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Multicenter Study
. 2025 Oct;125(10):960-971.
doi: 10.1055/a-2501-1369. Epub 2025 Jan 21.

Psychometric Validation of the Hemophilia Functional Ability Scoring Tool (Hemo-FAST)

Virginie Barbay  1 , Yohann Repessé  2 , Dominique Desprez  3 , Nicolas Drillaud  4 , Birgit Frotscher  5 , Marie-Léa Piel-Julian  6 , Sabine Castet  7 , Fabienne Genre-Volot  8 , Brigitte Tardy  9 , Annie Harroche  10 , Corinne Gandossi  11 , Meriem Zidi  11 , Sara Carlsson  12 , Nana Kragh  13 , Eva Bednar  14 , Claude Négrier  15 , Aurélien Lebreton  16
Affiliations
Multicenter Study

Psychometric Validation of the Hemophilia Functional Ability Scoring Tool (Hemo-FAST)

Virginie Barbay et al. Thromb Haemost. 2025 Oct.

Abstract

The Hemophilia Functional Ability Scoring Tool (Hemo-FAST), consisting of a patient-reported outcome (PRO) part and a clinician-reported outcome (ClinRO) part, was developed as a rapid and effective tool to assess functional mobility in clinical practice. This study (NCT04731701) aimed to validate the psychometric properties of Hemo-FAST for assessment of joint health in people with haemophilia (PwH).PwH A or B aged ≥18 years completed questionnaires including the PRO part of Hemo-FAST and the short-form 36 health survey (SF-36) during one study visit. Clinicians completed the Haemophilia Joint Health Score (HJHS) and the ClinRO part of Hemo-FAST at the same visit. Validation was performed using reliability, construct validity, and structure validity assessments.The study enrolled 180 PwH A or B from 14 centres across France. Estimated time to complete the PRO part was mean (standard deviation) 4.6 (5.4) minutes. PRO items showed good test-retest reliability (intraclass correlation coefficient value ≥0.70). Inter-rater values were >0.70 for 7/9 ClinRO items, indicating good reliability. All items (15 PRO; 9 ClinRO) had high internal consistency (Cronbach's coefficient alpha: 0.97). Hemo-FAST demonstrated convergent construct validity with HJHS and the SF-36 physical component and discriminant construct validity with the SF-36 mental health component. Hemo-FAST scores distinguished between subgroups of people with expected differences in joint health status, including by haemophilia severity (p < 0.0001).This study successfully validated Hemo-FAST as a rapid and reliable tool for the functional assessment of joint health in adults with haemophilia, both in clinical practice and clinical research settings.

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Conflict of interest statement

V.B. declares support for the present manuscript from Sobi to the medical writer for medical writing; grants or contracts from CSL Behring paid to their institution; consulting fees from Sobi and LFB; and support for attending meetings and/or travel from Sobi. Y.R. declares payment or honoraria from Sobi, LFB, and Roche, and support for attending meetings and/or travel from Sobi, CSL Behring, Roche, and Novo Nordisk. D.D. and B.F. declare no conflicts of interest. N.D. declares support for the present manuscript from Sobi to their institution; consulting fees from Sobi and Octapharma; participation on a Data Safety Monitoring Board or Advisory Board for Sobi; and support for attending meetings and/or travel from Novo Nordisk, LFB, and Roche. M.-L.P.-J. declares payment or honoraria from Novo Nordisk, and support for attending meetings and/or travel from Amgen, Novo Nordisk, Octapharma, LFB, CSL Behring, and Sobi. S.Cas. declares consulting fees from Sobi; payment for expert testimony from Sobi, Novo Nordisk, LFB, and Takeda; and support for attending meetings and/or travel from Novo Nordisk, Sobi, and Roche. F.G.-V. declares payment or honoraria for oral presentations from Sobi, Roche, Takeda, and CSL Behring; support for attending meetings and/or travel from Sobi, Roche, LFB, CSL Behring, and Pfizer; and participation on a Data Safety Monitoring Board or Advisory Board for Sobi, Pfizer, and LFB. B.T. declares grants or contracts from Sobi, Novo Nordisk, Roche, Takeda, CSL Behring, and Octapharma paid to their institution; payment or honoraria from Novo Nordisk, Sobi, and CSL Behring; and support for attending meetings and/or travel from Novo Nordisk, Roche, and CSL Behring. A.H. declares payment or honoraria for oral presentations from Sobi, Roche, LFB, Takeda, Novo Nordisk, CSL Behring, and Octapharma; support for attending meetings and/or travel from Sobi, Roche, LFB, Takeda, Novo Nordisk, CSL Behring, and Octapharma; and participation on a Data Safety Monitoring Board or Advisory Board for Sobi, Roche, LFB, Takeda, Novo Nordisk, CSL Behring, and Octapharma. C.G. and N.K. are shareholders of stock or stock options of Sobi, are employees of Sobi, and declare support for the present manuscript from Sobi. M.Z. is an employee of Sobi, and declares support for the present manuscript from Sobi. S.Car. is a shareholder of stock or stock options of Sobi, is an employee of Sobi, and a deputy member of the Sobi board of directors (employee representative). E.B. is a shareholder of stock or stock options of Sobi and is an employee of Sobi. C.N. declares support for the present manuscript from Sobi; grants or contracts from Novo Nordisk and Sobi; consulting fees from Sobi; and payment or honoraria from Novo Nordisk and Sobi. A.L. declares support for the present manuscript from Sobi; grants or contracts from Sobi, Takeda, Bayer, CSL Behring, LFB, Novo Nordisk, Octopharma, Pfizer, and Roche; consulting fees from LFB, Pfizer, Roche, and Sobi; payment or honoraria from LFB, Pfizer, Roche, Sobi, and Takeda; and support for attending meetings and/or travel from CSL Behring, Novo Nordisk, Octapharma, Roche, and Sobi.

Figures

Fig. 1
Fig. 1
Hemophilia Functional Ability Scoring Tool (Hemo-FAST) components. The pre-validation Hemo-FAST comprised 17 items in the patient-reported outcome (PRO) part whereas, following validation, the final Hemo-FAST PRO scores were calculated based on 15 questions. Time to complete the clinician-reported outcome (ClinRO) part of Hemo-FAST was not assessed in the study.
Fig. 2
Fig. 2
Hemophilia Functional Ability Scoring Tool (Hemo-FAST) scores at test and retest in the overall population and subgroups. ( A ) Total score and patient-reported outcome (PRO) score in the overall population ( n  = 180). ( B ) Total score by severity of haemophilia. ( C ) Total score by treatment regimen. (D) Total score by type of haemophilia. Scores were not calculated if more than 10% of the questions were unanswered. A score of 0 indicated the best possible joint health status and 100 indicated the worst joint health. SD, standard deviation.
Fig. 3
Fig. 3
Test–retest reliability of Hemophilia Functional Ability Scoring Tool (Hemo-FAST) patient-reported outcome (PRO) per question. *The sensitivity analysis set included people with haemophilia (PwH) who started the retest of Hemo-FAST PRO more than 30 minutes (and up to 169 minutes) after completing the first test. Q14 and Q17 are not shown because they were removed from the PRO part of the questionnaire following validation. Intraclass correlation coefficient (ICC) values <0.50 indicated poor reliability, values between 0.50 and <0.70 moderate reliability, and values ≥0.70 good reliability.
Fig. 4
Fig. 4
Construct validity—correlation of Hemophilia Functional Ability Scoring Tool (Hemo-FAST) with Haemophilia Joint Health Score (HJHS) and short-form 36 health survey (SF-36)—presented using Pearson's correlation coefficient. Convergent validity criteria were met if r > 0.50.
Fig. 5
Fig. 5
Construct validity of the Hemophilia Functional Ability Scoring Tool (Hemo-FAST) scale. ( A ) Hemo-FAST score stratified by Haemophilia Joint Health Score (HJHS) scores. ( B ) Distribution of Hemo-FAST score according to haemophilia severity. Boxplots showing means (squares), medians (bars inside each box), interquartile ranges (box limits), and minimum and maximum (bars) excluding outliers (triangles). An outlier was defined as a value more than 1.5 times of the interquartile range below the first quartile or above the third quartile. ( A ) Changes in joint health according to HJHS score were defined as: normal HJHS ≤3 (abnormal >3) for people with haemophilia (PwH) ≤50 years old; normal HJHS ≤8 (abnormal >8) for PwH >50 years old. ( B ) Mild haemophilia was defined as PwH with >5% to <40% basal factor VIII or IX; moderate haemophilia was defined as PwH with 1 to 5% basal factor VIII or IX; severe haemophilia was defined as PwH with <1% basal factor VIII or IX. F-statistic is the ratio of the variation between sample means and the variation within samples. F, F-statistic.
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