Table 2 Example of an IPSE study: the lived experience of hand involvement in patients with systemic sclerosis (SSc)
Context | SSc is a rare autoimmune chronic disease characterized by vascular injury, immune dysfunction, and an abnormal fibrotic process that can affect multiple organ systems including the skin, lungs, gastrointestinal tract, and cardiovascular system. Skin is always involved, sometimes with Raynaud syndrome and other frequent forms of hand involvement, associated with various often cumulative processes: joint, cutaneous, microvascular, bone, and nerve abnormalities. The repercussions of hand involvement play a central role in functional limitations and affect quality of life substantially. Existing quantitative research on hand involvement in SSc has mainly evaluated its functional, disabling, and esthetic aspects. These studies are based on preconceptions of how to define and measure hand involvement, preconceptions that do not come from patients’ lived experience. No published research has provided patients with the opportunity to describe their experiences and perspectives about the emotional challenges and areas of distress they face. |
Objective | To explore how patients with SSc experience the effect of their disease on their hands |
Stage 1 | Research group included - two dermatologists specialized in SSc: LV, a professor, and DH, a resident in this specialty; - three researchers expert in qualitative methods (JS, a male psychiatrist, ARL, a female professor of medicine and EM, a female psychologist). The group’s members were highly diverse especially in terms of knowledge, age, and background. The group started the research by meeting a group of patients (N = 5) recruited from LV outpatient practice, in the Caen University Hospital. All of them emphasized the central role of hand involvement in their daily lives. |
Stage 2 | The two clinicians reviewed the qualitative and quantitative literature systematically to confirm the relevance and originality of the study. They verified that no qualitative study had dealt with the specific experience of hand involvement in SSc, although a substantial qualitative literature has looked at the global experience of SSc patients as a whole. To maintain an inductive process, the other group members did not have access to this review until the practical phase of Stage 5. |
Stage 3 | The research group defined the inclusion and exclusion criteria, intended to attain this exemplarity. Inclusion criteria: - Age: 18 or older - Scleroderma diagnosis according to the ACR/EULAR criteria (European League Against Rheumatism, 2013) - Involvement of the hand (Raynaud phenomenon) - Agrees to participate in the research Exclusion criteria: - Age < 18 - Psychiatric disorders or impairments of cognitive function that would prevent a useful interview The group selected an appropriate study site: a multidisciplinary outpatient department, in the Caen University Hospital, where dermatologists, rheumatologists, vascular specialists, rehabilitation specialists, orthopedists, and internists assess and treat patients with SSc, especially those with hand involvement. The University of Paris-Descartes Council for Health Research Ethics Assessment approved the study protocol. All patients provided informed written consent before inclusion. Because patients had several consultations a month, a large amount of clinical and paracliniclal data were available: clinical elements, psychometric assessments, and additional examinations. Social and demographic data, Rodnan scores (of skin thickening), the Cochin Hand Function Scale (measuring functional involvement of the hand), and scores on the specific Scleroderma Health Assessment Questionnaire (SHAQ) QoL scale were collected for all included patients. These data were not to be analyzed but collected to present the clinical characteristics of the sample in detail. We used our purposive sampling strategy and included patients differing by age, social and family status, hand involvement, disease history, and comorbidities, continuously looking for potential heterogeneity. Data saturation was reached with 12 patients, but to ensure the theoretical sufficiency of our results, we included 21, reaching our preferred minimum sample size of 20 participants. |
Stage 4 | A researcher first met each participant to obtain his or her written consent and to collect social and demographic data and any relevant clinical scores. This facilitated the subsequent research interview, a few days later. It was clear that data for this study had to be collected in individual interviews as we wanted to reach an individual lived experience. We were nonetheless aware that patients might find it difficult, even intrusive, to talk about parts of their sick bodies. We therefore chose to provide them with visual narrative support: a photograph of a hand with Raynaud’s phenomenon, a characteristic disabling manifestation in 95% of SSc cases (Fig. 4). Photo-elicitation has not previously been used in any qualitative study of patients with SSc. The interview systematically began by asking participants for their reaction to the photograph. We conducted open-ended interviews, structured by areas to explore, chosen collectively by the group. These areas were selected after a review of two pilot interviews based solely on photo-elicitation: 1. Disease history, onset of the disease (potential question: How long have you had this disorder?) 2. Hand involvement (potential question: Can you tell me how your hands are damaged by this disease?) 3. Daily life (potential question: What bothers you the most on a daily basis?) 4. Emotion (potential question: How does it make you feel when you think about the current state of your hands?) 5. Care (potential question: What was helpful?) Each interview lasted from 45 to 60 min. They were conducted by two experienced researchers, from February 2015 through April 2016. |
Participants | This study included 21 participants: 18 women and 3 men. Their mean age was 60 years at the time of the interviews. 18 had limited cutaneous SSc and 3 diffuse cutaneous SSc. Its effect on their hands were diverse: all had Raynaud syndrome (n = 21), but some also presented joint involvement (n = 10), calcifications (n = 11), sclerosis (n = 15), telangiectases (n = 6), hyperkeratosis (n = 6), digital ulcers (n = 12), digital necrosis (n = 1), and sub/periungual hemorrhages (n = 6). The mean Rodman score was 8.1/51, the mean score on the Cochin Hand Function Scale was 15.6/85, and the mean SHAQ score 1.2/3. |
Stage 5 Structure of Experience | The analysis of the interviews enabled us to identify three central axes of experience: (1) my hands, myself; (2) my hands and others; and (3) treatments and strategies used by the participants. 1. My hands, myself (i) What I see of my disease through my hands: The patients considered the involvement of their hands was visible. They reported changes in the color, shape, and texture of their fingers, most often evoking emotions of rejection or disgust. Many described a major esthetic impact, saying their hands are ugly to look at, again with a strong emotional component. Finally, and especially, the participants considered this visible hand involvement as evidence of their disease and its prognosis, experiencing this damage as a permanent reminder of their illness and a witness of its development and its severity (ii) What I no longer feel: Most participants complained about the loss of sensitivity in their hands. They reported numbness, a feeling of skin tightness, or constantly cold hands. They underlined the daily effect of this loss of sensitivity. Numerous patients also complained about hand pain that handicapped them in their daily lives. (iii) What I can no longer do: The participants underlined what they could no longer do because of the impairment of their hands. All mentioned an impact on their work. Depending on their job, some felt helpless, unable to perform their work duties. Others found it impossible to keep their jobs. Some explained that they continued working, despite the pain. Finally, others directly linked the hand involvement to their difficulties in finding employment. Participants also reported major problems doing housework. Finally, many reported having had to give up hobbies or pleasures that mattered greatly to them, such as gardening, sewing, or playing music. The functional impact was always associated with emotions of anger, frustration, or sadness. 2. My hands and others (i) Others looking at their hands: Most patients considered that this hand involvement exposed their disease to the eyes of others. Some stressed the impossibility of hiding their hands from the sight of others; others mentioned esthetic discomfort that was greatest in social interactions and starting the instant they observed someone looking at their hands. They inferred the negative thoughts of others who looked at their hands. They imagined that these people had to find their hands strange or “dirty” and would worry about the potential risk of contagion. (ii) Interpersonal relationships: All participants reported an important impact on relationships, especially in interactions with those close to them. Many complained that they had to ask others for help every day because of their hand impairments. They reported experiencing unbearable dependence, more unbearable for some than the disease itself. They also reported a feeling of that they could no longer do these things themselves. Some said they accepted help only out of a feeling of resignation, while others preferred to refuse the assistance offered, even if they suffered. Less often, some patients said they were aware of their disability and had no trouble asking for help with some activities. These aspects of assistance and dependence were especially present in the participants’ descriptions of their relationships with those close to them. Some underlined essential support from their family, while others grumbled that their actions and behavior were constantly watched. Some participants found their family’s support inadequate and reported conflicts with them; they felt lonely and misunderstood facing a life encircled by restrictions. Finally, some reported especially what they could no longer do with those they were close to, special moments they could no longer share. Only two mentioned in this regard an effect of their hand impairment on their sex life. 3. Treatments and strategies (i)Perceived efficacy of the treatments prescribed: Many patients reported the direct efficacy of some drugs that made their symptoms disappear; nonetheless, they regretted the transience of their effect or that they could not use them more often. Similarly, surgical treatments were described as effective, helping to relieve the pain. They were sensitive to techniques that permitted functional improvements and judged their efficacy indirectly, by the lack of disease progression. Sometimes they found that neither medication nor physical therapy was effective. Others, however, considered that the treatments’ adverse effects outweighed their positive effects. (ii)Patients’ strategies: the participants reported the strategies that they used to battle hand damage on a daily basis and to preserve some pleasures. They had developed various means, most often material, to overcome these impairments: using a dishtowel to open a bottle, a nutcracker or a screwdriver to open jars, pliers to grasp small objects, etc.). All explained that they wore gloves, in summer as in winter, to prevent Raynaud’s phenomenon. Some complained about the discomfort these gloves engendered. They also described their strategies for maintaining some pleasures, by finding ways to preserve or adapt their hobbies and leisure activities. |
Stage 5 Practical Phase | The structure of lived experience has revealed that the intensity of the functional impact was related to “what I can no longer do” rather than to “what I cannot do.” The question of the visibility of their hand impairment, to themselves and to others, was crucial, with two original aspects revealed by our study: i) the permanent exposure of their disease to the eyes of others — both in social interactions and in more personal relationships — underlines its effect on relationships; ii) it serves as a permanent reminder of the disease to the patients, inducing constant concerns about their survival, their existence. This revealed the importance of other dimensions than functional impairment in the lived experience: esthetic, relational, emotional and existential. Practical implications for healthcare professionals: - PRO development: construction of the HaNDE scale, - Recommendations for clinicians: Clinicians should routinely evaluate hand functions that patients used in ways important to them and have now lost, the impact of the visibility of the disease due to this hand involvement, and should also explore the esthetic, emotional, relational, and existential issues that result. Our results also suggest the need for trust in the patient/physician relationship, to alleviate the patient’s distress in dimensions inaccessible to medication. |
Criteria of Rigor | Feedback to subjects of experience: In the study presented in this paper, we used all of these criteria to ensure the rigor of the analysis and thus the trustworthiness of the results. The feedback of “subjects of the experience” was conducted by presenting the research to a group of patients (N = 20) from the department of internal medicine at Cochin Hospital, a reference center for SSc. These patients all recognized their own experience in the structure we proposed. The question of the choices of the central axis of experience: only two participants mentioned the effect of hand involvement on sexual relations. We included this subject in our results to improve the patients’ lives, because this subject is difficult for patients to raise with doctors. Triangulation, reflexivity, attention to negative cases |
Limitations | 1. It took place in France, and caution is required in transposing our results to other places because medical care depends strongly on the organization of the medical system as well as on the country’s economy. 2. This was a single-center study and all patients recruited in this study were followed in specialized departments (dermatology, rheumatology, internal medicine, and others). It would be interesting to see the results of other studies reproducing this design in other medical settings, such as, for example, that of general medicine. However, we think that this study meets the aim of qualitative research: it may be transferable to other contexts. |