Concussion in sport is today regarded as both a public health issue and high profile injury concern in many contact and collision sports. This paper undertakes a comparative review of the current policies and practices of two high profile national sporting organisations of such sports—the Australian Football League (AFL) and Hockey Canada (HC)—in governing the issue as a regulatory concern. By examining the policies and practices of the AFL and HC, this study aims to identify common themes, divergent practices, and nuanced sport-specific approaches to develop understandings on the regulation and governance of this high profile sports injury. The paper aims to contribute to understanding concussion as a regulatory concern, while at the same time recognising the heterogeneity of sport and reinforcing nuanced understandings that align to specific social and cultural settings. We make recommendations based on regulatory and cultural legitimacy. The paper concludes that these NSOs are institutional actors with historical and cultural roots who assert regulatory legitimacy by steering and influencing behaviour and directing the regulatory agenda to manage and mitigate the harm associated with concussion. This paper examines the regulation of concussion in the context of sport governance. Our focus is on the role of the National Sporting Organisation (NSO) as the institutional actor steering and influencing behaviour by setting and directing the regulatory agenda. The paper contributes to understanding the regulatory role of NSOs in responding to concussion as a concern within their sport, recognising the heterogeneity of sport, and nuanced understandings that align to specific social and cultural settings.We acknowledge that other actors participate in this regulatory space. However, in the context of this paper, our goal is to identify and compare the policies, regulations and guidelines as regulatory mechanisms employed by the NSO to achieve the objective of managing and mitigating concussion within the sport. Thus, our research question is what are the similarities, differences, and nuances in the way the AFL and HC, as institutional actors, govern through regulation to manage and minimise the harm associated with concussion?To address this question, the paper proceeds as follows. First, we establish the foundations of the study by identifying relevant theories, conceptualising key terms and briefly framing the public and private dimensions of sport-related concussion. Next, we examine the specific governance characteristics of the AFL and HC as regulatory actors for their sport. As this paper focusses on concussion in sport as a regulatable concern, we then turn to consider the specific policies, regulations and guidelines deployed by each NSO to address the issue within their sport. Specifically, we examine responses across four areas: concussion prevention; concussion injury management; concussion education and advocacy, and concussion research. Our aim is to identify common themes, divergent practices, and nuanced sport-specific approaches. We then propose several recommendations and identify areas for future research consideration including a larger scale comparative study evaluating the effectiveness of NSOs' concussion strategies.As this paper involves a comparative analysis of the regulatory arrangements across two sports, we adopted a qualitative research methodology to understand the similarities, differences and nuances in the way institutional actors govern within their respective regulatory environments. We focus on the policies and practices that address the phenomena of interest , unions, NGOs, various levels of state and government actors, supra-governmental agencies, standard-setting organisations and global organisations  follows a federal model of governance, which aligns with their national governance systems, both of which have British colonial histories  McCrory, . For ourAnother challenge associated with concussion is the latent nature of the injury, often described as an “invisible injury” , and the other focussed on the beneficiaries—those who have a legitimate expectation of receiving the benefits. Indeed, Gostin and Wiley  and the recognised entity for the overall promotion and development of the sport (sport governance). This duality of roles derives primarily from the evolution and development of the sport. In recognition of this dual role, the AFL notes “Our obvious responsibility is to the national competition, but the AFL accepts responsibility for the development to the game, feeder structures and the overall health of the code”  was the original national governing body for the sport from 1906. Originally, members of the Council were delegates appointed by the various leagues which controlled the sport in their States and Territories. The NAFC was replaced by the Australian Football Foundation (AFF) to “administer football nationally on behalf of the AFL” , AFL (Northern Territory), AFLQ for the AFL Queensland, South Australian National Football League, AFL (TAS), AFL (Victoria), and West Australian Football Commission Inc (WAFC). These SSOs are not members of the AFL and have no voting power under the AFL's constitution. Instead, the SSOs are affiliated by agreement with the AFL to administer the sport within their geographical boundaries. These affiliation agreements authorise the affiliates to “promote, develop, manage and control Australian Football” in their state or territory .These affiliates are each separate legal entities. In Western Australia, the WAFC acts as “caretaker” of football throughout the State but also owns two elite level clubs based in the State. This means that in the case of Western Australia, the WAFC is both an affiliate and club owner, operating through a not-for-profit model. Arguably, this enables the WAFC to have greater governance participation in the AFL at least over league governance considerations through its voting power as a member.While each affiliate is recognised as a “Controlling Body” according to AFL Laws, the AFL regards itself as the “keeper of the code” and aims to “actively support” other levels . To illustrate, the AFL's 2013 Member Protection Policy states that “the AFL retains the exclusive power to amend and vary the The 2020 Laws of Australian Football (“AFL Laws”) apply to all matches of Australian Football as organised and conducted by a “Controlling Body”  with responsibility for the governance and administration of the sport in Canada and representation of the country in international competition . It has its roots in the Canadian Amateur Hockey Association (established 1914). The CAHA was formed to oversee amateur hockey at the national level in the country. It started with 21 delegates representing local and regional leagues across the country. Other leagues continued to join over the next 60 years, in large part to be able to vie for the national championship.The Canadian Hockey Association (CHA) was established by the federal government in 1968 specifically to oversee the national teams. The CAHA continued to exist and divided into two tiers based on competitive level. In 1975, the Tier I teams established the independent Canadian Major Junior Hockey League  and connected with the professional level National Hockey League (NHL), which had been in existence since 1917 . The CMJHL separated completely from the CAHA in 1980, retaining only a loose affiliation with the NSO which is now based only on its releasing players for national team participation in the World Junior Championships.In 1994 the CAHA and CHA merged to form HC, the official governing body with responsibility for the regulation and promotion of amateur hockey in Canada on a nation-wide basis, and management of national teams to participate in international competitions. Its mission is to “Lead, Develop and Promote Positive Hockey Experiences.”Today, HC has one class of Members, who are the 13 constituted amateur hockey federations representing each province and territory (“the Members”). The Members elect (and can remove) Board of Directors. They make proposals to, amend or repeal any articles, bylaws, regulations or playing rules of HC. The elected Board of Directors comprises individuals with no formal affiliation with any of the Member federations. Their duty is to regularly review HCs terms of reference and approve all partner agreements.In turn, the 13 provincial/regional/territorial federations are responsible for the governance and administration of amateur hockey within their jurisdictions, and for representing their constituents at HC meetings. They are empowered by HC to manage and foster hockey at the regional and grassroots levels. Each federation has several associational members that oversee minor level (community club) hockey. Within the federal model of sport governance, each level of HC operates hierarchically but semi-autonomously with regard to policy and practices governing hockey within their own jurisdiction. Each federation has in its by-laws something to the effect that the federation and its members, “[in] an unalterable provision… shall operate in a manner consistent with the by-laws, regulations and rules of Hockey Canada” .HC's only affiliation with the NHL professional league  is for the release of players for Canada's participation in the World Championships and Olympic Games.As NSO of the sport, HC has rule-making authority, subject to the constitutional voting rights of its Members as representatives of the 13 provincial/regional/territorial federations. Members have power to vote to accept or reject changes to the Bylaws, Regulations, and Playing Rules by Ordinary Resolution .The governance and constitutional voting rights of members within HC establishes the rule-making authority of HC as the NSO. Nonetheless, the 13 provincial/regional/territorial federations retain discretion over how they implement and enforce the HC ByLaws, Regulations and Rules.Before examining the various approaches and responses to concussion, it is important to briefly explain the differences between the sports of Australian football and ice hockey. We acknowledge that each sport is substantially different to the other and justifies a nuanced approach to injury prevention. We do, however, consider that once a concussive injury is sustained, there are synergies that exist in regard to injury management and return to play or practise protocols, a fact that underpins the principles-based approach established by the international Concussion in Sport Group (CISG) return to play approach  in the Peewee age group (11- and 12-year-olds) and younger. Although there was much concern about the implications of this new rule for players learning how to give and take a body cheque effectively, this change resulted in a 70% reduction in the risk of concussion in minor hockey across Canada. The rule prompted several community-level associations to consider extending the rule to older age groups, although it was ultimately supported in only one city league.Equipment modifications have also been mandated by HC, recognising the nuanced approach to injury prevention given the nature and extent of bodily contact in ice hockey. Canadian Safety Association-certified (CSA) helmets, full face caged protectors and mouthguards are required for all youth programs and recreational leagues , and for all goalkeepers  and older, HC must follow the regulations of the IIHF regarding playing rules and equipment requirements. These regulations are consistent across the two hockey bodies; for example, “clean” bodychecking is allowed and hits to the head are not . The collection of injury surveillance data aligned with early concerns about the cost burden of sports injury in general and their identification as a key health priority focus in 1997 . Any injury surveillance is ultimately dependent on self- or medical reporting, and is made more complex (and therefore less reliable) because of the difficulty diagnosing, and the reluctance in reporting  (ix)]. The latest version of the AFL and AFL Player Association Collective Bargaining Agreement now requires each club to have at least one doctor trained in the diagnosis and management of concussion in accordance with the AFL Concussion Guidelines At the amateur level, the AFL provides some information to assist with concussion injury management  Act]In another example of the semi-autonomous nature of HC's federal model of governance, the Ontario Hockey Federation (OHF) is legally required to also adhere to the guidelines set out by “Rowan's Law,” which is legislation enacted in that province in 2018 ” of their governance structures, that is further strengthened by their broader cultural legitimacy as the “social object [that is] right” (Batuev and Robinson, Yet the regulatory legitimacy of the AFL and HC—the credibility and acceptability as the rule-making authority— may be all the two NSOs have in common. At a fundamental level, they each have different governance models that align with their historical and cultural roots. The AFL, with the dual model of governance, and HC with the federated model, operate under very different rule-making systems that have implications for establishing the regulatory agenda regarding concussion. The AFL's responsibility for league governance attracts a greater proportion of its attention, certainly as far as regulating concussion and the role of clubs as voting members of the AFL captures the AFL somewhat in terms of regulatory oversight. These governance characteristics determine who participates in setting and directing the regulatory agenda and, indeed, the priority or otherwise of what is classified as a regulatable concern. HC involves direct member participation through constitutional voting rights, whereas the AFL Affiliates do not. Consequently, prioritising concussion as a regulatable concern is likely to have been more closely aligned to voting member interests in HC and, until very recently, a more marginal issue for AFL sport governance as a reflection of voting interests within the AFL.Importantly, the NSOs also have different regulatory target audiences in mind; where the AFL remains primarily focused at the elite level while HC takes a broader “whole of sport” approach. Consequently, they approach concussion prevention differently. The AFL is focused on elite level rules, compliance and enforcement, while HC is focused on all of sport rules and equipment, albeit with a particular focus on adolescents. They approach concussion research differently, with the AFL's financial investment in research and HC providing more in kind support through expert opinion. They also approach injury surveillance differently—where the AFL has an established injury surveillance system focused at the elite level while HC does not currently monitor concussion in hockey.When addressing their regulatory agendas for concussion management, the NSOs' collaborations and networks also differ. HC partners closely with public and other private agencies; while the AFL tends to operate autonomously. Consequently, this influences the broader sport-related concussion discussion in their country with HC directly involved in and shaping that conversation, while the AFL is involved more indirectly.Arguably, HC has played a much more active and direct role in collaborating with external stakeholders including state-actors, injury prevention charities and other sports. The HC cross-sport collaboration indicates a “whole of sport” approach, rather than the sharing of medical knowledge by chief medical officers as has been the case in the AFL.For the most part, the AFL has set, directed and controlled how it regulates concussion in the sport. Most of its efforts have been directed at the elite national competition level. Further, while there has been some collaboration with external stakeholders, the AFL has primarily focused within its sport.As noted by Hall , “CanadaBased on the above observations, we make two recommendations in regard to NSOs' policy-making and practices around sport-related concussion. The first recommendation is for NSOs to embrace their regulatory legitimacy—however designated and earned—in establishing and enacting a regulatory agenda with meaningful direction and influence over behaviour. Adopting Black's  conceptuTo illustrate this recommendation in the context of the AFL, we argue that it has the capacity and, importantly, regulatory legitimacy, to support greater engagement in support of stakeholders at the state and amateur levels of the sport. The Affiliates already acknowledge the rule-making authority and standard setting role of the AFL as the NSO. It is plausible, therefore, that the Affiliates already recognise the AFLs regulatory legitimacy over sport-related concussion.The AFL appears to have been constrained by the historical and cultural governance structures that give voting rights to the clubs operating at the elite level in the national competition, creating a form of capture in decision-making. Consequently, the AFL, while carrying dual governance responsibilities, has exercised its policy-making function by addressing concussion with a primary focus at the elite level. Perhaps this is unsurprising when considered in the context of the historical and cultural influences in the sport. However, we argue that by adopting a “whole of sport” approach to concussion regulation, there is potential to yield greater dividends by enhancing trust that playing the sport of Australian football, at any level, is optimally safe for participants.The second recommendation is for NSOs to build their broader cultural legitimacy through leadership by engaging in important public and private issues. As guardians of their sport, NSOs have capacity to build their cultural legitimacy which in turn underpins and enhances their regulatory legitimacy.To illustrate, there is strong evidence in support of the advocacy role of NSOs in raising awareness and mobilising resources to highlight concussion as a public health concern, particularly in youth sport. HC's participation in the FPTSC Concussion group and subsequent involvement in partnerships with non-profit groups and government agencies fostered greater cultural legitimacy across the sport. The AFL, on the other hand, has remained true to its historical and cultural league governance roots, without venturing beyond in any substantial way, to participate in coordinated public advocacy of sport-related concussion.In promoting the public health significance of sport-related concussion, there is much to be gained by stepping outside formal regulatory structures, creating opportunities to restore or enhance trust and cultural legitimacy. For example, when tracing the historical and cultural development of concussion in youth football in the United States, Bachynski  explainsThe above analysis establishes the various ways in which the AFL and HC as NSOs have asserted regulatory legitimacy over the field. Our research has identified the similarities, differences, and nuances in the way the AFL and HC, as institutional actors, govern through regulation to manage and minimise the harm associated with concussion. This comparative study has demonstrated that despite cultural and governance differences, the AFL and HC are both institutional actors asserting their regulatory authority by steering and influencing behaviour to manage and mitigate the harm associated with concussion in their sport.This research has not examined how these actors monitor compliance or enforcement of their concussion policies, regulations and guidelines. Nor does this study assess whether SSOs or affiliates accept the regulatory legitimacy of the NSOs. Future research in this area could proffer insights into gauging views of stakeholders as to the credibility and acceptability of the NSOs policy-making authority, and other key components of the regulatory policy process. Primary data collection, such as through interviews, could build on the foundational assessment, and comparison, provided here. Further, a larger scale comparative study could include an assessment of compliance motivations, enforcement mechanisms and evaluation methodologies to determine the “fitness for purpose” as evaluation goals.Despite the absence of a formal legislative mandate, both the AFL and HC assert regulatory legitimacy aligning with sociological institutionalism. They are institutional actors with strong historical and cultural roots in their sports. Their governance structures, albeit different in nature and form, provide them with the framework through which to exercise their rule making authority. These two sports, deriving from two systems, have the one common goal—to manage and mitigate the risk associated with the harm of concussion in their sport.The original contributions presented in the study are included in the article/supplementary material, further inquiries can be directed to the corresponding author/s.All authors listed have made a substantial, direct and intellectual contribution to the work, and approved it for publication.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Informal caregivers (ICGs) provide care to their family or friends in case of an illness, disability, or frailty. The caregiving situation of informal caregivers may vary based on the relationship they have with the care recipient (CR), e.g., being a spouse or being an adult child. It might be that these different ICGs also have different needs. This study aims to explore and compare the needs of different groups of ICGs based on the relationship they have with their CR. We conducted a systematic review, performing a search in the databases PubMed, CINAHL, and PsycINFO. We included studies with qualitative, quantitative, or mixed-method study designs. We analyzed the data using the thematic analysis method. We included 22 articles . The included articles reported the needs of ICGs taking care of a spouse , parent (adult child ICG), or sibling aged 18 years or above (adult sibling ICGs). We did not include other relationships due to the limited number of articles on these relationships. The most prominent needs reported by the spousal, adult child, and adult sibling ICGs were the need for information and need for support. The three groups differed in their needs as well. Adult child and adult sibling ICGs indicated a need to be acknowledged by the people around them for their role of carer, while they also needed to be seen as an individual having their own personal needs. Moreover, spousal ICGs indicated a unique need of redefining their role and relationship with their CR. Overall, the findings indicate that along with experiencing common needs, the investigated groups have unique needs as well. Knowing the needs of different groups of ICGs can help develop tailored solutions to improve the quality of life of the ICGs and their CR.Systematic Review Registration: [www.crd.york.ac.uk/prospero/], identifier [CRD42020188560]. With the gradual increase in life expectancy and longer periods of disability and chronic illness in people’s lifetime, there is increasing demand for informal caregivers (ICGs) in the health, social, and long-term care systems . ICGs prSpousal ICGs are often co-residing with the CR and are considered in most cases the primary caregivers . They arCaregiving may be challenging . Due to In the literature, the need for information and the need for support are expressed as the most prominent needs by ICGs . StudiesSince the caregiving situation of ICGs can vary based on the relationship they have with the CR, e.g., being a partner or being an adult child, it could be the case that different groups of ICGs also have different needs. As per our knowledge, there is no overview of the literature that compares the needs of several groups of ICGs based on the relationship they have with the CR. Therefore, this study aims to provide a systematic overview of the existing literature on the needs of specific ICGs groups based on their relationship with the CR. Knowledge about the common and unique needs of different groups of ICGs may help health care professionals to provide tailored support to ICGs that could help reduce ICGs’ burden. Moreover, the findings have the potential to help develop and implement solutions that are tailored to the unique needs of the ICGs based on their relationship with the CR.We conducted a systematic review following the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) guidelines . The pro((((“Caregivers”[Mesh] OR caregiv*[tiab] OR ((“Family”[Mesh] OR family[tiab] OR spous*[tiab] OR parent*[tiab] OR husband*[tiab] OR wife[tiab] OR wives[tiab] OR partner*[tiab] OR adult child*[tiab]) AND caring[tiab])))) AND )) AND ).In addition to the advanced search on the electronic databases, SD performed a reference check of the final shortlisted articles to identify the relevant studies that were not found in the database search.Two authors, SD and GF, independently performed the selection of studies in two phases. In the first phase, the titles and abstracts of all articles found in the electronic database search were screened. The author (GF) screened 10% of the retrieved articles in the first phase . To be iIn the second phase, we screened the shortlisted articles for full text. The author (GF) again screened 10% of the shortlisted articles in the second phase. The definition of need was kept broad, including personal  and care-related needs  of the ICGs. Articles describing an evaluation of an intervention program  were not considered. In this study, we define spousal ICGs as caregivers who were in an intimate relationship with their CR; they could be either married, living in a partnership, or unmarried, irrespective of whether they share the same household or not. The studies were excluded when both authors (SD and GF) were convinced that the study did not meet the inclusion criteria. Authors SD and GF experienced disagreement in 5% of the total number of articles screened by GF in the first phase and 2% in the second phase. These discrepancies were resolved with a detailed discussion between the researchers. If no consensus was reached, the articles were screened and discussed by the other authors to reach a consensus.SD and GF independently extracted the data from the selected studies. The researcher (GF) extracted the data from one-third of the selected studies. The following data were extracted and recorded in a table: author name, year of publishing, country, study aim, study design, sample characteristics, the relationship of the ICGs with their CR, illness of the CR, and type of needs expressed by the ICGs . We deveThe quality assessment was performed by two authors, SD and GF independently for all the 22 selected studies. After completing the phase both the authors discussed the discrepancies in selecting the studies. The discrepancies were resolved with a detailed discussion between the researchers. If no consensus was reached, the articles were screened and discussed by the other authors (MH and SD) to reach a consensus. The quality of included qualitative studies was accessed using the Critical Appraisal Skills Program checklist (CASP) . The cheThe quality of included quantitative studies was assessed using the Quality Assessment Tool for Quantitative Studies, recommended by Cochrane . The quaWe used thematic analysis, a technique of narrative synthesis , to syntn = 18) and quantitative (n = 4) studies. The included articles reported the needs of spousal ICGs, adult child and adult sibling ICGs aged 18 years or above. We found a limited number of articles that represented the relationships of parents, grandparents, and grandchildren; therefore, we did not include other relationships. Out of these 22 studies, (i) 11 articles reported the needs of spousal ICGs, (ii) five articles reported the needs of adult sibling ICGs (iii) three articles reported the needs of adult child ICGs, and (iv) three articles discussed the needs of both spousal and adult child ICGs with data analyzed separately. The sample sizes ranged from 6 to 139 ICGs for the qualitative studies and 68 to 862 ICGs for the quantitative studies. The studies were conducted in Australia, Canada, China, Denmark, France, India, Japan, Norway, Portugal, the Netherlands, United Kingdom, and United States.The results of the quality assessment of qualitative studies are presented in The assessment was conducted to improve transparency in the systematic review process. No study was excluded based on the evaluation.The synthesis of the 22 included qualitative and quantitative articles resulted in seven themes of needs . Out of The most prominent need expressed by the spousal, adult child, and adult sibling ICGs was the need for more information in both qualitative  and quanSpousal, adult child, and adult sibling ICGs indicated a need for information about the disease or illness in general , its treAdult child ICGs taking care of patients with a chronic illness indicated a need for information about the long-term implications of the illness so that they can gain control over the unpredictable nature of the illness . MoreoveAll three groups of ICGs expressed a need to be informed about the health condition of the CR . They waSpousal , adult cSpousal ICGs expressed a need to know about their roles and responsibilities as a caregiver . This neAll the three groups reported a need for support from family and friends . SpousalAdult child and adult sibling ICGs also experienced a lack of support from family and friends. They needed their social network to understand their efforts and struggles as caregivers . To gainAll three groups expressed a need for professional support, but the type of professional support needed varied for the three groups. Spousal ICGs mainly required respite care services such as day-care centers or assistance at home  or instiAdult child and adult sibling ICGs needed a professional counselor or health care worker whom they could trust to talk to Both spousal and adult sibling ICGs needed financial help and government benefits to support them in caregiving . They exAll three groups indicated a need to take a break from their caregiving role. They wanted time for socializing, pursuing their leisure activities and holidays . SpousalAll three groups expressed a need to take care of their physical and mental health. They all needed professional support to manage their mental health. A comparative study  on sibliSpousal ICGs taking care of CR with cancer , adult cThis need was indicated by the spousal  and adulThis sub-theme was indicated only by the spousal ICGs. They experienced a decrease in physical intimacy, such as kissing, hugging, touching since the treatment. Moreover, spousal ICGs did not want to disrupt the balance in their relationship by being just a ‘life coach’ for their CR . In one Studies reported that both adult child and adult sibling ICGs expressed a need to be acknowledged as a caregiver by the people around them, including friends, family, and health care workers . The famBoth adult child and adult sibling ICGs struggled to take time away from caregiving for personal activities. A common need for both adult child ICGs and adult sibling ICGs was a need to be more than just a carer and to be individuals who have their own feelings and personal needs . A partiThe goal of this systematic review was to explore the needs of different groups of ICGs based on the ICG-CR relationship. We compared spousal, adult child, and adult sibling ICGs to identify their common and unique needs. After performing a comprehensive search of the literature, we included 22 articles in our systematic review. The analysis showed that multiple articles from all three groups reported the need for information and need for support. All three groups needed information about the CR’s illness, health condition of their CR, and service availability such as home assistance and respite care services. All the groups also expressed a need for social support from family and friends and supportive care from professionals. Spousal and adult sibling ICGs also needed financial help to support them in caregiving. The three groups differed in their needs as well. It is noteworthy that all the qualitative articles targeting adult child ICGs and multiple articles targeting adult sibling ICGs indicated a need to be acknowledged by the people around them for their role of carer, while they also needed to be seen as an individual having their own personal needs. Moreover, multiple articles from spousal ICGs indicated a unique need of redefining their role and relationship with their CR. Before we discuss and interpret our findings, it is important to highlight possible biases that could have influenced our findings.Out of the 22 included articles, only three articles directly compared ICG groups  . Self-evIn addition, articles differed in the demographic characteristics of the participating ICGs, which could again influence the needs. For example, the needs of younger ICGs may differ from those of older ICGs, or the needs of ICGs residing with the CR may differ from those living far away. Thus, considering the scarcity of studies that directly compare different types of ICGs and the heterogeneity in the articles included, cautiousness in drawing conclusions is needed. Nevertheless, when we looked at the articles directly comparing the spousal and adult child ICGs, their findings seem to be in line with those of articles studying individual groups of ICGs. For example, the comparative study by All three groups indicated the need to know about the availability of services. This need was expressed by spousal and adult sibling ICGs in a qualitative study, and by adult child ICGs in a quantitative study, therefore the service needs for spousal and adult sibling ICGs were more descriptive in nature. They indicated the type of services they needed, such as home assistance and respite care, whereas the adult child ICGs only indicated a need to know about the services available and where to find them. The literature indeed suggests that ICGs lack awareness about the availability of services , and thiAll three groups expressed a need for social support from people around them and supportive care from the professionals. Interestingly, spousal and adult sibling ICGs expressed an additional need for financial support, which was not indicated by adult child ICGs. This need is in line with the literature that spousal ICGs experience higher financial strains than adult child ICGs and other relationships . This coThe need to be acknowledged was expressed only by the adult child and adult sibling ICGs. Our findings are in line with the literature that suggests that spousal ICGs by default are acknowledged as caregivers by society because of the nature of their relationship with the CR . Adult cAdult child ICGs expressed an additional need to detach themselves from the responsibilities of caregiving. The two articles that indicated this need included a population of mostly young adult caregivers (YACs), the age of these ICGs ranged from 18 to 30 years. As we know from previous literature, young adults in this age group are in their transition period between being young and at the same time entering adulthood, establishing themselves with respect to certain aspects of life such as education, career, relationships, and social life. This period may be more challenging for YACs who have to integrate caregiving with other aspects of life . We knowWe noticed several strengths and limitations of our review. Our review explored the needs of all the ICG-CR relationships in the literature, such as grandparental ICGs or extended family ICGs. After an extensive search of the literature, we ended up with multiple articles for spousal, adult child and adult sibling ICGs. Another strength is that all the included articles were checked for their quality using validated and standardized measures. Lastly, we pre-registered our review in PROSPERO before conducting it, thus, fostering the transparency of the systematic review process . A limitWe also found some strengths and limitations of the literature included in this review. One strength is that most of the articles were qualitative studies, which made the data descriptive in nature. The richness of data offers an in-depth understanding of the needs of different groups of ICGs, allowing us to make a good comparison between the needs of different groups of ICGs. We also found that most of the included qualitative articles were of high quality with respect to clarity and appropriateness of study aim, qualitative methods, recruitment strategies, data collection methods, and statement of findings. However, overall, quantitative articles were a mix of moderate and weak quality. It needs to be noticed that the quality is indicated based on the information reported in the included article and not on the actual quality of the studies. A limitation is that the included studies in this systematic review are mostly conducted in western countries, except for four studies that were conducted in Asian countries, that is, Japan , India  and TaiwMoreover, regardless that we explored all types of relationships in this review, we found only one article for another ICG-CR relationship, namely for parental ICGs taking care of an adult child aged 18 years or older . The limIn our study, we included all the needs of ICGs based on their relationship with the CR except for the needs of ICGs toward an intervention or program. Although, we encountered several articles on this topic while screening the articles, therefore, for future research, it would be interesting to compare the need for interventions and programs among ICGs groups. This will contribute in enriching and supporting the literature on needs of ICGs groups. Thus, help in tailoring the interventions and programs based on the unique needs of these ICGs specifically toward an intervention.The synthesis of the 22 included articles resulted in seven themes of needs among spousal, adult child, and adult sibling ICGs, i.e., a need for information, support, personal time, managing personal concern such as help with managing their health, to maintain their relationship with their CR, to be seen as carer, and to be acknowledged. The three groups of ICGs represented certain common as well as unique needs. By knowing the common and unique needs of the different groups of ICGs, we can offer more targeted and personalized support to the ICGs, and design targeted interventions in the future. This might help to improve the quality of life of both the caregivers and that of their care recipient.The original contributions presented in the study are included in the article/SD and GF screened the relevant article for eligibility based on the inclusion criteria. SD, AL, GF, and MH conducted the thematic analysis to synthesize the data for both qualitative and quantitative studies. SD, AL, and MH drafted the article. GF and GL commented, contributed, and edited the subsequent drafts. All authors contributed to the article and approved the submitted version.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


The aim of this paper is to analyse the heterogeneity and the performance of the value-creating strategies applied by dairy farms in their diversification initiatives. This is a pertinent topic to study because the Common Agricultural Policy provides a role for value-added diversification strategies as an alternative to ensure the economic viability of small farms. Furthermore, the fostering of specialised policy initiatives requires empirical evidence on specific diversification strategies. Nevertheless, there are still few quantitative studies analysing the factors that influence the success of value-added initiatives in terms of their contribution to economic performance, which may be due to the difficulty of obtaining databases such as the one handled in this study. Our findings confirm that the strategies are associated with different combinations of contexts and entrepreneurial capabilities. In fact, the results of this research present practical implications for future entrepreneurs and for policymakers, contributing towards understanding how an adequate alignment between contextual factors and entrepreneurial capabilities can enable the implementation and sustainability of new business initiatives in rural areas. We also conclude that the diversification ventures of dairy farms are an appropriate element for guaranteeing the survival of certain local breeds, making their use profitable.This paper explores different value-creating strategies (VCS) used by dairy farmers engaged in on-farm diversification ventures. In order to explicitly identify the informal strategies followed by 49 farmers in their value-added ventures, we applied a theoretically informed business model framework combining three dimensions: value proposition linked to local food, customer engagement via quality schemes and shorter supply chains, and the key capabilities of the entrepreneur. Using cluster analysis, four different types of VCS were identified and labelled as ‘Ecological’, ‘Single-product’, ‘Innovative’, and ‘Traditional’. Whilst we found that these strategies are influenced by contextual factors and the owner’s entrepreneurial skills, in general, we did not observe significant differences in performance between them. The results suggest that farmers respond entrepreneurially to sectorial changes adopting those VCS that tend to align with their entrepreneurial capabilities and context, thus enabling them to succeed with any of the strategies pursued. Hence, our work contributes towards clarifying the relationship between VCS, entrepreneurial capabilities, and context. This is important for farmers and policymakers because it reveals the diversity of farm management and the resilience of farm systems. As a result, the potential challenges for Single-product VCS and Traditional VCS are discussed. This study investigates the relationship between value-creating strategies (VCS), linked to contextual and personal factors, and economic performance in dairy farm diversification processes. The restructuring of the European agricultural sector resulting from globalisation and various Common Agricultural Policy (CAP) reforms has forced numerous farmers to deploy strategising and marketing skills, where the latter is required to adopt a stronger entrepreneurial orientation (EO) in order to guarantee the sustainability of their business . SpecifiRegarding the EU dairy sector, CAP reforms since 2003 have led to a reduction in intervention prices, so the price received by the farmer is determined to a greater extent by market forces, becoming more in line with world prices. Although the Spanish production of cow’s milk has grown from 2010 to 2020 by 27%, especially since the abolition of the quota system in 2015, Spain continues to present a deficit, with national production covering 77.5% of domestic consumption in 2020 . The maiThese changes in the dairy sector provide a role for value-added diversification strategies as an alternative for ensuring the economic viability of small farms . This isA substantial body of literature exists on agricultural diversification ,15,16 anExisting studies show that more diversified farmers have stronger entrepreneurial identities and employ more complex business strategies than farmers who do not diversify ,31. HoweAlthough previous studies have captured the different entrepreneurial approaches adopted by farmers . Considering the critical role of context on entrepreneurial activities ,43, thisRQ1: What types of VCS are used by dairy farmers engaged in on-farm diversification? RQ2: How are VCS associated with different contextual and personal factors?RQ3: Are there differences in performance between VCS?Our study focuses on the diversification ventures but not on the activity of the farm as a whole. Therefore, the data used in the empirical analysis refer fundamentally to these initiatives. Although, in the Spanish dairy sector, most of the production is sold directly to the processing industry, a certain number of farmers have embarked upon diversification strategies.This study centres on the four regions of Northern Spain , where 79.3% of the dairy farmers and 55.1% of Spanish dairy production were located in 2020 . We idenWe designed a questionnaire for the purpose of collecting information via personal interviews. For its design, we reviewed the literature ,50,51,52Each of the 49 farmers was interviewed on their own farm, a process lasting two hours on average. The meetings included informal discussion prior to and following the formal interview, which were recorded and transcribed. Prompts were utilised to ensure the data collected covered the research objectives, but discussions were allowed to flow as freely as possible. The data collection through face-to-face, in-depth interviews allowed us to complement the study with narrative elements important in the strategic formulation. Additionally, we were able to identify the farmer’s motivations for diversifying as well as analysing the context in which the initiative was developed. The data, collected in 2012, refer to 2011. The dairy farms visited reported an average raw milk production volume of 518,000 litres, which represented 178% of the average production per dairy farm in Spain. However, there was a large dispersion in this variable between the different VCS identified. The average volume of processed milk per farm amounted to 238,548 litres; 87.2% of the milk used for the elaboration of different products was cow milk, and the remainder was goat or sheep milk. The average investment dedicated to the processing and marketing of dairy products reached a value of EUR 257,500 per farm, 76% of this being allocated to assets related to milk processing and the rest to the marketing and distribution of products. The average number of workers per farm dedicated to activities related to the processing and commercialisation of products was 3.1, and these workers accounted for 64% of employment in the sampled farms.The average value of sales per farm was EUR 256,808. The average sales structure by product type indicated that cheese represented 54.1% of total sales, fresh milk 22.5%, yogurt 11.3%, and other products the remainder. The average sales structure in terms of marketing channels reflected sales to end consumers of 35.2%, with hospitality, shops, and large-scale distribution representing 21%, 30%, and 13.8% of sales, respectively. Products with an organic label accounted for an average of 26.8% of value-added venture sales, while sales with a PDO certification represented an average of 22.1% of sales per venture.Given that most of these farmers do not adopt a formal strategic planning approach , our empBMC is structured around four main areas: value proposition, customer, infrastructure management , and financial aspects. We adapted the framework to the objectives of this study by choosing ten strategic and operational indicators for three areas: value proposition, customer, and key activities. Firstly, for each farm we measured the ten indicators to explore the different groups of VCS. Next, we analysed the differences in the performance indicators between these groups. We excluded the financial area because our objective was to identify groups in line with the strategic and operative factors that explain the value creation. If we included performance variables in the cluster analysis, the groups would be defined by these variables. Finally, the revenue model is a result of the operative and strategic model . A value proposition can be defined as the “expression of the experience that a customer will receive from a supplier’s measurably value-creating offering” . As suchThe customer area is related to customer engagement, defining the targeted audience, the distribution channels, and the type of relationship the company maintains with their clients. The definition of the term ‘local food’ applied in this study refers to the quality dimension, as opposed to the geographical dimension, of the concept. Under this definition, the product is not necessarily consumed in the same region or locality of origin and “is identified and distinguished using product labels, certification systems and other production parameters such as artisanal, traditional, farm based, organic or natural to define and differentiate the quality of the specific product coming from a specific geographic area” . RegardiRegarding the key activities, the business model framework takes into account the internal capabilities that give rise to a competitive advantage. A firm’s competitive advantage may derive from either a better execution of certain activities within the value chain, from a better coordination of these activities, or from a better management of its networks . PreviouSmall firms, including farms, manage knowledge informally as part of their normal activities without the use of terminology and concepts of knowledge management . This idParticipative management is characterised by employees being involved and committed in the decision-making process, and this must be accompanied by a reward system linked to improving firm productivity and customer satisfaction ,49,50.For the dairy farms focused on value-added processing, innovation does not imply outlays of R&D, but instead it is implemented, for example, by increasing their portfolio with higher-value-added products or by differentiated packaging , or by tThe use of networks facilitates access to information, resources, and markets. Its importance will be greater for small and rural enterprises, which face less favourable conditions in terms of location, information sources, market proximity, and the possibility to develop their own R&D . VariousTwo types of variables were used: (1) factors extracted from a principal component analysis based on Likert-style questions (from 1 to 5) and (2) variables measured directly from interviews or calculated using the information gathered, as in the case of the economic indicators of costs and margins. Each construct or factor was measured through various items, using previous papers as a reference and, in some cases, adapting these to the characteristics of the dairy farms. The constructs used in the analysis and the references on which they are based are presented in Regarding performance, we used a qualitative measure based on the subjective perception of the interviewees, as well as three quantitative measures determined using accounting data. The factor denominated ‘performance’ was measured using three items, so that farmers were asked what they considered to be the situation of their farm compared with competitors in terms of (a) optimisation of the investment in assets, (b) competitiveness of the farm, and (c) profitability of the farm. In order to measure each item a scale was used, ranging from 1 ‘very inferior to competitors’ to 5 ‘very superior to competitors’. The three quantitative variables based on accounting data were margin per litre, profit per litre, and value added per worker.Entrepreneurial Orientation (EO) refers to a firm’s strategic orientation, reflecting the decision-making styles, practices, and methods that direct its activities . EO is dInnovativeness and product innovation reflects the firm’s tendency to embrace new ideas, favour change, and encourage experimentation . PreviouThe economic motivation behind agricultural diversification better reflects the willingness of the farmer to assume the risks of starting a business, with a higher risk of failure compared to social or lifestyle motivations .Given that the proactiveness of EO most closely resembles the ideas suggested by Miles and Snow’s  prospectPrevious research found that EO has a positive influence on the capability of an organisation to manage their knowledge ,72. On tAdditional information on the productive and commercial characteristics of the diversification initiatives has also been obtained.We used principal component analysis to obtain the constructs shown in The differences between VCS were first analysed using the Kruskal–Wallis test, which detects whether at least one of the VCS is different from the rest. Next, we used Dunn’s test to analyse the existence of significant differences between the specific pairs of the VCS. In the case of dummy variables, Pearson’s chi-square (χ2) test was used. We used the software IBM SPSS Statistics 24.p < 0.05) for factor analysis to be suitable, which occurred for all the factors. The first research question concerned the types of VCS used by farmers engaged in diversification initiatives. k, with k being the number of variables to be used in the classification, we decided to use five variables (25 = 32 < 49) instead of seven to perform the cluster analysis, which included number of product references, sales of fresh milk, sales of PDO, sales of organic products, and product innovation; the variables were standardised [Using cluster analysis, four types of VCS were identified in order to study their differing characteristics and performance. We used the K-means technique to carry out the cluster analysis, using variables from dardised . We deciIn order to determine the number of groups to be obtained with the cluster analysis, we applied the silhouette coefficient, which is a measure of both cohesion and separation, and the Akaike information criterion; both measures supported the choice of four groups see . In lineIn These ventures are based on organic farms with a high degree of orientation towards the environmental dimension. Direct sales represented 41.4% of total sales, proving to be the VCS with most sales outside the region and with the greatest weight of yogurt in its production mix. The farms of this VCS stood out because of their high motivation towards a direct relationship with the final consumer and, with this objective in mind, the development of initiatives to a much greater extent than the other VCS. Thus, in this VCS 63.4% of the farms adopted initiatives that favoured direct communication with the customer, such as agro-tourism, farm interpretation centres, guided tours of farms, museums, shops on the farm, etc., as represented by the variable relational diversification. The Ecological VCS is characterised by social motivations rather than seeking high-growth business opportunities. This VCS also had a high level of participative management, implying that employees were involved and committed in the decision-making process. In our sample, the Ecological VCS also presented the highest percentage of managers with university studies (54.5%).The Single-product VCS is characterised by its focus on fresh milk sales . The majority of farms of this cluster maintained vending machines as the principal channel for commercialising fresh milk. These farms had a low level of certification . They had a high percentage of direct sales, which requires a bigger marketing effort, observable in the form of a larger number of workers and investments dedicated to these activities, reflected in a greater weight of fixed costs for commercialisation purposes. These farms are based on a single product and offer a reduced number of product references. This VCS consisted of farms with high milk production , but the milk processed by the value-added venture represented just over 20% of overall production. In general, the milk production of these farms is based on intensive systems, characterised by highly productive cows, and a great dependence on both external inputs  and the dairy industry. The farms in this VCS only used cow’s milk, coming in almost 90% of the cases from Friesian cows. This contrasted with the other three VCS, where milk from goats and sheep was used to a greater or lesser degree together with that of other cow breeds, generally more rustic and adapted to the local environment, such as Galician Blond or Brown Swiss, which provide a differential quality to products, both physical  and intangible (image).The Single-product VCS comes with a high economic motivation, which tries to add value to milk production and seeks to reduce the great dependence on the dairy industry. Due to this, diversification in these farms, especially over recent years, tends to be dependent on push factors . The diversification is opportunity driven (“pull factor”) when new activities are started because the farmer has seen a business opportunity. The diversification is necessity driven (“push factor”) when the farmer has to diversify in order to secure family income or decrease risks caused by changes in the market situation .In general, according to their narratives, these farmers trust in the quality of their primary production, and in their technical skills and management capacity. However, they face difficulties in differentiating themselves from the dairy industry due to, among other reasons, the minimal transformation of production (they sell fresh milk), a narrow range of product references, as well as their defensive strategy (61.9% of the cases).These cases display a high degree of innovative behaviour. Their production featured a low level of product certification , with cheese representing their most important product . This VCS had the highest number of product references due to an innovative effort aimed at encompassing different segments of interest.The innovative VCS showed the highest value of milk processed . In some cases, this percentage exceeded 100%, requiring the purchase of milk from outside the farm to cover demand.Although this VCS presented a low level of products certified , farmers are able to use the farm’s resources and features in flexible and innovative ways to create value .These farmers are motivated by pull factors and are very oriented to the search of business opportunities, most of these ventures being run by prospectors and analysers. These ventures are innovative and proactive, seeking new market opportunities. Taking into account what was stated by the interviewees in relation to new projects, as a reflection of the EO of these ventures, several initiatives can be cited that try to take advantage of different market opportunities: increase in the product range, different product sizes, innovation in packaging, benefiting from regional tourism revitalisation plans, use of their own distribution to market other agricultural products, establishment of their own stores in cities for product sales, participation in benchmarking and innovation projects, etc.These initiatives are older than those of other VCS, being dedicated to the production of traditional cheese well-known in the market due to PDO protection. Since the PDO label is an instrument that reduces the asymmetric information problem between producers and consumers, if the collective reputation of the product is good, this label will be a powerful tool for signalling quality . This haMany of these farms produce cow, goat, and/or sheep milk and possess good technical skills in relation to the production of traditional cheeses, as they have extensive experience in dealing with consumers through participation in local markets.Most farms in the Traditional VCS develop defensive strategies, focusing on traditional products and established markets. They have a low level of participative management. Since the business model considers the key resources and activities for developing the value proposition delivered to consumers, it is possible to highlight the entrepreneurial capabilities involved and the context of each VCS, both summarised in x-axis reflects the context as the level of institutional protection, while the y-axis shows the level of entrepreneurial capabilities.Based on the analysis presented in previous sections, we have identified four VCS within the value-added initiatives. This can be illustrated graphically via two dimensions related, on the one hand, to the type of context and, on the other, to the level of entrepreneurial skills of the farmers. In the four VCS, we observed different contexts determined by factors such as regional location, dependence on the dairy industry, the level of compliance with the requirements to be able to use a quality certification as a PDO or the organic label, and the level of intensification of the farm’s productive system. Given that the context incorporates multiple dimensions, such as business, social, spatial, and institutional , in ordeFurthermore, our analysis describes different levels of entrepreneurial skills or capabilities between the four VCS. We show VCS as Single-product or Traditional, where technical and managerial skills tend to predominate, in contrast with Ecological and, especially, Innovative VCS, characterised by a high level of entrepreneurial skills.The use of the Miles and Snow typology allowed us to observe how the defensive character predominated in the Single-product and Traditional VCS, which is related to a low proactive character and, ultimately, to a low level of entrepreneurial skills ,80. TradIt is also observed that, in similar contexts, the personal characteristics of the entrepreneur determine whether the VCS adopted is different. In this sense, there are ventures that, due to the spatial context in which they operate, could belong to the Traditional VCS but, due to the farmer’s EO, fit better into the Innovative VCS. Likewise, farms that could be certified as organic are not and remain in the Traditional VCS because the entrepreneur prefers to continue with a defensive strategy and does not wish to affront changes in their productive system.The third research question explored differences in performance between VCS. According to the Kruskal–Wallis test, significant differences only existed for the variable margin per litre. Based on the results of Dunn’s test, the Ecological and Innovative VCS presented a greater margin per litre than the Single-product VCS, just as the Ecological also surpassed the Traditional. Nevertheless, no significant differences were found for the profit per litre, value-added per worker, and qualitative performance. The Single-product VCS corresponded to younger ventures focused on fresh milk, many of which were created from 2009 onwards for the distribution of milk to consumers through vending machines. The evolution from this VCS to others with greater value-added products and the use of different channels requires more entrepreneurial skills. Our results indicate that it is possible to add value to primary production through various VCS and with different EO. For example, Traditional VCS farms generate value with a lower level of EO because they meet the requirements for benefiting from the advantages of the PDO certification. On the other hand, Innovative VCS farms that mostly work with conventional milk and without the protection of a PDO or the organic label operate with the highest levels of EO in order to be successful in their economic activity.The growth of milk production experienced by Spain in recent years accompanied by a significant reduction in the number of farms has accentuated their specialisation, leading to more intensive production systems, with greater dependence on the dairy industry and external purchases. These types of specialised farms present a high market risk, being very vulnerable to changes in milk prices ,23. ValuThis study contributes to the field of agricultural entrepreneurship and farm diversification by uncovering the heterogeneity of the value-added strategies followed by dairy farmers in a changing environment and revealing how they seem to align their capabilities and contexts. To do this we adapt an investigative framework based on the BMC that shows the informal strategies followed by dairy farmers in their value-added ventures . Since tWe used quantitative and qualitative data from 49 Spanish dairy farms involved in diversification via the processing and marketing of dairy products . Our database offers suitable material in order to address the research questions posed. First, although all farmers are in the dairy sector and they elaborate similar products, the granularity of the data allows us to describe the features of their different VCS. The in-depth interviews have allowed us to complement the study with narrative elements important in strategic formulation and in context analysis. The combination of the farmers’ comments and the experience of the researchers helped to properly interpret the results obtained. Second, because the production and distribution costs linked to the activities of the processing and marketing of the dairy products are available, we can measure the real contribution of the value-added activities to the farm’s financial performance. While the collection of qualitative data allows us to capture the richness and diversity of the context, the use of quantitative methods offers legitimacy to our results . In thisFurthermore, this paper provides a worthwhile lesson by underlining the importance of developing management accounting systems in the context of farm diversification initiatives. The systematic collection of variables such as those used in this work permits the construction of datasets, currently unavailable, that allow a systematic evaluation of the effectiveness of diversification initiatives. These also represent an important source of information for monitoring the political measures proposed in the farm-to-fork strategy . AlthougOur study shows that there are contexts where success can be achieved with a weaker EO due to greater institutional protection provided by labels such as PDO . In other cases, where the conditions to take advantage of institutional protection are not fulfilled, acting with a greater EO is essential in order to differentiate products in the market and ultimately achieve success . The Single-product VCS showcases the risks and problems of operating in the market without institutional protection (via PDO certification or organic label) and with a weak EO.It should be borne in mind that barriers to entry and to mobility exist when operating with the Ecological, Innovative, and Traditional VCS. Hence, the Ecological VCS requires a milk production certification on the farm in accordance with the regulation established by the European Union. The aforementioned certification for a PDO is associated with certain geographical regions and production standards, whilst the Innovative VCS requires time for the adequate establishment of networks, product innovation, and a market presence through various channels with new product references. Our study reveals interesting results related to how value is produced and how it is apparently related to the farmer’s own values. In our sample, most of the farmers from Single-product and Traditional VCS expressed that they consider their future dependent on the high quality of their products. Nevertheless, they seem to pay scarce attention to the management of the intangible and symbolic aspects of quality agri-food products. In these cases, a reduced level of entrepreneurial skills was observed, with attention focused on a single resource . Precisely, the most specialised farms, with the highest production volume, corresponding to the Traditional VCS and especially the Single-Product VCS, were those that have a strong focus on production techniques (engineering approach), trying to improve labour productivity, while the Ecological and Innovative VCS presented a greater EO, assigning more importance to a marketing approach .With reference to the engagement in socio-material aspects, our typology reflects how farmers respond to the challenges and potential of rural activity in a diverse manner . The resMoreover, in line with more recent research , differeIn particular, in the case of the Single-product VCS, integrated by recent initiatives, despite weighing a high value on the product innovation factor, a low level of entrepreneurial skills is observed. This is related, among other things, to the low score in knowledge and participative management factors, the predominant character of the defensive strategy, and the reduced consideration of intangible assets. In the years prior to 2011, the reduction in the price received for milk sales to the dairy industry pushed a large portion of the farmers belonging to the Single-product VCS towards diversification in an attempt to maintain their income level, this despite a low level of entrepreneurial skills.Consequently, in an increasingly open and dynamic market, farms in the Single-product and Traditional VCS should try to create value strategies that enhance their market orientation. New products and product characteristics, services, brand names, or unique customer experiences may create additional value for farms. A suitable combination of tradition and innovation could be the source of greater business initiatives with very positive effects for regional development. In this process of the recombination of resources, there are farmers who contribute more than others to enhancing the value of the rural area, becoming agents of rural change, leveraging on spatial resource endowment and spatial bridging . These tThe development of skills for the implementation and management of diversification initiatives is a relevant issue, which should be treated appropriately based on the age and previous experience of the farmers, taking into account that older farmers could show greater resistance to change . Due to We understand that this work makes an important contribution to political decision makers, farm advisers, and farmers, by highlighting heterogeneity in VCS of diversification initiatives, which has made it possible to observe that different solutions and measures are required depending on the contexts and entrepreneurial skills present in each case. This has clear implications for guiding decision-making processes and public subsidies in order to improve the sustainability of farms.The use of dairy cows of more rustic breeds and adapted to the local environment, such as Galician Blond or Brown Swiss, provides tangible (more fat and protein) and intangible (image) elements that contribute to the differentiation of the value-added strategies followed by dairy farms. In this sense, the diversification ventures of dairy farms prove an appropriate element for guaranteeing the survival of certain local breeds as well as making their use profitable.This paper is focused on milk production, a traditional sector of European farming, which is currently facing major changes. We study the incorporation of value added to the agricultural product through food processing and direct marketing, a type of diversification promoted by the EU and supported by the CAP . Using tThe VCS are associated with contextual factors such as the region, compliance with the conditions for producing with a label of quality , and the level of intensification of the farm productive system. The characteristics of the entrepreneur and their motivations also influence how the entrepreneurial activity is carried out, in such a way that farms in similar contexts and with different entrepreneurial skills and motivations opt for different VCS. For example, some cases, due to their location, could belong to the Traditional VCS but, due to the level of entrepreneurial capabilities, instead belong to the Innovative VCS.In relation with the performance of each VCS, results reveal that there are no significant differences between the VCS in terms of profit per litre, value added per worker, and qualitative performance, while the Ecological and Innovative groups present the highest margins per litre. However, a more open market means potential challenges for Single-product VCS and Traditional VCS, which could be tackled with the incorporation of greater entrepreneurial skills.In terms of implications for practice, our results are useful not only for those farmers planning these types of diversification initiatives but also for the public administrations interested in promoting them. The analysis suggests that policymakers should consider the heterogeneity of agrarian diversification initiatives, given that the various VCS in turn exert differing impacts on the development of rural areas. Our typology of ventures could be useful as a starting point for discussions concerning how to support entrepreneurship in farms through policy measures . In thisFinally, this study presents several limitations, among which are the following: the small size of the sample, the availability of data for a single year only, and lack of information about the farmer’s family characteristics. Future work is required to analyse the evolution of the VCS for farm diversification over multiple years, as well as their possible effect on performance. In particular, since the VCS are not static, a detailed examination of their temporal evolution proves of great interest. Additionally, gathering more information regarding the characteristics of the farmer and his/her family is essential for analysing the role played by these in the VCS to be applied in agricultural diversification . The lat


The use of genome-wide sequencing in pediatric medicine and research is growing exponentially. While this has many potential benefits, the normative and empirical literature has highlighted various ethical issues. There have not been, however, any systematic reviews of these issues. The aim of this systematic review is to determine systematically the spectrum of ethical issues that is raised for stakeholders in in pediatric genome-wide sequencing.A systematic review in PubMed and Google Books (publications in English or German between 2004 and 2021) was conducted. Further references were identified via reference screening. Data were analyzed and synthesized using qualitative content analysis. Ethical issues were defined as arising when a relevant normative principle is not adequately considered or when two principles come into conflict.Our literature search retrieved 3175 publications of which 143 were included in the analysis. Together these mentioned 106 ethical issues in pediatric genome-wide sequencing, categorized into five themes along the pediatric genome-wide sequencing lifecycle. Most ethical issues identified in relation to genome-wide sequencing typically reflect ethical issues that arise in general genetic testing, but they are often amplified by the increased quantity of data obtained, and associated uncertainties. The most frequently discussed ethical aspects concern the issue of unsolicited findings.Concentration of the debate on unsolicited findings risks overlooking other ethical challenges. An overarching difficulty presents the terminological confusion: both with regard to both the test procedure/ the scope of analysis, as well as with the topic of unsolicited findings. It is important that the genetics and ethics communities together with other medical professions involved work jointly on specific case related guidelines to grant the maximum benefit for the care of the children, while preventing patient harm and disproportionate overload of clinicians and the healthcare system by the wealth of available options and economic incentives to increase testing.The online version contains supplementary material available at 10.1186/s12887-021-02830-w. Genome-wide sequencing, as whole exome or whole genome sequencing (WGS/ WES), can be used to identify variations in a person’s genetic code that might lead to impaired development and disease or disability, that might be ‘otherwise undetectable through clinical history, physical examination, and biochemical or metabolic tests’ . With geIn addition to the potential benefits of genome-wide sequencing in pediatrics, however, the empirical and normative literature has also highlighted a number of important regulatory and ethical challenges –24. ThesTo date, however, there have not been any systematic reviews of these ethical challenges. Although there have been previous review articles conducted on ethical issues in genome-wide sequencing , 28–30, With the increasing use of genome-wide sequencing, non-genetic medical specialties, such as pediatricians, are also increasingly confronted with it: They are often the first to see and know the affected patient and their families best; they provide pre- and post-test care; they are able to make a referral to a geneticist or, in some countries, can order genome-wide sequencing of their pediatric patients themselves. It is important that these health care practitioners have a comprehensive overview of ethical issues that may arise to guide their decision-making. This systematic qualitative review aims to determine systematically the spectrum of ethical issues that is raised for stakeholders in in pediatric genome-wide sequencing.The methods of the study are presented in accordance with the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) as far as they are applicable to qualitative analysis .To be included, publications had to describe and/or assess an ethical issue involved in genome-wide sequencing with children via either conceptual or empirical methods. The definition of ethical issues was based on principlism  which ha. Furthermore, publications needed to be a journal article, book or book chapter, or a national-level report published from April 14, 2003 to May 28, 2021. The date limit was added at the last step of the PubMed search, all other inclusion criteria were applied when screening Google Books and PubMed results. Publications before that date were excluded, because the Human Genome Project had not been completed [Due to the composition of our research team, only publications in English or German were includedompleted . The metThe search terms were developed through an iterative process, where combinations of key words and MeSH terms were piloted in PubMed and the results were assessed for inclusion of a known set of representative literature. This resulting combination of key words and MeSH terms included in the search strategy are presented in Table Based on the inclusion criteria, JE along with either SM, DS or BZ independently screened all titles and abstracts in order to assess their eligibility for inclusion for full text screening to insure inter-rater validity. Furthermore, JE and SM screened the back cover descriptions and tables of content of the Google Book’s hits and excluded those not containing any relevant chapters. In case of disagreement, consensus was reached discursively. Full texts of potentially eligible studies were then screened by JE again along with either SM or IK. Excel sheets were used for the entire screening process. Any discrepancies between reviewers during the screening process with regard to the inclusion/exclusion of articles was resolved by consensus.Included full texts were analyzed using conventional qualitative content analysis . Findingn = 137) were journal articles and the remaining (n = 6) were book chapters. The vast majority of included publications were published after 2014. A list providing full bibliographical information of all 143 publications is provided in Additional file The literature search identified 3175 publications of which 143 were included in the final analysis , whether there should be different forms of consent and directiveness in counselling depending on the urgency of the situation, the challenges of parental decision-making on behalf of their child, and the challenges to decide how much choice parents should have regarding what types of findings are received.Issues related to sequencing, analysis and interpretation: Here the ethical issues include challenges regarding the infrastructure, such as the risk of inconsistencies and variability due to different bioinformatics pipelines; challenges regarding the interpretation of variants due to the gap between the amount of data which are generated and the knowledge to use them in a clinical context; and the challenge to decide whether it should be actively searched for a certain list of disease-associated genes in every genome-wide sequencing.Issues related to communicating results: The main ethical issues repeatedly raised here relate to the challenge of reporting unsolicited findings (UFs), e.g. the risk of undermining the parents/participants/patients right not to know with an obligatory disclosure of certain UFs; the challenge of balancing the best interests of the child with the best interests of the family regarding the disclosure of UFs; the risk of the diagnosis negatively impacting the parent-child bonding; and the challenges of thoughtfully and effectively framing the results.Issues related to future use of data: These ethical relate mostly to the challenges of data sharing, storage and governance, such as the challenge to determine whose responsibility it is to initiate reanalysis, provide access and recontact patients/parents/participants , the risk to privacy and the risk of risk of genetic discrimination .A total of 106 distinct ethical issues in the application of genome-wide sequencing in a pediatric population were identified Table . The maiTo our knowledge, this is the first systematic qualitative review of the full spectrum of ethical issues in pediatric genome-wide sequencing discussed in the literature. Most ethical issues identified in relation to genome-wide sequencing typically reflect ethical issues that arise in general genetic testing in children –82, but The challenges surrounding UFs are a good example of this and were one of the most frequently discussed ethical issues in the literature. UFs have intensified tremendously in genome-wide sequencing, as the likelihood to generate them and their sheer number have increased a lot. These challenges are even bigger and more in pediatric genomic-sequencing as parents then have to make the decisions regarding the reception of UFs for their child. Issues frequently identified in this review include how much choice parents should be given regarding which findings they want to receive for their children; the risk to undermine the right not to know with an obligatory disclosure of UFs; the challenge of interpreting and balancing the best interests of the child with the best interests of the family regarding the disclosure of UFs  and In the discussion following the descriptive presentation of the ethical issues identified in the literature, such as ours, it should not be forgotten that these ethical issues have different qualities, risks and practical relevance and therefore require different solutions: First, there are ethical challenges, such as discrimination or unequal access to reimbursement, which could be solved by legal or policy measures . SecondWith the increasing use of genome-wide sequencing, non-genetic medical specialties, such as pediatricians, are also increasingly confronted with it: They are often the first to see and know the affected patient and their families best; they provide pre- and post-test care; they are able to make a referral to a geneticist or, in some countries, can order genome-wide sequencing of their pediatric patients themselves. However, studies show that given the complexity of genome-wide sequencing, pediatricians are often uncomfortable with it . Here, nWith the increased complexity and potentially difficult ethical decisions associated with genome-wide sequencing, the difficulty of responsibly designing and conducting the informed consent process also increases enormously. It has even been argued that the traditional idea of informed consent might no longer be feasible , 60, 61 Risk of irresponsible parental data sharing [Despite the long list of ethical issues, awareness of some appears to be higher than others, as these are discussed a lot more often, questions and challenges around UFs being the most prominent. This observation is supported by the systematic review of technology users view’s about clinical WES by Bertier et al. , which r sharing , 42, 66.whole exome or genome and furthermore, in most cases of clinical application only a part of the sequenced data is actually looked at. In addition, the term ‘whole’ runs the risk of obscuring the fact that a large part of the data collected cannot yet be meaningfully clinically interpreted and of raising unrealistic expectations not only for patient’s parents but also for non-geneticist medical physicians.What makes the discussion of the ethical aspects even more difficult, especially for other specialists/non-geneticists, is the confusing terminology. With regard to both the test procedure/ the scope of analysis, as well as with the topic of UFs , 96, 97,In light of the increased complexity of genome-wide sequencing, including of the ethical challenges, it is of great importance that the necessary resources, financially, in terms of personnel and also in terms of time, are available. This is the only way to ensure that genome-wide sequencing is used responsibly, that despite the described complexity the decisions of patients and parents are made as informed as possible, and that they are also well cared for in the long term. This also includes good cooperation between all professionals involved and sufficient further training opportunities, e.g. for pediatricians, as they are becoming increasingly involved in testing and will play a key role in providing information, support and follow-up for patients and their families. The comprehensive overview of ethical issues, provided in this review, can inform educational material and raise awareness among practitioners and serve as a check-list helping parents and their pediatricians to obtain more information.One limitation of this review might be seen in the fact that the searches were restricted to PubMed and Google Books with relevancy ranking. It is true that although the review is systematic, not all the existing literature dealing with ethical issues concerning genome-wide sequencing might have been included. However, this is not to be considered an overly disadvantageous factor and the approach was considered to be appropriate for various reasons: the search strategy allowed thematic saturation, and the publications that were finally analysed covered journals from all relevant fields ; additionally, former systematic reviews , 99 in tThis review gives a comprehensive overview of ethical issues in pediatric genome-wide sequencing which are discussed in the literature. It can inform educational material and raise awareness among practitioners. Ethical issues related to the analysis of human DNA in the context of clinical care and research have been discussed continually for the past 50 years. Most issues are not new as such but multiplied and amplified by genome-wide sequencing. This review is a first step to map the huge variety of issues. This is particularly important as awareness of the possibilities, but also the challenges, of genome-wide sequencing for children is becoming increasingly urgent also for other medical fields, i.e. non-geneticists. It highlights the importance that the medical genetics and ethics communities together with other medical professions involved work jointly on specific case related guidelines to grant the maximum benefit for the care of the children, while preventing patient harm and disproportionate overload of clinicians and the healthcare system by the wealth of available options and economic incentives to increase testing.Additional file 1. Bibliographical Information of Analysed Publications. A list providing full bibliographical information of all 87 publications included in the analysis of this systematic review.