Haemophilia. 2019;00:1–10. wileyonlinelibrary.com/journal/hae
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1 © 2019 John Wiley & Sons Ltd
1 | INTRODUCTION
Prophylaxis with regular infusion of clotting factor concentrates
(CFCs) has been considered the gold standard treatment for patients
with severe haemophilia.
1-3
However, adherence to prophylaxis in
haemophilia is very demanding and requires lifelong dedication
to prevent bleeding and, consequently, disability.
4
This has been
a primary factor underpinning the development of several novel
Received: 8 February 2019
|
Revised: 20 May 2019
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Accepted: 3 June 2019
DOI: 10.1111/hae.13811
ORIGINAL ARTICLE
Comparing objective and self‐reported measures of adherence
in haemophilia
Vanessa Giroto Guedes
1,2
| José Eduardo Corrente
2
| Albert Farrugia
3
|
Sylvia Thomas
4
| Patrick Alexander Wachholz
2
| Edison Iglesias de Oliveira Vidal
2
1
Blood Center of Marília Medical
School, FAMEMA, Marília, Brazil
2
Botucatu Medical School, São Paulo State
University – UNESP, Botucatu, Brazil
3
School of Surgery, University of Western
Australia, Crawley, Western Australia,
Australia
4
Radiology Department, Hospital
Clementino Fraga Filho, Federal University
of Rio de Janeiro ‐ UFRJ, Rio de Janeiro,
Brazil
Correspondence
Vanessa Giroto Guedes, Physiotherapy
Service of Blood Center of Marília Medical
School ‐ FAMEMA, Rua Lourival Freire,
nº 240, Bairro Fragata, Marília/SP – CEP:
17519-050.
Email: vanessagiroto@hotmail.com
Present address
Vanessa Giroto Guedes, Radiology
Department, Hospital Clementino Fraga
Filho, Federal University of Rio de Janeiro ‐
UFRJ, Rio de Janeiro, Brazil
Abstract
Aim: To compare subjective and objective measures of adherence to prophylaxis in
haemophilia.
Methods: In this cross-sectional study, we compared participants’ self-perceived ad-
herence and their estimate of the number of clotting factor concentrates (CFCs) that
had been missed over the last period of CFC dispensation with an objective measure
of adherence based on counts of CFC vials returned by participants.
Results: We included 29 out of 31 eligible patients in the study. There was no sig-
nificant correlation between self-perceived degree of adherence and the objective
classification of adherence (Rho: 0.10, 95% CI: −028 to 0.46, P: 0.61) and between the
classification of adherence based on the proportion of missed CFC doses assessed
by participants’ self‐report and objectively (Rho: 0.32, 95% CI: −0.01 to 0.59, P: 0.11).
Conversely, we found evidence of moderate correlation between the proportion of
missed CFC doses as assessed by participants’ self‐report and objectively (Rho: 0.56,
95% CI: 0.24 to 0.77, P: 0.003). Participants’ self-perceived adherence was 3 times
more likely to be rated as very good or good than it was for the objective assessment
to be classified as adherent or suboptimally adherent.
Conclusion: Our results showed significant discrepancies between subjective and
objective measures of adherence, which likely reflect the influence of social desir-
ability bias in self-reported measures and different concepts of adherence between
patients/caregivers and haemophilia experts. Additionally, our results allow us to hy-
pothesize that studies on adherence to prophylaxis in haemophilia relying exclusively
on information from self-reports and questionnaires may substantially overestimate
adherence levels.
KEYWORDS
Brazil, developing countries, haemophilia, medication adherence, patient compliance