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New insights into a spectrum of developmental malformations related to mTOR dysregulations: challenges and
A Mühlebner1, A Bongaarts1, H B Sarnat2
1Department of Neuropathology, Amsterdam UMC, University of Amsterdam, Amsterdam, The Netherlands.
Abstract:
In recent years the role of the mammalian target of rapamycin (mTOR) pathway has emerged as crucial for normal cortical development. Therefore, it is not surprising that aberrant activation of mTOR is associated with developmental malformations and epileptogenesis. A broad spectrum of malformations of cortical development, such as focal cortical dysplasia (FCD) and tuberous sclerosis complex (TSC), have been linked to either germline or somatic mutations in mTOR pathway-related genes, commonly summarised under the umbrella term 'mTORopathies'. However, there are still a number of unanswered questions regarding the involvement of mTOR in the pathophysiology of these abnormalities. Therefore, a monogenetic disease, such as TSC, can be more easily applied as a model to study the mechanisms of epileptogenesis and identify potential new targets of therapy. Developmental neuropathology and genetics demonstrate that FCD IIb and hemimegalencephaly are the same diseases. Constitutive activation of mTOR signalling represents a shared pathogenic mechanism in a group of developmental malformations that have histopathological and clinical features in common, such as epilepsy, autism and other comorbidities. We seek to understand the effect of mTOR dysregulation in a developing cortex with the propensity to generate seizures as well as the aftermath of the surrounding environment, including the white matter.
Insights
Dysregulation of the mammalian target of rapamycin (mTOR) pathway causes cortical developmental malformations and epilepsy. Studying tuberous sclerosis complex (TSC) offers insights into mTOR
Area of Science:
- Neuroscience
- Developmental Biology
- Genetics
Background:
- The mammalian target of rapamycin (mTOR) pathway is vital for normal cortical development.
- Aberrant mTOR activation is linked to developmental brain malformations and epilepsy.
- Conditions like focal cortical dysplasia (FCD) and tuberous sclerosis complex (TSC) are known as 'mTORopathies' due to mutations in related genes.
Purpose of the Study:
- To investigate the role of mTOR dysregulation in cortical development and seizure generation.
- To explore the impact of mTOR pathway abnormalities on the surrounding brain environment, including white matter.
- To utilize monogenetic diseases like TSC as models for understanding epileptogenesis and identifying therapeutic targets.
Main Methods:
- Review of existing literature on mTOR pathway involvement in cortical malformations.
- Analysis of genetic and neuropathological data from conditions such as FCD and TSC.
- Comparative study of mTORopathies to understand shared pathogenic mechanisms.
Main Results:
- Constitutive mTOR signaling activation is a common mechanism in developmental malformations with shared features like epilepsy and autism.
- Focal cortical dysplasia type IIb and hemimegalencephaly are identified as the same disease based on developmental neuropathology and genetics.
- Understanding mTOR's effect on the developing cortex and its environment is crucial for comprehending seizure propensity.
Conclusions:
- Dysregulation of the mTOR pathway is a key factor in a spectrum of developmental brain disorders.
- Shared mechanisms link mTORopathies, highlighting potential common therapeutic strategies.
- Further research into mTOR's role is essential for developing novel treatments for epilepsy and related comorbidities.
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