Sphingolipids as Regulators of Neuro-Inflammation and NADPH Oxidase 2

New Variant Mutation of Glucosylceramidase Beta (GBA) and Early Enzyme Replacement Therapy for Neuronopathic Gaucher Disease: A Case Report and Literature Review

(2020) Daiji Takajo et al.   Iranian Journal of Pediatrics

Role of 1‐Deoxysphingolipids in docetaxel neurotoxicity

(2020) Katrin Anne Becker et al.   JOURNAL OF NEUROCHEMISTRY

Lysosomal Ceramide Metabolism Disorders: Implications in Parkinson’s Disease

(2020) Silvia Paciotti et al.   Journal of Clinical Medicine

Sphingosine 1-Phosphate Receptor 2 Induces Otoprotective Responses to Cisplatin Treatment

(2020) Wei Wang et al.   Cancers

Modulation of radiation-induced damage of human glomerular endothelial cells by SMPDL3B

(2020) Alaa Abou Daher et al.   FASEB JOURNAL

Mechanism of Secondary Ganglioside and Lipid Accumulation in Lysosomal Disease

(2020) Bernadette Breiden et al.   INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

Multiple Sulfatase Deficiency: A Disease Comprising Mucopolysaccharidosis, Sphingolipidosis, and More Caused by a Defect in Posttranslational Modification

(2020) Lars Schlotawa et al.   INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

Fingolimod Affects Transcription of Genes Encoding Enzymes of Ceramide Metabolism in Animal Model of Alzheimer’s Disease

(2020) Henryk Jęśko et al.   MOLECULAR NEUROBIOLOGY

Polyunsaturated fatty acid deficiency affects sulfatides and other sulfated glycans in lysosomes through autophagy‐mediated degradation

(2020) Yaping Wang et al.   FASEB JOURNAL

Upregulating β-hexosaminidase activity in rodents prevents α-synuclein lipid associations and protects dopaminergic neurons from α-synuclein-mediated neurotoxicity

(2020) Oeystein R. Brekk et al.   Acta Neuropathologica Communications

The Role of Ceramide and Sphingosine-1-Phosphate in Alzheimer’s Disease and Other Neurodegenerative Disorders

(2019) Kinga Czubowicz et al.   MOLECULAR NEUROBIOLOGY

Exosomes mediate Zika virus transmission through SMPD3 neutral Sphingomyelinase in cortical neurons

(2019) Wenshuo Zhou et al.   Emerging Microbes & Infections

Inhibition of NADPH oxidase by apocynin prevents learning and memory deficits in a mouse Parkinson's disease model

(2019) Liyan Hou et al.   Redox Biology

Neuroinflammation in frontotemporal dementia

(2019) Fiona Bright et al.   Nature Reviews Neurology

SMPDL3b modulates insulin receptor signaling in diabetic kidney disease

(2019) A. Mitrofanova et al.   Nature Communications

Sphingosine Kinase 2 Potentiates Amyloid Deposition but Protects against Hippocampal Volume Loss and Demyelination in a Mouse Model of Alzheimer's Disease

(2019) Mona Lei et al.   JOURNAL OF NEUROSCIENCE

Sex-Related Abnormalities in Substantia Nigra Lipids in Parkinson’s Disease

(2018) T. N. Seyfried et al.   ASN Neuro

Acid Sphingomyelinase Down-Regulation Alleviates Vascular Endothelial Insulin Resistance in Diabetic Rats

(2018) Xin Li et al.   BASIC & CLINICAL PHARMACOLOGY & TOXICOLOGY

Zika virus infected primary microglia impairs NPCs proliferation and differentiation

(2018) Jin Wang et al.   BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS

Murine Sialidase Neu3 facilitates GM2 degradation and bypass in mouse model of Tay-Sachs disease

(2018) Volkan Seyrantepe et al.   EXPERIMENTAL NEUROLOGY

Stimulation of S1PR5 with A-971432, a selective agonist, preserves blood–brain barrier integrity and exerts therapeutic effect in an animal model of Huntington’s disease

(2018) Alba Di Pardo et al.   HUMAN MOLECULAR GENETICS

Hereditary sensory neuropathy type 1-associated deoxysphingolipids cause neurotoxicity, acute calcium handling abnormalities and mitochondrial dysfunction in vitro

(2018) Emma R. Wilson et al.   NEUROBIOLOGY OF DISEASE

Reversible Conformational Conversion of α-Synuclein into Toxic Assemblies by Glucosylceramide

(2018) Friederike Zunke et al.   NEURON

Analysis of age-related changes in psychosine metabolism in the human brain

(2018) Michael S. Marshall et al.   PLoS One

Sphingolipid Metabolism Is Dysregulated at Transcriptomic and Metabolic Levels in the Spinal Cord of an Animal Model of Amyotrophic Lateral Sclerosis

(2018) Alexandre Henriques et al.   Frontiers in Molecular Neuroscience

Neuronal sphingosine kinase 2 subcellular localization is altered in Alzheimer’s disease brain

(2018) Gaëlle Dominguez et al.   Acta Neuropathologica Communications

Cytoplasmic dynein regulates the subcellular localization of sphingosine kinase 2 to elicit tumor-suppressive functions in glioblastoma

(2018) Heidi A. Neubauer et al.   ONCOGENE

Zika virus infection modulates the metabolomic profile of microglial cells

(2018) Fodé Diop et al.   PLoS One

Sphingosine Kinase Activates the Mitochondrial Unfolded Protein Response and Is Targeted to Mitochondria by Stress

(2018) Sungjin Kim et al.   Cell Reports

The Peripheral Inflammatory Response to Alpha-Synuclein and Endotoxin in Parkinson's Disease

(2018) Alice J. White et al.   Frontiers in Neurology

FTY720/fingolimod increases NPC1 and NPC2 expression and reduces cholesterol and sphingolipid accumulation in Niemann-Pick type C mutant fibroblasts

(2017) Jason Newton et al.   FASEB JOURNAL

High-throughput analysis of sulfatides in cerebrospinal fluid using automated extraction and UPLC-MS/MS

(2017) Maria Blomqvist et al.   JOURNAL OF LIPID RESEARCH

Types A and B Niemann-Pick disease

(2017) Edward H. Schuchman et al.   MOLECULAR GENETICS AND METABOLISM

Sphingolipids and their metabolism in physiology and disease

(2017) Yusuf A. Hannun et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

Sphingosine 1-phosphate receptor modulation suppresses pathogenic astrocyte activation and chronic progressive CNS inflammation

(2017) Veit Rothhammer et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

De novo Synthesis of Sphingolipids Is Defective in Experimental Models of Huntington's Disease

(2017) Alba Di Pardo et al.   Frontiers in Neuroscience

Ror2 signaling regulates Golgi structure and transport through IFT20 for tumor invasiveness

(2017) Michiru Nishita et al.   Scientific Reports

Sphingosine-1-Phosphate (S1P) and S1P Signaling Pathway: Therapeutic Targets in Autoimmunity and Inflammation

(2016) Hsing-Chuan Tsai et al.   DRUGS

Neutral Sphingomyelinase-2 Deficiency Ameliorates Alzheimer's Disease Pathology and Improves Cognition in the 5XFAD Mouse

(2016) Michael B. Dinkins et al.   JOURNAL OF NEUROSCIENCE

CNS-accessible Inhibitor of Glucosylceramide Synthase for Substrate Reduction Therapy of Neuronopathic Gaucher Disease

(2016) John Marshall et al.   MOLECULAR THERAPY

Lysosomal trafficking defects link Parkinson's disease with Gaucher's disease

(2016) Yvette C. Wong et al.   MOVEMENT DISORDERS

Fingolimod: A Disease-Modifier Drug in a Mouse Model of Amyotrophic Lateral Sclerosis

(2016) Rosa Luisa Potenza et al.   Neurotherapeutics

Sphingosine Kinase 1 Cooperates with Autophagy to Maintain Endocytic Membrane Trafficking

(2016) Megan M. Young et al.   Cell Reports

Sphingosine 1-phosphate receptor 2 (S1P2) attenuates reactive oxygen species formation and inhibits cell death: implications for otoprotective therapy

(2016) Deron R. Herr et al.   Scientific Reports

Post-treatment with an ultra-low dose of NADPH oxidase inhibitor diphenyleneiodonium attenuates disease progression in multiple Parkinson’s disease models

(2015) Qingshan Wang et al.   BRAIN

Autoimmune Encephalitis and Its Relation to Infection

(2015) Arun Venkatesan et al.   Current Neurology and Neuroscience Reports

Elevation of plasma 1-deoxy-sphingolipids in type 2 diabetes mellitus: a susceptibility to neuropathy?

(2015) M. F. Dohrn et al.   EUROPEAN JOURNAL OF NEUROLOGY

Neurotoxic 1-deoxysphingolipids and paclitaxel-induced peripheral neuropathy

(2015) Rita Kramer et al.   FASEB JOURNAL

HSAN1 mutations in serine palmitoyltransferase reveal a close structure–function–phenotype relationship

(2015) Heiko Bode et al.   HUMAN MOLECULAR GENETICS

Fingolimod (FTY720) enhances hippocampal synaptic plasticity and memory in Huntington's disease by preventing p75NTRup-regulation and astrocyte-mediated inflammation

(2015) Andrés Miguez et al.   HUMAN MOLECULAR GENETICS

Functional characterization of enzymes catalyzing ceramide phosphoethanolamine biosynthesis in mice

(2015) Andreas Bickert et al.   JOURNAL OF LIPID RESEARCH

Frontotemporal dementia

(2015) Jee Bang et al.   LANCET

Unknown

(2015) Karen Ashe et al.   MOLECULAR MEDICINE

HDL-bound sphingosine-1-phosphate restrains lymphopoiesis and neuroinflammation

(2015) Victoria A. Blaho et al.   NATURE

Novel HSAN1 Mutation in Serine Palmitoyltransferase Resides at a Putative Phosphorylation Site That Is Involved in Regulating Substrate Specificity

(2015) Daniela Ernst et al.   NEUROMOLECULAR MEDICINE

The Variant p.(Arg183Trp) in SPTLC2 Causes Late-Onset Hereditary Sensory Neuropathy

(2015) Saranya Suriyanarayanan et al.   NEUROMOLECULAR MEDICINE

Sphingosine kinase 2 and sphingosine-1-phosphate promotes mitochondrial function in dopaminergic neurons of mouse model of Parkinson’s disease and in MPP+-treated MN9D cells in vitro

(2015) M. Sivasubramanian et al.   NEUROSCIENCE

Ceramides in Alzheimer’s Disease: Key Mediators of Neuronal Apoptosis Induced by Oxidative Stress and AβAccumulation

(2015) Maja Jazvinšćak Jembrek et al.   Oxidative Medicine and Cellular Longevity

Glycosphingolipids are modulators of disease pathogenesis in amyotrophic lateral sclerosis

(2015) James C. Dodge et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Cambinol, a Novel Inhibitor of Neutral Sphingomyelinase 2 Shows Neuroprotective Properties

(2015) Mariana Figuera-Losada et al.   PLoS One

Depletion of globosides and isoglobosides fully reverts the morphologic phenotype of Fabry disease.

(2014) Stefan Porubsky et al.   CELL AND TISSUE RESEARCH

Lowering Plasma 1-Deoxysphingolipids Improves Neuropathy in Diabetic Rats

(2014) Alaa Othman et al.   DIABETES

Deficiency of Sphingosine-1-phosphate Lyase Impairs Lysosomal Metabolism of the Amyloid Precursor Protein

(2014) Ilker Karaca et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Fibrillar Amyloid-  Peptides Activate Microglia via TLR2: Implications for Alzheimer's Disease

(2014) M. Jana et al.   JOURNAL OF IMMUNOLOGY

Sulfatide, A Major Lipid Component of Myelin Sheath, Activates Inflammatory Responses As an Endogenous Stimulator in Brain-Resident Immune Cells

(2014) S.-B. Jeon et al.   JOURNAL OF IMMUNOLOGY

Long term effects of enzyme replacement therapy in an Italian cohort of type 3 Gaucher patients

(2014) Annalisa Sechi et al.   MOLECULAR GENETICS AND METABOLISM

Anti-Inflammatory Effects of α-Galactosylceramide Analogs in Activated Microglia: Involvement of the p38 MAPK Signaling Pathway

(2014) Yeon-Hui Jeong et al.   PLoS One

PhotoImmunoNanoTherapy Reveals an Anticancer Role for Sphingosine Kinase 2 and Dihydrosphingosine-1-Phosphate

(2013) Brian M. Barth et al.   ACS Nano

The sirtuin inhibitor cambinol impairs MAPK signaling, inhibits inflammatory and innate immune responses and protects from septic shock

(2013) Jérôme Lugrin et al.   BIOCHIMICA ET BIOPHYSICA ACTA-MOLECULAR CELL RESEARCH

Systemic ceramide accumulation leads to severe and varied pathological consequences

(2013) Abdulfatah M. Alayoubi et al.   EMBO Molecular Medicine

Glycosphingolipids: synthesis and functions

(2013) Giovanni D'Angelo et al.   FEBS Journal

Expression machinery of GM4: the excess amounts of GM3/GM4S synthase (ST3GAL5) are necessary for GM4 synthesis in mammalian cells

(2013) Satoshi Uemura et al.   GLYCOCONJUGATE JOURNAL

Neuronal accumulation of glucosylceramide in a mouse model of neuronopathic Gaucher disease leads to neurodegeneration

(2013) T. Farfel-Becker et al.   HUMAN MOLECULAR GENETICS

Sphingomyelin synthase-related protein SMSr is a suppressor of ceramide-induced mitochondrial apoptosis

(2013) F. G. Tafesse et al.   JOURNAL OF CELL SCIENCE

Human and mouse neuroinflammation markers in Niemann-Pick disease, type C1

(2013) Stephanie M. Cologna et al.   JOURNAL OF INHERITED METABOLIC DISEASE

Specific changes of sulfatide levels in individuals with pre-clinical Alzheimer's disease: an early event in disease pathogenesis

(2013) Hua Cheng et al.   JOURNAL OF NEUROCHEMISTRY

A lipid storage-like disorder contributes to cognitive decline in HIV-infected subjects

(2013) V. V. R. Bandaru et al.   NEUROLOGY

Long term enzyme replacement therapy for Fabry disease: effectiveness on kidney, heart and brain

(2013) Saskia M Rombach et al.   Orphanet Journal of Rare Diseases

Beyond the cherry-red spot: Ocular manifestations of sphingolipid-mediated neurodegenerative and inflammatory disorders

(2013) Hui Chen et al.   SURVEY OF OPHTHALMOLOGY

Ceramide-Based Therapeutics for the Treatment of Cancer

(2012) Brian M. Barth et al.   Anti-Cancer Agents in Medicinal Chemistry

Contribution of brain inflammation to neuronal cell death in neuronopathic forms of Gaucher's disease

(2012) Einat B. Vitner et al.   BRAIN

Ceramide kinase deficiency improves diet-induced obesity and insulin resistance

(2012) Susumu Mitsutake et al.   FEBS LETTERS

NADPH Oxidase 1 Mediates  -Synucleinopathy in Parkinson's Disease

(2012) A. C. Cristovao et al.   JOURNAL OF NEUROSCIENCE

A Nonpolar Blueberry Fraction Blunts NADPH Oxidase Activation in Neuronal Cells Exposed to Tumor Necrosis Factor-α

(2012) Sally J. Gustafson et al.   Oxidative Medicine and Cellular Longevity

Lipid Raft Redox Signaling: Molecular Mechanisms in Health and Disease

(2011) Si Jin et al.   ANTIOXIDANTS & REDOX SIGNALING

Globotriaosylceramide is correlated with oxidative stress and inflammation in Fabry patients treated with enzyme replacement therapy

(2011) Giovana B. Biancini et al.   BIOCHIMICA ET BIOPHYSICA ACTA-MOLECULAR BASIS OF DISEASE

Inhibition of NADPH oxidase by glucosylceramide confers chemoresistance

(2011) Brian M. Barth et al.   CANCER BIOLOGY & THERAPY

Ceramide kinase regulates TNFα-stimulated NADPH oxidase activity and eicosanoid biosynthesis in neuroblastoma cells

(2011) Brian M. Barth et al.   CELLULAR SIGNALLING

Ceramide Kinase Regulates the Production of Tumor Necrosis Factor α (TNFα) via Inhibition of TNFα-converting Enzyme

(2011) Nadia F. Lamour et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Inhibition of NADPH oxidase promotes alternative and anti-inflammatory microglial activation during neuroinflammation

(2011) Sang-Ho Choi et al.   JOURNAL OF NEUROCHEMISTRY

BACE1 Activity Is Modulated by Cell-Associated Sphingosine-1-Phosphate

(2011) N. Takasugi et al.   JOURNAL OF NEUROSCIENCE

Neutral sphingomyelinase activation precedes NADPH oxidase-dependent damage in neurons exposed to the proinflammatory cytokine tumor necrosis factor-α

(2011) Brian M. Barth et al.   JOURNAL OF NEUROSCIENCE RESEARCH

Sphingosine-1-phosphate signaling and its role in disease

(2011) Michael Maceyka et al.   TRENDS IN CELL BIOLOGY

Sphingosine 1-phosphate receptor 1 and 3 are upregulated in multiple sclerosis lesions

(2010) Ruben Van Doorn et al.   GLIA

Identification and characterization of 15 novel GALC gene mutations causing Krabbe disease

(2010) Barbara Tappino et al.   HUMAN MUTATION

Fabry Disease Exhibiting Recurrent Stroke and Persistent Inflammation

(2010) Yoko Kikumoto et al.   INTERNAL MEDICINE

Hereditary Sensory Neuropathy Type 1 Is Caused by the Accumulation of Two Neurotoxic Sphingolipids

(2010) Anke Penno et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Intracellular localization of sphingosine kinase 1 alters access to substrate pools but does not affect the degradative fate of sphingosine-1-phosphate

(2010) Deanna L. Siow et al.   JOURNAL OF LIPID RESEARCH

Plasma membrane sphingomyelin hydrolysis increases hippocampal neuron excitability by sphingosine-1-phosphate mediated mechanisms

(2010) Eric Norman et al.   JOURNAL OF NEUROCHEMISTRY

Impaired Ganglioside Metabolism in Huntington's Disease and Neuroprotective Role of GM1

(2010) V. Maglione et al.   JOURNAL OF NEUROSCIENCE

Sphingolipids in Multiple Sclerosis

(2010) Arundhati Jana et al.   NEUROMOLECULAR MEDICINE

Intrathecal increase of sphingosine 1-phosphate at early stage multiple sclerosis

(2010) Alina Kułakowska et al.   NEUROSCIENCE LETTERS

Fabry disease

(2010) Dominique P Germain   Orphanet Journal of Rare Diseases

Post-Stroke Inhibition of Induced NADPH Oxidase Type 4 Prevents Oxidative Stress and Neurodegeneration

(2010) Christoph Kleinschnitz et al.   PLOS BIOLOGY

Targeting and Regulation of Reactive Oxygen Species Generation by Nox Family NADPH Oxidases

(2009) Thomas L. Leto et al.   ANTIOXIDANTS & REDOX SIGNALING

Glucocerebrosidase mutations in clinical and pathologically proven Parkinson's disease

(2009) Juliane Neumann et al.   BRAIN

Increased levels of inflammatory chemokines in amyotrophic lateral sclerosis

(2009) J. Kuhle et al.   EUROPEAN JOURNAL OF NEUROLOGY

Subcellular Origin of Sphingosine 1-Phosphate Is Essential for Its Toxic Effect in Lyase-deficient Neurons

(2009) Nadine Hagen et al.   JOURNAL OF BIOLOGICAL CHEMISTRY

Sphingomyelin synthase-related protein SMSr controls ceramide homeostasis in the ER

(2009) Ana M. Vacaru et al.   JOURNAL OF CELL BIOLOGY

FTY720 Reduces Inflammation and Promotes Functional Recovery after Spinal Cord Injury

(2009) Kangmin D. Lee et al.   JOURNAL OF NEUROTRAUMA

Proinflammatory cytokines provoke oxidative damage to actin in neuronal cells mediated by Rac1 and NADPH oxidase

(2009) Brian M. Barth et al.   MOLECULAR AND CELLULAR NEUROSCIENCE

Beneficial effects of anti-inflammatory therapy in a mouse model of Niemann-Pick disease type C1

(2009) David Smith et al.   NEUROBIOLOGY OF DISEASE

Oligodendrocyte Development and Myelin Biogenesis: Parsing Out the Roles of Glycosphingolipids

(2009) Nicole Jackman et al.   PHYSIOLOGY

Regulation of Histone Acetylation in the Nucleus by Sphingosine-1-Phosphate

(2009) N. C. Hait et al.   SCIENCE

Randomized, controlled trial of miglustat in Gaucher's disease type 3

(2008) Raphael Schiffmann et al.   ANNALS OF NEUROLOGY

Huntington's disease: from pathology and genetics to potential therapies

(2008) Sara Imarisio et al.   BIOCHEMICAL JOURNAL

Sphingomyelin synthases regulate production of diacylglycerol at the Golgi

(2008) Maristella Villani et al.   BIOCHEMICAL JOURNAL

The sphingolipid salvage pathway in ceramide metabolism and signaling

(2008) Kazuyuki Kitatani et al.   CELLULAR SIGNALLING

Intracerebroventricular infusion of acid sphingomyelinase corrects CNS manifestations in a mouse model of Niemann–Pick A disease

(2008) James C. Dodge et al.   EXPERIMENTAL NEUROLOGY

Decreased activities of lysosomal acid alpha-D-galactosidase A in the leukocytes of sporadic Parkinson's disease

(2008) Guanghua Wu et al.   JOURNAL OF THE NEUROLOGICAL SCIENCES

Niemann-Pick disease type C1 is a sphingosine storage disease that causes deregulation of lysosomal calcium

(2008) Emyr Lloyd-Evans et al.   NATURE MEDICINE

Ceramide Triggers Budding of Exosome Vesicles into Multivesicular Endosomes

(2008) K. Trajkovic et al.   SCIENCE