Atypical Auditory Brainstem Response and Protein Expression Aberrations Related to ASD and Hearing Loss in the Adnp Haploinsufficient Mouse Brain.
Gal Hacohen-KleimanOfer Yizhar-BarneaOlga TouloumiRoza LagoudakiKaren B AvrahamNikolaos GrigoriadisIllana GozesPublished in: Neurochemical research (2019)
Autism is a wide spread neurodevelopmental disorder with growing morbidity rates, affecting more boys than girls worldwide. Activity-dependent neuroprotective protein (ADNP) was recently recognized as a leading gene accounted for 0.17% of autism spectrum disorder (ASD) cases globally. Respectively, mutations in the human ADNP gene (ADNP syndrome), cause multi-system body dysfunctions with apparent ASD-related traits, commencing as early as childhood. The Adnp haploinsufficient (Adnp+/-) mouse model was researched before in relations to Alzheimer's disease and autism. Adnp+/- mice suffer from deficient social memory, vocal and motor impediments, irregular tooth eruption and short stature, all of which corresponds with reported phenotypes in patients with the ADNP syndrome. Recently, a more elaborated description of the ADNP syndrome was published, presenting impediments such as hearing disabilities in >ā10% of the studied children. Irregular auditory brainstem response (ABR) has been connected to ASD-related cases and has been suggested as a potential hallmark for autism, allowing diagnosis of ASD risk and early intervention. Herein, we present detriment hearing in the Adnp+/- mice with atypical ABR and significant protein expression irregularities that coincides with ASD and hearing loss studies in the brain.
Keyphrases
- autism spectrum disorder
- hearing loss
- intellectual disability
- attention deficit hyperactivity disorder
- case report
- mouse model
- working memory
- copy number
- endothelial cells
- type diabetes
- young adults
- magnetic resonance imaging
- systematic review
- computed tomography
- mental health
- small molecule
- magnetic resonance
- risk assessment
- skeletal muscle
- blood brain barrier
- induced pluripotent stem cells
- transcription factor
- amino acid