FormalPara Key Points

  • The Indigenous Australian Human Papillomavirus (HPV) Cohort Study examines the prevalence of oral HPV infection among Aboriginal and Torres Strait Islander people in South Australia. To our knowledge, this is the largest Indigenous HPV cohort study in the world to track oral HPV infection and monitor the early stages of oropharyngeal squamous cell carcinoma (OPSCC).

  • The study collected baseline data from 1011 participants. Follow-ups have been conducted at 12, 24, and 48 months and will also occur at 60 and 72 months.

  • Strong community engagement, employing Aboriginal staff, and yarning during the data collection have resulted in excellent recruitment and retention rates in this study.

Aboriginal and Torres Strait Islander peoples in Australia have a higher incidence of, and lower five-year survival rates from, oral cancer and oropharyngeal cancer compared to the general population [1]. Information on oral and oropharyngeal cancers among Aboriginal and Torres Strait Islander peoples is lacking, creating a void in understanding these diseases and their associated complications. Persistent high-risk human papillomavirus (HPV) infection is recognized as the leading cause of oropharyngeal cancer [2].

The Indigenous Australian Human Papillomavirus Cohort Study is a prospective longitudinal cohort study developed in partnership with Aboriginal communities in South Australia and funded by Australia’s National Health and Medical Research Council [3]. The primary aim of the study is to evaluate the population estimates of oncogenic genotypes of HPV in the mouth and oropharynx among Aboriginal and Torres Strait Islander communities of South Australia. The hypothesis informing the study states that the prevalence of oral HPV among Aboriginal and Torres Strait Islander Australians will be high compared with national-level estimates. The secondary aims include evaluating the rates of oropharyngeal carcinoma and the impact of HPV vaccinations, as well as evaluating the efficacy and cost-effectiveness of targeted extended HPV vaccinations among Aboriginal and Torres Strait Islander peoples.

The study is governed by an Aboriginal Reference Group, with data collected by trained Aboriginal research officers. Baseline eligibility included identifying as Aboriginal and/or Torres Strait Islander, being aged 18 years or over, and residing in South Australia. Participants were recruited from February 2018 to January 2019 across 11 South Australian sites, primarily through Aboriginal Community Controlled Health Organisations (ACCHOs).

Recruitment, Retention, and Follow-Up Strategies

Recruitment strategies were based on successful strategies utilized in the past, including (1) co-designing specific agreements with the chief ACCHOs, which were tailor-made to suit the specific needs of the communities; (2) consulting with Aboriginal community champions, some of whom were formerly involved in our research; (3) encouraging word-of-mouth spread of recruitment; (4) advertisements in local newspapers and radio shows; (5) flyers in post boxes in high-density Aboriginal locations; and (6) presentations to community groups in partnership with the community organizations. A snowballing technique was also used, with participants asked to contact any Aboriginal friends, family, and peers who may be interested in participating.

Follow-up strategies designed to ensure retention across the life of the study involved (1) retaining staff who were committed to following up participants notwithstanding challenges; (2) ensuring a regular communication link between participants and team to ensure accuracy of contact details; (3) ensuring the research officer always took the contact details of at least three key individuals (family members, friends, community members, organization managers) who would be informed about the whereabouts of participants should the current contact details (address, phone numbers) change; (4) sending birthday and Christmas cards and regular newsletters to participants; and (5) encouraging a one-on-one relationship between team members and participants with a genuine attempt to ensure participants saw the same team member at each phase of follow-up.

The recruitment and follow-up strategies were employed after extensive community consultations and, to a large extent, were successful based on pre-existing working relationships and the reputation of the research team. Although these strategies can be cited as successful, the significance of pre-existing relationships based on trust, reciprocity, and faith cannot be ignored. Successful community engagement was supported by ensuring the interactions with all participants were culturally safe. Team members facilitated self-determination by encouraging participants to answer questions themselves. Team members also ensured that the project aims, expected outcomes, risks, all participant rights, and consent were thoroughly explained.

The 1011 participants recruited at baseline represented 5% of Aboriginal and/or Torres Strait Islander South Australian adults eligible during the recruitment period: 8.2% of those eligible in non-metropolitan locations, and 3% of those eligible in metropolitan locations. Participants were followed up at 12 months (March 2019 to March 2020), with data obtained from 749 (74.1%) of the original 1011 participants. This follow-up was suspended early due to COVID-19 restrictions. Follow-up at 24 months ceased in December 2021, with data obtained from 815 participants. Across baseline and 12- and 24-month follow-ups, saliva samples to test for oral human HPV infection were collected using commercially available kits, and the retrieved DNA samples were tested for HPV detection. Further funding has been granted for 48-, 60-, and 72-month follow-ups. At the time of writing, the 48-month follow-up is in the fieldwork stage.

Strong Community Engagement

Strong Aboriginal partnership, engagement, and buy-in of the first study phase were reported against the Consolidated Criteria for Strengthening Reporting of Health Research involving Indigenous Peoples (CONSIDER) statement [4]. The success of the community engagement processes can be summarized as (1) engaging with ACCHOs as equal partners from early in the research process, (2) incorporating an Aboriginal Reference Group, (3) active promotion of the study by ACCHOs, (4) having a flexible agenda responsive to broader environment demands, (5) prioritizing Aboriginal leadership and self-determination while building team capacity, and (6) shifting the narrative from a deficit discourse to a strengths-based discourse.

Specific lessons learned during the first phase included (1) the need for active and wide Aboriginal community consultation initiated early in the research process, (2) strong and sustained Aboriginal capacity building, and (3) governance from an active Aboriginal Reference Group. These lessons were incorporated into subsequent phases of the research.

The presence of Aboriginal staff as representatives in each of the field sites has additional benefits. These representatives have developed their skills and reputations in the area of oral HPV infection, an area of Aboriginal health that has not previously been recognized. The participating Australian Indigenous communities now have a local contact, with study continuation facilitating improved HPV knowledge for both the ACCHO workforce and the broader community. Future phases of the study will continue to implement and strengthen these community engagement strategies.

Empowering Communities

It is essential that this cohort study, to our knowledge the largest Indigenous HPV cohort in the world to track oral HPV infection and monitor the early stages of oropharyngeal squamous cell carcinoma (OPSCC), continues in order to yield critical information that can be added to the management armamentarium of health and wellbeing recommendations for Indigenous Australians. This is especially relevant in light of the imminent rollout of self-sampling cervical HPV testing initiatives in Australia, as it opens doors for a similar self-sampling initiative for oral and oropharyngeal cancers.

The capacity-building element of the study is an ongoing strength and includes (1) naming Aboriginal CEOs of the partnering ACCHOs as co-authors on scientific papers; (2) Aboriginal community leaders attending international conferences, such as the International Indigenous Cancer conferences, to showcase the processes and outcomes of the study’s Indigenous engagement to international audiences; (3) upskilling Aboriginal organizations and staff in a new area of health knowledge aligned with oral cancer; (4) increasing the understanding of health staff/policy makers regarding fears among many Aboriginal and/or Torres Strait Islander peoples about the HPV vaccine, the general lack of awareness of the importance of regular cervical cancer screening, and the impact of HPV-related cancers for both men and women; (5) developing strong relationships with Aboriginal ethics organizations, with increased focus on cultural values when undertaking research in partnership with Aboriginal groups; (6) acknowledging the importance and value of participants’ time and remunerating this appropriately; (7) disseminating dental products to both study participants and their wider households and family groups; and (8) strong advocacy by all research staff with respect to navigating dental care pathways and cancer screening pathways for communities.

Strengths of the Study Design

This project was inspired by previous community engagement during which community members identified an increase in “throat sickness” (cancer) in the community. As Aboriginal and Torres Strait Islander peoples were involved in identifying the health priority, community interest and response were strong and all decisions were culturally safe and based on self-determination. Aboriginal leadership and governance ensured the cultural appropriateness of the strategies employed. The main strength of the Indigenous Australian HPV Cohort Study has been the engagement of South Australia’s Aboriginal communities. This has contributed to excellent recruitment and follow-up rates (approximately 74% at 12 months and 80.6% at 24 months), which are even more significant given the context of the research. This ongoing study is taking place over vast distances (traveling 700 km west, 400 km east, and 800 km north of the city of Adelaide) and involves highly disadvantaged participants who have, in the past, not always enjoyed positive research interactions. The fact that over 1000 participants were recruited in under 12 months demonstrates the widespread community support that exists for this research. It should also be noted that the intervention was conducted during the COVID-19 pandemic, thus limiting in-person interactions.

Strengths of the Research Team

One of the most significant aspects of research success and increased community engagement has been the research methodology employed. All members of the team were trained by an experienced Senior Aboriginal Researcher and were taught the principles of Relational Yarning, now a recognized culturally appropriate research methodology. The core values of the research team are based on prioritizing respect, relationships, advocacy, reciprocity, time, and gratitude. Aboriginal leaders continue to promote self-determination and encourage community members to trust and participate in the research. These leaders, combined with the diverse multidisciplinary skills of the research team, have played a major role in the success of the research. The members of the research team have continually recognized their own privilege, social position, and power, and situated themselves in ways that have ensured participants feel culturally safe, respected, heard, acknowledged, and appreciated at all times. They have ensured that interactions with participants are devoid of judgment or power differentials, placing the power back into the hands of the participants and communities. All research papers, presentations, and community conversations focus on a strengths-based narrative, practicing and reinforcing the principles of decolonizing research [5].