Laying the Groundwork: A Pilot Study Exploring Improved Endometriosis Management Strategies in Primary Healthcare

Endometriosis is an estrogen-dependent, benign, chronic inflammatory disease, characterised by endometrial-like tissue outside of the uterus. In Sweden, the diagnosis has been used for approximately 50 years. 1 , 2 Symptoms are multiple and diverse: dysmenorrhea, dyspareunia, pain in the abdomen often in the pelvic area, irregular bleeding, cystitis-like problems, bowel problems, infertility, depressive symptoms, feelings of ill health, tiredness, lack of energy and chronic pain. 3 , 4 Approximately 60% of the women suffering from endometriosis have symptoms. Symptoms vary depending on the organ that is affected. The correlation between the extent of the endometriosis and the amount of pain experienced by the patient is not strong. 4 Endometriosis has many differential diagnoses, including gynaecological problems such as myoma, polyps, infections, misplaced intra uterine device (IUD), but also lactose intolerance, Irritable Bowel Syndrome (IBS), Inflammatory Bowel Disease (IBD), chronic obstipation, colitis, depression, psychosocial problems and abuse, and musculoskeletal problems (disc hernia, fibromyalgia), thus making it difficult to diagnose. 4 Several explanatory models exist concerning the disease, and the pathogenesis is not conclusive. There is a high risk of contracting endometriosis if a first-degree relative suffers from it, indicating a hereditary factor. One theory is that there are different mechanisms/causes depending on where the endometriosis is located. 4 Among all women in childbearing age, about 2–10% suffer from endometriosis to a lesser or more serious extent. 4 , 5 In Sweden, that corresponds to approximately 200000 women Further, 25–70% of all women with menstrual and pelvic pain and 20–50% of all infertile women suffer from endometriosis. Some women with endometriosis never experience any symptoms (~20%). 4 Due to diagnostic difficulties, women with endometriosis usually have symptoms for many years (6–12 years) before receiving a diagnosis. 4 , 6 , 7 Some women with longstanding symptoms lack diagnosis. 4 , 6 , 7 Among younger women (<20 years), endometriosis is often underdiagnosed and time until diagnosis is longer compared to older women. 4 , 7 When the menstrual periods end at menopause, many women experience that the symptoms decrease and even disappear. 8 Several studies have shown that women with endometriosis have reduced quality of life, and that this is strongly associated with the amount of pain. Depression is common among patients with endometriosis and even more so among those women with both endometriosis and chronic pain. The time from debut of symptoms to diagnosis and the intensity of pain both correlate with decreased inability to work. 9 Except for microscopic analysis of a biopsy, there are no laboratory or other diagnostic methods that with certainty can identify endometriosis. The diagnosis is frequently made on the basis of symptom constellation, clinical findings, and hormonal treatment results, ie, as an ex juvantibus diagnosis. The diagnosis can be secured only after surgery and biopsy. Because the symptoms are so unspecific and the diagnostic process tends to be extended, it is often a forgotten diagnosis. 4 , 10 We have not identified any fully validated, symptom-based, patient-reported questionnaires for endometriosis. 11 There are several problems and challenges regarding the disease. In addition to the major problem of diagnostic difficulty, finding treatment is a challenge, and it is often difficult to ultimately retain good pain relief. Women with endometriosis cannot be cured, but only relieved from symptoms. 4 In Sweden, primary care centres (PCCs) are the first line of health care and most patients see a general practitioner (GP) as their first encounter with health care. GPs’ knowledge about endometriosis is limited, and this can be one of the reasons why they do not recognise the symptoms that may be caused by endometriosis, leading to diagnostic delay. 12–15 Current electronic applications concerning endometriosis are all targeting a patient already diagnosed with endometriosis, providing them with tools such as an endometriosis pain diary. 16 Educational interventions to increase the level of knowledge among staff members regarding specific diagnoses or treatments have been studied, and the results differ. Some interventions show increase of knowledge, and some do not. 17 , 18 Perhaps the key is not only to focus on the staff, but also on the patients seeking care for several clusters of rather unspecified symptoms. We set out to study if it is possible to enhance the general level of knowledge about endometriosis among staff at PCCs, and thereby facilitate identification of women with increased risk of having endometriosis. Our hypothesis was that this would make it possible to reach a diagnosis earlier and thus reduce suffering. To facilitate continuity in the risk identification and pedagogic process, a patient questionnaire/application targeting risk of having endometriosis was included as part of the intervention. Our hypothesis was that this could further increase staff awareness of endometriosis. Therefore, we aim to. First: To evaluate if a brief educational intervention for primary health care staff regarding endometriosis gives sustainable knowledge of endometriosis symptoms. Second: To evaluate if a brief questionnaire for women seeking care for common symptoms, aiming at increasing continuous awareness regarding endometriosis, is feasible in the Swedish primary care context.

This was conducted as a pilot study A pilot study was performed in the primary care context at three PCCs, strategically sampled regarding location (representing urban, rural and a mix between), number of patients listed and number of staff. An invitation letter explaining the different parts of the pilot study was sent to the PCCs (n=3, one public, two privately run). The pilot study was divided into two parts. The first part explored PCC staff knowledge of endometriosis at baseline and three months after participation in a brief information workshop at the PCC to evaluate if the educational intervention for primary health care staff regarding endometriosis gave sustainable knowledge of endometriosis symptoms. The second part evaluated if a brief questionnaire for women seeking care for common symptoms was feasible in the Swedish primary care context. Part I, staff education: A questionnaire consisting of six short questions testing knowledge about endometriosis was developed ( Appendix 1 ). Each question was coded as either correct (3 points), semi-correct (2 points), wrong (1point), or no answer (0 p). During a lunch meeting at the PCC, the staff were invited to fill in a written questionnaire. The questionnaire was then submitted to the study personnel before the correct answers were presented. After the questionnaire, a short lecture was given about endometriosis that took 40 minutes in total, with time for the staff to ask questions at the end. Three months after the first questionnaire and lecture, a follow up at the PCC was conducted, and the same questionnaire was distributed among the staff members in order to examine knowledge about endometriosis after 3 months, compared to baseline. Part II, patient questionnaire and patients’ perceptions of patient questionnaire: A patient questionnaire was developed consisting of 16 questions: 2 questions concerning age and number of children, and 14 questions based on known endometriosis signs/symptoms according to findings derived from a population study of women. 19 Two study nurses spent a week in the waiting room at each of the PCCs to test the patient questionnaire ( Appendix 2 ). The study personnel asked all female patients aged ~14-50 (in fertile age) in the waiting room if they wanted to participate. Those willing to participate received a written questionnaire to fill in. After having filled in the questionnaire, the patients also answered four questions concerning their perceptions about the adequacy, understandability, acceptability and feasibility of the questionnaire ( Appendix 2 ). All questions, both those regarding endometriosis signs/symptoms and those regarding the questionnaire, had dichotomous response options (YES/NO). Some questions had extra space for the participant to write comments if needed. Our intention was to collect 10 patient questionnaires at each PCC. Questionnaire for staff members at baseline and after 3 months. Patient questionnaire and patients’ perceptions of patient questionnaire at baseline. Statistical significance was accepted at p<0.05. Standard statistical methods were used for descriptive statistics. Continuous variables were analysed by independent-samples t -test and categorical variables or frequencies by Pearson chi-square test. In order to more easily compare questionnaires between baseline and the 3-month follow-up, we created an index of the total scores of all the questions and used this in the analyses. To compare the total index between baseline and 3-months follow-up for the three PCCs, we created a line graph. The statistical analyses were made using statistical software SPSS, version 25. The patients were not involved in setting the research questions or design, their contribution will help form the questionnaire to identify endometriosis in primary health care, used in the future. The study has been approved by the Regional Ethical Review Board in Gothenburg, Sweden (Dnr 733–18). All participants, both staff members and female patients in the waiting room, were provided with written and oral information and signed a written informed consent. This study was performed in alignment with the Declaration of Helsinki.

Three PCCs participated in this pilot study, and 43 staff members completed the knowledge questionnaire at baseline and 55 after 3 months. 106 patient participants completed the patient questionnaire. The primary care centres consisted of 1 publicly run PCC with ~ 9000 listed patients, 1 private urban with ~ 8000 listed patients, and 1 private/urban (city) PCC with ~9500 listed patients with multi-ethnic origin. The endometriosis questionnaire and lecture were conducted and completed at the PCCs with all staff available on that particular day - see Figure 1 for number of participants and Table 1 for participants’ professions. Table 1 Staff Composition for All PCCs, Showing Number of Individuals by Profession Among Those Who Responded to the Questionnaire at Baseline and After Three Months, Respectively Occupation Questionnaire Baseline Questionnaire 3 Months General practitioner 10 15 Registered nurse 15 20 Administrator 2 6 Psychologist/Curator 2 4 Occupational therapist/Physiotherapist 5 4 Assistant nurse 3 4 Student 2 1 Total 39 54 Did not state 4 1 Total all 43 55 Figure 1 Flow chart. Data collection at three PCCs from fall 2018 to spring 2019. Staff Composition for All PCCs, Showing Number of Individuals by Profession Among Those Who Responded to the Questionnaire at Baseline and After Three Months, Respectively Flow chart. Data collection at three PCCs from fall 2018 to spring 2019. The number of participating health professionals was higher at all PCCs after three months. The mean knowledge level about endometriosis at the three PCCs, both separately and in total, at baseline and after 3 months is presented in Figure 2 . The result shows sustained and improved levels of knowledge after 3 months at all 3 sites. Figure 2 Mean total score on endometriosis questionnaire by staff for the three participating PCCs at baseline and after 3 months, and total scores for all three PCCs combined (right). The outcome is presented in numbers based on the entire staff’s scores. There was an increase in scores at PCC 1 and PCC 3 at 3 months compared to baseline, but no increase at PCC 2. Mean total score on endometriosis questionnaire by staff for the three participating PCCs at baseline and after 3 months, and total scores for all three PCCs combined (right). The outcome is presented in numbers based on the entire staff’s scores. There was an increase in scores at PCC 1 and PCC 3 at 3 months compared to baseline, but no increase at PCC 2. The patient questionnaire was answered by 106 patients, but one questionnaire was excluded because the patient’s sex at birth was male, resulting in 105 questionnaires. Of the women, 73% had regular menstrual periods, 60% experienced pain in their stomach during their menstruation, 46% needed pain killers during menstruation, and 11% used other medicines besides pain killers for menstrual pain. Around 70% had ever been pregnant, and 5% reported infertility. The response rate for patient questions including questions concerning the construction of the questionnaire is shown in Table 2 . Table 2 Waiting Room Survey at the 3 PCCs, Patient Questionnaire Items and Frequency of Yes and No Answers. N=105 Question n Yes No Missing % Are your periods regular? 99 73.3% 21.0%  0 Have you ever been pregnant? 105 71.4% 28.6%  0 Infertility 105 4.8% 93.3% 1.9% Do you feel depressed? 105 32.4% 64.8% 2.9% Do you suffer from a general feeling of illness? 105 46.7% 52.4% 1.0% (Fatigue, lethargy, nausea)       Do you feel easily irritated? 105 38.1% 61.0% 1.0% Do you suffer from pain in the body? 105 33.3% 65.7% 1.0% Do you suffer from stomachache? 105 33.3% 64.8% 1.9% Do you have pain in your stomach During menstruation? 105 60.0% 35.2% 4.8% Do you need painkillers during Menstruation? 105 45.7% 49.5 4.8% Do you have any medicine for Menstrual pain besides painkillers? 105 11.4% 84.8% 3.8% Do you stay at home from work/school During your menstruation? 105 13.3% 83.8 2.9% Do you have trouble defecating? 105 22.9% 76.2 1.0% Do you have urinary tract/bladder problems? 105 16.2% 82.9% 1.0% Do you experience pain during intercourse? 105 10.5% 85.7% 3.8% Waiting Room Survey at the 3 PCCs, Patient Questionnaire Items and Frequency of Yes and No Answers. N=105 The mean age of the women who answered the patient questionnaire was 34.6 (range 13–58, SD 8.5) years. The majority (63%) had higher education, while 43% had middle and 5% lower education. Of the women, an overwhelming majority did not find that the questionnaire was difficult to complete, that the questions were complicated, or that the questions were perceived as being too sensitive to answer, and 91% understood all the questions ( Table 3 ). Table 3 Evaluation of Feasibility of the Patient Waiting Room Questionnaire in the Swedish Primary Care Context. Distribution of Patients’ Answers (n=105) Questionnaire questions n Yes n (%) No n (%) Missing n (%) Was the questionnaire difficult to complete? 105 5 (4.8) 99 (94.3) 1 (1) Were the questions complicated? 105 1 (1) 104 (99)  0 Were the questions sensitive to answer? 105 7 (6.7) 98 (93.3)  0 Did you understand all the questions? 105 96 (91.4) 9 (8.6)  0 Evaluation of Feasibility of the Patient Waiting Room Questionnaire in the Swedish Primary Care Context. Distribution of Patients’ Answers (n=105)

This study is conducted with real patients seeking treatment at primary health centres The methodology is easy replicated The study is limited to three primary health care centres There is a need for further development of the education and questionnaire This study helps to highlight and enhance the knowledge about an often forgotten diagnosis that has a large impact on women’s overall health This study is conducted with real patients seeking treatment at primary health centres The methodology is easy replicated The study is limited to three primary health care centres There is a need for further development of the education and questionnaire This study helps to highlight and enhance the knowledge about an often forgotten diagnosis that has a large impact on women’s overall health

The intervention with a questionnaire and lecture about endometriosis had a positive effect on general knowledge among the staff at the participating primary care centres after 3 months, albeit we do not know how long this positive effect will last. We could not see any negative effects among the participating centres. The patient questionnaire was feasible and easy to understand. These findings could be important for the development of an educational program together with an electronic application based on the patient questionnaire, so that the staff at the primary care centres will have more knowledge about endometriosis and thus be able to identify women with the disease earlier.

In this study that evaluated the effect of a brief educational intervention to staff at PCCs regarding endometriosis and the feasibility of a patient questionnaire, a higher mean level of knowledge about endometriosis was shown among the participating centres at follow up, indicating a remaining effect of the workshop education after 3 months. The patients’ perceptions of the questionnaire indicated adequacy, understandability, acceptability, and feasibility. Our method seems to be a successful way to enhance knowledge about endometriosis on the primary care level. Quibel 13 showed that general practitioners’ knowledge about endometriosis is limited, with possible direct consequences for the delay of the diagnosis. Further, van der Zanden 12 emphasised the need for educational programmes and awareness strategies for first-line medical professionals to be able to shorten the time to diagnosis for women with endometriosis. One of the most important ways to diagnose endometriosis is to ensure that the medical professional be on the alert for endometriosis when a woman seeks medical help for abdominal pain. 20 As endometriosis cannot be cured but only relieved, 4 it is important to start available treatment according to guidelines as soon as possible. There is no need to wait for a gynaecological referral to start treatment, when the GPs, midwives and nurses in primary care suspect endometriosis in their patients. When diagnosis, treatment and relief can be made at an early onset of disease, the gap of inequity can be reduced and the concept of primary care be strengthened for all. 21–23 Patients perceived that the patient questionnaire was easy to complete, was not complicated, did not contain sensitive questions and was easy to understand. These findings suggest that the patient questionnaire is feasible and applicable and may potentially be offered as an electronic application in the future. This is well in line with suggestions by Field 24 and Fung. 25 Both concluded that patient questionnaires can be an important part of clinical practice and help health care professionals to make decisions for individual patients, including targeted interventions that will improve patients’ care outcome. The present study did not evaluate if the women who answered the questionnaires had endometriosis or not. However, when evaluating the answers given, we could estimate that many women had abdominal pain of a degree that affected their lives.