Binaural interaction component of the auditory brainstem response in children with autism spectrum disorder

Elmoazen, Doaa; Sobhy, Ossama; Abdou, Rania M.; Abdelmotaleb, HebatAllah

doi:10.1016/j.ijporl.2019.109850

Cited by 17 publications

(12 citation statements)

References 33 publications

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“…Additionally, consistent with previous study, the BIC latency gets longer with increasing ITD (Ferber et al, 2016;Laumen et al, 2016;Benichoux et al, 2018). Interestingly, our work in mice with FXS is consistent with an increased latency of the BIC seen in a study in autistic people (ElMoazen et al, 2019), although they also see a decrease in the amplitude of the BIC. Our findings that the BIC latency is only significant in males at 0 ITD potentially suggest that there is overall slowing of binaural processing in the brain stem, which may ultimately impact binaural hearing, but that it is not dependent on ITD, which would be consistent with mice that do not rely as predominantly on ITD cues compared with other species.…”

Section: Discussionsupporting

confidence: 92%

Auditory Brain Stem Responses in the C57BL/6J Fragile X Syndrome-Knockout Mouse Model

Chawla

McCullagh

2022

Front. Integr. Neurosci.

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Sensory hypersensitivity, especially in the auditory system, is a common symptom in Fragile X syndrome (FXS), the most common monogenic form of intellectual disability. However, linking phenotypes across genetic background strains of mouse models has been a challenge and could underly some of the issues with translatability of drug studies to the human condition. This study is the first to characterize the auditory brain stem response (ABR), a minimally invasive physiological readout of early auditory processing that is also used in humans, in a commonly used mouse background strain model of FXS, C57BL/6J. We measured morphological features of pinna and head and used ABR to measure the hearing range, and monaural and binaural auditory responses in hemizygous males, homozygous females, and heterozygous females compared with those in wild-type mice. Consistent with previous study, we showed no difference in morphological parameters across genotypes or sexes. There was no significant difference in hearing range between the sexes or genotypes, however there was a trend towards high frequency hearing loss in male FXS mice. In contrast, female mice with homozygous FXS had a decreased amplitude of wave IV of the monaural ABR, while there was no difference in males for amplitudes and no change in latency of ABR waveforms across sexes and genotypes. Finally, males with FXS had an increased latency of the binaural interaction component (BIC) at 0 interaural timing difference compared with that in wild-type males. These findings further clarify auditory brain stem processing in FXS by adding more information across genetic background strains allowing for a better understanding of shared phenotypes.

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Section: Discussionsupporting

confidence: 92%

Auditory Brain Stem Responses in the C57BL/6J Fragile X Syndrome-Knockout Mouse Model

Chawla

McCullagh

2022

Front. Integr. Neurosci.

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show abstract

“…Additionally, consistent with previous work, the BIC latency gets longer with increasing ITD (Ferber et al, 2016;Laumen et al, 2016;Benichoux et al, 2018). Interestingly our work in FXS mice is consistent with increased latency of the BIC seen in a study in autistic people (ElMoazen et al, 2019), though they also see a decrease in amplitude of the BIC. Our findings that the BIC latency is only significant in males at 0 ITD potentially suggests that there is overall slowing of binaural processing in the brainstem, but that it is not dependent on ITD, which would be consistent with mice that do not rely as predominantly on ITD cues compared to other species.…”

Section: Binaural Hearingsupporting

confidence: 92%

Auditory Brainstem Responses in the C57BL/6J Fragile X Syndrome Knockout Mouse Model

Chawla

McCullagh

2021

Preprint

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Sensory hypersensitivity, especially in the auditory system, is a common symptom in Fragile X Syndrome (FXS), the most common monogenic form of intellectual disability. However, linking phenotypes across genetic background strains of mouse models has been a challenge and could underly some of the issues with translatability of drug studies to the human condition. This study is the first to characterize the auditory brainstem response (ABR), a minimally invasive physiological readout of early auditory processing that is also used in humans, in a commonly used mouse background strain model of FXS, C57BL/6J. We measured morphological features of pinna and head and used ABR to measure hearing range, monaural and binaural auditory responses in hemizygous males, homozygous females and heterozygous females compared to wildtype mice. Consistent with previous work we showed no difference in morphological parameters across genotypes or sexes. Male FXS mice had increased threshold for high frequency hearing at 64 kHz compared to wildtype males, while females had no difference in hearing range between genotypes. In contrast, female homozygous FXS mice had decreased amplitude of wave IV of the monaural ABR, while there was no difference in males for amplitudes and no change in latency of ABR waveforms across sexes and genotypes. Lastly, FXS males had increased latency of the binaural interaction component (BIC) at 0 ITD compared to wildtype males. These findings further clarify auditory brainstem processing in FXS by adding more information across genetic background strains allowing for a better understanding of shared phenotypes.

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“…These longer latency and lower amplitude responses have been attributed to the immaturity of brainstem circuits (Li et al, 2020 ). A recent study showed delays in speech-based ABRs (Chen et al, 2019 ) and reduced binaural interaction components (BIC) of the ABR in subjects with ASD (ElMoazen et al, 2020 ). The latter study also found a significant positive correlation between the amplitude of the BIC ABR waveform and both language and social scores in subjects with ASD (ElMoazen et al, 2020 ).…”

Section: Introductionmentioning

confidence: 99%

Central Auditory and Vestibular Dysfunction Are Key Features of Autism Spectrum Disorder

Mansour

Burchell

Kulesza

2021

Front. Integr. Neurosci.

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Autism spectrum disorder (ASD) is a neurodevelopmental disorder characterized by repetitive behaviors, poor social skills, and difficulties with communication. Beyond these core signs and symptoms, the majority of subjects with ASD have some degree of auditory and vestibular dysfunction. Dysfunction in these sensory modalities is significant as normal cognitive development depends on an accurate representation of our environment. The hearing difficulties in ASD range from deafness to hypersensitivity and subjects with ASD have abnormal sound-evoked brainstem reflexes and brainstem auditory evoked potentials. Vestibular dysfunction in ASD includes postural instability, gait dysfunction, and impaired gaze. Untreated vestibular dysfunction in children can lead to delayed milestones such as sitting and walking and poor motor coordination later in life. Histopathological studies have revealed that subjects with ASD have significantly fewer neurons in the auditory hindbrain and surviving neurons are smaller and dysmorphic. These findings are consistent with auditory dysfunction. Further, the cerebellum was one of the first brain structures implicated in ASD and studies have revealed loss of Purkinje cells and the presence of ectopic neurons. Together, these studies suggest that normal auditory and vestibular function play major roles in the development of language and social abilities, and dysfunction in these systems may contribute to the core symptoms of ASD. Further, auditory and vestibular dysfunction in children may be overlooked or attributed to other neurodevelopmental disorders. Herein we review the literature on auditory and vestibular dysfunction in ASD. Based on these results we developed a brainstem model of central auditory and vestibular dysfunction in ASD and propose that simple, non-invasive but quantitative testing of hearing and vestibular function be added to newborn screening protocols.

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Binaural interaction component of the auditory brainstem response in children with autism spectrum disorder

Cited by 17 publications

References 33 publications

Auditory Brain Stem Responses in the C57BL/6J Fragile X Syndrome-Knockout Mouse Model

Auditory Brain Stem Responses in the C57BL/6J Fragile X Syndrome-Knockout Mouse Model

Auditory Brainstem Responses in the C57BL/6J Fragile X Syndrome Knockout Mouse Model

Central Auditory and Vestibular Dysfunction Are Key Features of Autism Spectrum Disorder

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