Sharing results from complex disease genetics studies: a community based participatory research approach.

Abstract

Dissemination of research results to communities builds capacity of the community to understand and utilize the results. The objective of this manuscript was to propose a culturally appropriate approach to disseminate complex disease genetics research findings in small Alaska Native communities.
The Center for Alaska Native Health Research is a community-based participatory research project (CBPR) directed at understanding the interactions between genetic, nutritional and psychosocial risk factors for obesity, diabetes, and cardiovascular disease in Yup'ik Eskimos.
We have consulted with regional healthcare providers, tribal leaders, and university-, local-, and national-institutional review boards to identify potential mechanisms for sharing population-based genetics research results or progress.
We propose a six step CBPR-approach to conducting genetics research in isolated identifiable communities. This CPBR-approach includes generating a common research question, determining community interest, recruitment, capacity building, sharing power and control, avoiding group harm, and development of culturally appropriate dissemination procedures.
Research scientists and community members should both benefit from population-based genetics research. Although we are just beginning our discussions with regard to sharing genetics research progress and findings, we believe that it is essential move forward as co-researchers in the CBPR enterprise.

Full text

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International Journal of Circumpolar Health 66:1 2007
SHARING POPULATION-BASED GENETICS RESEARCH PROGRESS
SHARING RESULTS FROM COMPLEx DISEASE
GENETICS STUDIES: A COMMUNITY BASED
PARTICIPATORY RESEARCH APPROACH
Bert B. Boyer, Gerald V. Mohatt, Renee L. Pasker, Elaine M. Drew, Kathleen K. McGlone
Center for Alaska Native Health Research, Institute of Arctic Biology, University of Alaska Fairbanks, USA
ABSTRACT
Objectives. Dissemination of research results to communities builds capacity of the community 
to understand and utilize the results. The objective of this manuscript was to propose a culturally
appropriate approach to disseminate complex disease genetics research findings in small Alaska
Native communities.
Study Design. The Center for Alaska Native Health Research is a community-based participa-
tory research project (CBPR) directed at understanding the interactions between genetic, nutri-
tional and psychosocial risk factors for obesity, diabetes, and cardiovascular disease in Yup’ik 
Eskimos.
Methods. We have consulted with regional healthcare providers, tribal leaders, and university-, 
local-, and national-institutional review boards to identify potential mechanisms for sharing popu-
lation-based genetics research results or progress.
Results. We propose a six step CBPR-approach to conducting genetics research in isolated identi-
fiable communities. This CPBR-approach includes generating a common research question, deter-
mining community interest, recruitment, capacity building, sharing power and control, avoiding 
group harm, and development of culturally appropriate dissemination procedures. 
Conclusions. Research scientists and community members should both benefit from popu-
lation-based genetics research. Although we are just beginning our discussions with regard to 
sharing genetics research progress and findings, we believe that it is essential move forward as
co-researchers in the CBPR enterprise. (Int J Circumpolar Health 2007; 66(1):19-30)
Keywords: Yup’ik, Eskimo, confidentiality, family-based recruitment, reporting research results,
group harm

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The CANHR study overview
The Center for Alaska Natives Health Research 
(CANHR) study is a community-based partici-
patory  research  (CBPR)  project  focused  on 
identifying  current  risk  factors  for  obesity, 
diabetes, and cardiovascular disease in Alaska 
Natives living in southwest Alaska. The goal of 
our multidisciplinary and CBPR study design 
is to assess genetic, nutritional and behavioral 
risk factors by integrating the knowledge of the 
Center  researchers  with  the  local  knowledge 
of  community  stakeholders  and  residents.  A 
detailed description of the organizational devel-
opment and procedures of the CANHR study 
has been described elsewhere (?). In order to 
examine the interrelationships of genetic and 
environmental risk factors in the overall devel-
opment of obesity and diabetes, the CANHR 
study  team,  local  health  corporation,  and 
participating communities collaborated on the 
development of a study involving family-based 
recruitment and complex disease genetics that 
involved community input to shape all aspects 
of the genetics work. 
The goal of a CBPR-orientation to research 
is  to  involve  the  community  equally  in  all 
aspects of research, from design to dissemina-
tion of results (?). This approach has resulted 
from past studies failing to include members of 
small, identifiable communities in all aspects of
the research process, resulting in some commu-
nities being unwilling to participate in future 
research projects (?; ?). Rather than following 
a top-down, expert-driven model in which the 
researcher is distinctively empowered to deter-
mine research questions, methods, and results, 
a CBPR design has the advantage of working 
directly  with  communities  to  ascertain  their 
views on how the project should proceed.  From 
this perspective, research occurs as an iterative 
process that emphasizes the values of participa-
tion, cooperation, and accountability.   
A  CBPR  approach,  while  rarely  used  in 
genetics research, is fundamental to the ongoing 
success  of  the  CANHR  study.  Indeed,  there 
are active discussions throughout the scientific
community in regard to ethics of population-
based genetics research (5, 6), including how 
genetics studies should be developed in Native 
communities (7, 8), how family members should
be recruited into genetics studies (9, ?0), as well 
as how, and when, genetics study results should 
be reported back to participants and/or commu-
nities (??, ??).  In regard to sharing genetics 
results with participants, the National Bioethics 
Advisory  Commission  (NBAC)  recommends 
that  genetics  results  should  not  be  disclosed 
to  participants  unless  the  results  have  been 
confirmed; the findings have significant health
implications, and treatment, as well as medical 
counseling is available (??). 
Such  guidelines,  while  important,  present 
CBPR  studies  focused  on  population-based 
studies involving genetics of complex diseases 
with  an  ethical  dilemma.  Complex  diseases 
like obesity, type II diabetes, and cardiovas-
cular  disease  are  thought  to  result  from  the 
composite interactions of numerous genes and 
environmental factors. While population-based 
research on complex diseases will ultimately 
lead  to  enhanced  understanding  of  disease 
pathogenesis  and  improved  public  health, 
the path to this endpoint is complex and will 
require years of research from multiple scien-
tific disciplines (14). In the meantime, CBPR
researchers’, must acknowledge the importance 
of reporting back to participating communities 
in a timely manner, while contending with how 
to conduct complex disease genetics research 
within a CBPR framework when our research 

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findings have not been validated, nor conducted
in a clinically approved laboratory, and when 
there  is  no  treatment  available.  This  manu-
script  focuses  on  some  of  the  ethical  issues 
surrounding dissemination of complex disease 
genetics research findings within Alaska Native
communities, and how a CBPR framework may 
address these issues.
Ethical issues in the dissemination of complex
disease genetic results in a CBPR study
In general, dissemination of genetic data raises 
a  complicated  set  of  ethical  issues.    These 
issues include group harm, discrimination, stig-
matization, and privacy. Genetic data obtained
from members of isolated populations or iden-
tifiable groups is different from data collected
from large admixed population groups because 
it has the potential to harm not only research 
participants,  but  also  community  members 
not involved in the research project. If genetic 
results indicate that one population has a higher 
disease risk, this could lead to stigmatization
and  discrimination.  Therefore,  one  recom-
mended safeguard to reduce the risk of stig-
matization of socially identifiable populations
is to withhold the name of those communities 
in publications or presentations (?5). In addition 
to community protections, scientific investiga-
tions must also ensure privacy and confidenti-
ality of individual participants and their family 
member’s  such  that  the  ethical  principle  of 
beneficence is fulfilled (11).
Within a CBPR framework, an overriding 
objective  is  to  include  the  community  as  an 
equal partner in the research enterprise.  This 
includes sharing results that are both positive 
and negative.  However, if genetic data do not 
provide an immediate health benefit to partici-
pants,  should  researchers  withhold  results?   
Perhaps the most reasonable and ethical compro-
mise is to provide genetics research “progress 
updates”  to  participating  communities,  since 
individual participant feedback does not meet 
NBAC guidelines. Ultimately, we believe it is 
essential to determine the communities’ views 
on  ethical  issues  related  to  dissemination  of 
genetics research progress so that they have an 
active role in this important decision. 
CANHR and the CBPR approach
Employing  a  CBPR  approach,  CANHR 
researchers partnered with ten Native commu-
nities to conduct each phase of the study. In 
addition,  the  regional  Yukon  Kuskokwim 
Health Corporation (YKHC) played an inte-
gral  role  in  shaping  the  overall  process, 
including community recruitment and selec-
tion, individual participant recruitment, data 
gathering protocols, interpreting results, and 
developing procedures for sharing individual 
clinical results (?).
A CBPR approach to conducting
genetics research
Step 1:  Determining whether communities
wanted to participate in a genetics research
project.  Unlike other CBPR research under-
taken by the CANHR study team, which gener-
ated the research questions at the community 
level, the questions for the CANHR genetics 
study  derived  from  analyses  of  epidemio-
logical  data  and  tribal  health  meetings  that 
expressed concern about the increasing preva-
lence of chronic diseases. Because our center 
includes  genetics  research,  we  believe  it  is 
extremely  important  for  us  to  provide  some 
form  of  genetics  education  to  the  Alaska 
Native community leaders and health corpo-
ration representatives to assist their decision-

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making about participation in the study. At the 
same time, it was equally important to iden-
tify Alaska Native genetics research concerns 
before initiating data collection. 
To open the dialog of genetics research with 
the community leaders, we consulted with one 
of our Center’s external advisory committee 
members, Dr. Linda Burhansstipanov, Execu-
tive Director of the Native American Cancer 
Initiatives,  Inc. (http://natamcancer.org/), 
who has conducted many genetics education 
workshops throughout the country to Native 
American  groups.  Previously,  she  developed 
the Genetic Education for Native Americans 
(GENA) program and agreed to deliver a two-
day (?0 hour) workshop to tribal council and 
community  representatives  from  our  Yup’ik 
Eskimo study population, as well as to relevant 
ethics and human studies committee represen-
tatives  from  the  YKHC.  Approximately  ?0 
people participated in the two day workshop. 
The  workshop  introduced  topics  including 
basic cell and molecular biology, a review of 
genetic concepts including inheritance, cultural 
traditions, risks and benefits, and the Human
Genome  Project,  as  well  as  cultural  issues 
related to genetic research, and guidelines for 
culturally respectful genetic research. 
The  GENA  workshop  enhanced  commu-
nity members’ understanding of our approach 
to human genetics and effectively introduced 
CANHR scientists to the community. It also 
provided  a  key  opportunity  to  build  rapport 
through  an  open  dialogue  that  revealed  our 
genuine respect for Alaska Native culture and 
our  goal  to  work  collaboratively  for  cultur-
ally respectful protocols. Following the work-
shop,  the  YKHC  board  fully  embraced  the 
idea of the genetics research project. Indeed, 
the importance of genetics research was best 
summarized by a respected Elder and Emer-
itus Member of the YKHC Board of Directors 
when, in agreement, he stated, “I want to eat 
what my blood tells me to eat.” He summa-
rized his interpretation of the genetic research
as a vehicle for confirming that his blood had
been formed within his culture to resonate with 
certain  foods  and  could  instruct  the  people 
about what food would be healthy for them and 
lead to wellness.
Step 2: Community selection and partici-
pant recruitment.  YKHC medical staff and 
CANHR scientists met on several occasions 
to nominate a set of communities that did not 
overlap with other similar studies taking place 
in  the  region.  Communities  selected  were 
stratified by population size, geographic loca-
tion (coastal and river, which are thought to be 
related to types of diet), and religion (Moravian 
or Russian Orthodox), which are thought to be 
related to the prevalence of such risk factors 
as  smoking  and  alcohol  consumption).  Only 
those communities that gave community-wide 
consent participated. To date, ten communities 
have already participated. Additional commu-
nities have approved the CANHR study and 
have expressed an interest in future participa-
tion. While two communities denied participa-
tion, one of those two has since agreed to join 
the study. 
Step 3: Building research capacity within
communities.  Following  community  selec-
tion,  the  CANHR  project  hired  and  trained 
several staff members that have been instru-
mental to the success of our research center. 
These local community and regional residents 
acted  as  research  assistants  for  all  phases 
of the study. Our field research coordinator

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is a registered nurse and has participated in 
all aspects of organizing data collection and
training many of the staff that work directly 
with  participants.  Finally,  we  also  hired  a 
bilingual Yup’ik Eskimo research coordinator 
at the regional level to function as a liaison 
between the university, the health corporation, 
and  the  participating  communities.  Working 
together,  this  research  team  recruited  and 
enrolled  participants,  and  collected  blood, 
anthropometric, nutritional, physical activity, 
and psychosocial data. In addition, the team 
presented  the  research  results  back  to  the 
communities in both Yup’ik and English. 
Overall, the CBPR framework of working 
closely together throughout each part of the 
study provided constant checks and rechecks 
of the study as it proceeded.  For example, 
the  community  members  initiated  questions 
that led to changes in our research. We also 
raised questions for the community that helped 
us understand how to conduct our research in 
culturally  appropriate  ways.  As  a  result  of 
these  ongoing  interactions,  we  endeavored 
to develop a procedure (described below) for 
ensuring a balance between the scientific goals
of CANHR researchers and the community, 
and personal goals of Native participants.
Step 4: Sharing power and control. There 
are  many  reasons  why  Native  American 
communities are increasingly concerned about 
participating in genetics research projects (8). 
Some of the concerns raised when we began to 
work with Yup’ik Eskimo participants included 
being  over-researched,  not  hearing  from 
research  scientists  that  previously  initiated 
projects in their communities, fear of testing 
for drugs, apprehension about the potential for 
blood sample use that was not approved in the 
original study design, and potential for exclu-
sion from medical care or possible stigmati-
zation if a particular disease or mutation was
discovered that might be common in the Yup’ik 
community (group harm). Such concerns are 
shared by many indigenous communities (7).
Our  CBPR  approach  suggested  that  we 
needed to identify local concerns and develop 
culturally appropriate solutions in collabora-
tion with community members. For example, 
many of the community and health corpora-
tion  concerns  related  to  what  the  CANHR 
study  was  going  to  do  with  the  participant 
blood  samples,  and  how  CANHR  scien-
tists were going to report their results to the 
broader scientific community. We worked
with  members  of  our  external  advisory 
committee, the University of Alaska Institu-
tional Review Board (IRB), the Alaska Area 
IRB, the National Indian Health Service IRB, 
the YKHC human studies committee, and the 
Alaska  Centers  for  Disease  Control  Arctic 
Investigations Program to reach a consensus 
on how to share power and control over speci-
mens and information. As a result, we jointly 
developed protocols, consent forms, and iden-
tification procedures that adequately detailed
our methods of storage and approved use of 
the collected blood samples. Moreover, since 
collective stigmatization was of great concern
to  the  YKHC  Human  Studies  Committee, 
we agreed that any reporting of our research 
results in the form of abstracts, publications, 
and presentations would be approved first
by the YKHC to protect the individuals and 
communities  participating  in  our  research. 
Still, in spite of these efforts to share power 
and do no harm, the best methods to report 
our genetics research progress to the partici-
pating communities remain unclear. 

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Step 5: Avoiding group harm and stigmati-
zation.  Inclusion criteria required that partici-
pants be non-pregnant and of Alaska Native 
descent or married to an Alaska Native. If indi-
viduals that were not Alaska Native wanted to 
participate in the research project, they were 
allowed  to  do  so  and  they  received  direct 
feedback on their blood lipid profiles, body
measurements  and  overall  health.  However, 
their data was omitted from further analysis in 
the broader CANHR study. By restricting our 
analysis to Alaska Native Yup’ik Eskimos, our 
results directly relate to what we have learned 
about Yup’ik Eskimo health. At the same time, 
however, it is possible for our participants to 
be more readily exposed to the possibility of 
“group  harm,”  since  Yup’ik  Eskimos  come 
from a small region of Alaska and number less 
than ?5,000 people. 
The CANHR scientific team has taken
several  steps  to  counter  the  possibility  of 
potential  group  harm  in  our  studies.  First, 
we  spent  considerable  time  consulting  with 
the communities and the health corporation 
and working with several IRB committees to 
develop the final CANHR research operating
procedures. Second, we have severely limited 
access to the blood samples and data collected. 
All research done with existing samples has 
to be approved by the CANHR Director, the 
YKHC Executive Board and Human Studies 
Committee, and if the aims do not directly 
relate  to  obesity,  all  participants  must  be 
actively consented for the new study. Finally, 
before dissemination of any results can proceed 
to  the  publication  or  presentation  stage,  all 
research  must  be  approved  by  the  YKHC 
Human Studies Committee. These procedures 
were discussed with each tribal council and 
approved  prior  to  initiation  of  the  research 
projects. One of the first lessons we learned
in our efforts to maintain group confidenti-
ality is that our study participants preferred 
to  be  called  “Yup’ik  Eskimo,”  rather  than 
the more broadly defined “Alaska Native”
terminology. In addition, they decided that 
we should not disclose the names of the indi-
vidual communities, but rather use the more 
general  regional  descriptor  of  southwest 
Alaska, and Yukon Kuskokwim region.
As  the  CANHR  genetics  project  is  a 
family-based research project, and even with 
many protective strategies in place, we had 
to consider other issues specifically related to
genetic studies in isolated communities that 
involve family members (9, ?0). In terms of 
design, we believe that several characteristics 
of  the  Yup’ik  Eskimo  population  structure 
will facilitate the identification of obesity
susceptibility  genes.  The  reduced  number 
of  founders,  historical  bottlenecks,  as  well 
as the small population size, and limited
admixture should reduce the number of risk 
alleles contributing to complex disorders like 
obesity (16, 17). Moreover, the homogenous
environment in which our participants reside 
will  facilitate  the  control  of  intervening 
environmental factors (?8). Family member 
participation is facilitated by the fact that the 
communities we work in are generally less 
than 500 people in size and many individuals
in  nearby  communities  are  related  to  one 
another. 
During the design of the CANHR study, 
we  discussed  recruitment  strategies  with 
the local health corporation and ultimately 
agreed to invite everyone in the communi-
ties to participate. While our first wave of
participant  recruitment  in  the  communities 
ranged from ?0-?0% of all eligible partici-

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pants, we realized we also needed to discuss
how family members of enrolled participants 
(probands) might be invited to participate in 
the study. While we wanted to avoid placing 
undue  pressure  on  probands  to  encourage 
relatives to participate, we also did not want 
to introduce bias into our sampling methods 
from low participation rates (5). We consulted 
with the YKHC and ultimately decided that 
the  best  option  to  enhance  family  member 
recruitment would be to request that probands 
call their relatives, invite them to participate 
in  the  CANHR  study,  and  then  we  would 
schedule several return visits to the relevant 
communities so that we would be available 
if  relatives  decided  to  participate.  While 
other studies have asked probands to contact 
their  relatives  and  determine  if  they  were 
willing to be contacted by the research staff 
regarding participation, the YKHC thought 
this may put unnecessary pressure on rela-
tives to participate and constitute an invasion 
of their privacy. The process we used maxi-
mized privacy, but also likely reduced partic-
ipant  accrual  (?0).  Nevertheless,  protecting 
the privacy of family members that did not 
participate is equally important to protecting 
the privacy of those that did, since non-partic-
ipants could potentially suffer psychosocial 
harm, stigmatization, discrimination, and
other forms of group harm from being associ-
ated with participants in small communities 
that did participate. The overall objective of 
our procedures is to protect participants and 
non-participants equally from potential stig-
matization.
Step 6: Development of culturally appro-
priate dissemination procedures for clinical
results.  Before  we  could  begin  to  develop 
methods to share genetics results, we first
developed a process to disseminate clinical 
results to participants. These results included 
blood  lipids,  fasting  glucose  levels,  blood 
pressure,  nutritional  status,  and  physical 
measurements. At a community level, aggre-
gate results of the clinical biomarkers, phys-
ical  measurements,  behavioral,  and  nutri-
tional/physical  activity  project  have  been 
shared to maximize distribution of research
knowledge among participants. 
Once we obtained our first summary results
on Yup’ik health, we began to work closely 
with community members to develop cultur-
ally relevant formats in which our research 
results  could  be  shared  and  understood  by 
tribal  elders  and  community  members.  For 
instance,  the  request  for  formats  that  used 
cultural  symbols  rather  than  bar  graphs  or 
pie  charts  came  directly  from  members  of 
the tribal council in the first community
that hosted a dissemination presentation. An 
example of a research format results slide that 
we originally presented to the tribal council 
(Figure ?) is compared to the culturally rele-
vant slide developed in partnership with our 
community liaisons after feedback from the 
tribal council (Figure ?). In this example, bar 
graphs were replaced with a more meaningful 
diamond  pattern  design  that  is  commonly 
found  on  traditional  garments  worn  by 
community members.
Ultimately, the culturally relevant formats 
we  used  could  not  have  developed  without 
our strong relationship with the communities. 
Indeed, our rapport was such that we invited 
them to openly criticize our presentations
so that we could improve intelligibility and 
cross-cultural translation. In addition, instead 

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International Journal of Circumpolar Health 66:1 2007
SHARING POPULATION-BASED GENETICS RESEARCH PROGRESS
of presenting the results in English and then 
translating difficult concepts back to Yup’ik,
they requested that all of our presentations be 
made in Yup’ik by the local coordinator in the 
presence of at least one CANHR scientist who 
could assist in answering questions. Feedback 
from  the  tribal  leaders  has  been  extremely 
positive  with  regard  to  these  data-summary 
presentations, and similar presentations have 
now been made to the YKHC and the broader 
community of participants. 
Important considerations in reporting
progress from population-based
genetics studies
We have only recently received initial results 
from  the  genetics  research  and  must  now 
develop culturally appropriate ways to share 
our  research  progress  with  participating 
communities. In addition, before meaningful 
discussions  can take place, we also  need to 
identify  some  of  the  genetic  epidemiolog-
ical  considerations  that  should  be  discussed 
1
42
36
20
2
23
30
44
0
5
10
15
20
25
30
35
40
45
50
Underweight NormalOverweightObese
%
men
women
1% 2% 42% 23% 36% 30%20% 44%
Underweight Just Right HeavyVery Heavy
WomenMen
Figure 2. Culturally appropriate representation of the prevalence of overweight and obesity in Yup’ik
Eskimo men (n=288) and women (n=352). Underweight is defined as a BMI less than 18 kg/m2, just
right BMI is between 18-25 kg/m2, heavy BMI is between 25-30 kg/m2, and very heavy is defined as a
BMI greater than 30 kg/m2. Mann-Whitney: p<0.001.
Figure 1. Scientific representation of the prevalence of overweight and obesity in Yup’ik Eskimo men
and women. Underweight is defined as a BMI less than 18 kg/m2, normal BMI is between 18-25 kg/m2,
overweight BMI is between 25-30 kg/m2, and obese is defined as a BMI greater than 30 kg/m2.

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International Journal of Circumpolar Health 66:1 2007
SHARING POPULATION-BASED GENETICS RESEARCH PROGRESS
with our community partners. The long-term 
objective of these discussions is to maximize
benefit and understanding of genetics research
and our progress, while minimizing harm to
participating  individuals  and  communities 
and adhering to the guidelines of the National 
Bioethics Advisory Commission. These objec-
tives will foster a trusting research partnership 
with participating communities. 
Before  discussing  genetics  research  prog-
ress  with  our  participating  communities, 
we will first prepare a traditional scientific
presentation and share this presentation with 
CANHR  investigators  and  staff,  including 
Yup’ik Eskimos employed by CANHR. Next 
we will modify the presentation of research 
progress  based  on  feedback  from  CANHR 
investigators  before  presenting  the  revised 
progress  report  to,  and  holding  discussions 
with,  our  rural  community  research  assis-
tants. Suggested modifications from our rural
community research assistants will be incor-
porated into the revised presentation, and then 
we will hold a focus group meeting in one of 
our  participating  communities  to  ascertain 
their  understanding  of  the  genetics  progress 
report and incorporate suggestions to improve 
it. Based on all of the aforementioned feed-
back, a final presentation format for all future
presentations  will  be  developed  that  will 
incorporate community views on how genetics 
results should be disseminated in a culturally 
appropriate and understandable manner.
Population-based  genetics  studies  similar 
to the ongoing CANHR study involve geno-
typing participant samples for genetic variation 
(polymorphisms) that may be associated with 
the particular disease of interest. Population-
based  research  studies  investigating  genetic 
variation  associated  with  complex  diseases 
is significantly different from genetic testing
for Mendelian abnormalities in clinical labo-
ratories that are approved by Clinical Labo-
ratory Improvement Amendments (CLIA), or 
reporting  lab  results  from  standard  clinical 
practice.  In  population-based  studies  like 
those conducted in CANHR, the genetic tests 
are conducted solely for research purposes and 
are conducted in non-CLIA approved research 
laboratories  to  identify  genetic  risk  factors 
for  obesity.  Results  we  obtain  will  need  to 
be replicated in other populations and could 
only be moved from research to clinical use 
after  their  diagnostic,  prognostic,  and  treat-
ment value have been established (??). Once 
a genetic test is approved for clinical use, the 
National Bioethics Advisory Commission has 
recommended that the results should only be 
disclosed to individuals when: (1) the find-
ings are scientifically valid and confirmed;
(2) the findings have significant implications
for the subjects health; (?) a course of action 
or treatment is available; and (?) appropriate 
medical advice (genetic counsellor) or referral 
is provided (??). 
In general, the immediate clinical utility of 
population-based  genetics  studies  involving 
complex  diseases  like  obesity  and  diabetes 
are not currently at the stage where a genetic 
test will result in an improved health outcome 
(criterion  ?  above),  and,  therefore,  from  an 
ethical point of view,  the results  should  not 
be reported to individuals (?9). Nevertheless, 
in a community-based participatory research 
model, it is necessary to communicate some 
form of study progress back to participants, 
and  there  may  be  several  reasons  to  do  so, 
including the fact that relationships between 
researchers and participants are strengthened 
(?0), participants’ understanding of genetics is 

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International Journal of Circumpolar Health 66:1 2007
improved (??), and trust in the research process 
may be enhanced if participants are informed 
of the study outcomes and their contribution 
to public health (8).  
While  there  are  several  advantages  to 
sharing  genetics  research  progress  with 
participants, there are also several steps that 
need to be taken before genetic tests can be 
used to predict health outcomes (??, ??, ??). 
First and foremost, the descriptive epidemi-
ology and genetic associations must be repli-
cated in representative populations. Second, 
the  relationship  between  genotypes  and 
health outcomes for most common diseases 
is strongly influenced by environmental
factors that must be evaluated. Third, a thor-
ough assessment of the analytic validity (test 
accuracy), clinical validity (how well the test 
predicts  the  outcome),  and  clinical  utility 
(how useful the test is in preventing disease), 
of  complex  disease  gene  testing  must  be 
determined  in  clinical  trials  and  epidemio-
logic  studies  (??).  Finally,  the  ethical,  legal 
and social implications of genetic testing must 
be assessed. Despite these significant hurdles,
great strides are being made in this exciting 
new era of human genome epidemiology (??, 
??). Indeed, there is tremendous potential to 
develop public health interventions based on 
the identification of environmental factors
(behavioral,  nutritional,  and  psychosocial) 
that may reduce the risk of developing certain 
diseases. 
Conclusions
Beyond collaboration to co-researchers
Both scientific researchers and community
members will benefit from open discussions
related  to  human  research  participation  in 
general,  and  to  genetics  research  participa-
tion in particular. From our experiences thus 
far, many important questions have emerged. 
How do relatively small identifiable commu-
nities view research risks? How do they eval-
uate research outcomes and their potential for 
group benefit versus participation in research
and the possibility for group harm? Do various 
communities have specific cultural concerns
in relation to genetics research that may be 
resolved  through  mutual  education,  discus-
sion, and trust? How do they resolve personal 
interests in participation versus community or 
leadership opposition to participation?  What 
sense do they make of the results that we have 
obtained? In the end, we will work through 
interpreting  genetic  data  with  community 
members within our CBPR framework. 
While  there  are  many  considerations  to 
take into account with respect to reporting 
genetics results or study progress from popu-
lation-based  studies,  in  the  spirit  of  CBPR 
research, we believe it is essential to initiate 
discussions with our community partners – 
our co-researchers – on the issues surrounding 
dissemination.  How  we  ultimately  report 
study  progress  in  genetics  research  to  the 
community demands that they not only grasp 
our understanding of the results, but also work 
with us to make translatable sense of them. 
An explanation of the uncertainty of complex 
disease genetics results, the need for replica-
tion studies, and the fact that a genetic asso-
ciation does not equate to disease causality is 
essential knowledge to share. Perhaps prepa-
ration  of  culturally  meaningful  summary 
results or progress reports would be helpful, 
but  only  after  the  community  has  assisted 
us in interpreting the results. This includes 
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International Journal of Circumpolar Health 66:1 2007
understanding the importance of negative or 
inconclusive findings. Although such results
may  not  be  exciting  to  the  community,  we 
believe  that  we  should  work  through  these 
issues together. 
Within  its  infrastructure,  CANHR  has 
developed two core facilities to oversee the 
development of a successful methodology for 
this co-researcher interaction. The Cultural-
Behavioral/Intervention Core and the Genetics 
Core  collaborated  on  the  initial  genetics 
education  workshop  as  well  as  in  under-
standing how Yup’ik culture perceives blood, 
genetic transmission, and the appropriateness 
of reporting research findings. We are in the
process of planning how the two cores will 
engage in the future in a series of workshops 
and seminars with the Elders and leaders of the 
communities to address each question and to 
reach consensus on a Yup’ik ethics of genetics 
research. Our current objective is to do this in 
the context of understanding the results that 
we are now obtaining, to interpret the data 
together, and to decide, as co-researchers, the 
best way to disseminate these findings.
Acknowledgements
This publication was made possible by Grant 
Number P?0 RR?6??0 (Dr. Gerald Mohatt, 
PI)  from  the  National  Center  for  Research 
Resources  (NCRR),  a  component  of  the 
National Institutes of Health (NIH). In addi-
tion to the authors named at the beginning of 
the article, the CANHR Field Research Team 
includes: Andrea Bersamin, Anna Goropash-
naya, Scarlett Hopkins, Nick Hubalik, Cecile 
Lardon, Bret Luick, Eliza Orr, Elizabeth
Ruppert, and Chris Wolsko. The authors also 
thank the community field research assis-
tants  and  the  computer/data  management 
and  administrative  staff  members:  Michelle 
Dondanville,  Johanna  Herron,  Jean  James, 
Yongmei  Qin,  Judy  Romans,  and  Yichen 
Wang. Finally, the CANHR team would like 
to express our sincere appreciation to all of 
our study participants and their communities 
for welcoming us and teaching us so much 
about the Yup’ik way of life. Quyana!
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Bert Boyer
University of Alaska Fairbanks
Center for Alaska Native Health Research
Institute of Arctic Biology
311 Irving I Bldg.
Fairbanks, Alaska 99775
USA
Email: bert.boyer@uaf.edu
SHARING POPULATION-BASED GENETICS RESEARCH PROGRESS