Transcript
Page 1: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

Setting global research priorities for developmentaldisabilities, including intellectual disabilities and autism

M. Tomlinson,1 M. T. Yasamy,2 E. Emerson,3 A. Officer,2 D. Richler4 & S. Saxena2

1 Department of Psychology, Stellenbosch University, Stellenbosch, South Africa2 World Health Organization, Geneva, Switzerland3 Centre for Disability and Policy, University of Sydney, Sydney, Australia and Centre for Disability Research, Lancaster Univer-sity, Lancaster, UK4 Joseph P. Kennedy Jr Foundation, Toronto, Canada

Abstract

Objectives The prevalence of intellectual disabilities(ID) has been estimated at 10.4/1000 worldwidewith higher rates among children and adolescents inlower income countries. The objective of this paperis to address research priorities for development dis-abilities, notably ID and autism, at the global leveland to propose the more rational use of scarcefunds in addressing this under-investigated area.Methods An expert group was identified andinvited to systematically list and score researchquestions. They applied the priority setting method-ology of the Child Health and Nutrition ResearchInitiative (CHNRI) to generate research questionsand to evaluate them using a set of five criteria:answerability, feasibility, applicability and impact,support within the context and equity.Findings The results of this process clearly indi-cated that the important priorities for futureresearch related to the need for effective and effi-cient approaches to early intervention, empower-ment of families supporting a person withdevelopmental disability and to address preventablecauses of poor health in people with ID and autism.Conclusions For the public health and othersystems to become more effective in delivering

appropriate support to persons with developmentaldisabilities, greater (and more targeted) investmentin research is required to produce evidence of whatworks consistent with international human rightsstandards.

Keywords autism, developmental disabilities,global research priority setting, intellectual disabil-ities, low and middle income countries

Introduction

In recent years, WHO has expanded working in thearea of intellectual disabilities (ID)1 and autismthrough extending partnerships at the global levelwith key international stakeholders. Mapping ofpolicy and service provision for people with ID hasbeen completed in 147 countries (WHO 2007a) andboth ID and autism have been included in WHO’sflagship mental health Gap Action Programme(mhGAP) (WHO 2008, 2010). However, the moreWHO has been determined to work in this area, themore the dearth of scientific knowledge that hasrelevance to the situation of people with ID andautism in low and middle income countries (LMIC)has become obvious (Emerson et al. 2007, 2008,

Correspondence: Professor Mark Tomlinson, Department ofPsychology, Stellenbosch University, Private Bag X1, Matieland,Stellenbosch, 7602, South Africa (e-mail: [email protected]).

1 ‘Intellectual disabilities’ is synonymous with the outdated and tomany offensive ICD-10 term ‘mental retardation’.

Journal of Intellectual Disability Research doi: 10.1111/jir.12106

volume 58 part 12 pp 1121–1130 december 20141121

bs_bs_banner

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 2: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

2012; WHO 2011). One of WHO’s core functions isto shape the research agenda and stimulate the gen-eration, translation and dissemination of knowledge(WHO 2007b). Developing a global researchagenda for ID and autism could help to guideresearch activities towards priority areas.

Existing epidemiological research suggests thatthese developmental disabilities contribute to a highamount of disability worldwide. The prevalence ofID has been estimated at 10.4/1000 worldwide withhigher rates among children and adolescents and inLMIC (Maulik et al. 2011). The median prevalencefor pervasive developmental disorders (whichinclude autism and Asperger’s syndrome) has beenestimated at 6.2/1000 (Elsabbagh et al. 2012) withno difference by geographic region. We need toknow more about the prevalence, magnitude,impact, causes and correlates of these disabilities.

It is clear that people with disabilities, includingpeople with ID and autism, face extensive andwidespread discrimination and violations of theirhuman rights and fundamental freedoms (UnitedNations 2003, 2006, 2011; World HealthOrganization and the World Bank 2011). They also,partly as a result of these violations, are vulnerableto experiencing extensive health inequalities(Ouellette-Kuntz 2005). According to WHO’s Atlasof global resources for persons with ID (WorldHealth Organization 2007); most people with ID donot receive the services and supports they require.The global gap in services is accompanied and, inpart, attributable to the absence of relevant scien-tific knowledge. At present, we simply do not knowwhat the most cost-effective services are and whichservices models are applicable in different contexts.

Research can make a significant contribution toimproving the well-being of people with disabilitiesand to reducing the health inequalities faced by‘vulnerable’ groups (World Health Organization2008, 2011; World Health Organization and theWorld Bank 2011). However, most existing scien-tific knowledge is of ‘indirect relevance’ to peopleliving with developmental disabilities in LMIC andhas mostly been conducted in English speaking highincome countries (Emerson et al. 2007) a pattern ofinequity that is repeated across many areas of healthresearch (Global Forum for Health Research 2004).Much of the available knowledge is also insensitiveto issues of gender, culture and ethnicity (Yasamy

et al. 2011). There is a dual need for increasedresearch in LMIC countries, as well as more trans-lational and implementation research to establishthe extent to which the existing knowledge basefrom high income countries has relevance forLMIC. It is safe to say that current research in richcountries is not directly contributing to improvingthe situation in LMIC. Establishing global researchpriorities will help identify the appropriate balancebetween basic science, clinical research and publichealth research; a dire need in the area of ID andautism.

Methodology

The Child Health and Nutrition Research Initiative(CHNRI) methodology for setting priorities inhealth research investments was used to identifyglobal research priorities for ID and autism (Rudanet al. 2008). This methodology was adoptedbecause: (a) it is a carefully developed and docu-mented conceptual framework available in thepublic domain; (b) it has demonstrated usefulnessin several previous exercises (including disabilityand mental health); and (c) it has expanding use bypolicy makers, large donors and internationalorganisations (Tomlinson et al. 2007, 2009a,b;Rudan et al. 2010). The method also has the advan-tage of supporting the participation of a range ofstakeholders including those concerned with policy,with technical aspects of research, and with socialand community issues. The CHNRI process isdescribed below.1 The first step in the process was to gather a coregroup who oversaw the process. The authors of thispaper formed the core group and provided technicalinput.2 The core group defined the context in order toensure that recruitment of experts to participate inthe priority setting exercise was representative(equitable). The following principles guided theprocess.

a. Geographical focus: that there was adequaterepresentation from different regions of the worldincluding high income as well as LMIC.b. Gender representation: that gender equity wasan important considerationc. Researcher/NGO interface: that the processwas not dominated by academics/researchers and

1122Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 3: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

that there was representation from consumerorganisations, civil society, NGOs, community-based organisations, human rights groups andorganisations of persons with disabilities and theirfamilies.d. Problem focus: the exercise focused both onresearch related to mental disorders and psycho-social wellbeing/human rights of people withintellectual and developmental disabilities.

3 There are many possible criteria against whichresearch options can be judged. The CHNRI meth-odology recommends the use of a limited number inorder to ensure feasibility. Five criteria were identi-fied by the core group based on a review of generalresearch domains (see Box 1 for an outline of thespecific questions for each criteria). The criteriawere: (a) answerability; (b) feasibility; (c) applicabil-ity and impact; (d) support within the context; and(e) equity.4 Library searches and snowball sampling was usedto identify experts known for their work on develop-ment disabilities and representatives from civilsociety and service user organisations to indepen-dently generate research questions which theybelieved were priorities. Eligibility to participate towas based on meeting one of three criteria: (a) pub-lication in the scientific literature on ID or autism;(b) being a consumer of ID or autism services; (c)being a member of an organisation for persons withID or autism. All participants fulfilled at least oneof these criteria. This group of experts was thenasked to generate no more than five research ques-tions that they believed were a priority. This activitygenerated an initial list of 266 research questions.5 Questions were collated into a composite set ofquestions by eliminating overlap. The collationprocess was undertaken by the core group. Thisyielded 69 questions. Questions were grouped into anumber of sections in order to ease the scoringprocess. These were Basic Science and Epidemiol-ogy (11 questions); Prevention, Promotion, SocialOrganisation, Public Attitudes & Beliefs (14 ques-tions); Services & Supports – General (18 ques-tions); Services & Supports – Parents and Families(5 questions); Services & Supports – Identification/Screening & Early Intervention (5 questions);Services & Supports – Education (5 questions);Services & Supports – Access to Health (6 ques-tions); Services & Supports – Ageing (2 questions);

Services & Supports – Interventions (3 questions).We used the term ‘developmental disabilities’ tocover both ID and autism or other pervasive devel-opmental disabilities. Participants were asked inde-pendently to rate each of the 69 questions on eachof the five criteria.6 Scores for competing research options were gath-ered and computed. In this way, the proposedoptions received a score on each of the five criteriaranging from 0–100%. They represent a measure ofthe collective opinion of the experts scoring inde-pendently. Surowiecki has shown how once each inindividual in a rating process gets a right to expressjudgement that is treated equally as the judgementof any other individual, then the personal biasesthat those individuals bring into the process tend tocancel and dilute each other regardless who the par-ticipants are. What is left is information based onaccumulated knowledge, lifetime experience andcommon sense of those who took part (Surowiecki2004).

Results

The library search and snowballing approachyielded a list of 138 possible members of the expertgroup, of these 72 (52%) agreed to participate. Theexpert group was geographically diverse, with 23%from Asia, 17% from Europe, 21% from NorthAmerica, 12% from the Middle East, 9% fromAfrica, 9% from Latin America, 6% consideringthemselves international (WHO or internationalNGOs), and 4% from Australasia. Sixty-three par-ticipating members of the expert group contributedpotential research questions.

The final list of 69 research questions was sent tothe original group of 72 experts. Scoring took placeover a three-month period and was performed by 49

members; 68% of the original working group. Theexperts who completed scoring had almost a similarprofile to that of the original larger group. Theywere equally geographically diverse, with 18% fromAsia, 16% from Europe, 16% from North America,8% from the Middle East, 12% from Africa, 10%from Latin America, 12% considering themselvesinternational (WHO or international NGOs), and6% from Australasia. Experts were also asked todescribe their area of expertise – in some cases they

1123Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 4: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

Box 1 Questions and scoring instructions

Scoring instructions – Firstly, always keep in mind the context. It is not possible to predict all outcomes of healthresearch investments, and therefore the priorities should be set within a specific context, to influence a specificpolicy. Here, the context will be defined by space, time, population of interest. The space will be global, the timewill be the next 5–10 years, and the area of interest on setting priorities for developmental disabilities includingintellectual disabilities and developmental disabilities (including autism). Possible answers: Yes = 1; No = 0;Informed but undecided answer: 0.5; Not sufficiently informed: blank

CRITERION 1: ANSWERABILITY – Likelihood that the research question would be answerable and generatenew knowledge

1 Would you say the research question is clear and has well defined endpoints?2 Would you say that a study can be designed to answer the research question and to reach the pro-

posed endpoints of the research?3 Would you say that the research question would target a critical gap in knowledge, rather than just

advance an already existing level of knowledge?

CRITERION 2: FEASIBILITY – Is the research potentially doable in the majority of countries in the world

1 Taking into account the level of difficulty with undertaking the research (e.g. the complexity of theresearch, the infrastructure required and human factors involved), would it be possible to undertakethis research in most countries?

CRITERION 3: APPLICABILITY AND IMPACT – Likelihood that the knowledge generated through theproposed research would be implemented and have an impact on policy and practice

1 Taking into account the financial resources available to implement the research, would you say thatthe implementation of the end-points of the research would be affordable within the context ofinterest?

2 If basic science, is it likely that it will soon lead to significant improvement in practice?3 Based on the best available evidence and knowledge, do you believe that interventions and pro-

grammes developed or improved through the proposed research would be effective?

CRITERION 4: SUPPORT WITHIN THE CONTEXT – Likelihood that the proposed research would, inthe context of current policy and funding priorities, be likely to attract the necessary funding support.

1 Will the relevant public sectors such as health, social welfare and education or influential NGOs besupportive of the research in general?

2 Do you think the proposed research is likely to be prioritized for funding by agencies that fundresearch?

CRITERION 5: EQUITY – Likelihood that the proposed research would address underprivileged people,or provide solutions that would benefit people among all social strata equally.

1 Would you suggest that the proposed research would address primarily people that are among thepoorest globally?

2 Would you say that the proposed research would provide solutions that would improve the quality oflives of persons regardless of their social status?

1124Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 5: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

listed more than one. Primary areas of expertisewere academic researcher (37%); health or alliedhealth care professional (6%); basic scientist (4%);users (2%) (we define a user as a consumer ofdevelopmental disability, autism or ID services);non-governmental organisation (29%); rehabilita-tion (4%); public health (14%) and education orspecial education (8%). In addition, 12% orrespondents also stated that they were either theparent or immediate family member of a user.

The scoring of the 69 research questions resultedin the ranking of research topics based on the per-ceived likelihood that they would be answerable,feasible, have applicability and impact, obtainsupport or have an impact on equity. An overallresearch priority score (RPS) was calculated as themean of each criteria score. Mean scores on theseparate domains across the 69 items were all highlyinter-correlated (Table 1), while Table 2 shows themean RPS scores by research domain.

Following Collins et al. (2011) we grouped prior-ity areas into five goals: advance identification,screening and early intervention; improve aware-ness, prevention, and promotive interventions; iden-tify causes, prevalence, biomarkers, and risk andprotective factors; transform health system toimprove access and build human resource capacity;and improve support to parents and families. Illus-trative research questions for each goal are pre-sented in Table 3 (see Appendix S1 for the fullranking of all 69 research questions).

The goal of advancing early identification, screen-ing and early intervention achieved particularly highscores across the expert group. In this regard, therewas a focus on how health systems can improve inearly detection of developmental disabilities and

how to operationally define and identify people withdevelopmental disabilities. Linked to this was ser-vices and support related to identification, screeningand early intervention and included questions onthe availability, cost and efficacy of early interven-tion and rehabilitation programs for children withdevelopmental disabilities. In terms of improvingawareness and prevention (Goal B) prominent areasof focus were on the rights to health and educationof children together with a strong emphasis onstigmatisation, and the attitudes of both primaryhealth care workers as well as the broader societalattitudes towards the care of people with develop-mental disabilities and how these vary acrossregions and countries. Training emerged as a strongtheme – whether of primary health workers toreduce discrimination or establish how to train non-specialist health workers (community health workers

Table 1 Correlation (Pearson) between means domain scores and total score across items

FeasibilityApplicabilityand impact Support Equity Total

Answerability 0.759*** 0.653*** 0.807*** 0.618*** 0.877***Feasibility 0.742*** 0.638*** 0.647*** 0.873***Applicability and impact 0.659*** 0.777*** 0.874***Support 0.686*** 0.870***Equity 0.861***

*** P < 0.001.

Table 2 Mean RPS by research domain

DomainMeanRPS

Services and support: Identification, screening andearly intervention

71.6

Services and support: Access to health 62.0Services and support: Parents and families 60.9Prevention, promotion, social organisation, public

attitudes and belief60.9

Services and support: General 56.5Services and support: Ageing 53.8Services and support: Interventions 53.7Services and support: Education 53.5Basic science and epidemiology 53.0

RPS, research priority score.

1125Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 6: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

Table 3 Priority research areas

Illustrative research questions

Goal AAdvance identification,

screening and earlyintervention

• How can health systems improve in early detection (during infancy and early childhood) of developmentaldisabilities in low and middle income countries?

• What is the availability, cost and efficacy of early intervention and rehabilitation programs for childrenwith developmental disabilities in low and low-middle income countries?

• What is the effectiveness and efficiency of family-mediated early intervention on the future health andwell-being of children with developmental disabilities in low resource settings?

• How can we best operationally define and identify (screen for) people with developmental disabilities?• What are the most important priorities for investigation/assessment for children with developmental

disabilities in low resource rural settings?Goal BImprove awareness,

prevention and promotiveinterventions

• What are the most effective ways of promoting the rights to health and education of children withdevelopmental disabilities in low and low-middle income countries?

• What is the impact of global initiatives in disability (e.g. UNCRPD) on policies, laws and servicesfor people with developmental disabilities?

• How do public attitudes to developmental disabilities and the care of people with developmentaldisabilities vary across cultural and language groups and across types of developmental disabilities?

• What are the common experiences and attitudes of primary healthcare workers towards people withdevelopmental disabilities?

• What are the nature and forms of stigmatisation in different cultures and contexts (low, middle, highincome countries; rural/urban)?

Goal CIdentify causes, prevalence,

biomarkers, and risk andprotective factors

• What are the most common preventable causes of poor health among people with developmentaldisabilities?

• What are the most important nutritional needs of children with developmental disabilities in low resourcerural settings?

• What are the most common preventable causes of developmental disabilities (e.g., neurotoxins,undernutrition, infections, poverty, poor education)? How do these vary across regions and countries (andwithin countries)?

• What are the most common preventable causes of the social exclusion and reduced quality of life amongpeople with developmental disabilities? How do these vary across regions and countries (and withincountries)?

Goal DTransform health system to

improve access and buildhuman resource capacity

• How best can non-specialists be trained and supervised to work with people with developmentaldisabilities?

• What are the important preventive health measures and barriers to health care for people withintellectual disability and how successfully are these implemented?

• What are the most efficient ways of training health care workers to reduce the discriminationfaced by people with developmental disabilities in health care systems?

• How can assistive devices (computer software, hand-held devices, touch screen computers)support people with developmental disabilities to learn and communicate?

• What can be learned from a systematic study of public health systems and health care policyacross countries with regard to what has worked and what hasn’t with respect to prevention andintervention for developmental disabilities?

Goal EImprove support to parents

and families

• What are the most efficient ways of supporting and empowering parents/families of people withdevelopmental disabilities (in specific social and cultural contexts)?

• How can parents/families be most efficiently supported to provide a ‘healthy start’ in life for youngchildren with or at high risk of developmental disabilities in low resource settings?

• What are the most efficient interventions to support parents/families of children with disabilities tomanage their own stress?

• How does ‘family context’ interact with the behavioural challenges posed by children with developmentaldisabilities to produce either positive or negative impact on parents’ mental health and well-being?

• How do the dynamic mechanisms in early and ongoing family processes that affect child andparent outcome vary across culture and ethnic identity?

1126Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 7: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

for instance) to work with people with developmen-tal disabilities. Providing support to the parents andfamilies of children and people with developmentaldisabilities emerged as a particularly strong themewith a number of research questions relating to ser-vices and support research such as the most efficientways of supporting and empowering parents/familiesof people with developmental disabilities and tohow they can be most efficiently supported toprovide a ‘healthy start’ in life for young childrenwith or at high risk of developmental disabilities.Finally, the priorities recognise that there is muchstill to be learned in terms of identifying causes,prevalence studies and determining risk and protec-tive factors. Examples of this included questionsabout the most common preventable causes of poorhealth among people with developmental disabilitiesand how these vary across regions and countries(and within countries) as well as questions concern-ing the most common preventable causes of poorhealth, social exclusion and nutritional deficienciesamongst children and people with developmentaldisabilities.

Discussion

There is a dearth of scientific knowledge, other thanthat derived from basic science that has relevance tothe situation of people with developmental disabil-ities in LMIC (Emerson et al. 2007, 2008, 2012;World Health Organization and the World Bank2011). We applied a proven method (CHNRI meth-odology) to identify global research priorities in thisarea (Tomlinson et al. 2007, 2009a,b; Rudan et al.2008, 2010). The results of this process clearly indi-cated that the crucial priorities for future researchrelated to the need for effective and efficientapproaches to early intervention and to address pre-ventable causes of poor health among children withID and/or autism. Another goal of research in thisarea was improving support to parents and families,training of health workers and non-specialists, and astrong focus on improving awareness. The mainstrengths of this priority setting exercise were:(1) the use of a proven methodology (Tomlinsonet al. 2007, 2009a,b; Rudan et al. 2008, 2010);(2) the involvement of a significant number ofexperts with good gender balance and geographical

spread. The main limitations of the approach relateto validity of the CHNRI approach and potentialsampling biases. While 63 experts participated gen-erating the ‘research investment options’ that wererated, it is clearly impossible within such an exerciseto attain comprehensive coverage of all possibleresearch questions. As such, both the questions gen-erated and the ratings allocated are likely to reflectany biases in the identification, sampling and par-ticipation of experts. The nature of the CHNRImethodology as applied in this instance would havelimited the participation of experts who were notfluent in English and experts who have difficulty inmanipulating complex information. These biaseswould be expected to have a particular impact inlimiting the direct participation of ‘experts byexperience’ (e.g. people with ID and autism, manyfamily members). Nevertheless, the project was suc-cessful in eliciting research questions from 63

experts and ratings from 49 experts from aroundthe globe. The number of participants and the pro-tection against potential bias provided by theCHNRI approach (e.g. by limiting interactionbetween participants) does reduce the probabilitythat a similar group of experts would producematerially different results. As such, we believe thatthis research priority setting exercise provides animportant contribution to establishing a globalresearch agenda for ID and autism.

Finally, it is important to note that the prioritysetting exercise did not address the appropriatenessof particular research methodologies or paradigms.As has been argued elsewhere, there is clearly aneed to seek a balance between investment in basicscience, clinical research, strategic research andaction research (Yasamy et al. 2011). Agencies suchas WHO also support countries in developing rel-evant policies and plans and providing technicalassistance for implementing them. The presentexercise has provided grounds for the sound orien-tation of further research and service developmentparticularly in poor resource settings. Finally, whilethe focus of this exercise was on LMIC, the aim isto provide guidance in setting research prioritiesglobally. Different countries will certainly havedifferent needs and we are not arguing for thesame research questions to be employed withineach country. We are also not suggesting thatresearch conducted in rich countries has no

1127Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 8: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

relevance (through careful translational work) toLMIC.

In the area of ID and autism, there is also a par-ticular need to increase investment in participatoryor emancipatory research (i.e. research that is eitherco-produced with or controlled by organisations runby and for people with ID and/or autism). Thereasons for this are twofold. First, increasing invest-ment in participatory or emancipatory research isconsistent with obligations under the UN Conven-tion on the Rights of Persons with Disabilities forStates Parties and international organisations topromote the inclusion of people with disabilities inall aspects of society. Second, a growing body ofparticipatory or emancipatory research has docu-mented the particular benefits that ‘insider knowl-edge’ or ‘experts by experience’ may bring to thedevelopment of research questions, methods andinterpretation and dissemination of results(Ramcharan et al. 2004; Walmsley 2010; WorldHealth Organization and the World Bank 2011).

What was perhaps most striking about the resultsof the priority setting exercise was the emphasisplaced on research questions relating to providingmore effective and efficient support to children(specifically reflected in 9 of the top 25 priorities)and the importance of understanding/addressing thesocial exclusion, discrimination and disadvantagefaced by people with ID and/or autism (specificallyreflected in 11 of the top 25 priorities). In contrast,only three questions in the top 25 related to basicscience and epidemiology (one of these being theidentification of preventable causes of poor health).The prioritisation of these issues is consistent withtwo general trends in our understanding of disabilityand health. First, disability is increasingly beingconceptualised as a human rights issue in thatpeople with disabilities are at significantly increasedrisk of: (1) experiencing inequalities due to discrimi-nation in health care, employment, education, andlegal systems; (2) being subject to violations of theirdignity; and (3) being denied autonomy (e.g. bybeing subjected to involuntary sterilisation) (WorldHealth Organization and the World Bank 2011).Second, the growing emphasis within health policyover that last decade on addressing global, regionaland national inequalities in health has drawn atten-tion to the general importance of addressing thebroader social determinants of health associated

with inequalities in access to power, wealth, educa-tion and employment (Marmot and on behalf of theCommission on Social Determinants of Health2007; World Health Organization 2008, 2011).Within this context, a growing body of evidence hashighlighted the importance of early child develop-ment in setting the scene for the emergence ofhealth inequalities in later life (Irwin et al. 2007;Shonkoff 2010; Walker et al. 2011). Taken together,these trends stress the importance of taking abroader public health approach to understandingand addressing the health and well-being of peoplewith ID and/or autism; a perspective that is notablylacking in current research investment. Interestingly,health system research was found to be thecommon priority across all previous globalprioritisation exercises for mental health research(Yasamy et al. 2011). Interestingly, while many ofthe research priorities relate to LMIC many of theidentified priorities apply equally to higher incomecountries.

Evidence generated by research is clearly neces-sary but not sufficient for a real change on theground. For the public health and other systems tobecome more effective in delivering appropriatesupport to persons with developmental disabilities,existing health care and programmes need to bebased on evidence of what works consistent withinternational human rights standards, accompaniedby appropriate resource allocation (e.g. financial,training), and based on principles of accountability.

Acknowledgements

We would like to acknowledge the considerabletime and effort that our expert group committed tothis priority setting exercise.

Conflict of interest

MTY, AO and SS are World Health Organization(WHO) employees. WHO commissioned and sup-ported this work. However, the views expressed inthis article do not necessarily represent the deci-sions, policy, or views of WHO. We appreciate theShirley Foundation, Autistica represented by EileenHopkins and Autism Speaks represented by AndyShih, for supporting this work. MT is supported by

1128Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 9: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

grants from the National Institute on Drug Abuse(USA), the National Research Foundation (NRF)South Africa, and the Department for InternationalDevelopment (DFID), United Kingdom.

References

Collins P. Y., Patel V., Joestl S. S., March D., Insel T. R.,Daar A. S. et al. (2011) Grand challenges in globalmental health. Nature 475, 27–30.

Elsabbagh M., Divan G., Koh Y. J., Kim Y. S., KauchaliS., Marcin C. et al. (2012) Global prevalence of autismand other pervasive developmental disorders. AutismResearch 5, 160–79.

Emerson E., Fujiura G. T. & Hatton C. (2007) Interna-tional perspectives. In: Handbook on Developmental Dis-abilities (eds S. L. Odom, R. H. Horner, M. Snell & J.Blacher), pp. 593–613. Guilford Press, New York.

Emerson E., Mcconkey R., Walsh P. & Felce D. (2008)Intellectual disability in a global context. Journal ofPolicy and Practice in Intellectual Disability 5, 79–80.

Emerson E., Yasamy M. T. & Saxena S. (2012) Scalingup support for children with developmental disabilitiesin low- and middle-income countries. Journal of AppliedResearch in Intellectual Disabilities 25, 96–8.

Global Forum for Health Research (2004) The 10/90

Report on Health Research. Global Forum for HealthResearch, Geneva.

Irwin L. G., Siddiqi A. & Hertzman C. (2007) Early ChildDevelopment : A Powerful Equalizer. World HealthOrganisation, Geneva.

Marmot M. & on behalf of the Commission on SocialDeterminants of Health (2007) Achieving health equity:from root causes to fair outcomes. Lancet 370.

Maulik P. K., Mascarenhas M. N., Mathers C. D., DuaT. & Saxena S. (2011) Prevalence of intellectual disabil-ity: a meta-analysis of population-based studies. Researchin Developmental Disabilities 32, 419–36.

Ouellette-Kuntz H. (2005) Understanding health dispar-ities and inequities faced by individuals with intellectualdisabilities. Journal of Applied Research in Intellectual Dis-abilities 18, 113–21.

Ramcharan P., Grant G. & Flynn M. (2004)Emancipatory and participatory research: how far havewe come? In: The International Handbook of AppliedResearch in Intellectual Disabilities (eds E. Emerson, C.Hatton, T. Thompson & T. Parmenter), pp. 83–111.Wiley, New York & Chichester.

Rudan I., Chopra M., Kapiriri L., Gibson J.,Ann Lansang M., Carneiro I. et al. (2008) Setting prior-ities in global child health research investments: univer-sal challenges and conceptual framework. CroatianMedical Journal 49, 307–17.

Rudan I., Kapiriri L., Tomlinson M., Balliet M., CohenB. & Chopra M. (2010) Evidence-based priority settingfor health care and research: tools to support policy inmaternal, neonatal, and child health in Africa. PLoSMedicine 7, e1000308.

Shonkoff J. P. (2010) Building a new biodevelopmentalframework to guide the future of early childhood policy.Child Development 81, 357–67.

Surowiecki J. (2004) The Wisdom of Crowds: Why the ManyAre Smarter Than the Few and How Collective WisdomShapes Business, Economies, Societies and Nations. AnchorBooks, New York.

Tomlinson M., Chopra M., Sanders D., Bradshaw D.,Hendricks M., Greenfield D. et al. (2007) Setting prior-ities in child health research investments for SouthAfrica. PLoS Medicine 4, e259.

Tomlinson M., Rudan I., Saxena S., Swartz L., Tsai A. C.& Patel V. (2009a) Setting priorities for global mentalhealth research. Bulletin of the World Health Organization87, 438–46.

Tomlinson M., Swartz L., Officer A., Chan K. Y., RudanI. & Saxena S. (2009b) Research priorities for health ofpeople with disabilities: an expert opinion exercise.Lancet 374, 1857–62.

United Nations (2003) Standard rules on the equalization ofopportunities of persons with disabilities. United Nations,New York.

United Nations (2006) Convention on the Rights of Personswith Disabilities. United Nations, New York.

United Nations (2011) Status of the Convention on theRights of the Child: Report of the Secretary-General. UnitedNations, New York.

Walker S. P., Wachs T. D., Grantham-Mcgregor S., BlackM. M., Nelson C. A., Huff Man S. L. et al. (2011)Inequality in early childhood: risk and protective factorsfor early child development. Lancet 378, 1325–38.

Walmsley J. (2010) Research and emancipation: prospectsand problems. In: Learning Disasbility: A Life CycleApproach. (eds G. Grant & P. Ramcharan), pp. 489–501. Open University Press, Maidenhead.

WHO (2007a) Atlas: Global Resources for Persons with Intel-lectual Disabilities. World Health Organization, Geneva.

WHO (2007b) Basic documents, 46th edn. World HealthOrganization, Geneva.

WHO (2008) mhGAP: Mental Health Gap Action Pro-gramme – Scaling up care for mental, neurological, and sub-stance use disorders. World Health Organization, Geneva.

WHO (2010) mhGAP Intervention Guide for mental, neuro-logical and substance use disorders in non-specialized healthsettings. World Health Organization, Geneva.

WHO (2011) World Report on Disability. World HealthOrganization, Geneva.

1129Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd

Page 10: Setting global research priorities for developmental disabilities, including intellectual disabilities and autism

World Health Organization (2007) Atlas: Global Resourcesfor Persons with INtellectual Disabilities. World HealthOrganisation, Geneva.

World Health Organization (2008) Closing the gap in a gen-eration: Health equity through action on the social determi-nants of health. Final report of the Commission on the SocialDeterminants of Health. World Health Organization,Geneva.

World Health Organization (2011) Rio Political Declarationon Social Determinants of Health. World Health Organi-zation, Geneva.

World Health Organization and the World Bank (2011)World Report on Disability. World Health Organization,Geneva.

Yasamy M. T., Maulik P. K., Tomlinson M., Lund C.,Van Ommeren M. & Saxena S. (2011) Responsible gov-

ernance for mental health research in low resourcecountries. PLoS Medicine 8, e1001126.

Accepted 15 November 2013

Supporting information

Additional Supporting Information may be found inthe online version of this article at the publisher’sweb-site:

Appendix S1 All Research question ranked by TotalRPS.

1130Journal of Intellectual Disability Research volume 58 part 12 december 2014

M. Tomlinson et al. • Setting research priorities for developmental disabilities

© 2014 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and

John Wiley & Sons Ltd


Recommended