Bringing the Laboratory Home: PANDABox Telehealth-Based Assessment of Neurodevelopmental Risk in Children.

Bridgette L Kelleher,Nicole Witthuhn,Leonard Abbeduto,Wei Siong Neo,Taylor Halligan,Lisa Hamrick

doi:10.3389/fpsyg.2020.01634

Bridgette L Kelleher, Nicole Witthuhn + Show 4 more

Open Access

https://doi.org/10.3389/fpsyg.2020.01634

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Abstract

BackgroundAdvances in clinical trials have revealed a pressing need for outcome measures appropriate for children with neurogenetic syndromes (NGS). However, the field lacks a standardized, flexible protocol for collecting laboratory-grade experimental data remotely. To address this challenge, we developed PANDABox (Parent-Administered Neurodevelopmental Assessment), a caregiver-facilitated, remotely administered assessment protocol for collecting integrated and high quality clinical, behavioral, and spectral data relevant to a wide array of research questions. Here, we describe PANDABox development and report preliminary data regarding: (1) logistics and cost, (2) caregiver fidelity and satisfaction, and (3) data quality.MethodsWe administered PANDABox to a cohort of 16 geographically diverse caregivers and their infants with Down syndrome. Tasks assessed attention, language, motor, and atypical behaviors. Behavioral and physiological data were synchronized and coded offline by trained research assistants.ResultsPANDABox required low resources to administer and was well received by families, with high caregiver fidelity (94%) and infant engagement (91%), as well as high caregiver-reported satisfaction (97% positive). Missing data rates were low for video frames (3%) and vocalization recordings (6%) but were higher for heart rate (25% fully missing and 13% partially missing) and discrete behavioral presses (8% technical issues and 19% not enough codable behavior), reflecting the increased technical demands for these activities.ConclusionWith further development, low-cost laboratory-grade research protocols may be remotely administered by caregivers in the family home, opening a new frontier for cost-efficient, scalable assessment studies for children with NGS other neurodevelopmental disorders.

Highlights

Assessing developmental skills in children with neurogenetic syndromes (NGS) has become a pressing need given recent advances in clinical trials and the lack of appropriate outcome measures suitable for children with intellectual and developmental disabilities (IDD) (Budimirovic et al, 2017; Grieco et al, 2019)
The present study proposes a feasible and low-cost solution to this challenge – PANDABox – a Abbreviations: ASD, autism spectrum disorders; ECG, electrocardiograph; GAC, Gwet’s AC1; GUID, NIH Global Unique Identifier; IBI, interbeat interval; IDD, intellectual and developmental disabilities; Lab-TAB, Laboratory Temperament Assessment Battery; LENA, Language ENvironment Analysis; NGS, neurogenetic syndromes; Open Science Foundation (OSF), Open Science Framework; PANDABox, Parent-Administered Neurodevelopmental Assessment; VABS-3, Vineland Adaptive Behavior Scales, 3rd edition
The PANDABox protocol was (1) well received by both caregivers and infants, (2) required low resources to administer, and (3) generated high quality, integrated clinical, behavioral, and spectral data. These data suggest that with further development, low-cost laboratory-grade research protocols may be remotely administered by caregivers in the family home, opening a new frontier for cost-efficient, scalable assessment studies for children at risk for ASD and other neurodevelopmental disorders

Summary

Introduction

Assessing developmental skills in children with NGS has become a pressing need given recent advances in clinical trials and the lack of appropriate outcome measures suitable for children with IDD (Budimirovic et al, 2017; Grieco et al, 2019). Regular in-person assessments are necessary to assess patient safety during trials Supplementing these visits with remotely administered outcome assessments may reduce the number of clinic visits required for trials, reducing both cost and participation burden for patients. These alternate assessment approaches may benefit patients with low-incidence NGS who are often widely geographically distributed and do not live near trial sites. The field lacks a standardized, flexible protocol for collecting laboratory-grade experimental data remotely To address this challenge, we developed PANDABox (ParentAdministered Neurodevelopmental Assessment), a caregiver-facilitated, remotely administered assessment protocol for collecting integrated and high quality clinical, behavioral, and spectral data relevant to a wide array of research questions. We describe PANDABox development and report preliminary data regarding: (1) logistics and cost, (2) caregiver fidelity and satisfaction, and (3) data quality

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