the development, implementation and funding of various services for persons with developmental disabilities ( 55 ). Most intervention and service programs are developed and implemented within or linked to formal educational systems at local or community level. Programs are structured to meet the age appropriate differences in the needs of children, adolescents and adults with intellectual disability. For young children under 3 years of age, early intervention services provided by local community agencies are used and the Individualized Family Service Plan is implemented for these families. For children and adolescents between 3 and 16 years of age an Individualized Education Plan is developed to provide appropriate educational interventions in the least restrictive environment possible along with related support services. Between 14 and 16 years of age, plan for transition of services to adult oriented settings is considered. The Individualized Transition Plan includes consideration of adult oriented services, vocational training, and needs for independent living. When the adolescent graduates from formal educational system as a young adult, continued support services are provided based on the development and implementation of an Individualized Habilitation Plan. The scope and intensity of interventions and support services vary depending upon the needs of the person and the severity of the intellectual disability.

Conclusions

Significant limitations in both intellectual and adaptive functioning with onset during developmental period are the core features of intellectual disability. The level of adaptive functioning and the intensity of supports needed are the main criteria used to categorize intellectual disability levels of severity. The level and limitations of both the intellectual and adaptive functioning of a person are based on a comprehensive clinical evaluation as well as individually administered standardized tests of intellectual and adaptive functioning. The use of measured IQ in determining severity of intellectual disability has limitations, especially at higher levels of IQ and therefore IQ alone is not considered sufficient criteria to determine whether a person should be considered to have an intellectual disability nor to determine its severity. Psychosocial and environmental factors are the main contributors of mild intellectual disability, which comprises 85% of persons who have intellectual disability. The clinical symptoms and signs of intellectual disability depend on the age at initial presentation and the severity of the deficits; the more severe the deficits, earlier the age at presentation. An evaluation for an underlying specific cause for intellectual disability is recommended and such work-up includes genetic testing, metabolic testing and neuroimaging. The main strategies for management of persons who have intellectual disability are general medical care, treatment of comorbid conditions, treatment of behavioral symptoms, special education, vocational training, and community-based supports. In most countries, there are specific laws and acts that protect the rights of persons with disabilities as well as provide a framework and variable level of funding for their care.

Acknowledgments

Funding: None.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

Conflicts of Interest: DRP serves as the unpaid Deputy Editor-in-Chief of TP and the unpaid Guest Editor of the focused issue “Neurodevelopmental and Neurobehavioral Disorders in Children”. TP . Vol 9, Supplement 1 (February 2020). The other authors have no conflicts of interest to declare.