Content Validity of Sjögren’s Syndrome Symptom Diary and Functional Assessment of Chronic Illness Therapy-Fatigue in Patients with Sjögren’s

Patient Characteristics

A total of 12 patients with Sjögren’s were interviewed. All demographic and clinical sampling quotas implemented to promote heterogeneity and representation of the Sjögren’s population (Table 2) were met or exceeded, except for the education level category ‘completed high school or below only’ which was narrowly missed (target: ≥ 3; actual: 2). Patients were predominantly female (n = 8/12; 67%), with a mean age of 56.1 years (range, 20–80 years). Although Sjögren’s is more common in White individuals (hence the racial quota relating to White and non-White) [2], White and non-White individuals were equally represented (n = 6/12; 50%). Most patients (n = 10/12; 83%) were diagnosed with Sjögren’s within the last 10 years and classified as having moderate (n = 5/12; 42%) or high (n = 4/12; 33%) disease activity based on a Physician Global Assessment (PhGA) score. At screening, most patients (n = 9/12; 75%) had an unsatisfactory symptom state (≥ 5 ESSPRI score) [37], reporting symptoms of eye dryness (n = 12/12; 100%), tiredness/fatigue (n = 11/12; 92%), and mouth dryness (n = 8/12; 67%), among others.

Table 2 Demographic and clinical characteristics as reported by patients at screening (N = 12)Physician Characteristics

A total of ten physicians were interviewed, and all sampling quotas were met. Physicians were all rheumatologists from the US (n = 8/10; 80%), Germany (n = 1/10; 10%), and UK (n = 1/10; 10%), and five (n = 5/10; 50%) physicians were female. All physicians had been qualified for at least 10 years, treating patients with Sjögren’s for at least 5 years, and treating patients with Sjögren’s on a weekly (n = 9/10; 90%) or monthly (n = 1/10; 10%) basis at the time of interview. On average, physicians treated at least 20 patients with Sjögren’s per month and worked in a range of settings including academia (n = 8/10; 80%), private practice (n = 3/10; 30%), and/or hospital-based care (n = 2/10; 20%).

SSSDNew and Updated SSSD Items

In general, the new and updated SSSD items were well understood by all patients and most suggested it was easy to provide a response. Specifically, all patients (n = 12/12; 100%) understood the updated ‘tiredness’ item and 24-h recall period (n = 8/8; 100%), and most considered the item relevant to both their overall Sjögren’s experience and their experience of Sjögren’s in the past 24 h (n = 11/12; 92%). The 0–10 NRS was generally reported as an appropriate response scale (n = 11/12; 92%), and all patients suggested that it would be easy to remember their tiredness over the 24-h recall period (n = 12/12; 100%). All patients (n = 12/12; 100%) demonstrated a good understanding of the new ‘most bothersome’ and ‘most important to improve’ symptom items and most suggested it would be easy to choose a response for these items using the options provided (n = 10/12; 83%, and n = 10/10; 100%, respectively). All patients selected the same symptom as their most bothersome and most important to improve, with eye dryness selected the most frequently (n = 7/12; 58%). However, a third of patients (n = 4/12; 33%) reported a different symptom as their most bothersome, compared to the symptom they rated as most severe at the time of interview.

Meaningful Change on Individual SSSD Items

Meaningful change was explored in relation to patients’ 0–10 NRS score on each individual SSSD item at the time of interview. All patients (n = 12/12; 100%) reported that a two-point improvement on the eye dryness item would be important to them (Fig. 1). Similarly, most patients who experienced mouth dryness and muscle/joint pain reported that a two-point improvement would be meaningful to them (n = 7/9; 78%, range, 2–4 points and n = 7/11; 64%, range, 1–6 points, respectively). For the skin dryness and tiredness items, perceptions of a meaningful improvement varied, ranging from 1–6 to 1–5 points, respectively. Finally, of the three (n = 3/8; 38%) female patients who experienced genital dryness, two (n = 2/3; 67%) reported that a two-point improvement would be important to them (range, 2–5 points). The minimum amount of improvement that would be worth taking a treatment for was also explored during the patient interviews. Responses varied across patients, ranging from 1 to 6 points on the eye dryness and tiredness items, 1–5 points on the mouth dryness and muscle/joint pain items, 2–6 points on the skin dryness item, and 2–5 points on the genital dryness item.

Fig. 1figure 1

Meaningful improvement on individual SSSD items

Patients were also asked about meaningful worsening on each item. For the eye dryness item, all patients who were asked (n = 11/11; 100%) reported that worsening by two points would be important. Similarly, most patients suggested that a two-point worsening would be important on the mouth dryness (n = 9/12; 75%, range, 1–5 points), skin dryness (n = 6/12; 50%, range, 1–3 points), and tiredness (n = 9/12; 75%, range, 1–3 points) items. However, responses were more varied for the pain and genital dryness items, ranging from 1–8 to 1–7 points, respectively.

Meaningful Change on SSSD Total Score

Most patients reported that a two-point (n = 6/12; 50%) or one-point (n = 4/12; 33%) improvement in their total SSSD score would be meaningful (range, 1–7 points), mostly as it would improve how they feel (n = 6/10; 60%; Table 3). Patients also discussed the symptoms that would be most important to see improvement in for the change to be meaningful (Table 3). Eye dryness was reported most frequently (n = 7/12; 58%), followed by muscle/joint pain (n = 3/12; 25%), skin dryness (n = 1/12; 8%), and tiredness (n = 1/12; 8%). Of note, these findings were in line with patients’ responses to the ‘most important to improve’ item.

Table 3 Impact of improvement in SSSD total score on how patients feel/function and most important symptom to see change in

Most patients who were asked reported that a two-point (n = 5/11; 45%) or one-point (n = 3/11; 27%) improvement in their SSSD total score would be worth taking a treatment for (range: 1–4 points). Similarly, most patients who were asked reported that a one-point (n = 3/11; 27%) or two-point (n = 5/11; 45%) worsening would be important (range: 1–7 points).

Best Approach to Calculating an SSSD Score

Most physicians suggested that the most appropriate scoring approach would be tracking changes in individual SSSD item scores (n = 7/10; 70%), followed by tracking average total scores (n = 4/10; 40%) or tracking domain scores (n = 1/10; 10%). Of those who felt tracking changes in individual item scores would be optimal, three (n = 3/7; 43%) explained that patients’ experiences of symptoms assessed by SSSD vary greatly so should be tracked separately, and that improvements in individual symptoms may be masked by a total score including symptoms which are not relevant to their experience/may remain unchanged. Additionally, physicians noted that some symptoms may be influenced by environmental factors (i.e., skin dryness might be dependent on use of artificial heaters or lack of moisturizer use) so capturing individual data is important for monitoring long-term improvement (n = 2/7; 29%).

Interestingly, two physicians (n = 2/10; 20%) felt that a hybrid approach of tracking both individual item scores and average total score would be optimal. Both physicians felt it important to include an individual symptom score, given that improvement in one symptom does not equate to improvement in another, while acknowledging the importance of an overall total score, particularly in monitoring improvement longitudinally.

FACIT-F

Due to interview time constraints, FACIT-F was debriefed with n = 11/12 (92%) patients.

Understanding

FACIT-F items were generally well understood by patients (n ≥ 7/11; ≥ 64%, Fig. 2). However, some patients reported that they did not understand the term ‘listless’ (n = 4/11; 36%) in Item 3 (‘I feel listless [washed out]’), with one stating that they “have never actually heard that word before”. Of note, one of these patients was able to infer the meaning of this item from the term ‘washed out’, hence the total number of patients not understanding this item was three (n = 3/11; 27%).

Fig. 2figure 2

Overall understanding for each FACIT-F item

Due to interview time constraints, it was not possible to assess patients’ understanding of the response options and recall period for each individual FACIT-F item. However, as the response options and recall period are consistent across FACIT-F items, it was deemed sufficient to explore patients’ understanding in relation to Item 1 (‘Fatigue’) only. All patients who were asked demonstrated a good understanding of the FACIT-F response options (n = 11/11; 100%) and 7-day recall period (n = 6/6; 100%). Appropriateness and ease of the response options and recall period were also assessed more generally, considering all FACIT-F items. All patients who were asked reported that the response options were appropriate (n = 10/10; 100%) and most reported that it was easy to answer the questions using the response options (n = 4/6; 67%). All patients who were asked (n = 8/8; 100%) also reported that it was easy to remember their experience of fatigue over the 7-day recall period.

Relevance

Most FACIT-F items were relevant to most patients (Fig. 3). However, Item 10 (‘I am too tired to eat’) was not relevant to any patients (n = 0/11; 0%), and Item 2 (‘I feel weak all over’), Item 3 (‘I feel listless [washed out]’), and Item 11 (‘I need help doing my usual activities’) were relevant to less than half of patients (n = 5/11; 45%, n = 3/11; 27%, and n = 4/11; 36%, respectively).

Fig. 3figure 3

Overall relevance for each FACIT-F item

Throughout the interviews, four patients (n = 4/11; 36%) noted that it was difficult to distinguish whether their experiences of fatigue, as explored by FACIT-F, were caused by Sjögren’s or other reasons (i.e., age, co-morbid conditions, and/or general tiredness). Specifically, difficulties were raised in relation to Item 1 (‘fatigue’; n = 1/4; 25%), Item 2 (‘weak all over’; n = 1/4; 25%), Item 5 (‘trouble starting things’; n = 2/4; 50%), Item 6 (‘trouble finishing things’; n = 1/4; 25%) and Item 11 (‘need help doing usual activities’; n = 1/4; 25%).

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