Layout 1 [Qualitative Research in Medicine & Healthcare 2020; 4:9427] [page 95] Introduction An estimated four million people worldwide suffer from Parkinson’s Disease (PD), making it the second most common progressive neurodegenerative disorder after Alzheimer’s disease. PD is manifested by motor symp- toms such as tremor, involuntary movements (dyskinesia), postural instability, rigidity, freezing episodes or slowness of movement, and nonmotor symptoms like anosmia, de- mentia, gastrointestinal disturbances, abnormal urinary function, hallucinations, depression, anxiety, decreased li- bido, apathy, sleep disturbances or difficulties swallow- ing.1,2 All these symptoms have a large impact on PD patients’ physical and mental health, which can lead to loss of autonomy and self-esteem, altered relationships, and social isolation.3 Furthermore, PD does not present a challenge to patients only but also to their family care- givers.4 Nowadays, PD can be managed through a variety of oral pharmacological treatments such as Levodopa, dopamine agonists, transferase inhibitors or anticholiner- gic medication.1 Device-aided therapies such as Deep Brain Stimulation (DBS) or medicine pumps such as the duodopa or the apomorphine pump can be an option for PD patients who are refractory to oral treatment. Unlike oral therapies, infusion medicine therapies are based on constant drug delivery, which can reduce the burden of motor complications associated with PD. Currently, dif- ferent options are available for medicine pumps like the continuous subcutaneous apomorphine infusion, the in- trajejunal Trigel infusion or the intestinal gel infusion of Levodopa/Carbidopa, also known as the duodopa pump.5 This paper focuses on the duodopa pump (from now on the pump), which delivers levodopa/carbidopa-gel sus- pension directly in the duodenum through a plastic cas- sette linked to a pump that is connected to a tube inserted to the intestine through a small cut in the gut wall.6 This allows more stable plasma concentration of the drug than oral treatments, which improves patients’ quality of life. Living with Parkinson’s disease and connected to the duodopa pump: A qualitative study Yolanda María Chacón Gámez, Nikola Biller Andorno Institute of Medical Bioethics and History of Medicine, University of Zurich, Switzerland ABSTRACT Parkinson’s Disease (PD) is a complex and heterogeneous disease that has a major impact on the lives of patients and their family caregivers. The duodopa pump is one of the treatments options for advanced PD. The experience of patients with this treatment is largely unexplored, however. This study is based on the experiences of patients with PD as well as their family caregivers using the pump. We take a multimodal approach that includes narrative semi-structured interviews and drawings. This methodology helps par- ticipants, particularly those with difficulty articulating themselves, express their experience with the disease and their perception of the treatment. We believe that the use of drawings complementing semi-structured interviews offers additional insights into the participants’ perception of PD and the pump. In this article we focus on the following topics: the participants’ perception of PD and the pump, the reasons for choosing the pump over alternatives, and the advantages and disadvantages of this treatment for patients and family care- givers. Our findings show that the participants consider PD as a complex and unpredictable disease that changed their lives and affect everyone differently, while the pump makes the disease less changeable and more manageable. Correspondence: Yolanda María Chacón Gámez, Institute of Med- ical Bioethics and History of Medicine, University of Zurich, Wintherthurerstrasse 30, 8006, Zurich, Switzerland. E-mail: yolanda.chacon@ibme.uzh.ch Key words: Parkinson’s disease; duodopa pump; patients’ percep- tion; family caregivers’ perception; drawings. Acknowledgements: We would like to thank to the Spanish foun- dation “La Caixa” for their economic contribution to our study; to Prof. Dr. Stephan Bohlhalter, Dr. Florian Brugger, Dr. Stefan Hägele-Link, and the self-help groups of the Association Parkinson Schweiz for their cooperation in our study and their help in the se- lection of participants; and to Dr. Matthew Baker for his assistance and comments that greatly improved this manuscript. Contributions: YCG: Set-up of the study, data collection and analy- sis; literature review; manuscript write-up; manuscript editing; NBA: Research supervision; manuscript write-up; manuscript editing. Conflict of interest: The authors declare no conflict of interest. Funding: The Spanish foundation «La Caixa» has funded part of the study from which the data presented in this paper are drawn. Received for publication: 30 October 2020. Accepted for publication: 7 February 2021. This work is licensed under a Creative Commons Attribution Non- Commercial 4.0 License (CC BY-NC 4.0). ©Copyright: the Author(s), 2020 Licensee PAGEPress, Italy Qualitative Research in Medicine & Healthcare 2020; 4:95-106 doi:10.4081/qrmh.2020.9427 Qualitative Research in Medicine & Healthcare 2020; volume 4:95-106 No n- co mm er cia l u se on ly In 2016, a quantitative study about the preferences of PD disease patients for device-aid treatments was conducted with 401 patients in US. Its aim was to evaluate patients’ willingness to accept trade-offs among efficacy, tolerabil- ity, and convenience of alternative treatments. It showed that treatment delivered via portable infusion pump was preferred over DBS.7 However, there are many aspects of the user experience with medication pumps that we still do not know. To our knowledge, no study has been con- ducted that addresses patients’ lived experiences with medicine pumps for PD. Thus, the personal impact of these treatments on patients’ daily life is completely un- explored. We believe that we need to better understand how patients perceive medicine pumps and how they per- sonally cope with PD symptoms and the treatment with these devices. To do this, it is also relevant to incorporate the perspective of family caregivers as a valuable source of knowledge because they are sometimes more sensitive to changes in the patient than patients themselves.8 For these reasons, we consider it very important from both a clinical and an ethical point of view to conduct more qual- itative research to gain a richer understanding of patients’ and caregivers’ experiences, which will facilitate the con- tinuous improvement of patient-centred care for PD pa- tients. Our research fills this research gap by analysing the accounts of the experiences of a sample of PD patients treated with the pump and their caregivers. Materials and Methods The data presented in this study are part of a more ex- tensive qualitative study conducted in Switzerland be- tween 2018 and 2020, where we collected and explored a wide range of experiences of 44 patients with PD and aid- device treatments (DBS or the pump) and their caregivers. We conducted multimodal research including narrative semi-structured interviews and drawings. We identified four groups of experiences reported by patients and their caregivers: i) daily life and perception of PD and aid-de- vice treatments; ii) self-perception; iii) social interaction and partnership/family dynamics; iv) health needs of PD patients and interaction with the healthcare professionals. Due to the vast amount of data obtained during this study, in this article we will focus on the first category, which covers the perception of patients and caregivers of PD and the pump, the reasons for choosing the pump over other alternatives, and the advantages and disadvantages of this treatment for patients and caregivers. For better dissemi- nation, our results will be displayed on the website of the Swiss Database of Patient Experiences (www.dipex.ch) over the course of 2021. Our research group is part of the international Database of Patients Experiences research initiative (DIPEx International), which comprises fourteen countries. DIPEx represents a wide spectrum of diverse perspectives about the same topic (including deviant cases) and is addressed to a wide public (patients, care- givers, healthcare professionals and students).8-10 The aim of the DIPEx website is fourfold: i) empow- ering patients and family caregivers to talk about their per- sonal health experiences and to give first-hand information that health professionals not always have; ii) offering other patients and family caregiver emotional support and help with decision-making by offering them the possibility of knowing about the experiences of others in the same situation; iii) creating social awareness about how life suffering from a disease is; and iv) serving as training and educational tool for health sciences students and professionals. Study population A total of ten participants, including six patients treated (three men and three women) and four spouses (three women and one man) participated in the study section on experiences with PD and the pump. The characteristics of patients and caregivers are described in Table 1. Two of the patients lived in a nursing home and took part in the study alone. One of the participants was treated simultaneously with the pump and DBS. For the selection of participants, purposeful maximum variation sampling was performed to identify those individuals whose experiences were espe-cially informative and would vary from each other as much as possible. The following inclusion criteria were applied: i) pa- tients diagnosed with PD or caregivers providing care to a PD patient; ii) patients treated with the pump for at least six months or caregivers providing care to PD patients treated with the pump for at least six months. The follow- ing exclusion criteria were applied: i) lack of legal com- petency; ii) people experiencing moderate or severe dementia or substance addiction at the moment of the in- terview; iii) lack of physical and psychological resilience to participate in an interview or difficulties interacting with an interviewer. The sample was small because only a low number of [page 96] [Qualitative Research in Medicine & Healthcare 2020; 4:9427] Article Table 1. Description of patients and caregivers of the interview and details of their interviews. All participants were interviewed in 2019 except for the last participant, who was interviewed in 2020. Group Number Average ageat interview Average years with PD Average yearswith the pump Average interview duration Patients 6 73 15.6 4.1 91 Caregivers 4 70.7 —- —- 57 No n- co mm er cia l u se on ly patients in Switzerland are treated with this therapy. De- spite the small sample, we reached data saturation as we did not observe new themes when analysing the last in- terviews. We recruited the participants with the help of neurologists, PD nurses, and patient support groups be- longing to the Swiss Association Parkinson’s Switzerland. We conducted this study in collaboration with the Depart- ments of Neurology of the Cantonal Hospitals in St Gallen and Lucerne. Informed consent process The participants were informed in great detail about the study before participating. The day of the interview, they signed the informed consent form that allowed us to proceed with the interview and its subsequent analysis for our research project. As part of the form, study partici- pants were also given the opportunity to express their preference between being video- or audiotaped to partic- ipate. After the interview, participants received a second informed consent form, through which they could accept or reject the use of the interview for the DIPEx website and choose how they wanted the information to be pre- sented on the website (video, audio or test). To this aim, the participants received a copy of the interview tran- scripts as well for verification. Although no personal information (such as personal names or locations) will appear on the website that could lead to the identification of participants, those who have chosen to have their interview used for the website in audio or video format are aware that they may be recog- nised through their image or voice by people who may be able to identify them. It is therefore important that the par- ticipants had the opportunity of verifying the transcripts to decide what they would and would not like to be public information. Given that after the transcript review, not all participants may wish that some parts of their interviews would be made public, the final number of participants being included in the DIPEx platform could be lower than the number of participants in the study. Data collection We applied a multimodal approach that includes the conduction of narrative semi-structured interviews and the collection of drawings. This multimodal methodology al- lows to collect data about the experience with the disease and the treatment incorporating both language-based and nonverbal communication, which helps participants, par- ticularly those with difficulties to articulate themselves, to express their individual experiences and give them meaning.11,12 We collected the data of the presented dataset collecting drawings and conducting ten narrative semi- structured interviews in the language the interviewees felt most comfortable with. Seven were in German (with some parts in Swiss German), two in Italian and one in English and the first author conducted all of them. The first author is fluent in the three languages used in the study (German, Italian and English) and conducted all the interviews with- out the language being a barrier between the interviewer and the interviewee. Each interviewee was interviewed separately, but some of them were in company of the spouse and others were alone, depending on how they felt most comfortable. The interviews took place either at the participants’ homes or in the nurse homes where they live. To conduct the semi-structured interviews, we em- ployed the DIPEx methodology, which is an established qualitative methodology developed by the research group of the department of Public Health of the University of Oxford.9 Therefore, all interviews started with a general question to give the participants the freedom to put their personal narrative into words. This question is as follows: “Could you explain your experience with Parkinson’s and the duodopa pump from the beginning? How did every- thing start?”. Once the participants had finished telling their stories, we asked a series of semi-structured ques- tions that we separated into two blocks. The first one in- cluded questions about the participants’ experiences with PD and with previous treatments like oral medication, their daily life with PD as patients or caregivers and their perception of PD as disease including questions about their drawing of the disease. The second section included questions about, their daily life with the pump, their per- ception of the pump including questions about their draw- ing if the treatment, the advantages and disadvantages of the pup over previous treatments as well as their reasons to choose the pump over other treatments. Additional as- pects (e.g. self-perception, relationships…) were ad- dressed during the interviews, but as it was mentioned in the introduction, they will not be presented in this paper. To enrich the participants’ oral account, we used draw- ings in this study as complementary qualitative method- ology for several reasons. First, to draw allows the participants to visually show the consequences of chronic health conditions and share the emotions that their condi- tion provoke on them.13-15 Second, the drawings offer ad- ditional insights to the researcher into the patients’ and caregivers’ experiences with PD and the pump because they provide access to nonverbal meanings and to a qual- itatively different aspect of the participants’ experiences.16 Third, we believe that a multimodal approach is an effec- tive way to work with vulnerable participants and address sensitive topics.17 At the beginning of each interview, both patients and caregivers were invited to draw how they visually per- ceived the disease and the pump. Although other authors usually ask the participants to draw after the interview,18 we decided to ask our participants to draw before con- ducting the interview to provide our participants with the opportunity to reflect on their own history and circum- stances before recounting it, because we considered that this preliminary reflection would contribute to enriching the results obtained from the study. The participants re- [Qualitative Research in Medicine & Healthcare 2020; 4:9427] [page 97] Article No n- co mm er cia l u se on ly ceived a sketching pencil, a set of twelve colour pencils, an eraser and two pieces of paper (one to draw their visual representation of PD and another for the pump). Five par- ticipants opted not to draw due to physical incapability or the lack of a visual image of the disease or therapy. During the interview, the participants who drew were asked dif- ferent questions about the drawings they had made. It was not necessary for the participants to know how to draw well. It only mattered that their drawing had a meaning for them that they could later explain during the interview. Data analysis We performed our data analysis using a modified ap- proach of grounded theory, which required a continuous back and forth between data collection and analysis.9,19 To proceed with the analysis, the interviews were transcribed verbatim by our team of transcribers. After the partici- pants’ verification, we fed the transcripts and the drawings into the qualitative data analysis software MAXQDA, which allowed us to perform multimodal analysis. After- wards, we thematically coded data using a coding scheme (coding tree), which we primarily created based on the in- terview guide and continuously enriched with topics emerging from the transcripts and the drawings (mix cod- ing).20 Every time that we created a new category or code, we reviewed all coded segments to ensure homogeneity within the entire data set. This way, the concepts that we used to develop the final coding tree stem from the col- lected participants’ life experiences through the semi- structured interviews and the drawings, which we subsequently systematized, categorized and analysed fol- lowing the coding tree.9 The final coding tree was checked by other members of our team to ensure trustworthiness. The coding tree was created in English to allow the dis- cussion about it with other members of the team who do not speak German or Italian, but all data were coded and analysed in its original language by the first author. The second author supervised the analysis. Further analysis will select relevant material for our website www.dipex.ch using the One Sheet Of Paper (OSOP) method, which involves reading through each section of coded data for each topic and summarize on a single sheet of paper the key points of all interviews in re- lation to the same topic.9 The resulting text will constitute the summary of the different topics for the website. The website will be organized in the categories derived from the coding tree (e.g. PD symptoms, patients’ or caregivers’ difficulties with PD, work life with PD…), and all texts, video and audio clips on the website will be classified along these categories.9,10 Ethical concerns and data management The study participants were not at risk of any physical harm and did not directly benefit from the study. We con- ducted this study in compliance with the current version of the Declaration of Helsinki, the ICH-GCP and ISO EN 14155 (as far as applicable) as well as all national legal and regulatory requirements. The study has been reviewed by the ethics review committee of the Canton of Zurich, which considered that it did not fall under the Swiss Law on Human Subjects Research and, after consultation with the national working group of Swiss ethics review com- mittees, issued a nation-wide waiver. Subsequently, our study proposal went through our Institute’s internal ethics review process (CEBES). We handled all data confiden- tially and only persons who are directly involved in the data collection, transcription or data analysis had access to them. Data are protected by copyright and belong to the Institute (the name will be provided when the manu- script is accepted for publication), which can use the col- lected data for research and teaching purposes including publications or broadcasting. We performed data anonymization and we saved the data of each participant giving them a code (e.g. Patient1, Caregiver1...) and deleting from the transcripts any infor- mation that that could lead to the personal identification of participants. In order to present our results in this paper, we translated the selected quotations of the interviews that were not conducted in English using the DeepL Pro Trans- lator. The translated data were not stored by DeepL and all translations were compliant with our data protection regu- lations. The final translations were verified by a native speaker to ensure accurate translation of the original quotes. Results Patients’ and caregivers’ perception of PD The reactions of the different participants when receiv- ing the diagnosis were very different from each other. While most participants, both among patients and care- givers, stated that they were not surprised by the diagnosis of PD, one participant stated that she felt devastated when she received the PD diagnosis: “That was a shock to me because I didn’t know how it would play out, how it would progress. The diag- nosis blows your mind. It smashes you and you feel disappointed. The truth is difficult to accept. The di- agnosis is difficult to accept (…).” (patient 6). “What was the most difficult thing for you to ac- cept?” (author 1) “That it is incurable.” (patient 6). Some of the participants even expressed relief at the diagnosis because they were aware that something was wrong and did not know what. Others admitted not being aware of what it meant to have PD. One of the patients said the following: [page 98] [Qualitative Research in Medicine & Healthcare 2020; 4:9427] Article No n- co mm er cia l u se on ly “I had no idea that it affected the whole body. I thought I just end up with tremor one day. I had no idea what it meant. And a lot of people don’t know what it means. Even medical people (…) I was ig- norant. I didn’t know what was ahead of me. So, they probably thought I was just cheerful by na- ture, but I was ignorant (…).” So, you didn’t feel shocked? (autor 1) No. I just carried on. You know and then slowly or suddenly I couldn’t do things.” (Patient 4). This feeling of discovering that living with PD was much harder than they expected at the time of diagnosis was shared by other participants. When explaining their experiences with the disease, different participants ex- pressed things such as: “This is a terrible disease” (care- giver 3);“It’s just a break in life” (caregiver 1) ;“I believe that I have been a victim of this disease for a long time” (Patient 1) or “Every day I also see that there is a change in the worse possible sense” (Patient 5). One of the care- givers explained that his wife went through a very difficult period with the disease two years ago when she consid- ered applying for assisted suicide (which is legally al- lowed in Switzerland). Despite the difficulty of living with this disease chronically either as a patient or as a family caregiver, most expressed relief that PD is not a fatal disease. For instance, one of the patients, who also suffered from cancer, said: “Parkinson’s makes you sick, but you don’t die” (…) Cancer was worse than Parkinson’s because it was eating me.” (Patient 5). Another experience shared by virtually all patients and caregivers is the impression that PD changes your life but not in the same way for everyone because everybody’s PD is different. Not only can each person have different symptoms, but each patient faces them differently, which means that each of them goes through the disease in a dif- ferent way. While some responded to PD symptoms in a more physical way by trying to be active most of the day as they experienced less symptoms if they were focused on some activity, others dealt with the symptoms rather in a mental way by trying to think positively about the disease. One of the patients mentioned that facing PD with a sense of humour is the most important thing for her to not feel swamped by the disease and described how to find the fun side of the disease. “I can’t live without a sense of humour/ you can’t live without. You’ve got to see the other side as well (…) My mother had a very good sense of humour and was always giggling. So, maybe I grew up with that mentality (…) But since I have Parkinson’s, I laugh much more. When I come together with friends and I tell them these stories that happened to me, and they’re very serious stories. We should really cry, but we just end up laughing (…). In re- habilitation, another woman who had deep brain stimulation and we were always laughing at one an- other, you know, because uhm/ these/ all these hitches, all things that didn’t work (…) And we had so much fun. (…) If you don’t get drowned by the situation, it’s very funny.” (patient 4). If we look at the way some patients and caregivers vi- sually captured the disease on paper through their draw- ings, we get more extra information about how they perceive the disease. In Figure 1 we can see how patient 4 visualises her feeling that her motor skills fluctuate during the day through the use of different colours. She represented PD symptoms as a figure in blue, and the effect of the oral medication consisting of levodopa as different figures in red, orange, and yellow. Although this treatment improved her motor skills, its effect was not stable because it was wearing off and she began to experience off-periods. Therefore, she represented movement through various fig- ures that are intermingled during the day with the PD symptoms. “Well, the colours for me, the red, orange are like dynamo, like movement and dynamics. And the blue is the more rigid, is a rigid position, a rigid / the rigid leg. That’s how I depict Parkinson’s, rigidity.” (patient 4). Rigidity is for you the definition of Parkinson’s? (first author). Yeah. Stiffness (patient 4) (…) [Qualitative Research in Medicine & Healthcare 2020; 4:9427] [page 99] Article Figure 1. Patient’s 4 perception of PD. No n- co mm er cia l u se on ly So, the blue figure is the underlying disease? (first author). Yeah, yeah. And that’s the release with something like levodopa. These colours there are the move- ment (pointing at the figures in red, orange and yellow).” (patient 4). The unpredictability of PD is reflected, in two differ- ent ways, in Figure 2 from the perspective of one the care-givers. On the one hand, as PD is degenerative disease, there is always the big question of how the disease will develop in the long term and how life will be then, which is un- predictable. On the other hand, patients and caregivers need to cope with the unpredictability of the daily fluctu- ations of PD symptoms, which makes daily life difficult. “That’s the Parkinson’s. That is, yes, exactly, the Parkinson’s. You don’t know what to do next. (….). And here, what’s difficult with Parkinson’s is the fluctuations (pointing at the curve) (caregiver 3). And what does the straight-line mean? (first author). This is a medicine. Different drugs. (…) I don’t know where the limit is that the medication stops working. Doctors don’t know that either. With one person it takes longer to reach it and with others less. We need to live every day.” (caregiver 3). Patients’ and caregivers’ perception of the pump In Figure 3, we see how patient 4 perceives the effects of the pump differently in comparison to oral treatment. While in Figure 1 we could observe several figures in different colours reflecting movement, which contrasted with the blue figure representing PD, in Figure 3 we can see how patient 4 perceives the effect of PD as a single orange figure that is jumping and contrasts with the rigid- ity of the figure in blue. This representation reflects her perception of the effect of the pump being more stable and consistent than the effect of oral medication, which gives her greater motor mobility throughout the day. “This is a stooping figure (pointing at the blue fig- ure). That’s what Parkinson’s does to most people. But this is what I feel like when I feel when I fed/ fed the pump (pointing at the orange figure). You know I feel much livelier. Everything goes upwards instead of downwards. That’s a mix of/that’s what I feel like with the pump, okay? (…) Just, you know, not dragged downwards, but moving upwards. I can move more easily and uhm I can walk very quickly when I’m/when I’m feeling good.” (patient 4). Although she was very satisfied with the pump as treatment, she described a sense of dependence on the pump to be able to move properly. “I’m now living with uhm uhm an attachment to me (…) Now I’m not dependent on tablets, but I’m dependent on the cassette, am I not?” Looking at the caregivers’ perception of the treatment, one of them compares their experience with the pump in comparison to oral medication in Figure 4. “Those are the drugs (pointing at the circles on the top of the drawing, which represent the hours his wife had to take her pills every day). She had to take 8 pills a day (…). Every day she was suffering from Parkinson’s disease from morning to night, because she was constantly reminded that she has Parkinson’s disease. And with the pump we turn it on and off.” (caregiver 3). The pills taking routine was replaced by the pump cleaning, activation and deactivation routine, which is car- ried out by the patient’s husband. He explained that each [page 100] [Qualitative Research in Medicine & Healthcare 2020; 4:9427] Article Figure 2. Caregiver’s 3 perception of PD. Figure 3. Patient’s 4 perception of the pump. No n- co mm er cia l u se on ly morning at 06.00, he takes the levodopa/carbidopa-gel out of the fridge to let it warm up a bit before putting it in the cassette of his wife’s pump at 08.00 to switch on the pump on for the day. In the evening, he switches the pump off at 18.00, and at 20.00 he cleans the tube that goes into the patient’s intestine. While caregiver 3 was explaining his drawing, he mentioned that he started a diary since his wife was treated with the pump. He used it to keep track of the involuntary movements that his wife suffers throughout the day to accordingly adjust the dose of med- ication put into the cassette. “The pump is much simpler. But the adjustment is very, very difficult (…) An adjustment can be good for three or four days and suddenly it’s nothing. There must be other events and then there can be fluctuations. But I think it’s much less today than it was before with the medication (…) I take some minutes to write down every day the symptoms and the duration because you can’t keep every- thing in your head. Mr. (Name_1) is our doctor and gave us free hand in varying the dosage. He gave us some recommendations and said, “try it once”. I am also aware that every Parkinson is different. Every person reacts differently. This is very diffi- cult. But that’s what it is with these drugs.” The reasons to choose the pump over other treatments Most participants chose the pump as a treatment be- cause oral treatment was no longer effective for manage- ment of their PD symptoms and their quality of life was very poor. Most of them expected to enjoy greater mobil- ity with the pump and noted that the PD symptoms de- creased with this treatment (e.g. less uncontrolled movements or freezing episodes). For one of the patients, the reason to choose the pump over oral treatment was that the side-effects of drugs taken orally were unbearable. His wife explained that the treatment with dopamine ag- onists caused him to have, as a side effect, a serious sex addiction, which led him to spend a great amount of money in sexual relations with several prostitutes. “He took drugs, various drugs. At first, they fluctu- ated and also people change. He was not the same person anymore, because with the pills, it always goes high and then it goes down again and then up again (…) He got stronger and stronger medication over the years (…) And the pills have very bad side effects. Especially the Sifrol that was what he got. And then many unpleasant things happened. So vi- olently. And that was the reason why/I immediately told the doctor (…) He cheated on me for years with prostitutes. That was a disaster (…). That’s why doctor (name_1) said that with the pump would be much better.” (caregiver 1). Two patients were offered the possibility to choose DBS. One of them was doubtful at first but ended up re- jecting it because she would not have felt comfortable with a device inside her brain. Her husband explained that he did not know how he would have reacted if his wife would have chosen DBS instead the pump because DBS is a treatment he could not morally agree with. “At first she was not determined. For her it was an opportunity. And for this reason, we were informed at the hospital. How much that I influenced her in her decision, I do not know of course. But for me it was never an issue. This is a catastrophe for me. (…) Drilling holes in the brain, that’s not possible. A person is not a machine where you can screw in a screw. For me there are limits in all medicine. And it borders on ethics (caregiver 3). So deep stimulation is not an ethical treatment for you? (first author). No, no, this is a mechanical treatment (caregiver 3). Why is DBS not ethical? I am not a religious person, not at all, but I think that many things are wanted, somewhere wanted it must be so. And we have to learn to accept a disease, not to accept, but to come to terms with it. That is also a difference for me. Accepting it means doing noth- ing else, isn’t it? But coming to terms with it means that we simply cannot expect to be able to correct everything, everything. (…) From my point of view, it is irresponsible (caregiver 3). What is irresponsible? (first author ). Drill holes there. That might take millimeters, mil- limeters. Millimeters and then it happened. And then I would have a woman who couldn’t do any- thing anymore, maybe. The risk was also much too big for me.” (caregiver 3). Another patient, who was not offered DBS, nonethe- less was of the same the opinion as caregiver 3. He men- tioned that if he had been offered DBS as a therapeutic option for him, he would have not accepted anyway be- cause of ethical reasons. [Qualitative Research in Medicine & Healthcare 2020; 4:9427] [page 101] Article Figure 4. Caregiver’s 3 perception of the pump. No n- co mm er cia l u se on ly “The brain stimulation was out of question for me also for ethical reasons. I find it delicate when the skull is sawed open.” (patient 1). The other patient who was offered DBS as treatment did not consider the option of being treated with it because she also was too afraid of having a hole in her skull. “I find it grotesque; I mean having a whole board in your head. And it doesn’t last, it doesn’t, it does- n’t make you immune from the results (...). Is it worth it?” (patient 4). (…) Would have been stressful or disturbing for you to be treated with it though? (first author). (…) I can’t imagine what it’s like having a hole in your head. And they shave away your hair and you don’t know whether it’s going to be a success (…) It’s very invasive, isn’t it? (…) I think it’s incredible people are prepared to do that. (…) Would you do it? (patient 4). The four couples who participated in the study ex- plained that they assessed all the options together and jointly made the decision to try with the pump. The three caregivers felt very involved in the decision-making process, and all three patients felt supported by their care- givers. All participants (including the two patients who participated in the study without the participation of a caregiver) expressed satisfaction with the decision taken. Several of them explained that a major reason for deciding on the pump was that it was an easy treatment to stop if it did not work well, whereas stopping DBS would have been more complicated. In addition, before any perfora- tion was made in the wall of the duodenum, the effective- ness of the treatment was tested by inserting a tube through the patient’s mouth temporarily for a few days. This was evaluated positively by patients and caregivers, although one patient felt discomfort after one day and asked for the definitive tube to be inserted through the duodenum. Advantages and disadvantages of the pump All participants expressed having noticed a marked improvement when they started being treated with the pump and a better quality of life. One patient was being treated at the same time with DBS and the pump and it was planned to remove the pump when DBS would start to work properly for the patient. At the time of the inter- view DBS was not controlling the PD symptoms to the level expected by the doctors. The patient was also more satisfied with the improvement achieved with the pump than with DBS, although he acknowledged that a proper management of the PD symptoms was lately also being difficult with the pump as well. Despite this, he considers the pump as a good transition between the pills and DBS, because he could not imagine going from oral treatment straight into DBS. Some of the advantages mentioned by participants were better sleep (without waking up more than once or twice during the night), a better management of PD symptoms than with pills and feeling better during the day. The relief of not having to take pills every three or four hours was shared by both patients and caregivers, as the daily routine revolved around the time the pills needed to be taken. One of the caregivers explained that her husband could eat better now that he didn’t have to take so many pills a day. Two of the caregivers mentioned that the pump calmed their spouses greatly and they were more psychologically stable than when their treatment consisted of oral medica- tion. In one of the cases, the reason for the increased sta- bility was that her husband no longer suffered from sexual addiction caused by oral medication and did not seek sex with prostitutes, which markedly improved the relationship between them. In the other case, the family caregiver ex- plained that her husband had great fluctuations in his mood and suffered from deep depression when he was on oral medication. Once the patient received treatment with the pump, he experienced no ups and downs. However, participants also reported some disadvan- tages they found with the pump as therapy. For example, one caregiver mentioned some disadvantages that were shared by other participants: “You are simply not that free or flexible anymore (…) Yeah, it’s just that you’re just sort of fixated. In theory one could do anything, but I find it too deli- cate. An example is swimming. One can go and cover it with special plasters. The people in charge of the pump say of course, it’s no problem, you can do it. But I am too sceptical. Nowadays in these swimming pools, or, with all these chemicals and chlorine, you don’t know what can happen, so I just don’t trust it. That’s the puncture that goes in, and what if some bacteria get in there. So, you’re just restricted with sports (…) And then with yoga one can’t do some things anymore because otherwise you lie on the pump, don’t you? One can still do ex- ercises, but not many. If the pump were smaller, that would be better. And also, with travelling, you al- ways have to take everything with you. I always have medicine in my handbag, if the pump breaks down, or if he needs an extra dosage. Then I also must always have batteries with me in case the pump runs out of battery.” (caregiver 1) The impossibility of pursuing some activities with the pump, that patients pursued before starting treatment, was described by other participants. For example, patient 5 used to exercise daily with a stationary bike and had to stop doing it when she started to be treated with the pump. Pa- tient 2 mentioned that he had to stop swimming, as previ- ously mentioned by caregiver 1, although he was also told [page 102] [Qualitative Research in Medicine & Healthcare 2020; 4:9427] Article No n- co mm er cia l u se on ly that swimming was possible after removing the pump and tube. However, his reason for stopping swimming was not due the fear to bacterial infections but to the concern that during the time he did not have the pump, he would have PD symptoms that he could not control otherwise. The large size and weight of the pump was also an as- pect regarded as an inconvenience by most of the patients. One of the patients described having to carry such as a large device as feeling like being a kangaroo with a pouch. The pump size and weight were also reasons why some participants had to stop doing certain physical activities like riding a stationary bike or doing yoga. Since doing physical activity is considered by patients to be essential in coping with PD, it poses a problem for them not to be able to do physical activities that they previously did be- cause of the size of the pump. Patient 1 related the large size of the pump to a lack of technological development of the pump by the industry. He considers that the com- panies that make this pump should pay more attention to the daily needs of PD patients. “If you already have such a disease, then you should make your day as easy as possible for the patients and then you are abandoned by the indus- try.” (patient 1). Only one of the patients explained that for him the pump size is not a problem as over time one gets used to it and does not notice it anymore. Finally, diverse accounts regarding the care of the pump were shared. While one caregiver reported that the daily cleaning of the tube con- necting the patient’s duodenum to the pump was very la- borious and time-consuming, another caregiver stated that this task was very bearable for him and was not a burden in his daily life. The two other caregivers explained that they did not take care of the cleaning of the tube because their husbands could still take care of it. In the case of the two women living in a nursing home, the tube cleaning was assumed by the nurses working in the centre. Discussion PD is a very heterogeneous disease not only in the symptoms it can present but also in the progression of the disease over time.21 This heterogeneity can cause prob- lems in diagnosing the disease and also in finding the right treatment for each patient. As PD is a chronic and degen- erative disease, patients try different medical treatments throughout their lives. From a pharmacological point of view, there are a wide variety of oral treatments to manage PD (e.g. Levodopa, dopamine agonist, transferase in- hibitors, anticholinergic medication).1 However, pharma- cological management of PD can be especially challenging when motor complications can no longer be controlled by changes in oral treatment or by combina- tions of drugs. Worsening symptoms due to disease pro- gression or increasing side-effects caused by some oral treatments have an undeniable impact on the quality of life of patients and their families, as the life and daily rou- tines of caregivers is also affected in such situations.22,23 Different aid-device therapies such as DBS or medicine pumps are available if conventional pharmacological treatments fail. These therapies differ in their invasive- ness, side-effect profile, and the need for nursing care and work differently in each patient.24 Our results reveal that the duodopa pump is consid- ered by our participants as a treatment capable of min- imising the signs of PD by facilitating the daily life of patients and their caregivers. Some of the described ad- vantages of this treatment were better management of PD symptoms than with oral treatments, fewer side-effects, increased comfort due to not having to take pills all the time, improved quality of sleep, and greater psychological stability. All this translates into an improved quality of life of patients and caregivers. Nevertheless, some discom- forts in daily life with the pump have also been described: e.g. difficulties in practicing sports like swimming or yoga, the need of greater advanced planning when travel- ling, the discomfort due to the great weight and size of the device, the time consuming task of cleaning the tube connected to the intestine or the feeling of being tied to a device. Nonetheless all participants were satisfied with the choice of being treated with the pump, reducing the size and weight of the device could help to alleviate the above-mentioned discomforts and further improve the quality of life of patients and their caregivers. Further- more, our results show that the pump could be a therapeu- tic alternative for those people who are reluctant to undergo DBS because of insurmountable fear, due to eth- ical reasons or who would not feel comfortable with a de- vice deep in their brains for other reasons. Our study also provides relevant information about the participants’ perception of PD as a disease that elicits het- erogeneous reactions both in the acceptance of the diag- nosis and in coping with the disease in their everyday life.25-27 While some patients received the diagnosis with relief or were not impacted by the diagnosis of PD due to lack of knowledge about the disease and not knowing ex- actly what they were facing, one patient described the di- agnosis as devastating and difficult to accept. What all participants seem to agree on is that PD ended up being a very difficult disease to deal with, which is different for everyone. Therefore, each patient deals with the disease in a different way by establishing different coping strate- gies (e.g. by being active, facing the disease with sense of humour, or thinking positively). The caregivers also need to establish their own coping strategies (e.g. estab- lishing mechanisms like a diary to understand how PD symptoms develop over the course of a day for their loved ones ), because they play a fundamental role in the daily management of the disease.28-30 Our results also show that PD is accounted for as a [Qualitative Research in Medicine & Healthcare 2020; 4:9427] [page 103] Article No n- co mm er cia l u se on ly less bad disease to cope with than other diseases, such as cancer, because it is not considered a terminal disease. This finding seems to contrast with the perception of other chronic patients, who consider that the consequences of Chron’s disease are similarly strong as those of patients with cancer.31 However, other study about the patients’ ex- periences with multiple sclerosis show that the diagnosis of this disease dissipates fears of acute danger, which is comparable to what our participants expressed when con- sidering that PD is not a fatal disease. Another similarity between the perception of our participants with PD and the patients with multiple sclerosis participating in the Reenen et al. study is that the initial relief is challenged by the realization that PD is a disease that progresses and worsens over time.32 This coincides with the difficulty of our participants to cope with the disease progression and its unpredictability that characterises PD both in the man- ifestation of its symptoms and in its progression. Our multimodal approach including narrative semi- structured interviews and drawings allowed us to obtain an additional insight into the participants’ perception of PD and the effects of the pump. The integration of drawings as a complementary methodology to the interviews enabled us to better understand the complexity of the reality that they live in dealing with their disease and its treatment. For in- stance, the use of curves and lines allowed caregiver 3 to represent in Figure 2 the fluctuation of their wife’s symp- toms during the day and the uncertainty it causes due to not knowing what will happen next. Through the use of colour, patient 4 could describe the phenomenon of wearing-off of dopaminergic drugs through different figures in yellow, red and orange tones that overlap each other in Figure 1, and in Figure 4 the contrast between the PD symptoms and the ef- fects of the pump. The body drawn in orange that is jumping reflects her perception of the effects of the pump as contin- uous and unchanging, which not only makes her feel more alive but also more confident, by providing her more control over her illness than the oral medication she was taking be- fore starting the treatment with the pump. This sense of greater control over the disease is also apparent from Figure 4, in which caregiver 3 pictures the pump as a device that figuratively allowed his wife to turn the PD on and off. We decided to adopt a multimodal approach to explore how different modes could make meaning in ways that are creative and that goes beyond the pre-established meaning of words.33,34 Although the use of drawings to explore the way in which people understand and experi- ence different diseases was traditionally limited and mostly confined to the paediatric field,18,35 their use has slowly emerged in the last decade as a valuable tool com- plementing semi-structured interviews for investigating patients’ perceptions about their illness and treatments that are difficult to verbalize.36 For instance, Scott explores the concept of AIDS among women living with HIV adopting a multimodal approach including interviews, free lists and drawings.37 Besser et al., asked their participants to draw during the interviews to have a further insight into how patients view osteoporosis and its consequences in their daily lives, which elicited powerful emotions from the participants.38 Kirkham et al., in their study about experi- ences with chronic pain, used the drawings to explore the participants’ individual representations of their pain, which they consider an invisible condition and difficult to describe with words.39 This description of the pain through the colour coincides with the representation of the motor skills fluctuations typical of PD and the effects of the pump on the improvement of its symptoms depicted by patient 4 through the drawing of body figures in dif- ferent colours. Strengths and study limitations Our study applies the narrative DIPEx approach, which allowed us to explore in-depth the perception of patients and caregivers of PD and the management of it with the pump. To our knowledge, it is the first study ex- ploring their personal experiences with this treatment and its impact on the patients’ and caregivers’ daily life. The perspective of caregivers provided us with valuable infor- mation about PD and the advantages and disadvantages of the pump that patients cannot always offer. Despite the small sample, the study has managed to collect results that reflect different types of circumstances, experiences, im- pressions and opinions of both patients and caregivers. Some participants, both patients and caregivers, experi- enced a beneficial emotional effect for having been lis- tened to and for their personal stories having been taken into account for research. Furthermore, we believe that the use of drawings as complementary methodology to narrative semi-structured interviews elicited the further verbal data because they allowed the participants to reflect on what they have drawn and express these reflections during the interview.16 The possibility of expressing them- selves through drawings also gave them the opportunity to express emotions that would not have been externalised by words alone and facilitated the communication of dif- ficult experiences.40 Therefore, our multimodal research including narrative semi-structured interviews and draw- ings offer additional insights into the participant’s expe- riences not only with the disease but also with the treatment, which led to a more complete data analysis. We identified three possible limitations in our study. Firstly, the participants were only interviewed after being treated with the pump and not before they started receiving this treatment. Although all participants were asked ques- tions about their daily lives and how they were doing before treatment, those who have lived with PD and the pump for a longer period of time may have lost perspective on the before and after. Secondly, we could have missed some negative experiences with the pump due to the fact, that from our experience, people who have had bad experiences are often more reluctant to share their experiences than those who have had positive experiences with the treat- [page 104] [Qualitative Research in Medicine & Healthcare 2020; 4:9427] Article No n- co mm er cia l u se on ly ment. Likewise, the perception of the pump users opting against DBS needs to be further investigated. Thirdly, our findings may be linked to a specific cultural setting, which could make it difficult to extrapolate them to other coun- tries. It would therefore be valuable to carry out this study in other countries so that the results can be compared. Conclusions PD is a complex and heterogeneous disease that changes the lives of patients and their caregivers from the moment of diagnosis affecting all facets of their daily lives.26,29 From a clinical perspective, finding the right treatment for each patient is neither an easy task. Medi- cine pumps are one of the options along with DBS for ad- vanced PD when oral medication stops working. However, the user experience with these medication pumps is largely unexplored. Since the patient’s percep- tion of the chosen therapy may also play a role in the final outcome,41 it is important to better understand how pa- tients, and with their caregivers, perceive medicine pumps and their experiences with these pumps. Therefore, the presented study describes the experiences of a small group of patients and caregivers with PD and the pump. The analysed experiences offer valuable insights into the lived experience of living with PD and being treated with the pump, the caregivers’ perspective of this situation and the reasons that led the participants to choose the pump as therapy over other alternatives. We conducted multimodal research using drawings and narrative semi-structured interviews with the aim of explor- ing the complexity of the individual experience with a chronic degenerative disease treated with a device-aid ther- apy. Drawing give the participants the opportunity to con- vey emotions through the use of colour and shape that give words a new dimension. Furthermore, multimodal research methods allow us, the researchers, to look at the patients’ and caregivers’ experiences from more than one perspec- tive, which offer us the possibility of capturing the sensate aspect of the illness. Therefore, we advocate for more re- search done following a multimodal approach, which en- ables that the illness experience and the process of communicating it to another person are embodied.11 Our findings show that the participants consider PD as an unpredictable disease that changed their lives, while the pump makes the disease more manageable, which im- proves the life quality of patients and caregivers. Some disadvantages of the treatment have also been pointed out by the participants. Where there was most agreement was that the size and weight of the pump was excessive, lim- iting the ability to perform certain activities. Therefore, further technological developments leading to a reduction of the pump size and weight would be desirable. The impression of being dependent on or tied to the device was also reported and hence, future research is needed to explore this topic further. It would also be rel- evant to carry out a comparative study between different countries to assess whether the cultural component plays a role in patients’ and caregivers’ perception of medication pumps. As a final conclusion, our findings show that de- spite the drawbacks, our participants were satisfied with the improvements that the pump has brought to the pa- tients’ physical and mental health. From the perspective of our participants, these improvements could not have been achieved otherwise. References 1. Hayes MT. Parkinson’s Disease and Parkinsonism. Am J Med 2019;132:802-7. 2. Kulisevsky J, Oliveira L, Fox SH. Update in therapeutic strategies for Parkinsonʼs disease: Curr Opin Neurol 2018;31:439-47. 3. Soundy A, Stubbs B, Roskell C. The Experience of Parkin- son’s Disease: A Systematic Review and Meta-Ethnography. Sci World J 2014;2014:613592. 4. Calne SM. The Psychosocial Impact of Late-Stage Parkin- sonʼs Disease. J Neurosci Nurs 2003;35:306-13. 5. 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