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Analysis of TWIST1’s role in meningeal and cortical development
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Ho-Nguyen, Khue Tu. Analysis of TWIST1’s role in meningeal and cortical development. Retrieved from https://doi.org/doi:10.7282/t3-760g-ss09

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TitleAnalysis of TWIST1’s role in meningeal and cortical development
NameHo-Nguyen, Khue Tu (author); Tischfield, Max (chair); Abraira, Victoria (member); Zhang, Huaye (member); Shumyatsky, Gleb (member); Rutgers University; School of Graduate Studies
Date Created2022
Other Date2022-10 (degree)
SubjectNeurosciences, Developmental biology, Cortical development, Cortical gyrification, Craniosynostosis, Embryonic, Meningeal development
Extent1 online resource (85 pages) : illustrations
DescriptionMeningeal development and cortical development are intertwined processes. We previously reported hypoplastic meningeal development in a Twist1 mouse model of craniosynostosis, a skull malformation with high instances of neurodevelopmental delays even with early surgical interventions. It is unknown how the loss of Twist1 loss meningeal development and whether Twist1 loss has a secondary effect on neurodevelopment. In my work, I show that the loss of Twist1 in the mesenchyme and meninges leads to a decrease in proliferation of these cells, particularly in the dorsolateral forebrain. Changes to early meningeal development caused by Twist loss has a profound effect on corticogenesis, in which the dorsolateral cortex develops cortical folding structures resembling gyrification during late corticogenesis that persists into adulthood, giving a normally smooth mouse brain an abnormally bumpy appearance. Embryonic cortical folding develops independent of neural progenitor proliferation and have abnormal neuronal distribution in the radial direction, with higher propensity to be closer to the pia. The early appearance of gyrus/sulcus is temporally correlated with a localized reduction in meningeal retinoic acid signaling at E16.5. Maternal supplementation of retinoic acid partially rescues the macroscopic bumps associated with loss of Twist1 in the meninges. Taken together, my results show that Twist1’s role in meningeal proliferation is important to normal cortical development and contributes to the maintenance of secondary lissencephaly in mice.
NotePh.D.
NoteIncludes bibliographical references
Genretheses
Persistent URLhttps://doi.org/doi:10.7282/t3-760g-ss09
LanguageEnglish
CollectionSchool of Graduate Studies Electronic Theses and Dissertations
Organization NameRutgers, The State University of New Jersey
RightsThe author owns the copyright to this work.
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