Challenges included the burden on snack volunteers to plan, procure, and prepare foods; the competition the program posed for school priorities; limited funding; and a lack of clear purpose for the program. Suggestions for improvement included providing adequate and sustained resources and an integration of the program into the curriculum. Data obtained from 15 on-site visits, 7 General Information Forms, and 59  Weekly Logbooks confirmed the data obtained from interviews. This research provides insights into the challenges of volunteer-led school snack programs in Canada and may guide policy makers, practitioners, and researchers in the development of a universal, nationally funded school food program.The purpose of this study was to assess the implementation of a school snack program for children in elementary schools. School-level program volunteers’ experiences were explored using semi-structured interviews. Fieldnotes were taken during on-site school visits. Quantitative data were collected through a General Information Form and Weekly Logbooks. Seven elementary schools in Southwestern Ontario were invited and agreed to participate. Interviews ( School food programs (SFP) have been identified as promising strategies for fostering children’s healthy development . In CanaAlthough different types of SFP have been implemented, Canada remains one of the few industrialized countries without a universal, nationally funded SFP . The resIn Southwestern Ontario, the Ontario Student Nutrition Program (OSNP) is housed within the Windsor-Essex site of the Victorian Order of Nurses (VON). VON is 1 of 14 lead agencies in the provincial Student Nutrition Ontario program that administers provincial grant funds to support the development and implementation of SFP across the province . The OSNIf a universal, nationally funded SFP is to be implemented in Canada, then a better understanding of the implementation practices and processes of existing SFP is clearly needed. To date, few SNP in Canada have undergone process evaluations ,14,15. TPh1); Phase II  (Ph2); and Phase III (Winter 2018) (Ph3) to mirror the evaluation of the intervention arm of the larger study.This research is part of a larger study that involved 60 elementary schools. Of these, 30 schools implemented the usual practice model of the OSNP while the remaining 30 schools received an enhanced intervention program that involved central food procurement and the delivery of foods to schools. The evaluation of the intervention arm of this study has been published elsewhere . AlthougTo be eligible to participate in the study, schools were required to serve food 3–5 days a week. Of the 30 schools implementing the usual practice model of the OSNP, seven were invited to participate in this evaluation. All participating schools served children in full-day kindergarten through Grade 8, and schools in both urban and rural areas were selected to capture diversity in school characteristics. School size ranged from approximately 100 to 900 students. All personnel , teachers (T), educational assistants (EA), and volunteers (V)) either directly involved with or aware of their school’s food program were invited to participate in the study. Recruitment occurred primarily by placing a recruitment flyer and the study’s Letter of Information in the staff lounge. Principals also made recruitment announcements on the day of the interviews. Any school-level participants who expressed interest in the study were interviewed. This study was approved on 29 November 2016 by the Western University Non-Medical Research Ethics Board (#108549) and the research and evaluation offices of the London District Catholic School Board and the Thames Valley District School Board. Written and/or verbal informed consent was obtained at the beginning of each interview.To gain a deep understanding of school-level staff and volunteer perspectives, we employed a descriptive, exploratory approach with the intention to advance knowledge translation between researchers, practitioners (stakeholders), policy makers, and the public and to provide suggestions for program improvement . We alsoQualitative data were obtained through interviews and field notes from on-site visits. Interviews were conducted at the end of each phase of SFP implementation during school hours on dates determined in collaboration with the school principals. A semi-structured interview guide was adapted to reflect each participant’s involvement with the OSNP and to allow role-specific concepts to be explored . On-siteQuantitative data were derived from a General Information Form and Weekly Logbooks. Participants recorded data on general school and program characteristics  on the General Information Form, which was administered once at the beginning of the program. Volunteers directly involved in food preparation recorded quality aspect measures  in Weekly Logbooks.All interviews were audio recorded, transcribed verbatim by student volunteers, and verified by one of the researchers. Interviews were analyzed using inductive content analysis . To incrSeveral strategies enhanced the trustworthiness of the data. The semi-structured interview guide was employed to achieve reliability and consistency in data collection. Member checking was conducted during interviews to confirm participants’ perspectives. An audit trail, including a reflexive journal, was kept as documentation of the decisions made during the analytical processes . Validitn = 3); T (n = 10); EA (n = 6); and V (n = 8). In addition, 15 on-site visits were conducted (MRI). Each school was observed at least twice during their 10-week phase of implementation for approximately 2–3 h per visit. Each quotation is identified by participant role, participant number, and phase of implementation. Thus, ‘EA3_Ph2’ represents Educational Assistant, Participant #3, Phase 2. Seven General Information Forms (one per school) and 59 out of 70 Weekly Logbooks  were collected and analyzed.All seven invited schools that were implementing the usual practice model of the OSNP agreed to participate in this study . In totaMost participants expressed appreciation for the OSNP and stated that it was a valuable and integral part of their school. Participants felt that the program supported and filled a need in their school community by feeding hungry children. Some participants also stated that the program showed children that they were cared for and provided support to parents and caregivers. In addition, most participants valued the universality of the program as it eliminated any potential stigma that some children may feel when accessing the program.“It’s a multi-pronged situation, that they’re feeding themselves, they’re getting the nutrition, and they know that somebody cares about them”EA1_Ph1“It gives students who might not necessarily come out and say they’re hungry a chance to get something to eat…there’s not so much stigma attached to it”EA6_Ph3When asked about the successful aspects of snack programs, most participants focused on the benefits or impacts on their school communities. Frequently mentioned was the fact that the OSNP created excitement among children, with a few participants commenting that children expected or relied upon the program. This was confirmed by Weekly Logbook data from snack personnel, which suggested that children seemed to enjoy the snack 100% of the time (data not shown).“I get “thank yous”, I even get applauses sometimes, because the kids are excited; they’re happy”EA3_Ph2“There’s more bathroom visits I think than any other day of the week because they wanna know what I am up to”V8_Ph3“And they don’t have to worry about it, it’s not something special. They know they can count on it and it’s a comfort to them”EA4_Ph3Most participants also mentioned that the snack program provided and exposed children to a variety of healthy foods that might not be offered to them at home. In turn, participants highlighted the impacts of this provision and exposure. Most participants stated that it encouraged children to try new items, helped them to focus on their learning, and led to enhanced socialization within the classroom. This latter aspect was confirmed during field visits, where children were seen discussing food items and their willingness to try unfamiliar foods.“The kids won’t always eat stuff at home, but at least they are trying it here”V8_Ph3“Getting them to a place where they need to be, where they’re available to learn”T1_Ph1“It definitely starts the day off on a good note…it settles them and gets them ready for the day”T3_Ph2“Food is social, and social is part of learning”AS3_Ph2“[I hear] kids talking [mimicking voices] “Have you tried this before?” “No.” Then the other kid would say, “I love it”, so then they’ll try it too, just because”T2_Ph2With respect to socialization, a few participants also expressed that the OSNP provided additional opportunities to build relationships with their students while also providing opportunities for non-school program volunteers  to become part of the school community.“Meets the needs of building a relationship with the student, ‘cause they see that this is a safe place and sort of a place that cares for them…it fosters the education”T1_Ph1“Everyone enjoys the parents coming in. It kind of feels like a family. Or their kids go here and they want to feel part of the school. I love that”V3_Ph3Finally, a few participants stated that the OSNP presented opportunities for enhanced learning, such as promoting both good manners, nutrition, and leadership, by including children in food preparation and delivery tasks.The main challenge mentioned by most participants was the high burden placed on personnel/volunteers to implement the program. While most participants commented on the dedication of program volunteers, they also acknowledged the large responsibility put on volunteers to plan, purchase, prepare, and deliver snacks to students. Not only did participants express difficulty in finding dedicated volunteers for the program, but some also expressed that when school personnel  were involved, it competed with their official school duties. These findings were confirmed during field observations. The site observer (MRI) witnessed 1–2 h of preparation time and heard comments from program volunteers regarding the additional time needed to shop for food .“It’s just a little snack, but it is a lot of work to prep it and plan it”V1_Ph2“Nobody wants to take responsibility of organizing that because there’s a lot of effort and time [so] EAs come every morning early [to do that] on top of everything else. And its volunteer work. They’re stressed out, they need support”T6_Ph3“We’d have to do [grocery shopping] on our own time or after school and then I’d have to store it at my house for the weekend”EA6_Ph3Another challenge mentioned by most participants was inadequate funding, which placed additional constraints on program implementation. Some participants expressed that while they tried to adhere to nutritional guidelines, funding often limited their purchases to less expensive non-perishable items . Other participants commented that the lack of funding to support infrastructure, such as storage and preparation utensils, had an impact on food choices . This necessitated that participants either bring supplies from home, often at their own cost, or fundraise/find donations, which led to an increased need to purchase food items  more frequently. Field observations confirmed this lack of infrastructure by noting limited space for food preparation and storage.“I think money is the first and foremost challenge. We work the best we can, by getting donations, or bringing in our own stuff”EA2_Ph1“You still end up defaulting to more prepared foods over fresh fruits and vegetables”AS3_Ph2“I literally have to go every single day to buy the snack”V3_Ph3“What I like least is often I am out of pocket”T5_Ph3While most participants felt the program was appreciated by school staff, some mentioned that the lack of clear guidance regarding the purpose of the program led to some tensions within the school. Some participants expressed that they were unsure as to the intended program recipients , the expectations regarding how much each child should receive, the program’s role in feeding children, and the competition with mandated instructional time.“Some teachers just don’t do it, or they say their kids don’t need it”T6_Ph3“It’s a delicate area because we are not parents, I don’t feel we should be the ones telling them they have to eat it”T9_Ph3“We’re supposed to teach this many minutes, they don’t account for a snack in that time”T7_Ph3“I know some teachers feel that’s it not my job, and it is our job in a way, because it is in the curriculum to teach healthy eating, right?”T7_Ph3“It’s taking time away from where I need to be in my classroom”T6_Ph3Although only mentioned by a few participants, some minor issues regarding the variety and quality of foods provided were mentioned . This was confirmed by the Weekly Logbook data, which revealed that the quality, freshness, and appearance of snacks were rated as “very good” to “excellent” by 82%, 97%, and 98% of respondents, respectively (data not shown).All participants stated the need for adequate and sustained resources to ensure program feasibility and fidelity in the future. Most participants mentioned the need for enhanced funding to support a greater variety of snacks, including some that are pre-packaged, to alleviate the burden of preparation. Many participants commented not only on the need for more dedicated staff, but also for resources to support those staff, such as educational workshops , access to a pre-planned menu with some flexibility to choose food items, and opportunities to improve economies of scale.“More money is always wonderful”V4_Ph3“It’d be so much better if we had more help”V2_Ph2“Reading the ingredients, learning the first few words on the ingredient list are really important, learning how to buy in bulk for cheap when you are feeding 520 people you gotta find cheap and just organizational skills and multi tasking on how to do this”V4_Ph3“We are all doing procurement and delivery on a small scale”AS3_Ph2Another suggestion mentioned by many participants was the need to clearly communicate the role of the food program within the school environment. Participants stated that if clear links to existing curriculum were made, it might help to better communicate the expectations for the program. Some participants also commented that children’s involvement in food preparation would be welcome; however, the feasibility regarding supervision would have to be considered.“Communication is key. Working together is key and the culture has to change a little bit”T6_Ph3“We have to do a better job at educating teachers and understanding that this is why we’re doing it”T10_Ph3“It’s [student involvement] not really built into the program, but I think administration would support it. I think its just a difficult thing to do with supervision”T1_Ph1A few participants mentioned the need to improve the visibility of the program in the hopes that it might lead to better buy-in by all stakeholders, including not only school staff and parents, but also by society in general, which ultimately could lead to more funding.“Making it [the program] more visible in schools, would go a long way”AS2_Ph1“You have to change the mindset of the public, right? And then have the parents be understanding of the importance, so they want to fight for it…without the public’s passion behind it, politics won’t change”EA1_Ph1This study examined the perspectives and experiences of school-level volunteers involved with a SFP in Southwestern Ontario. While the values and successes of the program were overwhelmingly acknowledged by all volunteers, some challenges were discussed, along with potential solutions for future program improvement and sustainability.The primary value of the OSNP identified by these participants was the influence they perceived it had on children’s dietary consumption and exposure to a variety of healthy, and sometimes unfamiliar, foods. These impacts of a SFP on children’s eating patterns have been well documented in the literature. Studies in the US and Europe have shown that these programs can increase the consumption of healthy foods, such as FV . They haIn addition to the impact on eating behaviors, the universality of the OSNP was acknowledged by participants as a means of lowering the potential stigma that can often be associated with programs that simply target children from food insecure households . AlthougInterestingly, participants identified learning opportunities for children with the OSNP , and the potential for greater learning in the future. They commented that the sharing of food promoted socialization and dialogue with their students. Aarestup et al. (2014) reported a sense of “hygge” or “cozy” with their SFP, which represents a safe, low-key, intimate form of socialization . The shaIn addition to social skill development, participants discussed the potential of the OSNP to support food literacy education. Food literacy, by definition, goes beyond an understanding of nutrition and food skills to a greater understanding of the complexities of food and its interaction with health and the environment . TherefoFurther to the above values and successes of the program, participants also viewed the OSNP as a way to support and/or care for their students—a value that is not often mentioned in the literature. Potter et al. 2011) reported that children felt cared for by snack personnel 11 report. Taken tDespite the many values and successes of the SFP, some challenges were mentioned. Predominant among them was the burden put on snack program volunteers to plan, procure, and prepare food. Typically, universal, nationally funded programs have access to food service staff and facilities and employ centralized food procurement practices ,20. ThatAnother challenge of the OSNP was that it often contributed to competition among school priorities for school staff. For those school staff directly involved with the program (mostly EAs), the time needed to prepare snacks was shown to impede upon their existing contractual responsibilities . For teachers, the inclusion of food into the classroom can pose different challenges, such as disruption of learning time and trepidation regarding the role of a teacher vs. the parent in feeding students. This tension between competing school priorities is a common phenomenon discussed in SFP literature ,20,21,27Finally, and central to addressing many of the challenges presented with the OSNP, was the need for sustained and adequate funding. Funding is a common facilitator and/or barrier mentioned in school snack literature ,27,31. NThe strengths of this study include a variety of data sources and methods of data collection. Multiple researchers also conducted data analysis, enhancing credibility. The limitations of this study include the fact that the study investigated volunteer-led snack programs in only one region of the country; therefore, the results may not be transferable to other school snack programs.The findings of the present study confirm that SFP offered in OSNP offer many benefits to children. Participants provided many in-depth insights into the potential values and successes of the OSNP that extend beyond providing children with the provision of healthy foods. These include the potential of these programs to influence children’s learning, engender a sense of community belonging, and enhance life skill development. Despite its successes and the dedication of school-level volunteers to deliver the OSNP, the sustainability of this program is uncertain due to its many challenges. These challenges are mainly attributed to inadequate resources, which lead to inconsistencies in program implementation and possible inefficiencies . If a universal, nationally funded SFP is to be considered, policy makers and practitioners will need to assess the political will to invest in such programs. Adequate and sustained funding will be needed to ensure that schools have the proper physical infrastructure and human resources to deliver a SFP that provides appealing and healthy foods to children at the appropriate level to have any impact on their dietary behaviors. This study also demonstrates that an evaluation of best practices needs to be conducted to assess the parameters of a nationally funded program . In conclusion, SFP represent a means by which multiple sectors—public health, government, education, and the private sector—can intersect to not only meet the basic needs, education, and life skill development of children, but also to have holistic benefits in the community by supporting the local food economy, building community beyond the school environment, and providing potential employment opportunities.


NUP214-ABL1 fusion gene is a constitutively active tyrosine kinase that can be detected in 6% of T-cell acute lymphoblastic leukemia (T-ALL) patients, and it can also be found in B-cell acute lymphoblastic leukaemia (B-ALL). However the NUP214-ABL1 fusion in acute myeloid leukemia (AML) has not yet been reported. Up to now, the sensitivity of NUP214-ABL1-positive patients to tyrosine kinase inhibitor (TKI) is still controversial. Here we report the first case of an AML patient carrying NUP214-ABL1 fusion gene. The conventional AML chemotherapy regimen for the patient was successful. Identification of additional AML patients with NUP214-ABL1 fusion gene will provide treatment experience and prognostic evaluation.The  ABL1 rearrangement, the NUP214-ABL1 is the second common fusion in haematologic malignancies. About 6% of T-cell acute lymphoblastic leukaemia (T-ALL) patients carry this fusion gene (NUP214-ABL1 fusion gene has never been reported in acute myeloid leukaemia (AML).Identifying genetically targetable abnormalities that may respond to targeted therapy is clinically important in the context of precision medicine. Among all the fusion genes involving ion gene . HoweverNUP214-ABL1, a constitutively active tyrosine kinase, is a potential target of tyrosine kinase inhibitors (TKIs)  . Howeverperience –11. Somepatients , 10–13.  relapse –9.NUP214-ABL1-positive patients were diagnosed retrospectively and had already received classical treatment .Given that many 9/L , haemoglobin of 92 g/l, and platelets 10×109/l. Lactate dehydrogenase (LDH) was increased to 2080 U/l (reference <245U/l). D-dimer was quantified at 1730 ug/L (reference range 0-700 ug/L). Ultrasonography revealed lymphadenopathy and splenomegaly. Bone marrow (BM) aspiration revealed 78% blast cells  82.5×10CEBPA (NM_004364.3) had double mutations  and NRAS (NM_002524.5) had a point mutation (p.G13D). NGS also showed the presence of an NUP214 (NM_005085.4)-ABL1 (NM_007313.2) fusion gene (fusion of NUP214 exon 34 and ABL1 exon 3) (NUP214-ABL1 fusion (NGS (a panel of 88 genes) revealed that NUP214-ABL1 fusion and explore whether there is amplification of the ABL1 gene, interphase fluorescence in situ hybridization (FISH) was performed using the BCR/ABL1 dual-colour dual-fusion probe and the ABL1 break-apart probe . Both probes demonstrated normal signal patterns. No amplification or the split signal of ABL1 was detected  with a minimal residual disease (MRD) under 0.01%. After that, he was treated with high-dose cytarabine for consolidation therapy and is currently in his third phase of consolidation therapy. To date, he was in CR status.NUP214-ABL1 fusion gene detected by NGS in a patient with AML. As far as we know, this is the first report of an AML patient with NUP214-ABL1 fusion.In this study, we demonstrated the ABL1 translocation is difficult to detect through conventional cytogenetic and FISH analysis, such as the NUP214-ABL1 fusion in our case. The major cause might be that both NUP214 and ABL1 are located at the edges of 9q34, and FISH may not successfully detect the fusion of these two genes, as reported in a previous study  fused to exon 2 of ABL1  in National Genomics Data Center (Nucleic Acids Res 2021), China National Center for Bioinformation/Beijing Institute of Genomics, Chinese Academy of Sciences. The accession number is: HRA000867. Please access it from the following link: The studies involving human participants were reviewed and approved by the Research Ethics Committee of the First Affiliated Hospital, College of Medicine, Zhejiang University. The patients/participants provided their written informed consent to participate in this study.H-PW wrote the manuscript. H-PW, J-JH, Q-YZ, LW, J-HL, and J-SH performed the research and analyzed the data. W-ZX provided samples and clinical data. JJ and H-HZ critically revised the manuscript. 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.


This study explored the process and early outcomes of work undertaken by a program to increase Aboriginal people’s awareness of, and access to, the National Disability Insurance Scheme (NDIS). This ‘Access Program’ was implemented through the Aboriginal Community Controlled Sector in the remote Kimberley region of Western Australia. Access Program staff were interviewed to explore the strengths, challenges, and future directions of the program. The demographics, primary disability types, and NDIS access outcomes for clients who engaged with the program in the first 12 months of its implementation have been described. The Access Program engaged with 373 clients during the study period and assisted 118 of these to achieve access to the NDIS. The program was reported as successful by staff in its aim of connecting eligible people with the NDIS. Vital to this success was program implementation by the Aboriginal Community Controlled Sector. Staff in these organisations held community trust, provided culturally appropriate services, and utilised strengths-based approaches to overcome barriers that have historically hindered Aboriginal people’s engagement with disability services. Our results demonstrate the Access Program is a successful start in increasing awareness of, and access to, the NDIS for Aboriginal people in the Kimberley region. Much work remains to assist the large number of Aboriginal people in the Kimberley region believed to be eligible for NDIS support who are yet to achieve access. In Australia, Aboriginal and Torres Strait Islander people experience disability at twice the rate of other Australians . HistoriLaunched in 2013 and progressively rolled out since 2016, the National Disability Insurance Scheme (NDIS) was developed to provide a better funding model for all Australians with permanent and significant disabilities . The NDIThe NDIS restructures funding from services to individuals promoting greater ‘choice and control’ . HoweverThe governing body of the NDIS, the National Disability Insurance Agency (NDIA), has recognised the different needs of certain groups in accessing the NDIS ,15. The The Kimberley NDIS “Access Program” was developed after a $4.6 million investment by the NDIA into Aboriginal Community Controlled Health Services in regional and remote Western Australia to improve awareness of, and access to, the NDIS for Aboriginal people . This AcThe Kimberley Access Program is nested within the NDIA funded Kimberley Supports programs managed by Kimberley Aboriginal Medical Services (KAMS) and delivered by a consortium of regional Aboriginal Community Controlled Organisations and Aboriginal Community Controlled Health Services.This research aims to explore the work being undertaken by Kimberley Access Program staff who are tasked with identifying and connecting eligible Aboriginal community members to the NDIS. We aim to contribute to an understanding of the challenges, successes and future directions of ensuring equitable access to the NDIS for Aboriginal people in the Kimberley.A mixed methods approach  was usedThis research was undertaken at the request of KAMS. KAMS, established in 1986, is the peak Aboriginal Community Controlled Health Organisation in the Kimberley region and is committed to progressing an evidence base for its emergent disability activities. All consortium organisations involved in the Access Program were consulted in the development of the research proposal and provided written support for the project. Consortium organisations were provided with a copy of the manuscript to review prior to submission of the article for publication. This process of feedback ensured the publication is reflective of the experiences and perceptions of the organisations within the consortium.The project was endorsed by the Kimberley Aboriginal Health Planning Forum Research Subcommittee prior to receiving formal ethics approval from the Western Australian Aboriginal Health Ethics Committee (Project 970). Both of these committees include Aboriginal leadership and exist to support and promote research that reflects and is responsive to the needs of Aboriginal people ,28.The Kimberley region see , in the RCCs and EACPs are employed by eight consortium organisations based in four towns in the Kimberley region. RCCs and EACPs have provided support to potential NDIS participants in at least 12 remote communities and six Kimberley towns. The RCC role includes building community-level awareness and understanding of the NDIS and engaging with individuals and families who may be eligible for the NDIS. RCCs then support clients through the NDIS access process. RCCs refer to EACPs who assist clients to apply for the NDIS and access NDIS funded services.Once informed consent has been obtained, the EACP gathers the necessary evidence from the client to complete an NDIS access request form. EACPs may access the medical record with patient consent to find appropriate supporting evidence for their client’s application. When medical records are held in non-consortium clinics or historical records are required, EACPs may submit applications to other services for release of information. If appropriate evidence for an application is not available in the medical record, EACPs may organise functional assessments for their clients with an allied health professional or general practitioner. The process of gathering evidence can be time consuming and labour intensive. The EACP submits the completed form and supporting documentation to the NDIA’s National Access Team for remote areas in Western Australia on behalf of their client. Clients may consent to EACPs acting as their delegate to coordinate communication between themselves and the NDIA throughout this process. If a client does not qualify for access to the NDIS after their access request form has been reviewed by the NDIA (termed “access not met”), the EACP may assist them to appeal the decision or link the client with other support services as appropriate. If the client does meet access to the NDIS on review by the NDIA (termed “access met”), the EACP will assist the participant to prepare for a planning meeting with the NDIA (termed “pre-planning”). RCCs and/or EACPs may assist with coordinating planning meetings and may attend with the participant as per their wishes. EACPs and RCCs may be employed by different Aboriginal Community Controlled Organisations and Aboriginal Community Controlled Health Services but work in partnership to support both individual and community level outcomes.A purposive sampling frame of the Access Program staff was used for the qualitative component of the study. Twenty Access Program staff were employed at the time of data collection (October 2020 to January 2021) and were invited to participate. In addition to Access Program staff, managers and executives were invited to participate to gain additional background and perspectives on the Access Program.Quantitative analysis included data from a total of 373 potential NDIS participants who had engaged with the Access Program between 31 October 2019 and 16 November 2020.Quantitative analysis utilised a database provided by the Regional Access Program Coordinator. This data set consisted of de-identified information from all participants referred to an EACP from 31 October 2019 to 16 November 2020. Variables within the data set included age, community of residence, source of referral, primary disability, secondary disability, and stage of NDIS application. The primary disability listed was categorised to protect confidentiality using an impairment of functioning approach . The catReferrals to the EACP were summarised by referral source, outcome of referral, category of disability, and time to referral outcome. Descriptive analysis was completed in Stata Statistical Software: Release 16 .In total, 14 semi-structured interviews were conducted with 11 Access Program staff and three program managers/executives. Of these interviews, eight were conducted face to face (including two group interviews with five and two participants) while six were conducted over video conference. Interviews were conducted during working hours at staff members’ places of work. All interviews were undertaken by a female non-Aboriginal clinical researcher (CW) and voice recorded with written participant consent.Interviews were professionally transcribed into individual Microsoft Word documents. These were imported into NVivo 12  and then coded and analysed using a directed qualitative content analysis approach . All intn = 102) or being confirmed as a pre-existing NDIS participant (n = 16). Eighty-eight clients (24%) did not access the NDIS because of not meeting access requirements (n = 72) or self-withdrawing from the referral process (n = 16).Between 31 October 2019 and 16 November 2020 a total of 373 people were referred to an EACP in the Kimberley region. Characteristics of these clients are outlined in The remainder  had referrals in progress. Of these, 66  were in early engagement with Access Program staff, 76  were having evidence gathered, 23  had completed their Access Request Form, and the referral status for two  clients was unknown.Intellectual , physical , and psychiatric disability  categories were the most common primary disability type recorded for participants referred to an EACP .Time from EACP referral to formal submission for NDIS access was highly variable, ranging from 1 to 265 days. Similarly, time from formal referral to access approval ranged from 1 to 83 days.Of the 20 Access Program staff eligible to participate, a total of 11 program staff were interviewed. Nine of the interviewees were employed in access program delivery (RCC or EACP) and two were access program coordinators. The remaining nine Access Program staff did not respond to an invitation to be involved or were not available when the interview took place. An additional three interviews were held with consortium executives or managers. Out of the 14 interviews conducted, four staff members interviewed were Aboriginal and three were male. All staff who participated in the interviews provided written informed consent.“I think one of the hardest parts of it [engaging potential clients] is, people not realising themselves that they have a disability…people are just so resilient in communities and they just get on with things… So, when you go and speak to people…‘No, we’re right’. They think they’re not disabled, that it’s normal.”Remote Community ConnectorWhen reflecting on the Access Program, staff discussed Aboriginal people’s perceptions and experiences of disability. Many staff reported that disability is often ‘not recognised’ by the individual or by the individual’s family group. High levels of resiliency and normalisation of the disability were raised as contributing factors:“…a lot of people have got that stigma ‘well I don’t want my child being labelled as disabled’ and we’re explaining to them it’s not being labelled as ‘disabled’ it’s just getting the support that they may need at school to help them learn and help them. So trying to explain that to a lot of the community so that they don’t have that stigma …the shame factor…”Evidence, Access and Coordination of Planning CoordinatorAdditionally, Access Program staff discussed the fear and stigma that Aboriginal community members associated with concepts of disability:“It’s a sensitive issue disability. You’ve got a lot of people who have had permanent disabilities for a long time or born with [a disability]. It doesn’t change their standing in community or their decision making or where they sit within their family.”Kimberley Consortium Executive/ManagerOther staff discussed how disability often had a ‘different meaning’ within Aboriginal communities and was often not seen as central to a person’s identity:Staff discussed how it was important to be aware of these differing perceptions of disability and not to approach disability as a deficit or something to be ‘fixed’ with potential clients and their families. Staff reflected on the need for conversations to be enquiry based, have a focus on recognising the strength and ‘resilience’ of an individual and family, and include key family members.“The beauty of your RCCs, their skill set isn’t necessarily sitting in front of a computer and gathering evidence…They’ve got the ability to actually engage people around NDIS.”Kimberley Consortium Executive/Manager“I run around, try and find the clients, go to this house and that house, finally find them, and then I’ll do meetings under a tree, sitting there having a yarn just like it’s nothing else.”Remote Community ConnectorMany staff reflected that historically the Kimberley region has experienced a lack of available or suitable disability support services. This was perceived to complicate people’s engagement with the NDIS as trust had been broken and people were reticent to ‘waste their time’ with another service. Engagement at the individual and community level was perceived as essential to work through barriers to accessing disability services and promoting awareness of the NDIS. RCCs and EACPs described engagement as requiring high levels of flexibility, commitment, and support:“Well a lot of it is just knowledge of the community… And knowledge of actual people. You know like maybe potential participants because you know them in a social [setting]… and working here at [name of service] you see people coming in and coming out and you know they may have a disability”.Evidence, Access and Coordination of Planning CoordinatorAccess staff described their knowledge of the community as helping them to make targeted visits to certain families:“They know the name [Organisation Name], they know the brand already and they know we’ll be there… We don’t go away. So, we’re in.”Kimberley Consortium Executive/ManagerStaff discussed the positive impacts of having Aboriginal Community Controlled Organisations deliver the Access Program. These organisations were described as trusted, culturally appropriate, and holding pre-existing relationships with clients. This in turn enhanced the acceptability of the Access Program staff working to raise awareness and facilitate access to the NDIS:“During this time, you liaise or become the person that connects the NDIA planners to the participant and help with organising the meetings and setting that up. Making sure that it’s conducted in an appropriate manner, make sure everyone’s or the person is comfortable and is best prepared as it can be. Quite often I sit in on these meetings to support and also go through, basically prompt the person if there’s something that’s been missed.”Evidence, Access and Coordination of Planning CoordinatorAccess Program staff described the engagement with clients as enduring with many EACPs continuing to work with clients after access had been met. This was to ensure clients had appropriate support and advocacy when engaging with NDIA staff to develop their support plan:It was identified that Access Program staff also met with client frustration when timeframes for access decisions from the NDIA were lengthy, access to the NDIS was denied, or the appropriate disability support services were unavailable in communities.“…so if there’s a birth certificate missing or the dates don’t match Centrelink and different things like that, instead of rejecting it and sending a letter straight to the participant, they’ll talk to the Evidence and Access person who’s on the third-party consent form.”Access Program CoordinatorStaff reflected on how the program had provided a powerful advocacy service for the region that had resulted in a shift in the way that NDIA reviewed Kimberley NDIS applications. Access Program staff reported instances when the NDIA were responsive to issues affecting clients’ access to the NDIS. EACPs discussed being an important source of contact for the NDIA to clarify issues in applications in order to facilitate meeting access and avoid lengthy decision-making delays for clients:“…it’s about being that person to liaise with the NDIA to say ‘Hey, this person might not be ready or they might not be in community, or they haven’t done their pre-planning’… we had someone with acute psychosis in the mental health here and they still thought it was appropriate to have a planning meeting while they were there and not in a healthy state of mind in a familiar environment… They’re [the NDIS] very keen to get things happening really soon. A bit too soon in some cases.”Evidence, Access and Coordination of Planning CoordinatorDespite the responsiveness of the NDIA with application review, Access Program staff still reported that the access pathway to the NDIS could be challenging for clients due to bureaucratic inflexibility. For example, there could be pressure from the NDIA to quickly move a client to the planning stage after access being met despite a client not yet being ready to proceed. Locally based Access Program staff were well positioned to advocate for clients to proceed at their own pace:Access Program staff commonly reported that NDIA resources were not culturally appropriate for Aboriginal people in the Kimberley. ‘Easy read’ versions of booklets provided by the NDIA did not meet the cultural needs of remote Aboriginal people, and Access Program staff created their own resources as a result of this gap. Access Program staff and managers reflected that NDIA training was not targeted to the unique needs of the Kimberley Access Program workforce, and the onus on developing responsive and appropriate training became an issue for the consortium to resolve.Staff noted that confusion existed amongst communities regarding the eligibility requirements for the NDIS. In particular, a common perception was that those on a government-issue disability support pension would be automatically eligible for the NDIS. Access staff noted they were required to explain that while both supports came from the federal government and included the word ‘disability’ the eligibility criteria are very different.“Sometimes I’m finding that it’s [the referral is for] health reasons but…there’s not a disability, so we’ve got to get our people to understand the difference as well about what a disability is.”Evidence, Access and Coordination of Planning CoordinatorSeveral staff reflected on the diversity of the program’s workforce with many coming from outside the health and disability sector. While the diversity was seen as a strength of the program, it highlighted the need for ongoing training and a baseline knowledge in disability:“There is maybe a lot more people in the Kimberley who would be eligible for NDIS. I think there’s probably still a lot more work to be done in the way disability is described and talked about, just generally, so that community members awareness starts to lift…”Kimberley Consortium Executive/ManagerOverall staff felt positive about the program and the regional efforts that had been made in terms of training, program set up, and clients meeting access. Program managers noted the value of the large consortium of community controlled organisations delivering the program. Specifically, partners brought value to the table in terms of local knowledge and the ability to implement place-based engagement responses across the vast and remote Kimberley region. The program was frequently described as ‘successful’ by staff. Despite this, there was a strong sense that the work had just begun with staff voicing fears that the project would not be funded after the current contract ends in the middle of 2021:This research has demonstrated the importance of the Kimberley Access Program as described by a review of audit data and consultation with program providers. The program positively contributes to evidence that place-based approaches to engagement are successful in promoting awareness of and access to the NDIS for a population that experiences profound social inequality and exclusion . CongrueAccess Program staff and managers discussed the importance of Aboriginal Community Controlled Organisations leading engagement. Aboriginal-led approaches were identified as best placed to overcome the historical and systemic factors that contribute to low levels of engagement by Aboriginal people with a disability ,13,43. TWhile the proportion of those with a physical disability who engaged with the Access Program was lower than observed in the national dataset, the proportion of those with non-physical disabilities  was higher . It has With relatively low rates of self-referrals to the Access Program, referral data indicates that engagement has occurred largely from RCCs, again, using their local knowledge. Another important source of referral is through clients engaging with other local organisations including the health sector, child and family services, and the justice services. The Access Program has demonstrated a promising brokerage role between these organisations and the NDIS.Despite the promising success of the Kimberley Access program, evidence suggests that for Aboriginal people in rural and remote Australia more work needs to be done . The NDIThis study is the first of its kind for the Kimberley region. It provides a regional level understanding of the NDIA funded Access Program and a profile of the individuals who are engaging with the program. It is the first study to explore the novel role of the EACP, which is unique to Western Australia. We note as a limitation that the qualitative component of this study did not include any potential or active NDIS participants that had engaged with the Access Program. Missing data in the quantitative analysis limited the conclusions that could be drawn from its analysis. It is recommended that future research engage with potential and active NDIS participants to explore their NDIS journey from access through to the provision of support they receive via their NDIS funded plan.As a ‘once in a generation’ reform, the NDIS presents the opportunity for Australians living with a disability to access disability support services to reach their goals with greater choice and control. For Aboriginal and Torres Strait Islander people living in remote Australia, access to and engagement with disability services has historically been limited. Successfully implementing the NDIS with this priority population, who are disproportionately burdened with disability, is in the interest of both the NDIA and the Aboriginal Community Control Sector. The Kimberley Access program has demonstrated success in engaging remote Aboriginal clients who are eligible for the NDIS. Overall, this study demonstrates how the two parts of the Access Program work both together, and with other community services, to support clients in accessing the NDIS.


The lymphatic drainage was classified into echelon-1 , echelon-2  and echelon-3 . The pattern of spread was classified according to all permutations of echelon-1, echelon-2, and echelon-3 EBUS-TBNA findings.This is a single-center study based on our preceding investigation, including all consecutive patients with initial diagnosis of stage IIIA-C NSCLC, receiving concurrent radiochemotherapy (12/2011–06/2018). Inclusion criteria were curative treatment intent, p < 0.0001, exact symmetry test). In a multivariable logistic model for EBUS-positivity at echelon-3, prognostic factors were PET-positivity at echelon-3 (Hazard ratio (HR) = 12.1; 95%-CI: 3.2–46.5), EBUS-TBNA positivity at echelon-2  and left-sided tumor location . There were significantly less combined ipsilateral upper (LN-stations 2 and 4) and lower (LN-station 7) mediastinal involvements (16.8% of patients) with EBUS-TBNA than with PET . EBUS-TBNA detected a lobe specific heterogeneity between the odds ratios of LN-positivity in the upper versus lower mediastinum , while PET did not (p = 0.19).In total, 180 patients were enrolled. Various patterns of LN-spread could be identified. Skip lesions with an involved echelon distal from an uninvolved one were detected in less than 10% of patients by both EBUS-TBNA and PET. The pattern with largest asymmetry was detected in cases with EBUS-TBNA- or PET-positivity at all three echelons (Frequent patterns of LN-metastatic spread could be defined by EBUS-TBNA and PET and discrepancies in the pattern were seen between both methods. EBUS-TBNA showed more lobe and tumor laterality specific patterns of LN-metastases than PET and skipped lymph node stations were rare. These systematic relations offer the opportunity to further refine multi-parameter risk of LN-involvement models for target volume delineation based on pattern of spread by EBUS-TBNA and PET.The online version contains supplementary material available at 10.1186/s13014-021-01904-4. Precise detection of the loco-regional pattern of tumor spread is of utmost importance for delineating the radiation target volume. The primary goal is to maximize effectiveness of radiotherapy while minimizing the treated volume in order to spare surrounding normal tissue. The balancing act between the optimal therapeutic benefit and possible long-term sequelae is a key issue in radiotherapy.18F-FDG PET-scans did not show a higher effectiveness of radiotherapy including elective nodal irradiation over involved field radiotherapy alone in locally advanced non-small cell lung cancer (NSCLC) [18F-FDG PET/CT.Evidence from older randomized trials with  (NSCLC) . HoweverFirst the PET-plan trial was able to confirm the non-inferiority of involved field lymph node irradiation compared to a conventional target group including limited elective nodal irradiation at the primary endpoint of locoregional progression . Hence, 18F-FDG-uptake, such as granulomatous inflammation, necrosis, as well as lymphoid infiltrates and anthracotic macrophages [18F-FDG PET/CT in comparison to histopathologic results, EBUS-TBNA with pathologic confirmation of mediastinal involvement is recommended in potentially curable NSCLC [Nevertheless, mediastinal LNs are known to be PET-positive also due to non-malignant causes. Acute or chronic infectious and inflammatory processes may result in LN enlargement with an elevated rophages . Due to le NSCLC , 8.18F-FDG PET/CT from echelon-1, to echelon-2 and -3 lymph nodes in patients with locally advanced NSCLC, treated with neoadjuvant or definitive radiochemotherapy [In our preceding per lymph node analysis at this institution’s lung cancer database, we found a rising FDR of otherapy . There aotherapy , 11. Occotherapy , 13. Skiotherapy , 15.In the present study, we analyzed the agreement of the intra-patient pattern of nodal spread by EBUS-TBNA as well as PET, from the primary tumor to the upper and contralateral mediastinum. A prognostic model was built for echelon-3 involvement by EBUS depending on the pattern of spread revealed by PET and EBUS at the more proximal LN stations and the localization of the primary tumor. In addition, differences in the lobe-specific pattern of spread detected by EBUS and PET were assessed.All consecutive patients with histopathologically proven NSCLC stage IIIA-C (according to AJCC/UICC/TNM 8th edition) presented in an academic lung cancer center for radiation oncology from December 2011 to June 2018 were enrolled in this study. This is a further evaluation of the exact intra-patient pattern of nodal spread complementing our previous study with a per lymph node analysis over all patients .18F-FDG PET/CT diagnostic information was obtained before start of treatment. Mandatory exclusion criterion was a previous cancer diagnosis or corresponding treatment.In all patients classified for a potentially curative concept at initial diagnosis, EBUS-TBNA and 18F-FDG, PET/CT imaging was performed on the PET/CT Biograph mCT scanner . Two certified board members created the principal nuclear medicine report (PET-report): a nuclear medicine physician and a radiologist. SUVmax measurements were conducted in all EBUS-TBNA sampled LN stations, both for EBUS-positive (with tumor cells proven) and EBUS-negative .After intravenous injection of 250–400 MBq th TNM classification, as well as ipsilateral involvement of station 2. This classification also allows sub-analyses of the different stations of echelon-3 involvement depending on the involvement of echelon-1 and -2.EBUS-TBNA was regularly done in a systematic manner. All detectable LN stations larger than 5 mm  were sampled and entered into the study database according to the definition of the IASLC  lymph node map . PET-pos18F-FDG PET/CT. Patterns of spread were classified according to echelon-1, echelon-2, and echelon-3 positivity or negativity by EBUS-TBNA or 18F-FDG PET/CT.PET-positivity or EBUS-positivity was assigned to an echelon if at least one LN was positive in that echelon. At lymph node stations 5 and 6, surgical staging plays a decisive role for the exact staging before resection, especially in patients with EBUS-TBNA negative mediastinum, CT-morphologically indolent but PET-positive LN stations 5 and 6 or with primary tumor location in the left upper lobe , 19–21. This study was approved by the local Ethics committee of the Medical Faculty (19-9056-BO).p-values are provided for two-sided hypotheses.Statistical analysis was performed using SAS software version 9.4, SAS/STAT 14.3  . The proA total of 180 patients met the inclusion criteria of this study. Patients’ characteristics along with numbering/ranking of patterns of spread are shown in Table 18F-FDG PET/CT. PET-positivity was observed in 72.2%, 67.8% and 32.8% of patients at echelon-1, -2, and -3, respectively. EBUS-TBNA samples were taken from echelon-1, -2, and -3 in 139, 174 and 144 patients, respectively. The prevalence of EBUS-positive echelons in PET-positive echelons that were analyzed by EBUS-TBNA was 99.2%, 81.8%, and 29.1% in echelon-1, -2, and -3, respectively. The intra-patient spread of LN metastases detected by EBUS-TBNA or PET across echelon-1 to -3 was classified into patterns 1.1–1.6, as shown in Table The aim of this study was to evaluate the concordance in the intra-patient pattern of spread in those LN echelons which can be assessed by both methods, EBUS-TBNA and s = 0.67; 95%-CI: 0.56–0.77). The dependence of the number of EBUS-positive LNs on the number of PET-positive LNs per patient is shown in Fig. The cross-tabulation of the involved LNs per patient according to EBUS- and PET-criteria excluding LN stations 5 and 6 per patient is depicted in Table p < 0.0001, exact symmetry test). The weighted kappa coefficient as a measure of inter-staging agreement between two examination methods is κ = 0.37 (95%-CI: 0.23–0.51).Table p = 0.013, exact symmetry test), 1.3  and 1.6 . These results are shown in Fig. p = 0.056, exact symmetry test).Subsequently, we analyzed which of the patterns showed significant differences in symmetry and found significant asymmetry in patterns 1.2 . In the remaining 39 patients, no asymmetries were observed between the examination methods across patterns 3.1–3.4.As a validation cohort, we analyzed the pattern of spread in the 37 patients with EBUS-sampled echelon-2 and -3 but with an untested echelon-1 . Therefore, we analyzed the dependence of EBUS-positivity in echelon-3 on factors determining the intra-patient pattern of lymphatic spread by PET and EBUS in echelons more proximal to the primary tumor, i.e. PET-positivity in echelon-1, -2 and -3 as well as EBUS-positivity in echelon-1 and -2, and laterality of the primary tumor as prognostic factors using univariable and multivariable logistic regression with forward selection. EBUS-positivity at echelon-2, PET-positivity in echelon-3 and laterality became significant prognostic factors in univariable and multivariable analysis for EBUS-positivity in echelon-3. The respective odds ratios (OR) for EBUS positivity in echelon-3 are shown in Table Table th TNM classification. All factors that were found to be significant for echelon-3 involvement by EBUS-TBNA or PET using multivariable logistic regression were also found to be predictive of N3 involvement when repeated with the same logistic regression model. The respective odds ratios (OR) for N3 involvement using multivariable analysis are shown in Additional file A supplementary analysis revealed that 97% of the echelon-3 lymph nodes examined were located in the contralateral mediastinum or hilum and only 3% were ipsilateral station 2 lymph nodes, with the former defined as N3 involvement and the latter as N2 involvement according to the 8p = 0.0012, χ2-test).In addition, we analyzed the dependence of the probability of PET-positivity in echelon-3 on PET-positivity in echelon-1 and -2 and laterality of the primary tumor as prognostic factors and found that PET-positivity in echelon-2 alone was significant by multivariable logistic regression with forward selection .Of the 66 patients with left-sided tumors and EBUS-proven echelon-2 and -3 LNs, 13 had echelon-3 EBUS-positivity, all of them at LN station 4R, 1 also at station 10R, and 1 also at station 2R. Twenty-six of these 66 patients had PET-positivity at echelon-3, 20 at station 4R, 1 at station 2R and 9 in the contralateral hilum. Of the PET-positives at stations 4R, 2R, and the contralateral hilum, 11, 0 and 1 were EBUS-positive at these stations, respectively. The respective fraction of EBUS-positives among PET-positives at station 4R for left-sided tumors was 11/20 (55%) and for the remaining left-sided and the right-sided PET-positive patients at echelon-3, EBUS-positivity at echelon-3 was observed on average in 5/32, i.e.15.6% . In particular, there was significantly less combined upper and lower mediastinal involvement (16.8%) with EBUS compared to PET (38.9%), . EBUS detected more involvement at the lower mediastinum in lower-lobe tumors (53.3%) than in upper/middle lobe tumors (25.0%), . In parallel, EBUS detected more upper mediastinal involvement in upper/middle lobe tumors (61.4%) than in lower lobe tumors (26.7%), . The odds ratio of EBUS-positivity in the upper mediastinum compared to the lower mediastinum differed significantly between upper and middle lobe tumors  and lower lobe tumors  (Breslow-Day test for homogeneity of odds ratios: p = 0.0021). This could not be defined for PET (Breslow-Day test for homogeneity of odds ratios: p = 0.19). The contralateral mediastinum at stations 4 and 2 was significantly more frequently involved by EBUS-TBNA for left-sided tumors than for right-sided tumors . This difference was not observed by PET .In addition, we analyzed the dependence of the pattern of lymphatic spread by EBUS and PET on the lobe of the primary tumor and therefore used a grouping of LN zones underlying lobe-specific LN dissection, i.e. in the lower mediastinum (station 7), the ipsilateral upper mediastinum  and the contralateral mediastinum/ hilum . With reAdditional discrepancies between invasive staging and PET/CT may exist in stations 5 and 6, but no data are available from this series. In the present study, 37 patients had PET-positive lymph nodes at stations 5 and 6. Only 2 of them had right-sided tumors and echelon-2 involvement was detected in both by EBUS-TBNA. Nineteen patients had PET-positive stations 5 and 6 as well as a negative mediastinal EBUS-TBNA at echelon-2 and -3. Seventeen of these patients had EBUS-positive ipsilateral hilar lymph node metastases and an enlargement of the target volume to stations 5 and 6 was considered moderate. These data show that the potential risk of a false-positive PET-finding at stations 5 and 6 to change the radiation target volume by adding more volume is rather low.18F-FDG PET/CT for NSCLC staging. LN staging pathways are based on the knowledge of lymphatic drainage and mediastinal anatomy.This is a large consecutive case-series study at an academic lung cancer center demonstrating the potential change of radiotherapy protocols depending on primary diagnostic procedures EBUS-TBNA and Research work on lymphatic drainage originates from surgical, pathological and anatomical studies and, more recently, from imaging techniques. The mediastinal N2 status is the subject of a broad investigation in lung cancer. It represents a major challenge in the clinic and requires profound clinical diagnostics of mediastinal spread followed by consequent selection of the most appropriate local therapy , 24. The18F-FDG PET/CT. When considered independently, neither modality provided exact information alone about the spread of LN metastases. Combining these two modalities allowed defining the spread of LN metastasis more precisely.Here we followed the classification based on study results of Riquet et al. and I. Caplan, which we also used in our previous work . The cliPrimary, this study detected significant differences in the intra-patient pattern of lymph node involvement defined by EBUS-TBNA and PET/CT. A pattern with all lymph node echelons involved was found significantly more often by PET than by EBUS-TBNA.In addition, EBUS-TBNA revealed a higher frequency of contralateral mediastinal involvement for left-sided tumors compared to right-sided tumors and a preferential involvement of ipsilateral superior mediastinal lymph nodes of upper and middle lobe tumors compared to lower lobe tumors with a preferential involvement of subcarinal nodes. These patterns were not detected by PET diagnostics. Skip lesions were seen in less than 10% of patients in this study with both staging procedures, however at a high prevalence of hilar lymph node metastases with both methods.Such lobe and side specific spread of lymph node metastases as found in this study by EBUS-TBNA were also detected in specimens from large surgical series , 11, 29.In this study, a model of the risk of LN metastatic involvement of the echelon-3 lymph nodes in the EBUS-TBNA was built as a function of PET-positivity in proximal lymph node stations, as well as EBUS positivity in proximal lymph node stations and laterality of the primary tumor location. Factors that were dependent on the pattern of spread became significant in this model. Sophisticated models for determining the probability of malignant involvement, based on multi-parameter imaging and minimally invasive spread diagnostics, have the potential to improve the accuracy of target volume delineation in radiotherapy planning of stage III NSCLC. This allows a further refinement of the current standard of including all PET-positive lymph nodes in the target volume , 5. PET/The negative predictive value of EBUS-TBNA in PET-positive lymph nodes is of particular importance for the exclusion of a lymph node station from the target volume. The negative predictive value was 91% for PET-positive nodes in the study of Bauwens et al. (2008) at a prevalence of mediastinal lymph node metastases of 58% and 89% in the study of Taverner et al. (2016) at a prevalence of 11% , 39. HowThere are some limitations in this study. PET-positivity at stations 5 and 6 was not included in further analysis as these LN stations were not accessible with EBUS-TBNA. To correctly detect mediastinal involvement in the left para-aortic mediastinum, a preoperative minimal surgical staging may be a necessary diagnostic step , 19.Our investigation points out systematic asymmetries between the intra-patient patterns of lymph node spread in stage III NSCLC detected by EBUS-TBNA and PET. For a single lymph node station, an assessment of the risk of involvement depending on the pattern of lymphatic metastases detected by PET and by EBUS-TBNA in other lymph node echelons can help to include or exclude these regions in the target volume. As genuine for a radiotherapy study, we did not have surgical assessment to validate the spread pattern by EBUS-TBNA. Surgical staging is only available for selected cases. An additional mediastinoscopy cannot generally reduce the false negative rate by EBUS-TBNA, provided that EBUS-TBNA is performed by an experienced team .In summary, morphologically inconspicuous PET-positive echelon-3 lymph nodes should still be assessed critically. If the prevalence in a given lymph node station is known to be low with respect to the localization of the primary tumor and the pattern of LN spread by PET/CT and EBUS-TBNA, then the negative predictive value of EBUS-TBNA is so high that it should be considered for radiotherapy planning. Furthermore, this study points out the low risk of involvement of PET-positive echelon-3 lymph node stations depending on the pattern of more proximal nodal spread.18F-FDG PET/CT information, it is possible to identify the intra-patient spread of lymph node metastases along the lymphatic chain in mediastinum. PET/CT does not only function as a search test and EBUS-TBNA as a confirmatory test. Moreover, both diagnostic tools complement each other, and combined may further adjust the target volume.With the knowledge gained from the combined EBUS-TBNA and This study underlines the importance of combined PET/CT and EBUS-TBNA diagnostics for radiotherapy treatment planning. Mediastinal patterns of lymph node metastases differed by EBUS-TBNA and PET. EBUS-TBNA showed a lobe and tumor laterality specific pattern of spread and skipping of echelon-2 was rarely seen. These systematic relations provide the opportunity to further refine multi-parameter risk models for lymph node involvement to delineate target volume based on patterns of spread by EBUS-TBNA and PET.Additional file 1: Table 4S. The respective odds ratios (OR) for EBUS-positivity in echelon-3 excluding ipsilateral LN station 2 according to the prognostic factors from multivariable analysis


Digital health research encompasses methods from human-computer interaction and health research.This paper aims to describe how these methods were combined to develop HeLP-Diabetes: Starting Out, a web-based structured education program for people newly diagnosed with type 2 diabetes.in the wild testing in the National Health Service with people newly diagnosed with type 2 diabetes, and further revisions. We adopted an iterative user-centered approach and followed steps from the human-computer interaction design life cycle and the Medical Research Council guidelines on developing and evaluating complex interventions.The development process consisted of three phases: initial design for effectiveness, optimization for usability, and in the wild testing provided data on uptake of and barriers to use. The study suggested low uptake and completion of the program, but those who used it seemed to benefit from it. The qualitative findings suggested that barriers to use included an expectation that the program would take too long. This informed refinements to the program.The initial design process resulted in an 8-session program containing information and behavior change techniques targeting weight loss, being more active, and taking medication. The usability testing was highlighted at an early stage, where changes needed to be made to the language and layout of the program. The The use of interdisciplinary methods resulted in an iterative development process and refinements to the program that were based on user needs and data on uptake. The final intervention was more suitable for a definitive evaluation than the initial version. The description of our approach informs other digital health researchers on how to make interventions more sensitive to user needs. The research methods used in HCI, such as health research, are largely empirical . However, health research tends to use a sequential approach, based on the methods used in pharmacological drug development, culminating in a randomized controlled trial to determine its effectiveness . In HCI the user -4. Accepthe user . A decisThe Medical Research Council (MRC) has published guidelines for health researchers researching complex interventions to help them adopt appropriate methods. The 2006 MRC framework suggested a nonlinear approach to the development and evaluation of complex interventions, with four key stages: (1) development, (2) feasibility and piloting, (3) evaluation, and (4) implementation [These 4 stages involve using evidence and theory to develop complex interventions, then testing them with a series of pilot studies aimed at key design uncertainties, before moving on to an exploratory and then a definitive evaluation . MRC besIterative development and evaluation are also features of the HCI development life cycles. A life cycle is the sequence of activities that occurs from the initial concept, through to the eventual phasing out and replacement . The proThe challenges of interdisciplinary work across HCI and health have been highlighted by Pagliari  and BlanThis paper focuses on this development phase, with the aim of highlighting the importance of careful user-centered design and early-stage usability testing before further evaluation. We have reported how we tackled this, by describing the 3 phases of development and describing the methods from HCI and the methods from health that we incorporated at each phase. The aim of reporting our methods is to guide other researchers in developing similar interventions. Historically, complex interventions, such as diabetes self-management programs, have not been well described ,13. BettOur approach was also informed by guidelines on evaluating DHIs , which din the wild testing stages.There are other examples where the process of combining interdisciplinary methods have been documented -19. The Guidelines for evaluating DHIs recommend starting by defining the problem to be addressed, namely the health need that the DHI is intended to address and the population who could benefit from the DHI. For HeLP-Diabetes: Starting Out (HDSO), the health need that is being addressed is the provision of a structured education for people with type 2 diabetes mellitus (T2DM). T2DM is an international priority, affecting approximately 425 million people worldwide. T2DM places a considerable burden on patients in terms of premature morbidity and mortality and on health services, in terms of cost. Both these burdens can be reduced by structured self-management education, which can improve patient knowledge, self-care behaviors, metabolic control, psychological outcomes, and health care costs -23. In tIt is an NHS policy that all patients diagnosed with T2DM are offered structured education . GeneralHowever, QOF payment and QISMET accreditation require a structured program  aimed at newly diagnosed patients. HeLP-Diabetes was not structured , and it was not aimed at newly diagnosed patients but at patients at all stages of their diabetes journey. Therefore, we decided to develop a web-based structured course that could gain QISMET accreditation and meet the QOF requirements. The established courses that the GPs could refer patients to and gain QOF remuneration were all group-based and face-to-face; thus, a web-based structured course would provide an alternative that could potentially bypass some of the barriers to uptake described earlier. The interdisciplinary development process required the skills of a multidisciplinary team of patients, GPs, diabetes nurse specialists, and health and HCI researchers. KP and JR formed part of the HeLP-Diabetes team, and KP and SP formed part of the HDSO team. EM led both teams.in the wild testing and further revisions.This paper aims to describe how we combined the methods described in the MRC and HCI guidelines, using the development of the HDSO program as a worked example. The three stages of development we undertook were as follows: (1) phase 1—initial design, (2) phase 2—optimizing for usability, and (3) phase 3—For each stage of development, we have described the methods from HCI, the methods from health, and how we combined the two. The evaluation of the final intervention for feasibility, acceptability, and impact is described elsewhere .The first steps of the HCI design process involve understanding the user context and requirements. This took place during the development of the HeLP-Diabetes website, before the development of the HDSO-structured program.wants) and features needed to help improve health outcomes (needs). The HeLP-Diabetes team conducted focus groups with patients and health professionals  to collect this information.Understanding the contexts in which people live, work, and learn allows designers to develop products that fit easily into users’ everyday lives. Products that are easy to use are more likely to be acceptable to patients and more widely taken up. Extensive work in establishing the requirements of patients with T2DM went into the development of HeLP-Diabetes, the precursor to HDSO, and has been reported by Dack et al . User rethinking aloud while undertaking prespecified tasks , a technique common and unique to HCI [The content identified as necessary in the focus groups was integrated by the design team and then reviewed by a participatory design group consisting of patients with T2DM. The content went through several iterations and was put through usability testing. Usability testing is commonly used in software engineering and HCI research. It has been described as “representative users attempting representative tasks in representative environments, on early prototypes or working versions of computer interfaces” . Usabilie to HCI . UsabiliSystematic reviews of web-based diabetes self-management interventions -42 have The aim of the structured program was not only to impart knowledge but also to empower and encourage people newly diagnosed with T2DM to improve their self-efficacy (self-confidence in self-management) and emotional well-being by learning about living a healthy lifestyle, making the most of the NHS and staying motivated. There were many theories and theoretical models which related to the aims of the program. These included the Corbin and Strauss model for the work of living with a long-term condition  and behaMedical management: adopting healthy behaviors , working with health professionals , and taking medicines. Emotional management: addressing the negative emotions associated with being diagnosed with a long-term condition.patient role and managing the impact of one’s diagnosis on relationships with friends, family, and colleagues.Role management: coming to terms with the disruption to one’s sense of self, including adjusting to the Behavior change theories were used because they can help predict how and when behavior change occurs . BehavioGuidelines on evaluating DHIs  recommenIn the long term, the proximal outcomes of knowledge, motivation, self-efficacy, and emotional distress combine to enable behavior change and better glycemic control. We opted not to measure long-term outcomes but focus on the short-term outcomes, including registration, use, and change in these 3 proximal outcomes.Findings from the work on user requirements (HCI) and evidence, theory, and causal modeling of the intervention  were combined to select appropriate BCTs and develop the content, format, and structure of the program.The usability testing of the HDSO program was conducted using questionnaires emailed to 5 patient volunteers. The questionnaires were written by the GPs and diabetes specialist nurses in the HDSO team and included the following four items: (1) the title of the session , (2) the information contained in the session , (3) the visual design of the session , and (4) any other specific suggestions.in the wild testing with patients in the NHS.Usability testing formed part of the early phase work recommended by the MRC and was undertaken to test the initial design with target users, before the exploratory Combining the phased approach advocated by the MRC and making changes to the program based on the results of 1 round of usability testing meant that the early development of the program was iterative. This was a key strength of this research. We did not immediately proceed to evaluation after the initial design of the program but undertook cycles of testing, refinement, retesting, and further refinement until we were more confident that the intervention fulfilled user needs and did not need major changes. This was recommended by both the MRC guidelines used by health researchers and the HCI lifecycle models used by HCI researchers. The refinements made to the design of the program were based on the results of the usability testing (and therefore based on user needs and experience).in the wild testing.The purpose of the next stage of testing and refinement was to evaluate the design of the program against user requirements. This was undertaken using in the wild is a term used for research conducted in natural settings. It is increasingly used in HCI to understand how people react to and integrate technologies in their everyday lives over a period [in the wild studies nearly always provide unexpected findings regarding what humans do when confronted with a digital intervention; these can be the most informative findings [Research a period ,49. In sin the wild testing was to understand more about how patients were using the HDSO program in their everyday lives, including their experiences and views of the problems they encountered in using the program. Therefore, mixed methods were used in this study. Quantitative data were collected on the number of patients registering for and completing the program, patient characteristics, and changes in questionnaire scores. The questionnaires administered were the Problem Areas in Diabetes measuring diabetes-related distress [The aim of the distress , and thedistress . Questiolocus of expertise differs. In health and social science research, researchers typically start with their own expertise rather than the user’s expertise, and the design interventions that (it is hoped that) users will engage with [user-centered approaches to address the challenge of low uptake and adherence, with a focus on understanding and accommodating the perspectives of the people who will use the intervention [Qualitative methods are used in both health and HCI research, but there are some important differences in the approaches used. For example, the age with . In HCI rvention . In healin the wild testing of the HDSO program. The setting for the in the wild testing was GP practices. This was the natural setting for this study as referrals to structured education for T2DM patients occurs in primary care. Practices in 2 London boroughs that had taken part in a HeLP-Diabetes implementation study and practices in 2 London boroughs that were interested in commissioning the HDSO program participated in the study. The program was offered to these practices for free as an alternative to established face-to-face diabetes structured education courses that were already commissioned.Methods from HCI and health were combined to conduct the The study was submitted to the Health Research Authority (HRA) for NHS Research Ethics Committee (REC) for review. Secondary analysis of information collected as part of normal care was excluded from the REC review by the HRA as long as the patients were not identifiable . TherefoA total of 15 practices agreed to offer the HDSO program to patients for the study. The program was offered to patients as an NHS service; therefore, there were no formal inclusion and exclusion criteria. Practices were informed that the target population of the intervention was adults (aged ≥18 years) with T2DM diagnosed in the last 9 months and asked to offer the program to everyone in this population. Practices were asked to identify eligible patients by running a search of the electronic medical records. Practices were sent registration packs to mail out to eligible patients. A total of 322 packs were mailed out. The registration pack contained information about the HDSO program , how to register, and a reply slip. Patients interested in using the program returned a reply slip to the HDSO administrator with their contact details. Patients were then telephoned by the HDSO administrator who collected baseline demographic data  and created a username and password for the program. The HDSO administrator also confirmed whether they were happy to take part in research interviews and securely sent SP the ID numbers of all the patients who agreed. The username and password for the HDSO program were then emailed to the patient, along with information on who to contact if there were any problems.We used qualitative telephone interviews with patients to explore their experience of using the program. We took an HCI approach of rapid data collection and used the data to inform optimization before further evaluation.One of the members of the HDSO team (SP) contacted patients who registered for the HDSO program but did not start or complete it. We were unable to contact patients who did not register for the program, as they did not provide us with their details or consent to be contacted. The patients who did not start or complete the program were contacted, because we were particularly interested in the problems encountered with the program. The telephone calls were semistructured and lasted approximately 10 minutes. Questions included “What would help you to use the program more regularly?”The interviews were carried out by telephone by SP, and written notes were taken rather than audio-recording and transcribing because the data needed to be collected and analyzed quickly to inform the program optimization. Note-taking is a recognized form of recording , and altEthical approval was obtained from the HRA (reference number: 159488). Data on registrations, completed sessions, and questionnaire scores were excluded from the HRA REC review, because of a clause that states that secondary analysis of information collected as part of normal care is excluded from REC review by the HRA, as long as the patients are not identifiable .Results from the focus groups showed that patients needed help in managing the complexities of living with diabetes, such as managing the impact that irregular working hours had on diet and blood sugar, impact on relationships and social life, and support in dealing with the profound negative emotions caused by the diagnosis, which included anger, guilt, shame, and despair. The tools to help them manage these tasks included high quality, detailed information, personal stories from other people with similar experiences, and quizzes to test knowledge and provide feedback. Health professionals had similar perceptions of patient needs. Both patients and health professionals wanted HeLP-Diabetes to be interactive and visual , to be easy to use, and have a positive tone .These results were combined with the results of the usability testing to create HeLP-Diabetes, a website containing 560 pages of information divided into 8 sections, which patients at any stage of their illness journey, could dip in and out of.We used selected content from the HeLP-Diabetes website  to construct the HDSO-structured program, which was needed to meet the QISMET and QOF requirements described in the introduction.The causal modeling process helped us postulate that information and BCTs targeting healthy eating, weight loss, activity levels, smoking, alcohol consumption, and medication intake, would help users achieve the intended outcomes. Modules targeting these behaviors were therefore selected from the HeLP-Diabetes website to be integrated into the HDSO. The modules contained BCTs, including goal-setting, action-planning, self-monitoring, and feedback on performance . These BIn addition to the BCTs from HeLP-Diabetes, self-assessment questionnaires and feedback were added as new components to the HDSO-structured course. The questionnaires assessed self-efficacy (self-confidence) in self-management, diabetes-related distress, and diabetes knowledge. These were positioned in the course in weeks 1 and 8 (before and after the program), thereby allowing users to reflect on the change in their scores.Personalized emails were also added as new components to the HDSO-structured course. These were added to encourage motivation and engagement. A systematic review by Alkhaldi et al  on the eA curriculum was needed to structure the content and components of the program and to achieve accreditation as a structured course. By identifying relevant theory (as suggested by the MRC guidelines), we decided that the program would follow a spiral curriculum based on the Harden and Stamper spiral curriculum model . This moThe qualitative work that was conducted to establish user requirements for HeLP-Diabetes showed that users wanted information to be presented using text, images, and videos. These formats were therefore used to present information in the HDSO program and included videos of others living with diabetes. The text was written for people with a reading age of 12 to correspond with 80% of the population in the United Kingdom .The result was an 8-session program containing information presented as text, images, and videos, and BCTs including goal-setting, action-planning, self-monitoring, and feedback on performance. Each session was designed to take approximately 40 to 50 minutes to complete and for people to complete 1 session per week. A screenshot of the HDSO program showing a video giving an introduction to T2DM is shown in Responses to the questionnaires described earlier were reviewed by the team. The relevant changes were made where there was agreement among responses from the patient volunteers. Where there was disagreement in responses from the patient volunteers, the suggested changes were discussed among the team members, and a consensus decision was made as to whether to make the changes. Changes were made to the language and layout of each session of the program, including making text and titles clearer and easier to understand for users.Examples of questionnaire responses and changes made to the program, as a result, are given in We made changes to the design and content of the program based on the questionnaire responses. This ensured that the development of the program was grounded in user needs. The changes included clearer subtitling, advice about physical activity made more concise, a more appropriate number of videos of people’s stories included, and links to support and advice organizations added.During the study, 24 people registered for the HDSO program. Quantitative data were collected on program use, questionnaire scores, and characteristics. Of the 24 people registered, 3 (13%) people completed the program, 13 (54%) people started the program but did not complete it, and 8 (33%) people did not start the program. The data suggested low uptake and completion, but those who used it seemed to benefit from it (mean self-efficacy in self-management scores and diabetes knowledge scores increased).The telephone interview responses were analyzed using a thematic analysis approach, and a list of barriers to completing the program emerged from the data. These are listed in These themes were used to inform the refinement and optimization of the program as discussed in the next section. The ideas were followed up in subsequent interviews conducted as part of the evaluation of the final intervention and reported elsewhere .The themes identified from the interviews suggested that there were patient and program factors which influenced program use. Patient factors such as ambivalence were difficult to address. However, we were able to shorten the program and provide users with quicker access to the program with web-based registration.Following a discussion among the HDSO team, the following changes were agreed upon:managing my diabetes when I’m ill, working with diabetes, and driving with diabetes were taken out of the main course and moved to the fifth bonus session. The final 4-session intervention is described elsewhere [getting started, self-management, improving my health and well-being, and taking control of my diabetes.Reducing the number of sessions in the program: evidence from systematic reviews of engagement with digital behavior change interventions  and reselsewhere  and contReducing the number of questionnaires: we decided to reduce the number of questionnaires from 3 to 2 by removing the AdKnowl questionnaire. The AdKnowl (knowledge) questionnaire  was remoWeb-based self-registration: it was decided to change to web-based registration to save time and to make it easier for patients to access the program quickly. The self-registration page included a demographic questionnaire, which allowed for the collection of baseline data. Telephone support from the HDSO team was still available for those who had difficulty registering on the web or using the program.We decided to offer the program to everyone with T2DM and not just people who were newly diagnosed. The HDSO program was developed in line with the national clinical guidelines for GPs advising them to offer patients with T2DM structured education at and around the time of diagnosis . Howeverin the wild testing allowed us to identify low completion rates, which were not picked up in the usability testing because it was conducted with highly motivated patient volunteers. The methods from health and the MRC framework for complex interventions emphasized the impact of the intervention. This helped us to understand the potential effectiveness of the intervention. The iterative development process that we went through with the intervention, in contrast to traditional piloting and feasibility studies conducted in health research [We have described the stages of the development of a web-based structured education program for people newly diagnosed with T2DM, HDSO. Methods from HCI and health research were used in combination at every stage. Methods from HCI put more emphasis on understanding user requirements and determining uptake. The research , resulteresearch . Randomiresearch .The existing reporting of complex behavior change interventions is limited, and this prevents successful replication of successful interventions . ReviewsThe strength of this research is the use of methods from both health and HCI. We combined the theory of living with long-term conditions and a user-centered design to understand and meet user requirements. This meant that users of different age groups, education levels, and ethnic backgrounds could use the intervention, as demonstrated by the analysis of the usage data from subsequent studies . A weaknAnother weakness of our approach was patient involvement. More extensive patient involvement could have been used in the design of the interview guide for the telephone interviews in the user experience study and data analysis. The patient volunteers could also have been asked to make their own suggestions for refinements to the first iteration of the program, instead of relying solely on the data.This paper describes how interdisciplinary methods can be used to develop a web-based structured education program for people newly diagnosed with T2DM. Methods were combined from human-computer research and health research. The reporting of the development processes for DHIs needs to continue, especially when interdisciplinary methods are used, for researchers to be able to learn from each other and create user-centered interventions.


Social isolation affects one in five older adults, significantly increases the risk of premature death from all causes and is associated with higher rates of depression, anxiety and suicide. Covid-19 has exacerbated social isolation, including among older adults that reside in senior apartments. In response, a Friendly Caller Program was developed to foster intergenerational social connections among university students and residents in a large housing community that serves older adults aged 62 and older who have limited income and have mobility impairments. This study aimed to evaluate the Friendly Caller Program from the perspective of the older adult. An online survey includes questions about the participants’ demographic characteristics, physical and mental health self-assessment, social support, and ways in which the Friendly Caller Program has affected these areas of their life. An open-ended question assesses older adult participant expectations of the Friendly Caller Program. Results describe the population currently being served by this program and indicate that the program has a positive influence on participants’ feelings of safety, support and ability to function. Suggestions for future research include assessing university student perceptions about the benefits of participation as callers in the program and creating a toolkit that can guide other universities to create similar programs in partnership with housing communities that serve older adults.