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

In vulvar neoplasia, substantial improvements in therapy and care have been attained during the last
decades.1 Nevertheless even minor surgical interventions cause multiple symptoms and side effects
that impact women’s quality of life.2 The term ‘vulvar neoplasia’ includes vulvar intraepithelial
neoplasia (VIN) and vulvar cancer.3, 4 Vulvar neoplasia is a rare condition with increasing incidence
rates in the last 3 decades in Europe and the US.5, 6 The incidence of vulvar neoplasia in Germany and
Switzerland is about 2 to 7 per 100’000 women per year.7, 8 Surgical treatment is the standard therapy
for women with vulvar neoplasia.9, 10 The prevalence of surgery related complications, such as wound
infections, is estimated to be between 5% to 45%.2, 11 Surgery-related complications are one type of
adverse event and a substantial number of these complications are preventable.12, 13 Surgery-related
complications in women with vulvar neoplasia result in a variety of physical and psychosocial
problems,2, 14 and contribute to high health care costs.15, 16 Modified surgical procedures such as the
replacement of radical vulvectomy by less wide local excision often mean shorter hospitalizations.17
Furthermore oncology care in general is shifting to the outpatient setting.17 Thus, after discharge
women with vulvar neoplasia and surgical treatment are confronted with the need to assess, evaluate
and manage surgery-related symptoms without the support of the inpatient care team.
Symptoms are defined as patient’s perceived changes in biopsychosocial functioning, sensations, or
cognition,18, 19 e.g. bleeding, shame, and fear. A patient’s symptom experience includes 2 common
dimensions with symptom occurrence, frequency and severity as the cognitive dimension, and
symptom distress as the emotional dimension.20 Dodd et al.18 developed a generic symptom
management model to understand how patients experience and manage symptoms, to provide health
care professionals with direction for selecting clinical interventions, and to inform research. The
model provides a framework for understanding the relationship between aspects of symptom
experience, symptom management strategies and patient outcomes. In a patient-centred health care
approach, this model provides appropriate guidance for treatment and care of patients with vulvar
neoplasia. It is crucial to understand symptom experience in order to specify symptom assessment
strategies and identify the focus for symptom management.18
Symptom assessment should be part of the general screening for adverse symptom events. This can be
undertaken by collecting Patient-Reported Outcome (PRO) data.21 A PRO is any report of the patient’s
health status that comes directly from the patient, such as his/her symptom experience.22 The
assessment of both objective and subjective data allows for better supportive care.23
In clinical practice complications and symptoms after vulvar surgical treatment are an immense
challenge for affected women because they are often discharged before their surgical wound has
healed. Despite this, for this patient population there is limited research about the prevalence and risk
factors for different types of short-term wound complications and about women’s experiences during
the first 6 months following surgical treatment. Furthermore, no structured symptom assessment
instrument is available and there is little guidance for the assessment of symptom experience in
women with vulvar neoplasia and surgical treatment. To date, nursing care and research are hampered
by effective symptom assessment following vulvar surgical treatment. An effective assessment will
allow nurses to better assign management strategies and to be able to evaluate the improvement in
assessed symptoms. With the goal of improving care of women with vulvar neoplasia and surgical
treatment, this international, multicenter research project aimed to develop a symptom assessment
instrument for patients with vulvar neoplasia who have undergone surgical treatment.
To address this aim we used a mixed-methods research approach and pragmatism guided this project.24
This project consisted of a series of quantitative and qualitative studies with equal weighting of both
methods using data of more than 180 women recruited from four Swiss (Zurich, Basel, Berne, St.
Gallen) and four German University Hospitals (Berlin, Dusseldorf, Freiburg, Munich) from the years
1997 to 2011. The first study of the research project was a cross-sectional investigation focusing the
clinical perspective. With a sequential exploratory two-phase strategy, in studies 2 to 5 we intended to
give voice to the patients’ experiences by the means of a qualitative approach in the first phase. Their
experiences were then systematically analyzed in the quantitative component of the project (the
second phase).
First, we conducted a cross-sectional study on the period prevalence and risk factors for postoperative
short-term wound complications in vulvar cancer patients in Switzerland. Data were collected
retrospectively from medical records from 2007 to 1997 using an investigator-developed data
collection instrument to assess risk factors preoperatively and perioperatively, and wound
complications that occurred within the first 30 postoperative hospital days. The period prevalence of
wound complications was 45.4% (49/108), showing at least 1 of 8 assessed wound complications per
patient within the median hospital duration of 11 days. The period prevalences for the wound
complication were: 31.5% for dehiscence, 12% for hematoma, 6.5% for necrosis, 5.6% for infections,
4.6% for seroma, 1.9% for lymph cysts, 1.9% for malignant wounds, and 0% for disturbed tissue
formation. 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) were
significantly associated with short-term wound complications. The results of this retrospective study
showed, that the high prevalence of short-term wound complications indicates a need for systematic
wound assessment and early risk management, especially after hemivulvectomy, radical vulvectomy,
and inguinal lymphadenectomy.25
Second, in order to understand the women’s perspective, we conducted a qualitative study exploring
the symptom experience in 20 women with vulvar precancer or cancer and surgical treatment in one
Swiss and two German University Hospitals. Narratives showed eight key themes composing the
essence of women’s symptom experience: delayed diagnosis, disclosed disease, disturbed woman’s
self-image, changed vulva care, experienced wound-related symptoms, evoked emotions, affected
interpersonal interactions and feared illness progression. The identified themes were consistent across
different surgical procedures. The pattern present in all narratives was that women experienced a lack
of information with regard to the above interrelated themes and that all of them used strategies to
handle their situation, which affected their distress level. The communication, assessment and
treatment of symptoms were hampered by the society’s and the health system’s tendency to overlook
these symptoms and leave them in the realm of the unspeakable. We recommend that health care
professionals need new strategies to support these women to recognize, assess and evaluate the
seriousness of symptoms, and to communicate their symptom experience in order to minimize
potentially preventable symptom-related distress.26
Third, we focused on the perspective of women with VIN during their illness trajectory by conducting
a secondary data analysis of the qualitative study. Eight narratives showed women’s experiences
during their course of illness occurred in five phases: (1) There is something unknown; (2) One
knows, what it is; (3) It is treated and should heal; (4) It has effects on daily life; (5) Meanwhile it
works. These phases showed many women (1) had a late or incorrect diagnosis; (2) despite having a
diagnosis women did not know what they suffered; (3) experienced a high uncertainty in decisionmaking
during treatment, (4) VIN had an impact on the physical and psychosocial level; and (5) that
the women learned over time to deal with their illness. Central for these women during their course of
illness was a sense of ‘Hope and Fear’. It reflects the fear of recurrence but also the trust in healing.
Women’s experiences were particularly influenced by the feeling of ‘embarrassment’ and by ‘dealing
with professionals’. Current care seems to lack adequate support for women with VIN to manage these
phases. We recommend that new models of counseling and providing information need to be
developed and evaluated.
Fourth, a new Patient-Reported Outcome instrument for WOMen with vulvAr Neoplasia (WOMANPRO)
was developed according to the PRO guidelines and based on literature searches, expert
feedback (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 were
recruited from one Swiss and two German University Hospitals. To our knowledge, this is the first
PRO measure designed specifically to assess post-vulvar-surgery symptoms, informational needs and
related distress after hospital discharge. The revised WOMAN-PRO showed an excellent item and
scale Content Validity Index (CVI=1.0) and has the potential to support women in recognizing,
assessing, and evaluating the seriousness of post-surgical symptoms and in communicating their
symptom experience to health care providers. This can decrease women’s uncertainty about symptoms
and reassure their decision-making about when to seek health care provider evaluation. It can provide
clinical experts with systematic information about key symptoms from a patient perspective, and
women’s unmet informational needs.
In the fifth part of this research project, we identified the occurrence and distress associated with postsurgery
symptoms of women with vulvar neoplasia measured with a Patient-Reported Outcome
instrument (WOMAN-PRO). The study was a prospective cross-sectional survey conducted in 8
Hospitals in Switzerland and Germany. Outpatients (n=54) rated the occurrence of each of 31
symptoms, and the degree to which the symptoms distressed them. The average number of symptoms
reported per patient was 20 (SD 5.02) with a range of 10 to 31 symptoms on a 0 to 3 scale. The 3 most
prevalent wound-related symptoms were ‘swelling’ (n=46), ‘drainage’ (n=46) and ‘pain’ (n=43). The
3 most prevalent difficulties in daily life were ‘sitting’ (n=52), ‘wearing clothes’ (n=48) and ‘carrying
out my daily activities’ (n=43). ‘Tiredness’ (n=51), ‘insecurity’ (n=44) and ‘feeling that my body has
changed’ (n=42) were the 3 most prevalent psychosocial symptoms. The most distress symptoms were
‘sitting’ (Mean 1.98, SD 0.90), ‘carrying out my daily activities’ (Mean 1.79, SD 0.89), and ‘open spot
(e.g. opening of skin or suture)’ (Mean 1.79, SD 0.92), which were on average reported to be ‘quite a
bit’ distressing. In this study we also examined the reliability of the instrument using a Cronbach’s
alpha coefficient. For the items representing wound-related symptoms and difficulties in daily life
alpha was 0.70, and it was 0.87 for items representing psychosocial symptoms. An alpha of 0.70 or
above reflects adequate reliability.27 The WOMAN-PRO data (1) show high symptom prevalence and
distress, (2) call for a comprehensive symptom assessment, (3) may allow identification of areas for
symptom management. If the results of further psychometric testing are promising, the WOMAN-PRO
will provide an outcome measure for clinical trials.
The results of the research project contributed to the evidence on women with vulvar neoplasia and
surgical treatment. First, it added knowledge to support nurses and physicians ability to identify
patients at risk for post-surgical wound complications. Second, it provided, for the first time, a
conceptual model of symptom experience in affected women. Third, it established, for the first time,
evidence to help understand the VIN patient’s experiences during her illness trajectory. Fourth, the
development of the WOMAN-PRO instrument with a good content validity has the potential to
contribute to a valid assessment of symptoms, informational needs and related distress in women
diagnosed with vulvar neoplasia and treated surgically. Fifth, WOMAN-PRO data showed a high
symptom prevalence and distress, and provided preliminary evidence that the WOMAN-PRO
instrument offers a feasible, targeted screening instrument to support systematic symptom assessment.
We conclude that our research project added to the existing knowledge on complications, symptoms
and associated distress of women with vulvar neoplasia and surgical treatment, confirmed the given
need for further research on (a) implementation strategies for comprehensive symptom assessment and
(b) the identification of areas in symptom management to support early recognition of symptoms and
decrease symptom related distress in women with vulvar neoplasia and surgical treatment.