Content validation of observer-reported sickle cell pain diaries (SCPD-CS and SCPD-CN): results from interviews with caregivers

Michelle K. White,Denise D’Alessio,Patricia Stebbins,April M. Foster,Danielle St. Pierre,Cory Saucier,Miranda Bailey,Avery Rizio

doi:10.1186/s12955-021-01888-5

Abstract

BackgroundPatients with sickle cell disease (SCD) experience daily pain and acute episodes known as sickle cell pain crises (SCPCs). The Sickle Cell Pain Diary-Caregiver Report (SCPD-C) is an observer-reported diary for use by caregivers of children ages < 12 years with SCD. This study reports on the content validity of the SCPD-C.MethodsThe SCPD-C was developed based on a literature review, measurement expert input, and a patient advisory board including clinicians. Three rounds of interviews (including both concept elicitation and cognitive debriefing methodologies) were conducted with caregivers of children with SCD aged < 12 to evaluate the content validity of the SCPD-C.ResultsAcross three rounds of interviews, caregivers confirmed concepts in the SCPD-C and described observed impacts that were important and were added. Overall, caregivers evaluated the SCPD-C as easy to understand, with some minor adaptations for clarity. Additionally, the diary was split into two versions based on the child’s age and school enrollment status (SCPD-CS for school-aged and SCPD-CN for non-school age children).ConclusionsCaregivers provided valuable input that led to important additions and changes to the measures. The SCPD-CS and SCPD-CN are appropriate and fit-for-purpose observer-reported outcome measures of SCPC-related pain frequency and severity, and impacts on health-related quality of life.

Highlights

Sickle cell disease (SCD) is a genetic, progressive disease affecting approximately 100,000 children and adults in the United States (US) [1]
sickle cell disease (SCD) affects African Americans and Hispanic Americans in the US, as 1 out of every 365 African-Americans are born with SCD and 1 out of every 16,300 Hispanic Americans are born with SCD [1,2,3]
Cognitive debriefing A cognitive debriefing (CD) approach was used in all three rounds of interviews to test the relevance and comprehensibility of each element of the diary

Summary

Introduction

Sickle cell disease (SCD) is a genetic, progressive disease affecting approximately 100,000 children and adults in the United States (US) [1]. Clinical features of SCD include pain, fatigue, cognitive difficulty, Vaso-occlusive crises, often referred to by patients as sickle cell pain crises (SCPCs), are a hallmark complication of SCD and are associated with increased risk of morbidity and mortality [8]. SCPCs are characterized by acute, often debilitating pain, and have been reported as the most problematic complication of SCD in children and adults [5]. The Sickle Cell Pain Diary-Caregiver Report (SCPD-C) is an observer-reported diary for use by caregivers of children ages < 12 years with SCD. This study reports on the content validity of the SCPD-C

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