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Movement Disorders (revue)

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Progressive delayed-onset dystonia after cerebral anoxic insult in adults

Identifieur interne : 000733 ( PascalFrancis/Curation ); précédent : 000732; suivant : 000734

Progressive delayed-onset dystonia after cerebral anoxic insult in adults

Auteurs : Mikko Kuoppam Ki [Royaume-Uni] ; Kailash P. Bhatia [Royaume-Uni] ; Niall Quinn [Royaume-Uni]

Source :

RBID : Pascal:03-0038718

Descripteurs français

English descriptors

Abstract

The basal ganglia, especially the globi pallidi (GP), are highly vulnerable to generalized cerebral anoxia/hypoxia. We report on 2 new cases with delayed-onset generalized dystonia due to cerebral anoxia. The onset of dystonia in both of our patients was delayed by about 2 months. In both cases, the unusual feature was the progressive worsening and the spread of dystonia over many years after delayed onset. Dystonia progressed for 16 years in Case 1 and for 4 years in Case 2. Furthermore, initial magnetic resonance imaging (MRI) scan of Case I showed mild changes of the internal capsule sparing the basal ganglia. Years later, in line with clinical progression, the follow-up MRI scan showed isolated bilateral lesions involving the entire GP. MRI scans in Case 2 showed bilateral lesions of caudate and lentiform nuclei. There may be several mechanisms underlying delayed and progressive symptoms after time-limited brain anoxia. We hypothesize that anoxia-induced excitotoxicity resulting in mitochondrial dysfunction and subsequent apoptosis may explain, at least partly, the delayed-onset and progressive extrapyramidal syndromes seen in these patients.
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A11 01  1    @1 KUOPPAMÄKI (Mikko)
A11 02  1    @1 BHATIA (Kailash P.)
A11 03  1    @1 QUINN (Niall)
A14 01      @1 Sobell Department of Motor Neuroscience and Movement Disorders, Institute of Neurology, Queen Square @2 London @3 GBR @Z 1 aut. @Z 2 aut. @Z 3 aut.
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C01 01    ENG  @0 The basal ganglia, especially the globi pallidi (GP), are highly vulnerable to generalized cerebral anoxia/hypoxia. We report on 2 new cases with delayed-onset generalized dystonia due to cerebral anoxia. The onset of dystonia in both of our patients was delayed by about 2 months. In both cases, the unusual feature was the progressive worsening and the spread of dystonia over many years after delayed onset. Dystonia progressed for 16 years in Case 1 and for 4 years in Case 2. Furthermore, initial magnetic resonance imaging (MRI) scan of Case I showed mild changes of the internal capsule sparing the basal ganglia. Years later, in line with clinical progression, the follow-up MRI scan showed isolated bilateral lesions involving the entire GP. MRI scans in Case 2 showed bilateral lesions of caudate and lentiform nuclei. There may be several mechanisms underlying delayed and progressive symptoms after time-limited brain anoxia. We hypothesize that anoxia-induced excitotoxicity resulting in mitochondrial dysfunction and subsequent apoptosis may explain, at least partly, the delayed-onset and progressive extrapyramidal syndromes seen in these patients.
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C03 03  X  FRE  @0 Arrêt cardiocirculatoire @5 07
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C03 03  X  SPA  @0 Paro cardiocirculatorio @5 07
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C03 07  X  ENG  @0 Pallidum @5 13
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C03 11  X  FRE  @0 Oxygène @2 NC @2 FX @5 27
C03 11  X  ENG  @0 Oxygen @2 NC @2 FX @5 27
C03 11  X  SPA  @0 Oxígeno @2 NC @2 FX @5 27
C07 01  X  FRE  @0 Appareil circulatoire pathologie @5 53
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C07 01  X  SPA  @0 Aparato circulatorio patología @5 53
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C07 02  X  SPA  @0 Músculo estriado patología @5 61
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C07 03  X  ENG  @0 Nervous system diseases @5 62
C07 03  X  SPA  @0 Sistema nervioso patología @5 62
C07 04  X  FRE  @0 Trouble neurologique @5 63
C07 04  X  ENG  @0 Neurological disorder @5 63
C07 04  X  SPA  @0 Trastorno neurológico @5 63
C07 05  X  FRE  @0 Mouvement involontaire @5 64
C07 05  X  ENG  @0 Involuntary movement @5 64
C07 05  X  SPA  @0 Movimiento involuntario @5 64
C07 06  X  FRE  @0 Extrapyramidal syndrome @5 65
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C07 06  X  SPA  @0 Extrapiramidal síndrome @5 65
N21       @1 020
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<s5>63</s5>
</fC07>
<fC07 i1="04" i2="X" l="ENG">
<s0>Neurological disorder</s0>
<s5>63</s5>
</fC07>
<fC07 i1="04" i2="X" l="SPA">
<s0>Trastorno neurológico</s0>
<s5>63</s5>
</fC07>
<fC07 i1="05" i2="X" l="FRE">
<s0>Mouvement involontaire</s0>
<s5>64</s5>
</fC07>
<fC07 i1="05" i2="X" l="ENG">
<s0>Involuntary movement</s0>
<s5>64</s5>
</fC07>
<fC07 i1="05" i2="X" l="SPA">
<s0>Movimiento involuntario</s0>
<s5>64</s5>
</fC07>
<fC07 i1="06" i2="X" l="FRE">
<s0>Extrapyramidal syndrome</s0>
<s5>65</s5>
</fC07>
<fC07 i1="06" i2="X" l="ENG">
<s0>Extrapyramidal syndrome</s0>
<s5>65</s5>
</fC07>
<fC07 i1="06" i2="X" l="SPA">
<s0>Extrapiramidal síndrome</s0>
<s5>65</s5>
</fC07>
<fN21>
<s1>020</s1>
</fN21>
<fN82>
<s1>PSI</s1>
</fN82>
</pA>
</standard>
</inist>
</record>

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