Introduction

Indigenous Peoples in Canada are diverse and include First Nations, Inuit and Métis. Indigenous nations have many different languages, dialects, cultural practices/beliefs, and unique experiences; however, all share experiences with colonization. Historically and presently, Canada has a concerning record of failing to equitably care for and protect Indigenous children in the context of health research and otherwise.

Residential schools operated for decades with the intent of cultural and biological genocide.1,2 When Canada became a country in 1867, churches were already operating residential schools, and the last residential school did not close until the late 1990s.2 Indigenous children in residential schools were likely to experience physical, sexual, mental, emotional and spiritual abuse.2 While in residential schools, Indigenous children were exploited through nutrition experiments, as detailed by Mosby.3 Furthermore, many died of tuberculosis due to “poor ventilation and poor standards of care from school officials”.1 It is difficult to accurately estimate how many children died in residential schools due to poor record keeping.4 Canadian leaders were aware of the deaths occurring in residential schools; Dr Peter Bryce, chief medical officer for Indian Affairs at the time, raised alarm about the deaths of Indigenous children as a result of residential schools in his 1906 report.4 Poor physical, mental, and emotional health outcomes have been directly linked to residential schooling.5 Medical experimentation on Indigenous children also occurred in racially segregated Indian Hospitals as described by Dr Maureen Lux’s work, who also details the experimentation on Indigenous infants to test the experimental bacille Calmette–Guérin vaccine for tuberculosis.6 The Truth and Reconciliation Commission of Canada called on the Canadian government to acknowledge that the current state of Indigenous health in Canada is directly a result of previous Canadian government policies including residential schools.2

Due to the devastating impacts of colonial violence driving ongoing and historical trauma, Indigenous children are currently the highest represented group in Canada’s child welfare system, with 52% of children in foster care in Canada being Indigenous, despite accounting for only 7.7% of the child population.7,8 Yet, the Canadian Government has been found by the Canadian Human Rights Tribunal to be continuing to discriminate against First Nations children through underfunding Child and Family Services Federally, as compared to the Provincial provision of care for non-First Nations families.9

In response, many guidelines have been developed to guide the conduct of ethical health research involving Indigenous people in Canada. Briefly, some key milestones include the following: in 1996, The Royal Commission on Aboriginal Peoples (RCAP) issued the first research ethics statement.10 This statement, guidelines from the Canadian Institutes of Health Research (CIHR), and others informed the current Tri-Council policy statement on Indigenous health research and ethical conduct of research in Canada (TCPS2 Chapter 9), which was established in 2010.11,12 Other statements to guide researchers include the OCAP (ownership, control, access, possession) framework, OCAS (ownership, control, access and stewardship) principles, the First Nations Ethics Guide on Research and Aboriginal Traditional Knowledge, First Nations Regional Longitudinal Health Survey: Code of Research Ethics, and the Inuit Tapiriit Kanatami: National Inuit Strategy on Research.13,14,15,16,17,18 The only guideline which must be adhered to in most institutional Research Ethics Board (REB) review is the TCPS2. The previous CIHR guidelines were specific to Indigenous health research, rather than Indigenous research in general, leading to some scholars noting that having the CIHR guidelines specific to health research strengthened the ethics process beyond the more general TCPS2.19,20 Some Indigenous communities also have their own REBs with their own requirements.21,22,23

Despite these statements available for guidance there are a number of issues which still arise in research involving Indigenous children.24 A review of research from 2000–2010 concerning Inuit child, youth and maternal health found “the agenda for the past decade of Canadian Inuit health research has not mapped well to the realities of the population’s demographic, or to the prevailing health statuses, of these population groups”, and the emphasis in research did not match the priority issues identified by Inuit organizations.25 Furthermore, a 2014 review found a lack of community participation in research on neurodevelopmental disorders in Indigenous children in Canada, contravening ethical principles important to Indigenous health research such as OCAP (ownership, control, access, possession).14,26 In fact, one study identified in this review examining children for Fetal Alcohol Spectrum Disorder (FASD) had a community decline participation after consent had not been sought.26,27 Finally, a 2003 review of Indigenous health research in Canada found that Indigenous children were under-researched relative to their proportion of the population.28

At worst, pediatric Indigenous health research has physically harmed Indigenous children, and at best, it may be inconsistent with the realities of this population or misaligned with the priorities of their communities. Many scholars have highlighted the important role that Indigenous researchers play in ensuring that research is based in the priorities of Indigenous communities, interpreted in the context of lived experience, and that it serves to empower self-determination.24,29,30 This is even more critical when the research is focused on children who are incapable of consenting to research for themselves.

Objective

The primary objective of this research is to systematically review the existing pediatric Indigenous Health research in Canada to determine the prevalence of disclosed Indigenous community, organization and/or researcher participation. Secondary objectives include delineating the role those Indigenous participants played in the research, and identifying trends in participation over time, by type of research included, language, population, geographical location, age of children studied, and category of topic being studied.

Methods

Definitions and approach

In this paper we reference Canada for ease, while acknowledging that this land is known by other names in Indigenous Nations. However, given that Indigenous individuals may or may not identify as Canadian, the authors instead refer to them as residing in Canada. Indigenous in this paper references First Nations, Inuit and Métis, who are the original people residing in Canada. This research was a collaboration between Indigenous and non-Indigenous researchers (authors), with participation at every step of the research process. Further description of this partnership and self-locations statements from authors most significantly engaged in the research process are outlined in the author contributions section. This review was not registered, and a protocol paper was not prepared.

Data sources

The search was constructed with assistance from a librarian scientist. Embase and Medline were searched on April 15, 2017. On July 16, 2020 the search was updated, and a search of Cochrane Library was added. The searches of Medline and Embase were constructed based on search strings developed and validated by the University of Alberta (see Supplementary materials for search strategy).31 Articles were extracted into EndNote (version X9.3.3), and duplicates were removed automatically using the de-duplication function.

Study selection

Each abstract was independently reviewed by two separate reviewers (S.H., J.R., R.M.). Independent review of 9.5% full texts was completed in duplicate by S.H. and J.R., to permit comparison and consensus building with regard to agreement and exclusion criteria. The remaining full texts were then reviewed by either S.H. or J.R. independently. The French articles reviewed by one reviewer (S.M.) for content, and consensus about inclusion were discussed between S.M. and S.H.

Studies were excluded per the list outlined in Fig. 1. An exception to the exclusion criterion “includes participants >18 years of age” were studies about breastfeeding, qualitative studies including adults but about a pediatric issue, and studies describing children and/or youth but never mentioning an age range. Exclusion criterion “Indigenous children are an aside” identified papers where Indigeneity was not intentionally examined as a factor and no conclusions could be drawn about Indigenous children specifically.

Fig. 1: PRISMA 2020 flow diagram.
figure 1

The PRISMA flow diagram for the systematic review detailing the database searches, the number of abstracts screened, the full texts retrieved, the records excluded and reasons for exclusion.

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Data extraction

Of the articles included in full-text review, 7.8% of articles had data extracted in duplicate by S.H. and J.R. using a piloted data abstraction sheet, S.H. then independently completed the remaining data abstraction. Any issues were resolved by S.H. and J.R. through consensus.

Once articles were accepted for inclusion, Indigenous participation was recorded as either not disclosed, Indigenous researcher as author, Indigenous researchers as non-author, Indigenous organization, Indigenous community or any combination of these four types of participation. Categories were not mutually exclusive. Indigenous researcher as author was recorded if any author had an Indigenous community listed as their affiliation, or if the article explicitly stated ≥1 author was Indigenous or a member of a participating community or a citizen of an Indigenous nation. Researcher professional profiles were not sought to further discern if authors were Indigenous, as explained in the discussion section below. Indigenous researchers as non-author was recorded if the article mentioned any Indigenous or community researcher participation in any part of the research project but never specified if these team members were authors. Indigenous organization was recorded if the article included affiliations with Indigenous organizations or if the paper described participation of an Indigenous organization (including governance organizations). If reviewers were unsure of whether an organization was Indigenous-led, organization websites were checked for evidence, such as having Indigenous community members as the majority of the board of directors. Papers noting Band Council or other local government approval/support were recorded as Indigenous organization participation, not Indigenous community participation. Indigenous community was recorded if the article described community participation including, but not limited to, community advisory boards, receiving and incorporating guidance from elders, community engagement/consultation sessions, using a community-based participatory research methodology, or community ownership/leadership of the research project. Author affiliations with or approvals from community entities such as local health centers/nursing stations or schools in communities were not counted as any type of participation, since individuals employed by these entities are not necessarily from communities or representing community interests. Any issues or disagreements were resolved by S.H. and J.R. by consensus.

Other datapoints recorded included trends in participation over time, participation by type of research, language, population studied, geographic area, age of children studied, and category of topic. Year was recorded as <1980, 1980–1990, 1991–2000, or 2011–present. These ranges were chosen because of the introduction of RCAP in the late 1990s and the TCPS2 Chapter 9 in 2010, anticipating that changes in approaches to Indigenous health research may have followed. Type of research was recorded as primary, review, or other (e.g., analysis). Primary research included any study, quantitative, qualitative, or mixed methods, where primary data was collected from participants. Language was recorded as English or French. Population was recorded as either First Nations, Inuit, Métis, some combination of these groups, Indigenous people (First Nations, Inuit or Métis) and Indigenous children in other countries, or pan-Indigenous descriptor used (e.g., Indigenous, Aboriginal, Native, Indian, etc.). Indigenous nations were described too inconsistently for further breakdown and analysis (e.g., by language group, by European terms, or by Indigenous terms). Geography was recorded as province and/or territory if supplied, region if unspecified, or Canada (province or territory unspecified) with or without other countries. Papers prior to 1999 specifying Northwest Territories were checked for a description of region to confirm if authors were referring to present day Nunavut or Northwest Territories. If no region was specified, Northwest Territories was recorded. Age was recorded as infant <1, child 1–12, youth 13–18, any combination of these or age not stated, or adults (breastfeeding or qualitative about pediatric issue).

Topic was recorded as described in Table 1. Papers were assigned a category of topic based on the outcome of interest of the paper rather than exposure. If applicable, a subcategory was assigned. While topics were mutually exclusive, subcategories were not. For example, a paper including both suicide and addiction as outcomes of interest would be assigned to the neurology, behavior, mental health topic, but counted in both suicide and addiction subcategories. A subcategory assignment means a paper included that subcategory as an outcome of interest, not that the paper was necessarily exclusively about that outcome of interest. Subcategories were chosen based on the authors’ opinion of what may be considered especially stigmatized topics. The percentage of papers about a topic within each year category was additionally described.

Table 1 Topic categorization of papers.
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The template data collection form, including data extracted from included studies and used for all analyses, can be found in Supplementary materials. Analyses can be made available from the authors upon request.

Data analysis

Quantitative descriptive analyses, including proportions, percentages were calculated in Microsoft Excel (version 16.49, 2021) for all categories and subcategories of data extracted. The PRISMA checklist was utilized to ensure study adherence to these principles. No study risk of bias assessment, reporting bias assessment or assessment of certainty was performed on the included studies, as the information extracted was not based on the results of these studies, but rather how and when these studies were conducted. As such, a determination of quality was not of relevance to the measures of interest.

Results

Literature search and study selection

The search strategy identified 25,227 records. After removing 7475 duplicates, 17,752 records were title and abstract screened. From these, 15,696 were excluded and 14 were not retrieved leaving 2042 full-text articles to be assessed for eligibility. A total of 798 studies were included in the final review. The PRISMA flow diagram is shown in Fig. 1.

For title and abstract screening Cohen’s κ coefficient was 0.95 (S.H. and R.M.), 0.72 (S.H. and J.R.) and 0.72 (S.H. and E.L.) for each reviewing pair. For full-text screening Cohen’s κ coefficient was 0.86 (S.H. and J.R.).

Indigenous participation

Across all studies, 46.1% of papers mentioned at least one type of Indigenous participation. Table 2 shows the breakdown of types of participation being disclosed, with organization or government being the most common type (62.8%) and Indigenous researcher as author being the least common (10.9%).

Table 2 Type of Indigenous participation in pediatric Indigenous health research in Canada.
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Participation by year

There were 57 articles from prior to 1980, 80 from 1980 to 1990, 100 from 1991 to 2000, 241 from 2001 to 2010 and 320 from 2011 to the search date. The proportion of papers with disclosed Indigenous participation has been increasing over time as demonstrated in Fig. 2 from 5.3% prior to 1980 to 60% from 2011 to 2020.

Fig. 2: Indigenous participation in Canadian pediatric Indigenous health research by decade.
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The figure demonstrates gradual improvement of Indigenous participation in Indigenous child health research over time.

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Research type and language

Table 3 displays paper characteristics, as well as Indigenous participation by paper characteristic. The most common type of paper was primary research (87.7%), and most papers lacked participation across all types of research. Most studies were published in English (98.6%). Indigenous participation was noted in 46.6% of English studies and 9.1% of French studies.

Table 3 Participation by paper characteristics.
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Population

First Nations children exclusively were the most studied population (31.8%), followed by papers using only a pan-Indigenous term (e.g., Indigenous, Aboriginal, Native, Indian) (26.6%), then 21.8% about exclusively Inuit children. The least studied population was exclusively Métis children (0.1%), who were otherwise grouped with First Nations children (2.8%) or both First Nations and Inuit children (4.6%). Only 17.9% of papers using a pan-Indigenous term included Indigenous participation.

Geography

Geographical categories were not mutually exclusive. The most included areas of Canada were Ontario (23.9%), closely followed by Québec (23.1%), then Canada with province or territory unspecified with or without other countries (19.7%). Yukon (1.5%) and New Brunswick (1.6%) were the least included provinces and territories. Québec was the only jurisdiction where most papers disclosed participation (60.9%).

Age groups

Most papers included children in broad or unstated age groups (63.1%), followed next by the child (1–12 years) age group (15.2%). Studies that pertained to adults demonstrated the most Indigenous participation (83.3%) followed by the child age group (63.6%). The infant (<1 year) age group demonstrated the least participation (39.8%).

Research topics

The most researched outcome of interest topic was Nutrition, Lifestyle and Anthropometrics (22.9%) and the least researched topic was Injury (1.5%). Subcategory breakdowns for the neurological, behavior, mental health and nutrition, lifestyle and anthropometrics topics can be seen in Figs. 3 and 4, respectively. Shifting topic priorities are visualized in Fig. 5, demonstrating the percentage of papers about each topic in each time range. Prior to 1980, the most commonly researched topic was infectious disease (29.8%), decreasing steadily over time (8.1% after 2011), while papers examining nutrition, lifestyle and anthropometrics as well as neurological, behavior and mental health have been generally increasing over time.

Fig. 3: Neurological, behavior, mental health subcategory participation (n = 155).
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This figure demonstrates the percentage of participation of Indigenous researchers in pediatric Indigenous health research as it relates to addiction/substance use, FASD, suicide and the rest of the neurological, behaviour and mental health category. 

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Fig. 4: Nutrition, lifestyle and anthropometrics subcategory participation (n = 183).
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This figure demonstrates the percentage of participation of Indigenous researchers in pediatric Indigenous health research as it relates to obesity, type 2 diabetes and the rest of the nutrition, lifestyle and anthropometrics category. 

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Fig. 5: Distribution of health topics within decade categories.
figure 5

This figure demonstrates the distribution of Indigenous child health topics reported on during each decade time block, showing the shifting reporting priorities over time. Prior to 1980 the most reported topics were in the infectious disease category, and most recently were in the neurological, behaviour and mental health category, as well as the nutrition, lifestyle and anthropometrics category. 

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Papers about Environmental outcomes demonstrated the most Indigenous participation (75.0%), while papers about Genetics demonstrated the least (7.3%). The Fetal Alcohol Spectrum Disorder subcategory is displayed in Fig. 3 within the neurological, behavior, mental health topic, and it demonstrated the least participation within that category (33.3%). Other subcategories and papers with no assigned subcategory included similar levels of participation of ≥50.0%. Within the Nutrition, Lifestyle and Anthropometrics topic (Fig. 4), the Obesity subcategory included the most participation at 82.1%.

Discussion

Indigenous participation in pediatric Indigenous health research in Canada has been increasing over time, but has only reached 60% in the most recent decade, despite clear guidelines stating the necessity for involvement. Participation of Indigenous organizations/government was more common than participation of Indigenous researchers, as was Indigenous participation in the Québec region, in older rather than younger children studied, and in studies of environmental outcomes. While some of these findings may be due to feasibility, non-Indigenous researchers must involve Indigenous partners in all studies moving forward. Indigenous participation in every stage of pediatric research focused on Indigenous children is critical to ensure the research objectives are focused on community priorities, the conduct of the study is informed by historical context, the results are reviewed through the lens of lived experience and that the knowledge translation plan is culturally appropriate and feasible.

Indigenous health research conducted appropriately has potential to support reconciliation efforts in Canada, but to do so requires Indigenous participation and leadership to support self-determination of Indigenous communities.29,32,33 This work must be responsive to the Truth and Reconciliation Commission of Canada’s calls to action and enact the United Nations Declaration on the Rights of Indigenous Peoples framework for building relationships.2,29,34 Research has historically “furthered the colonial agenda”, and will likely continue to do so without prioritizing Indigenous self-determination.32 This review demonstrates that pediatric researchers in Canada must continue to build systems for accountability in order to achieve 100% Indigenous participation in all pediatric Indigenous health research, which is the only way forward.

Conducting pediatric Indigenous health research without Indigenous partners can have harmful effects. Harms can range from failing to provide the necessary context (colonization, racism, ongoing colonial policies) in order to understand a health disparity, to interpreting the findings in a blatantly white-supremacist and racist perspective.20 This review identified examples across the spectrum. A paper on ethnic disparities in oral health, published recently, commented on the poor oral health of the Indigenous children in the sample, without comment about potential root causes, such as the destruction of Traditional Indigenous food systems, lack of fluoridated water in many communities or the difficult to navigate non-insured health benefits.35,36 This could potentially lead readers to believe that this disparity is a purely physiological racial difference. On the other end of the spectrum, in a paper from 1967, discussing infant mortality, the author states “Interestingly, abuse of alcohol, usually regarded as a besetting sin of Indian parents, was reported in only…of the group of inefficient parents”.37 The “drunken Indian” stereotype this statement perpetuates harm, and stereotypes such as this relating to Indigenous adults being portrayed as irresponsible may have contributed to Indigenous children being wrongly apprehended during the Sixties Scoop or presently.38,39,40 These reports were just two chosen from a plethora of possible examples, but highlight the harms that could have been avoided, and the missed opportunities to identify actual solutions, through inclusion of Indigenous perspectives on the research team.

Researcher as author was the least commonly reported type of participation we identified. While author’s professional profiles were not consulted to delve more deeply into whether they publicly disclosed Indigenous identity, this finding may highlight the importance of researchers explicitly outlining Indigenous participation within publications themselves. As such, while some papers may have included Indigenous authors, they would have been missed if this was not specified within the paper. Authors may wish to consider explicitly stating what Indigenous participation was involved, or journals may consider requesting this information, so that readers may assess the ethics and trustworthiness of a paper’s conclusions related to Indigenous children. Concurrently, it is possible that this finding reflects a need for training more Indigenous scholars, and ensuring that existing Indigenous scholars are in leadership roles, and are well-supported in such roles.41 A few examples of such initiatives include CIHR’s Indigenous Mentorship Network Program, and McMaster University Indigenous Research Institute’s Indigenous Undergraduate Summer Research Scholar’s program.42,43

An incidental and concerning finding of this review was the inclusion of only one single paper about exclusively Métis children. Métis children were otherwise only researched when grouped with other Indigenous children, demonstrating a lack of Métis-specific pediatric research. Although many Indigenous populations share historical and present experiences of colonial violence and racism, Métis people have distinct cultural and historical experiences which may impact the health of Métis children differently than First Nations or Inuit children. Métis have distinct heritage, language, cultural practices, spiritual beliefs, and unique experiences of stress and marginalization.44,45 Lack of Métis-specific research is a well-described ongoing issue on the field of Indigenous health research.46

Québec was also the second most studied province/territory behind Ontario, which is in alignment with Ontario having the highest absolute population of Indigenous people.47 Interestingly, Quebec was the only province/territory where most papers included Indigenous participation. The stronger trend of participation in Québec may be due to established governance bodies in the province, such as the Cree Board of Health and Social Services of James Bay, Nunavik Regional Board of Health and Social Services, or Nunavik Nutrition and Health Committee, which were frequently involved in overseeing research. It may also be related to the establishment of longitudinal community lead pediatric research programs such as the Kahnawake Schools Diabetes Prevention Project. Building long-standing and trusting relationships between researchers and communities and/or organizations appears to empower communities to ask questions of relevance to their children’s health.

Papers about Indigenous infants less than one year of age disclosed the least participation of all reported age groups. We hypothesize that this may be because diseases of infancy and infant outcomes tend to be reported from a more epidemiologic perspective, and researchers may falsely believe it is less important to consider community perspectives in this context.

Paper topic analysis demonstrated some interesting trends. For example, the Canadian Pediatric Society highlights that unintentional injuries is the leading cause of death among Indigenous children and youth in Canada, yet injury was the least researched outcome topic.48 Potentially, research may be driven more by researcher interests than realities of prominent health concerns for these children and their communities. In addition, research concerning Environmental topics as outcomes demonstrated the most participation of any topic. It is possible that research questions in this area are more frequently motivated by Indigenous stakeholder concerns, and that this area of work may serve as an example for models of participation. Finally, emphasizing participation in subcategories of potentially stigmatizing areas of research is critical. For example, FASD, while a particularly stigmatized health issue, demonstrated little participation. Indigenous participation in research concerning stigmatized or stereotyped health conditions is especially integral to avoid misrepresentation of root causes of these health challenges.20

This is the first fulsome review of Indigenous participation in pediatric Indigenous health research in Canada. Approaching this work with a systematic and comprehensive search strategy, over 25 thousand records were identified, and almost 800 studies were included. Yet, one potential limitation is that only MEDLINE, Embase and Cochrane Library were searched. Cochrane, considered the global authority on systematic reviews in medicine, recommends searching these three databases as standard practice in medical research.49 There is a possibility of Indigenous health research being published in journals that are not indexed in these databases. Indigenous focused databases such as the Indigenous Studies Portal, and nursing/allied health focused databases such as CINHAL were intentionally excluded as we wished to examine literature most likely to be performed and read by clinician-researchers in medicine. Had these additional databases been included, we hypothesize that more papers may have more Indigenous participation, as Indigenous methodologies have often been considered less valid when held to Western standards of many medical journals.24 Future reviews may wish to intentionally include Indigenous research journals to gain a more inclusive view of participation in this published body of work.

Other limitations include the potential underestimation of true participation of Indigenous researchers as authors. As previously described, there is the possibility that some researchers were Indigenous, but if not disclosed within the paper itself, they were not recorded as such. We used a categorical approach to measure types of participation. While this ensured a clear analysis, participation is often nuanced, ranging from tokenistic to full and meaningful partnership. As most papers did not provide clear details of participation we were not able to abstract this information reliably, and as such could not include this in our analysis. Moving forward, we urge researchers to include author self-location statements and/or statements about engagement and roles within projects in order to better understand this nuance. Furthermore, we excluded papers that were not exclusively about children less than 18 years of age, apart from papers about breastfeeding, to manage the volume of studies included. Had papers with both children and adults been included, the results may have varied from those identified. With regard to the categorization of time, we chose to categorize years in straightforward decades based on the influence of RCAP and the TCPS2, recognizing that while these milestones had significant impacts there would be various other contributing factors influencing Indigenous participation at various points in time. Finally, the authors were not able to retrieve all full texts; however, with only 14 reports of 2056 unable to be retrieved, we do not feel this would have impacted our results significantly.

Conclusions

Indigenous participation in pediatric Indigenous health research in Canada has improved over time. Moving forward, non-Indigenous researchers need to support this imperative by ensuring fulsome and meaningful partnerships with Indigenous stakeholders and by giving up positions of power and privilege to make space for Indigenous scholar leaders. Researchers can work to increase Indigenous participation in their research by seeking relevant Indigenous organizations concerned with their topic of interest, communities or governing bodies that will be primary users of findings or who are the source of research participants, and/or by partnering with Indigenous scholars. Authors should disclose how Indigenous partners were involved in the work, so that readers may use this information to appraise the ethics of published work. Risks of absence of participation include perpetuating racist stereotypes, misrepresenting communities, and generating research that is not practically helpful or of interest to Indigenous children and their communities.