Creating and validating a patient-reported outcome instrument for women with vulvar neoplasia after surgical treatment : a mixed-methods project

摘要

In vulvar neoplasia, substantial improvements in therapy and care have been attained during the lastuddecades.1 Nevertheless even minor surgical interventions cause multiple symptoms and side effectsudthat impact women’s quality of life.2 The term ‘vulvar neoplasia’ includes vulvar intraepithelialudneoplasia (VIN) and vulvar cancer.3, 4 Vulvar neoplasia is a rare condition with increasing incidenceudrates in the last 3 decades in Europe and the US.5, 6 The incidence of vulvar neoplasia in Germany andudSwitzerland is about 2 to 7 per 100’000 women per year.7, 8 Surgical treatment is the standard therapyudfor women with vulvar neoplasia.9, 10 The prevalence of surgery related complications, such as woundudinfections, is estimated to be between 5% to 45%.2, 11 Surgery-related complications are one type ofudadverse event and a substantial number of these complications are preventable.12, 13 Surgery-relatedudcomplications in women with vulvar neoplasia result in a variety of physical and psychosocialudproblems,2, 14 and contribute to high health care costs.15, 16 Modified surgical procedures such as theudreplacement of radical vulvectomy by less wide local excision often mean shorter hospitalizations.17udFurthermore oncology care in general is shifting to the outpatient setting.17 Thus, after dischargeudwomen with vulvar neoplasia and surgical treatment are confronted with the need to assess, evaluateudand manage surgery-related symptoms without the support of the inpatient care team.udSymptoms are defined as patient’s perceived changes in biopsychosocial functioning, sensations, orudcognition,18, 19 e.g. bleeding, shame, and fear. A patient’s symptom experience includes 2 commonuddimensions with symptom occurrence, frequency and severity as the cognitive dimension, andudsymptom distress as the emotional dimension.20 Dodd et al.18 developed a generic symptomudmanagement model to understand how patients experience and manage symptoms, to provide healthudcare professionals with direction for selecting clinical interventions, and to inform research. Theudmodel provides a framework for understanding the relationship between aspects of symptomudexperience, symptom management strategies and patient outcomes. In a patient-centred health careudapproach, this model provides appropriate guidance for treatment and care of patients with vulvarudneoplasia. It is crucial to understand symptom experience in order to specify symptom assessmentudstrategies and identify the focus for symptom management.18 udSymptom assessment should be part of the general screening for adverse symptom events. This can beudundertaken by collecting Patient-Reported Outcome (PRO) data.21 A PRO is any report of the patient’sudhealth status that comes directly from the patient, such as his/her symptom experience.22 Theudassessment of both objective and subjective data allows for better supportive care.23udIn clinical practice complications and symptoms after vulvar surgical treatment are an immenseudchallenge for affected women because they are often discharged before their surgical wound hasudhealed. Despite this, for this patient population there is limited research about the prevalence and riskudfactors for different types of short-term wound complications and about women’s experiences duringudthe first 6 months following surgical treatment. Furthermore, no structured symptom assessmentudinstrument is available and there is little guidance for the assessment of symptom experience inudwomen with vulvar neoplasia and surgical treatment. To date, nursing care and research are hamperedudby effective symptom assessment following vulvar surgical treatment. An effective assessment willudallow nurses to better assign management strategies and to be able to evaluate the improvement inudassessed symptoms. With the goal of improving care of women with vulvar neoplasia and surgicaludtreatment, this international, multicenter research project aimed to develop a symptom assessmentudinstrument for patients with vulvar neoplasia who have undergone surgical treatment.udTo address this aim we used a mixed-methods research approach and pragmatism guided this project.24udThis project consisted of a series of quantitative and qualitative studies with equal weighting of bothudmethods using data of more than 180 women recruited from four Swiss (Zurich, Basel, Berne, St.udGallen) and four German University Hospitals (Berlin, Dusseldorf, Freiburg, Munich) from the yearsud1997 to 2011. The first study of the research project was a cross-sectional investigation focusing theudclinical perspective. With a sequential exploratory two-phase strategy, in studies 2 to 5 we intended toudgive voice to the patients’ experiences by the means of a qualitative approach in the first phase. Theirudexperiences were then systematically analyzed in the quantitative component of the project (theudsecond phase).udFirst, we conducted a cross-sectional study on the period prevalence and risk factors for postoperativeudshort-term wound complications in vulvar cancer patients in Switzerland. Data were collectedudretrospectively from medical records from 2007 to 1997 using an investigator-developed dataudcollection instrument to assess risk factors preoperatively and perioperatively, and woundudcomplications that occurred within the first 30 postoperative hospital days. The period prevalence ofudwound complications was 45.4% (49/108), showing at least 1 of 8 assessed wound complications perudpatient within the median hospital duration of 11 days. The period prevalences for the woundudcomplication were: 31.5% for dehiscence, 12% for hematoma, 6.5% for necrosis, 5.6% for infections,ud4.6% for seroma, 1.9% for lymph cysts, 1.9% for malignant wounds, and 0% for disturbed tissueudformation. Two of the 14 risk factors examined, the type surgical therapy (OR, 2.6; 95% confidence interval [CI], 1.34-5.14) and inguinofemoral lymphadenectomy (OR, 3.0; 95% CI, 1.03-8.76) wereudsignificantly associated with short-term wound complications. The results of this retrospective studyudshowed, that the high prevalence of short-term wound complications indicates a need for systematicudwound assessment and early risk management, especially after hemivulvectomy, radical vulvectomy,udand inguinal lymphadenectomy.25udSecond, in order to understand the women’s perspective, we conducted a qualitative study exploringudthe symptom experience in 20 women with vulvar precancer or cancer and surgical treatment in oneudSwiss and two German University Hospitals. Narratives showed eight key themes composing theudessence of women’s symptom experience: delayed diagnosis, disclosed disease, disturbed woman’sudself-image, changed vulva care, experienced wound-related symptoms, evoked emotions, affectedudinterpersonal interactions and feared illness progression. The identified themes were consistent acrossuddifferent surgical procedures. The pattern present in all narratives was that women experienced a lackudof information with regard to the above interrelated themes and that all of them used strategies toudhandle their situation, which affected their distress level. The communication, assessment andudtreatment of symptoms were hampered by the society’s and the health system’s tendency to overlookudthese symptoms and leave them in the realm of the unspeakable. We recommend that health careudprofessionals need new strategies to support these women to recognize, assess and evaluate theudseriousness of symptoms, and to communicate their symptom experience in order to minimizeudpotentially preventable symptom-related distress.26udThird, we focused on the perspective of women with VIN during their illness trajectory by conductinguda secondary data analysis of the qualitative study. Eight narratives showed women’s experiencesudduring their course of illness occurred in five phases: (1) There is something unknown; (2) Oneudknows, what it is; (3) It is treated and should heal; (4) It has effects on daily life; (5) Meanwhile itudworks. These phases showed many women (1) had a late or incorrect diagnosis; (2) despite having auddiagnosis women did not know what they suffered; (3) experienced a high uncertainty in decisionmakingudduring treatment, (4) VIN had an impact on the physical and psychosocial level; and (5) thatudthe women learned over time to deal with their illness. Central for these women during their course ofudillness was a sense of ‘Hope and Fear’. It reflects the fear of recurrence but also the trust in healing.udWomen’s experiences were particularly influenced by the feeling of ‘embarrassment’ and by ‘dealingudwith professionals’. Current care seems to lack adequate support for women with VIN to manage theseudphases. We recommend that new models of counseling and providing information need to beuddeveloped and evaluated.udFourth, a new Patient-Reported Outcome instrument for WOMen with vulvAr Neoplasia (WOMANPRO)udwas developed according to the PRO guidelines and based on literature searches, expertudfeedback (n=9) and patient interviews (n=20). Thirty-seven items were first pilot-tested by patients (n=6) and experts (n=6). The revised 36 items were pilot-tested by patients (n=4). Participants wereudrecruited from one Swiss and two German University Hospitals. To our knowledge, this is the firstudPRO measure designed specifically to assess post-vulvar-surgery symptoms, informational needs andudrelated distress after hospital discharge. The revised WOMAN-PRO showed an excellent item andudscale Content Validity Index (CVI=1.0) and has the potential to support women in recognizing,udassessing, and evaluating the seriousness of post-surgical symptoms and in communicating theirudsymptom experience to health care providers. This can decrease women’s uncertainty about symptomsudand reassure their decision-making about when to seek health care provider evaluation. It can provideudclinical experts with systematic information about key symptoms from a patient perspective, andudwomen’s unmet informational needs.udIn the fifth part of this research project, we identified the occurrence and distress associated with postsurgeryudsymptoms of women with vulvar neoplasia measured with a Patient-Reported Outcomeudinstrument (WOMAN-PRO). The study was a prospective cross-sectional survey conducted in 8udHospitals in Switzerland and Germany. Outpatients (n=54) rated the occurrence of each of 31udsymptoms, and the degree to which the symptoms distressed them. The average number of symptomsudreported per patient was 20 (SD 5.02) with a range of 10 to 31 symptoms on a 0 to 3 scale. The 3 mostudprevalent wound-related symptoms were ‘swelling’ (n=46), ‘drainage’ (n=46) and ‘pain’ (n=43). Theud3 most prevalent difficulties in daily life were ‘sitting’ (n=52), ‘wearing clothes’ (n=48) and ‘carryingudout my daily activities’ (n=43). ‘Tiredness’ (n=51), ‘insecurity’ (n=44) and ‘feeling that my body hasudchanged’ (n=42) were the 3 most prevalent psychosocial symptoms. The most distress symptoms wereud‘sitting’ (Mean 1.98, SD 0.90), ‘carrying out my daily activities’ (Mean 1.79, SD 0.89), and ‘open spotud(e.g. opening of skin or suture)’ (Mean 1.79, SD 0.92), which were on average reported to be ‘quite audbit’ distressing. In this study we also examined the reliability of the instrument using a Cronbach’sudalpha coefficient. For the items representing wound-related symptoms and difficulties in daily lifeudalpha was 0.70, and it was 0.87 for items representing psychosocial symptoms. An alpha of 0.70 orudabove reflects adequate reliability.27 The WOMAN-PRO data (1) show high symptom prevalence anduddistress, (2) call for a comprehensive symptom assessment, (3) may allow identification of areas forudsymptom management. If the results of further psychometric testing are promising, the WOMAN-PROudwill provide an outcome measure for clinical trials.udThe results of the research project contributed to the evidence on women with vulvar neoplasia andudsurgical treatment. First, it added knowledge to support nurses and physicians ability to identifyudpatients at risk for post-surgical wound complications. Second, it provided, for the first time, audconceptual model of symptom experience in affected women. Third, it established, for the first time,udevidence to help understand the VIN patient’s experiences during her illness trajectory. Fourth, theuddevelopment of the WOMAN-PRO instrument with a good content validity has the potential toudcontribute to a valid assessment of symptoms, informational needs and related distress in womenuddiagnosed with vulvar neoplasia and treated surgically. Fifth, WOMAN-PRO data showed a highudsymptom prevalence and distress, and provided preliminary evidence that the WOMAN-PROudinstrument offers a feasible, targeted screening instrument to support systematic symptom assessment.udWe conclude that our research project added to the existing knowledge on complications, symptomsudand associated distress of women with vulvar neoplasia and surgical treatment, confirmed the givenudneed for further research on (a) implementation strategies for comprehensive symptom assessment andud(b) the identification of areas in symptom management to support early recognition of symptoms anduddecrease symptom related distress in women with vulvar neoplasia and surgical treatment.