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Address correspondence to: Marie T. Williams, PhD, Innovation, Implementation And Clinical Translation in Health (IIMPACT), School of Health Sciences, City East Campus, University of South Australia, North Terrace, Adelaide, 5000 South Australia, Australia.
Innovation, Implementation And Clinical Translation in Health (IIMPACT), School of Health Sciences, University of South Australia, Adelaide, South Australia, Australia
Innovation, Implementation And Clinical Translation in Health (IIMPACT), School of Health Sciences, University of South Australia, Adelaide, South Australia, AustraliaDepartment of Kinesiology and Physical Education, Faculty of Education, McGill University, Montreal, Canada
School of Rehabilitation Sciences, McMaster University, Hamilton, CanadaDepartment of Respiratory Medicine, West Park Healthcare Centre, Toronto, Canada
Explanations provided by health professionals may underpin helpful or harmful symptom beliefs and expectations of people living with chronic breathlessness.
Objectives
This study sought perspectives from health professionals with clinical/research expertise in chronic breathlessness on priority issues in chronic breathlessness explanations and research.
Methods
Authors (n = 74) of publications specific to chronic breathlessness were invited to a three-round Delphi survey. Responses to open-ended questions (Round 1 “What is important to: include/avoid when explaining chronic breathlessness; prioritize in research?”) were transformed to Likert scale (1–9) items for rating in subsequent rounds. A priori consensus was defined as ≥70% of respondents rating an item as important (Likert rating 7–9) and interquartile range ≤2.
Results
Of the 31 Round 1 respondents (nine countries, five professional disciplines), 24 (77%) completed Rounds 2 and 3. Sixty-three items met consensus (include n = 28; avoid n = 9; research n = 26). Explanations of chronic breathlessness should use patient-centered communication; acknowledge the distress, variability, and importance of this sensation; emphasize current management principles; clarify maladaptive beliefs and expectations; and avoid moral culpability and inappropriate reassurance. Research priorities included the need 1) for a comprehensive understanding of breathlessness science; 2) to optimize, explore, and develop effective interventions, both pharmacological and nonpharmacological; and 3) determine effective models of care including strategies for education and training of health professionals and people caring for, or living with, chronic breathlessness.
Conclusion
These consensus-based concepts for chronic breathlessness explanations and research provide a starting point for conversations between patients, carers, clinicians, and researchers within the chronic breathlessness community.
This Delphi survey of international professionals with expertise in chronic breathlessness identified key concepts for breathlessness explanations and research. When explaining chronic breathlessness: Acknowledge impact, Avoid blame, Believe something can be done, Clarify maladaptive beliefs. Research priorities build on progress made in breathlessness science to translate findings to optimal practice.
Introduction
Chronic breathlessness is a distressing, disabling daily symptom which persists despite optimal treatment for many people with chronic respiratory, cardiovascular, and neuromuscular conditions.
Although almost always pathophysiological in origin, an individual's awareness of breathlessness is informed by sensory, cognitive, and psychological factors.
Within predictive coding frameworks, an individual's interpretation of breathlessness is influenced by sensory input and symptom expectations or priors.
and cognitive factors (past experiences, learned associations, beliefs). Breathlessness beliefs and expectations are also informed by disease- or symptom-specific information provided by health professionals, friends, family, and increasingly, the Internet.
Illness beliefs and expectations influence an individual's coping and health behaviors where unfavorable or maladaptive beliefs are associated with worse health outcomes.
A health professional's beliefs and expectations of a clinical condition can profoundly influence patient beliefs, expectations, and behaviors, especially in chronic conditions where perceptions of moral culpability exist.
Physiotherapists' beliefs and attitudes influence clinical practice in chronic low back pain: a systematic review of quantitative and qualitative studies.
In other fields, notably chronic low back pain, health care professionals have been reported to have the strongest and most enduring influence on patients' attitudes and beliefs.
The association between health care professional attitudes and beliefs and the attitudes and beliefs, clinical management, and outcomes of patients with low back pain: a systematic review.
clinicians do not discuss this frightening symptom with patients due to a perceived inability to offer effective palliation and lack of specific training or awareness of appropriate services. Consequently, the explanations used (or avoided) by health professionals for the mechanisms of problematic symptoms may contribute both positively and negatively to patients' beliefs and expectations.
publications focused on breathlessness science have progressively increased (Scopus 2013–2018 median 97 publications per annum; 2006–2011 median 61 per annum). More recently, the need for better education and research specific to chronic breathlessness has been identified by people living with chronic breathlessness, informal carers, and health professionals.
Six key topics informal carers of patients with breathlessness in advanced disease want to learn about and why: MRC phase I study to inform an educational intervention.
Cowan K on behalf of the James Lind Alliance Mesothelioma priority Setting Partnership Steering Committee Research priorities in mesothelioma: a James Lind alliance priority setting partnership.
ATS/ERS task Force for COPD research An Official American Thoracic Society/European respiratory Society statement: research questions in chronic obstructive pulmonary disease.
Although a “breathlessness curriculum” may include a range of topics, one underexplored yet potentially impactful aspect of better breathlessness education is how chronic breathlessness is explained. In the absence of current recommendations, we sought the perspectives of health professionals with clinical and/or research expertise in chronic breathlessness via a Delphi consensus survey to identify priority issues in explanations and research for chronic breathlessness.
Methods
This study used a three-round Delphi survey process informed by Diamond et al.,
Using the Delphi technique to determine which outcomes to measure in clinical trials: recommendations for the future based on a systematic review of existing studies.
potential participants were identified based on three criteria: 1) recognized expertise in chronic breathlessness (dyspnea), evidenced by recent publications or involvement in key breathlessness-related consensus guidelines; 2) multiprofessionality; and 3) broad geographic spread. This sampling frame aimed to recruit clinicians/researchers active in generating knowledge in breathlessness science. Rather than undertaking two separate surveys for explanations and research priorities, we opportunistically combined these questions into one survey to reduce responder burden. Historically, Delphi surveys have comprised 15 to 20 participants, with fewer participants potentially leading to underrepresentation of opinion.
Evaluation of nine consensus indices in Delphi foresight research and their dependency on Delphi survey characteristics: a simulation study and debate on Delphi design and interpretation.
We planned to identify at least 70 potential expert participants, with a minimum target of 20 completing each survey round.
Recruitment
Using the author search function of Scopus, original articles published from 2012 to 16th April 2018 that included “breathlessness” or “dyspn(o)ea” within the title or key words were identified. Authors were cross-checked against key source documents for chronic breathlessness: two national statements (U.S.,
E-mail addresses of identified authors were searched via public websites/corresponding author lists in publication.
Delphi Procedure
Chronic breathlessness was defined within the survey as “breathlessness that persists despite optimal treatment of underlying pathology and results in disability.
The initial survey was pilot-tested by research team members locally and residing outside of Australia for access, clarity, wording and time to complete. Further details on survey processes are provided in Appendix I.
Round 1
Round 1 of this Delphi survey comprised three sections: 1) study information, including consent processes and individual identification code; 2) responder demographics; and 3) a series of open-ended questions
Using the Delphi technique to determine which outcomes to measure in clinical trials: recommendations for the future based on a systematic review of existing studies.
Information that is important to include when explaining chronic breathlessness to a person living with this symptom includes …;
2)
Information or specific terms which should not be included (i.e., avoided) when explaining chronic breathlessness to a person living with this symptom includes …; and
3)
In your opinion, which three to five questions should be priorities for breathlessness research in the next five years?
Round 1 Data Management
Verbatim responses to each of the three open-ended questions were reviewed and independently coded. Statements were developed into items suitable for rating on a nine-point Likert scale (1 = Least Important to 9 = Most Important) and prospectively grouped as “Least Important” (1–3), “Neither Least nor Most Important” (4–6), and “Most Important” (7–9). A priori criteria for consensus agreement required ≥70% of respondents to rate an item as “Most Important” with an interquartile range (IQR) of the median Likert score ≤2.
Rounds 2 and 3
In both rounds, respondents were invited to rate items for importance. In Round 2, respondents could suggest additional items (free text), and where this did not duplicate an existing item, a Likert style statement was created for rating in Round 3. New items and items not reaching a priori consensus in Round 2 were retained for rerating in Round 3.
As part of Round 3, participants were provided detailed feedback on items that did/did not meet consensus in Round 2. Participants were encouraged to review and consider feedback before rating items in Round 3.
Using the Delphi technique to determine which outcomes to measure in clinical trials: recommendations for the future based on a systematic review of existing studies.
Participant demographic data were analyzed descriptively. All items meeting a priori consensus criteria (Rounds 2 and 3) were collated. Items meeting the first consensus criterion (i.e., ≥70% participant agreement as “Most Important”), but not the second dispersion measure (i.e., IQR ≤ 2), were also retained for inclusion in the final recommendations (to account for response rates between rounds
Evaluation of nine consensus indices in Delphi foresight research and their dependency on Delphi survey characteristics: a simulation study and debate on Delphi design and interpretation.
). Response stability was assessed by comparing the percentage of identical items in both Rounds 2 and 3, which consistently did not meet consensus. Concepts reaching consensus for chronic breathlessness explanations and research priorities were drafted, reviewed by the research team for duplication of concepts and language style, and provided to Delphi respondents for information and feedback (n = 4 responded, no amendments suggested).
Results
Of the 80 potential experts in chronic breathlessness identified, 74 individuals (53% male) were invited to participate in Round 1 (April 2018). Participant flow and item evolution are presented in Fig. 1. Demographic details for respondents and comparison between survey rounds are presented in Table 1. By close of Round 1, 33 responses had been received (45% response rate), of which two respondents provided no data beyond consent (followed up with alternative to online survey without response). With the exception of therapists and scientists, respondents reflected the proportion of authors (country and professional disciplines) included in the original invitation list. Most respondents had direct clinical contact with patients over the previous six months (42% in nonclinical roles) and had been involved in a conversation with a patient about chronic breathlessness at least once a day (13%), week (32%), or month (26%). Self-reported expertise in chronic breathlessness was high (median score = 84, IQR = 20).
Fig. 1Delphi process—participant and item flow. *Round 1 response rate reflects “worse case” as we cannot confirm that all invitations were delivered and read by their intended recipients.
What Is Important to Include or Avoid in an Explanation of Chronic Breathlessness for a Person Living With This Symptom?
In total, 79 items were generated and rated (important to include n = 56; avoid n = 23) in Rounds 2 and 3. Of these, 40 identical items (include n = 25; avoid n = 15) were rated in both rounds because they did not reach consensus in Round 2. Response stability was high (item did not meet consensus in either round: include = 21/25, 84%; avoid = 15/15, 100%). At the end of Round 3, 28 items (include) and nine items (avoid) met a priori consensus (Appendix I).
Concepts important to include in chronic breathlessness explanations emphasized initiating or establishing beneficial and adaptive beliefs/expectations (e.g., “something can be done,” “not harmful in itself,” “treatment options to reduce impact”). Concepts important to avoid in chronic breathlessness explanations emphasized reducing or extinguishing maladaptive beliefs/expectations (e.g., “breathlessness should be avoided”, “nothing more can be done”, “reflects low levels of oxygen/will always be relieved by oxygen”). Concepts important to include or avoid in chronic breathlessness explanations are presented in Table 2 and Table 3, respectively.
Table 2Concepts Important to Include in an Explanation of Chronic Breathlessness for a Person Living With This Symptom
Conversations About Chronic Breathlessness Should Be Continuous and Ongoing When Explaining Chronic Breathlessness to a Person Living With This Symptom, It Is Important …
To:
Use person-centered communication:
•
Speak to the level of understanding of the person
•
Include partners and/or carers in the conversation
•
Encourage the person to express their fears and concerns
Derived from 28 items reaching a priori consensus and six items meeting ≥70% but IQR >2.
Ask what activities result in breathlessness (and tie explanations to these activities) Assure that all reversible (treatable) causes of breathlessness have been sought
Table 3Concepts Important to Avoid in an Explanation of Chronic Breathlessness for a Person Living With This Symptom
Conversations About Chronic Breathlessness Should Be Continuous and Ongoing When Explaining Chronic Breathlessness to a Person Living With This Symptom, It Is Important to …
Avoid:
•
Blaming/shaming the person or permitting the person to blame/shame themselves
Research Priorities for Breathlessness in the Next Five Years
In total, 82 items were rated across Rounds 2 and 3. Of these, 55 items were rated in Rounds 2 and 3 as they did not reach consensus in Round 2 (response stability = 51/55, 93%). At the end of Round 3, 26 items met consensus (Table 4; details can be found in Appendix I).
Table 4Breathlessness Research Priorities for the Next Five Years
Domain
Specific Item
Basic science of breathlessness
Identify and understand:
•
Neural markers of breathlessness
•
Key pathways involved in central processing
•
Differences in central processing between diseases
•
Pathological mechanisms of breathlessness between diseases
•
Physiological and behavioral mechanisms underpinning relief of breathlessness (pharmacological and nonpharmacological)
•
Role/impact of anxiety, stress, and mood on breathlessness and management
Pharmacological approaches for breathlessness relief
Validate and standardize pharmacological approaches using known compounds Determine:
•
Optimal pharmacological management
•
Optimal opioid management
•
Long-term effects of low-dose opioids
•
Predictors of benefit from opioids
•
Alternatives to opioids (e.g., cannabinoids)
Nonpharmacological approaches for breathlessness relief
Develop simple, user-friendly tools for daily self-management Identify and understand:
•
Optimal models for self-management for patients and carers
Items meeting consensus reflected a range of domains: basic science; pharmacological; nonpharmacological; assessment; psychosocial; comparative studies; organization and delivery of care; and education and training. Research questions ranged from mechanistic (e.g., neural markers, key pathways in central processing), confirmatory (e.g., predictors for benefit from opioids), exploratory (e.g., identify novel alternatives to opioids, patient/carer self-management), translational (e.g., behavior change in patients and health professionals, integrated breathlessness service models), to “big picture aspirational” (e.g., creation of multicenter and national collaborative(s)).
Discussion
This Delphi survey of multidisciplinary international researchers and clinicians resulted in a number of consensus-based agreements. In summary, using a foundation of patient-centered communication, explanations for chronic breathlessness should
1.
Acknowledge the distress, variability, and importance of this sensation;
2.
Emphasize current management principles;
3.
Clarify maladaptive beliefs and expectations; and
4.
Avoid moral culpability and inappropriate reassurance.
Research priorities encompassed the need to
1.
Develop a more comprehensive understanding of breathlessness science;
2.
Optimize, explore, and develop further effective interventions, both pharmacological and nonpharmacological; and
3.
Determine effective models of care including strategies for education and training of health professionals and people living, or carer for someone, with chronic breathlessness.
In survey research, there is no universally agreed threshold for adequacy of response rate (often considered as a proxy for representativeness of the target audience), though journal editors have been reported to have informal policies for acceptable response rates when considering publication.
Response rates for Delphi approaches are likely to be lower than cross-sectional surveys due to survey iterations which impose a greater degree of respondent commitment and burden. In multidisciplinary Delphi studies for breathlessness, participation rates are commonly lower where potential participants have been “cold contacted” from publication databases (Round 1 invited/accepted: present study 33/74 [45.6%], 31/68 [45.6%],
). Although response rates vary, the number of respondents commencing and participating in this present study is consistent with prior Delphi surveys concerning breathlessness.
Explaining Chronic Breathlessness to a Person Living With This Symptom
Providing an explanation of chronic breathlessness may be part of diagnostic and/or therapeutic conversations in a variety of clinical settings. There is a growing evidence base indicating that both “what” and “how” health explanations are communicated and impact patient beliefs, perceptions, and health behaviors.
Physiotherapists' beliefs and attitudes influence clinical practice in chronic low back pain: a systematic review of quantitative and qualitative studies.
The intent of this Delphi survey was not to devise a “one size fits all” generic script for explaining chronic breathlessness in a single conversation with a person living with this symptom. As commented by a number of respondents, the content of chronic breathlessness explanations is part of ongoing conversations and needs to be customized to the individual and her/his caregiver(s). In the absence of any specific patient scenario or disease context, respondents faced a difficult task when considering what is important to include or avoid in a generic explanation. Nonetheless, there was a clear agreement for a range of key concepts perceived to be important when explaining chronic breathlessness.
Studies of both patient and carer groups report the need for clearer explanations about the disease and symptoms such as breathlessness.
Six key topics informal carers of patients with breathlessness in advanced disease want to learn about and why: MRC phase I study to inform an educational intervention.
Few items concerning specific physiological/neurophysiological mechanisms of breathlessness were volunteered within this Delphi for inclusion in explanations (none of which reached consensus). This might reflect 1) the lack of specific patient and context detail provided to respondents, 2) a move away from ascribing chronic breathlessness as a direct result of specific organ pathology, and/or 3) a perception that this degree of information is not always essential/important in an explanation of breathlessness.
Respondents in this study were likely to represent “change-agents” of breathlessness science, many of whom challenge the therapeutic nihilism related to chronic breathlessness.
Accordingly, over half of the items reaching consensus (include) described management principles reflecting a proactive focus on treatment possibilities and evidence-based management approaches (drug and nondrug options, eliminating other contributing factors, changing reactions to breathlessness). These items also lay a foundation for building adaptive/beneficial beliefs and expectations of breathlessness. The strongest recommendations for concepts to avoid in an explanation of breathlessness were shame and hopelessness, which have been associated with diagnosis concealment, delayed medical help-seeking, and greater symptom severity in people with chronic lung conditions.
Items reaching consensus (avoid) might also reflect the commonest, unhelpful beliefs or expectations respondents of this Delphi come across in clinical or research practice (e.g., inconsistent relationships between chronic breathlessness and arterial oxygen saturation, value of supplemental oxygen in normoxemia, harm/tissue damage). Wherever possible, the existence of these maladaptive beliefs should be explored with patients and caregivers when explaining chronic breathlessness.
While there is still much to do, research priorities reaching consensus in this present study suggest new priorities are emerging. Greater refinement is proposed for pharmacological interventions (alternatives, optimizing, predicting benefits, safety profiles, and target groups) and a growing appreciation of the education and service needs of people living, or caring for a person, with chronic breathlessness.
These emerging priorities reflect the translation of breathlessness science into clinical practice at both an organizational (breathlessness services) and individual (patient, carer, and health professional) level, with a greater emphasis on comprehensive education and culture change among health professionals.
Strengths and Limitations
This study used an a priori definition of consensus based on agreement and dispersion, transparent reporting of data management and synthesis into recommendations, and independence of the researchers through noncontribution to data or recommendations. Our target of 20 respondents per round was met, retention rates (response rate Round 2 = 27/33, 82%; Round 3 = 24/31, 77%) were acceptable and participants who completed all rounds did not differ markedly from those who discontinued. We chose to “cold call” experts irrespective of professional discipline, based on predefined eligibility criteria (authorship) but recognize that the pool of health professionals with expertise in chronic breathlessness is much wider. In suggesting priorities for breathlessness research (Round 1), respondents may have focused on chronic breathlessness — though the responses suggest a broader interpretation (Appendix I). Respondents underrepresented therapists and basic scientists indicating that views expressed within the survey rounds may not fully represent all those originally invited. The findings of this Delphi survey should not be interpreted to mean that only those items reaching consensus are worthy of consideration. Items that did not reach a priori consensus criteria indicate that, for this specific responder group, the views for any given item were simply less harmonious.
Conclusion
Chronic breathlessness is a complex symptom, which many health professionals, let alone people living with or caring for someone with this symptom, find challenging to understand and manage. The findings of this study provide a researcher-clinician perspective on a framework for explaining chronic breathlessness and research priorities. Important concepts when explaining chronic breathlessness might be summarized as follows: A-Acknowledge the distress/impact and Avoid blaming; B-Believe that something can be done; and C-Clarify maladaptive expectations and beliefs. The research priorities generated by this study reflect the progress and translation of breathlessness science into clinical practice. Whether people living, or caring for someone, with chronic breathlessness volunteer and endorse similar concepts and priorities as those identified by expert health professionals remains to be seen.
Disclosures and Acknowledgments
The research team would like to thank all Delphi respondents for their time and perseverance in participating in this study. The following respondents provided permission to be acknowledged and named: Peter Allcroft, Claudia Bausewein, Sara Booth, Patricia Davidson, Magnus Ekström, John Hansen-Flaschen, Ann Hutchinson, Rhys Hurst, Suzanne C. Lareau, Robert Lansing, Paula Meek, Capucine Morelot-Panzini, Richard A. Mularski, Mark B. Parshall, Richella Ryan, Richard M. Schwartzstein, Thomas Similowski, Steffen T. Simon, Tracy Smith, Anna Spathis, Andreas Von Leupoldt, and Janelle Yorke.
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.
Appendix I. Method Detail
Round 1 Survey and Process
Survey rounds were prospectively planned to be electronically disseminated (Survey Monkey) for time efficiency and geographic reach. The Round 1 survey included three sections comprising study information (including consent processes and individual identification code), responder demographics, and a series of open-ended questions. Respondents were invited to provide general information concerning professional discipline, country of employment, current involvement in clinical management of people living with chronic breathlessness and self-rate their expertise in chronic breathlessness on a 0-100 scale (“I would rate my expertise in chronic breathlessness” as: 0 = Do not consider myself an expert to 100 = Extensive understanding of theoretical and/or clinical aspects). Respondents were invited to outline their usual explanation of chronic breathlessness for a person living with the symptom (open-ended), whether this explanation would change if the person had a specific chronic condition (yes/no) and if yes, how the explanation would be modified (open ended). The basis for subsequent Delphi survey rounds focused on the three remaining open-ended questions:
1.
Information that is important to include when explaining chronic breathlessness to a person living with this symptom includes …;
2.
Information or specific terms which should not be included (i.e., avoided) when explaining chronic breathlessness to a person living with this symptom include …; and
3.
In your opinion, which three to five questions should be priorities for breathlessness' research in the next five years?
In Round 1 (and subsequent Round 2 and 3 surveys), respondents were provided with an opportunity to provide further comments or clarification at the end of the survey. The Round 1 survey was pilot-tested with research team members locally and residing outside of Australia for access, clarity, wording, and time to complete. After minor modifications, Round 1 survey was finalized.
Potential expert participants were invited to participate in this Delphi survey by e-mail, which included an embedded link to the Explain Chronic Breathlessness Delphi Survey. Each recipient of the invitation e-mail was allocated a unique identification code, encouraged to respond within 14 days, with follow-up e-mails sent after one week and the day before the closing date of the survey.
Round 1 Data Management
A single member of the research team (M. T. W.) downloaded the survey data into Excel, descriptively analyzed responses to demographic questions, and allocated a new identification code to each responder for use during coding of verbatim responses to the three open-ended Delphi questions (important to include in an explanation, should not be included, and research priorities). The same member of the research team (M. T. W.), reviewed each respondent's verbatim response to each Delphi question and identified and coded separate concepts within the response. On completion, all original verbatim responses and provisionally coded concepts within each response were independently reviewed by two team members (K. N. J./D. B.) with disagreements resolved by discussion. The final set of coded concepts for responses was randomized for each of the three research questions. Working in teams of two (M. T. W. and H. L., K. N. J. and D. B.), each member was instructed to independently work through the list of coded statements for each research question, group similar concepts, and iteratively develop a category or domain names for similar concepts. On completion, teams met to discuss and resolve discrepancies, finalized a domain name for similar concepts, and developed statements for each concept suitable for rating using a nine-point Likert scale (1 = Least Important to 9 = Most Important) in subsequent survey rounds.
The creation of Likert style statements suitable for rating was based on three principles: 1) the statement wherever possible should use the exact language used by the respondent (modifications used only for grammar and tense); 2) statements should reflect unique concepts provided by respondents, and where it was arguable that two statements contained similar but not identical concepts, separate statements would be included for rating; and 3) only statements and ideas volunteered by respondents would be included in the statements (i.e., research team members responsible for coding and creating Likert statements could not contribute to statements).
A Priori Consensus Definition for Rounds 2 and 3
Within the 1–9 point scale, ratings were grouped into three important categories:
1.
“Least Important” (1–3);
2.
“Neither Least nor Most Important” (4–6); and
3.
“Most Important” (7–9).
A priori criteria for consensus agreement required ≥70% of respondents to rate an item within the Most Important category (rating 7 to 9) AND the median score had an interquartile range (IQR) ≤2.
Round 2 Survey and Process
All respondents to Round 1 were invited to participate in Round 2 (e-mail with embedded link to survey). The Round 2 survey included three sections: Section 1 included clarification of respondent queries (research team, involvement of patients, survey process), a general description of respondents, and processes used for developing items; Section 2 included items concerning what should and should not be included in an explanation of chronic breathlessness to a person living with this symptom; and Section 3 included items for research priorities for breathlessness over the next five years. Opportunities to suggest additional items or comments were provided for each key question. Respondents were encouraged to respond within 14 days (with follow-up e-mails at one week and the day before the closing date of the survey) with this deadline extended up to six weeks due to a respondent's commitments to several international conferences.
Round 2 Data Management
For each item within the survey, the percentage of respondents rating within the three important categories, item median, and IQR were calculated. Items meeting a priori consensus were identified and removed from the Round 3 survey. New items suggested by respondents were reviewed and, where an exact duplicate did not exist, allocated to an appropriate domain and a new Likert style statement was created. A summary of Round 2 item ratings including which items had met or did not meet a priori consensus was created to provide feedback to participants as a basis for Round 3.
Round 3 Survey, Process, and Data Management
Round 3 survey dissemination followed the same process as Round 2, with the exception that participants were provided with feedback via the Round 2 results summary and invited to review and reflect upon these results before rating in Round 3. Respondents were encouraged to respond within 14 days (with follow-up e-mails at one week and the day before the closing date of the survey) with this deadline extended up to eight weeks due to a respondent's commitments to several international conferences. For each item within the survey, the percentage of respondents rating within the three important categories, item median, and IQR were calculated. Items meeting a priori consensus from Round 2 and 3 were identified and collated.
Process for Moving From Round 3 Results to Final Recommendation Documents
All items meeting a priori consensus criteria (≥70% of respondents rating as “Most Important” [score 7 to 9 with IQR ≤ 2]) AND meeting the first consensus criterion (≥70% agreement as “Most Important”), but not the dispersion criterion (IQR ≥ 2), were collated for inclusion in recommendations. This conservative approach accounted for the potential impact of the slightly smaller response rate in Round 3 on the IQR and the planned survey where consensus-based items would be rated by health professional and people living, or caring for someone, with breathlessness.
A single member of the research team (M. T. W.) reviewed each item and drafted a recommendation statement based on the following principles: 1) wherever possible, use the original wording of the item; 2) retain all items meeting consensus unless concepts or content are duplicated; and 3) use a consistent language style for coherence and readability. Provisional recommendations statements were then reviewed by the same researcher (M. T. W.) to identify potential duplication of items and arranged in a logical order for two separate documents: Explaining chronic breathlessness, Recommendations for information which should be included and avoided; and Research Priorities for chronic breathlessness. The worksheets used to draft recommendation statement for each item, item decisions (potential duplicate and position within recommendation document), and the provisional recommendations documents were independently reviewed by research team members (K. N. J, D. B., D. J., H. L.) with the final recommendations revised based on reviewer feedback.
All Delphi respondents completing Round 1 surveys were provided with the final version of the Recommendation documents with an invitation to provide feedback (four participants responded but no additional feedback provided).
Data Management and Synthesis
Profession representativeness of respondents (ratio of invited to consented/completed surveys in each round) and response rates (number responded and number of completed surveys/number invited) were calculated. Response stability was assessed by percentage of items rated in both Rounds 2 and 3 which consistently did not meet consensus (i.e., same items in both rounds).
Appendix Table 1Final Results for Question 1 “What Is Important to Include in an Explanation of Chronic Breathlessness for a Person Living With This Symptom?”
Item history. 0 = new item for Round 3; 1 = item did not reach consensus in Round 2 and was included in Round 3 for rerating; 2 = item reached consensus in Round 2 and was not included in Round 3.
Item
n
Important Ratings % of Respondents
Dispersion Measures
Least (1–3)
Mid (4–6)
Most (7–9)
Median
IQR
Person-centered communication
2
Speak to the level of understanding of the patient that may vary somewhat between individuals
27
0
0
100
9
1
0
To include closest supporters/partner/carer in the conversation to see and understand partner's feelings
24
4
8
88
8
1.12
0
Emphasize to the patient the importance of expressing his/her fears and concerns
24
4
21
75
8
2.25
0
Use the term ‘breathlessness' rather than ‘dyspnea’ even in the questions/descriptors
24
8
17
75
8
2.5
1
Include everything, but delivered in an appropriate time frame depending on patients wish for information, ability to understand, and general condition
24
17
17
67
7.5
3
1
Use both a medical term and a plain language term
23
35
30
35
5
4
Acknowledge and validate (new domain Round 3)
0
That it is a legitimate concern to bring to clinical attention
24
4
8
88
8.5
1.25
0
Limitations, disability, and impact on daily life
24
0
17
83
8
1
0
The distress/suffering caused by this symptom and impact on emotions
24
8
13
79
8
2
0
End-of-life fears
24
13
25
63
7
2.25
0
A fear of suffocation
24
8
33
58
8
3
0
How difficult it is for carers to watch and manage
24
25
21
54
7
4.5
0
That the sensation is always unpleasant or uncomfortable
24
33
25
42
5
4
Personal, not harmful but persists despite optimal treat:
1
That it includes an emotional component
23
4
9
87
8
2
2
That being breathless from exertion is not harmful
27
11
7
81
8
2
1
That it is not harmful in itself but should be reported if breathlessness becomes more frequent or severe
24
0
29
71
8
2.25
1
That all reversible (treatable) causes of breathlessness have been sought
24
13
17
71
7.5
2.25
1
Why their breathlessness persists despite best treatment of the underlying disease (s)—(or why best treatment cannot be in particular case)
24
8
29
63
8
3
1
That in most instances does not closely reflect hypoxemia or suffocation
24
8
33
58
7.5
3.25
1
That breathlessness is a personal, lived experience of a sensation
24
13
33
54
7.5
3.5
1
That it is very different to acute breathlessness
24
21
33
46
6
2.25
1
That breathlessness is part of most people's normal life, is an expected reaction to a situation but chronic breathlessness is out of proportion to activity, provoked by lesser activities/stress and feels worse.
24
13
42
46
5.5
4
Mechanisms, aggravating and relieving factors
2
The vicious circle of decreased activity, compounded by muscle inactivity and deconditioning and further worsening breathlessness
26
4
12
85
9
1.75
1
That it can be exacerbated by unhelpful emotions/thoughts such as anxiety/worry
23
4
13
83
7.5
2
1
That it is multifactorial, not just due to the underlying condition
23
9
13
78
7.5
2.5
2
An enquiry about what activities lead to breathlessness and then tie the explanation to the individual
26
0
23
77
7.5
2
2
That related factors can precipitate or alleviate the sensation
26
8
19
73
7
1.75
1
The lung pathology and the exact vicious circles of emotions and behaviors that are relevant to that individual patient
24
25
13
63
7
4.25
1
That the brain is central to sensation
23
9
39
52
6.5
3
1
That breathlessness reflects the mismatch between the need and ability to breathe
24
17
33
50
6.5
3
1
That it lasts longer because of underlying disease
24
46
21
33
5
4.25
1
That there are different physiological origins
24
25
46
29
5.5
3.25
Symptomology (variability and trajectory)
2
It varies over time and in relation to activities and events of the person's life
25
4
16
80
8
1
1
It can fluctuate during the day depending on activity/fatigue/emotion
24
4
25
71
8
2
1
It is something that people can live with for many years
24
0
29
71
7
2.5
1
It does not always feel the same way
24
13
29
58
7
2
1
It can be continuous (does not go away, present 24 hours a day) or episodic breathlessness or both
24
4
42
54
7
3
1
The term chronic (means over a long period)
24
17
29
54
7
4
1
There will be a continual decline in functional status and what that may look like
24
46
25
29
4
5
Management principles
2
It is something that people can live with and self-manage
27
0
7
93
8
1
2
The importance of staying active
27
4
7
89
9
1
2
It is not possible to eliminate it completely but that it may be possible to eliminate some of the things contributing to it and it is possible to change reaction to it, cope, adapt, and self-manage
27
0
11
89
9
1
2
Something that can be done (even if the underlying condition cannot be improved any further)
27
0
11
89
9
1
2
The importance of promoting and expanding positive things in life
27
0
15
85
8
1.5
2
There are evidence-based techniques that can lessen sensation/impact, for example, fan
27
0
15
85
8
1
2
There are treatment options to manage dyspnea (to relieve the distress/intensity/impact) and improve quality of life
26
0
15
85
9
2
1
Paradoxically, pushing your activity to be short of breath causes improved shortness of breath as you become conditioned (for some diagnoses)
24
0
17
83
8
2
0
Loved ones should be included in education/self-management strategies
24
0
17
83
8
1
0
It is possible to live life well even with breathlessness
24
0
17
83
8
2
2
It is very important to manage panic response to be able to tolerate additional breathlessness during exercise
27
0
19
81
8
2
2
The breathlessness is unlikely to go away fully, rather it will feel easier to live with and it should be possible to do more before getting to a certain level of breathlessness.
27
4
15
81
8
2
2
We cannot take the breathlessness away but we can look to impact how we think, feel, and behave.
27
4
15
81
8
2
2
Strategies may include nonpharmacological approaches including psychotherapeutic-like approaches
27
4
15
81
8
2
2
Management requires participation from the patient, their family, and the medical team, and that communication is necessary
27
4
19
78
8
2
1
Support groups are available so know they are not alone
24
8
38
54
7
2
0
There may be barriers toward opioid treatment (new item)
24
21
38
42
5
2.5
Round 2 included 43 items; 18 items reached consensus (not included in Round 3) and 13 new items were suggested. Round 3 included 38 items; 10 items reached consensus (six of which were new items. Overall, 56 items were rated across the two rounds with 28 items reaching consensus. See table footnotes for further explanation of results presentation.
Each item was rated on a 1–9 point scale. Responses were grouped into three important categories:
Least Important (1–3)
Neither Least nor Most Important (4–6)
Most Important (7–9).
Items are ordered within each domain by percentage of respondents rating within the “Most important” category.
Green indicates consensus criteria achieved; orange indicates items where ≥70% of respondents rated the item within the same Important Category but interquartile range criterion (≤2) was not achieved.
a Item history. 0 = new item for Round 3; 1 = item did not reach consensus in Round 2 and was included in Round 3 for rerating; 2 = item reached consensus in Round 2 and was not included in Round 3.
Appendix Table 2Final Results for Question 2 “What Is Important to Avoid in an Explanation of Chronic Breathlessness for a Person Living With This Symptom?”
Item history. 0 = new item for Round 3; 1 = item did not reach consensus in Round 2 and was included in Round 3 for rerating; 2 = item reached consensus in Round 2 and was not included in Round 3.
Item
n
Important Ratings % of Respondents
Dispersion Measures
Least (1–3)
Mid (4–6)
Most (7–9)
Median
IQR
Chronic breathlessness is not
2
That breathlessness is “in your head,” only in the mind and therefore imaginary/not as “hard” as other measurements such as lung function
27
4
4
93
9
1
2
A sign of impending death
27
4
7
89
9
1.5
1
That the oxygen saturation in the blood is definitely low or can be relieved by oxygen as a first-choice treatment option
24
4
17
79
9
2
1
(Making it sound like) a separate ‘new condition’ or that chronic breathlessness is some sort of fancy syndrome that the person has to learn to understand
24
21
13
67
8
3.25
1
We do not know why people get breathless as the correlation to other clinical tests (breathing frequency, heart rate, saturation, lung function) is low
24
21
29
50
6.5
4
1
A result of anxiety
24
25
33
42
6
4.25
Blaming and hopelessness
2
Conveying that chronic breathlessness is shameful
27
4
0
96
9
0
2
Saying it is hopeless
27
7
0
93
9
0
2
Saying anything that implies that there is nothing (more) that can be done for it
27
7
4
89
9
1
0
Blaming the person or letting them blame themselves and at the same time encouraging them to actively take part in their own self-management with support from others
24
13
8
79
8
2
1
Saying “it's up to you, your responsibility”
24
8
17
75
8
2.25
Specific terms
1
“Resistant to treatment”
24
21
21
58
7.5
4.25
1
Overmedicalizing the issues
24
13
33
54
7
4
1
“Intractable”
23
17
30
52
6.5
5
1
“Panic” or “panic disorder”
24
50
21
29
3.5
5.25
1
Descriptors (e.g., cannot breathe in, cannot breathe out, feeling of suffocation) as this defeats the purpose of identifying the varying perceptions patients experience under the term breathless
24
46
38
17
4
3.25
1
Palliative/hospice care and/or death
24
54
38
8
3
3.25
Inappropriate reassurance/assurance
2
The idea of taking breathlessness away completely
27
7
7
85
8
2
2
Saying that all breathlessness should be avoided
27
11
7
81
9
1.5
1
Raising false hopes
24
8
21
71
7
2.25
1
Being overly reassuring about the harmlessness of chronic breathlessness but it is important that they do not misinterpret this and start ignoring acute exacerbation that need treatment
24
17
29
54
7
2.25
1
Focusing on deep/big breaths when recovering
24
21
38
42
5.5
3
1
Conveying information about time frame
24
38
33
29
4
5
Round 2 included 22 items; seven items reached consensus (not included in Round 3) and one new item was suggested. Round 3 Included 16 items; two items reached consensus (one of which was a new Item). Overall, 23 items were rated across the two rounds with nine items reaching consensus. See table footnotes for further explanation of results presentation.
Each item was rated on a 1–9 point scale. Responses were grouped into three important categories:
Least Important (1–3)
Neither Least nor Most Important (4–6)
Most Important (7–9).
Items are ordered within each domain by percentage of respondents rating within the “Most Important” category.
Green indicates consensus criteria achieved; orange indicates items where ≥70% of respondents rated the item within the same important category but interquartile range criterion (≤2) was not achieved.
a Item history. 0 = new item for Round 3; 1 = item did not reach consensus in Round 2 and was included in Round 3 for rerating; 2 = item reached consensus in Round 2 and was not included in Round 3.
Item history. 0 = new item for Round 3; 1 = item did not reach consensus in Round 2 and was included in Round 3 for rerating; 2 = item reached consensus in Round 2 and was not included in Round 3.
Item
n
Important Ratings % of Respondents
Dispersion Measures
Least (1–3)
Mid (4–6)
Most (7–9)
Median
IQR
Basic science
2
Underlying pathological mechanisms of breathlessness in COPD versus ILD, heart failure, etc.
27
11
11
89
7
1
2
Key pathways involved in the central processing of chronic breathlessness
27
4
15
81
8
2
2
Differences in central processing of breathlessness between different diagnoses
27
4
19
78
7
1
2
Mechanisms of nonpharmacological approaches, for example, fan therapy
26
4
19
77
7
2
2
Neural markers of breathlessness
27
11
15
74
7
1.5
2
Physiological mechanisms for dyspnea relief
27
11
15
74
7
2
1
Behavioral mechanisms for dyspnea relief
23
0
30
70
7
3
1
Role of pulmonary opioid receptors in modulating the sensation of breathlessness
23
13
30
57
7
3
1
Role of the primitive brain in mechanisms of breathlessness
23
9
39
52
6.5
2
1
Contribution of hyperventilation to the experience of chronic breathlessness
23
9
43
48
6
2
1
Relationship between hypoxemia and breathlessness (and variability among individuals)
23
9
48
43
6
2
0
Role of microbiome lung and gut and inflammation in SOBr
23
48
30
22
3.5
2.5
Pharmacological
1
Identification of novel alternatives to opioid analgesics for relief of breathlessness (e.g., cannabinoids)
23
4
4
91
8.00
1.25
2
Determine the optimal pharmacological management of breathlessness
27
7
7
85
8.00
2
2
Pharmacovigilance and long-term effect of low dose of opioids on dependence and tolerance
27
4
15
81
8.00
1.5
1
Clinical trials with opioids to optimize this treatment option (which opioid? starting dose? short acting or long acting?)
23
0
22
78
8.00
1.25
2
Predictors for benefit from opioids
27
4
22
74
7
1.5
2
Validate and standardize pharmacological approaches using known compounds
27
19
11
70
8.00
1.5
1
Pharmacovigilance of drug interventions
23
0
35
65
7.00
2
1
Role of antidepressants in management of breathlessness
23
13
26
61
7.00
3.25
1
Individual and community adverse effects of long-term opioid therapy for chronic breathlessness
23
9
48
43
6.00
3
1
Benefit of supplemental oxygen for people who experience hypoxemia only when physically active
23
17
39
43
6.00
3
Nonpharmacological
1
How can patients and carers be supported to self-manage their breathlessness?
23
0
9
91
8
2
2
Behavioral approaches to ease breathlessness (beside activity)
27
0
15
85
8
2
2
How to change behavior in patients, for example, become more active
27
0
19
81
8
2
1
Long-term follow-up of early intervention with nonpharmacological techniques
23
0
26
74
7
2
1
Brief neuromodulation strategies to help shortness of breath
23
4
26
70
7
2.25
1
Comparative effectiveness of programmatic/comprehensive approaches using nonpharmacological strategies for breathlessness
23
0
35
65
7
2.25
1
Cost-effectiveness of complex breathlessness interventions (mainly nonpharmacological interventions)
23
4
30
65
7
2
1
Development/optimizing of educational programs about breathlessness
23
0
39
61
7
2.25
1
Role of rehabilitation in severe lung disease
23
4
39
57
5
3
1
Validate and homogenize nonpharmacological therapies
23
17
26
57
7
4
1
Development of nonpharmacologic treatment (at spinal or cortical level)
23
13
52
35
5
3
1
Effect of mechanical ventilation on breathlessness
22
27
41
32
5
4
1
Nutritional approaches to decrease demands on the system to reduce breathlessness
23
30
43
26
6
3.5
1
Development of a fully portable noninvasive ventilator to enhance the exercise tolerance of people with chronic breathlessness
22
14
64
23
5
2
1
The role of wind instruments for breathlessness
23
48
30
22
3.5
4
Pharmacological and/or nonpharmacological
1
Identify pharmacological and nonpharmacological interventions that relieve chronic breathlessness with an acceptable safety profile and net clinical benefit in people with relatively long expected survival (not near death)
23
0
13
87
8.00
2
1
Identification of pharmacologic and nonpharmacologic strategies to alleviate breathlessness by optimizing the psycho-physiological benefits of physical activity and/or rehabilitative exercise training
23
0
22
78
8
2.25
2
Identify which interventions are effective in people with short expected survival/palliative care and how they be administered including optimal dosing
27
0
26
74
7.00
1.5
1
Symptom-specific methods of alleviating breathlessness without modifying the underlying disease (e.g., opioids, cannabinoids, transcutaneous electrical nerve stimulation, etc.)
23
4
30
65
7.50
2.5
1
Identify which elements should be included in short-term interventions, for example, for patients in hospital
23
0
43
57
7.00
2.25
1
Comparing the relative contributions of nonpharmacological and pharmacological approaches
23
13
35
52
6.50
3
Psychosocial
2
Impact of anxiety, stress, and mood on breathlessness and management
27
0
22
78
8
2
1
Role of anxiety in chronic breathlessness
23
0
30
70
7
2
1
Relationship between symptoms and emotional consequences
23
0
39
61
7
3
1
Individual differences in coping mechanisms
23
13
30
57
7
3
1
How to get patients to talk about breathlessness, coping
23
13
39
48
6
3
1
Mutual interactions of breathlessness with social aspects (family, caregivers, friends, etc.)
23
4
48
48
5.5
3
0
Dynamics of doctor-patient interactions for optimal treatment
23
9
43
48
5.5
2.35
1
Exploring if complex interventions impact stress levels
23
22
35
43
5.5
5.5
1
Impact of breathlessness on specific (which?) daily activities
23
13
57
30
6
4
Comparative studies (within and between conditions)
1
Main subcategories of chronic breathlessness and whether these need separate trials and treatments
23
17
30
52
6.5
4.25
1
Comparative studies across diagnoses such as COPD, ILD, heart failure, MND/ALS
23
13
39
48
6
2.25
1
Relationship of breathlessness to other symptoms and how this differs across diagnoses
23
4
57
39
6
2
1
Characteristics of breathlessness in noncardiopulmonary diseases
23
13
52
35
6
2
1
Differences in how breathlessness is described in patients with the same diagnosis
23
22
52
26
5
2.75
1
Relationship between breathlessness and nonobstructive emphysema (normal spirometry; reduced DLCO; but still very short of breath)?
23
30
48
22
4.5
3
Assessment
2
Developing simple, user-friendly tools for daily self-management
27
4
11
85
8
1.5
1
Development of a common core outcome set of measures for clinical practice and research
23
9
17
74
8.00
3
0
Determining which interventions caregivers have provided that have reduced breathlessness in patients.
22
5
27
68
7
4.25
1
Establish a standardized breathlessness assessment tool of universal/routine use in clinical care and research settings
23
13
22
65
7
4
1
Standardized tests for exertional breathlessness in clinical practice
23
13
26
61
7
3.75
1
Measures of breathlessness in daily life standardized for level of exertion/activity to appropriately capture treatment effects—1 Least Important
23
4
35
61
7
3
1
Exploring assessment methods, both inpatient and outpatient
23
4
48
48
6
4
1
Validation of the two multidimensional questionnaires of dyspnea in the main languages (French, Spanish, German, Italian, Portuguese)
23
4
57
39
6
3
1
Symptom clusters—and how we assess/manage clusters that include breathlessness
23
17
48
35
6
3
0
Greater emphasis on replication and on clinical implications of symptom clusters (i.e., to the extent that we can identify replicable clusters within or across diagnoses, what difference to they make for approaches to treatment)
23
17
57
26
5
2.25
1
Breathlessness descriptions at different stages of disease
23
26
57
17
5
3
Organization and delivery of care
2
Best models of integration of breathlessness services to help people with SOB and all disease states
27
4
15
81
8
1.5
2
Best way to tailor management of breathlessness to individual patients and their carers
27
4
19
78
8
1.5
2
Exploring and evaluating effective treatments/process of management for episodic breathlessness
27
4
22
74
8.00
1.5
0
Creation of multicenter and national collaborative(s) to facilitate increased and more efficient research in this area.
23
4
22
74
7.00
3
1
Identifying when palliative care should be initiated with chronic breathlessness
23
17
30
52
6.50
2.5
0
All patients benefit from a “palliative approach” (skills held by all health professionals) but identifying which patients would benefit from a “palliative care service” (skills provided by people with more detailed training for a smaller group of patients with more complex needs)
23
9
39
52
6.50
3
Health professional education and training
2
Facilitating a greater understanding of the effects of breathlessness among clinicians
27
0
19
81
8
2
2
Facilitating behavior change in clinicians, that is, use evidence base and measure breathlessness
25
0
20
80
8
2
2
Training programs for nurses and doctors and caretakers specifically for the needs of patient with chronic breathlessness
27
7
15
78
8
2
1
Raise awareness of physicians, whatever their background
23
4
30
65
7
3.25
Miscellaneous
1
Can we learn from individuals with chronic breathlessness more about how to manage it successfully?
23
9
26
65
7
3
1
Why are some patients bothered by breathlessness and others not?
23
4
35
61
5
3
1
Effect of paralysis in ICU and breathlessness
23
26
43
30
5
4
Round 2 included 76 items; 21 items reached consensus and six new items were suggested. Round 3 included 61 items; five items reached consensus (none of which were new items). Overall, 82 items were rated across the two rounds with 26 items reaching consensus. See table footnotes for further explanation of results presentation.
Each item was rated on a 1–9 point scale. Responses were grouped into three important categories:
Least Important (1–3).
Neither Least nor Most Important (4–6).
Most Important (7–9).
Items are ordered within each domain by percentage of respondents rating within the “Most Important” category.
Green indicates consensus criteria achieved; orange indicates items where ≥70% of respondents rated the item within the same importance category but interquartile range criterion (≤2) was not achieved.
a Item history. 0 = new item for Round 3; 1 = item did not reach consensus in Round 2 and was included in Round 3 for rerating; 2 = item reached consensus in Round 2 and was not included in Round 3.
Physiotherapists' beliefs and attitudes influence clinical practice in chronic low back pain: a systematic review of quantitative and qualitative studies.
The association between health care professional attitudes and beliefs and the attitudes and beliefs, clinical management, and outcomes of patients with low back pain: a systematic review.
Six key topics informal carers of patients with breathlessness in advanced disease want to learn about and why: MRC phase I study to inform an educational intervention.
Using the Delphi technique to determine which outcomes to measure in clinical trials: recommendations for the future based on a systematic review of existing studies.
Evaluation of nine consensus indices in Delphi foresight research and their dependency on Delphi survey characteristics: a simulation study and debate on Delphi design and interpretation.
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