Raras
Buscar doenças, sintomas, genes...
Ataxia espinocerebelosa tipo 42
ORPHA:458803CID-10 · G11.8OMIM 616795DOENÇA RARA

Doenças de repetição de trinucleotídeos, também conhecidas como doenças de expansão de microssatélites, são um conjunto de mais de 50 doenças genéticas causadas por expansão de repetição de trinucleotídeo, um tipo de mutação em que repetições de três nucleótidos aumentam o número de cópias até cruzarem um limiar acima do qual se tornam instáveis. Dependendo de sua localização, a repetição instável do trinucleotídeo pode causar defeitos em uma proteína codificada por um gene; alterar a regulação da expressão gênica; produzir um RNA tóxico, ou levar à instabilidade cromossômica. Em geral, quanto maior a expansão, mais rápido o aparecimento da doença e mais grave a doença se torna.

Mantido por Agente Raras·Colaborar como especialista →

Introdução

O que você precisa saber de cara

📋

Ataxia espinocerebelosa tipo 42 é uma doença neurológica autossômica dominante, causada por mutações no gene CACNA1G. Manifesta-se com espasticidade, hipertonia, convulsões e atraso global do desenvolvimento, além de características faciais como olhos profundamente inseridos e nariz curto.

Publicações científicas
8.166 artigos
Último publicado: 2026 Apr 16

Escala de raridade

CLASSIFICAÇÃO ORPHANET · BRASIL 2024
<1 / 1 000 000
Ultra-rara
<1/50k
Muito rara
1/20k
Rara
1/10k
Pouco freq.
1/5k
Incomum
1/2k
Prevalência
0.0
Worldwide
Casos conhecidos
25
pacientes catalogados
Início
Adolescent
+ adult, childhood, elderly
🏥
SUS: Cobertura mínimaScore: 15%
CID-10: G11.8
🇧🇷Dados SUS / DATASUS
PROCEDIMENTOS SIGTAP (2)
0202010694
Sequenciamento completo do exoma (WES)genetic_test
0301070040
Atendimento em reabilitação — doenças rarasrehabilitation
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Entender a doença

Do básico ao detalhe, leia no seu ritmo

Preparando trilha educativa...

Sinais e sintomas

O que aparece no corpo e com que frequência cada sintoma acontece

Partes do corpo afetadas

🧠
Neurológico
28 sintomas
👁️
Olhos
6 sintomas
😀
Face
3 sintomas
🦴
Ossos e articulações
3 sintomas
🧬
Pele e cabelo
3 sintomas
🫘
Rins
2 sintomas

+ 24 sintomas em outras categorias

Características mais comuns

100%prev.
Ataxia
Frequência: 15/15
90%prev.
Morfologia anormal do cerebelo
Muito frequente (99-80%)
90%prev.
Anormalidade do neurodesenvolvimento
Muito frequente (99-80%)
80%prev.
Disartria
Muito frequente (99-80%)
76%prev.
Marcha instável
Muito frequente (99-80%)
55%prev.
Ataxia da marcha
Frequente (79-30%)
72sintomas
Muito frequente (4)
Frequente (9)
Ocasional (22)
Sem dados (37)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 72 características clínicas mais associadas, ordenadas por frequência.

Ataxia
Frequência: 15/15100%
Morfologia anormal do cerebeloAbnormal cerebellum morphology
Muito frequente (99-80%)90%
Anormalidade do neurodesenvolvimentoNeurodevelopmental abnormality
Muito frequente (99-80%)90%
DisartriaDysarthria
Muito frequente (99-80%)80%
Marcha instávelUnsteady gait
Muito frequente (99-80%)76%

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa1desde 2026
Total histórico8.166PubMed
Últimos 10 anos100publicações
Pico202115 papers
Linha do tempo
2026Hoje · 2026📈 2021Ano de pico
Publicações por ano (últimos 10 anos)

Encontrou um erro ou informação desatualizada? Sugira uma correção →

Genética e causas

O que está alterado no DNA e como passa nas famílias

Genes associados

1 gene identificado com associação a esta condição. Padrão de herança: Autosomal dominant.

CACNA1GVoltage-dependent T-type calcium channel subunit alpha-1GDisease-causing germline mutation(s) inAltamente restrito
FUNÇÃO

Voltage-sensitive calcium channels (VSCC) mediate the entry of calcium ions into excitable cells and are also involved in a variety of calcium-dependent processes, including muscle contraction, hormone or neurotransmitter release, gene expression, cell motility, cell division and cell death. The isoform alpha-1G gives rise to T-type calcium currents. T-type calcium channels belong to the 'low-voltage activated (LVA)' group and are strongly blocked by mibefradil. A particularity of this type of c

LOCALIZAÇÃO

Cell membraneCytoplasm

VIAS BIOLÓGICAS (2)
NCAM1 interactionsSmooth Muscle Contraction
MECANISMO DE DOENÇA

Spinocerebellar ataxia 42

A form of spinocerebellar ataxia, a clinically and genetically heterogeneous group of cerebellar disorders. Patients show progressive incoordination of gait and often poor coordination of hands, speech and eye movements, due to degeneration of the cerebellum with variable involvement of the brainstem and spinal cord. SCA42 is a slowly progressive, autosomal dominant form with variable severity.

OUTRAS DOENÇAS (2)
spinocerebellar ataxia type 42spinocerebellar ataxia 42, early-onset, severe, with neurodevelopmental deficits
HGNC:1394UniProt:O43497

Variantes genéticas (ClinVar)

448 variantes patogênicas registradas no ClinVar.

🧬 CACNA1G: NM_018896.5(CACNA1G):c.2911-8C>A ()
🧬 CACNA1G: NM_018896.5(CACNA1G):c.2859C>A (p.Tyr953Ter) ()
🧬 CACNA1G: NM_018896.5(CACNA1G):c.2754+1G>A ()
🧬 CACNA1G: NM_018896.5(CACNA1G):c.4705+22G>T ()
🧬 CACNA1G: NM_018896.5(CACNA1G):c.1847C>T (p.Pro616Leu) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 79 variantes classificadas pelo ClinVar.

12
63
4
Patogênica (15.2%)
VUS (79.7%)
Benigna (5.1%)
VARIANTES MAIS SIGNIFICATIVAS
CACNA1G: NM_018896.5(CACNA1G):c.4180C>T (p.Arg1394Trp) [Likely pathogenic]
CACNA1G: NM_018896.5(CACNA1G):c.4931A>T (p.Lys1644Met) [Likely pathogenic]
CACNA1G: NM_018896.5(CACNA1G):c.757G>A (p.Val253Met) [Conflicting classifications of pathogenicity]
CACNA1G: NM_018896.5(CACNA1G):c.4781A>G (p.Tyr1594Cys) [Uncertain significance]
CACNA1G: NM_018896.5(CACNA1G):c.4663C>T (p.Arg1555Trp) [Uncertain significance]

Vias biológicas (Reactome)

2 vias biológicas associadas aos genes desta condição.

Diagnóstico

Os sinais que médicos procuram e os exames que confirmam

Carregando...

Tratamento e manejo

Remédios, cuidados de apoio e o que precisa acompanhar

Carregando informações de tratamento...

Onde tratar no SUS

Hospitais de referência no Brasil e o protocolo oficial do SUS (PCDT)

🇧🇷 Atendimento SUS — Ataxia espinocerebelosa tipo 42

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Selecione um estado ou use sua localização para ver resultados.

Dados de DATASUS/CNES, SBGM, ABNeuro e Ministério da Saúde. Sempre confirme a disponibilidade diretamente com o estabelecimento.

Pesquisa ativa

Ensaios clínicos abertos e novidades científicas recentes

Pesquisa e ensaios clínicos

Nenhum ensaio clínico registrado para esta condição.

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Publicações mais relevantes

Timeline de publicações
3.160 papers (10 anos)

Mostrando amostra de 100 publicações de um total de 3.160

#1

Monitoring the Progression of Pre-Ataxic Gait in SCA2 with Inertial Sensors Over Four Years.

Cerebellum (London, England)2026 Mar 25

The search for digital biomarkers of gait ataxia is a key research priority in spinocerebellar ataxias (SCAs), especially in the early stages when traditional clinical scales are less effective. Despite existing evidence supporting the effectiveness of suitable digital biomarkers, their use in assessing early disease progression remains limited. This study was aimed to evaluate the progression of digitally measured gait ataxia features in preclinical SCA2. Twenty-seven preclinical carriers of the SCA2 mutation were monitored four times over four years. Participants completed a 10-meter walking test (back and forth) using six body-worn inertial measurement units. We assessed stride-to-stride means and variability of eight gait features indicative of subtle abnormalities in SCA2 carriers, alongside the Scale for the Assessment and Rating of Ataxia (SARA). Mean stride-to-stride variables demonstrated significant progression more frequently than variability measures, with means primarily exhibiting non-linear patterns and variability metrics showing mainly linear trajectories. Significant progression of mean stride-to-stride variables was also observed in unconverted carriers. CAG repeat length significantly influences progression of some gait kinematics in preclinical SCA2 carriers. Notably, several digitally measured gait parameters required smaller sample sizes to detect progression in hypothetical clinical trials than the SARA clinical scale. This study confirmed the progressive deterioration of subtle gait function in preclinical SCA2 and highlighted the clinical utility of digitally derived metrics for tracking longitudinal changes at early disease stages. These digital measures may provide more sensitive and reliable biomarkers of disease progression than conventional clinical rating scales, supporting their potential use in future clinical trials.

#2

Propranolol for Tremors in Spinocerebellar Ataxia Type 12: A Randomized Clinical Trial.

Movement disorders : official journal of the Movement Disorder Society2026 Feb

Spinocerebellar ataxia type 12 (SCA12) is a neurodegenerative disorder with prominent upper limb tremors. No trials have evaluated tremor-targeted therapies in this population. We evaluated long-acting propranolol for efficacy and safety in reducing tremors and improving ataxia and quality of life in SCA12. In this single-center, randomized, double-blind, placebo-controlled trial, 60 genetically confirmed SCA12 patients (aged 18-65 years; TETRAS PS [The Essential Tremor Rating Assessment Scale Performance Subscale] upper limb component ≥2) were randomized 1:1 to receive extended-release propranolol or placebo. Propranolol was escalated by 40 mg/week (placebo by one tablet/week) up to 240 mg/day, limiting side effects or ≥30% reduction in TETRAS PS/ADL (Activities-of-Daily-Living Subscale) + PS. The stable dose was continued for 8 weeks (maintenance phase). Primary outcomes were changes from baseline in TETRAS PS and ADL + PS at maintenance weeks 4 and 8. Secondary outcomes included changes in the Scale for the Assessment and Rating of Ataxia (SARA), RAND 36-Item Short-Form Health Survey (SF-36) domains, and accelerometric tremor parameters. Propranolol significantly reduced TETRAS PS (LSM ± SE: -4.4 ± 0.3 at week 4; -4.42 ± 0.4 at week 8) and ADL + PS (-5.5 ± 0.63 at week 4; -5.56 ± 0.62 at week 8; P < 0.001 vs. placebo). Significant improvements were also noted in SARA and five of nine SF-36 domains at both time points. Accelerometry confirmed reductions in tremor peak frequency and power in all positions except rest. Fatigue and headache were the commonest adverse effects, with no discontinuations due to adverse events. Propranolol effectively reduced tremors and improved quality of life in SCA12, with good tolerability. © 2025 International Parkinson and Movement Disorder Society.

#3

Multidimensional abnormal gait analysis and biomarker identification for patients with spinocerebellar ataxia type 3 using an Azure Kinect-based motion capture system.

Gait &amp; posture2026 Jan

Spinocerebellar Ataxia Type 3 (SCA3), the most common hereditary ataxia in China, is characterized by progressive gait dysfunction. While quantitative gait analysis provides critical insights into movement disorder management, conventional motion capture systems are often cost-prohibitive and impractical for clinical use. We propose using the markerless Azure Kinect, a cost-effective and portable tool for gait analysis, to detect SCA3-specific gait patterns and identify gait parameters associated with disease severity and duration. We enrolled 38 patients with SCA3 patients and 42 healthy controls (HCs). Gait was recorded using an Azure Kinect. Multiple gait parameters were computed and compared with t-tests/Mann-Whitney U tests. The receiver operating characteristic (ROC) analysis identified discriminatory biomarkers, while Pearson's test assessed gait-clinical characteristic associations. Patients with SCA3 exhibited increased mediolateral margins of stability (MOS, p < 0.01), wider step width (p < 0.001), shorter stride length (p = 0.003), slower gait speed (p = 0.007), and reduced hip/knee/ankle joint angles (p < 0.05) compared to HCs. Step width demonstrated the highest diagnostic accuracy (AUC = 0.878, cutoff = 0.197). Increased medial-lateral MOS was negatively correlated with step length (r = -0.52∼-0.45, P < 0.005). Minimal hip frontal angles negatively correlated with SARA scores (r = -0.46, p = 0.004) and disease duration (r = -0.35, p = 0.028), reflecting a progressive cerebellar degeneration. In SCA3, gait abnormalities such as increased step width and shortened stride length indicate compensatory adaptations exist to enhance dynamic stability. Step width is identified as a sensitive biomarker for SCA3 screening.

#4

Predictive models for ataxia progression and conversion in spinocerebellar ataxia type 1 and 3.

Brain : a journal of neurology2025 Oct 28

The READISCA study aims to prepare for clinical trials in SCA1 and SCA3. Hence, we searched for predictive variables of ataxia onset (phenoconversion) and progression. Individuals with SCA1 or SCA3 and controls were enrolled from 2018-2021 in US and Europe. Clinical scores, MRI measures and NfL levels were assessed annually for 5 years. In the pre-ataxic group at baseline, we compared phenoconverters with non-converters. A Bayesian mixed model was used to model the longitudinal progression of clinical scores and NfL levels. The impact of data-driven selected baseline variables (demographic, clinical, MRI) on the expected SARA progression was tested. Forty-three controls, 55 SCA1 and 124 SCA3 carriers were included; a subset of the cohort (n=109) had MRI data. Converters from pre-ataxic to ataxic stages represented 5/22 (22%) and 12/38 (32%) for SCA1 and SCA3. Converters were more depressed (PHQ9: 3.9±2.9 vs 2.3±2.6 p = 0.04), had higher plasma NfL levels (17.6±5.7 pg.mL-1 vs 11.1±5.9, p<0.0001), more cerebellar white matter atrophy (1.44±0.12 % of total intracranial volume vs 1.54±0.16, p=0.032) and more INAS signs (1.8±1.3 vs 0.7±0.8, p = 0.002). All clinical scores except CCAS significantly worsened during the study. NfL levels significantly increased in non-converters and ataxic SCA3 (1.06±0.33 pg.mL-1/year, p=0.002 and 0.57±0.21, p=0.01) but not in controls and ataxic SCA1 (0.31±0.26, p=0.24 and 0.26±0.42, p=0.55). In the best predictive model of SARA progression after 1 year (R2=0.54), factors linked with faster progression were higher functional stage (p<0.001), higher CCFS score (p=0.002), and higher total creatine in cerebellar white matter (p=0.026). Factors significantly linked to conversion, namely NfL levels, depression, and lower motor neuron involvement, differ from those driving disease progression. NfL levels and lower motoneuron signs could be used as predictors of phenoconversion and MRI variables as ataxia progression predictors. Psychological care should be provided in the pre-ataxic phase of the disease.

#5

An Investigation of Corticospinal Tract Microstructural Integrity in ARSACS Using a Profilometry MRI Analysis: Results From the PROSPAX Study.

European journal of neurology2025 Apr

Spasticity represents a core clinical feature of Autosomal Recessive Spastic Ataxia of Charlevoix-Saguenay (ARSACS) patients. Nonetheless, its pathophysiological substrate is poorly investigated. We assessed the microstructural integrity of the corticospinal tract (CST) using diffusion MRI (dMRI) via profilometry analysis to understand its possible role in the development of spasticity in ARSACS. In this multi-center prospective study, data of 37 ARSACS (M/F = 21/16; 33.4 ± 12.4 years) and 29 controls (M/F = 13/16; 42.1 ± 17.2 years) acquired within the PROSPAX consortium were collected from January 2021 to October 2022 and analyzed. Differences in terms of global CST microstructural integrity were probed, as well as a possible spatial distribution of the damage along the tract via profilometry analysis. Possible correlations between clinical severity, including the Spastic Paraplegia Rating Scale (SPRS), were also tested. A significant global involvement of the CST was found in ARSACS compared to controls (all tests with p < 0.001), with a spatially defined pattern of more pronounced microstructural integrity loss occurring right below and above the pons, a structure that was also confirmed to be thickened in these patients (p < 0.001). A bilateral negative correlation emerged between the microstructural integrity of the CST and clinical indices of spasticity expressed via SPRS (p = 0.02 for both CSTs). A clinically meaningful microstructural involvement of CST is present in ARSACS patients, with a spatially defined pattern of damage occurring right below and above a thickened pons. An evaluation of the microstructure of this bundle might serve as a possible biomarker in this condition.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC3.032 artigos no totalmostrando 98

2026

Monitoring the Progression of Pre-Ataxic Gait in SCA2 with Inertial Sensors Over Four Years.

Cerebellum (London, England)
2025

Changes in Gait After Training for Individuals With Cerebellar Ataxia.

Archives of rehabilitation research and clinical translation
2026

Propranolol for Tremors in Spinocerebellar Ataxia Type 12: A Randomized Clinical Trial.

Movement disorders : official journal of the Movement Disorder Society
2025

Predictive models for ataxia progression and conversion in spinocerebellar ataxia type 1 and 3.

Brain : a journal of neurology
2026

Multidimensional abnormal gait analysis and biomarker identification for patients with spinocerebellar ataxia type 3 using an Azure Kinect-based motion capture system.

Gait &amp; posture
2025

Electrophysiological classification of CACNA1G gene variants associated with neurodevelopmental and neurological disorders.

Frontiers in pharmacology
2025

Nerve Fiber Bundle Damage in Spinocerebellar Degeneration on Diffusion Tensor Imaging.

Current medical imaging
2025

Spastic Ataxia Composite (SPAXCOM): A Scale to Evaluate the Progression of Subjects with Spasticity and Ataxia.

Movement disorders : official journal of the Movement Disorder Society
2025

Sex Differences in Spinocerebellar Ataxia Type 1: Clinical Presentation and Progression.

Cerebellum (London, England)
2025

An Investigation of Corticospinal Tract Microstructural Integrity in ARSACS Using a Profilometry MRI Analysis: Results From the PROSPAX Study.

European journal of neurology
2025

Diplopia in the Spinocerebellar Ataxias: Prevalence, Risk Factors, and Association with Falls.

Neuro-ophthalmology (Aeolus Press)
2025

Natural compounds as therapeutic candidates for spinocerebellar ataxia type 1: a computational approach.

In silico pharmacology
2025

Associations between CAG repeat size, brain and spinal cord volume loss, and motor symptoms in spinocerebellar ataxia type 3: a cohort study.

Orphanet journal of rare diseases
2024

Purkinje Cell Dendritic Swellings: A Postmortem Study of Essential Tremor and Other Cerebellar Degenerative Disorders.

Cerebellum (London, England)
2024

An MRI evaluation of white matter involvement in paradigmatic forms of spastic ataxia: results from the multi-center PROSPAX study.

Journal of neurology
2024

Generation of an Induced pluripotent stem cell (iPSC) line (IGIBi011-A) from a Spinocerebellar ataxia type 12 gait dominant patient.

Stem cell research
2024

On the Cut-Off Value of the Anteroposterior Diameter of the Midbrain Atrophy in Spinocerebellar Ataxia Type 2 Patients.

Brain sciences
2024

Spinocerebellar ataxia type 2 has multiple ancestral origins.

Parkinsonism &amp; related disorders
2024

Toward a Better Understanding of Walking Speed in Ataxia of Charlevoix-Saguenay: a Factor Exploratory Study.

Cerebellum (London, England)
2024

Exonic trinucleotide repeat expansions in ZFHX3 cause spinocerebellar ataxia type 4: A poly-glycine disease.

American journal of human genetics
2024

Dilemma in differentiation of spinocerebellar ataxia type 17 from Huntington's disease: comorbidity or independent disease?

The International journal of neuroscience
2023

Case report: Short-term efficacy and changes in 18F-FDG-PET with acute multi-target stimulation in spinocerebellar ataxia type 3 (SCA3/MJD).

Frontiers in neurology
2023

Peripheral polyneuropathy in children and young adults with ataxia-telangiectasia.

European journal of neurology
2024

Computational identification and molecular dynamics simulation of potential circularRNA derived peptide from gene expression profile of Rheumatoid arthritis, Alzheimer's disease, and Atrial fibrillation.

Journal of biomolecular structure &amp; dynamics
2023

First families with spinocerebellar ataxia type 7 in Poland.

Neurologia i neurochirurgia polska
2023

Case report: Variants in the ERCC4 gene as a rare cause of cerebellar ataxia with chorea.

Frontiers in genetics
2022

The roles of HSP40/DNAJ protein family in neurodegenerative diseases.

Zhejiang da xue xue bao. Yi xue ban = Journal of Zhejiang University. Medical sciences
2022

Effect of speech therapy on quality of life in patients with spinocerebelar ataxia type 3.

Arquivos de neuro-psiquiatria
2023

Genotype-Phenotype Correlations for ATX-TBP (SCA17): MDSGene Systematic Review.

Movement disorders : official journal of the Movement Disorder Society
2023

The frequency of non-motor symptoms in SCA3 and their association with disease severity and lifestyle factors.

Journal of neurology
2023

Primary degeneration of oculomotor, motor, and somatosensory systems and auditory and visual pathways in spinocerebellar ataxia type 7: A clinicopathological study in a Japanese autopsy case.

Neuropathology : official journal of the Japanese Society of Neuropathology
2022

[Spinocerebellar ataxia type 8 in Russian patients].

Zhurnal nevrologii i psikhiatrii imeni S.S. Korsakova
2022

Simple and clear differentiation of spinocerebellar degenerations: Overview of macroscopic and low-power view findings.

Neuropathology : official journal of the Japanese Society of Neuropathology
2022

Cannabis use in patients with Autosomal Recessive Spastic Ataxia of Charlevoix-Saguenay.

Journal of clinical neuroscience : official journal of the Neurosurgical Society of Australasia
2022

An autopsy case of amyotrophic lateral sclerosis with striatonigral and pallidoluysian degeneration and cat's-eye-shaped neuronal nuclear inclusions.

Neuropathology : official journal of the Japanese Society of Neuropathology
2022

Clinical, Radiological, and Genetic Profile of Spinocerebellar Ataxia 12: A Hospital-Based Cohort Analysis.

Tremor and other hyperkinetic movements (New York, N.Y.)
2022

Sleep disorders among Aboriginal Australians with Machado-Joseph Disease: Quantitative results from a multiple methods study to assess the experience of people living with the disease and their caregivers.

Neurobiology of sleep and circadian rhythms
2022

Case Report: Late-Onset Autosomal Recessive Cerebellar Ataxia Associated With SYNE1 Mutation in a Chinese Family.

Frontiers in genetics
2022

Progressive Depletion of B and T Lymphocytes in Patients with Ataxia Telangiectasia: Results of the Italian Primary Immunodeficiency Network.

Journal of clinical immunology
2022

Safety and efficacy of riluzole in spinocerebellar ataxia type 2 in France (ATRIL): a multicentre, randomised, double-blind, placebo-controlled trial.

The Lancet. Neurology
2021

Tripeptidyl Peptidase 1 (TPP1) Deficiency in a 36-Year-Old Patient with Cerebellar-Extrapyramidal Syndrome and Dilated Cardiomyopathy.

Life (Basel, Switzerland)
2022

Impaired Oligodendrocyte Maturation Is an Early Feature in SCA3 Disease Pathogenesis.

The Journal of neuroscience : the official journal of the Society for Neuroscience
2022

X-Linked TLR7 Deficiency Underlies Critical COVID-19 Pneumonia in a Male Patient with Ataxia-Telangiectasia.

Journal of clinical immunology
2022

Detection Methods and Status of CAT Interruption of ATXN1 in Korean Patients With Spinocerebellar Ataxia Type 1.

Annals of laboratory medicine
2021

Spastic paraplegia type 46: novel and recurrent GBA2 gene variants in a compound heterozygous Italian patient with spastic ataxia phenotype.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2021

Episodic Vestibulocerebellar Ataxia Associated with a CACNA1G Missense Variant.

Case reports in neurology
2021

Cervical Spinal Cord Degeneration in Spinocerebellar Ataxia Type 7.

AJNR. American journal of neuroradiology
2021

Variants in Genes of Calpain System as Modifiers of Spinocerebellar Ataxia Type 3 Phenotype.

Journal of molecular neuroscience : MN
2021

Spinocerebellar ataxia type 2 from an evolutionary perspective: Systematic review and meta-analysis.

Clinical genetics
2021

Two novel missense variants in SPTBN2 likely associated with spinocerebellar ataxia type 5.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2021

Ataxia with oculomotor apraxia type 2 (AOA2): an eye movement study of two siblings.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2021

Biallelic RFC1-expansion in a French multicentric sporadic ataxia cohort.

Journal of neurology
2021

Evidence for Non-Mendelian Inheritance in Spastic Paraplegia 7.

Movement disorders : official journal of the Movement Disorder Society
2021

A novel SACS p.Pro4154GlnfsTer20 mutation in a family with autosomal recessive spastic ataxia of Charlevoix-Saguenay.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2020

Progression of Cerebellar Atrophy in Spinocerebellar Ataxia Type 2 Gene Carriers: A Longitudinal MRI Study in Preclinical and Early Disease Stages.

Frontiers in neurology
2020

Zonisamide can ameliorate the voltage-dependence alteration of the T-type calcium channel CaV3.1 caused by a mutation responsible for spinocerebellar ataxia.

Molecular brain
2021

Spinocerebellar ataxia type 48: last but not least.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2020

Isolation and Identification of a Novel Anti-protein Aggregation Activity of Lignin-Carbohydrate Complex From Chionanthus retusus Leaves.

Frontiers in bioengineering and biotechnology
2021

Early-onset severe spinocerebellar ataxia 42 with neurodevelopmental deficits (SCA42ND): Case report, pharmacological trial, and literature review.

American journal of medical genetics. Part A
2021

Genetic profile and clinical characteristics of Chinese patients with spinocerebellar ataxia type 2: A multicenter experience over 10 years.

European journal of neurology
2020

Familial spontaneous pneumothorax and Machado-Joseph disease.

Oxford medical case reports
2020

Frequency of Spinocerebellar Ataxia type 1, 2, 3,6 and 7 and clinical profile of Spinocerebellar Ataxia type 3 in Malaysia.

Cerebellum &amp; ataxias
2020

The discriminative capacity of CSF β-amyloid 42 and Tau in neurodegenerative diseases in the Chinese population.

Journal of the neurological sciences
2020

Clonidine GH stimulation test to differentiate MSA from idiopathic late onset cerebellar ataxia: a prospective, controlled study.

Journal of neurology
2020

Assessment of ventilatory function in patients with spinocerebellar ataxia type 2.

Arquivos de neuro-psiquiatria
2020

Congenetic Hybrids Derived from Dearomatized Isoprenylated Acylphloroglucinol with Opposite Effects on Cav3.1 Low Voltage-Gated Ca2+ Channel.

Journal of medicinal chemistry
2020

Infantile-Onset Syndromic Cerebellar Ataxia and CACNA1G Mutations.

Pediatric neurology
2019

Carboxyl Terminus of Hsp70-Interacting Protein Is Increased in Serum and Cerebrospinal Fluid of Patients With Spinocerebellar Ataxia Type 3.

Frontiers in neurology
2019

Ataxic phenotype with altered CaV3.1 channel property in a mouse model for spinocerebellar ataxia 42.

Neurobiology of disease
2019

Prediction of Survival With Long-Term Disease Progression in Most Common Spinocerebellar Ataxia.

Movement disorders : official journal of the Movement Disorder Society
2019

Extra-Cerebellar Signs and Non-motor Features in Chinese Patients With Spinocerebellar Ataxia Type 3.

Frontiers in neurology
2019

Nerve ultrasound as a diagnostic tool for sensory neuronopathy in spinocerebellar ataxia syndrome.

Clinical neurophysiology : official journal of the International Federation of Clinical Neurophysiology
2019

A significant inflation in TGM6 genetic risk casts doubt in its causation in spinocerebellar ataxia type 35.

Parkinsonism &amp; related disorders
2019

Transglutaminase diseases: from biochemistry to the bedside.

FASEB journal : official publication of the Federation of American Societies for Experimental Biology
2019

The movement disorder spectrum of SCA21 (ATX-TMEM240): 3 novel families and systematic review of the literature.

Parkinsonism &amp; related disorders
2019

Treatment of intractable resting tremor of spinocerebellar ataxia 42 with zonisamide.

Journal of the neurological sciences
2019

A case series of hereditary cerebellar ataxias in a highly consanguineous population from Northeast Brazil.

Parkinsonism &amp; related disorders
2018

A case of a novel CACNA1G mutation from a Chinese family with SCA42: A case report and literature review.

Medicine
2018

Sleep apnea in Machado-Joseph disease: a clinical and polysomnographic evaluation.

Sleep medicine
2020

Body composition in Spinocerebellar ataxia type 3 and 10 patients: Comparative study with control group.

Nutritional neuroscience
2018

Expanding the global prevalence of spinocerebellar ataxia type 42.

Neurology. Genetics
2018

Validity and Reliability of Outcome Measures Assessing Dexterity, Coordination, and Upper Limb Strength in Autosomal Recessive Spastic Ataxia of Charlevoix-Saguenay.

Archives of physical medicine and rehabilitation
2018

Lethal form of spinocerebellar ataxia type 7 with early onset in childhood.

Archives de pediatrie : organe officiel de la Societe francaise de pediatrie
2017

Increased biological activity of protein Kinase C gamma is not required in Spinocerebellar ataxia 14.

Molecular brain
2017

Neurological phenotypes in spinocerebellar ataxia type 2: Role of mitochondrial polymorphism A10398G and other risk factors.

Parkinsonism &amp; related disorders
2017

Stance instability in preclinical SCA1 mutation carriers: A 4-year prospective posturography study.

Gait &amp; posture
2017

SCA42 mutation analysis in a case series of Japanese patients with spinocerebellar ataxia.

Journal of human genetics
2017

Polyglutamine expansion of ataxin-3 alters its degree of ubiquitination and phosphorylation at specific sites.

Neurochemistry international
2016

Trinucleotide repeat expansion of TATA-binding protein gene associated with Parkinson's disease: A Thai multicenter study.

Parkinsonism &amp; related disorders
2016

Patterns of motor signs in spinocerebellar ataxia type 3 at the start of follow-up in a reference unit.

Cerebellum &amp; ataxias
2017

Non-motor and Extracerebellar Features in Spinocerebellar Ataxia Type 2.

Cerebellum (London, England)
2015

Mutation analysis of 6 spinocerebellar ataxia (SCA) types in patients from southern Turkey.

Turkish journal of medical sciences
2015

The Social Amoeba Dictyostelium discoideum Is Highly Resistant to Polyglutamine Aggregation.

The Journal of biological chemistry
2015

A Probable Korean Case of Autosomal Recessive Spastic Ataxia of Charlevoix-Saguenay.

The Canadian journal of neurological sciences. Le journal canadien des sciences neurologiques
2016

Polyglutamine aggregation in Huntington's disease and spinocerebellar ataxia type 3: similar mechanisms in aggregate formation.

Neuropathology and applied neurobiology
2015

Cerebellar neurochemical alterations in spinocerebellar ataxia type 14 appear to include glutathione deficiency.

Journal of neurology
2015

Novel candidate blood-based transcriptional biomarkers of Machado-Joseph disease.

Movement disorders : official journal of the Movement Disorder Society
2015

DNA damage primes the type I interferon system via the cytosolic DNA sensor STING to promote anti-microbial innate immunity.

Immunity
Ver todos os 3.032 no EuropePMC

Associações

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Comunidades

Grupos ativos de quem convive com esta doença aqui no Raras

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Doenças relacionadas

Doenças com sintomas parecidos — ajudam quem ainda está buscando diagnóstico

Referências e fontes

Bases de dados externas citadas neste artigo

Publicações científicas

Artigos indexados no PubMed ligados a esta doença no grafo RarasNet — título, periódico e PMID direto da fonte, sem intermediação de IA.

  1. Monitoring the Progression of Pre-Ataxic Gait in SCA2 with Inertial Sensors Over Four Years.
    Cerebellum (London, England)· 2026· PMID 41876820mais citado
  2. Propranolol for Tremors in Spinocerebellar Ataxia Type 12: A Randomized Clinical Trial.
    Movement disorders : official journal of the Movement Disorder Society· 2026· PMID 41261874mais citado
  3. Multidimensional abnormal gait analysis and biomarker identification for patients with spinocerebellar ataxia type 3 using an Azure Kinect-based motion capture system.
    Gait &amp; posture· 2026· PMID 41138689mais citado
  4. Predictive models for ataxia progression and conversion in spinocerebellar ataxia type 1 and 3.
    Brain : a journal of neurology· 2025· PMID 41150672mais citado
  5. An Investigation of Corticospinal Tract Microstructural Integrity in ARSACS Using a Profilometry MRI Analysis: Results From the PROSPAX Study.
    European journal of neurology· 2025· PMID 40241303mais citado
  6. Therapeutic targeting of blood-derived protein infiltration to modulate neuroinflammation in cerebellar ataxia.
    J Neuroinflammation· 2026· PMID 41992320recente
  7. Association Between Cerebellar Metabolic Markers and Activities of Daily Living in Patients With Spinocerebellar Ataxia Type 3.
    Mol Genet Genomic Med· 2026· PMID 41983302recente
  8. Long term administration of selective NMDA GluN2B receptor blocker Ro25-6981 attenuates neurodegeneration in mouse model of spinocerebellar ataxia type 1 (SCA1).
    Cell Death Discov· 2026· PMID 41974656recente
  9. Cell-Based Therapies for Spinocerebellar Degenerations: A Systematic Review of Human Clinical Evidence.
    Cerebellum· 2026· PMID 41968234recente
  10. Comprehensive Review of Anesthetic Strategies for Patients With Neurodegenerative Diseases.
    Med Sci Monit· 2026· PMID 41964193recente

Bases de dados e fontes oficiais

Identificadores e referências canônicas usadas para montar este verbete.

  1. ORPHA:458803(Orphanet)
  2. OMIM OMIM:616795(OMIM)
  3. MONDO:0014776(MONDO)
  4. GARD:17811(GARD (NIH))
  5. Variantes catalogadas(ClinVar)
  6. Busca completa no PubMed(PubMed)
  7. Q55345906(Wikidata)

Dados compilados pelo RarasNet a partir de fontes abertas (Orphanet, OMIM, MONDO, PubMed/EuropePMC, ClinicalTrials.gov, DATASUS, PCDT/MS). Este conteúdo é informativo e não substitui avaliação médica.

Conteúdo mantido por Agente Raras · Médicos e pesquisadores podem colaborar

Ataxia espinocerebelosa tipo 42
Compêndio · Raras BR

Ataxia espinocerebelosa tipo 42

ORPHA:458803 · MONDO:0014776
Prevalência
<1 / 1 000 000
Casos
25 casos conhecidos
Herança
Autosomal dominant
CID-10
G11.8 · Outras ataxias hereditárias
Início
Adolescent, Adult, Childhood, Elderly
Prevalência
0.0 (Worldwide)
MedGen
UMLS
C4225205
Repurposing
1 candidato
taltirelinthyrotropin releasing hormone receptor agonist
EuropePMC
Wikidata
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