Barriers and facilitators experienced in collaborative prospective research in orthopaedic oncology

Statistical analysis

We used a qualitative descriptive approach for analysis, a method that aims to provide a descriptive summary of the data, in the language of the participants, with minimal theoretical interpretation.⁶ Transcripts and field notes from the discussion were analysed using conventional qualitative analysis, with codes being derived directly from the data rather than using preconceived categories.⁷ Four investigators (SR, MS, MG and PS) independently undertook line-by-line coding of the field notes and transcript, and met to develop the list of codes by consensus. They also assessed saturation of the data and agreed when saturation was achieved (i.e. when no new information was being provided). The research team reviewed the list of codes, discussed the relationships between them and organised them into categories, a process known as axial coding.⁸ The qualitative software programme NVivo, version 10.0 (QSR International, Doncaster, Australia) was used for data management and analysis. Quantitative demographic data were analysed using descriptive statistics.

The need for collaborative research

All participants valued collaborative research and agreed that it is needed in orthopaedic oncology due to the rare nature of bone tumours:

“[t]here are just things that we cannot answer by ourselves given the nature of [the] disease.” And: “We work on difficult problems; these are ultra-rare diseases and without collaboration, it’s difficult for me to envisage how there’s going to be meaningful numbers and clinical research to really drive the field forward”.

Surgeons described how collaboration on multicentre trials is “long overdue – it’s something that we’ve been talking about for years and years” and is necessary in order to generate evidence:

If you’re a teaching hospital and your residents say, “Dr. X, why do you keep antibiotics on until the drain is out?” And my best answer is, “Shut up, kid, that’s the way I do it.” [group laughter] …I’m supposed to be an academic physician - that’s not okay.

One participant acknowledged that “evidence-based medicine is the future” while another commented on the lack of evidence-based research in the field:

I think that at this past meeting that 95% of the papers are retrospective studies. There was practically [no] randomised, controlled, multi-centre….this is what has to be done to really get evidence going in our field.

One surgeon explained: “We’ve heard a lot of information from retrospective stud[ies] and you can’t base all of your decisions on retrospective studies.”

Motivational factors for participating in collaborative research

The participants described various factors that motivated them to participate in collaborative research including proof of principle, relevance of the research question, the opportunity for learning how to do larger-scale research and wanting to be involved in a collaborative research community. As PARITY is the first RCT undertaken by orthopaedic oncologists, some discussed the need to establish that the orthopaedic oncological community can successfully complete an RCT as a reason for participating, “It’s a really good study to demonstrate that this is actually possible on an international basis.” And:

The PARITY trial is unprecedented. I think having a bunch of surgeons get together, involved in a truly randomised, controlled trial… it’s bit of a proof of principle project…this is trailblazing and I think it’s exciting for all of us who are involved.

One surgeon described how the relevance of the research question motivated him to participate and reflected on how it might motivate others to participate:

One of the best things for me, to get involved in this program…is the good question … [infection is a] very important problem, that all of us, independent of the country we work in, in the hospital we work in, we have that problem…if you do musculoskeletal oncology, you get infections…if you get a good question to be answered, people would get involved.

A junior surgeon described how the opportunity to learn about the research process was a motivation for him:

I think, as one of the younger members of the collaboration, it’s an extremely valuable learning opportunity just to see what goes into this process… just to know what goes into it and all the work and the time and barriers that there are.

A number of participants spoke of the desire to be part of a research community as motivation:

[A]fter a while, everybody’s participating and there’s almost this sense that you have to be a part of it, that this is something big and different and practice-changing going on and you really want yourself and your institution to be making a contribution.

Eventually [someone else is] going to come up with some idea that we’re all going to want to trial. Or maybe I’ll ask a basic science question that I think is suitable for the larger membership…how am I going to expect the membership to participate if I haven’t voted with my feet? …As we move forward, we’re all going to have ideas that we want to see asked this way. So, I think it’s important to support each other in this.

Barriers to participating in collaborative research

While participants highlighted the overwhelming need and motivation for participating in collaborative research, all described the many barriers to their participation, including those from sites with existing research infrastructure and prior research experience. Some spoke of the difficulties presented by involvement in collaborative research:

I never envisioned how difficult it would be to do this… Lawyers are involved; exchange of information; all that stuff gets into play…it sounds like, in a perfect world, it would be easy but it really is not.

One of the things that I’ve learned from this is that these types of studies are really hard…I can’t think of a simpler question with a less toxic intervention that what we’re doing right now and it has been incredibly difficult, I think, for everybody involved to participate … So, yes, it can be done but we really have to think about the questions we’re asking and make sure that they’re going to be important if we want to be successful.

There was a collective sense of fatigue expressed by surgeons, and an articulated sense of needing to “fight” to overcome barriers and participate in this kind of research. One participant explained that: “There’s always more roadblocks, you figure you’re trying to do something for a good cause, you know, decrease rates of infection and it all makes sense but you just keep hitting blocks along the way. It gets a little tiring after a while.”

The barriers which were discussed were classified into the following: funding; personal barriers; institutional barriers; trial barriers and administrative barriers. Limited funding was identified by several surgeons as a barrier to their participation. One explained that:

The financial limitations - it probably all goes back to that…… if something’s going to make you money, usually you can find people to help you do it so it’s difficult to advocate something just on scientific merit, especially if in our institution being in PARITY actually costs us money rather than not even bringing in money.

[Our institution] just create[s] new barriers so we cannot go through the process. There’s some legal stuff and they just said that we cannot get money from elsewhere in our country but they don’t want to give us money…

Personal barriers were related to demanding workloads and limited time:

For most people that I’ve been talking to about this trial [they] would maybe, in principle, very much like to participate but fear that they are already so much drowning in work and don’t have local finance and research support that it’s really very difficult to envision that, with all the work that you already have to take responsibility for that you can take on another responsibility to contribute on a reasonable basis.

The staff is supportive of all this but it’s been more time consuming than I had imagined. I haven’t had the time to dedicate to it but I think the amount of time that it needs.

Institutional barriers included a lack of research staff and infrastructure, and a lack of recognition. Some surgeons specifically identified the lack of research staff:

I was kind of smiling when I looked at the registration form specifying the number of staff surgeons that we’ve got in our department, which are eight…And the number of allocated research personnel is zero… that I think is the most prominent barrier for us.

Frankly, most people don’t have the research support except at specific institutions to do it and you really need to have a study coordinator, at the very minimum to do these sorts of studies.

Trial barriers included the lack of relevance of the protocol to their practices (related to the type of antibiotic) and the lack of enthusiasm from interdisciplinary partners. One surgeon explained how the country in which he works uses a different antibiotic, and how that in turn influences the relevance of the research question:

PARITY approached this by trying to simplify the question and really get two homogeneous groups but then that goes into detriment, especially with European centres, where for example, the findings of Cefazolin being two grams post-op, that’s not what we’re going to do… So for us, as European centres, it’s going to answer a worldwide question about length of antibiotics but not about our own antibiotic or current clinical practice.

A number of surgeons described how a barrier exists when the research question doesn’t resonate with other disciplines:

The ID guys might get excited that some long term benefit is there if we kind of understand our usage of antibiotics better but folks who are used to seeing oncologic studies with really clear outcomes that you’re trialing a new drug, I mean, just trying to pitch them that this is an important topic was really hard.

It’s an awkward position to be in because orthopaedic departments generally don’t know how to run randomised clinical trials so the support staff is not within your department. And then the cancer centres don’t understand what you’re doing anyway. It’s kind of hard to advocate that this is actually a cancer study.

Administrative barriers included the large amount of paperwork related to participating in an RCT and, for some participants, the time involved in translating the trial documents into the language spoken in their country.

Facilitators for participating in collaborative research

Facilitating factors for participating in research were organised into the following: leadership; institutional facilitators; authorship; trial set-up; and Methods Centre support. Leadership at different levels was seen as a facilitator. One surgeon described how the support of leadership from professional societies influenced his participation in the PARITY trial:

When we hear our Society leadership at the podium every year talking about [PARITY] and how important it is and how exciting it is - it’s hard not to buy into that and I think having the leadership buy in was certainly a factor for me in trying to implement this in our institution.

A junior surgeon shared how the support of the fellowship directors had a positive influence:

The other part, as a younger guy going through is seeing a lot of the Fellowship directors jumping on board and being really enthusiastic about PARITY ….you sort of see these people as the leaders and the teachers in the field and it seems meeting after meeting, they’re involved, they’re supportive of it….it’s a really nice way to kind of pyramid down from [the Principal Investigator for the trial] to the Fellowship directors to the younger members of the Society.

Many spoke highly of the leadership shown by the Principal Investigator for the PARITY trial and how it is a facilitator for collaborative research: “[Her] enthusiasm is infectious for this, there’s no question…I think that [her] enthusiasm and her drive and passion for this is a huge enabling factor in seeing its success.”

Institutional facilitators included research infrastructure, industry to subsidise funding from an investigator-initiated trial, and supportive colleagues. One surgeon described how the research infrastructure enabled his site to participate:

For our division of orthopaedics there are six surgeons - two of us, oncology surgeons…and we’ve got three and a half full time clinical research staff and two and a half of those dedicated to oncology so I will admit, freely admit, that is a huge enabling factor for us that permitted us to participate right off the bat.

Another participant explained how his site did not have a lot of infrastructure, but used funding from industry to offset the financial losses resulting from participating in investigator-initiated studies:

At our institution, there’s a long track record for people doing industry studies. And because of that, we’ve been able to support clinical research coordinators within the department… I’ve been able to do a few industry-based studies that support my research coordinator and that has basically funded her to do trials like this where we’re not really going to make any money on it.

Supportive colleagues were identified as facilitators by some participants. One described how having an advocate at his institution facilitated his institution’s involvement with PARITY:

I have a colleague who’s a medical oncologist who starts a lot of clinical trials and brings a lot of money into the department…he understands the importance of our institution being a part of a big effort like this. That it would be good in the long run for our institution even though there’s no financial compensation and he actually championed that and said “Look guys, I know we’re losing money but we have to do this and we’re doing it.”

Authorship was identified as a facilitator for participation at both the individual and at trial level:

I’d be lying if I said authorship didn’t matter. It does. Most of us are in academic centres. The currency that I deal in is with publications…participating in studies like this adds gravitas to my list of publications so from a completely selfish standpoint, it is helpful.

Authorship is really helpful and also gives a confirmation of the validity of the process and the validity of the trial.

The process of how the PARITY trial was set up and presented to surgeons was also identified as facilitator:

I think a lot of the reason that PARITY, specifically, was successful is that it was well set up and vetted before it started.

The setup process was transparent in that the people who were eventually going to be participants saw the evolution of the question.

This is a question that, I think, [the Principal Investigator for the PARITY] demonstrated that there’s equipoise for. If you go through that process, then you realize we don’t know what the right answer is and if you buy into that, then you can enrol patients.

Support from the PARITY Methods Centre was described as a “fundamental” facilitator by offering assistance with the legal and regulatory processes related to trial participation. One surgeon explained that, “to have a consistent contact is certainly helpful, and I would say, critical, in a situation like this.”

Strengths and limitations

Musculoskeletal tumours are rare and often described as ‘orphan’ diseases. The strength of this study is that, to date, orthopaedic oncologists have not explored these areas of qualitative research. This study also might be used to investigate research into other rare diseases, in which there may be similar challenges in conducting RCTs. Moreover, this study’s infrastructure is based on PARITY, which is, for the first time, capturing and exploring the experiences of orthopaedic oncological surgeons in different stages of involvement in RCTs. It therefore represents a step forward for future large-scale research in orthopaedic oncology and other ‘orphan’ conditions.

The information for the study was collected from a focus group discussion. The group was multinational and, given the qualitative nature of the research, this was beneficial and enriched the information obtained. However, a limitation of this study is that some international investigators may not have been able to attend the discussion for logistical reasons of time and cost of travel to the United States. Nevertheless, the discussion eliminates the challenges encountered in multicentre studies by relying on information collected at a single time, without the requirement for continued participation and contribution.

Although important challenges to collaboration have been encountered though this qualitative research study, most have been overcome thanks to a growing interest in maintaining a solid collaborative network, which strives to find answers of common benefit to orthopaedic oncologists. The focus group strategy implemented, using PARITY as the means to answer our research question, is therefore an example of how a scientific society can produce and share knowledge with the goal of overcoming challenges and consolidating scientific efforts to facilitate future large-scale research studies.