The Subjective Experience of Living with Parkinson’s Disease: A Meta-Ethnography of Qualitative Literature

Search strategy and study selection

In collaboration with a medical librarian (LS) we performed a comprehensive search of the bibliographic databases PubMed, Embase.com, PsycINFO (via Ebsco), Cinahl (via Ebsco) and Scopus, spanning all entries up until March 11, 2019. Search terms included both controlled terms and free text terms. The terms ‘Parkinson disease’ AND ‘experience’ (including synonyms and closely related words) were used as index terms or free-text words. For the complete search string, we refer to the Supplementary Material. The search was performed without language or date restrictions.

We included studies with a qualitative design, in-cluding interviews, focus groups and photovoice studies, describing the subjective experience of PD from a PwP’s perspective. Qualitative historical studies and case studies, as well as articles that focused on a particular aspect of PD, such as a single symptom, or experiences with interventions, were excluded. Our study sample consisted of people with self-reported or medically confirmed PD. Studies using mixed samples were only included if the results for PwP were reported separately.

Duplicates were removed from the search results. Titles and abstracts were first screened for eligibility by two authors independently (SR and ES), and full text articles of potentially eligible studies were read carefully. Disagreement over the eligibility of particular studies was resolved through discussion, including a third author (KR). SR and ES independently assessed the quality of included studies using the Consolidated Criteria for Reporting Qualitative Research (COREQ), which are used to evaluate three domains: 1) reporting on the research team and its reflexivity, 2) the rigor of the study design, and 3) the quality of the analytical process and resultant findings [8]. The quality of articles with a COREQ score lower than 13 out of 32 were discussed by SR, ES and KR, with a main focus on COREQ domain 2 and 3, and articles of insufficient quality were excluded.

Data collection and analysis

For this study, we used the meta-ethnographic approach, which was introduced by Noblit and Hare (1988) as a qualitative counterpart to the quantitative systematic review [9]. However, while quantitat-ive reviews deal with the evaluation or summarization of ‘objective knowledge’ (i.e., measurable facts), the meta-ethnographic approach is interested in ‘constructed knowledge’: concepts and perceptions, that are— according to the philosophical position of objective idealism— collectively shared. Rather than trying to ascertain the veracity of the results of the studies under review, the meta-ethnography aims to provide a synthesis that is mindful of both the commonalities of the constructed knowledge under review and the context in which this knowledge was generated.

The process of data-collection started with SR and ES independently reading the included articles, and collating the first order themes, i.e., the the-mes and subthemes reported by the authors of the article, in an Excel file (Microsoft Excel for Windows, 2016). The first order themes were compared by the two authors, and disagreements regarding interpretation of particular themes were resolved through discussion.

The analysis of the first order themes was performed by SR and KR, using the principles of the constant comparative method [10]. Through repeated comparison of the first order themes, similar concepts were rearranged into second order themes. Simultaneously, representational differences that are indicative of consequential contextual variation were identified.

As a final step in the meta-ethnography, SR and KR analyzed the relationship between the second order themes. Potential relationships between the second order themes were assessed in light of the thematic relations reported in the original studies (first order relations). Through this exercise, the researchers ar-rived at a third order construct, i.e., a model, of the subjective experience of living with PD and the factors that influence this experience, based on a synthesis of the reviewed literature. To decrease the influence of subjective bias, the second order themes and model were discussed by the two authors repeatedly, bearing the following criteria in mind:

As a final step, we performed a member check by presenting the results to two patient-researchers from the Dutch Parkinson’s disease Association (RH and KV).

Systematic search

A total of 71 full-text articles were assessed for eligibility, of which 48 were excluded, resulting in a total of 23 included studies. Based on insufficient quality, we excluded three studies more studies, resulting in a final number of 20 articles in the meta-ethnography. The selection procedure is shown in the PRISMA flow diagram (Fig. 1).

Fig. 1

Flow chart.

Critical appraisal

The mean total COREQ score of included studies was 18/32, with a range between 13 and 22. Included studies scored lowest on COREQ domain 1 (mean score of 2/8, range 0 – 4), with most studies failing to report details about the researchers and their previous relationship with the study participants. While the study design was presented clearly in most studies, reasons for non-participation, the presence of non-participants during interviews and whether the researchers took field notes, were not reported in multiple studies, resulting in a mean score of 9/15 on COREQ domain 2 (range 7 – 12). Data-analysis and reporting of findings was rated overall as sufficient, with a mean score of 6/9 on COREQ domain 3 (range 2 – 8), although most studies failed to report details on the use of software, and whether they performed a member-check.

Details of the included studies are provided in Table 1. Across all studies, a total of 279 PwP were included. Of these PwP, 137 were female and 142 male. The mean age and age range was not disclosed in all studies, but reported ages of participants varied from 32 to 95 years.

Data analysis and synthesis

The extraction of data resulted in a total of 227 first order themes. Nineteen first order themes could not be categorized because they did not describe specific content (e.g., “complexity”), or because they were not specific enough (e.g., the first order theme “challenging perspectives” referred to acceptance of the diagnosis, decreased physical activity, curtailment of social activities and emotional losses). These themes were excluded from the meta-analysis. Through comparison, the 208 included first order themes were all categorized into five second order themes: 1) Awareness (n = 17 first order themes), 2) Disruption (n = 35), 3) Adjustment (n = 77), 4) The external environment (n = 44), and 5) The changing self (n = 34). A full description of these themes and subthemes is provided in the codebook in Table 2.

Following our comparative analysis, we identified two overarching thematic relationships. A first set of themes relates to disruptions (i.e., physical and mental symptoms, negative affect and cognitions caused by PD), the way in which these disruptions may be alleviated or exacerbated by the external (i.e., social, professional and medical) environment, as well as the adjustments (coping strategies) employed by PwP. A second cluster of themes stresses the evolving nature of life with PD and includes awareness of PD and changes in self-perception and identity (the changing self), as the disease progresses over time.

Our “model of dialectic change” (see Fig. 2) encompasses both relationships. Central to this model is the dialectic between stability and instability, as well as the ongoing negotiation between the external environment and the self. Inspired by Hegel’s dialectics (2010) [11], our model holds that this dialectic is comprised of three states: stability, instability, and unity. A sense of stability results from closely held beliefs about the self and the role of the self within the personal environment. Instability arises when the individual is presented with a contradictory truth that renders these prior beliefs untenable. The arrival at a new equilibrium requires a process of concurrent negation and preservation, whereby the original understanding about the self and the social roles is somehow unified with its contradiction.

Fig. 2

Model of dialectic change.

The start of this dialectic process is generally marked by the diagnosis, which necessitates that PwP in some way reconcile their diagnosis of PD with their prior concept of the (healthy) self [12, 13], and periodically resurfaces when symptoms exacerbate and/or interrupt the personal status quo. PwP frequently respond to the resulting instability through adjustments that are aimed at maintaining the extant self-concept [13– 16]. These adjustments can be psychological strategies, e.g., employing relativism or downward comparison [17– 19], but often address the external environment. For example, PwP may start a new form of treatment, implement new daily routines, or rely more on support from their family [9, 12, 13, 17– 22].

However, if external adjustments prove incapable of preventing changes in physical or cognitive functioning that preclude the performance of tasks or roles that the person with PD strongly identifies with, the self-concept rather becomes the focus of the dialectic process [16, 23], e.g., a person with PD who considered his independence as an important personal trait will need to redefine his self-concept when he becomes increasingly dependent on others due to physical limitations. This requires a ‘sifting and sorting’ of previously held beliefs about the self and identification of alternative or novel representative traits [13– 15, 19].

Notable is that our “model of dialectic change” suggests that it is not necessarily the prevalence or intensity of symptoms, but rather the degree of perceived stability that primarily determines a PwP’s quality of life. Stability in turn depends on the degree to which PwP are able to sustain roles, activities and closely held beliefs about the self [13– 15, 19]. The interaction between symptoms, physical and cognitive limitations, and individual notions of stability highlighted by our model suggests it does not necessarily follow that instability is more frequent or intense in the later stages of PD. However, during late stage PD, potentially successful coping strategies do become less numerous and/or effective [24], e.g., motor symptoms might no longer be sufficiently reduced by medications, and social interactions may be hampered by cognitive or speech problems. The-refore, episodes of instability may become more dominant in later disease stages [16, 25, 26].

In sum, our model conceptualizes life with PD as a transformative journey, wherein PwP employ strategies intended to stabilize their changeable relationship with their external environment, as well as actively engage in a continuous process of reconstituting the self.

Our member check with two patient-researchers of the Dutch Parkinson’s disease association (RH and KV) affirmed that the results of our meta-ethnography summarize their experience of living with PD. However, as they pointed out, the model is not specific to the experience of living with PD; it is also be app-licable to the process of adjustment after life-events during an average person’s life. However, both pat-ient-researchers emphasized this does not detract from its value, as people living with PD are people first and patients second.