Developmental Skills of Individuals with Angelman Syndrome Assessed Using the Bayley-III

Anjali Sadhwani; Anne Wheeler; Angela Gwaltney; Sarika U Peters; Rene L Barbieri-Welge; Lucia T Horowitz; Lisa M Noll; Rachel J Hundley; Lynne M Bird; Wen-Hann Tan

doi:10.1007/s10803-020-04861-1

Developmental Skills of Individuals with Angelman Syndrome Assessed Using the Bayley-III

J Autism Dev Disord. 2023 Feb;53(2):720-737. doi: 10.1007/s10803-020-04861-1. Epub 2021 Jan 30.

Authors

Anjali Sadhwani^#^{1

2}, Anne Wheeler^#³, Angela Gwaltney³, Sarika U Peters⁴, Rene L Barbieri-Welge⁵, Lucia T Horowitz⁶, Lisa M Noll^{7

8}, Rachel J Hundley⁴, Lynne M Bird^#^{9

10}, Wen-Hann Tan^#^{11

12}

Affiliations

¹ Department of Psychiatry, Boston Children's Hospital, Boston, MA, 02115, USA. Anjali.sadhwani@childrens.harvard.edu.
² Harvard Medical School, Boston, MA, USA. Anjali.sadhwani@childrens.harvard.edu.
³ RTI International, Research Triangle Park, NC, USA.
⁴ Department of Pediatrics, Vanderbilt University Medical Center, Nashville, TN, USA.
⁵ Developmental Evaluation Clinic, Rady Children's Hospital - San Diego, San Diego, CA, USA.
⁶ Greenwood Genetic Center, Greenwood, SC, USA.
⁷ Psychology Service, Texas Children's Hospital, Houston, TX, USA.
⁸ Baylor College of Medicine, Houston, TX, USA.
⁹ Division of Dysmorphology/Genetics, Rady Children's Hospital - San Diego, San Diego, CA, USA.
¹⁰ Department of Pediatrics, University of California, San Diego, San Diego, CA, USA.
¹¹ Harvard Medical School, Boston, MA, USA.
¹² Division of Genetics and Genomics, Boston Children's Hospital, Boston, MA, 02115, USA.

^# Contributed equally.

Abstract

We describe the development of 236 children with Angelman syndrome (AS) using the Bayley Scales of Infant and Toddler Development, Third Edition. Multilevel linear mixed modeling approaches were used to explore differences between molecular subtypes and over time. Individuals with AS continue to make slow gains in development through at least age 12 years of age at about 1-2 months/year based on age equivalent score and 1-16 growth score points/year depending on molecular subtype and domain. Children with a deletion have lower scores at baseline and slower rate of gaining skills while children with UBE3A variant subtype demonstrated higher scores as well as greater rates of skill attainment in all domains. The developmental profiles of UPD and ImpD were similar.

Keywords: Child development; Developmental disabilities; Language development disorders; Motor skills disorders; Neurodevelopmental disorders.

MeSH terms

Angelman Syndrome* / diagnosis
Angelman Syndrome* / genetics
Autism Spectrum Disorder*
Child
Child Development
Developmental Disabilities / diagnosis
Humans
Infant
Motor Skills

Abstract

MeSH terms

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