Is a specialist breathlessness service more effective and cost-effective for patients with advanced cancer and their carers than standard care? Findings of a mixed-method randomised controlled trial

Farquhar et al. BMC Medicine 2014, 12:194 http://www.biomedcentral.com/1741-7015/12/194 RESEARCH ARTICLE Open Access Is a specialist breathlessness service more effective and cost-effective for patients with advanced cancer and their carers than standard care? Findings of a mixed-method randomised controlled trial Morag C Farquhar1,2*, A Toby Prevost3, Paul McCrone4, Barbara Brafman-Price5, Allison Bentley5, Irene J Higginson6, Chris Todd2 and Sara Booth7 Abstract Background: Breathlessness is common in advanced cancer. The Breathlessness Intervention Service (BIS) is a multi-disciplinary complex intervention theoretically underpinned by a palliative care approach, utilising evidence-based non-pharmacological and pharmacological interventions to support patients with advanced disease. We sought to establish whether BIS was more effective, and cost-effective, for patients with advanced cancer and their carers than standard care. Methods: A single-centre Phase III fast-track single-blind mixed-method randomised controlled trial (RCT) of BIS versus standard care was conducted. Participants were randomised to one of two groups (randomly permuted blocks). A total of 67 patients referred to BIS were randomised (intervention arm n = 35; control arm n = 32 received BIS after a two-week wait); 54 completed to the key outcome measurement. The primary outcome measure was a 0 to 10 numerical rating scale for patient distress due to breathlessness at two-weeks. Secondary outcomes were evaluated using the Chronic Respiratory Questionnaire, Hospital Anxiety and Depression Scale, Client Services Receipt Inventory, EQ-5D and topic-guided interviews. Results: BIS reduced patient distress due to breathlessness (primary outcome: −1.29; 95% CI −2.57 to −0.005; P = 0.049) significantly more than the control group; 94% of respondents reported a positive impact (51/53). BIS reduced fear and worry, and increased confidence in managing breathlessness. Patients and carers consistently identified specific and repeatable aspects of the BIS model and interventions that helped. How interventions were delivered was important. BIS legitimised breathlessness and increased knowledge whilst making patients and carers feel ‘not alone’. BIS had a 66% likelihood of better outcomes in terms of reduced distress due to breathlessness at lower health/social care costs than standard care (81% with informal care costs included). Conclusions: BIS appears to be more effective and cost-effective in advanced cancer than standard care. (Continued on next page) * Correspondence: 1 Primary Care Unit, Department of Public Health and Primary Care, University of Cambridge, Institute of Public Health, Robinson Way, Cambridge CB2 0SR, UK 2 School of Nursing, Midwifery & Social Work, University of Manchester, Jean McFarlane Building, Oxford Rd, Manchester M13 9PL, UK Full list of author information is available at the end of the article © 2014 Farquhar et al.; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated. Farquhar et al. BMC Medicine 2014, 12:194 http://www.biomedcentral.com/1741-7015/12/194 Page 2 of 13 (Continued from previous page) Trial registration: RCT registration at ClinicalTrials.gov NCT00678405 (May 2008) and Current Controlled Trials ISRCTN04119516 (December 2008). Keywords: Breathlessness, Cancer, Advanced disease, Randomised controlled trial, Complex intervention, Mixed methods Background Breathlessness (dyspnoea) is a common distressing symptom of advanced cancer, impacting physically, emotionally and socially on patients and families [1]. Occurring in 90% of lung cancer and 50% to 70% of all cancers, its prevalence increases rapidly towards the end of life [2]. The experience of breathlessness is complex [3]. Given its multifactorial causes and multidimensional impacts [2], and absence of a single effective palliative treatment, complex interventions are indicated. Early intervention models used non-pharmacological single-disciplinary approaches [4-6]. More recent models are multi-disciplinary [7], utilising evidence-based pharmacological [8-10] and non-pharmacological [11-13] component interventions [14,15]. Few have been evaluated with randomised controlled trial (RCT) methodology. The Breathlessness Intervention Service (BIS) is a multi-disciplinary complex intervention combining nonpharmacological and pharmacological interventions to support breathless patients with advanced disease, theoretically underpinned by a palliative care approach [16-18]. Developed and evaluated [1,19-22] using the Medical Research Council (MRC) framework for complex interventions [23], it has undergone a Phase III RCT with two sub-protocols: one for advanced cancer and one for advanced non-malignant disease (differing service model for each) [24]. This paper reports the findings of the subprotocol for advanced cancer in relation to the following research questions: 1. Is BIS more effective than standard care for patients with intractable breathlessness from advanced malignant disease? 2. Does it reduce patient and carer distress due to breathlessness and increase patients’ sense of mastery of the symptom? 3. What are the experiences and views of those who use BIS (patients and their informal carers)? 4. Is BIS cost-effective? Methods A detailed study protocol [24] and detailed intervention description [16,17] are published elsewhere. Box 1 outlines the two-week intervention for advanced cancer (intervention duration determined by disease trajectory). The BIS team comprises: a palliative care medical consultant (with dedicated clinical sessions and a research interest in breathlessness), a clinical specialist occupational therapist (lead clinician for the service), a clinical specialist physiotherapist and an administrator. Each professional contributes their individual strengths and skills in particular areas, but all are able to deliver the core interventions outlined in Box 1, using a psychologically-informed approach. At a weekly multidisciplinary team meeting cases are allocated to the most appropriate professional based on information derived from the referral; many patients receive visits from at least two professionals on the team. The intervention is delivered predominantly in the homesetting with visits typically lasting 1 to 1.5 hours. Visits include interventions relevant to that person (outlined in Box 1) and formulation of an individually-tailored exercise plan, for example, walking incrementally increasing distances in their local environment using a handheld fan to man (...truncated)