Scientific literature offers several recommendations on ethical, legal and cultural considerations for research in low resource settings and for recruitment of minority populations. Experiences with community engagement and informed consent for genetic studies in Africans  and for obtaining DNA samples from four populations including the Yorubas from Ibadan (Nigeria) have also been reported . However, there is no report on issues that should be considered when recruiting entire families in rural and urban settings with genetic research as the main goal.
Successful recruitment of diverse populations such as the Yorubas for genetic studies is important for advancement in biomedical research. The Yoruba ethnic group was chosen for this linkage analysis approach because we were expecting that large families with keloids could be recruited. Due to the expected locus heterogeneity for causative variants leading to keloids [32–34], recruitment from the Yoruba population seemed ideal for finding significant linkage because of relatively low admixture [35, 36] compared to many other populations.
To attain acceptance of a genetic study within multiple generations of a family, it is important to appreciate social, religious, and cultural beliefs that may impact participation. The Yoruba ethnic group that occupies mostly the western part of Nigeria highly values their cultural practices and religious beliefs. Although most Yorubas are Christians or Muslims, they retain their practice of worshiping different gods such as Sango (god of thunder), Obatala also known as Orisa-nla (the shaper, former, maker), Yemoja (god of the river) and Ogun (god of iron). All these beliefs create perceptions about the possible causes for a disease and often determine the approach to treatment. Traditional healers still have high influence on people’s decisions. Part of their health beliefs is that illnesses may originate from one or a combination of three forces; the magical practices (witches or sorcerers), natural events (environmental, hereditary) and supernatural occurrence (destiny or the individual’s double spirits) [37–39]. It has also been documented that many Nigerians approach treatment of diseases by utilizing Western medicine in combination with treatments offered by traditional healers and “spiritual” centers .
For example, one study reported that some Yorubas identified environmental factors such as diet and use of unorthodox traditional herbs as the cause for cancer . The interest in experimenting with different treatments could explain why some of the participants in our keloid study reported treatment of their keloids with a combination of traditional herbs and Western medicine and why they were willing to contribute to science to find better treatment or cure. However, stigmatizing and/or painful diseases such as cleft lip and palate, cancer or keloids are by some perceived to be in part due to a “punishment” for evil deeds or result from retaliation by a jealous foe [37, 38, 40]. As a result, many Nigerians are weary of participating in studies that have the potential of “affecting” them or other family members, especially if they donate blood, saliva or hair samples. Recruitment was also affected by challenges that had already been described in other cultural environments [30, 41]. Such issues included economic factors, lack of trust, different cultural background of researchers and study participants, or issues with time and transportation needed to reach remotely located family members.
Meeting regulatory and administrative requirements (IRB, ethics committees and FWA) as well as staff training was less of a problem compared to recruitment challenges. Research staff took the Collaborative Institutional Training Initiative (CITI) training for good clinical practice before they engaged in recruitment. An online education module for ethics training on research based on the Nigerian Code for Health Research Ethics has recently been developed and validated. Future studies will require research staff to take this module that encompasses the Nigerian cultural and social principles .
Our experience with this study confirmed the need to allocate extra time for all stages of the study. Extra time was needed to educate the public and prospective research participants about the study and to make up for delays due to travel logistics when recruiting relatives residing outside a proband’s home town or when family members were not available for recruitment in a timely manner. Additional challenges arose during the rainy season when treacherous road conditions led to extended delays. Unreliable public transport slowed down recruitment throughout the study period. While purchasing a car dedicated to the study might have been the most economical way to travel to recruitment sites, administrative barriers and uncertain liability issues prevented us from doing so.
Working with an institution with established research infrastructure versus a site without any research infrastructure or experience with foreign collaboration should be taken into consideration when planning a study as this will likely impact timely study initiation, staff recruitment, and research coordination. There is recognition that capacity building in African countries must be addressed for successful integration of human genetic studies . Nigerians and specifically the Yorubas are generally not research-naïve. Participation of Yorubas or any underserved population in genetic studies can be enhanced by community involvement from inception of the study as well as by regular feedback , especially when the study design involves participation of family members from several generations. Our study plan included the assistance from probands or other individuals with keloids and contact with community leaders, kings or chiefs where applicable. Interaction with members of the oldest generation or children of old family members may be difficult when the culture of scientists and recruiters is different from that of the participants. To address these and other issues, our study involved research staff from the local community who were born and raised in the Yoruba culture. Visits to kings, chiefs, churches and mosques were useful to convince the community that blood and saliva samples would not be used for voodoo or juju. It was important and reassuring to be accompanied by research participants from their community at such meetings to demonstrate that nothing bad had happened to them since recruitment. It was also important to ensure that clinic or hospital administrators were aware of this study so that they could then encourage the population to participate. We were allowed to recruit in local palaces, churches and health centers/hospitals. Additionally, the research staff was available for recruiting during special holidays when relatives would return home for family celebrations. Cultural sensitivity during study coordination was ensured by a member of the US team that was born and raised in the Yoruba community.
Genetic analysis identified non-blood relatives in some families. Some pedigrees had to be updated several times as additional information became available. While scientists require accurate information about a participant’s position in a family it is more important for the traditional Yoruba family where individuals fit within a family than how they are biologically related. When obtaining family histories, we found that “adopted” distant relatives were sometimes identified as children or parents. We also encountered situations where friends of the family were reported as members of the family. These situations required that research staff approached a certain member of the family with clear questions to obtain accurate information before establishing a pedigree. During DNA analysis we still identified participants who did not fit in the family structure. The need for confidentiality in these situations was discussed among researchers and participants were either removed from the pedigree or in rare instances the family was eliminated from analysis. With increasing education, the community may become more aware of inheritance and biological relationships within a family and better understand the significance of genetic family studies. Until then, investigators should be prepared to address these factors in their study plan.
Even though remuneration is always a difficult topic for recruitment in low resource settings, it is still an incentive for participants who otherwise may not be able to travel to a recruitment site or miss some work/earning to volunteer for a study “without immediate benefit” to them. Other studies also reported that some participants expected more payment for their contribution , while others preferred to have free treatment/cure in lieu of money . Investigators should be aware of the potential for abuse by opportunists that may impact recruitment. Our research protocol, for example, required that all study participants be treated and compensated equally.
In a review of Project SuGar, the author identified some dynamics that negatively affected recruitment of African-American families into genetic studies and offered suggestions to enhance recruitment . Impacting factors included economic barriers, core values including cultural, spiritual and social factors, as well as health practices of the community. Previous studies have demonstrated that the majority of Nigerians would participate in research activities. Several approaches have been recommended or used to engage, recruit and retain Nigerians for genetic studies . Such methods include the use of community advisory councils, incentives, local partners/champions, focus groups, awareness through public forums and less cumbersome wording during the informed consent process. Our study employed many of these suggestions.
Knowledge of genetic variations and their roles in disease pathology and for treatment outcome is significant in the quest to reduce health disparities. While there seems to be a recent increase in genetic studies conducted in African countries, the majority is usually in collaboration with and led by Western scientists. African countries must continue to be involved and take a leadership role in genetic studies so their people can benefit from the advances of the genomic and the post-genomic era. One could argue that the Yoruba people still have to offer a lot to science. Nigerians have been called upon to embrace the opportunity of improving their people’s quality of life with a timely response to technology gaps in genomics . Initiatives such as the MalariaGen  and the Human Heredity and Health in Africa (H3Africa) project, among others, have begun to address this concern (H3Africa: Human heredity and Health in Africa, http://www.h3africa.org/).