Sage Journals: Discover world-class research

Abstract

Interest in the applications of music in health, social care and community settings are growing worldwide. Rare diseases are a major challenge for public health, particularly in paediatric populations. Previous research has demonstrated music and music medicine benefit children and young people’s health and wellbeing; however, this is under-researched in the paediatric rare disease context. This scoping review was conducted to explore music therapy and music medicine as tools to support children with rare diseases’ health and wellbeing within healthcare settings. The review investigated the current research on how music is used for children and young people with rare diseases and complex conditions and synthesised the resulting evidence. Results from this review suggest that music therapy and music medicine are beneficial for the health and wellbeing of children with rare diseases and complex conditions, including reduced anxiety, stress and pain during clinical procedures, and increased communication, social and coping skills and enhanced quality of life.

Keywords

hospital music medicine music therapy music-based interventions paediatric rare disease

Introduction

A rare disease is characterised as a life-threatening or chronically debilitating disease with a prevalence of less than five people in 10,000 (Kole et al., 2021). Incidences of rare disease vary globally, but overall, an estimated 300 million people have a rare disease (Kole et al., 2021). Rare diseases are common collectively, but their low prevalence typically coincides with a lack of medical expertise, knowledge and research (EURODIS, 2022). Most rare diseases therefore represent a substantial unmet medical need and a major challenge for European public health (European Commission, 2017).

Approximately, 75% of rare diseases begin in childhood and are lifelong (Bavisetty et al., 2013). The complex and chronic nature of these diseases often causes significant impacts on children’s development, health and wellbeing, both physically and psychosocially (Kole et al., 2021). Previous research demonstrates that arts based activities like music are useful in disease and health management and treatment, through supporting child development and promoting the health and wellbeing of those with chronic conditions (Fancourt and Finn, 2019). Therefore, there is potential benefit in the use of music interventions for paediatric rare disease populations.

Music interventions are significant for children with rare, complex medical conditions in hospitals, offering a unique and therapeutic avenue for holistic care (Burrai et al., 2020). Music can provide comfort, emotional expression and joy for these young patients whose medical journeys are often marked by extended hospital stays and complex treatments (Pérez-Núñez et al., 2021; Sampaio, 2023). Moreover, for children with rare diseases, whose conditions may be isolating and challenging to understand, music interventions can serve as an accessible non-verbal language, fostering connection and communication. Without specialised treatments tailored to each rare condition, music becomes a versatile tool that can be adapted to these children’s individual needs and preferences, contributing to their emotional wellbeing and enriching their overall quality of life during their hospitalisation (Sampaio, 2023).

Interest in Music Therapy (MT) and Music Medicine (MM) has been expanding in paediatric and adult healthcare settings (Schmid and Ostermann, 2010). Yet, the literature suggests there is broad confusion and misunderstanding of which music-based interventions constitute MT and which, MM. MT is defined as ‘an established health profession in which music is used within a therapeutic relationship to address physical, emotional, cognitive, and social needs of individuals’ (American Music Therapy Association, 2024). Referral to a MT service is based on clinical need and is unrelated to patient’s previous enjoyment in music or musical skill. Therapy begins with a process of assessment and having identified areas of need. Following this, individualised treatment programmes are then created using music in a prescriptive way to meet identified patient goals. MT, like psychotherapy, offers the client a confidential space to explore their inner conflicts in a contained, safe way. The entire process is built on a foundation of trust and mutual respect between client and therapist, and it is from this relationship that patient’s positive outcomes derive (McIvor, 2023). A number of different approaches may be utilised by the therapist, dependent on their training, to address the client’s needs. These include psychodynamic, humanistic, behavioural, neurological, and community-based approaches (Darrow, 2008; Wheeler, 2015). An eclectic approach draws on theories and interventions from various schools of psychology to best support the client in meeting their identified goals. Intervention type, whether music listening, songwriting, verbal reflection and so on, is selected based on targeted goal areas and the therapist continually reviews and updates the approach based on clinical observations, client progress and engagement.

MM has been described as the use of music within a healthcare setting, delivered by a professional other than an MT. MM typically involves the use of music listening to support anxiety reduction in the medical setting. The literature does not outline a process of assessment within MM, nor is there a systematic therapeutic process in place (Bradt et al., 2021). Although MM may have therapeutic benefits, MM is not MT. This distinction is important for many reasons and is often misrepresented in the public domain.

The use of MT and MM in hospital settings has clinical benefits. Improved overall hospital experience and pain management was associated with MT in Roseen et al. (2017) and with MM in Ames et al. (2017). Improved sleep quality of patients was associated with MM in Momennasab et al. (2018). In palliative care settings, MT and MM have been reported to create more supportive care environments. In Pommeret et al. (2019), palliative cancer patients experienced improved satisfaction, comfort and social ties through a MM intervention. In Preissler et al. (2016), MT interventions assisted in identifying the psychosocial needs of palliative cancer patients.

In addition, there is an increasing evidence base for the use of music interventions with children in hospitals (Bradt, 2013). Prior research has demonstrated that the use of MT can improve coping skills (Moola et al., 2020; Stegemann et al., 2019), mood, communication, social skills, and quality of life (Johnson et al., 2021) and can reduce pain and anxiety during clinical procedures (Klassen et al., 2008; Stegemann et al., 2019). MM has also demonstrated benefits in pain and anxiety reduction during hospitalisation (Johnson et al., 2021; Stegemann et al., 2019). While there is a growing body of evidence regarding the beneficial effects of music for children and young people, more rigorous research is still needed on using MT and MM with children living with rare diseases.

Methods

Research questions

This scoping review of the literature aimed to map out the existing studies about the use of MT and MM with children who have rare diseases or comparable complex conditions in the healthcare setting. The study also aimed to explore and summarise the diversity of these studies, answering the following questions:

What types of MT and MM interventions were used?

In what healthcare settings have these studies been conducted?

Who were the MT and MM target population?

What types of research designs and methods were used?

Which outcomes have been described in the literature?

What were the roles and significant impacts of MT and MM on participants’ health and wellbeing?

Protocol and registration

The study was based on the Joanna Briggs Institute’s methodology for scoping reviews (Peters et al., 2020) and followed the original framework of Arksey and O’Malley’s (2005) five-stage scoping review framework, including consultations with relevant stakeholders, as proposed by Levac et al. (2010). The study protocol is available at Karpavičiūtė et al. (2022). This study was conducted as part of the SAMPI research project which aims to enable children and young people living with rare diseases to express their experience of living with a rare disease and help to identify the factors that enhance, inhibit and impact their lives (Karpavičiūtė et al., 2022).

Eligibility criteria

While the focus of the SAMPI project is on children with rare diseases, due to the limited number of articles on MT and MM with this cohort, children with complex conditions were also included. This is because there are similar disease burdens between these populations: rare conditions are often chronic and are of high complexity (Somanadhan et al., 2020). Studies that included adults were excluded. As the United Nations Department of Economic and Social Affairs (2013) recognises those up to the age of 24 years old as young people, studies with similar age ranges were included if the population was defined as young people, not adults, and children were also included in the studied population. Studies that looked at children with specific needs related to the autism spectrum or related disorders were excluded due to the prevalence of MT and MM among this population. Articles that included diagnostic tests commonly undergone by children and young people with rare diseases and complex conditions were included due to their relevance to this population. The period was limited from January 2010 to May 2022 to reveal present-day literature. The inclusion and exclusion criteria are detailed in Table 1.

Table 1.

Inclusion and exclusion criteria.

Inclusion criteria	Exclusion criteria
Children and young people (as defined by the studies) with rare, complex or chronic conditions.	Non-peer reviewed documents, restricted access publications, and grey literature (including book chapters, book reviews, policy reports, bibliographies, technical reports, conference proceedings and reports, accessible dissertations and theses, research protocols and reports, commentaries, editorials and letters).
Discussion or application of MT or MM.	Articles not in the English language.
Healthcare settings (any country).	Studies that focus on Autism.
Articles published between 1st January 2010 and October 2020 (updated search performed between September 2020 and May 2022).

Search

The databases searched included CINAHL Complete, Medline Complete, the Psychological and Behavioural Sciences Collection, and PubMed/PMC. The initial search was performed in October 2020, with an updated search performed in May 2022 to enhance the timeliness of this review. However, the Psychological and Behavioural Sciences Collection could not be accessed during the updated search; this limitation was accepted due to the limited number of returned articles from the updated search. The search strategy used in both the original and updated searches is included in Appendix 1. Non-peer reviewed literature and grey literature were not included.

Selection of sources of evidence

In the original search, four researchers (AS, SK, AO and SS) screened the studies by title and abstract independently, following recommendations provided by Levac et al. (2010). This involved a multidisciplinary team approach to selection of included articles, with discussions between researchers during the abstract review process regarding arising challenges or uncertainties. This collaborative approach added transparency and rigour to the study selection process, enhancing the reliability and validity of the overall review. In the original search, the fifth researcher (TK) resolved any disagreements through discussion and review. In the updated search, two researchers (NB and SSi) screened by title and abstract independently; these authors also reviewed the included articles from the original search. This continuity in the team ensured consistency and familiarity with the inclusion criteria, promoting a cohesive and reliable screening process. Where any disagreements arose, a third researcher (SS) resolved conflicts through discussion and review.

Data charting and results synthesis

Two researchers performed data extraction according to Braun and Clarke’s (2006) method of thematic analysis. This approach was used as it is a flexible and accessible approach that enables a rich analysis of the data, supporting the identification of similarities, differences and unexpected insights (Braun and Clarke, 2006). Relevant information was identified and coded in the studies according to emergent themes. Due to the inductive nature of this process, codes that related to each other were reviewed and grouped together as a theme. These themes were then presented to the research team for discussion and validation.

The quality of the studies included in this scoping review was assessed using the Mixed Methods Appraisal Tool (MMAT). The MMAT is a critical appraisal tool designed for the methodological appraisal of systematic and scoping mixed studies reviews (Hong et al., 2018).

Results

Study selection

From a total of 448 identified articles, a total of 19 studies were included in this review. A summary of the search process using a PRISMA diagram (Page et al., 2021) is included in Figure 1 below.

Figure 1.

PRISMA flow diagram: search process summary.

Study characteristics

Most of the studies included in this review used quantitative study designs (n = 15). Studies that used qualitative methods (n = 2) were based on interviews conducted with participants or parents of participants. Two studies employed mixed methods research designs. The study designs of each article are specified in Table 2. Most studies were published from 2014 on and were based in the United States, Canada and Europe. Most studies (n = 17) were conducted in a hospital setting; one study was based in a MT Research Centre and one study focused on home care. The populations studied included children and young people with various cancers, cystic fibrosis (CF), neurological conditions, Sanfilippo syndrome, and chronic diseases including rheumatoid arthritis. Overall, participants ranged in age from 0 to 24 years, though most studies focused on children below 18 years.

Table 2.

Characteristics of included MT studies.

Author (year)	Facilitated by	Characteristics of intervention	Setting	Sample	Research design	Outcome measured
Bringas et al. (2015)	Professional Music Therapist	Music listening, music for physical engagement (games/exercises to music).	Clinic	34 children, with Static Lesions of the Central Nervous System or other neurological disorders.	Two-armed parallel group design.	Effectiveness of music therapy as part of neurorestoration therapy; brain plasticity changes; attention and communication in neurologically impaired children.
Irons et al. (2012)	Professional Music Therapist	Singing, including diaphragmatic breathing and vocal warm-ups.	Hospital	30 hospitalised children with Cystic Fibrosis (from 7 to 17 years, mean age 11.6 years).	Dual centre double blinded parallel RCT.	Respiratory muscle strength, spirometry and quality of life.
Liu et al. (2019)	Professional Music Therapist.	Mindfulness-based stress reduction (MBSR) combined with music therapy; music listening.	Hospital	101 patients diagnosed with osteosarcoma (aged 10–21 years).	RCT	Pain, anxiety, and sleep quality scores.
Pérez-Núñez et al. (2021)	Professional Music Therapist.	Music listening, active music participation (e.g. vocal/instrumental).	Music Therapy Research Centre	Three children diagnosed with MPS III receiving music therapy treatment.	Intra-subject repeated measures design.	Illness-related distress, social integration, spiritual perspective, family environment, coping, hope-derived meaning and resilience.
Robb et al. (2014)	Board-certified Music Therapist (MT-BC).	Lyric writing; music listening; storyboarding to music; active music participation (e.g. vocal/instrumental); creation of therapeutic music video.	Hospital	113 adolescents and young adults aged 11–24 years undergoing myeloablative Hematopoietic Stem Cell Transplant.	Multisite RCT.	Courageous coping, social integration, and family environment.
Robb et al. (2017)	Board-certified Music Therapist (MT-BC) delivered the first session, parents thereafter.	Active music engagement (responding to music).	Hospital	16 children (ages 3–8 years) with cancer and their parents.	Pilot randomised trial.	Changes in parent emotional distress and patient emotional distress.
Scheufler et al. (2021)	Board-certified, Music Therapist (MT-BC).	Active music engagement, live patient-selected music and music-assisted relaxation.	Hospital	48 youth (ages 10–18) participating in a 40/week hospital-based intensive interdisciplinary pain treatment programme.	Quant design–questionnairesQuasi-Experimental	Anxiety and relaxation.
Uggla et al. (2018)	Professional Music Therapist.	Music listening; active music participation (e.g. vocal/instrumental).	Hospital	29 patients aged 0–17 years who underwent Hematopoietic Stem Cell Transplant.	Randomised clinical pilot study.	Health-related quality of life
Uggla et al. (2019)	Professional Music Therapist.	Music listening; active music participation (e.g. vocal/instrumental).	Hospital	Six families of children (aged 1–18 years) who underwent Hematopoietic Stem Cell Transplant.	Qualitative design with both an inductive and deductive approach.	Experiences of a music therapy intervention.
Yates et al. (2018)	Board-certified, master’s level practitioner (MT).	Active music engagement through instrument play, music for physical engagement, music for relaxation, instrument lessons, and musical books.	Hospital	15 caregivers of 14 paediatric blood and marrow transplant recipients.	Qualitative design; interpretivist investigation	Experiences of a music therapy intervention.

More than half of the studies (n = 10) reference a Music Therapist implementing the interventions provided in the studies. Five studies referred to their interventions as MT but were not designed or delivered by a Music Therapist. The remaining articles (n = 4) were classed as music-based interventions and did not feature any input from Music Therapists. The articles that did not feature a Music Therapist were therefore classified in this review as MM. Table 2 describes the included articles that featured MT according to the review questions, whereas Table 3 describes the included articles that featured MM. A more detailed summary table of the included studies is available as Supplementary Table A.

Table 3.

Characteristics of included MM articles.

Author (year)	Facilitated by	Characteristics of intervention	Setting	Sample	Research design	Outcome measured
Hasanah et al. (2020)	No Music Therapist involved.	Based on guidelines and patient preferences no other description provided.	Hospital	34 children with leukaemia aged 6–18 years undergoing IV-line insertion.	Pre-experimental study with one group pre-test and post-test design.	Changes in cortisol levels to indicate stress.
Karbandi et al. (2020)	No Music Therapist involved.	Music listening.	Hospital	83 children with chronic diseases aged 8–12 years.	RCT.	Anxiety (measured by Spence Children’s Anxiety Scale (SCAS)).
Kesselman et al. (2016)	No Music Therapist involved.	Music listening.	Hospital	44 children between the ages of 1–10 years old.	Between measures design with control group.	Anxiety (measured by heart rate).
Lin et al. (2020)	No Music Therapist involved.	Music listening.	Hospital and Home.	50 Ventricular Septal Defect patients (mean ages for music group and control group were 7.3 ± 1.0 and 6.9 ± 1.2, respectively).	Between-subjects experimental design.	Chronic pain, quality of life and quality of sleep.
Lin et al. (2012)	No Music Therapist involved.	Music listening.	Hospital	39 Taiwanese children diagnosed with epilepsy (aged 2–17 years).	Repeated measures design.	Changes in epileptiform discharges.
Lin et al. (2014)	No Music Therapist involved.	Music listening.	Hospital	48 children who experienced their first unprovoked seizure with epileptiform discharges.	RCT	Seizure recurrence; changes in epileptiform discharges.
Montero et al. (2020)	No Music Therapist involved.	Music listening (during airway clearance therapy sessions).	Hospital Spain	54 children with Cystic Fibrosis aged of 2–17 years.	RCT	Enjoyment and perception of time of airway clearance therapy.
Nguyen et al. (2010)	No Music Therapist involved.	Music listening.	Hospital	40 children (aged 7–12 years) with leukaemia.	RCT followed by interviews with open ended questions.	Pain scores, vital signs.
Sabzevari et al. (2017)	No Music Therapist involved.	Music listening during medical procedure.	Hospital	100 children from 7 to 14 years undergoing an endoscopy.	Double-blind clinical trial.	Pain levels and anxiety, vital signs.

According to the MMAT, overall, the quality of included studies was medium, with less than half of the studies being deemed of the highest quality. The principal areas of concern arising from the formal quality appraisal are the underreporting of complete outcome data, outcome assessors not being blinded to the intervention, and participant non-adherence to the assigned intervention. An overview of the results of the MMAT screening can be found in Supplementary Table B.

Thematic analysis

This review adheres to the thematic analysis method proposed by Braun and Clarke (2006), categorising findings into thematic groups. We undertook a preliminary reading of the included studies and developed initial patterns and themes based on the primary outcomes reported. Findings were then structured into thematic categories (Braun & Clarke, 2006). Incorporating thematic analysis into a scoping review enhances the qualitative depth of the review process, offering a nuanced understanding of the literature and contributing to generating insights that may inform both research and practice.

Following the analysis of the data, MT and MM were determined to have effects over two overarching domains:

Physiological and clinical support

Psychological wellbeing

While these domains are interconnected, they are reported for MT and MM individually to illustrate the similarities and differences in effect for these interventions.

MT

MT positively affects physiological and clinical support

MT was shown to have a positive impact on clinical symptoms including heart rate (Uggla et al., 2018), respiratory status (Irons et al., 2012), brain plasticity (through electroencephalogram) and attention (Bringas et al., 2015). It was also demonstrated to improve sleep quality (Liu et al., 2019; Robb et al., 2017) and attention levels (Pérez-Núñez et al., 2021). Reduced levels of pain perception were measured directly through accredited pain scales (Liu et al., 2019) and behavioural coding tools (Robb et al., 2017), or indirectly through qualitative interviews with patients or caregivers (Pérez-Núñez et al., 2021; Uggla et al., 2019; Yates et al., 2018). Three common mechanisms for MT to reduce pain perception emerged from the study: MT as a distraction from pain (Uggla et al., 2019), MT for mood improvement and reduced pain perception (Robb et al., 2017; Yates et al., 2018), and MT for relaxation/reduced anxiety for reduced pain perception (Liu et al., 2019; Pérez-Núñez et al., 2021). One study focused on pain alleviation through establishing long-term coping measures using MT (Scheufler et al., 2021). One study did not find a statistically significant difference in pain perception between the MT intervention and control groups (Uggla et al., 2018).

MT improves psychological wellbeing

The benefits of MT to reduce anxiety were reported in five studies using accredited anxiety scales ( Liu et al., 2019; Scheufler et al., 2021; Uggla et al., 2018; Yates et al., 2018) or associated clinical signs such as blood pressure (Pérez-Núñez et al., 2021). Stress reduction was evaluated in one study (Robb et al., 2017) which found lower emotional distress following MT. However, this study focused on music support delivered by parents following an initial MT session and found that the challenge of this activity could increase parental emotional distress. MT was found to have a positive impact on mood as reported by parents and caregivers (Robb et al., 2017; Uggla et al., 2018; Yates et al., 2018).

Emotional state was measured in four studies, demonstrating higher scores in four psychological variables which included physical, social, emotional and cognitive levels (Pérez-Núñez et al., 2021), enhanced emotional state (Irons et al., 2012) and activation of positive emotions (Uggla et al., 2019) after MT sessions. Combining mindfulness-based stress reduction with MT was reported to significantly alleviate psychological complications in participants in Liu et al. (2019).

MT was reported to improve health-related quality of life of participants in Uggla et al. (2018). A similar finding was reported in Irons et al. (2012) but this was not statistically significant. Three studies did not measure quality of life but discussed how the MT interventions in the studies could positively impact quality of life (Liu et al., 2019; Pérez-Núñez et al., 2021; Yates et al., 2018). The benefits of MT for building social connections and increasing the relationships between the patient and parents and healthcare professional, and creating a safe space, was reported in six studies (Bringas et al., 2015; Pérez-Núñez et al., 2021; Yates et al., 2018; Robb et al., 2014; Robb et al., 2017; Uggla et al., 2019). The importance of the therapeutic relationship during MT was also highlighted in Uggla et al. (2019).

MT was also beneficial for self-expression and creativity through different mechanisms including lyric writing (Robb et al., 2014), creative decision-making (Robb et al., 2017), learning and playing songs for self-expression (Uggla et al., 2019), and increased communication and creativity due to the stimulation of the MT session (Pérez-Núñez et al., 2021).

The positive effects of MT for children and young people’s ability to cope with their illness was discussed in five studies (Irons et al., 2012; Robb et al., 2014, 2017; Uggla et al., 2019; Yates et al., 2018) through providing a break in monotony and sense of normality, and encouraging engagement.

MM

MM positively affects physiological and clinical support

Similar to MT, MM was shown to positively affect clinical symptoms through music listening, including systolic and diastolic blood pressure (Sabzevari et al., 2017), heart rate (Kesselman et al., 2016; Nguyen et al., 2010) and respiratory rate, and oxygen saturation (Nguyen et al., 2010). Also noted were the positive impact of music listening on epileptiform discharges in children and young people with epilepsy (Lin et al., 2012, 2014) and improved sleep quality (Lin et al., 2020). MM was also demonstrated to reduce pain perception generally (Lin et al., 2020), and before and during diagnostic procedures such as endoscopy (Sabzevari et al., 2017) or lumbar puncture (Nguyen et al., 2010). The mechanisms of MM to reduce pain perception included distraction (Nguyen et al., 2010) and reduced anxiety (Sabzevari et al., 2017).

MM supports psychological wellbeing

Five studies considered the impact of MM on anxiety (Karbandi et al., 2020; Kesselman et al., 2016; Nguyen et al., 2010; Sabzevari et al., 2017). Reduced anxiety was reported through accredited anxiety scales (Karbandi et al., 2016; Nguyen et al., 2010), through associated clinical symptoms such as blood pressure and heart rate (Kesselman et al., 2016; Sabzevari et al., 2017) or visible relaxation (Pérez-Núñez et al., 2021). Reduced anxiety levels due to MM use were reported during diagnostic interventions (Kesselman et al., 2016; Nguyen et al., 2010; Sabzevari et al., 2017) and during treatment procedures (Hasanah et al., 2020). Stress reduction was measured through lower cortisol levels in Hasanah et al. (2020), though the results were not statistically significant. Montero-Ruiz et al. (2020) measured participants’ increased enjoyment and decreased perception of time taken of their treatment routine when listening to specifically recorded music. The impact of MM on quality of life was only measured in one study (Lin et al., 2020), which concluded that MM improves medium-to-long term quality of life postoperatively.

MM was noted in one study to assist self-management through enhancing enjoyment and reducing the perceived time taken of a medical treatment, therefore, improving compliance and establishing it as a positive experience (Montero-Ruiz et al., 2020).

Discussion

Results from this review indicate that engaging in MT and MM benefits children and young people with rare diseases during hospitalisation. The most reported outcome in the publications included in this review was the positive impact of MT and MM on anxiety and stress reduction, and improvement in mood and psychological wellbeing. This is supported by the literature, as Stegemann et al. (2019) have demonstrated the positive effects of MT and MM on levels of stress parameters as observed in heart and respiratory rates in children and young people in healthcare contexts, as well as improving mood and regulation. Klassen et al. (2008) also report that music is effective in reducing anxiety and pain during clinical procedures in children and young people, which is similarly described in this review.

The reported benefits of MT and MM for pain perception are also in line with the literature; for example, Moola et al. (2020) found in their review that MT had a positive impact on pain perception in children and youth with disabilities and chronic conditions in outside hospital settings. Benefits to clinical symptoms, including reduced systolic blood pressure and heart rate are similarly noted in Kobus et al. (2022). Findings in this review around self-management and coping with an illness are echoed in previous research which has reported MT as effective in improving communication and social skills in childhood and adolescence (Moola et al., 2020; Stegemann et al., 2019).

This scoping review has demonstrated that the benefits of MT and MM are not limited to the paediatric population living with rare diseases as parents and caregivers can both benefit from these types of interventions (Pérez-Núñez et al., 2021; Yates et al., 2018). This is supported in the literature (Edwards, 2016) and demonstrates the importance of MT as child and family-centred approaches to care. However, placing parents in facilitation roles for these interventions has the potential to negatively affect their emotional states. Thus, removing caregivers from the role of facilitator and returning to the Music Therapist-delivered intervention provides more benefits for caregivers, for example, the opportunity to relax and enjoy what was reported as a normalising, shared experience (Robb et al., 2017).

While in this review both MT and MM were demonstrated to have similar benefits, there was a lack of clear differentiation between the two. The clearest difference between MT and MM was illustrated in the types of interventions used – while MT interventions involved several different methods of engaging with the music (e.g. active music engagement, participation in music-making, lyric writing, music listening, etc.), MM studies primarily encompassed music listening only without the involvement of a Music Therapist. The types of interventions highlight the key characteristics that separate the two approaches. MT studies involved the development of a therapeutic relationship between the participant and the Music Therapist to attain patient goals, while MM was used in a more generic singular way through utilising music listening experiences. This may be significant when identifying the differences between the uses of MT and MM in clinical settings and may contribute to avoiding the misrepresentation of MM as MT in the literature in the future.

In addition to the reported benefits of MT, the findings from this review also identify MT and MM as unique tools which can support various clinical procedures and patient goals in hospital settings. Some of the studies included in this review consisted of using music to support the client’s engagement and acceptance of medical interventions (Montero-Ruiz et al., 2020), Mindfulness Based Stress Reduction (MBSR; Liu et al., 2019) and distraction cards (Karbandi et al., 2020). As each child with a rare disease presents in a unique way, further research is needed to determine the effect of using music and other creative therapy modalities in the treatment of children with rare diseases.

It is clear from this review that there is limited knowledge of using music interventions to support children and young people with rare diseases based in Europe, especially the United Kingdom and Ireland. No articles identified for review were based in the United Kingdom or Ireland. This suggests a gap in the policy and healthcare pathways for rare diseases in these countries. As this population faces significant challenges, including physical and intellectual disabilities, frequent hospitalisations, stigmatisation and challenges in developing coping strategies and resilience, among others (Somanadhan et al., 2023), the use of music-based interventions could contribute to improving both physical and psychological impacts of living with rare diseases. Music Therapists are trained to work therapeutically and sensitively with individuals with complex medical needs and as such, are well placed to devise and support healthcare pathways by offering an alternative, capacity driven and family-centred intervention for children with rare disease in healthcare settings.

It is important to note that five of the studies that featured MM referred to the intervention as MT (Hasanah et al., 2020; Karbandi et al., 2020; Lin et al., 2020; Montero-Ruiz et al., 2020; Nguyen et al., 2010). However, as clarity was not provided as to whether a Music Therapist was involved, these studies were categorised in this review as MM. The presence of a Music Therapist in delivering a music-based intervention defines MT, due to the relational nature of the intervention. This is not to say that there is a hierarchy of music-based interventions; this review demonstrates the opposite by highlighting the clear benefits of both MT and MM. While MT can facilitate therapeutic change, MM can also be utilised for affirmation and health promotion, with both being valuable tools for health. Both can provide physiological, physical, emotional or relational experiences depending on the context and needs of the patient (Bonde, 2011). However, to ensure the safety of both vulnerable patients availing of MT and working Music Therapists, it is crucial that MT is recognised as an allied health profession. This ensures MT services are delivered by a qualified, regulated Music Therapist, promoting the highest professional standards among Music Therapists (Keating, 2020). To this end, a recommendation of this review is for the global protected use of the professional title, Music Therapist and term, Music Therapy.

Study limitations

There are several potential limitations to this review. These include limitations of inclusion criteria, such as English-language requirements, date of publication and lack of grey literature, which may have led to the exclusion of relevant literature. Furthermore, the diverse interventions and methodologies of the included studies made the comparison of study results more difficult. However, these limitations are not considered significant enough as to misrepresent the research in this field and are therefore accepted by the authors.

Conclusion

This scoping review investigated the current research on how music is utilised for children and young people with rare diseases and complex conditions in paediatric healthcare settings and synthesised the resulting evidence of the impact of MT and MM. Evidence on the use of MT and MM in paediatric populations is still emerging, but results from this study suggest that using MT and MM is beneficial for the health and wellbeing of children with rare diseases and complex conditions. These benefits include reduced anxiety, stress and pain during clinical procedures, as well as an increase in communication, social and coping skills and quality of life. Parents and caregivers of children and young people with rare diseases and complex conditions can also benefit from the use of MT and MM in paediatric healthcare settings.

Based on the findings, the authors propose that the involvement of professional Music Therapists in MM research would be of particular benefit given their area of expertise and support more accurate reporting on the use of MT and non-MT interventions, which would enhance emerging research in this area.

Supplemental Material

sj-docx-1-bjm-10.1177_13594575241258820 – Supplemental material for The impact of music on health and wellbeing of children and young people with rare diseases in healthcare settings: A scoping review

Supplemental material, sj-docx-1-bjm-10.1177_13594575241258820 for The impact of music on health and wellbeing of children and young people with rare diseases in healthcare settings: A scoping review by Shannon Sinnott, Niamh Buckle, Alison Sweeney, Simona Karpavičiūtė, Amy O’Neill, Thilo Kroll and Suja Somanadhan in British Journal of Music Therapy

Footnotes

Author contributions

SS, TK, AS, AO contributed to conceptualisation (ideas; formulation or evolution of overarching research goals and aims). NB, SSi, SK, SS contributed to data curation (management activities to annotate (produce metadata), scrub data and maintain research data (including software code, where it is necessary for interpreting the data itself) for initial use and later re-use). NB, SSi contributed to formal analysis (application of statistical, mathematical, computational, or other formal techniques to analyse or synthesise study data). SS, AS contributed to Funding acquisition (acquisition of the financial support for the project leading to this publication). NB, SSi, SS, AS, AO contributed to Investigation (conducting a research and investigation process, specifically performing the experiments, or data/evidence collection). TK, SS contributed to Methodology (development or design of methodology; creation of models). SS, AS contributed to Project administration (management and coordination responsibility for the research activity planning and execution). SS, AS contributed to Resources (provision of study materials, reagents, materials, patients, laboratory samples, animals, instrumentation, computing resources, or other analysis tools). N/A contributed to Software (programming, software development; designing computer programmes; implementation of the computer code and supporting algorithms; testing of existing code components). SS, TK contributed to Supervision (oversight and leadership responsibility for the research activity planning and execution, including mentorship external to the core team). SS, TK contributed to Validation (verification, whether as a part of the activity or separate, of the overall replication/reproducibility of results/experiments and other research outputs). NB, SSi, AS, SS contributed to Visualisation (preparation, creation and/or presentation of the published work, specifically visualisation/data presentation). NB, SSi contributed to Writing – original draft (preparation, creation and/or presentation of the published work, specifically writing the initial draft (including substantive translation). NB, SSi, AS, TK, SS contributed to Writing – review & editing (preparation, creation and/or presentation of the published work by those from the original research group, specifically critical review, commentary or revision – including pre- or post-publication stages).

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: We would like to express our sincere gratitude to our funder, Children’s Health Foundation at Children’s Health Ireland, for their support.

ORCID iD

Niamh Buckle

Supplemental material

Supplemental material for this article is available and can be accessed by contacting the authors.

Author biographies

Shannon Sinnott has a Masters in Psychology and has worked with different clinical populations, including adults with intellectual disabilities and children. She is currently working as an assistant psychologist at the Irish Prison Service and is due to commence her doctoral studies in Clinical Psychology at the end of the year. At the time of this review, Shannon Sinnott worked with the SAMPI team as a research assistant at Children’s Health Ireland.

Niamh Buckle is a PhD Student studying at University College Dublin, Ireland. She has a Bachelor of Science in Children’s and General Nursing from University College Dublin, Ireland, and a Master of Science in Global Health and Development from University College London, United Kingdom. Her research interests include global health, socioeconomic determinants of health, health inequalities, rare diseases, and child and adolescent health.

Alison Sweeney is a senior Music Therapist at Children’s Health Ireland, Temple Street where she works as part of the neurosurgery, neurorehabilitation and palliative care teams. Alison holds additional training in specialised Music Therapy assessment programmes (MATADOC and OOMPA) as well as a postgraduate diploma in integrative adolescent psychotherapy.

Dr Simona Karpavičiūtė is an arts for health and cross-sectorial/ cross-disciplinary researcher, project manager and creative thinker.

Dr Amy O’Neill holds a Masters in Art Therapy from Munster Technological University and a PhD in Psychology from Trinity College Dublin. Her research interests relate to art therapy as a support for children living with chronic illness. She currently holds the role of School Completion Programme Coordinator addressing educational disadvantage and using art therapy to support the emotional well-being of children at risk of early school leaving and their families.

Dr Thilo Kroll is a Professor of Health Systems Management at University College Dublin and has experience conducting research into the social dimensions of health and well-being with a particular emphasis on public health perspectives on disability and health topics. His research interests and passion are focused on systems-related topics in global public health and inclusion, from the individual to health and population level service systems. Specialties: Disability and Public Health, Health Systems, Psychology, Ageing and Rehabilitation Research, Social Dimensions of Health, Wellbeing and Participation, Health Inequalities, Environment and Access, Violence and Health, Inclusive research design, methodologies and data collection approaches.

Dr Suja Somanadhan is an Associate Professor and Researcher at UCD School of Nursing, Midwifery & Health Systems at University College Dublin, Ireland. With a profound commitment to advancing the health and well-being of children and their families living with rare and complex medical conditions, Dr Somanadhan’s research focuses on Patient Public Involvement (PPI) and service user engagement in healthcare to inform policy and practice. She brings extensive working experience in these areas, aiming to bridge the gap between research, healthcare delivery, and the needs of patients and families.

References

American Music Therapy Association (2024) About music therapy & AMTA. Available at: https://www.musictherapy.org/about/ (accessed 5 January 2024).

Ames

Shuford

Yang

, et al. (2017) Music listening among postoperative patients in the intensive care unit: A randomized controlled trial with mixed-methods analysis. Integrative Medicine Insights 12: 1178633717716455.

Arksey

O’Malley

(2005) Scoping studies: Towards a methodological framework. International Journal of Social Research Methodology 8: 19–32.

Bavisetty

Grody

Yazdani

(2013) Emergence of pediatric rare diseases: Review of present policies and opportunities for improvement. Rare Diseases 1: e23579.

Bonde

(2011) Health musicing – music therapy or music and health? A model, empirical examples and personal reflections. Music and Arts in Action 3(2): 120–140.

Bradt

(2013) Pain management with children. In: Bradt

(ed.) Guidelines for Music Therapy Practice in Paediatric Care. Gilsum, NH: Barcelona Publishers, pp. 15–65.

Bradt

Dileo

Myers-Coffman

, et al. (2021) Music interventions for improving psychological and physical outcomes in people with cancer. The Cochrane Database of Systematic Reviews 10(10): CD006911. https://doi.org/10.1002/14651858.CD006911.pub4

Braun

Clarke

(2006) Using thematic analysis in psychology. Qualitative Research in Psychology 3(2): 77–101.

Bringas

Zaldivar

Rojas

, et al. (2015) Effectiveness of music therapy as an aid to neurorestoration of children with severe neurological disorders. Frontiers in Neuroscience 9: 427–427.

10.

Burrai

Forton Magavern

Micheluzzi

, et al. (2020) Effectiveness of music to improve anxiety in hemodialysis patients: A systematic review and meta-analysis. Holistic Nursing Practice 34(6): 324–333.

11.

Darrow

(2008) Introduction to Approaches in Music Therapy, Second Edition. Silver Spring, MD: American Music Therapy Association.

12.

Edwards

(2016) The Oxford Handbook of Music Therapy. Oxford: Oxford University Press.

13.

European Commission (2017) Rare diseases: A major unmet medical need. Available at: https://data.europa.eu/doi/10.2777/749056 (accessed 31 August 2022).

14.

EURODIS (2022) What is a rare disease? Available at: https://www.eurordis.org/information-support/what-is-a-rare-disease/ (accessed 22 August 2022).

15.

Fancourt

Finn

(2019) What Is the Evidence on the Role of the Arts in Improving Health and Well-Being? A Scoping Review (Health Evidence Network Synthesis Report 67). Copenhagen: WHO Regional Office for Europe.

16.

Hasanah

Mulatsih

Haryanti

, et al. (2020) Effect of music therapy on cortisol as a stress biomarker in children undergoing IV-line insertion. Journal of Taibah University Medical Sciences 15: 238–243.

17.

Hong

Pluye

Fàbregues

, et al. (2018) Mixed methods appraisal tool (MMAT) version 2018 user guide. Available at: http://mixedmethodsappraisaltoolpublic.pbworks.com/w/file/fetch/127916259/MMAT_2018_criteria-manual_2018-08-01_ENG.pdf

18.

Irons

Kenny

McElrea

, et al. (2012) Singing therapy for young people with cystic fibrosis: A randomized controlled pilot study. Music and Medicine 4(3): 136–145.

19.

Johnson

Berry

Bradley

, et al. (2021) Examining the effects of music-based interventions on pain and anxiety in hospitalized children: An integrative review. Journal of Pediatric Nursing 60: 71–76.

20.

Karbandi

Soltanifar

Salari

, et al. (2020) Effect of music therapy and distraction cards on anxiety among hospitalized children with chronic diseases. Evidence Based Care: Quarterly Journal of Mashhad School of Nursing and Midwifery 9: 15–22.

21.

Karpavičiūtė

Sweeney

O’Neill

, et al. (2022) The use of music for children and adolescents living with rare diseases in the healthcare setting: A scoping review study protocol. HRB Open Research 4: 52.

22.

Keating

(2020) Music therapy in Ireland: An overview of music therapy on Irish soil. Connect 2 Music Therapy, 15 July. Available at: https://www.connect2musictherapy.com/post/music-therapy-in-ireland-an-overview-of-music-therapy-on-irish-soil

23.

Kesselman

Bergen

Stefanov

, et al. (2016) Impact of music in reducing patient anxiety during pediatric ultrasound. Pediatric Reports 8: 6349–6349.

24.

Klassen

Liang

Tjosvold

, et al. (2008) Music for pain and anxiety in children undergoing medical procedures: A systematic review of randomized controlled trials. Ambulatory Pediatrics 8: 117–128.

25.

Kobus

Buehne

Kathemann

, et al. (2022) Effects of music therapy on vital signs in children with chronic disease. International Journal of Environmental Research and Public Health 19: 6544.

26.

Kole

Hedley

(2021) Recommendations for the rare 2030 foresight study: The future of rare diseases starts today (Report). Paris: EURODIS-Rare Diseases Europe.

27.

Levac

Colquhoun

O’Brien

(2010) Scoping studies: Advancing the methodology. Implementation Science 5: 69.

28.

Lin

Lee

Wei

, et al. (2012) Mozart K.545 Mimics Mozart K.448 in reducing epileptiform discharges in epileptic children. Evidence-Based Complementary and Alternative Medicine 2012: 607517–607516.

29.

Lin

Lee

Wei

, et al. (2014) Mozart K.448 listening decreased seizure recurrence and epileptiform discharges in children with first unprovoked seizures: A randomized controlled study. BMC Complementary and Alternative Medicine 14: 17–17.

30.

Lin

Huang

, et al. (2020) Effect of music therapy on the chronic pain and midterm quality of life of patients after mechanical valve replacement. Annals of Thoracic and Cardiovascular Surgery 26(4): 196–201.

31.

Liu

Gao

Hou

(2019) Effects of mindfulness-based stress reduction combined with music therapy on pain, anxiety, and sleep quality in patients with osteosarcoma. Revista Brasileira de Psiquiatria 41: 540–545.

32.

McIvor

DSH

(2023) Exploring the client-therapist relationship in music therapy: A qualitative study in adult mental healthcare. Voices: A World Forum for Music Therapy 23(3): 3931.

33.

Momennasab

Ranjbar

Najafi

(2018) Comparing the effect of listening to music during hemodialysis and at bedtime on sleep quality of hemodialysis patients: A randomized clinical trial. European Journal of Integrative Medicine 17: 86–91.

34.

Montero-Ruiz

Fuentes

Pérez Ruiz

, et al. (2020) Effects of music therapy as an adjunct to chest physiotherapy in children with cystic fibrosis: A randomized controlled trial. PLOS ONE 15: e0241334–e0241334.

35.

Moola

Moothathamby

Naganathan

, et al. (2020) A scoping review of music therapy in the lives of children and youth with disabilities and chronic conditions in non-acute medical and community-based settings: A novel consideration for art therapists? [Étude de portée de la musicothérapie dans la vie des enfants et des jeunes ayant des incapacités et des états chroniques en milieux médical et communautaire de soins non actifs: Une nouvelle considération pour les art-thérapeutes?]. Canadian Art Therapy Association Journal 33: 17–28.

36.

Nguyen

Nilsson

Hellström

, et al. (2010) Music therapy to reduce pain and anxiety in children with cancer undergoing lumbar puncture: A randomized clinical trial. Journal of Pediatric Oncology Nursing 27: 146–155.

37.

Page

McKenzie

Bossuyt

, et al. (2021) The PRISMA 2020 statement: An updated guideline for reporting systematic reviews. BMJ 372: n71.

38.

Pérez-Núñez

Lázaro

Amayra

, et al. (2021) Music therapy and Sanfilippo syndrome: An analysis of psychological and physiological variables of three case studies. Orphanet Journal of Rare Diseases 16: 486.

39.

Peters

MDJ

Godfrey

McInerney

, et al. (2020) Chapter 11: scoping reviews (2020 version). In: Aromataris

Munn

(eds) JBI Manual for Evidence Synthesis. Available at: https://jbi-global-wiki.refined.site/space/MANUAL/4687342/Chapter+11%3A+Scoping+reviews (accessed 31 August 2022).

40.

Pommeret

Chrusciel

Verlaine

, et al. (2019) Music in palliative care: A qualitative study with patients suffering from cancer. BMC Palliative Care 18: 78–78.

41.

Preissler

Kordovan

Ullrich

, et al. (2016) Favored subjects and psychosocial needs in music therapy in terminally ill cancer patients: A content analysis. BMC Palliative Care 15: 48–48.

42.

Robb

Burns

Stegenga

, et al. (2014) Randomized clinical trial of therapeutic music video intervention for resilience outcomes in adolescents/young adults undergoing hematopoietic stem cell transplant: A report from the Children’s Oncology Group. Cancer 120: 909–917.

43.

Robb

Haase

Perkins

, et al. (2017) Pilot randomized trial of active music engagement intervention parent delivery for young children with cancer. Journal of Pediatric Psychology 42(2): 208–219.

44.

Roseen

Cornelio-Flores

Lemaster

, et al. (2017) Inpatient massage therapy versus music therapy versus usual care: A mixed-methods feasibility randomized controlled trial. Global Advances in Health and Medicine 6: 2164957X17735816.

45.

Sabzevari

Kianifar

Jafari

, et al. (2017) The effect of music on pain and vital signs of children before and after endoscopy. Electronic Physician 9: 4801–4805.

46.

Sampaio

ASC

(2023) Music-based interventions in rehabilitation of children and adolescents with chronic diseases: Sharing an experience from a Brazilian public hospital. Frontiers in Rehabilitation Sciences 4: 1116914.

47.

Scheufler

Wallace

Fox

(2021) Comparing three music therapy interventions for anxiety and relaxation in youth with amplified pain. The Journal of Music Therapy 58: 177–200.

48.

Schmid

Ostermann

(2010) Home-based music therapy – a systematic overview of settings and conditions for an innovative service in healthcare. BMC Health Services Research 10: 291–291.

49.

Somanadhan

Nicholson

Dorris

, et al. (2020) Rare disease research partnership (RAinDRoP): A collaborative approach to identify research priorities for rare diseases in Ireland. HRB Open Research 3: 13.

50.

Somanadhan

O’Donnell

Bracken

, et al. (2023) Children and young people’s experiences of living with rare diseases: An integrative review. Journal of Paediatric Nursing 68: e16–e26.

51.

Stegemann

Geretsegger

Phan Quoc

, et al. (2019) Music therapy and other music-based interventions in pediatric health care: An overview. Medicines 6: 25.

52.

Uggla

Bonde

Hammar

, et al. (2018) Music therapy supported the health-related quality of life for children undergoing haematopoietic stem cell transplants. Acta Paediatrica 107: 1986–1994.

53.

Uggla

Mårtenson Blom

Bonde

, et al. (2019) An explorative study of qualities in interactive processes with children and their parents in music therapy during and after paediatric hematopoietic stem cell transplantation. Medicines 6: 28.

54.

United Nations Department of Economic and Social Affairs (UNDESA) (2013) Factsheet: Definition of youth. Available at: https://www.un.org/esa/socdev/documents/youth/fact-sheets/youth-definition.pdf (accessed 28 June 2022).

55.

Wheeler

(2015) Music Therapy Handbook. New York: The Guilford Press.

56.

Yates

Beckmann

Voss

, et al. (2018) Caregiver perceptions of music therapy for children hospitalized for a blood and marrow transplant: An interpretivist investigation. Global Advances in Health and Medicine 7: 2164956118788853–2164956118788853.

Supplementary Material

Please find the following supplemental material available below.

For Open Access articles published under a Creative Commons License, all supplemental material carries the same license as the article it is associated with.

For non-Open Access articles published, all supplemental material carries a non-exclusive license, and permission requests for re-use of supplemental material or any part of supplemental material shall be sent directly to the copyright owner as specified in the copyright notice associated with the article.

0.00 MB

0.02 MB