Search results for " sensory neuropathy"

showing 7 items of 7 documents

The EGR2 gene is involved in axonal Charcot-Marie-Tooth disease

2015

Background and purpose A three-generation family affected by axonal Charcot−Marie−Tooth disease (CMT) was investigated with the aim of discovering genetic defects and to further characterize the phenotype. Methods The clinical, nerve conduction studies and muscle magnetic resonance images of the patients were reviewed. A whole exome sequencing was performed and the changes were investigated by genetic studies, in silico analysis and luciferase reporter assays. Results A novel c.1226G>A change (p.R409Q) in the EGR2 gene was identified. Patients presented with a typical, late-onset axonal CMT phenotype with variable severity that was confirmed in the ancillary tests. The in silico studies sho…

AdultMaleEarly Growth Response Protein 2In silicomedicine.disease_causeCharcot-Marie-Tooth diseaseSeverity of Illness Indexhereditary motor sensory neuropathywhole exome sequencingYoung AdultCharcot-Marie-Tooth DiseasemedicineEGR2 geneHumansExomeeducationGeneExomeExome sequencingEarly Growth Response Protein 2Genetic testingAgedGeneticsAged 80 and overeducation.field_of_studyMutationmedicine.diagnostic_testbusiness.industryMiddle AgedPhenotypeAxonsPedigreePhenotypeNeurologyMutationFemaleNeurology (clinical)business
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Hereditary motor sensory neuropathy type II with neurofilament accumulation: new finding or new disorder?

1985

Peroneal muscular atrophy is now known to be heterogeneous and to be due to various underlying genetic mechanisms. Exploring this heterogeneity further, we report on a German kinship with the clinical, genetic, and nerve conduction features of hereditary motor and sensory neuropathy type II (HMSN type II) but whose sural nerves on biopsy were found to show infrequent axonal swellings with neurofilament accumulations not previously described. The dominant inheritance and absence of kinky hair set this disorder apart from giant axonal neuropathy. There was no history of toxic exposure to industrial chemicals. We conclude that the disorder either is a new type of HMSN or is HMSN type II with p…

AdultMaleNeurofilamentAdolescentBiopsyCardiomyopathyNeural ConductionCardiovascular System03 medical and health sciences0302 clinical medicineSural NerveEvoked Potentials SomatosensoryBiopsymedicineHumansAxonHereditary Sensory and Autonomic NeuropathiesChildCytoskeleton030304 developmental biologyGiant axonal neuropathyAged0303 health sciencesmedicine.diagnostic_testbusiness.industryElectromyographyPeroneal muscular atrophyMiddle Agedmedicine.diseaseAxonsPedigreeMicroscopy ElectronMuscular Atrophymedicine.anatomical_structureNeurologyEvoked Potentials VisualFemaleNeurology (clinical)AbnormalityHereditary motor and sensory neuropathybusinessNeuroscience030217 neurology & neurosurgeryAnnals of neurology
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Genetics of the Charcot-Marie-Tooth disease in the Spanish Gypsy population: the hereditary motor and sensory neuropathy-Russe in depth

2012

Four private mutations responsible for three forms demyelinating of Charcot-Marie-Tooth (CMT) or hereditary motor and sensory neuropathy (HMSN) have been associated with the Gypsy population: the NDRG1 p.R148X in CMT type 4D (CMT4D/HMSN-Lom); p.C737_P738delinsX and p.R1109X mutations in the SH3TC2 gene (CMT4C); and a G>C change in a novel alternative untranslated exon in the HK1 gene causative of CMT4G (CMT4G/HMSN-Russe). Here we address the findings of a genetic study of 29 Gypsy Spanish families with autosomal recessive demyelinating CMT. The most frequent form is CMT4C (57.14%), followed by HMSN-Russe (25%) and HMSN-Lom (17.86%). The relevant frequency of HMSN-Russe has allowed us to inv…

GeneticsProbandcongenital hereditary and neonatal diseases and abnormalitiesWeaknesseducation.field_of_studyHaplotypePopulationBiologymedicine.diseaseTooth diseaseDistal sensory lossGeneticsmedicinemedicine.symptomeducationHereditary motor and sensory neuropathyFounder mutationGenetics (clinical)Clinical Genetics
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Association of Variants in the SPTLC1 Gene with Juvenile Amyotrophic Lateral Sclerosis

2021

Key Points Question What genetic variants are associated with juvenile amyotrophic lateral sclerosis (ALS)? Findings In this family-based genetic study, exome sequencing was performed in 3 patients diagnosed with juvenile ALS and failure to thrive; this identified de novo variants in SPTLC1 (p.Ala20Ser in 2 patients and p.Ser331Tyr in 1 patient). Variants in SPTLC1 are a known cause of hereditary sensory and autonomic neuropathy, type 1A, and these data extend the phenotype associated with this gene. Meaning De novo variants in the SPTLC1 gene are associated with juvenile ALS, a fatal neurological disorder.

Hereditary sensory neuropathy; L-serine; Mutations; Deoxysphingolipids; AccumulationEnzyme complexJuvenile amyotrophic lateral sclerosisSerine C-Palmitoyltransferase/dk/atira/pure/subjectarea/asjc/2700/2728Whole Exome Sequencing0302 clinical medicineMedicineFamily historyAmyotrophic lateral sclerosisChildIndex caseExome sequencingOriginal Investigation0303 health sciencesNeurosciences and neurology3. Good healthChild PreschoolFailure to thriveFemalemedicine.symptomLife Sciences & BiomedicineL-SERINECommentsHumanAdultmedicine.medical_specialtyAdolescent; Adult; Amyotrophic Lateral Sclerosis; Child; Child Preschool; Female; Genetic Predisposition to Disease; Humans; Mutation; Serine C-Palmitoyltransferase; Whole Exome Sequencing; Young AdultAdolescentClinical NeurologyNO03 medical and health sciencesYoung AdultDEOXYSPHINGOLIPIDSInternal medicineExome SequencingOnline FirstHumansJuvenileGenetic Predisposition to DiseasePreschool030304 developmental biologyACCUMULATIONScience & TechnologySPTLC1business.industryMUTATIONSResearchAmyotrophic Lateral Sclerosis3112 Neurosciencesmedicine.diseaseHEREDITARY SENSORY NEUROPATHYjuvenileMutation3111 BiomedicineNeurology (clinical)Neurosciences & NeurologyALSgeneticbusiness030217 neurology & neurosurgeryAmyotrophic Lateral Sclerosi
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Complexity of the Hereditary Motor and Sensory Neuropathies

2015

Early-onset hereditary motor and sensory neuropathies are rare diseases representing a broad clinical and genetic spectrum. Without a notable familial history, the clinical diagnosis is complicated because acquired causes of peripheral neuropathy, such as inflammatory neuropathies, neuropathies with toxic causes, and nutritional deficiencies, must be considered. We examined the clinical, electrophysiological, and pathologic manifestations of a boy with an initial diagnosis of chronic inflammatory demyelinating polyneuropathy. The progression of the disease despite treatment led to a suspicion of hereditary motor and sensory neuropathy. Genetic testing revealed the presence of the MPZ p.D90…

MalePathologymedicine.medical_specialtyChronic inflammatory demyelinating polyneuropathySensory systemDiseaseBioinformaticsSural NervemedicineHumansGenetic testingmedicine.diagnostic_testbusiness.industrymedicine.diseasePhenotypePeripheral neuropathyChild PreschoolClinical diagnosisMutationPediatrics Perinatology and Child HealthMutation (genetic algorithm)Disease ProgressionNeurology (clinical)Hereditary Sensory and Motor NeuropathybusinessHereditary motor and sensory neuropathyMyelin P0 ProteinHeLa CellsJournal of Child Neurology
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Neuropathologic and morphometric studies in hereditary motor and sensory neuropathy type II with neurofilament accumulation.

1986

Histological, electron microscopic and morphometric data on sural nerve, muscle, and skin biopsies of three patients affected by autosomal dominant hereditary motor and sensory neuropathy type II with neurofilament accumulation, whose neurological, cardiological and electrophysiological data have been provided in a previous paper disclosed focally enlarged myelinated axons, due to aggregation of neurofilaments in sural nerves of all 3 biopsied patients, as well as densely packed clusters of filaments in occasional non-myelinated axons without axonal enlargement, in several fibroblasts and endothelial cells in muscle and particularly in skin. This accumulation of filaments was less pronounce…

Pathologymedicine.medical_specialtyNeurofilamentNeurologyIntermediate FilamentsSural nerveDermatologyBiologylaw.invention03 medical and health sciences0302 clinical medicineSural NervelawCharcot-Marie-Tooth DiseasemedicineHumansHereditary Sensory and Autonomic NeuropathiesIntermediate filamentCytoskeletonMyelin Sheath030304 developmental biologySkinMotor Neurons0303 health sciencesGeneral NeuroscienceMusclesGeneral MedicineAnatomymedicine.diseaseAxonsPsychiatry and Mental healthElectrophysiologyMicroscopy ElectronMuscular Atrophynervous systemUltrastructureNeurology (clinical)Electron microscopeHereditary motor and sensory neuropathy030217 neurology & neurosurgeryItalian journal of neurological sciences
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Vitamin D in the Prevention and Treatment of Diabetic Neuropathy.

2022

Neuropathy is one of the most important complications of diabetes. According to recent advances, vitamin D deficiency might play a role in the development and progression of diabetic neuropathy. Moreover, therapeutic vitamin D supplementation has the potential to improve this condition. The aim of the present review was to summarize new data available in this area.The PubMed database was searched for articles written in English and published through September 2021, using combinations of the following key words: vitamin D, diabetes, diabetes mellitus, diabetic neuropathy, polyneuropathy, peripheral neuropathy, cardiac autonomic neuropathy, supplementation, and therapy.A number of studies hav…

PharmacologyDiabetes Mellitus Type 2Diabetic NeuropathiesHumansPharmacology (medical)cardiovascular autonomic neuropathy sensory neuropathy vitamin DVitaminsVitamin DVitamin D DeficiencyClinical therapeutics
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