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Insights into Human Neurodegeneration: Lessons Learnt from Drosophila pp 55–88Cite as

Understanding Neurodegeneration and Neuroprotection Through Genetic Screens in Drosophila

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Abstract

Drosophila genetic screens have been invaluable in understanding neurodegenerative diseases (NDD) and neuronal maintenance. The modeling of several human neurodegenerative diseases such as Alzheimer’s and Parkinson’s in Drosophila and subsequent modifier genetic screens for neurodegenerative phenotypes have been instrumental in identifying the molecular mechanisms of neurodegeneration as well as the cellular function of genes implicated in neurodegeneration. For instance, studies on Drosophila homologs of PINK1 and PARKIN, genes implicated in Parkinson’s disease, identified their roles in mitochondrial quality control. Interestingly, unbiased genetic screens for fly mutants with neurodegenerative phenotypes have also identified many genes implicated in neurodegenerative diseases and have led to the discovery of novel players regulating neuronal health and maintenance. Drosophila has emerged as a valuable screening platform for validating the pathogenicity of variants identified through whole-genome sequencing of patients with neurodegenerative diseases and has thus fast-tracked the identification of causative mutations. With rapid and consistent development of genome editing technologies, together with amenability for genetic screens, Drosophila will continue to serve as a great system to study neurodegeneration.

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Abbreviations

ALS:

Amyotrophic lateral sclerosis

APOE:

Apolipoprotein E

APP:

Amyloid precursor protein

CMT:

Charcot-Marie-Tooth disease

EMS:

Ethyl methyl sulfonate

ER:

Endoplasmic reticulum

ERG:

Electroretinogram

FATP:

Fatty acid transport proteins

FHM:

Familial hemiplegic migraine

iPSC:

Induced pluripotent stem cells

LD:

Lipid droplet

MCTs:

Monocarboxylate transporters

ND:

Neurodegeneration/neurodegenerative

NDD:

Neurodegenerative diseases

NGS:

Next-generation sequencing

NTE:

Neuropathy target esterase

PD:

Parkinson’s disease

PDF:

Pigment-dispersing factor

RDP:

Rapid-onset dystonia-parkinsonism

TCA Cycle:

Tricarboxylic acid cycle

UAS:

Upstream activating sequence

UPRmt:

Mitochondrial unfolded protein response

WES:

Whole exome sequencing

WGS:

Whole-genome sequencing

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Acknowledgments

We thank Neha Singhal, Nele Haelterman, and Megan Campbell for critical comments. MJ is supported by intramural funding from TIFR and Ramalingaswami Fellowship, DBT, India.

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  1. Aishwarya S Mandya and Rajit Narayanan Cheramangalam contributed equally with all other contributors.

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  1. Tata Institute of Fundamental Research Hyderabad, Hyderabad, Telangana, India

    Aishwarya S Mandya, Rajit Narayanan Cheramangalam & Manish Jaiswal

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  1. Department of Molecular and Human Genetics, Banaras Hindu University, Varanasi, Uttar Pradesh, India

    Mousumi Mutsuddi

  2. Department of Molecular and Human Genetics, Banaras Hindu University, Varanasi, Uttar Pradesh, India

    Ashim Mukherjee

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Mandya, A.S., Cheramangalam, R.N., Jaiswal, M. (2019). Understanding Neurodegeneration and Neuroprotection Through Genetic Screens in Drosophila. In: Mutsuddi, M., Mukherjee, A. (eds) Insights into Human Neurodegeneration: Lessons Learnt from Drosophila. Springer, Singapore. https://doi.org/10.1007/978-981-13-2218-1_3

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