Implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource-constrained settings: a systematic review of adoption, cost and acceptability

Objectives Mortality associated with sickle cell disease (SCD) is high in many low- and middle-income countries (LMICs). Hydroxyurea, a medicine to effectively manage SCD, is not widely available in resource-constrained settings. We identified and synthesised the reported implementation outcomes for...

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Main Authors: Joyce Gyamfi, Temitope Ojo, Dorice Vieira, Nessa Ryan, Lotanna Dike, Obiageli Nnodu, Emmanuel Peprah
Format: Article
Language:English
Published: BMJ Publishing Group 2020-11-01
Series:BMJ Open
Online Access:https://bmjopen.bmj.com/content/10/11/e038685.full
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author Joyce Gyamfi
Temitope Ojo
Dorice Vieira
Nessa Ryan
Lotanna Dike
Obiageli Nnodu
Emmanuel Peprah
author_facet Joyce Gyamfi
Temitope Ojo
Dorice Vieira
Nessa Ryan
Lotanna Dike
Obiageli Nnodu
Emmanuel Peprah
author_sort Joyce Gyamfi
collection DOAJ
description Objectives Mortality associated with sickle cell disease (SCD) is high in many low- and middle-income countries (LMICs). Hydroxyurea, a medicine to effectively manage SCD, is not widely available in resource-constrained settings. We identified and synthesised the reported implementation outcomes for the therapeutic use of hydroxyurea for SCD in these settings.Design Systematic review.Data sources PubMed, Embase, Cochrane, Web of Science Plus, Global Health, CINAHL, and PsycINFO were searched February through May 2019 without any restrictions on publication date.Eligibility criteria We included empirical studies of hydroxyurea for management of SCD that were carried out in LMICs and reported on implementation outcomes.Data extraction and synthesis Two reviewers independently assessed studies for inclusion, carried out data extraction using Proctor et al.’s implementation and health service outcomes, and assessed the risk of bias using ROBINS-I (Risk Of Bias In Non-randomised Studies - of Interventions).Results Two cross-sectional surveys (n=2) and one cohort study (n=1) reported implementation of hydroxyurea for SCD management, namely regarding outcomes of adoption (n=3), cost (n=3) and acceptability (n=1). These studies were conducted exclusively among paediatric and adults populations in clinical settings in Nigeria (n=2) or Jamaica (n=1). Adoption is low, as observed through reported provider practices and patient adherence, in part shaped by misinformation and fear of side effects among patients, provider beliefs regarding affordability and organisational challenges with procuring the medicine. There was no difference in the cost of hydroxyurea therapy compared with blood transfusion in the paediatric population in urban Jamaica. Risk of bias was low or moderate across the included studies.Conclusions This review rigorously and systematically assessed the evidence on implementation of hydroxyurea in resource-constrained settings such as LMICs. Findings suggest that knowledge regarding implementation is low. To address the know-do gap and guide clinical practice, implementation research is needed. Integrating effective interventions into existing health systems to improve hydroxyurea uptake is essential to reducing SCD-associated mortality.PROSPERO registration number CRD42020155953.
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spelling doaj-art-004d23b9f4ea4b8daa761fe9c9bdff872025-08-20T02:33:01ZengBMJ Publishing GroupBMJ Open2044-60552020-11-01101110.1136/bmjopen-2020-038685Implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource-constrained settings: a systematic review of adoption, cost and acceptabilityJoyce Gyamfi0Temitope Ojo1Dorice Vieira2Nessa Ryan3Lotanna Dike4Obiageli Nnodu5Emmanuel Peprah6New York University School of Global Pubic Health, New York, NY, USASchool of Global Public Health, Department of Social and Behavioral Sciences, ISEE Lab, New York University, New York, New York, USASchool of Global Public Health, Department of Social and Behavioral Sciences, ISEE Lab, New York University, New York, New York, USASchool of Global Public Health, Department of Social and Behavioral Sciences, ISEE Lab, New York University, New York, New York, USANew York University School of Global Pubic Health, New York, NY, USACentre of Excellence for Sickle Cell Disease Research & Training (CESRTA), University of Abuja, Abuja, Nigeria16 School of Global Public Health, New York University, New York, New York, USAObjectives Mortality associated with sickle cell disease (SCD) is high in many low- and middle-income countries (LMICs). Hydroxyurea, a medicine to effectively manage SCD, is not widely available in resource-constrained settings. We identified and synthesised the reported implementation outcomes for the therapeutic use of hydroxyurea for SCD in these settings.Design Systematic review.Data sources PubMed, Embase, Cochrane, Web of Science Plus, Global Health, CINAHL, and PsycINFO were searched February through May 2019 without any restrictions on publication date.Eligibility criteria We included empirical studies of hydroxyurea for management of SCD that were carried out in LMICs and reported on implementation outcomes.Data extraction and synthesis Two reviewers independently assessed studies for inclusion, carried out data extraction using Proctor et al.’s implementation and health service outcomes, and assessed the risk of bias using ROBINS-I (Risk Of Bias In Non-randomised Studies - of Interventions).Results Two cross-sectional surveys (n=2) and one cohort study (n=1) reported implementation of hydroxyurea for SCD management, namely regarding outcomes of adoption (n=3), cost (n=3) and acceptability (n=1). These studies were conducted exclusively among paediatric and adults populations in clinical settings in Nigeria (n=2) or Jamaica (n=1). Adoption is low, as observed through reported provider practices and patient adherence, in part shaped by misinformation and fear of side effects among patients, provider beliefs regarding affordability and organisational challenges with procuring the medicine. There was no difference in the cost of hydroxyurea therapy compared with blood transfusion in the paediatric population in urban Jamaica. Risk of bias was low or moderate across the included studies.Conclusions This review rigorously and systematically assessed the evidence on implementation of hydroxyurea in resource-constrained settings such as LMICs. Findings suggest that knowledge regarding implementation is low. To address the know-do gap and guide clinical practice, implementation research is needed. Integrating effective interventions into existing health systems to improve hydroxyurea uptake is essential to reducing SCD-associated mortality.PROSPERO registration number CRD42020155953.https://bmjopen.bmj.com/content/10/11/e038685.full
spellingShingle Joyce Gyamfi
Temitope Ojo
Dorice Vieira
Nessa Ryan
Lotanna Dike
Obiageli Nnodu
Emmanuel Peprah
Implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource-constrained settings: a systematic review of adoption, cost and acceptability
BMJ Open
title Implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource-constrained settings: a systematic review of adoption, cost and acceptability
title_full Implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource-constrained settings: a systematic review of adoption, cost and acceptability
title_fullStr Implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource-constrained settings: a systematic review of adoption, cost and acceptability
title_full_unstemmed Implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource-constrained settings: a systematic review of adoption, cost and acceptability
title_short Implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource-constrained settings: a systematic review of adoption, cost and acceptability
title_sort implementation of the therapeutic use of hydroxyurea for sickle cell disease management in resource constrained settings a systematic review of adoption cost and acceptability
url https://bmjopen.bmj.com/content/10/11/e038685.full
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