Raras
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Ataxia espinocerebelosa tipo 45
ORPHA:589527CID-10 · G11.8CID-11 · 8A03.16OMIM 617769DOENÇA RARA

Ataxia cerebelosa autossómica dominante rara, caracterizada por ataxia dos membros e da marcha lentamente progressiva e de início tardio, disartria e nistagmo variável. Os exames de neuroimagem cerebral demonstram atrofia cerebelosa.

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Introdução

O que você precisa saber de cara

📋

Ataxia cerebelosa autossômica dominante rara, caracterizada por ataxia dos membros e da marcha lentamente progressiva e de início tardio, disartria e nistagmo variável. Os exames de neuroimagem cerebral demonstram atrofia cerebelosa.

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
7
pacientes catalogados
Início
Adult
🏥
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

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Sinais e sintomas

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

Partes do corpo afetadas

🧠
Neurológico
4 sintomas
👁️
Olhos
1 sintomas

+ 3 sintomas em outras categorias

Características mais comuns

100%prev.
Disartria
Obrigatório (100%)
100%prev.
Ataxia da marcha
Obrigatório (100%)
100%prev.
Nistagmo de batimento para baixo
Obrigatório (100%)
100%prev.
Ataxia de membro
Obrigatório (100%)
100%prev.
Atrofia cerebelar
Obrigatório (100%)
100%prev.
HP:0003596
Obrigatório (100%)
8sintomas
Muito frequente (6)
Sem dados (2)

Os sintomas variam de pessoa para pessoa. Abaixo estão as 8 características clínicas mais associadas, ordenadas por frequência.

DisartriaDysarthria
Obrigatório (100%)100%
Ataxia da marchaGait ataxia
Obrigatório (100%)100%
Nistagmo de batimento para baixoDownbeat nystagmus
Obrigatório (100%)100%
Ataxia de membroLimb ataxia
Obrigatório (100%)100%
Atrofia cerebelarCerebellar atrophy
Obrigatório (100%)100%

Linha do tempo da pesquisa

Publicações por ano — veja quando o interesse científico cresceu
Anos de pesquisa11
Total histórico8.166PubMed
Últimos 10 anos99publicações
Pico202416 papers
Linha do tempo
20202015Hoje · 2026📈 2024Ano 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.

FAT2Protocadherin Fat 2Disease-causing germline mutation(s) inRestrito
FUNÇÃO

Involved in the regulation of cell migration (PubMed:18534823). May be involved in mediating the organization of the parallel fibers of granule cells during cerebellar development (By similarity)

LOCALIZAÇÃO

Cell membraneCell junctionGolgi apparatus, trans-Golgi network

MECANISMO DE DOENÇA

Spinocerebellar ataxia 45

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. SCA45 is a slowly progressive, autosomal dominant form with onset in adulthood.

EXPRESSÃO TECIDUAL(Tecido-específico)
Cerebelo
223.9 TPM
Cérebro - Hemisfério cerebelar
179.8 TPM
Esôfago - Mucosa
38.2 TPM
Skin Not Sun Exposed Suprapubic
31.1 TPM
Skin Sun Exposed Lower leg
28.4 TPM
INTERAÇÕES PROTEICAS (1)
OUTRAS DOENÇAS (1)
spinocerebellar ataxia 45
HGNC:3596UniProt:Q9NYQ8

Variantes genéticas (ClinVar)

70 variantes patogênicas registradas no ClinVar.

🧬 FAT2: NM_001447.3(FAT2):c.4625C>A (p.Thr1542Asn) ()
🧬 FAT2: NM_001447.3(FAT2):c.1150G>A (p.Val384Met) ()
🧬 FAT2: NM_001447.3(FAT2):c.7652G>A (p.Arg2551Lys) ()
🧬 FAT2: NM_001447.3(FAT2):c.12853G>A (p.Gly4285Arg) ()
🧬 FAT2: NM_001447.3(FAT2):c.6088C>T (p.Arg2030Trp) ()
Ver todas no ClinVar

Classificação de variantes (ClinVar)

Distribuição de 5,215 variantes classificadas pelo ClinVar.

1043
1043
3129
Patogênica (20.0%)
VUS (20.0%)
Benigna (60.0%)
VARIANTES MAIS SIGNIFICATIVAS
FGF14: NM_004115.4(FGF14):c.353G>T (p.Gly118Val) [Pathogenic]
GRM1: NM_001278064.1(GRM1):c.[2652_2654delGAA;2660+2T>G] [Pathogenic]
PUM1: NM_001020658.2(PUM1):c.1544dup (p.Asn516fs) [Likely pathogenic]
PUM1: GRCh38/hg38 1p35.2(chr1:30936422-30948423)x3 [Pathogenic]
TDP1: NM_018319.4(TDP1):c.1317G>A (p.Leu439=) [Uncertain significance]

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 45

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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

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Publicações mais relevantes

Timeline de publicações
3.160 papers (10 anos)

Mostrando amostra de 99 publicações de um total de 3.160

#1

The insertion of an ATTTC repeat in an Alu element hyperactivates a neurodevelopmental enhancer in spinocerebellar ataxia type 37.

Cell reports2026 Mar 22

Alu elements are evolutionarily very old primate-specific interspersed repeat elements that constitute ∼11% of the human genome. They are a source of short tandem repeats (STRs), which often expand in size and cause inherited neuromuscular and neurodegenerative disorders. How expanded STR insertion mutations within Alu STRs culminate in disease remains unknown. Here, we report an Alu STR located in an intron of DAB1 that functions as a neurodevelopmental enhancer. We demonstrate that an ATTTC repeat insertion in this DAB1 Alu STR, known to cause spinocerebellar ataxia type 37 (SCA37), hyperactivates a neurodevelopmental DAB1 enhancer. Importantly, we show that neurons derived from SCA37 subjects have higher levels of DAB1 expression and that DAB1 overexpression causes abnormal axonal pathfinding in vivo. Overall, these results establish that neuronal dysregulation of a developmental DAB1 Alu STR enhancer contributes to SCA37 pathogenesis, an unexplored mechanism likely acting in many Alu STR diseases, potentially reshaping the therapeutic landscape.

#2

Remote Assessment of Ataxia Severity in SCA3 Across Multiple Centers and Time Points.

Annals of clinical and translational neurology2026 Jan 22

Spinocerebellar ataxia type 3 (SCA3) is a genetically defined ataxia. The Scale for Assessment and Rating of Ataxia (SARA) is a clinician-reported outcome that measures ataxia severity at a single time point. In its standard application, SARA fails to capture short-term fluctuations, limiting its sensitivity in trials. To overcome this, we employed SARAhome, a video-based, self-administered tool for high-frequency, remote ataxia assessment. We assessed feasibility and validity of SARAhome in 65 SCA3 patients from seven centers. Participants recorded SARAhome twice daily for 14 days using a mobile e-health app. We analyzed adherence, intraindividual fluctuations and their predictors, and evaluated sensitivity to change in a longitudinal substudy of 11 patients. Adherence to the study protocol was generally high (80.2%) with valid scores in 79.2% of 1459 recordings. Maximum adherence occurred over a 4-day period (84.8%). Fluctuations ranged 3.0 points between lowest and highest scores (IQR: 2.5-4.5) and 1.0 point based on score IQRs (IQR: 0.5-1.5), corresponding to 10.7% and 3.6% of the maximal SARAhome score. Fluctuations showed rough agreement with patient global impression. Greater disease severity and longer CAG repeats were associated with smaller relative fluctuations. Over a median follow-up of 411 days, SARAhome showed higher sensitivity to change than conventional SARA (SRM: 0.67 vs. 0.37). SARAhome is a feasible, innovative video-based tool for remote, high-frequency monitoring of ataxia severity. A 4-day recording effectively captures relevant fluctuations and enhances sensitivity to change, supporting its use in future SCA3 trials.

#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

The impact of interrupted ATXN10 expansions on clinical findings of spinocerebellar ataxia type 10.

Journal of neurology2025 Mar 11

Spinocerebellar ataxia type 10 (SCA10), due to an ATTCT repeat expansion in ATXN10, has variable expressivity and the role of presence (ATTCTint +) and absence (ATTCTint-) of interruptions in the repeat is not clear. We aimed to describe the relations between ATTCTint + and age at onset, seizures, and neurologic severity in ataxic and non-ataxic carriers from Brazil. Family, age at onset (AO), and seizures data plus DNA were obtained from symptomatic carriers already diagnosed in Porto Alegre, Curitiba, and São Paulo, Brazil. Patients and their relatives were invited to be evaluated through Scale of Assessment and Rating of Ataxia (SARA) and other clinical scales; a SARA > 2.5 classified subjects as ataxic carriers. Repeat-primed PCR (RP-PCR) defined the expansions with (ATTCTint +) or without (ATTCTint-) interruptions. Comparisons were performed for a p level of 0.05. Among 78 ataxic carriers, earlier AO (p = 0.039) and higher occurrences of epilepsy (p < 0.0001) were seen in subjects with ATTCTint + than in those with ATTCTint-. Clinical scales were worse in 34 ataxics than in 7 non-ataxics and 10 related controls (p = 0.006) and did not discriminate non-ataxics from controls. The 11 ataxic ATTCTint + carriers had higher SARA scores per year of disease duration than the 23 ATTCTint- carriers (r = 0.879, beta = 0.45, p = 0.0001). ATTCTint + carriers had worse clinical findings than ATTCTint- carriers: earlier AO, more seizures, and worse ataxia scores. Interruptions in the expanded repeat have a real impact in SCA10 phenotype.

#5

Cerebellar cognitive affective syndrome in patients with spinocerebellar ataxia type 10.

PloS one2025

Spinocerebellar ataxia type 10 (SCA10) is an autosomal dominant cerebellar ataxia, characterized by epilepsy, ataxic symptoms, and cognitive impairments linked to Cerebellar Cognitive Affective Syndrome (CCAS). The Cerebellar Cognitive Affective Syndrome Scale (CCAS-S) has been developed to identify CCAS across various cerebellar pathologies. To determine whether patients with SCA10 exhibit CCAS using the CCAS-S, and to compare its effectiveness with the Montreal Cognitive Assessment (MoCA). A secondary objective was to evaluate the effect of demographic and clinical data on CCAS-S performance. Fifteen patients with SCA10 and fifteen matched controls underwent assessments using the CCAS-S, the MoCA, the Scale for the Assessment and Rating of Ataxia (SARA), and the Center for Epidemiologic Studies Depression Scale (CES-D). Diagnostic accuracy was analyzed using ROC curve analysis, comparing total and subcategory scores between groups. Demographic and clinical data were examined for relations with CCAS-S scores. The CCAS-S effectively distinguished cognitive impairments in SCA10 patients, showing satisfactory sensitivity and specificity (AUC of 0.83). Although no significant differences were found in the AUCs between CCAS-S and MoCA (p =  0.45), the CCAS-S demonstrated a significantly larger effect size in the comparison between patients and control group (d =  2.33). Cognitive performance was poorer in patients than in controls (p =  < 0.001), with depressive symptoms and age having a significant impact on CCAS-S outcomes. Patients with the SCA10 mutation exhibit CCAS. Besides the significant cognitive impairment, also detected by MoCA, the CCAS-S score was significantly affected by indicators of depressive mood and age, highlighting the importance of considering these variables during outcome analyses.

Publicações recentes

Ver todas no PubMed

📚 EuropePMC3.032 artigos no totalmostrando 99

2026

The insertion of an ATTTC repeat in an Alu element hyperactivates a neurodevelopmental enhancer in spinocerebellar ataxia type 37.

Cell reports
2026

Remote Assessment of Ataxia Severity in SCA3 Across Multiple Centers and Time Points.

Annals of clinical and translational neurology
2025

Interpretable machine learning for differentiating SCA3 and MSA-C using gait and postural features from wearable sensors.

Journal of neuroengineering and rehabilitation
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

Naphthyridine carbamate dimer ligand induces formation of Z-RNA-like fold of disease-related RNA and exhibits a molecular glue characteristics in crystal lattice formation.

Nucleic acids research
2025

Spinocerebellar Ataxia Type 27B can be Suspected Based on Clinical Phenotype: The Massachusetts General Hospital Ataxia Center Experience.

Cerebellum (London, England)
2025

The impact of interrupted ATXN10 expansions on clinical findings of spinocerebellar ataxia type 10.

Journal of neurology
2025

Cerebellar cognitive affective syndrome in patients with spinocerebellar ataxia type 10.

PloS one
2025

Predicting Which Mitophagy Proteins Are Dysregulated in Spinocerebellar Ataxia Type 3 (SCA3) Using the Auto-p2docking Pipeline.

International journal of molecular sciences
2025

Effects of trace element dysregulation on brain structure and function in spinocerebellar Ataxia type 3.

Neurobiology of disease
2025

Demystifying the Etiology of ILOCA in the Genomic Era: A Narrative Review.

Cerebellum (London, England)
2025

Novel Intermediate ATXN10 Alleles in the Healthy Peruvian Population: A Matter of Indigenous American Ethnic Origin.

Cerebellum (London, England)
2024

Spinocerebellar Ataxias: Phenotypic Spectrum of PolyQ versus Non-Repeat Expansion Forms.

Cerebellum (London, England)
2024

Spinocerebellar ataxia type 27B (SCA27B) in India: insights from a large cohort study suggest ancient origin.

Neurogenetics
2024

Imbalanced optimal feedback motor control system in spinocerebellar ataxia type 3.

European journal of neurology
2024

MRI-ARSACS: An Imaging Index for Autosomal Recessive Spastic Ataxia of Charlevoix-Saguenay (ARSACS) Identification Based on the Multicenter PROSPAX Study.

Movement disorders : official journal of the Movement Disorder Society
2024

CACNA1A variant associated with generalized dystonia.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2025

Clinical Presentation and Neuro-Ophthalmological Features in Spinocerebellar Ataxia Type 3: A Case Report and Literature Review.

Journal of neuro-ophthalmology : the official journal of the North American Neuro-Ophthalmology Society
2024

Rare association between spinocerebellar ataxia and amyotrophic lateral sclerosis: a case series.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2024

Dynamic molecular network analysis of iPSC-Purkinje cells differentiation delineates roles of ISG15 in SCA1 at the earliest stage.

Communications biology
2024

Co-existence of Myelin Oligodendrocyte Glycoprotien Antibody-associated Disease (MOGAD) and Spinocerebellar Ataxia type 1 (SCA1): A case report.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2024

Novel heterozygous PRPH2 variant identified in a patient with spinocerebellar ataxia type 14 and macular dystrophy.

Ophthalmic genetics
2024

Compressed cerebellar functional connectome hierarchy in spinocerebellar ataxia type 3.

Human brain mapping
2024

Cranial Nerve Thinning Distinguishes RFC1-Related Disorder from Other Late-Onset Ataxias.

Movement disorders clinical practice
2024

Subcellular localization and ER-mediated cytotoxic function of α1A and α1ACT in spinocerebellar ataxia type 6.

Biochemical and biophysical research communications
2024

Spinocerebellar ataxia type 2 has multiple ancestral origins.

Parkinsonism &amp; related disorders
2024

Autosomal recessive spino-cerebellar ataxia type 10 (SCAR10): clinical presentation associated with c.289delA ANO10 gene variant.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2024

Three Hertz postural leg tremor impairs posture maintenance in multiple system atrophy-cerebellar type.

Neurological sciences : official journal of the Italian Neurological Society and of the Italian Society of Clinical Neurophysiology
2023

A boy with a progressive neurologic decline harboring two coexisting mutations in KMT2D and VPS13D.

Brain &amp; development
2023

A novel ELOVL4 variant, L168S, causes early childhood-onset Spinocerebellar ataxia-34 and retinal dysfunction: a case report.

Acta neuropathologica communications
2023

Composite autonomic severity scoring in spinocerebellar ataxia type 1 and 2.

Journal of clinical neuroscience : official journal of the Neurosurgical Society of Australasia
2024

Temporal Relationship between Impairment of Cerebellar Motor Learning and Deterioration of Ataxia in Patients with Cerebellar Degeneration.

Cerebellum (London, England)
2023

Systematic assessment of plasma biomarkers in spinocerebellar ataxia.

Neurobiology of disease
2023

Age-related differences of cerebellar cortex and nuclei: MRI findings in healthy controls and its application to spinocerebellar ataxia (SCA6) patients.

NeuroImage
2023

Gene therapy for selected neuromuscular and trinucleotide repeat disorders - An insight to subsume South Asia for multicenter clinical trials.

IBRO neuroscience reports
2023

Baseline Clinical and Blood Biomarkers in Patients With Preataxic and Early-Stage Disease Spinocerebellar Ataxia 1 and 3.

Neurology
2023

Spinocerebellar ataxia type 31: A clinical and radiological literature review.

Journal of the neurological sciences
2023

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

Movement disorders : official journal of the Movement Disorder Society
2022

Long-read sequencing identified intronic (GGCCTG)n expansion in NOP56 in one SCA36 family and literature review.

Clinical neurology and neurosurgery
2023

Cerebello-cerebral resting-state functional connectivity in spinocerebellar ataxia type 3.

Human brain mapping
2022

Moyamoya associated with Turner syndrome in a patient with type 2 spinocerebellar ataxia-Occam's razor or Hickam's dictum: a case report.

BMC neurology
2022

Cerebellar glutamatergic system impacts spontaneous motor recovery by regulating Gria1 expression.

NPJ Regenerative medicine
2022

Compounds activating VCP D1 ATPase enhance both autophagic and proteasomal neurotoxic protein clearance.

Nature communications
2023

Thymidine Kinase 2 and Mitochondrial Protein COX I in the Cerebellum of Patients with Spinocerebellar Ataxia Type 31 Caused by Penta-nucleotide Repeats (TTCCA)n.

Cerebellum (London, England)
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
2022

Amyotrophic lateral sclerosis associated with a pathological expansion in the ATXN7 gene.

Amyotrophic lateral sclerosis &amp; frontotemporal degeneration
2021

Neuropathology of SCA34 showing widespread oligodendroglial pathology with vacuolar white matter degeneration: a case study.

Acta neuropathologica communications
2021

Midbrain atrophy related to parkinsonism in a non-coding repeat expansion disorder: five cases of spinocerebellar ataxia type 31 with nigrostriatal dopaminergic dysfunction.

Cerebellum &amp; ataxias
2021

The electrophysiological footprint of CACNA1A disorders.

Journal of neurology
2023

Wheelchair mobility, motor performance and participation of adult wheelchair users with ARSACS: a cross-sectional study.

Disability and rehabilitation. Assistive technology
2021

Selective forces acting on spinocerebellar ataxia type 3/Machado-Joseph disease recurrency: A systematic review and meta-analysis.

Clinical genetics
2020

Association of serum neurofilament light and disease severity in patients with spinocerebellar ataxia type 3.

Neurology
2020

Generation of human iPS cell line IBCHi002-A from spinocerebellar ataxia type 3/Machado-Joseph disease patient's fibroblasts.

Stem cell research
2020

Generation of induced pluripotent stem cell line (CSUXHi002-A) from a patient with spinocerebellar ataxia type 1.

Stem cell research
2020

Comorbid argyrophilic grain disease in an 87-year-old male with spinocerebellar ataxia type 31 with dementia: a case report.

BMC neurology
2020

Extending the Phenotypic Spectrum Associated with STUB1 Mutations: A Case of Dystonia.

Movement disorders clinical practice
2020

Supratentorial and Infratentorial Lesions in Spinocerebellar Ataxia Type 3.

Frontiers in neurology
2019

In Human and Mouse Spino-Cerebellar Tissue, Ataxin-2 Expansion Affects Ceramide-Sphingomyelin Metabolism.

International journal of molecular sciences
2019

Novel compound heterozygous mutations in the WWOX gene cause early infantile epileptic encephalopathy.

International journal of developmental neuroscience : the official journal of the International Society for Developmental Neuroscience
2019

The structural basis of lipid scrambling and inactivation in the endoplasmic reticulum scramblase TMEM16K.

Nature communications
2019

Investigating PUM1 mutations in a Taiwanese cohort with cerebellar ataxia.

Parkinsonism &amp; related disorders
2019

Spinocerebellar ataxias in Southern Brazil: Genotypic and phenotypic evaluation of 213 families.

Clinical neurology and neurosurgery
2019

Identification of a novel mutation in the CACNA1C gene in a Chinese family with autosomal dominant cerebellar ataxia.

BMC neurology
2019

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

Neurobiology of disease
2019

Prevalence and clinicoradiological features of spinocerebellar ataxia type 34 in a Japanese ataxia cohort.

Parkinsonism &amp; related disorders
2019

Heterozygous missense variants of SPTBN2 are a frequent cause of congenital cerebellar ataxia.

Clinical genetics
2019

Clinical and molecular studies in two new cases of ARSACS.

Neurogenetics
2019

Short Review: Investigating ARSACS: models for understanding cerebellar degeneration.

Neuropathology and applied neurobiology
2018

Coiled-coil structure-dependent interactions between polyQ proteins and Foxo lead to dendrite pathology and behavioral defects.

Proceedings of the National Academy of Sciences of the United States of America
2018

Serum neurofilament light is increased in multiple system atrophy of cerebellar type and in repeat-expansion spinocerebellar ataxias: a pilot study.

Journal of neurology
2020

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

Nutritional neuroscience
2018

Evaluation of Various Movement Disorders in Patients of Genetically Proven Spinocerebellar Ataxia: A Study from a Tertiary Care Center in Northern India.

Annals of Indian Academy of Neurology
2018

Incidentalome in Neurogenetics: Pathogenic Variant of NSD1 in a Patient With Spinocerebellar Ataxia (SCA).

Frontiers in genetics
2018

A diagnostic decision tree for adult cerebellar ataxia based on pontine magnetic resonance imaging.

Journal of the neurological sciences
2018

Optical coherence tomography in autosomal recessive spastic ataxia of Charlevoix-Saguenay.

Brain : a journal of neurology
2018

The progression rate of spinocerebellar ataxia type 2 changes with stage of disease.

Orphanet journal of rare diseases
2018

Inositol 1,4,5-trisphosphate receptors and neurodegenerative disorders.

The FEBS journal
2017

Impact of disease duration on functional status of patients with spinocerebellar ataxia type 2.

Arquivos de neuro-psiquiatria
2018

Identification of a Splicing Mutation in ITPR1 via WES in a Chinese Early-Onset Spinocerebellar Ataxia Family.

Cerebellum (London, England)
2017

Developmental YAPdeltaC determines adult pathology in a model of spinocerebellar ataxia type 1.

Nature communications
2017

Sequence configuration of spinocerebellar ataxia type 8 repeat expansions in a Japanese cohort of 797 ataxia subjects.

Journal of the neurological sciences
2017

A novel gain-of-function mutation in the ITPR1 suppressor domain causes spinocerebellar ataxia with altered Ca2+ signal patterns.

Journal of neurology
2017

Knockdown and replacement therapy mediated by artificial mirtrons in spinocerebellar ataxia 7.

Nucleic acids research
2018

Calcium Signaling, PKC Gamma, IP3R1 and CAR8 Link Spinocerebellar Ataxias and Purkinje Cell Dendritic Development.

Current neuropharmacology
2017

Cancer in Machado-Joseph disease patients-low frequency as a cause of death.

Cancer genetics
2017

Regulatory Role of RNA Chaperone TDP-43 for RNA Misfolding and Repeat-Associated Translation in SCA31.

Neuron
2017

Gene dosage effect in spinocerebellar ataxia type 6 homozygotes: A clinical and neuropathological study.

Journal of the neurological sciences
2017

Rats with a missense mutation in Atm display neuroinflammation and neurodegeneration subsequent to accumulation of cytosolic DNA following unrepaired DNA damage.

Journal of leukocyte biology
2017

Natural History of Spinocerebellar Ataxia Type 31: a 4-Year Prospective Study.

Cerebellum (London, England)
2016

Genome-wide association study for hereditary ataxia in the Parson Russell Terrier and DNA-testing for ataxia-associated mutations in the Parson and Jack Russell Terrier.

BMC veterinary research
2016

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

Parkinsonism &amp; related disorders
2015

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

Turkish journal of medical sciences
2016

Liver Disease in Pediatric Patients With Ataxia Telangiectasia: A Novel Report.

Journal of pediatric gastroenterology and nutrition
2015

Abnormalities in synaptic dynamics during development in a mouse model of spinocerebellar ataxia type 1.

Scientific reports
2015

Population genetics and new insight into range of CAG repeats of spinocerebellar ataxia type 3 in the Han Chinese population.

PloS one
2015

Central auditory processing in patients with spinocerebellar ataxia.

Hearing research
2015

Psychiatric disorders, spinocerebellar ataxia type 3 and CAG expansion.

Journal of neurology
2015

Decreased metabotropic glutamate receptor type 1 availability in a patient with spinocerebellar ataxia type 6: A (11)C-ITMM PET study.

Journal of the neurological sciences
2015

Altered p53 and NOX1 activity cause bioenergetic defects in a SCA7 polyglutamine disease model.

Biochimica et biophysica acta
Ver todos os 3.032 no EuropePMC

Associações

Organizações que acompanham esta doença — pra ter apoio e orientação

Associação brasileira dedicada a Doença de Machado-Joseph.

Comunidades

Grupos ativos de quem convive com esta doença aqui no Raras

Ainda não existe comunidade no Raras para Ataxia espinocerebelosa tipo 45

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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. The insertion of an ATTTC repeat in an Alu element hyperactivates a neurodevelopmental enhancer in spinocerebellar ataxia type 37.
    Cell reports· 2026· PMID 41871099mais citado
  2. Remote Assessment of Ataxia Severity in SCA3 Across Multiple Centers and Time Points.
    Annals of clinical and translational neurology· 2026· PMID 41568670mais 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. The impact of interrupted ATXN10 expansions on clinical findings of spinocerebellar ataxia type 10.
    Journal of neurology· 2025· PMID 40067487mais citado
  5. Cerebellar cognitive affective syndrome in patients with spinocerebellar ataxia type 10.
    PloS one· 2025· PMID 40029932mais 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:589527(Orphanet)
  2. OMIM OMIM:617769(OMIM)
  3. MONDO:0033480(MONDO)
  4. GARD:22353(GARD (NIH))
  5. Variantes catalogadas(ClinVar)
  6. Busca completa no PubMed(PubMed)

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 45
Compêndio · Raras BR

Ataxia espinocerebelosa tipo 45

ORPHA:589527 · MONDO:0033480
Prevalência
<1 / 1 000 000
Casos
7 casos conhecidos
Herança
Autosomal dominant
CID-10
G11.8 · Outras ataxias hereditárias
CID-11
Início
Adult
Prevalência
0.0 (Worldwide)
MedGen
UMLS
C4540400
Repurposing
1 candidato
taltirelinthyrotropin releasing hormone receptor agonist
EuropePMC
Papers 10a
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