Health-related quality of life in ANCA-associated vasculitis and item generation for a disease-specific patient-reported outcome measure

Joanna C Robson^{1

2}, Jill Dawson³, Peter F Cronholm⁴, Nataliya Milman⁵, Katherine S Kellom⁶, Susan Ashdown⁷, Ebony Easley⁸, John T Farrar⁹, Don Gebhart^{10

11}, Georgia Lanier^{10

11}, Carol A McAlear¹², Jacqueline Peck⁷, Raashid A Luqmani⁷, Judy A Shea¹³, Gunnar Tomasson¹⁴, Peter A Merkel^{10

11}

Affiliations

¹ Department of Rheumatology, Faculty of Health and Applied Sciences, University of the West of England, Bristol, UK.
² School of Clinical Sciences, University of Bristol, Bristol, UK.
³ Department of Population Health (HSRU), University of Oxford, Oxford, UK.
⁴ Department of Family Medicine and Community Health, University of Pennsylvania, Philadelphia, PA, USA.
⁵ Division of Rheumatology, Department of Medicine, University of Ottawa, Ottawa Hospital Research Institute, Ottawa, ON, Canada.
⁶ PolicyLab, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
⁷ Nuffield Department of Orthopaedics, Rheumatology and Musculoskeletal Sciences, University of Oxford, Oxford, UK.
⁸ Department of Family Medicine and Community Health, Mixed Methods Research Laboratory, University of Pennsylvania, Philadelphia, PA, USA.
⁹ Biostatistics and Epidemiology, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.
¹⁰ Division of Rheumatology, Department of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
¹¹ Department of Biostatistics and Clinical Epidemiology, University of Pennsylvania, Philadelphia, PA, USA.
¹² Vasculitis Research, University of Pennsylvania, Philadelphia, PA, USA.
¹³ School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
¹⁴ Department of Public Health Sciences, University of Iceland, Reykjavik, Iceland.

PMID: 29379322
PMCID: PMC5759851
DOI: 10.2147/PROM.S144992

Health-related quality of life in ANCA-associated vasculitis and item generation for a disease-specific patient-reported outcome measure

Joanna C Robson et al. Patient Relat Outcome Meas. 2018.

. 2018 Jan 4:9:17-34.

doi: 10.2147/PROM.S144992. eCollection 2018.

Authors

Affiliations

¹ Department of Rheumatology, Faculty of Health and Applied Sciences, University of the West of England, Bristol, UK.
² School of Clinical Sciences, University of Bristol, Bristol, UK.
³ Department of Population Health (HSRU), University of Oxford, Oxford, UK.
⁴ Department of Family Medicine and Community Health, University of Pennsylvania, Philadelphia, PA, USA.
⁵ Division of Rheumatology, Department of Medicine, University of Ottawa, Ottawa Hospital Research Institute, Ottawa, ON, Canada.
⁶ PolicyLab, Children's Hospital of Philadelphia, Philadelphia, PA, USA.
⁷ Nuffield Department of Orthopaedics, Rheumatology and Musculoskeletal Sciences, University of Oxford, Oxford, UK.
⁸ Department of Family Medicine and Community Health, Mixed Methods Research Laboratory, University of Pennsylvania, Philadelphia, PA, USA.
⁹ Biostatistics and Epidemiology, Hospital of the University of Pennsylvania, Philadelphia, PA, USA.
¹⁰ Division of Rheumatology, Department of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
¹¹ Department of Biostatistics and Clinical Epidemiology, University of Pennsylvania, Philadelphia, PA, USA.
¹² Vasculitis Research, University of Pennsylvania, Philadelphia, PA, USA.
¹³ School of Medicine, University of Pennsylvania, Philadelphia, PA, USA.
¹⁴ Department of Public Health Sciences, University of Iceland, Reykjavik, Iceland.

PMID: 29379322
PMCID: PMC5759851
DOI: 10.2147/PROM.S144992

Abstract

Objective: The antineutrophil cytoplasmic antibody (ANCA)-associated vasculitides (AAVs) are multisystem diseases of the small blood vessels. Patients experience irreversible damage and psychological effects from AAV and its treatment. An international collaboration was created to investigate the impact of AAV on health-related quality of life (HRQoL), and develop a disease-specific patient-reported outcome measure to assess outcomes of importance to patients.

Methods: Patients with AAV from the UK, USA, and Canada were interviewed to identify salient aspects of HRQoL affected by AAV. The study was overseen by a steering committee including four patient research partners. Purposive sampling of interviewees ensured representation of a range of disease manifestations and demographics. Inductive analysis was used to identify themes of importance to patients; these were further confirmed by a free-listing exercise in the US. Individual themes were recast into candidate items, which were scrutinized by patients, piloted through cognitive interviews and received a linguistic and translatability evaluation.

Results: Fifty interviews, conducted to saturation, with patients from the UK, USA, and Canada, identified 55 individual themes of interest within seven broad domains: general health perceptions, impact on function, psychological perceptions, social perceptions, social contact, social role, and symptoms. Individual themes were constructed into >100 candidate questionnaire items, which were then reduced and refined to 35 candidate items.

Conclusion: This is the largest international qualitative analysis of HRQoL in AAV to date, and the results have underpinned the development of 35 candidate items for a disease-specific, patient-reported outcome questionnaire.

Keywords: ANCA-associated vasculitis; eosinophilic granulomatosis with polyangiitis; granuloma-tosis with polyangiitis; microscopic polyangiitis; patient-reported outcomes; quality of life.

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Conflict of interest statement

Disclosure Dr Robson and Professor Luqmani were supported in part by the National Institute for Health Research Musculoskeletal Biomedical Research Unit, Oxford, UK. Dr Robson was supported by a National Institute for Health Research (NIHR) clinical lectureship. Dr Milman was supported by a UCB/Canadian Rheumatology Association/Arthritis Society postgraduate rheumatology fellowship award and a research fellowship from the Department of Medicine at the Ottawa Hospital. The authors report no other conflicts of interest in this work.

Figures

**Figure 1**
Smith’s Salience Indices of patient’s free-listed and ranked terms. **Notes:** (A) Terms at AAV diagnosis; (B) terms during AAV remission; (C) terms during AAV flare; (D) comparison across AAV activity states. **Abbreviations:** AAV, associated vasculitis; labs, laboratory tests.

**Figure 2**
Conceptual framework for the AAV-PRO. **Notes:** The scope of the developing measure was defined by the steering committee and evolved during development to include symptom severity, the impact of problems and limitations imposed by patients’ ANCA-associated vasculitis and treatment on their work and domestic roles, family and social interactions (including activities and interests outside the home), and psychological state. **Abbreviations:** AAV, ANCA-associated vasculitis; ANCA, antineutrophil cytoplasmic antibody; PRO, patient-reported outcome.

See this image and copyright information in PMC

References

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Health-related quality of life in ANCA-associated vasculitis and item generation for a disease-specific patient-reported outcome measure

Affiliations

Health-related quality of life in ANCA-associated vasculitis and item generation for a disease-specific patient-reported outcome measure

Authors

Affiliations

Abstract

Conflict of interest statement

Figures

References

Grants and funding

LinkOut - more resources

Full Text Sources

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