Abstract
Objective.
The health effects of SAPHO syndrome and chronic nonbacterial osteomyelitis (CNO) have not been well studied. We assessed health-related quality life (HRQOL) in adults with SAPHO-CNO and performed a review of EQ-5D outcomes amongst similar chronic rheumatic and inflammatory diseases.
Methods.
We enrolled patients in the first US-based SAPHO-CNO prospective registry and assessed their HRQOL using the EQ-5D index and EQ-5D VAS. A focused scoping review was performed of EQ-5D related outcomes amongst disease cohorts with phenotypic similarities to SAPHO-CNO.
Results.
In the 138 SAPHO-CNO participants, mean age (standard deviation) was 37.7 (15.0) years with a mean disease duration of 6.4 (6.2) years. All subjects had musculoskeletal involvement (44% having spine involvement) and 48% had skin involvement. Mean EQ-5D-5L index value was 0.75 (0.16) and mean EQ-VAS was 63.0 (21.0). Most patients were impacted by pain/discomfort (86%), difficulty in carrying out usual activities (65%) and anxiety/depression (54%); over one-third reported problems in all five EQ-5D domains. These SAPHO-CNO adults had similarly reduced EQ-5D and VAS values, and similar high proportion of perceived problems, as other inflammatory disease cohorts.
Conclusion.
The HRQOL among adults with SAPHO-CNO is low, with more than half of patients experiencing an impact in perceived pain, performing usual activities, and feelings of anxiety or depression. Future therapies should target the multi-dimensional nature of SAPHO-CNO, targeting HRQOL as a major outcome.
Key Indexing Terms: SAPHO syndrome, chronic recurrent multifocal osteomyelitis, health-related quality of life, cohort study, scoping review
INTRODUCTION.
Chronic nonbacterial osteomyelitis (CNO), an umbrella term that includes SAPHO (synovitis, acne, pustulosis, hyperostosis, and osteitis) syndrome and chronic recurrent multifocal osteomyelitis (CRMO), is a painful and disabling autoinflammatory disease primarily targeting bones, joints and skin.1,2 Untreated, severe sterile inflammation of multiple bones can lead to irreversible damage, chronic pain and disability in more than 30% of SAPHO-CNO patients.3 Additionally, a significant delay in diagnosis is commonly observed in both pediatric and adult cohorts.3–5 Despite the availability of recent expert consensus recommendations for the treatment of CNO, there are no FDA-approved therapies for adults with SAPHO-CNO.6 A significant gap remains in our knowledge of the etiology, disease prognosis, drug effectiveness and outcomes in SAPHO-CNO. Thus, there is a need to improve our understanding of SAPHO-CNO to improve patient outcomes.
Children and adults with SAPHO-CNO suffer from decreased quality of life (QOL), although there is very little information quantifying this impact, particularly in adults, with only a few published studies. Children with long-standing CRMO reported impairments in all QOL domains measured by the Short Form 36 (SF-36) questionnaire.7 Additionally, children with CNO and their families experienced a significant impact on relationships, school, work, and well-being.4 Adults with sternocostoclavicular hyperostosis (SCCH), a spectrum of SAPHO-CNO, reported worse functioning and physical and mental health scores compared to healthy controls.8 Additionally, a small survey study of adults with SAPHO in Germany reported limitations in all SF-36 QOL dimensions, and a recent larger study in adults with SCCH/CNO showed impairments in nearly all SF-36 domains compared to the Dutch population, highlighting low QOL in adults with SAPHO-CNO.5,9 Thus, HRQOL is an important core area impacting individuals living with SAPHO-CNO across the age spectrum.10
Measures of general health and health-related QOL (HRQOL) assess an individual’s perceived health status and overall physical and emotional well-being.11 A widely used modern measure of HRQOL in chronic conditions and rheumatic diseases is the EQ-5D, which assesses health in five key domains.11,12 The EQ-5D has well-established psychometric properties, and has been used successfully in clinical trials, health survey and outcomes research in a broad range of rheumatic conditions.11,13,14 Although comprehensive catalogues of EQ-5D values have been assembled for common rheumatic diseases, measures of HRQOL with EQ-5D are lacking in rarer chronic rheumatic and autoinflammatory diseases such as SAPHO-CNO.13,15,16
In the current study, we aimed to measure HRQOL with the EQ-5D-5L instrument in adults from the baseline visit of the SAPHO-CNO Study, a prospective longitudinal US-based cohort study. We wished to contextualize these results, and thus performed a scoping review of contemporary literature of EQ-5D in chronic rheumatic and inflammatory disease (CRID) cohorts. Our overarching goal was to highlight the importance of reduced HRQOL in adult SAPHO-CNO amongst relevant CRIDs.
METHODS.
The Adult SAPHO-CNO Study: Design and Study Population.
The SAPHO-CNO Study (SCS) is a novel prospective longitudinal observational study with accompanying biospecimen collection designed to propel our understanding of the longitudinal course of SAPHO-CNO in adults. The SCS, established in October 2021 at the University of Iowa, has been enrolling all consecutive adults (18 years of age and older) who are diagnosed with SAPHO-CNO, as defined by the Benhamou and/or Kahn criteria, and who are able to provide informed consent (Supplementary Table 1).17,18 Exclusion criteria include infectious osteitis, malignant bone conditions, and non-inflammatory bone lesions.
We used cross-sectional data from the baseline visit of the SCS from all enrolled subjects who completed their study visit between October 2021 and January 2025. At baseline, subjects undergo a comprehensive clinical evaluation by rheumatologists (the core clinician investigator members of the research team). Participants complete comprehensive questionnaires on demographics, symptoms, treatments, and validated patient-reported outcome (PRO) measures on key QOL and disease impact domains informed by our ongoing qualitative work with SAPHO-CNO patients and experts.10 For this study, we extracted subject information on age, sex at birth, self-reported ethnicity and race, disease characteristics, current medications, comorbidities, patient-reported global assessments of disease activity in SAPHO-CNO (overall, MSK, skin) by the numeric rating scale (NRS, ranging from 0 well controlled to 10 poorly controlled), and patient-reported QOL as measured by the EQ-5D-5L instrument. This study has received approval from the University of Iowa IRB (HawkIRB; IRB ID# 202107229) through the single site IRB (SMART IRB) reliance mechanism required by the National Institutes of Health.
EQ-5D-5L.
Subjects self-completed the latest version of the EQ-5D instrument (EQ-5D-5L) at the baseline visit, which assesses health in five dimensions (mobility, self-care, usual activities, pain/discomfort, anxiety/depression).11,12,19 Each dimension in the EQ-5D-5L instrument has five levels of response: no problems, slight problems, moderate problems, severe problems, and unable to do (for mobility, self-care, usual activities) or extreme problems (for pain/discomfort and anxiety/depression). The EQ-5D index value reflects the subject’s health state profile, based on societal preference weights for the health state, and ranges from negative values (where 0 is the health state equivalent to death) to 1 (the value of full health), with higher values indicating better HRQOL. In this study, we used the United States value set to calculate the EQ-5D-5L index values. The EQ-5D VAS scoring is based on a vertical VAS ranging from 0 (worst health imaginable) to 100 (best health imaginable), with higher values indicating better health.11,12
Data Analysis (SAPHO-CNO Cohort).
We calculated descriptive statistics for the cross-sectional data on adults with SAPHO-CNO from the SCS. For continuous variables (i.e., age, disease duration), we calculated means and standard deviations; categorical variables are presented as counts and proportions. Exploratory correlational analyses were performed between EQ-5D scores and disease features. All analyses were performed with SAS (version 9.4).
Scoping Review of EQ-5D Literature in Chronic Rheumatic and Inflammatory Diseases.
To describe the current landscape, we performed a focused scoping literature review on the use of EQ-5D for assessing HRQOL over the last 5 years. We included CRIDs to contextualize our findings more broadly amongst disease cohorts given the small amount of data in adult SAPHO-CNO and HRQOL.
Our initial systematic search in Pubmed on EQ-5D and SAPHO-CNO, including wide terminology used for this disease, did not yield any articles for inclusion in the study (Supplementary Table 2).20 Next, we performed the following broad systematic search of publications indexed to PubMed: 1) Timeframe: limited to the last 5 years (from 1/1/2020 to 1/1/2025) to be reflective of our adult SAPHO-CNO cohort; 2) Measure for HRQOL: EQ-5D instrument; 3) Diseases of interest: CRIDs with phenotypic similarity to SAPHO-CNO including rheumatoid arthritis (RA), psoriatic arthritis (PsA), ankylosing spondylitis (AS) or axial spondyloarthritis (AxSpA), autoinflammatory syndromes, inflammatory skin diseases including severe acne, psoriasis (PsV), palmoplantar pustulosis (PPP), hidradenitis suppurativa (HS), inflammatory bowel disease including Crohn disease (CD) and ulcerative colitis (UC), and sarcoidosis; and 4) Language: English (Supplementary Table 3).
The first author (AL) manually reviewed all abstracts and/or full manuscripts and their supplement to screen the articles for additional a priori inclusion criteria: 1) Specific use of the EQ-5D-5L instrument with each study required to report either the EQ-5D-5L index or EQ-5D-5L VAS or both; 2) Study design: cross-sectional observational or survey study design; 3) Disease cohorts: to be representative of disease spectrum. Exclusion criteria were older versions of EQ-5D instrument (i.e. EQ-5D-3L), interventional study designs, and disease subgroups (i.e. limited only to active disease or with a specific comorbidity). The first author (AL) extracted key variables from all included studies on author and year of publication, country/region, study population, study design, diagnostic criteria, sample size, EQ-5D index value, EQ-5D VAS and proportions of responses in five EQ-5D domains.
We assembled the EQ-5D-5L results (means with SD or medians with IQR for the EQ-5D index value and EQ-5D VAS) per each study, and for each CRID. We also summarized the ranges of the EQ-5D results from the studies per disease group. Additionally, we constructed histograms to summarize the distribution of levels of perceived problems (on the 5-item scale) for each of the 5 domains of the EQ-5D-5L instrument for adults with SAPHO-CNO and disease cohorts from the literature.
RESULTS.
SAPHO-CNO Cohort Baseline Characteristics.
A total of 138 adults with SAPHO-CNO were included in this cross-sectional study and their baseline characteristics are summarized in Table 1. The mean age of subjects was 38 (15) years at enrollment into the SCS, with a mean age at diagnosis of SAPHO-CNO of 31 (17) years and ranging from 4 to 70 years of age at diagnosis. The subjects had a mean disease duration since diagnosis of 6.4 (6.2) years. The SCS cohort was predominantly female non-Hispanic white, but also included important racial representations from Asian, Black/African American, and American Indian or Alaska Native populations. Subjects experienced a mean symptom duration of 12.0 (8.3) years, defined as time from onset of any SAPHO-CNO related symptom to enrollment. Importantly, the mean time to diagnosis, defined as the time from onset of any SAPHO-CNO related symptom to diagnosis, was 5.3 (7.3) years.
Table 1.
Baseline demographics and disease characteristics of adults with SAPHO-CNO.
| Variable | Adults with SAPHO-CNO, N=138. |
|---|---|
| Age at enrollment, mean (SD), years | 38 (15) |
| Age at diagnosis, mean (SD), years | 31 (17) |
| Female biological sex (%) | 123 (89) |
| Non-Hispanic (%) | 131 (95) |
| Race (%) | |
| White | 123 (89) |
| Asian (Asian Indian, Chinese, Other Asian) | 6 (4) |
| Black or African American | 4 (3) |
| American Indian or Alaska Native | 3 (2) |
| Other | 5 (4) |
| SAPHO-CNO Disease characteristics (%) | |
| Disease duration (years) | 6.4 (6.2) |
| Time to diagnosis (years) | 5.3 (7.3) |
| Symptom duration (years) | 12.0 (8.3) |
| Musculoskeletal involvement (%) | 138 (100) |
| Inflammatory arthritis (ever) | 61 (46) |
| Spine involvement (ever) | 59 (44) |
| Skin involvement (%), any | 64 (48) |
| PPP | 36 (26) |
| PsV | 35 (25) |
| Severe Acne | 16 (12) |
| HS | 15 (11) |
| Current Medications (%) | |
| NSAID | 74 (54) |
| Biological DMARD | 49 (36) |
| Conventional DMARD | 30 (22) |
| Bisphosphonate IV | 12 (9) |
| Steroid | 10 (7) |
| Targeted synthetic DMARD | 8 (6) |
| Immunosuppressant | 3 (2) |
| Patient Global Assessment of Disease Activity (NRS) | |
| Overall SAPHO-CNO, mean (SD) | 3.93 (3.06) |
| MSK SAPHO-CNO, mean (SD) | 3.99 (3.21) |
| Skin SAPHO-CNO*, mean (SD) | 2.97 (2.86) |
| Comorbidities (%) | |
| Anxiety | 70 (51) |
| Depression | 49 (36) |
| Tobacco use (current or past) | 37 (27) |
| Chronic pain | 29 (21) |
| Malignancy | 7 (5) |
| Diabetes mellitus | 5 (4) |
| EQ-5D-5L index value, mean (SD) | 0.75 (0.16) |
| EQ-5D-5L VAS, mean (SD) | 63.0 (21.0) |
Means (SD) and counts (%) are presented unless otherwise stated. SAPHO: Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis; CNO: chronic nonbacterial osteomyelitis; PPP: Palmoplantar pustulosis; PsV: Psoriasis vulgaris; HS: Hidradenitis suppurativa; NSAID: non-steroidal anti-inflammatory drug; DMARD: disease modifying anti-rheumatic drug; IV: intravenous; NRS: numeric rating scale for patient-reported disease activity ranging from 0 (=well controlled) to 10 (=poorly controlled); MSK: Musculoskeletal; EQ-5D-5L: EuroQol-5 Dimension 5-length instrument; VAS: visual analogue scale. EQ-5D index value ranges from negative values (where 0 is the health state equivalent to death) to 1 (the value of full health), with higher values indicating better HRQOL. EQ-5D VAS ranges from 0 (worst health imaginable) to 100 (best health imaginable).
For subjects with SAPHO-CNO related skin manifestations (n=64).
All subjects had MSK involvement; approximately 46% had prevalent inflammatory arthritis and 44% had spine involvement due to SAPHO-CNO. Almost half of the subjects (48%) reported skin involvement, most commonly PPP (26%) and PsV (25%), followed by severe acne (12%) and HS (11%). Current medications were NSAIDs (54%), biological DMARDs (36%) and conventional DMARDs (22%) followed by oral steroids (7%), targeted synthetic DMARDs (6%) and traditional immunosuppressants (2%). Approximately 9% of subjects were treated with intravenous bisphosphonates within a year of their baseline visit. The mean overall and MSK disease activity by NRS were 3.93 (3.06) and 3.99 (3.21) respectively. In the subgroup of subjects with skin involvement (n=64), the mean skin disease activity by NRS was 2.97 (2.86). Medical comorbidities included anxiety (51%), depression (36%) and chronic pain (21%), while current or prior tobacco use was reported in 27%.
Quality of Life in the SAPHO-CNO Cohort.
We observed a mean EQ-5D-5L index value of 0.75 (0.16), and a mean EQ-5D-5L VAS of 63.0 (21.0), with higher scores indicating better HRQOL (Table 1). Subjects with inflammatory arthritis, spine and skin involvement had numerically lower mean EQ-5D index and VAS scores (Supplementary Table 4). Additionally, the EQ-5D index and VAS scores showed moderate negative correlation with overall and MSK disease activity, and weak negative correlation with skin disease activity in SAPHO-CNO (Supplementary Table 5). We observed that more than one-third of adults with SAPHO-CNO reported problems in all 5 EQ-5D domains, with each EQ-5D domain having five levels of responses ranging from no problems to extreme problems (Figure 1, and Supplementary Table 6). Most remarkably, 86% had problems with pain/discomfort, 65% had problems with carrying out their usual activities, and most subjects had problems with anxiety/depression (54%). For these 3 domains, subjects’ reported problems were proportionally rated higher at “moderate” to “extreme” levels. In contrast, 43% and 33% of subjects had problems with mobility and self-care, respectively, and almost all perceived problems were categorized as either “slight” or “moderate” for these 2 domains.
Figure 1.
Distribution of levels of perceived problems in each of the 5 domains of the EQ-5D-5L instrument among SAPHO-CNO, and chronic rheumatic and inflammatory disease cohorts*.
SAPHO: Synovitis, Acne, Pustulosis, Hyperostosis and Osteitis; CNO: chronic nonbacterial osteomyelitis; SpA: spondyloarthritis; MO: Mobility; SC: Self-Care; UA: Usual Activities; PD: Pain/Discomfort; AD: Anxiety/Depression. *The SAPHO-CNO cohort is from the current study; Behçet disease cohort data is from Murray 2021, axial SpA data is from Neilson 2022, and hidradenitis suppurativa data is from Bato 2021.
Scoping Review of EQ-5D Literature in Chronic Rheumatic and Inflammatory Diseases.
The scoping review focused on the use of the EQ-5D for assessing HRQOL in CRIDs. A total of 365 studies were identified, with 47 original studies included (Figure 2).21–52 The most common reasons for exclusion of articles were interventional or other non-observational study design, incorrect disease, incorrect EQ-5D instrument or missing summary results, and unrepresentative disease cohorts. Of the 47 included studies, 11 were in RA, 9 in AS/AxSpA, 7 in PsA, 7 in IBD, 6 in PsV, 2 in acne, 2 in HS, 2 in sarcoidosis, and 1 in Behçet disease (BD). Surprisingly, only 3 studies reported the proportion of perceived problems on the 5-item scale for each domain of the EQ-5D-5L instrument.22,36,37
Figure 2.
Study Selection Flowchart for Literature Review on EQ-5D in Chronic Rheumatic and Inflammatory Diseases.
EQ-5D: EuroQol-5 Dimension instrument; VAS: visual analogue scale; SpA: spondyloarthritis.
Summary of EQ-5D index values and EQ-5D VAS ranges from all included studies are shown in Tables 2 and 3. The distribution of levels of perceived problems in 5 domains of the EQ-5D-5L are presented in Figure 1 and Supplementary Table 6 from the 3 studies reporting this data.22,36,37 In studies of inflammatory arthritis, both the EQ-5D index value ranges and the EQ-5D VAS ranges were very similar across RA, PsA, and AS/AxSpA cohorts (Table 3). In contrast, the EQ-5D index value and EQ-5D VAS were lower in a single study of BD. Three inflammatory skin conditions had differing ranges for the EQ-5D, with HS having the lowest EQ-5D index and EQ-5D VAS values followed by acne and PsV. The ranges of EQ-5D index and EQ-5D VAS values were lower in sarcoidosis than IBD.
Table 2.
Comparative review of the EQ-5D index value and EQ-5D VAS across chronic rheumatic and inflammatory diseases.
| Disease | Author | Country | N | EQ-5D index value | EQ-5D VAS |
|---|---|---|---|---|---|
| RA | |||||
| Wan 2024 | China | 171 | 0.586 (0.279) | 47.3 (19.6) | |
| Feng 2024 | China | 314 | 0.57 (0.40) | ||
| Haridoss 2024 | India | 320 | 0.54 (0.36) | 63.05 (18.54) | |
| Chamorro-de-Vega 2024 | Spain | 107 | 0.72 (0.25) | 67.5 (22.5) | |
| Sakai 2023 | Japan | 4902 | 0.90 (0.77–1.00) | ||
| Kontodimopoulos 2022 | Greece | 112 | 0.565 | ||
| Thiele 2022 | Germany | 7589 | 0.77 (0.2) | ||
| Ramirez 2022 | Spain | 107 | 0.72 (0.2) | 67.5 (22.5) | |
| Al-Jabi 2021 | Palestine | 300 | 0.56 (0.33–0.74) | 60 (50–77.5) | |
| Glintborg 2021 | Denmark | 8168 | 0.80 (0.72–0.86) | ||
| Hashimoto 2020 | Japan | 85 | 0.810 (0.157) | ||
| PsA | |||||
| Chiowchanwisawakit 2024 | Thailand | 117 | 0.9 (0.1) | ||
| Chamorro-de-Vega 2024 | Spain | 103 | 0.73 (0.24) | 70.3 (no SD) | |
| Coyle 2024 | Europe | 503 | 63.4 (22.03) | ||
| Ramirez 2022 | Spain | 103 | 0.78 (0.2) | 70.3 (19.3) | |
| Kontodimopoulos 2022 | Greece | 55 | 0.59 | ||
| Macfarlane 2021 | UK | 162 | 0.639 (0.22) | ||
| Glintborg 2021 | Denmark | 2068 | 0.77 (0.66–0.83) | 73.79 (18.73) | |
| AS/AxSpA | |||||
| Yu 2024 | China | 238 | 0.74 (0.27) | ||
| Chamorro-de-Vega 2024 | Spain | 108 | 0.71 (0.21) | 69.0 (no SD) | |
| Kontodimopoulos 2022 | Greece | 45 | 0.675 | ||
| Thiele 2022 | Germany | 1504 | 0.77 (0.2) | ||
| Macfarlane 2021 | UK | 596 | 0.652 (0.231) | ||
| Kiltz 2022 | Germany | 191 | 0.6 (0.2) | 52.2 (22.2) | |
| Neilson 2022 | UK | 1965 | 0.693 (0.26) | ||
| Glintborg 2021 | Denmark | 1758 | 0.77 (0.71–0.85) | ||
| Ramirez 2022 | Spain | 108 | 0.71 (0.2) | 69.0 (19.9) | |
| Behçet disease | |||||
| Murray 2021 | UK | 103 | 0.519 (0.315) | 59.04 (23.8) | |
| Acne | |||||
| Szabo 2025 | Hungary | 23 | 0.82 (0.74–0.90) | ||
| Mahfouz 2023 | Saudi Arabia | 334 | 0.771 (0.24) | ||
| Hidradenitis suppurativa | |||||
| Koumaki 2023 | Greece | 70 | 0.75 (0.21) | 62.48 (21.12) | |
| Bato 2021 | Hungary | 198 | 0.76 (0.30) 0.86 (0.71–0.96) | 64.29 (22.68) 70 (50–80) | |
| Psoriasis | |||||
| Szabo 2025 | Hungary | 47 | 0.88 (0.68–1.00) | ||
| Arora 2024 | India | 198 | 69.5 (21.9) | ||
| Chamorro-de-Vega 2024 | Spain | 107 | 0.87 (0.17) | 76.9 (17.6) | |
| Lysen 2024* | Netherlands | 501 | 0.85 (0.74–0.82) | 80 (66–85) | |
| Ramirez 2022 | Spain | 114 | 0.87 (0.2) | 76.9 (17.6) | |
| Su 2020 | China | 321 | 0.91 (0.10) | ||
| Sarcoidosis | |||||
| Bloem 2021 | Netherlands | 59 | 0.74 (0.23) | 62.7 (19.6) | |
| Bloem 2020 | Netherlands | 58 | 0.75 (0.23) | 63.0 (19.7) | |
| IBD | |||||
| Roukas 2024 | UK | 8486 | 0.76 (0.23) | ||
| Barnes 2024 | Australia | 553 | 0.79 (0.15) | 64.47 (19.9) | |
| Chamorro-de-Vega 2024 | Spain | 146 | 0.81 (0.20) | 72.8 (no SD) | |
| Latteur 2024 | Sweden | 9769 | 0.79 (0.21) | 74.48 (19.48) | |
| Ramirez 2022 | Spain | 146 | 0.81 (0.2) | 72.8 (19.7) | |
| Gao 2022 | China | 202 | 0.85 (0.12) | ||
| Hagelund 2020 | Denmark | 302 | 0.77 (0.20) | ||
EQ-5D: EuroQol-5 Dimension; VAS: visual analogue scale; RA: rheumatoid arthritis; PsA: psoriatic arthritis; AS: ankylosing spondylitis; AxSpA: axial spondyloarthritis; IBD: inflammatory bowel disease. The EQ-5D index and VAS are shown as mean (SD) or median (interquartile range). EQ-5D index value ranges from negative values (where 0 is the health state equivalent to death) to 1 (the value of full health), with higher values indicating better HRQOL. EQ-5D VAS ranges from 0 (worst health imaginable) to 100 (best health imaginable), with higher values indicating better health.
This study included 44% with psoriatic arthritis.
Table 3.
Summary of the EQ-5D index value and EQ-5D VAS ranges in SAPHO-CNO and chronic rheumatic and inflammatory diseases.
| Disease | EQ-5D index | EQ-5D VAS |
|---|---|---|
| SAPHO-CNO* | 0.75 (0.16) | 63.0 (21.0) |
| RA | 0.54–0.90 | 47.3–67.5 |
| PsA | 0.59–0.9 | 63.4–73.8 |
| Ankylosing Spondylitis/Axial SpA | 0.6–0.77 | 52.2–69.0 |
| Behçet | 0.52 | 59.0 |
| Acne | 0.77–0.82 | |
| Hidradenitis Suppurativa | 0.75–0.76 | 62.5–64.3 |
| Psoriasis | 0.85–0.91 | 69.5–80.0 |
| Sarcoidosis | 0.74–0.75 | 62.7–63.0 |
| IBD | 0.76–0.85 | 64.5–74.5 |
EQ-5D: EuroQol-5 Dimension; VAS: visual analogue scale; RA: rheumatoid arthritis; PsA: psoriatic arthritis; SpA: spondyloarthritis; IBD: inflammatory bowel disease. EQ-5D index value ranges from negative values (where 0 is the health state equivalent to death) to 1 (the value of full health), with higher values indicating better HRQOL. EQ-5D VAS ranges from 0 (worst health imaginable) to 100 (best health imaginable), with higher values indicating better health.
For SAPHO-CNO, the EQ-5D index and EQ-5D VAS are shown as mean (standard deviation).
EQ-5D for HRQOL Across SAPHO-CNO and Related Diseases.
Adults with SAPHO-CNO had a mean EQ-5D index value that was generally similar to the reported range from studies in RA, PsA, AS/AxSpA, HS and sarcoidosis, while it was lower than the reported range in acne, PsV and IBD (Tables 2 and 3). Adults with SAPHO-CNO had a mean EQ-5D VAS within the reported range of studies in PsA, AS/AxSpA, HS and sarcoidosis, while it was lower than the reported range in RA, PsV and IBD. Both the mean EQ-5D index value and EQ-5D VAS were slightly higher in adults with SAPHO-CNO relative to a single study in BD.
In Figure 1 and Supplementary Table 6, we summarized the proportion of perceived problems in each domain of the EQ-5D-5L instrument (on the 5-item scale) in SAPHO-CNO, and BD, AxSpA and HS cohorts from the literature.22,36,37 Mobility problems were generally similar for SAPHO-CNO and HS, while AxSpA and BD patients reported numerically more “severe problems”. Only 1% of SAPHO-CNO, AxSpA and BD reported problems in the “unable to walk” category. Problems in self-care were comparable between SAPHO-CNO and AxSpA, while patients with BD reported numerically more “moderate” (21%) to “severe” (8%) problems. Across all 4 diseases, none reported problems in the category of “unable to wash or dress” for self-care. For usual activities, a similar proportion across categories of problems was seen for SAPHO-CNO and AxSpA, while BD patients reported numerically more problems in the “severe” (13%) and “unable to” (7%) categories. Problems in the domain pain/discomfort were prevalent across all 4 diseases: 93% in AxSpA, 87% in BD, 86% in SAPHO-CNO, and 77% in HS reported at least “slight pain”. For SAPHO-CNO, AxSpA and HS, similar proportions were reported across categories of pain (from “slight” to “extreme” pain), while BD patients reported a numerically higher proportion of “severe pain” (24%). Lastly, for the domain of anxiety/depression, SAPHO-CNO subjects reported similar proportions of problems across all 5 categories as AxSpA and HS, while BD patients reported numerically higher proportions in the “severely” (9%) and “extremely” (7%) categories.
DISCUSSION.
Compared to the general population, QOL is reduced in individuals living with CRIDs but remains understudied in rare autoinflammatory diseases. In this study, we aimed to measure HRQOL in adults with SAPHO-CNO and to discuss our findings relative to disease cohorts with phenotypic similarities to SAPHO-CNO. By using data from the SAPHO-CNO Study, a novel prospective longitudinal US-based cohort study, we found that more than one-third of subjects reported problems in all EQ-5D domains. Most notably, adults with SAPHO-CNO had similar range of EQ-5D values and levels of perceived problems when compared with disease cohorts (RA, PsA, AS/AxSpA, HS, sarcoidosis) from our literature review. Our results highlight the importance of addressing both active disease and reduced HRQOL in adults living with SAPHO-CNO.
We observed that HRQOL was reduced in adults with SAPHO-CNO, relative to the general US population norms.53 In a representative sample of 1134 individuals aged 35–44 from the US general population, Jiang et al. observed a mean EQ-5D index value of 0.84 (0.21) and EQ-5D VAS of 78.1 (15.4).53 Comparatively, the mean EQ-5D index value and VAS were both lower in our study. In addition, we observed that at least a third of adults with SAPHO-CNO reported problems in all 5 domains measured by the EQ-5D. The majority of subjects reported problems with pain/discomfort, performing usual activities, and anxiety/depression, and frequently rated these problems as “moderate” to “extreme”. Problems were also reported for mobility and self-care, although less frequently and more likely to be rated as “slight” or “moderate”. In comparison, the sampled US adults by Jiang et al. had proportionally fewer problems in all 5 domains.53 Thus, our findings show that HRQOL is reduced compared with the general population, and confirm that all measured QOL domains are highly impactful for adults living with SAPHO-CNO.5,9
Our novel findings using the EQ-5D-5L instrument to measure and characterize HRQOL in the SCS study cohort expand the current literature on QOL in SAPHO-CNO by presenting a larger study, with adult patients.4,7,10 Our study is the first to use the EQ-5D-5L instrument in a large well-characterized cohort. Three prior adult population studies, which used a well-established legacy measure (SF-20 and SF-36) to assess QOL, have reported decreased QOL in adults with SCCH and SAPHO-CNO, but did not use US populations.5,8,9 This makes our study unique.
In a cross-sectional study of 52 adults with SCCH, patients had worse scores compared to healthy controls on the physical, social and role functioning, pain, and physical and mental health domains measured by the SF-20 questionnaire.8 SAPHO patients were found to have limitations in all 8 dimensions of QOL as measured by the SF-36 questionnaire in a cross-sectional survey study of 64 adults with SAPHO syndrome in Germany.5 These participants also reported high pain and impairments in activities, with musculoskeletal symptoms having the highest impact on well-being.5 Adult SCCH/CNO patients showed impairments in nearly all aspects of QOL measured by the SF-36 with a delay in diagnosis (>5 years) identified as one of the major determinants of QOL in a cross-sectional study from the Netherlands.9 Finally, a single study from China that focused on the effects of SAPHO on work productivity reported a similar mean EQ-5D index value as our study; however, the authors did not specify which EQ-5D instrument was used, and neither the EQ-5D VAS nor the proportions of perceived problems in the five domains were reported.15 Overall, prior studies demonstrate low QOL across multiple domains in different adult populations, thus highlighting the importance of identifying predictors of this key outcome in SAPHO-CNO.10
We also aimed to contextualize our EQ-5D findings through a descriptive comparison to important CRID cohorts, as no prior adult SAPHO-CNO studies met inclusion criteria. Adults with SAPHO-CNO had mean EQ-5D index and VAS similar to reported ranges in inflammatory arthritis (RA, PsA, AS/AxSpA), inflammatory skin disease (HS) and sarcoidosis; additionally, both were also numerically lower than the reported ranges in acne, PsV and IBD. The proportion of problems in each of 5 domains in the EQ-5D were similar across SAPHO-CNO and AxSpA. Additionally, similarities were seen for the domains of pain/discomfort and anxiety/depression for SAPHO-CNO and HS. This confirms that QOL is reduced in adult SAPHO-CNO with clinical manifestations spanning musculoskeletal and skin involvement. In contrast, compared to a single study in BD, HRQOL was slightly better in adult SAPHO-CNO. These observed EQ-5D differences may be attributable to longer disease duration and multisystem involvement in this UK-based BD cohort.36 Although catalogues of HRQOL measured by EQ-5D exist for common chronic conditions, it is imperative to describe and quantify HRQOL in rarer autoinflammatory diseases.13 Hence, publications in BD and autoinflammatory diseases, along with our findings and prior QOL studies using SF-36 are important in adding to the literature on reduced QOL in rare rheumatic diseases.5,8,9,16,36,54,55
Our study is not without limitations. This is an observational cohort study, with inherent limitations around bias in patients who agree to participate, as well as potential recall bias when patients report symptom duration and disease characteristics. This bias was minimized by confirmation of time-related data with subjects, thorough review of medical records, and clinical confirmation of disease manifestations by the study’s core investigators. Despite its increasing use in clinical research, the newer EQ-5D instrument may not fully capture the multidimensional construct of QOL. The SF-36, a legacy instrument, may provide more insight into broader domains of QOL, and would be well suited for further psychometric analyses in our cohort. We did not assess factors associated with low QOL, as this was not the aim of the study; in the future, potential directions will include longitudinal measurements of QOL by SF-36 and identifying predictors of QOL trajectories in our cohort. Our literature review may have missed some studies of EQ-5D in SAPHO-CNO and CRIDs. Finally, we did not perform a robust quantitative analysis of our QOL findings to the literature cohorts, as a direct comparison would be difficult given the heterogeneity in the distributions of demographics, disease characteristics, comorbidities, and treatments across diseases. Instead, we provided a thorough descriptive comparison to contextualize our QOL findings amongst important CRIDs.
In conclusion, in this cross-sectional study, we showed that QOL is reduced in a novel observational cohort of adults with SAPHO-CNO. More than one-third of participants reported QOL problems in all measured domains, with more than half experiencing impact from perceived pain, performing usual activities, and feelings of anxiety or depression. We demonstrated that adults living with SAPHO-CNO have a comparable reduction in QOL to other relevant CRID cohorts with musculoskeletal and skin manifestations. Thus, our study highlights the importance of addressing the multi-dimensional nature of SAPHO-CNO, targeting HRQOL as a major outcome.
Supplementary Material
The source(s) of support in the form of grants or industrial support:
This work was funded by the National Institutes of Health (K23 AR082966 to A.L., P50 AR080612 to R.D., P30 AR072577 to D.H.S., UM1TR004403 to the University of Iowa).
Footnotes
Conflict of interest:
D.H.S. has received research contracts unrelated to the current study from Amgen, CorEvitas, and Janssen, and he also receives royalties from unrelated chapters in UpToDate. The remaining authors declare no conflicts of interest relevant to this article.
Statement of ethics and consent:
This study has received approval from the University of Iowa institutional review board (HawkIRB; ID# 202107229) through the single site IRB (SMART IRB) reliance mechanism required by the National Institutes of Health. We obtained written informed consent from all subjects enrolled in this study.
Contributor Information
Aleksander Lenert, Division of Immunology, Department of Internal Medicine, Carver College of Medicine, University of Iowa, 200 Hawkins Drive, Iowa City, IA 52242, USA..
Jenna Thomason, Division of Rheumatology, University of Washington, Rheumatology Clinic at Harborview, 325 9th Ave, Seattle, WA 98104, USA..
Melanie H. Smith, Weill Cornell Medicine, Hospital for Special Surgery, 525 E 71st St, 8th Floor, New York, NY 10021, USA..
Helena Abodeely H.S., Division of Immunology, University of Iowa, 200 Hawkins Drive, Iowa City, IA 52242, USA..
Sandy D. Hong, Division of Pediatric Rheumatology, University of Iowa Stead Family Children’s Hospital, 200 Hawkins Drive, Iowa City, IA 52242, USA..
Courtney E. Kremer, Division of Pediatric Rheumatology, University of Iowa Stead Family Children’s Hospital, 200 Hawkins Drive, Iowa City, IA 52242, USA..
Petar S. Lenert, Division of Immunology, Department of Internal Medicine, Carver College of Medicine, University of Iowa, 200 Hawkins Drive, Iowa City, IA 52242, USA..
T. Shawn Sato, Department of Radiology, University of Iowa Hospitals and Clinics, 200 Hawkins Drive, Iowa City, IA 52242, USA..
Sandra Hansmann, Department of Pediatrics, Division of Pediatric Rheumatology and autoinflammation reference center Tuebingen (arcT), University Hospital Tuebingen, Hoppe-Seyler-Str. 1, 72076 Tuebingen, Germany..
Melissa Oliver, Division of Pediatric Rheumatology, Indiana University School of Medicine, 705 Riley Hospital Dr, Indianapolis, IN 46202, USA..
Yongdong (Dan) Zhao, Division of Pediatric Rheumatology, Department of Pediatrics, University of Washington School of Medicine, Seattle Children’s Hospital, 4800 Sand Point Way NE, Seattle, WA, 98105, USA..
Jonathan Templin, Department of Psychological and Quantitative Foundations, College of Education, University of Iowa, 240 Lindquist Center, Iowa City, IA 52242, USA..
Mary Vaughan-Sarrazin, Department of Internal Medicine, University of Iowa Carver College of Medicine, 200 Hawkins Drive, Iowa City, IA 52242, USA..
Robyn Domsic, Division of Rheumatology & Clinical Immunology, University of Pittsburgh, 3601 5th Avenue, Suite 2B, Pittsburgh, PA 15213, USA..
Daniel H. Solomon, Division of Rheumatology, Inflammation, and Immunity, Brigham and Women’s Hospital and Harvard Medical School, 75 Francis Street, Boston, MA 02115, USA..
Polly J. Ferguson, Division of Pediatric Rheumatology, Carver College of Medicine, University of Iowa Stead Family Children’s Hospital, 200 Hawkins Drive, Iowa City, IA 52242, United States..
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