Author response: Ciliary neuropeptidergic signaling dynamically regulates excitatory synapses in postnatal neocortical pyramidal neurons

Tereshko, Lauren; Gao, Ya; Cary, Brian A; Turrigiano, Gina G.; Sengupta, Piali

doi:10.7554/elife.65427.sa2

2021

DOI: 10.7554/elife.65427.sa2

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Author response: Ciliary neuropeptidergic signaling dynamically regulates excitatory synapses in postnatal neocortical pyramidal neurons

Lauren Tereshko

Ya Gao

Brian A Cary

et al.

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2024

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Shared and unique consequences of Joubert Syndrome gene dysfunction on the zebrafish central nervous system

Noble,

Masek,

Hofmann

et al. 2024

Preprint

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Joubert Syndrome (JBTS) is a neurodevelopmental ciliopathy defined by a highly specific midbrain-hindbrain malformation, variably associated with additional neurological features. JBTS displays prominent genetic heterogeneity with >40 causative genes that encode proteins localising to the primary cilium, a sensory organelle that is essential for transduction of signalling pathways during neurodevelopment, among other vital functions. JBTS proteins localise to distinct ciliary subcompartments, suggesting diverse functions in cilium biology. Currently, there is no unifying pathomechanism to explain how dysfunction of such diverse primary cilia-related proteins results in such a highly specific brain abnormality. In order to identify the shared consequence of JBTS gene dysfunction, we carried out transcriptomic analysis using zebrafish mutants for the JBTS-causative genes cc2d2aw38, cep290fh297, inpp5ezh506, talpid3i264 and togaram1zh510 and the Bardet-Biedl syndrome-causative gene bbs1k742. We identified no commonly dysregulated signalling pathways in these mutants and yet all mutants displayed an enrichment of altered gene sets related to central nervous system function. We found that JBTS mutants have altered primary cilia throughout the brain, however do not display abnormal brain morphology. Nonetheless, behavioural analyses revealed reduced locomotion and loss of postural control which, together with the transcriptomic results, hint at underlying abnormalities in neuronal activity and/or neuronal circuit function. These zebrafish models therefore offer the unique opportunity to study the role of primary cilia in neuronal function beyond early patterning, proliferation and differentiation.

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Shared and unique consequences of Joubert Syndrome gene dysfunction on the zebrafish central nervous system

Noble,

Masek,

Hofmann

et al. 2024

Preprint

View full text Add to dashboard Cite

show abstract

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Author response: Ciliary neuropeptidergic signaling dynamically regulates excitatory synapses in postnatal neocortical pyramidal neurons

Cited by 1 publication

References 0 publications

Shared and unique consequences of Joubert Syndrome gene dysfunction on the zebrafish central nervous system

Shared and unique consequences of Joubert Syndrome gene dysfunction on the zebrafish central nervous system

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