Navigating the diagnostic journey of epidermolysis bullosa - a qualitative study on the lived experiences and needs of parents and patients.

BACKGROUND: Epidermolysis bullosa (EB) concerns a heterogeneous group of rare genetic skin fragility disorders that result in chronic blistering and wounding. EB significantly impacts the daily lives of patients with EB, as well as their families. While advances in diagnostics are improving the speed and accuracy of EB diagnosis, little is known about the experiences and needs of parents and patients throughout their diagnostic journey. OBJECTIVES: In this qualitative study, we explore the parent and patient perspective on the EB diagnostic trajectory to gain an in-depth understanding of their lived experiences and needs. METHODS: Participants were parents of paediatric patients (n=18), and adult patients (n=8) recruited from the Dutch EB Registry. After purposive sampling, they participated in semi-structured interviews via video-calls to discuss their personal diagnostic trajectory and the subsequent impact of EB diagnosis on their (family) life. Applying a constructivist approach, a reflexive thematic analysis was executed to facilitate a dynamic and iterative process, involving inductive open coding of transcripts and constant comparison of data. RESULTS: Ten major themes were developed, representing three distinct groups: (i) parents of children with JEB and RDEB, (ii) parents of children with EBS and DDEB, and (iii) adult patients with localized EB. The EB diagnostic process appeared to have a diversity of emotional consequences, varying from desperation and uncertainty about the future, to clarification and confirmation. The urgent need for a timely diagnosis and accurate prognosis was emphasized, particularly by parents of children with extensive presentation. Both parents and patients expressed shortcomings in clinical practice, with severity ratings in current EB disease terminology in particular seeming to have an adverse impact on illness perception, healthcare-seeking behaviour, research participation, and engagement in peer support. CONCLUSIONS: This study describes the lived experience and needs of parents of children and adult patients during the diagnostic process of EB. We show an urgent need to accelerate diagnostics and imply that the EB community should continue working towards ever faster diagnosis, public awareness, and education. While guiding patients along the diagnostic journey, clinicians should focus their support strategies on tailored medical communication, while refraining from value-connoted wording.

The Working Alliance Inventory's Measurement Properties: A Systematic Review.

Measurement properties of the Working Alliance Inventory (WAI) and its various translations and adaptations for specific target groups have been investigated for over 30 years. No systematic review analyzing studies on measurement properties of the WAI has been conducted to date. COnsensus-based Standards for the selection of health Measurements INstruments (COSMIN) were developed for conducting high-quality systematic reviews on measurement properties in a transparent and standardized way. Aim of this study was to systematically review studies on measurement properties of the WAI, and its adapted versions, within psychotherapy, and other healthcare contexts using COSMIN criteria. PsycINFO, Medline, and EMBASE were searched (1989-2021). In all phases of the review procedure, study selection, data extraction, risk of bias assessment, rating of the quality of measurement properties, and rating of the quality of evidence for measurement properties, disagreement between reviewers was resolved by discussion. Results on validity, internal structure, reliability, construct validity, and responsiveness were analyzed. In total 66 studies were included. In most studies, evidence for measurement properties was according to COSMIN criteria, insufficient, lacking, or conflicting. Content validity was rated insufficient because neither patients nor healthcare professionals were involved in the development and validation process. Hence evidence for content validity of the WAI is unknown. Conflicting evidence was found for structural validity. Evidence for internal consistency could not be established. Limited evidence was found for inter-rater reliability and convergent validity. Conflicting evidence was also found for test-retest reliability and divergent validity. COSMIN criteria exposed persistent problems in validation studies of the WAI. These findings may indicate that measurement properties of the WAI are not up to current standards, or that COSMIN criteria may be less appropriate for assessing measurement properties of the WAI, or it could indicate both. The results of this systematic review suggest that WAI outcomes should be interpreted with caution and further research is needed regarding the content validity and hypotheses development. For the future, the theoretical framework underlying the measurement of the working alliance needs to be studied in psychotherapy and other health contexts, and tested in methodologically sound studies. Systematic Review Registration: https://www.crd.york.ac.uk/PROSPERO/, identifier CRD42019051428.

Participants' unspoken thoughts and feelings negatively influence the therapeutic alliance; a qualitative study in a multidisciplinary pain rehabilitation setting.

PURPOSE: This study aimed to explore factors that influence participants' perceptions of the therapeutic alliance with healthcare professionals; their participation in the alliance; and their commitment to treatment in a multidisciplinary pain rehabilitation setting. MATERIALS AND METHODS: A qualitative research-design was used and 26 participants in a multidisciplinary pain rehabilitation program were interviewed in-depth. RESULTS: Initially, participants reported to be satisfied with their healthcare professionals. After deeper reflection on the therapeutic alliance, several unspoken thoughts and feelings and relational ruptures emerged. Almost all participants mentioned a history of disappointing and fragmented healthcare, and they reported on how this affected their cognitions, perceptions, and beliefs about the current program. Participants felt insufficiently empowered to voice their concerns and regularly chose to avoid confrontation by not discussing their feelings. They felt a lack of ownership of their problems and did not experience the program as person-centered. CONCLUSIONS: Several factors were found that negatively influence the quality of therapeutic alliance (agreement on bond) and efficacy of the treatment plan (agreement on goals and tasks). To improve outcomes of pain rehabilitation, healthcare professionals should systematically take into account the perceptions and needs of participants, and focus more on personalized collaboration throughout the program offered.Implications for rehabilitationDifferences in perceptions and experiences of pain, together with differences in beliefs about the causes of pain, negatively influence the therapeutic alliance.When participants and healthcare professionals operate from different paradigms, it is important that they negotiate these differences.From the perspective of participants, a clear-cut organization of healthcare that encourages collaboration is required.It is important to focus on personalized collaboration from the start and during treatment, and to recognize and discuss disagreement on diagnosis and treatment plans.During this collaboration, healthcare professionals should systematically take into account the perceptions and needs of the participants.

Between the Lines: A Qualitative Phenomenological Analysis of the Therapeutic Alliance in Pediatric Physical Therapy.

Aim: To explore perceptions and preferences of children, parents, and physical therapists regarding the therapeutic alliance in pediatric physical therapy in a rehabilitation setting.Methods: Qualitative phenomenological analysis of interviews with children (n = 10), their parents (n = 10), and physical therapists (n = 10).Results: Three themes were identified: importance of trust in the physical therapist, transparency in sharing information, and negotiation concerning goals and tasks of treatment. Parents considered trust in the therapist's relational skills of greater importance to the therapeutic alliance than the therapist's technical skills. Although the physical therapists showed a strong willingness to meet the needs of children and parents, they seemed unaware of the emotional impact of positional inequality and the differences in roles and tasks of children, parents, and therapists during the treatment.Conclusion: All participants emphasized the importance of the quality of the therapeutic alliance. Nevertheless, positional inequality and differences in roles and tasks appeared to influence negotiation about goals and tasks of treatment. Children and parents are in a dependent relationship with the physical therapist. Physical therapists are challenged to find the right balance between their professional position and input on the one hand, and the emotional needs of child and parents on the other hand.

Identifying Patients with COPD in Need for Psychosocial Care Through Screening with the HSCL-25 and the CCQ Mental State.

High levels of psychological distress are documented in patients with COPD. This study investigates the extent to which patients with a high score on the Hopkins Symptoms Checklist-25 (HSCL-25) or with a high score on the Mental State scale of the Clinical COPD Questionnaire (CCQ) endorse a need for psychosocial care, and investigates several characteristics of patients with a need. Outpatients with COPD of the Department of Pulmonary Diseases of a University Medical Center were assessed with the HSCL-25, CCQ and a question on need for psychosocial care. For patients indicating a need, the percentage of patients with HSCL-25 ≥39 was compared with the percentage of patients with CCQ Mental State >2 and tested with a Chi-square. In total 323 patients participated; 57% of them were distressed according to the HSCL-25 (≥39) and 20% according to the CCQ Mental State (>2); 28% reported a need for psychosocial care. For patients reporting a need for psychosocial care a higher percentage was identified by the HSCL-25 than by the CCQ Mental State (χ2 = 9.41, p <. 002) and they were younger than patients without a need (t = 4.48, p <. 001). No differences existed for sex, FEV1, FEV1% predicted or medical comorbidities. The HSCL-25 identified more patients in need than the CCQ Mental State scale. However, not all patients with a need were identified. No relationship was found between need for psychosocial care and illness variables or comorbidities. Distress screening is questioned as the most effective way to identity patients with COPD in need for psychosocial care.

Is implementing screening for distress an efficient means to recruit patients to a psychological intervention trial?

OBJECTIVES: Psychological interventions show greater efficacy when evaluated with distressed patients. We report on the feasibility of implementing screening for recruiting distressed cancer patients to a randomized controlled trial of problem-solving therapy (PST), characteristics associated with enrolment, and time investment and challenges of implementing screening. METHODS: Three medical settings implemented screening of patients, directly after cancer treatment (T1) and 2 months later (T2), using Hopkins Symptom Checklist-25 and one question about need for services. Distressed patients indicating need for services were interviewed. Eligible patients were offered the possibility to participate in the trial. Consenting patients were randomized to PST or waitlist. RESULTS: At T1, 366 of 970 screened patients (37%) scored above the cutoff and at T2, 208 of 689 screened patients (30%). At either or both T1 and T2, 423 patients reported distress, of whom 215 indicated need for services. Only 36 (4% of 970) patients consented to trial participation. Twenty-seven patients needed to be screened to recruit a single patient, with 17 h required for each patient recruited. Barriers to screening were time constraints and negative attitudes of oncology staff towards screening. CONCLUSIONS: Implementing screening proved inefficient for recruiting distressed cancer patients post-treatment to a randomized controlled trial on PST, with need for services being much less than anticipated. Consecutively screening patients did not result in a sample representative of the larger pool of distressed patients, which may lower generalizability. An adequately powered intervention trial using screening requires a feasibility study establishing recruitment rates and dedicated, funded staff assistance.

Lower levels of trust in one's physician is associated with more distress over time in more anxiously attached individuals with cancer.

OBJECTIVE: In the present study, we investigated individual differences in the outcome of patient-physician trust when confronted with cancer from an attachment theoretical perspective. We expected that lower levels of trust are associated with more emotional distress and more physical limitations within the first 15 months after diagnosis, especially in those who score relatively high on attachment anxiety. No such association was expected for more avoidantly attached individuals. METHOD: A group of 119 patients with different types of cancer (breast, cervical, intestinal and prostate) completed questionnaires concerning trust (short version of the Wake Forest Physician Trust Scale) and attachment (Experiences in Close Relationship scale Revised) at 3 months after diagnosis. Emotional distress (Hospital Anxiety and Depression Scale) and physical limitations (physical functioning subscales of the European Organization for Research and Treatment of Cancer, Quality of Life Questionnaire-C30) were assessed at 3, 9 and 15 months after diagnosis. To test the hypotheses, multiple hierarchical regression analyses were performed. RESULTS: Lower levels of trust were associated with more emotional distress and more physical limitations at 3, 9 and 15 months after diagnosis in more anxiously attached patients, but not in less anxiously attached patients. DISCUSSION: These results indicate an attachment-dependent effect of trust in one's physician. Explanations and clinical implications are discussed.

Do patients trust their physician? The role of attachment style in the patient-physician relationship within one year after a cancer diagnosis.

BACKGROUND: The degree of trust in and satisfaction with the physician has been shown to have important implications for treatment outcomes. This study aims to examine individual differences in patients' trust, satisfaction and general distress from an attachment theoretical perspective. MATERIAL AND METHODS: One hundred and thirty recently diagnosed cancer patients of three medical hospitals were extensively interviewed by trained psychologists to assess attachment style. Patients completed standardized questionnaires three and nine months after diagnosis to assess trust, satisfaction and distress. t-tests and repeated measures ANOVAs were used to examine differences between securely and insecurely attached patients and changes over time. A mediation model based on a bootstrapping method was used to examine whether trust mediated between attachment and satisfaction, and attachment and distress. RESULTS: Insecurely attached patients (N = 45, 35%) reported less trust in and satisfaction with their physician, and reported more general distress than securely attached patients three and nine months after diagnosis (p < 0.05). Trust and distress levels did not change over time. Trust mediated between attachment and satisfaction, but not between attachment and distress. CONCLUSION: Insecurely attached patients trusted their physician less than securely attached patients, and in turn were less satisfied with their physician. Their higher levels of general distress were not related to their lower levels of trust. Attachment theory provides a framework to interpret differences in patients' trust, satisfaction and distress, and may help physicians respond in such a way that their patients feel secure, which in turn is expected to result in better health outcomes.

The concept and measurement of meaning in life in Dutch cancer patients.

We investigated the psychometric properties of a Dutch translation of the Personal Meaning Profile in a heterogeneous group of cancer patients. Our study resulted in a relatively short scale consisting of 39 of the 57 original items, divided into 5 factors, labeled 'relation with God'; 'dedication to life'; 'fairness of life'; 'goal-orientedness' and 'relations with other people', which can be summed to a total score of the experience of meaning in life. The internal consistency of the total scale as well as of its sub dimensions was high. The experience of meaning in life was positively related to feelings of psychological well-being and negatively to feelings of distress. Furthermore, the experience of meaning in life was also related to trait-like characteristics as personality. Future research can investigate its appropriateness for other populations than cancer patients, and if and how the experience of meaning in life eventually changes as a result of existential threats.

Psychometric properties of the Dutch version of the posttraumatic growth inventory among cancer patients.

In the current study, we investigated the psychometric properties of a Dutch translation of the posttraumatic growth inventory in a heterogeneous group of cancer patients. Its original five-factor structure was maintained. The internal consistency of the total scale, as well as its subdimensions, was satisfactory. As expected, the experience of posttraumatic growth was positively related to: emotional expression about the illness, openness to experience, and feelings of innerness. Furthermore, the scale appeared to be sensitive for the demographics age and gender. The experience of posttraumatic growth was not related to negative feelings such as avoidance, anxiety, depression, and neuroticism. Our Dutch translation of the instrument appeared to be a sound measure for the experience of posttraumatic growth in cancer patients.

Male sexuality after cancer treatment--needs for information and support: testicular cancer compared to malignant lymphoma.

Testicular cancer (TC) as well as malignant lymphoma (ML), both have nowadays an excellent prognosis. However, both types of cancer may be diagnosed at young adulthood and patients may experience sexual concerns. In this article the need for information and support concerning sexuality will be explored, and the traumatic impact of cancer diagnosis with respect to this will be considered. A total of 264 patients with testicular cancer, median age 36 (S.D. 9.7) years, and 50 patients with malignant lymphoma, median age 42 (S.D. 11.7) years returned a questionnaire concerning sexual functioning; four items assessed the need for information or support concerning sexuality, at diagnosis and at follow-up. It appeared that more than half of the patients with testicular cancer reported a lack of information and support concerning sexuality during treatment; 67% of them still had a need for information at follow-up. These rates were significantly lower for patients with malignant lymphoma. Especially patients with testicular cancer who suffered sexual dysfunction reported extremely high needs for information and support. According to these findings it can be concluded that more attention should be paid to the doctor-patient communication with respect to sexual concerns in general, and especially where it concerns patients with testicular cancer.