The proposed work complies with ethical principles in human studies. A data collection notebook was developed for the pregnant women with RD and without RD, which contained information about Sociodemographic variables, General Clinical Data, Current and Previous Obstetric History, and also 8 PROM’s (BASDAI, BASFI, EQ5D, EVA, FACIT, HAQ, SF-36 and HADS) that were acquired on the online platform REUMA.pt.
For 5 months (since October of 2019 and February of 2020), and after obtaining informed consent, the previous document was filled out by a sample of 20 low-risk pregnant women without RD, who attended the Maternal-Fetal Outpatient Clinic at Hospital Santa Maria, Centro Hospitalar Universitário Lisboa Norte EPE, and by a sample of 20 pregnant women with RD who resorted to the Multidisciplinary Rheumatology and Obstetrics consultation at the same department. Rheumatic
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inflammatory diseases included systemic lupus erythematosus, rheumatoid arthritis, Sjogren’s syndrome, ankylosing spondylitis, phospholipid antibody syndrome, Still’s disease, and various vasculitis as Takayasu disease, Cogan syndrome and Behçet’s disease.
Initially we considered delivering the SF-36 PROM by email and/or mail to be completed by patients at home. However, it was later inserted in the data collection notebook at the beginning of the collection of questionnaires in person because we realized it did not consume much time to complete along with the other PROM’s and patients actually preferred this approach. For that reason, only 34 women filled it.
In order to compare the group of Pregnant Women without RD with the group of Pregnant Women with RD, we proceeded to the calculation, descriptive statistics for variables divided into 5 domains: Sociodemographic and General Clinical Data, Current and Previous Obstetric History, Physical Function and Fatigue, Quality of life related with General Health and Mental Health.
Statistic data was obtained using the SPSS software (IBM SPSS Statistics for Windows 26.0). For categorical variables (Sociodemographic, General Clinical Data, Current and Previous Obstetric History, HAQ support activities and instruments, Anxiety and Depression) we calculated proportions and applied the Chi-Square homogeneity test to compare proportions of the different groups. When more than 80%
of cells had expected frequencies ≤ 4 cells we used the Fisher Exact Test. For Age, Menarche Age and Physical Function, Fatigue, Quality of life related with General Health and Mental Health Outcomes we calculated means, standard deviations, and applied the t-Student two-samples test to assess differences in the two groups.
The 8 PROM’s that were used are describe bellow:
3.1. BASDAI (Bath Ankylosing Spondylitis Disease Activity Index) (Appendix 3) BASDAI is a specific instrument that measures patient-reported disease activity in patients with ankylosing spondylitis. It contains 6 items such as back pain, fatigue, peripheral joint pain and swelling, localized tenderness, and the duration and severity of morning stiffness, scaled (0–10) or visual analogue scale (0–10 cm) anchored by adjectival descriptors “none” and “very severe.” Duration of morning stiffness is anchored by a time scale (0–2 or more hours)26,27.
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The scores for the last 2 questions (severity and duration of morning stiffness) are averaged, and then the result is averaged with the remaining 4 question scores to provide a final score out of 10. The final score varies from 0 (no disease activity) to 10 (maximal disease activity). A cut off of 4 is used to define active disease26–29.
The BASDAI has been the most frequently used measure of disease activity in clinical trials30–32 and is recommended to assess response to anti–tumor necrosis factor therapies in ankylosing spondylitis patients26,27. It also has been used to analyse the disease course of patients with ankylosing spondylitis during and after pregnancy33.
3.2. BASFI (Bath Ankylosing Spondylitis Functional Index) (Appendix 4)
BASFI is a specific instrument used to define and monitor physical functioning in patients with ankylosing spondylitis. It includes 10 items, with 8 items assessing activities such as bending, reaching, changing position, standing, turning, and climbing steps, and the 2 other items assess the patients’ ability to cope with everyday life.
Numeric response scale (0–10) or visual analogue scale (0–10 cm) anchored by adjectival descriptors “easy” and “impossible”29–31.
The mean of the individual items scores is calculated to provide the overall score. Score range is from 0 (no functional impairments) to 10 (maximal impairment)29,34. It is the most widely used functional index for assessment of ankylosing spondylitis patients, primarily in studies of disease impact20,35 and in clinical trials30,31.
3.3. EQ5D (EuroQol – 5 dimensions) (Appendix 5)
It is a standard instrument developed by EuroQol Group, that can measure health status. And so, it provides an easy, generic measure of health for clinical and economic assessment. It includes the following 5 dimensions: mobility, self-care, usual activities, pain/discomfort, and anxiety/depression. Each dimension has 3 levels: no problems, some problems, extreme problems36. One of the ways to present the results of EQ‐5D‐
3L is by a quality of life (QoL) score, obtained from a coefficient calculus, an interval scale where “1” represents “full health” and “0” represents “dead”. However, a negative value is possible when some severe health states are considered worse than being dead3738.
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It has been used to evaluate the quality of life of pregnant women with a full-term birth from the first trimester to the 9th month, comparing physiological, simple or complex pathological pregnancies39.
3.4. EVA/VAS (Visual Analogue Scale) (Appendix 6)
It consists of a line with the extremities defining “no pain at all” (0) and “pain as worse as it could be” (10). The patient is expected to mark his pain level on the line between the two extremities40. The distance between the extremity “0” and the mark, defines the subject’s pain score. Scores less than 3.4 indicate “mild pain”, between 3.5-6.7 “moderate pain” and higher than 3.5-6.7 “severe pain”40. This tool was first used in psychology by Freyd in 192341 and it is used in DAS 28 to access patients with rheumatoid arthritis.
3.5. FACIT (Functional Assessment of Chronic Illness Therapy) (Appendix 7) FACIT is an instrument designed to assess fatigue/ tiredness and its impact on daily activities and functioning in several chronic diseases. It includes items such as weakness, tiredness, lack of energy, listlessness. This scale (previously called the Functional Assessment of Cancer Therapy-Fatigue (FACT-F)) was originally developed to assess cancer-related fatigue. More recently this instrument is been used validly in numerous chronic conditions such as systemic lupus erythematosus, rheumatoid arthritis and psoriatic arthritis42.
Responses of “not at all”(0); “a little”(1); “somewhat”(2); “quite a bit”(3); and
“very much”(4) are presented for each question43. All items are then summed to create a single fatigue score with a range from 0 to 52. Items can be reverse scored when appropriate to provide a scale in which higher scores represent better functioning or less fatigue42.
3.6. HAQ (Health Assessment Questionnaire) (Appendix 8)
It is controversial, but some authors say that it should be considered a generic instrument.44 It is one of the most widely used comprehensive, validated, patient-oriented outcome assessment instruments44,45. The maximum scores in each category are summed (0–24) and divided by the number of categories scored to generate a score from 0–3. The value of the score can be interpreted in terms of three categories: from 0
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to 1: mild difficulties to moderate disability; from 1 to 2: disability moderate to severe and from 2 to 3: severe to very severe disability46.
It was used to ascertain the influence of pregnancy on disease activity in women with rheumatoid arthritis during pregnancy and postpartum47. And now, it has been most extensively utilized and validated in rheumatoid arthritis43, for assessing physical function in rheumatoid arthritis trials44,45.
3.7. SF-36 (Short Form Health Survey 36 Item) (Appendix 9)
It is a generic instrument, applicable to healthy individuals or any health condition, with the aim of measuring and evaluating the health status of populations and individuals with or without disease, monitor patients with multiple conditions and compare patients with different conditions and compare the health status of patients with that of the general populations.
Composed of 36 items, grouped into 8 dimensions: Physical Function, Physical Performance, Pain, General Health, Vitality, Social Function, Emotional Performance and Mental Health. Scores by dimensions are presented on a positive orientation scale from 0% (worst health) to 100% (best health)48–50.
For the validation of this scale in the Portuguese version, a sample of 930 pregnant or postpartum women was used51. It has also been applied in a study to evaluate the quality of sleep in pregnant women49 and study the patient’s experience of the disease course, and HRQoL during and after pregnancy in women with rheumatoid arthritis and ankylosing spondylitis1.
3.8. HADS (Hospital Anxiety and Depression Scale) (Appendix 10)
Consists of 2 subscales designed to identify and quantify depression and anxiety in physically ill patients52. Is a questionnaire composed of statements relevant to either generalised anxiety (questions number 1, 3, 5, 7, 9, 11 and 13) or depression (questions number 2, 4, 6, 8, 10, 12 and 14).
Each question is answered on a four-point (0–3) response category and so the scores varied between 0 to 21 for anxiety and for depression. An analysis of scores on the two subscales allows provision of information that a score of 0 to 7 for either subscale could be regarded as being in the normal range, a score of 11 or higher indicating probable presence of the mood disorder and a score of 8 to 10 being just
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suggestive of the presence of the respective state. Further work indicated that the two subscales, anxiety and depression, were independent measures53.
A recent review of the literature on the validity of the HADS distinctly indicates that it is a well-performed questionnaire when assessing the severity of the anxiety and depression disorders symptoms in both somatic, psychiatric and primary care patients and even in the general population54. It has been used to investigate the occurrence of symptoms of depression and anxiety in early and late pregnancy55.