SingStrong – singing for better lung health in pulmonary fibrosis: A feasibility study

1 Introduction

Pulmonary fibrosis (PF) is a chronic, progressive and typically life-limiting disease characterised by scarring and stiffening of the lung parenchyma, reduced capacity for gaseous exchange and marked respiratory compromise for the patient [1]. Of the main two types of PF –familial and idiopathic pulmonary fibrosis (IPF)- the latter is more common, with a life expectancy of between three and five years post diagnosis [1]. PF currently affects up to three million people globally, with numbers on the increase [2]. There is a marked incidence of serious co-morbidity with PF including pulmonary emphysema, gastroesophageal reflux, lung cancer, pulmonary hypertension and sleep disorders, in addition to the acute exacerbation of the underlying condition [3]. The mechanism of the aberrant fibrosis in PF remains unclear, with proffered theories including repeated micro-injury to a genetically predisposed alveolar epithelium, followed by an abnormal healing response hallmarked by excessive collagen deposition [4]. There is no cure for PF, with current pharmaceutical management capable only of slowing functional decline and disease progression [4]. With the lack of curative therapies, strategies to optimise quality of life (QoL) and the biopsychosocial wellbeing of this population must be a priority.

Breathlessness, cough, reduced exercise endurance, fatigue, anxiety, depression and weight loss are among the most common problems faced by this population [5].The impact of the COVID-19 pandemic has further complicated the management of these symptoms. The risk factors for poor outcomes in COVID-19 are common in people with PF, who already have compromised pulmonary reserves. It is probable that the prognosis for PF patients with COVID-19 is more ominous than for the general population, or cohorts with other chronic conditions [2]. Furthermore, the requirement for people with PF to limit their social interactions and cocoon due to their health status, is likely to also have marked adverse effects on their mental as well as physical health [6].

Robust evidence supports pulmonary rehabilitation (PR) as a preferred conservative strategy to address the challenges faced by people with PF [7], and early access to a bespoke PR programme is advocated in the Irish Thoracic Society’s position statement on the management of PF [8]. However, the curtailment of in-person PR classes has adversely impacted this important pillar of care for this population, even as many service providers attempt to move classes online. There are currently mixed results on the feasibility and acceptability of virtual PR classes, with available studies being of small cohort sizes and of variable methodological quality [9, 10]. In addition, many of these studies involve patients with Chronic Obstructive Pulmonary Disease (COPD), with results that may or may not be generalisable to PF patients with restrictive breathing patterns. Even in pre-pandemic times, availability of, and compliance with PR was historically low in Ireland with many patients unwilling or unable to engage with services when offered [11]. Therefore alternative approaches that address the biopsychosocial issues associated with PF, while embodying tenets of exercise and education inherent in PR, are worthy of investigation.

Singing interventions in lung pathologies aim to improve the efficiency of respiration through optimal recruitment of the primary muscles of respiration, while simultaneously enhancing QoL and ameliorating psychological distress [12]. A systematic review exploring the benefits of singing for lung health found that such interventions have the potential to improve bio-psychological health-related (HR) QoL, without incurring significant side effects [13]. In patients with PF, impaired gas exchange has been identified as the primary cause of exercise intolerance, with associated extra-pulmonary consequences including reduced health-related QoL, fatigue, and/or anxiety and depression [14]. Community based programmes that use singing and breathing control to address these respiratory and broader QoL issues have been successfully delivered to cohorts with COPD [15], with recent evidence from the United Kingdom reporting benefits from online classes including a reduction in depression and greater balance confidence in a COPD cohort [16]. However, it is unclear if such an online intervention would be feasible in a PF population, who typically have more complex presentations and poorer prognoses than patients with COPD. This feasibility study therefore aimed to evaluate the clinical efficacy and acceptability of an online group singing and breathing programme (SingStrong for Lung Fibrosis) for people with PF.

2 Methods

This project was conducted in partnership with the Irish Lung Fibrosis Association (ILFA), the national patient organisation for lung fibrosis in the Republic of Ireland. Founded in 2002, ILFA support patients and families living with PF. The 12-week SingStrong programme (including one bank holiday with no class) ran over the summer of 2021 and was delivered online via Zoom^TM due to the ongoing COVID-19 pandemic. SingStrong was established in 2018 by authors RC and CM, who have successfully delivered similar classes to people with COPD, those with Long COVID and chronic lung conditions in both in-person and online settings [15].

3 Results

4 Discussion

PF is a progressive, debilitating and typically fatal disease that affects every facet of the patient’s life and wellbeing. Interventions that address the biopsychosocial wellness and QoL of the patient are advocated. The ongoing COVID-19 pandemic has proved challenging for the PF community who are vulnerable to adverse outcomes from the virus, and for whom social isolation associated with government guidance to cocoon has become the norm. Online interventions offer access to treatment options for this cohort, although the efficacy of this is not clear. This study examined the clinical efficacy and acceptability of an online singing and breathing retraining programme (SingStrong) for a PF population of convenience in Ireland. Results indicate some modest clinical gains, with high attendance rates reflecting participant enjoyment and perceived benefits in managing their lung health.

A key fundamental underpinning the efficacy of singing for better lung health is the utilisation of the cardiorespiratory system during persistent singing training, resulting in enhanced respiratory muscles and an optimized breathing mode [12]. Breathing pattern retraining has shown to improve multiple physiological outcomes for COPD patients [25], but there is a paucity of evidence in this area in PF. The impact of the SingStrong programme on respiratory and other common symptoms in this study was evaluated using self-complete questionnaires and showed variable results. The symptom and impact domains of the SGRQ showed small non-significant deteriorations, with the activity domain and overall total score showing small improvements although these were not statistically significant. In contrast to a ten-week singing intervention in COPD involving participants with comparable SGRQ baseline scores to the current study, statistically significant improvements were noted in all scores except the activity domain, which also improved slightly [26]. The comparative capacity for clinical improvement in COPD and PF may be limited however, given the underlying physiology of both conditions and the greater severity of PF. In addition, in the current study there were no clinically meaningful changes that met the criteria for PF using the SGRQ. In contrast to COPD where the MCID is just four points, a recent update on the issue noted a much higher MCID of 11 points in PF [24]. However that study notes that “real-world” PF MCID values are likely to be variable and dependent upon factors such as disease severity.

The IPF-PROM questionnaire in contrast showed significant improvements in outcomes for participants with a medium effect size (> 0.6). This is likely due to the bespoke nature of the outcome measure, which addresses issues more particular to PF, in contrast to the more generic nature of the SGRQ. The main domains of the IPF-PROM relate to physical and affective breathlessness, emotional wellbeing and fatigue [22], in comparison to the strong functional focus of the SGRQ. Given the emphasis of the SingStrong intervention on breathlessness and holistic wellbeing, the positive outcome using the IPF-PROM questionnaire is unsurprising. It is also arguably a better reflection of the participant experience of the intervention.

This study also found positive results regarding self-reported QoL, general well-being and urge to cough. Similar results were reported in a small 12-week study of ten participants with IIP [17]. The study by Russell et al. reported self-reported improvements in physical, psychological well-being, cognitive and social functioning, although spirometry measures were unchanged. Research indicates that after dyspnoea, extra-pulmonary problems such as poor HRQoL, depression and anxiety are the most distressing disease manifestations for patients with PF [14]. Singing interventions have previously been found to be effective in addressing several of these issues in cohorts with chronic respiratory cohorts, in particular COPD [15, 27–29]. In a 2010 study of singing in chronic respiratory conditions by Lord and colleagues [28], COPD patient participants reported benefits in numerous domains including physical sensation, general well-being, community/social support and achievement/efficacy. Over 80% of respondents also reported “a marked physical difference” due to the singing intervention. Notably, this was in the absence of any improvement in several objective parameters including single breath counting, breath hold time or shuttle walk distance. Similarly in a post-lung cancer cohort, improvements in several QoL domains, depression, anxiety, confidence and self-esteem were recorded in the absence of any positive change in lung function, respiratory muscle strength or fatigue [30]. It would therefore appear that singing interventions such as that in the current study may provide meaningful, life-enhancing gains even in the absence of objective clinical change. Proposed mechanisms for this include chemical changes such as upregulation of oxytocin, immunoglobulin A, and endorphins, which improves immune function and increases feelings of happiness [31]. Alternative hypotheses purport that benefits may be derived from engagement in an enjoyable activity with individuals dealing with the same challenges, thus creating a community of support and understanding [30]. This theory is supported by the level of enjoyment noted by participants in this study, who also reported good overall programme efficacy in helping them managing their lung health.

Rates of attendance and adherence to the programme were also encouraging. Class attendance trended upwards from the initial one or two weeks, with number of class attendees increasing by approximately 180% from week 1 to week 12. The reasons for this are unknown but word of mouth is likely to have contributed given that participants were recruited from a single source (ILFA). The growth in attendance also reflects the exceptionally high levels of enjoyment reported by the participants in the SingStrong programme, and their enthusiasm for additional future programmes. This is further supported by the qualitative feedback provided by the participants. On the whole, this was largely positive and reflects perceived benefits in their mastery of breathlessness and relaxation techniques. Several participants also alluded to the fun and friendly atmosphere created in the class as well as the sense of community and support derived therein. Popularity of singing for lung health problems has been consistently reported in the literature [32, 33], in contrast to PR where rates of adherence and retention are poor on the whole [34]. Although not explicitly evaluated, these findings also support satisfaction with an online environment that is comparable to in-person attendance reported at previous in-person SingStrong programmes for participants with COPD [15].

5 Limitations

This was a small population of convenience drawn from one source, and findings may not be generalizable to other settings. A sample size calculation should have been performed in advance of the study to add rigour. In particular, the strong positive influence of the vocal coach may skew results and may not be replicable elsewhere. Due to the pandemic, it was not possible to obtain objective clinical measures such as spirometry, which may have added to the study. The severity of disease was not established and may have had a bearing on the ability of the participants to participate in the intervention. Finally, although volunteers were sought for a follow-up focus group, only three participants stepped forward. The authors agreed that this would not provide a balanced and diverse pool of feedback and as such have not included those findings in this script. The addition of such qualitative data would have greatly enhanced any insights into the strengths and weaknesses of the programme in this particular cohort. A related issue is the limited capacity of the interpretation of the inferential tests applied in this study, which do not reflect the typical primary aims of feasibility studies.

6 Conclusion

Singing for better lung health offers a viable treatment adjunct for people with PF. Given the prognosis and available treatments for PF, it is debatable whether significant objective clinical improvements are achievable in these patients, or whether maintenance of current health status should be the aim. The importance of more holistic factors such as self-efficacy, HRQoL and positive mental health may therefore assume a greater priority. Programmes such as “SingStrong” which offer an enjoyable, community-based intervention, in addition to targeted breathing retraining, appear to be acceptable in an online setting, and are capable of engendering positive perceived changes to health and HRQoL.