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Accepted for/Published in: JMIR mHealth and uHealth

Date Submitted: Jan 21, 2019
Open Peer Review Period: Jan 25, 2019 - Mar 14, 2019
Date Accepted: Apr 23, 2019
(closed for review but you can still tweet)

The final, peer-reviewed published version of this preprint can be found here:

A Multidimensional Electronic Hydroxyurea Adherence Intervention for Children With Sickle Cell Disease: Single-Arm Before-After Study

Creary S, Chisolm D, Stanek J, Hankins J, O'Brien SH

A Multidimensional Electronic Hydroxyurea Adherence Intervention for Children With Sickle Cell Disease: Single-Arm Before-After Study

JMIR Mhealth Uhealth 2019;7(8):e13452

DOI: 10.2196/13452

PMID: 31397291

PMCID: 6705009

ENHANCE—(ElectroNic Hydroxyurea AdhereNCE): A multi-dimensional, electronic adherence intervention that improves hydroxyurea adherence in children with sickle cell disease

  • Susan Creary; 
  • Deena Chisolm; 
  • Joseph Stanek; 
  • Jane Hankins; 
  • Sarah H O'Brien

ABSTRACT

Background:

Hydroxyurea is a disease-modifying medication for patients with sickle cell disease (SCD). Despite demonstrated efficacy, hydroxyurea non-adherence in clinical practice is common and results in worse health outcomes for non-adherent patients. Mobile Directly Observed Therapy (Mobile DOT) is a pilot-tested, electronic, multidimensional hydroxyurea adherence intervention for children with SCD. Mobile DOT includes sending daily text message reminders to take hydroxyurea, participants recording and sending daily videos that captured their hydroxyurea administrations for the research team to review, providing electronic personalized feedback to participants about their adherence, and providing small monetary incentives to participants if they achieve high hydroxyurea adherence.

Objective:

The aim of this study was to determine if Mobile DOT increases hydroxyurea adherence in children with SCD and explore its impact on hematologic and clinical outcomes.

Methods:

This was a single-arm, 6-month intervention study of patients with SCD on hydroxyurea who were ≤19 years and reported daily access to an electronic device. Participants’ hydroxyurea adherence when their received Mobile DOT was compared to their adherence 6 months before and after receiving Mobile DOT. Adherence was measured using pharmacy dispensing records and laboratory and clinical outcomes were tracked using participants’ electronic medical records.

Results:

55 (60.4%) children who were sequentially approached enrolled and 34 engaged with the 6-month intervention. With Mobile DOT, engaged participants’ median hydroxyurea adherence rate increased from 61.7% to 84.4% (p<0.001) and significantly more (66.7% vs. 30.3%, P= .002) achieved ≥80% hydroxyurea adherence compared to baseline. Engaged participants’ laboratory studies, including their median mean corpuscular volume (MCV) and fetal hemoglobin (HgbF) levels, improved significantly with Mobile DOT (P= .001 and P= .04, respectively) but adherence, MCV, and HgbF returned to baseline 6 months after discontinuing the intervention. Hospitalizations and other measured clinical outcomes occurred infrequently during the entire study. Non-engagement was associated with being female and having a recent SCD complication and not having sufficient electronic data, not being able to quickly complete Mobile DOT each day, and not perceiving benefit from the intervention may have also decreased engagement.

Conclusions:

Mobile DOT shows promise as an effective intervention for some children with SCD. Modifications that increase engagement and sustain high adherence could increase the impact that Mobile DOT has on clinical outcomes in children with SCD.


 Citation

Please cite as:

Creary S, Chisolm D, Stanek J, Hankins J, O'Brien SH

A Multidimensional Electronic Hydroxyurea Adherence Intervention for Children With Sickle Cell Disease: Single-Arm Before-After Study

JMIR Mhealth Uhealth 2019;7(8):e13452

DOI: 10.2196/13452

PMID: 31397291

PMCID: 6705009

Per the author's request the PDF is not available.

© The authors. All rights reserved. This is a privileged document currently under peer-review/community review (or an accepted/rejected manuscript). Authors have provided JMIR Publications with an exclusive license to publish this preprint on it's website for review and ahead-of-print citation purposes only. While the final peer-reviewed paper may be licensed under a cc-by license on publication, at this stage authors and publisher expressively prohibit redistribution of this draft paper other than for review purposes.