Goal Attainment Scaling for haemophilia (GAS-Hem): Testing the feasibility of a new patient-centric outcome measure in people with haemophilia

J. C. Roberts, S. Lattimore, Michael Recht, S. Jackson, D. Gue, S. Squire, K. S. Robinson, V. Price, M. Denne, S. Richardson, K. Rockwood

Research output: Contribution to journalArticlepeer-review

4 Scopus citations

Abstract

Introduction: To address the need for a patient-reported outcome that can measure clinically and personally meaningful change in people with haemophilia (PwH) on prophylaxis, an approach based on Goal Attainment Scaling (GAS) was developed: the GAS-Hem. Aim: To establish real-world feasibility of GAS-Hem in PwH. Methods: Patients aged 5-65 years were enroled from four North American centres for a 12-week study. The primary outcome was the proportion of participants who completed GAS-Hem interviews at baseline, 6 and 12 weeks. GAS-Hem scores were obtained by subject- and clinician-rated goal attainment at Weeks 6 and 12, and compared with quality of life (QoL) measures and annualized bleed rate (ABR) for construct validity. Goals were evaluated qualitatively for content validity. Responsiveness was calculated using standardized response means (SRM). Results: Forty-two participants set 63 goals. Participants preferred to define (37/63) their own goals or further individualize (23/63) from the GAS-Hem menu. Thirty of the 37 self-defined goals were matched to goals on the GAS-Hem menu. The most common goal areas were: weight, exercise and nutrition (n = 17); leisure activities (n = 8); and joint problems (n = 7). Both participant- and clinician-rated GAS-Hem scores at 6 weeks (n = 40) and 12 weeks (n = 41) demonstrated satisfactory goal attainment (SRM [subject-rated] at 12 weeks for adult and paediatric groups was 1.25 and 1.16, respectively). Correlations of GAS-Hem scores with QoL measures and ABR were uniformly small. Conclusion: GAS-Hem was feasible and tapped constructs not captured by ABR or QoL measures.

Original languageEnglish (US)
JournalHaemophilia
DOIs
StateAccepted/In press - Jan 1 2018

Keywords

  • Goal attainment scaling
  • Haemophilia A
  • Outcome measure
  • Patient engagement
  • Patient-centred
  • Prophylaxis

ASJC Scopus subject areas

  • Hematology
  • Genetics(clinical)

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