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Music Therapy for Children With Autistic Spectrum Disorder and/or Other Neurodevelopmental Disorders: A Systematic Review.

ABSTRACT: Background: Several studies have reported contradictory results regarding the benefits of music interventions in children and adolescents with neurodevelopmental disorders (NDDs), including autism spectrum disorder (ASD). Methods: We performed a systematic review according to the PRISMA guidelines. We searched the Cochrane, PubMed and Medline databases from January 1970 to September 2020 to review all empirical findings, except case reports, measuring the effect of music therapy on youths with ASD, intellectual disability (ID), communication disorder (CD), developmental coordination disorder (DCD), specific learning disorder, and attention/deficit hyperactivity disorder (ADHD). Results: Thirty-nine studies (N = 1,774 participants) were included in this review (ASD: n = 22; ID: n = 7; CD and dyslexia: n = 5; DCD: n = 0; ADHD: n = 5 studies). Two main music therapies were used: educational music therapy and improvisational music therapy. A positive effect of educational music therapy on patients with ASD was reported in most controlled studies (6/7), particularly in terms of speech production. A positive effect of improvisational music therapy was reported in most controlled studies (6/8), particularly in terms of social functioning. The subgroup of patients with both ASD and ID had a higher response rate. Data are lacking for children with other NDDs, although preliminary evidence appears encouraging for educational music therapy in children with dyslexia. Discussion: Improvisational music therapy in children with NDDs appears relevant for individuals with both ASD and ID. More research should be encouraged to explore whether oral and written language skills may improve after educational music therapy, as preliminary data are encouraging.

SUBMITTER: Mayer-Benarous H

PROVIDER: S-EPMC8062803 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:Autism spectrum disorders (ASD) represent a phenotypically heterogeneous group of patients that strongly intertwine with other neurodevelopmental disorders (NDDs), with genetics playing a significant role in their etiology. Whole exome sequencing (WES) has become predominant in molecular diagnostics for ASD by considerably increasing the diagnostic yield. However, the proportion of undiagnosed patients still remains high due to complex clinical presentation, reduced penetrance, and lack of segregation analysis or clinical information. Thus, reverse phenotyping, where we first identified a possible genetic cause and then determine its clinical relevance, has been shown to be a more efficient approach. WES was performed on 147 Slovenian pediatric patients with suspected ASD. Data analysis was focused on identifying ultrarare or "single event" variants in ASD-associated genes and further expanded to NDD-associated genes. Protein function and gene prioritization were performed on detected clinically relevant variants to determine their role in ASD etiology and phenotype. Reverse phenotyping revealed a pathogenic or likely pathogenic variant in ASD-associated genes in 20.4% of patients, with subsequent segregation analysis indicating that 14 were de novo variants and 1 was presumed compound heterozygous. The diagnostic yield was further increased by 2.7% by the analysis of ultrarare or "single event" variants in all NDD-associated genes. Protein function analysis established that genes in which variants of unknown significance (VUS) were detected were predominantly the cause of intellectual disability (ID), and in most cases, features of ASD as well. Using such an approach, variants in rarely described ASD-associated genes, such as SIN3B, NR4A2, and GRIA1, were detected. By expanding the analysis to include functionally similar NDD genes, variants in KCNK9, GNE, and other genes were identified. These would probably have been missed by classic genotype-phenotype analysis. Our study thus demonstrates that in patients with ASD, analysis of ultrarare or "single event" variants obtained using WES with the inclusion of functionally similar genes and reverse phenotyping obtained a higher diagnostic yield despite limited clinical data. The present study also demonstrates that most of the causative genes in our cohort were involved in the syndromic form of ASD and confirms their comorbidity with other developmental disorders.

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Music Therapy for Children With Autistic Spectrum Disorder and/or Other Neurodevelopmental Disorders: A Systematic Review.

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