Perceptions and experiences of control among people living with motor neurone disease: a systematic review and thematic synthesis

Figures & data

Table 1. Inclusion and exclusion criteria.

Inclusion criteria	Exclusion criteria
Sample Studies investigating adults with MND of any type.	Articles which are not about MND. Articles which include MND but the data for those with MND cannot be separated from other illnesses in the study.
Phenomenon of interest Articles exploring plwMND’s perceptions or experiences of control or any domains related to control in their day to day lives. Control includes any mention of control or autonomy or agency or any control constructs such as self-efficacy or mastery.	Articles that do not focus on daily living e.g., those that focus solely on diagnostic process or end of life care. Articles which make no mention of control, or any aspects related to control in their report Articles that focus on a particular institution or intervention or development of a scale.
Design Primary studies of any type of qualitative method or a systematic review of qualitative methodologies. Qualitative data or mixed methods if qualitative data clearly separated Any type of data collection e.g., interviews, focus groups however data must come directly from the plwMND Systematic reviews of qualitative or mixed methods studies	Quantitative research or mixed methods if qualitative data cannot be isolated. Studies where data came from carers or health professionals
Research type Peer-reviewed journal articles	Books, theses, conference proceedings, grey literature, case studies, editorials, letters and other non-peer-reviewed articles Any primary research articles already included in a relevant systematic review. Articles not in English Articles published before 1999

Figure 1. PRISMA 2020 flow diagram.

The PRISMA flow chart shows that searched retrieved 6755 publications of which 468 were duplicates, 6287 were screened by title and abstract, 91 were screened by full text with 70 excluded. 20 were included in the review.

Table 2. Characteristics of included studies.

Study	Location	Study aims	Sample details	MND Type	Point in MND journey	Methodology and data collection
Ando et al. [28]	UK	To explore patients’ perceptions and use of NIV over time	5 People (m = 4) 51–75 years	3 bulbar onset 2 limb onset	Illness duration 23–237 months	Phenomenology Semi structured interviews over a year
Ando et al. [29]	UK	To explore why some plwMND refuse or withdraw from NIV	9 People (m = 7) 40–79 years	3 bulbar onset 6 limb onset	Unable to ascertain	Phenomenology Semi structured interviews
Ando et al. [30]	UK	To understand factors important to QOL and explore how plwMND maintain and/or improve QOL	26 People (m = 14) 39–83 years	13 limb onset 13 bulbar onset	Illness duration 1–110 months	Semi structured interviews
Cave and Bloch [31]	England	To investigate what plwMND consider when deciding whether or not to bank their voice And what are the expectations of significant communication partners	21 people 12 plwMND (m = 6) 30–90 years 9 significant communication partners	Not specified	Time since diagnosis 1–12 months	Semi structured interviews
Cipolletta et al. [32]	Italy	To identify illness trajectories in pwMND	18 people (m = 8) 43–84 years	12 ALS spinal form 5 bulbar form 1 atypical form	Time since diagnosis range a few days to 4 years	Mixed methods Grounded Theory for qualitative section Semi structured interviews
Foley et al. [33]	Ireland	To investigate how and why plwMND engage with healthcare services	34 people (m = 17) 37–81 years	Not specified	Duration since onset of symptoms 4 months − 13 years	Grounded Theory Interviews
Foley et al. [34]	Ireland	To identify key psychosocial processes that underpin how plwMND engage with healthcare services	34 people (m = 17) Age ranges 20–39 years to > 80 years	26 spinal onset 6 bulbar onset 2 respiratory onset	Disease duration 4–169 months	Grounded Theory Interviews
Greenaway et al. [35]	UK	To investigate the decision-making processes involved in accepting or declining NIV	21 people (m = 13) 41–76 years	Not specified	Duration of disease 6–60 months	Method not specified used an inductive thematic analysis approach. Semi structured interviews
Gysels and Higginson [36]	UK	To explore and compare the lived experience of breathlessness in patients with COPD, heart failure, cancer and MND	48 people. plwMND = 10 (m = 9) Age range 24–77	Not specified	Not specified	Method not specified a form of framework analysis used Semi structures interviews
Harris [37]	UK	Aims to answer the question “what does it mean to be a person living through the illness trajectory of MND”	4 people (m = 3) 55–70 years	3 ALS 1 PMA	Time since diagnosis 14 months − 10 years	Hermeneutic phenomenology Life story interviews
Harris et al. [38]	UK	To explore the meaning of living with uncertainty in pwMND	4 people gender not specified Age range not specified	Not specified	Had been receiving treatment for MND for 3–6 months	Hermeneutic phenomenology Interviews
Hocking et al. [39]	New Zealand	To shed light on the experience of living with MND	7 people (m = 3) 48–72 years	Not specified	Time since diagnosis 8 months − 4 years	Phenomenology Interviews
Hogden et al. [40]	Australia	To identify factors influencing decision making in MND	14 people (m = 7) 40–77 years	Not specified	Time since diagnosis 2–93 months	Not specified but data analysed thematically
Mistry and Simpson [41]	UK	Exploring the transitional process from diagnosis to living with MND	7 people (m = 3) 57–75 years	Not specified	All diagnosed within previous 6 months	Phenomenology Interviews
Ozanne et al. [42]	Sweden	To investigate how plwMND create meaning despite their diagnosis	14 people (m = 7) 42–80 years	Not specified	Duration of disease 2–13 years	Not specified but used qualitative content analysis Semi structured interviews
Pavey et al. [43]	UK and Australia	Investigating technology and response to bodily limitation in MND	42 people (m = 31) 42–74 years	Not specified	Not specified	Phenomenology 10 face to face interviews others conducted via email to allow for those with communication difficulties
Pinto et al. [44]	UK	To understand the emotional impact of living with MND and explore what impacts experiences of emotional distress and wellbeing	35 people 25 plwMND (m = 15) 39 − 80 years	ALS limb = 18 ALS bulbar = 1 PLS = 4 PBP = 1 PMA =1	Time since diagnosis 2 months − 17 years	Semi structured interviews
Sakelleriou et al. [18]	Includes studies from UK, Sweden, USA, New Zealand, Ireland, Australia Canada	To answer the question “What is known about people’s experiences of living with MND?”	257 people gender not specified 25–85 years	Not specified	Time since diagnosis 1 month − 3 years	Followed CRD systematic review procedures for search, quality appraisal, data extraction, analysis and synthesis
Weeks et al. [45]	UK	To explore needs and preferences of plwMND about psychological interventions and how these may be adapted for this populaton	37 people 15 plwMND (m = 9) Age range 56–75 years	ALS = 10 PLS = 4 PBP = 1	Not specified	Semi structured topic guides
Yuan et al. [46]	China	To gain understanding of the illness experience of als and meaning attached to these	20 people(m = 13) Age range 28–72 years	ALS =20	Time since diagnosis 6–60 months	Semi structured interviews

Table 3. Overview of themes and study support at subtheme unit.

Analytical theme	Sub themes and supporting studies
A diagnosis of MND leads to a significant disruption of previously held control beliefs	Control beliefs about health [29,32,34,36–39,41,44–46]
	Control beliefs about fate [30,32,36,38–42,46]
	Control beliefs about identity [18,29–33,39,41–44,46]
	Control beliefs about the body [18,29–30,32–35,39–42,44–46]
PlwMND use a range of control strategies to attempt to retain control in their lives	Retaining control through developing their own illness beliefs [18,28,29,31,33–35,38–40,43,44]
	Retaining control through focusing on the present [18,29–36,38,40,42,44]
	Retaining control through focusing upon personal values [18,29–31,33,36,40–43,44,46]

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