Search results for "lipofuscin"

showing 10 items of 62 documents

Current state of clinical and morphological features in human NCL.

2004

The neuronal ceroid lipofuscinoses (NCL) are large group of autosomal recessive lysosomal storage disorders with both enzymatic deficiency and structural protein dysfunction. Previously, diagnosis of (NCL) was based on age at onset clinicopathological (C‐P) findings described 4 forms, classified as infantile (INCL) (2), late‐infantile (LINCL) (5), juvenile (JNCL) (6), and adult (ANCL) most patients with NCL have progressive ocular and cerebral dysfunvtion, including cognitive/motor dysfunction and uncontrolled seizures. After reviewing 520 patients with NCL, we found that about 104 (20%) did not fit this classification of NCL With further research, 4 additional forms have been recognized: F…

AdultPathologymedicine.medical_specialtymedicine.disease_causeArticlePathology and Forensic MedicineEpilepsyNeuronal Ceroid-LipofuscinosesGenotypemedicineHumansPalmitoyl protein thioesteraseAge of OnsetChildInclusion BodiesMutationbiologyTripeptidyl-Peptidase 1General NeurosciencePPT1Infantmedicine.diseasePhenotypeCLN8Child PreschoolMutationbiology.proteinNeurology (clinical)Age of onsetBrain pathology (Zurich, Switzerland)
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Ultrastructure of the Retina in Adult Neuronal Ceroid Lipofuscinosis

1998

A 33-year-old woman died of biopsy-proven adult neuronal ceroid lipofuscinosis (NCL) or Kufs’ disease marked by fingerprint and curvilinear lipopigments in neural and nonneural cell types. She had never experienced visual impairment or shown electroretinographic abnormalities. At autopsy, her retina appeared intact without degeneration at the light-microscopic level, but nerve cells in different layers were loaded with lipopigments of the granular type. This appears to be the third ultrastructural study of the retina in a patient with adult NCL, a former one showing preservation of the retina, another retinal degeneration. Thus, only further molecular genetic data will clarify the nosology …

AdultRetinal Ganglion CellsRetinal degenerationCell typePathologymedicine.medical_specialtyHistologyAutopsyDegeneration (medical)BiologyRetinaAdult neuronal ceroid lipofuscinosisFatal OutcomeNeuronal Ceroid-LipofuscinosesmedicineHumansKufs diseaseRetinaPigments BiologicalAnatomymedicine.diseaseLipidsMicroscopy Electronmedicine.anatomical_structureUltrastructureFemaleAnatomyCells Tissues Organs
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Photobleaching effects onin vivoskin autofluorescence lifetime

2015

The autofluorescence lifetime of healthy human skin was measured using excitation provided by a picosecond diode laser operating at a wavelength of 405 nm and with fluorescence emission collected at 475 and 560 nm. In addition, spectral and temporal responses of healthy human skin and intradermal nevus in the spectral range 460 to 610 nm were studied before and after photobleaching. A decrease in the autofluorescences lifetimes changes was observed after photobleaching of human skin. A three-exponential model was used to fit the signals, and under this model, the most significant photoinduced changes were observed for the slowest lifetime component in healthy skin at the spectral range 520 …

AdultTime FactorsMaterials scienceLightPhotochemistryBiomedical EngineeringHuman skinAbsorption (skin)LipofuscinBiomaterialsNuclear magnetic resonanceFlavinsIntradermal NevusmedicineHumansNevusskin and connective tissue diseasesPhospholipidsSkinInflammationPhotobleachingbusiness.industryEquipment DesignMiddle AgedHandmedicine.diseasePhotobleachingFluorescenceAtomic and Molecular Physics and OpticsElectronic Optical and Magnetic MaterialsAutofluorescenceSpectrometry FluorescenceOxyhemoglobinsPicosecondFlavin-Adenine DinucleotideNevus IntradermalOptoelectronicsbusinessJournal of Biomedical Optics
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Lipofuscin Hypothesis of Alzheimer’s Disease

2011

The primary culprit responsible for Alzheimer’s disease (AD) remains unknown. Aβ protein has been identified as the main component of amyloid of senile plaques, the hallmark lesion of AD, but it is not definitively established whether the formation of extracellular Aβ deposits is the absolute harbinger of the series of pathological events that hit the brain in the course of sporadic AD. The aim of this paper is to draw attention to a relatively overlooked age-related product, lipofuscin, and advance the hypothesis that its release into the extracellular space following the death of neurons may substantially contribute to the formation of senile plaques. The presence of intraneuronal Aβ, sim…

Aβ proteinNeurofibrillary tanglesAmyloidAmyloidCognitive Neurosciencelcsh:Geriatricslcsh:RC346-429LipofuscinLipofuscinLesionExtracellularMedicineOriginal Research ArticleSenile plaquesPathologicallcsh:Neurology. Diseases of the nervous systembusiness.industryMacular degenerationAlzheimer's diseaseMacular degenerationmedicine.diseaseBiochemistry of Alzheimer's diseaselcsh:RC952-954.6Psychiatry and Mental healthmedicine.symptombusinessAlzheimer’s diseaseNeuroscienceDementia and Geriatric Cognitive Disorders Extra
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The neuronal ceroid-lipofuscinoses: A historical introduction

2013

AbstractThe neuronal ceroid-lipofuscinoses (Batten disease) collectively constitute one of the most common groups of inherited childhood onset neurodegenerative disorders, and have also been identified in many domestic and laboratory animals. The group of human neuronal ceroid-lipofuscinoses currently comprises 14 genetically distinct disorders, mostly characterised by progressive mental, motor and visual deterioration with onset in childhood or adolescence. Abnormal autofluorescent, electron-dense granules accumulate in the cytoplasm of nerve cells, and this storage process is associated with selective destruction and loss of neurons in the brain and retina. The present paper outlines near…

Batten diseaseHistoryBatten diseaseDiseaseBiology03 medical and health sciences0302 clinical medicineNeuronal Ceroid-LipofuscinosesmedicineHumansNeurodegenerationMolecular Biology030304 developmental biologyNeuronal Ceroid-Lipofuscinoses0303 health sciencesRetinaNeurodegenerationHistory 19th CenturyHistory 20th Centurymedicine.disease3. Good healthAgeingmedicine.anatomical_structureNerve cellsNeuronal ceroid-lipofuscinosisMolecular genetic classificationMolecular MedicineNeuronal ceroid lipofuscinosisIdentification (biology)Neuroscience030217 neurology & neurosurgeryBiochimica et Biophysica Acta (BBA) - Molecular Basis of Disease
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Immunelectronmicroscopic characterization of T4 and T8 lymphocytes and natural killer cells in neuronal ceroid-lipofuscinosis.

1995

CD4+, CD8+, and CD56+ cells were isolated with the immunomagnetic separation technique from peripheral blood mononuclear cells (PBMC) of 3 patients with neuronal ceroid-lipofuscinosis : one patient each with infantile (INCL), late infantile (LINCL), and juvenile (JNCL) neuronal ceroid-lipofuscinoses, all studied by light (LM) and electron (EM) microscopy. To compare the pathology of these cells with affected cells in other types of lysosomal diseases, the separation was also performed with PBMC of 1 patient with mucolipidosis (ML) type II, 2 patients with mucopolysaccharidosis (MPS) type I, and 4 patients with MPS type III. Disease-specific lysosomal inclusions were identified in CD4+, CD8+…

CD4-Positive T-LymphocytesAdolescentMucolipidosisLymphocyteMucopolysaccharidosisInfantBiologyCD8-Positive T-LymphocytesMucopolysaccharidosesmedicine.diseaseImmunomagnetic separationMolecular biologyPeripheral blood mononuclear cellKiller Cells Naturalmedicine.anatomical_structureNeuronal Ceroid-LipofuscinosesChild PreschoolImmunologymedicineHumansNeuronal ceroid lipofuscinosisLysosomesMicroscopy ImmunoelectronGenetics (clinical)CD8American journal of medical genetics
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Characterization of T–cell subclasses and NK–cells in lysosomal disorders by immuno–electron microscopy

1994

Previous studies have shown that B and T lymphocytes are affected in lysosomal disorders. The aim of this study was to investigate the involvement of subclasses of T lymphocytes and natural killer cells in lysosomal diseases. CD4+, CD8+, and CD56+ cells were immunomagnetically separated from peripheral blood mononuclear cells in 10 patients with various lysosomal diseases--including one patient each with infantile, late infantile, and juvenile neuronal ceroid-lipfuscinoses, two patients with mucopolysaccharidosis (MPS) type I and four patients with MPS type III, and one patient with mucolipidosis type II; all lymphocytes were studied by light and electron microscopy. Respective vacuolar or …

CD4-Positive T-LymphocytesPathologymedicine.medical_specialtyHistologyT-LymphocytesMucopolysaccharidosisT cellImmunoblottingCD8-Positive T-LymphocytesBiologyPathology and Forensic MedicineNatural killer cellPhysiology (medical)Lysosomal storage diseasemedicineHumansMicroscopy ImmunoelectronT lymphocyteMucopolysaccharidosesmedicine.diseaseKiller Cells Naturalmedicine.anatomical_structureNeurologyNeuronal ceroid lipofuscinosisNeurology (clinical)I-cell diseaseLysosomesCD8Neuropathology and Applied Neurobiology
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Untersuchungen an isoliertem, unver�ndertem Lipofuscin aus Herzmuskulatur

1955

1. Nach einer kritischen Betrachtung der heutigen Kenntnisse uber das Lipofuscin und einer Abwagung der einzelnen Untersuchungs-methoden wird die besondere Bedeutung chemischer Untersuchungen an isoliertem Lipofuscin hervorgehoben. 2. Es wird eine Methode zur Isolierung reinen, unveranderten Lipofuscins aus Herzmuskulatur angegeben, die bestimmte physikalische Eigenschaften von Lipofuscin fur die Isolierung ausnutzt. Chemische Methoden zur Isolierung erscheinen wenig geeignet, da durch sie das Pigment denaturiert wird. 3. Bei mikroskopischen Untersuchungen zeigt das isolierte Lipofuscin das gleiche optische und farberische Verhalten wie innerhalb der Zelle. Es kann daher sicher identifizier…

ChemistryMyocardium metabolismCell BiologyGeneral MedicineMolecular BiologyMolecular biologyPathology and Forensic MedicineLipofuscinVirchows Archiv f�r Pathologische Anatomie und Physiologie und f�r Klinische Medizin
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Prenatal Ultrastructural Diagnosis in the Neuronal ceroid-lipofuscinoses

1994

Summary The neuronal ceroid-lipofuscinose (NCL) are autosomal-recessive disorders in childhood of unknown enzymatic origin. They can be recognized by the presence of abnormal lipopigments identified by electron microscopy. Based on the study of circulating lymphocytes, individual clinical subtypes of NCL can be correlated. Prenatal diagnosis of NCL with the electron microscope is now feasible for the infantile (Finnish) from (INCL) and late-infantile form (LINCL). INCL-specific granular lipopigments are present in endothelial cells of biopsied chorion stroma vessels of homozygously affected fetuses. In LINCL, disease-typical curvilinear bodies can be identified in uncultured amniotic fluid …

FetusPathologymedicine.medical_specialtyAmniotic fluid cellsCurvilinear bodiesPrenatal diagnosisChorionCell BiologyBiologyAmniotic FluidPathology and Forensic MedicineMicroscopy ElectronStromaNeuronal Ceroid-LipofuscinosesPregnancyPrenatal DiagnosisUltrastructuremedicineHumansFemaleElectron microscopicNeuronal Ceroid-LipofuscinosesPathology - Research and Practice
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7th International Congress on Neuronal Ceroid‐Lipofuscinoses (NCL‐98) 13–16 June 1998, Dallas, USA

1998

General NeuroscienceInternational congressPolitical scienceLibrary scienceEnvironmental ethicsNeurology (clinical)Meeting ReportPathology and Forensic MedicineNeuronal Ceroid-Lipofuscinoses
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