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. 2019 Jan 29;9(2):27.
doi: 10.3390/brainsci9020027.

A Pilot Quantitative Evaluation of Early Life Language Development in Fragile X Syndrome

Debra L Reisinger  1 , Rebecca C Shaffer  2   3 , Ernest V Pedapati  4   5   6 , Kelli C Dominick  7   8 , Craig A Erickson  9   10
Affiliations

A Pilot Quantitative Evaluation of Early Life Language Development in Fragile X Syndrome

Debra L Reisinger et al. Brain Sci. .

Abstract

Language delay and communication deficits are a core characteristic of the fragile X syndrome (FXS) phenotype. To date, the literature examining early language development in FXS is limited potentially due to barriers in language assessment in very young children. The present study is one of the first to examine early language development through vocal production and the language learning environment in infants and toddlers with FXS utilizing an automated vocal analysis system. Child vocalizations, conversational turns, and adult word counts in the home environment were collected and analyzed in a group of nine infants and toddlers with FXS and compared to a typically developing (TD) normative sample. Results suggest infants and toddlers with FXS are exhibiting deficits in their early language skills when compared to their chronological expectations. Despite this, when accounting for overall developmental level, their early language skills appear to be on track. Additionally, FXS caregivers utilize less vocalizations around infants and toddlers with FXS; however, additional research is needed to understand the true gap between FXS caregivers and TD caregivers. These findings provide preliminary information about the early language learning environment and support for the feasibility of utilizing an automated vocal analysis system within the FXS population that could ease data collection and further our understanding of the emergence of language development.

Keywords: automated vocal analysis; fragile X syndrome; language development.

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Conflict of interest statement

R.C.S. receives funding from Fulcrum Therapeutics. C.A.E. has received current or past funding from Confluence Pharmaceuticals, Novartis, F. Hoffmann-La Roche Ltd., Seaside Therapeutics, Riovant Sciences, Inc., Fulcrum Therapeutics, Neuren Pharmaceuticals Ltd., Alcobra Pharmaceuticals, Neurotrope, Zynerba Pharmaceuticals, Inc., and Ovid Therapeutics Inc. to consult on trial design or development strategies and/or conduct clinical trials in FXS or other neurodevelopmental disorders. C.A.E. is additionally the inventor or co-inventor on several patents held by Cincinnati Children’s Hospital Medical Center or Indiana University School of Medicine describing methods of treatment in FXS or other neurodevelopmental disorders. E.V.P. has received research support by the National Institutes of Health (NIMH), American Academy of Child and Adolescent Psychiatry, and Cincinnati Children’s Hospital Research Foundation. He is a clinical trial site investigator for the Marcus Autism Center (clinical trial, Autism). He receives compensation for consulting for Proctor & Gamble. He receives book royalties from Springer. There are no conflicts of interest with the current manuscript. K.C.D. has received research support from the National Institute of Neurological Disorders and Stroke (NINDS), American Academy of Child and Adolescent Psychiatry, and Cincinnati Children’s Hospital Medical Center. She is a clinical trial site investigator for F. Hoffman-La Roche Ltd. and Ovid Therapeutics. There are no conflicts of interest for the current manuscript.

Figures

Figure 1
Figure 1
Infants and toddlers with fragile X syndrome (FXS) vocalizations per hour plotted in comparison to their chronologically age-matched (A) and developmentally age-matched (B) typically developing (TD) peers with trend lines.
Figure 2
Figure 2
Infants and toddlers with fragile X syndrome (FXS) conversational turns per hour plotted in comparison to their chronologically age-matched (A) and developmentally age-matched (B) typically developing (TD) peers with trend lines.
Figure 3
Figure 3
Caregivers’ of infants and toddlers with FXS adult word count per hour plotted in comparison to the average adult word count per hour in TD caregivers (M = 1024.75).
See this image and copyright information in PMC

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