Literature DB >> 30713867

The Clinical Spectrum of Autosomal-Dominant Episodic Ataxias.

Stefan Kipfer1, Michael Strupp2.   

Abstract

Autosomal-dominant episodic ataxias (EAs) represent a clinically and genetically heterogeneous group of disorders characterized by recurrent episodes of cerebellar ataxia (CA). Ataxia episodes are usually of short duration and often triggered by specific stimuli. There are currently seven classified subtypes of EA. EA types 1 and 2 have the highest prevalence and are therefore the clinically most relevant. Between attacks, EA 1 is associated with myokymia. In EA 2, often an interictal downbeat nystagmus with other cerebellar ocular dysfunctions is present; patients with EA 2 may display slowly progessive ataxia and vermian atrophy. EA 1 and 2 are both channelopathies, affecting the potassium channel gene, KCNA1, in EA 1 and the PQ calcium channel-encoding gene, CACNA1A, in EA 2. The types EA 3 to 7 are very rare and have to be further elucidated. Here, we review the historical, clinical, and genetic aspects of autosomal-dominant EAs and their current treatment, focusing on EA 1 and 2.

Entities:  

Keywords:  CACNA1A; CACNB4; KCNA1; SLC1A3; channelopathy; episodic ataxia

Year:  2014        PMID: 30713867      PMCID: PMC6353419          DOI: 10.1002/mdc3.12075

Source DB:  PubMed          Journal:  Mov Disord Clin Pract        ISSN: 2330-1619


  71 in total

1.  Clinical, genetic, and expression studies of mutations in the potassium channel gene KCNA1 reveal new phenotypic variability.

Authors:  L H Eunson; R Rea; S M Zuberi; S Youroukos; C P Panayiotopoulos; R Liguori; P Avoni; R C McWilliam; J B Stephenson; M G Hanna; D M Kullmann; A Spauschus
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2.  The clinical spectrum of familial hemiplegic migraine associated with mutations in a neuronal calcium channel.

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Journal:  N Engl J Med       Date:  2001-07-05       Impact factor: 91.245

3.  A novel mutation in the human voltage-gated potassium channel gene (Kv1.1) associates with episodic ataxia type 1 and sometimes with partial epilepsy.

Authors:  S M Zuberi; L H Eunson; A Spauschus; R De Silva; J Tolmie; N W Wood; R C McWilliam; J B Stephenson; J P Stephenson; D M Kullmann; M G Hanna
Journal:  Brain       Date:  1999-05       Impact factor: 13.501

4.  Loss-of-function EA2 mutations are associated with impaired neuromuscular transmission.

Authors:  J Jen; J Wan; M Graves; H Yu; A F Mock; C J Coulin; G Kim; Q Yue; D M Papazian; R W Baloh
Journal:  Neurology       Date:  2001-11-27       Impact factor: 9.910

5.  Expression in mammalian cells and electrophysiological characterization of two mutant Kv1.1 channels causing episodic ataxia type 1 (EA-1).

Authors:  F Bretschneider; A Wrisch; F Lehmann-Horn; S Grissmer
Journal:  Eur J Neurosci       Date:  1999-07       Impact factor: 3.386

6.  An autosomal dominant disorder with episodic ataxia, vertigo, and tinnitus.

Authors:  J L Steckley; G C Ebers; M Z Cader; R S McLachlan
Journal:  Neurology       Date:  2001-10-23       Impact factor: 9.910

7.  High prevalence of CACNA1A truncations and broader clinical spectrum in episodic ataxia type 2.

Authors:  C Denier; A Ducros; K Vahedi; A Joutel; P Thierry; A Ritz; G Castelnovo; T Deonna; P Gérard; J L Devoize; A Gayou; B Perrouty; T Soisson; A Autret; J M Warter; A Vighetto; P Van Bogaert; S Alamowitch; E Roullet; E Tournier-Lasserve
Journal:  Neurology       Date:  1999-06-10       Impact factor: 9.910

8.  Recent progress on the molecular organization of myelinated axons.

Authors:  Steven S Scherer; Edgardo J Arroyo
Journal:  J Peripher Nerv Syst       Date:  2002-03       Impact factor: 3.494

9.  Coding and noncoding variation of the human calcium-channel beta4-subunit gene CACNB4 in patients with idiopathic generalized epilepsy and episodic ataxia.

Authors:  A Escayg; M De Waard; D D Lee; D Bichet; P Wolf; T Mayer; J Johnston; R Baloh; T Sander; M H Meisler
Journal:  Am J Hum Genet       Date:  2000-04-04       Impact factor: 11.025

10.  Hereditary myokymia and periodic ataxia.

Authors:  D H VanDyke; R C Griggs; M J Murphy; M N Goldstein
Journal:  J Neurol Sci       Date:  1975-05       Impact factor: 3.181

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  4 in total

1.  A Novel KCNA1 Mutation in an Episodic Ataxia Type 1 Patient with Asterixis and Falls.

Authors:  Geum Bong Lee; Ga Yeon Kim; In Hwa Jeong; Namhee Kim; Jae Woo Kim
Journal:  J Clin Neurol       Date:  2021-04       Impact factor: 3.077

Review 2.  Paroxysmal Movement Disorders.

Authors:  Susan Harvey; Mary D King; Kathleen M Gorman
Journal:  Front Neurol       Date:  2021-06-11       Impact factor: 4.003

Review 3.  Episodic Ataxias: Faux or Real?

Authors:  Paola Giunti; Elide Mantuano; Marina Frontali
Journal:  Int J Mol Sci       Date:  2020-09-05       Impact factor: 5.923

4.  A novel pathogenic CACNA1A variant causing episodic ataxia type 2 (EA2) spectrum phenotype in four family members and a novel combined therapy.

Authors:  Josef Penkava; S Ledderose; S Chahrokh-Zadeh; A Munzig; Zu Eulenburg; D Huppert; M Strupp; S Becker-Bense
Journal:  J Neurol       Date:  2020-09-10       Impact factor: 4.849

  4 in total

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