Modeling Parkinson’s Disease in Drosophila: What Have We Learned for Dominant Traits?

α-synuclein Induces Mitochondrial Dysfunction through Spectrin and the Actin Cytoskeleton

(2018) Dalila G. Ordonez et al.   NEURON

Robust kinase- and age-dependent dopaminergic and norepinephrine neurodegeneration in LRRK2 G2019S transgenic mice

(2018) Yulan Xiong et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Vps35 in cooperation with LRRK2 regulates synaptic vesicle endocytosis through the endosomal pathway in Drosophila

(2017) Tsuyoshi Inoshita et al.   HUMAN MOLECULAR GENETICS

Parkinson's Disease Is Not Simply a Prion Disorder

(2017) D. James Surmeier et al.   JOURNAL OF NEUROSCIENCE

Prying into the Prion Hypothesis for Parkinson's Disease

(2017) Patrik Brundin et al.   JOURNAL OF NEUROSCIENCE

α-synuclein toxicity in neurodegeneration: mechanism and therapeutic strategies

(2017) Yvette C Wong et al.   NATURE MEDICINE

Animal models of α-synucleinopathy for Parkinson disease drug development

(2017) James B. Koprich et al.   NATURE REVIEWS NEUROSCIENCE

The Complicated Relationship between Gaucher Disease and Parkinsonism: Insights from a Rare Disease

(2017) Elma Aflaki et al.   NEURON

A novel iron (II) preferring dopamine agonist chelator D-607 significantly suppresses α-syn- and MPTP-induced toxicities in vivo

(2017) Banibrata Das et al.   NEUROPHARMACOLOGY

VPS35, the Retromer Complex and Parkinson’s Disease

(2017) Erin T. Williams et al.   Journal of Parkinsons Disease

Ambroxol effects in glucocerebrosidase and α-synuclein transgenic mice

(2016) Anna Migdalska-Richards et al.   ANNALS OF NEUROLOGY

Lovastatin protects neurite degeneration inLRRK2-G2019Sparkinsonism through activating the Akt/Nrf pathway and inhibiting GSK3β activity

(2016) Chin-Hsien Lin et al.   HUMAN MOLECULAR GENETICS

The contribution of mutantGBAto the development of Parkinson disease inDrosophila

(2016) Gali Maor et al.   HUMAN MOLECULAR GENETICS

Human R1441C LRRK2 regulates the synaptic vesicle proteome and phosphoproteome in aDrosophilamodel of Parkinson’s disease

(2016) Md Shariful Islam et al.   HUMAN MOLECULAR GENETICS

Impaired striatal dopamine release in homozygous Vps35 D620N knock-in mice

(2016) Nobutaka Ishizu et al.   HUMAN MOLECULAR GENETICS

Rab7 induces clearance of α-synuclein aggregates

(2016) Elisabeth Dinter et al.   JOURNAL OF NEUROCHEMISTRY

A Drosophila Model of Neuronopathic Gaucher Disease Demonstrates Lysosomal-Autophagic Defects and Altered mTOR Signalling and Is Functionally Rescued by Rapamycin

(2016) Kerri J. Kinghorn et al.   JOURNAL OF NEUROSCIENCE

Melatonin attenuates hLRRK2-induced sleep disturbances and synaptic dysfunction in a Drosophila model of Parkinson's disease

(2016) XICUI SUN et al.   Molecular Medicine Reports

Defects in trafficking bridge Parkinson's disease pathology and genetics

(2016) Asa Abeliovich et al.   NATURE

Advances in GBA-associated Parkinson's disease – Pathology, presentation and therapies

(2016) Melinda Barkhuizen et al.   NEUROCHEMISTRY INTERNATIONAL

A LRRK2-Dependent EndophilinA Phosphoswitch Is Critical for Macroautophagy at Presynaptic Terminals

(2016) Sandra-Fausia Soukup et al.   NEURON

Deubiquitinase Usp8 regulates α-synuclein clearance and modifies its toxicity in Lewy body disease

(2016) Zoi Alexopoulou et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

LRRK2 regulates retrograde synaptic compensation at the Drosophila neuromuscular junction

(2016) Jay Penney et al.   Nature Communications

Glucocerebrosidase Deficiency in Drosophila Results in α-Synuclein-Independent Protein Aggregation and Neurodegeneration

(2016) Marie Y. Davis et al.   PLoS Genetics

Phosphoproteomics reveals that Parkinson's disease kinase LRRK2 regulates a subset of Rab GTPases

(2016) Martin Steger et al.   eLife

Ambroxol chaperone therapy for neuronopathic Gaucher disease: A pilot study

(2016) Aya Narita et al.   Annals of Clinical and Translational Neurology

Parkinson disease-linked GBA mutation effects reversed by molecular chaperones in human cell and fly models

(2016) Alvaro Sanchez-Martinez et al.   Scientific Reports

Inhibition of alpha-synuclein aggregation by multifunctional dopamine agonists assessed by a novel in vitro assay and an in vivo Drosophila synucleinopathy model

(2016) Deepthi Yedlapudi et al.   Scientific Reports

LRRK2 Pathways Leading to Neurodegeneration

(2015) Mark R. Cookson   Current Neurology and Neuroscience Reports

Phosphoproteomic screening identifies Rab GTPases as novel downstream targets of PINK1

(2015) Y.-C. Lai et al.   EMBO JOURNAL

Glucocerebrosidase deficiency accelerates the accumulation of proteinase K-resistant α-synuclein and aggravates neurodegeneration in aDrosophilamodel of Parkinson's disease

(2015) Mari Suzuki et al.   HUMAN MOLECULAR GENETICS

VPS35pathogenic mutations confer no dominant toxicity but partial loss of function inDrosophilaand genetically interact withparkin

(2015) Bilal R. Malik et al.   HUMAN MOLECULAR GENETICS

Lrrk regulates the dynamic profile of dendritic Golgi outposts through the golgin Lava lamp

(2015) Chin-Hsien Lin et al.   JOURNAL OF CELL BIOLOGY

Ambroxol-induced rescue of defective glucocerebrosidase is associated with increased LIMP-2 and saposin C levels in GBA1 mutant Parkinson's disease cells

(2015) Giulia Ambrosi et al.   NEUROBIOLOGY OF DISEASE

GBA mutations and Parkinson disease: When genotype meets phenotype

(2015) S. W. Scholz et al.   NEUROLOGY

Attenuation of neuromotor deficits by natural antioxidants of Decalepis hamiltonii in transgenic Drosophila model of Parkinson’s disease

(2015) S.R. Jahromi et al.   NEUROSCIENCE

Glucocerebrosidase is shaking up the synucleinopathies

(2014) Marina Siebert et al.   BRAIN

Ribosomal Protein s15 Phosphorylation Mediates LRRK2 Neurodegeneration in Parkinson’s Disease

(2014) Ian Martin et al.   CELL

The Glycolytic Enzyme, GPI, Is a Functionally Conserved Modifier of Dopaminergic Neurodegeneration in Parkinson’s Models

(2014) Adam L. Knight et al.   Cell Metabolism

Novel ethyl methanesulfonate (EMS)-induced null alleles of the Drosophila homolog of LRRK2 reveal a crucial role in endolysosomal functions and autophagy in vivo

(2014) M. W. Dodson et al.   Disease Models & Mechanisms

LRRK2 localizes to endosomes and interacts with clathrin-light chains to limit Rac1 activation

(2014) A. M. Schreij et al.   EMBO REPORTS

Walking deficits and centrophobism in an α-synuclein fly model of Parkinson's disease

(2014) A. Y. Chen et al.   GENES BRAIN AND BEHAVIOR

Rab11 modulates α-synuclein-mediated defects in synaptic transmission and behaviour

(2014) Carlo Breda et al.   HUMAN MOLECULAR GENETICS

Retromer-dependent neurotransmitter receptor trafficking to synapses is altered by the Parkinson's disease VPS35 mutation p.D620N

(2014) L. N. Munsie et al.   HUMAN MOLECULAR GENETICS

LRRK2 pathobiology in Parkinson's disease

(2014) Ian Martin et al.   JOURNAL OF NEUROCHEMISTRY

Vacuolar protein sorting 35 (Vps35) rescues locomotor deficits and shortened lifespan in Drosophila expressing a Parkinson’s disease mutant of Leucine-rich repeat kinase 2 (LRRK2)

(2014) Radek Linhart et al.   Molecular Neurodegeneration

Large-scale meta-analysis of genome-wide association data identifies six new risk loci for Parkinson's disease

(2014) Mike A Nalls et al.   NATURE GENETICS

VPS35 dysfunction impairs lysosomal degradation of α-synuclein and exacerbates neurotoxicity in a Drosophila model of Parkinson's disease

(2014) Emiko Miura et al.   NEUROBIOLOGY OF DISEASE

α-Synuclein interacts with the switch region of Rab8a in a Ser129 phosphorylation-dependent manner

(2014) Guowei Yin et al.   NEUROBIOLOGY OF DISEASE

Enhanced ubiquitin-dependent degradation by Nedd4 protects against α-synuclein accumulation and toxicity in animal models of Parkinson's disease

(2014) Sian E. Davies et al.   NEUROBIOLOGY OF DISEASE

Olfactory Deficits in an Alpha-Synuclein Fly Model of Parkinson’s Disease

(2014) Alex Y. Chen et al.   PLoS One

Increasing microtubule acetylation rescues axonal transport and locomotor deficits caused by LRRK2 Roc-COR domain mutations

(2014) Vinay K. Godena et al.   Nature Communications

In vivo evidence of pathogenicity of VPS35 mutations in the Drosophila

(2014) Hua-shan Wang et al.   Molecular Brain

Dopaminergic expression of the Parkinsonian gene LRRK2-G2019S leads to non-autonomous visual neurodegeneration, accelerated by increased neural demands for energy

(2013) Samantha Hindle et al.   HUMAN MOLECULAR GENETICS

DNAJC13 mutations in Parkinson disease

(2013) Carles Vilariño-Güell et al.   HUMAN MOLECULAR GENETICS

The Function of α-Synuclein

(2013) Jacob T. Bendor et al.   NEURON

RAB7L1 Interacts with LRRK2 to Modify Intraneuronal Protein Sorting and Parkinson’s Disease Risk

(2013) David A. MacLeod et al.   NEURON

Unfolded protein response in Gaucher disease: from human to Drosophila

(2013) Gali Maor et al.   Orphanet Journal of Rare Diseases

Expression of Human Gaucher Disease Gene GBA Generates Neurodevelopmental Defects and ER Stress in Drosophila Eye

(2013) Takahiro Suzuki et al.   PLoS One

LRRK2 GTPase dysfunction in the pathogenesis of Parkinson's disease

(2012) Yulan Xiong et al.   BIOCHEMICAL SOCIETY TRANSACTIONS

Ambroxol as a pharmacological chaperone for mutant glucocerebrosidase

(2012) Inna Bendikov-Bar et al.   BLOOD CELLS MOLECULES AND DISEASES

ArfGAP1 Is a GTPase Activating Protein for LRRK2: Reciprocal Regulation of ArfGAP1 by LRRK2

(2012) Y. Xiong et al.   JOURNAL OF NEUROSCIENCE

LRRK2 kinase activity mediates toxic interactions between genetic mutation and oxidative stress in a Drosophila model: Suppression by curcumin

(2012) Dejun Yang et al.   NEUROBIOLOGY OF DISEASE

LRRK2 Controls an EndoA Phosphorylation Cycle in Synaptic Endocytosis

(2012) Samer Matta et al.   NEURON

FlyAtlas: database of gene expression in the tissues of Drosophila melanogaster

(2012) S. W. Robinson et al.   NUCLEIC ACIDS RESEARCH

The Mitochondrial Chaperone Protein TRAP1 Mitigates α-Synuclein Toxicity

(2012) Erin K. Butler et al.   PLoS Genetics

A Mutation in VPS35, Encoding a Subunit of the Retromer Complex, Causes Late-Onset Parkinson Disease

(2011) Alexander Zimprich et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Translation Initiator EIF4G1 Mutations in Familial Parkinson Disease

(2011) Marie-Christine Chartier-Harlin et al.   AMERICAN JOURNAL OF HUMAN GENETICS

VPS35 Mutations in Parkinson Disease

(2011) Carles Vilariño-Güell et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Recent Advances in the Genetics of Parkinson's Disease

(2011) Ian Martin et al.   Annual Review of Genomics and Human Genetics

Gaucher Disease Glucocerebrosidase and α-Synuclein Form a Bidirectional Pathogenic Loop in Synucleinopathies

(2011) Joseph R. Mazzulli et al.   CELL

Transcriptional responses to loss or gain of function of the leucine-rich repeat kinase 2 (LRRK2) gene uncover biological processes modulated by LRRK2 activity

(2011) Elena V. Nikonova et al.   HUMAN MOLECULAR GENETICS

Roles of the Drosophila LRRK2 homolog in Rab7-dependent lysosomal positioning

(2011) M. W. Dodson et al.   HUMAN MOLECULAR GENETICS

Inhibitors of LRRK2 kinase attenuate neurodegeneration and Parkinson-like phenotypes in Caenorhabditis elegans and Drosophila Parkinson's disease models

(2011) Zhaohui Liu et al.   HUMAN MOLECULAR GENETICS

Pre-Fibrillar α-Synuclein Mutants Cause Parkinson's Disease-Like Non-Motor Symptoms in Drosophila

(2011) Madhu Babu Gajula Balija et al.   PLoS One

LRRK2 signaling pathways: the key to unlocking neurodegeneration?

(2011) Daniel C. Berwick et al.   TRENDS IN CELL BIOLOGY

LRRK2 G2019S in the North African Population: A Review

(2010) Hani T.S. Benamer et al.   EUROPEAN NEUROLOGY

Parkinson's disease-related LRRK2 G2019S mutation results from independent mutational events in humans

(2010) Suzanne Lesage et al.   HUMAN MOLECULAR GENETICS

LRRK2 Kinase Regulates Synaptic Morphology through Distinct Substrates at the Presynaptic and Postsynaptic Compartments of the Drosophila Neuromuscular Junction

(2010) S. Lee et al.   JOURNAL OF NEUROSCIENCE

LRRK2 G2019S Mutation Induces Dendrite Degeneration through Mislocalization and Phosphorylation of Tau by Recruiting Autoactivated GSK3 

(2010) C.-H. Lin et al.   JOURNAL OF NEUROSCIENCE

Drosophila Histone Deacetylase 6 Protects Dopaminergic Neurons against α-Synuclein Toxicity by Promoting Inclusion Formation

(2010) Guiping Du et al.   MOLECULAR BIOLOGY OF THE CELL

Pathogenic LRRK2 negatively regulates microRNA-mediated translational repression

(2010) Stephan Gehrke et al.   NATURE

The role of leucine-rich repeat kinase 2 (LRRK2) in Parkinson's disease

(2010) Mark R. Cookson   NATURE REVIEWS NEUROSCIENCE

Dopamine-dependent neurodegeneration in Drosophila models of familial and sporadic Parkinson's disease

(2010) Florian Bayersdorfer et al.   NEUROBIOLOGY OF DISEASE

Genetic Animal Models of Parkinson's Disease

(2010) Ted M. Dawson et al.   NEURON

GTPase Activity Plays a Key Role in the Pathobiology of LRRK2

(2010) Yulan Xiong et al.   PLoS Genetics

Pre-fibrillar α-synuclein variants with impaired β-structure increase neurotoxicity in Parkinson's disease models

(2009) Damla Pinar Karpinar et al.   EMBO JOURNAL

Leucine-rich repeat kinase 2 interacts with Parkin, DJ-1 and PINK-1 in a Drosophila melanogaster model of Parkinson's disease

(2009) K. Venderova et al.   HUMAN MOLECULAR GENETICS

Tyrosine and serine phosphorylation of α-synuclein have opposing effects on neurotoxicity and soluble oligomer formation

(2009) Li Chen et al.   JOURNAL OF CLINICAL INVESTIGATION

The role of ubiquitin linkages on α-synuclein induced-toxicity in aDrosophilamodel of Parkinson’s disease

(2009) Francesca K. M. Lee et al.   JOURNAL OF NEUROCHEMISTRY

Parkin Protects against LRRK2 G2019S Mutant-Induced Dopaminergic Neurodegeneration in Drosophila

(2009) C.-H. Ng et al.   JOURNAL OF NEUROSCIENCE

Parkinson's disease

(2009) Andrew J Lees et al.   LANCET

Genome-wide association study reveals genetic risk underlying Parkinson's disease

(2009) Javier Simón-Sánchez et al.   NATURE GENETICS

Rapamycin activation of 4E-BP prevents parkinsonian dopaminergic neuron loss

(2009) Luke S Tain et al.   NATURE NEUROSCIENCE

Multicenter Analysis of Glucocerebrosidase Mutations in Parkinson's Disease

(2009) E. Sidransky et al.   NEW ENGLAND JOURNAL OF MEDICINE

Cathepsin D expression level affects alpha-synuclein processing, aggregation, and toxicity in vivo

(2009) Valerie Cullen et al.   Molecular Brain

Phosphorylation of 4E-BP by LRRK2 affects the maintenance of dopaminergic neurons in Drosophila

(2008) Yuzuru Imai et al.   EMBO JOURNAL

Induction of the Phase II Detoxification Pathway Suppresses Neuron Loss in Drosophila Models of Parkinson's Disease

(2008) K. Trinh et al.   JOURNAL OF NEUROSCIENCE

High doses of nicotinamide prevent oxidative mitochondrial dysfunction in a cellular model and improve motor deficit in aDrosophilamodel of Parkinson's disease

(2008) Haiqun Jia et al.   JOURNAL OF NEUROSCIENCE RESEARCH

Phenotype, diagnosis, and treatment of Gaucher's disease

(2008) Gregory A Grabowski   LANCET

Phenotype, genotype, and worldwide genetic penetrance of LRRK2-associated Parkinson's disease: a case-control study

(2008) Daniel G Healy et al.   LANCET NEUROLOGY

Dispensable role of Drosophila ortholog of LRRK2 kinase activity in survival of dopaminergic neurons

(2008) Danling Wang et al.   Molecular Neurodegeneration

Wingless secretion promotes and requires retromer-dependent cycling of Wntless

(2008) Fillip Port et al.   NATURE CELL BIOLOGY

Wingless secretion requires endosome-to-Golgi retrieval of Wntless/Evi/Sprinter by the retromer complex

(2008) Xavier Franch-Marro et al.   NATURE CELL BIOLOGY

Superoxide dismutase overexpression protects dopaminergic neurons in a Drosophila model of Parkinson's disease

(2008) Jose A. Botella et al.   NEUROBIOLOGY OF DISEASE

A Drosophila model for LRRK2-linked parkinsonism

(2008) Z. Liu et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

The Retromer Complex Influences Wnt Secretion by Recycling Wntless from Endosomes to the Trans-Golgi Network

(2007) Tatyana Y. Belenkaya et al.   DEVELOPMENTAL CELL