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      Right Sensory Alien Hand Phenomenon from a Left Pontine Hemorrhage

      case-report

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          Abstract

          Background

          Acute onset of a sensory alien hand phenomenon has been observed only from a supratentorial lesion involving the non-dominant hand, mostly from a right posterior cerebral artery infarction. A single acute vascular lesion resulting in a dominant hand sensory alien hand syndrome has not been previously documented.

          Case Report

          A 78-year old right-handed woman exhibited right sensory alien hand phenomenon from a left pontine hemorrhage. Disturbance of proprioceptive input and visuospatial perception are likely to play a role in manifesting the sign.

          Conclusions

          Dominant-hand sensory alien hand phenomenon may occur in an acute setting from a left pontine hemorrhage.

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          Most cited references7

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          Secondary cervical dystonia associated with structural lesions of the central nervous system.

          Mark LeDoux, Aisling Brady (2002)
          We tested the hypothesis that structural lesions of the central nervous system (CNS) associated with cervical dystonia more commonly involve the cerebellum and its primary afferent pathways than basal ganglia structures. Cervical dystonia is the most common focal dystonia, the majority of cases are idiopathic, and only a small percentage of patients have a family history of dystonia or other movement disorders. Pathophysiological mechanisms operative in solely or predominantly appendicular dystonias such as writer's cramp and Oppenheim's dystonia, respectively, may not be directly applicable to axial dystonias. The localization of structural lesions of the CNS associated with secondary cervical dystonia may provide some insight into the neural structures potentially involved in primary cervical dystonia. The National Library of Medicine Gateway (from 1960) and a clinical database maintained by the senior author (from 1999) were searched for cases of secondary cervical dystonia associated with structural lesions of the CNS. Search terms included one or more of the following: dystonia, torticollis, cervical, secondary, and symptomatic. Lesion localization and type, patient age, patient gender, head position, occurrence of sensory tricks, and associated neurological findings were tabulated for each case. Structural lesions associated with cervical dystonia were most commonly localized to the brainstem and cerebellum. The remaining cases were equally divided between the cervical spinal cord and basal ganglia. Although inconsistent, head rotation tended to be contralateral to lesion localization. Additional neurological abnormalities were present in the majority of patients with secondary cervical dystonia. The relative paucity of basal ganglia pathology and concentration of lesions in the brainstem, cerebellum, and cervical spinal cord in patients with secondary cervical dystonia suggests that dysfunction of cerebellar afferent pathways may be important to the pathophysiology of primary cervical dystonia. Copyright 2002 Movement Disorder Society
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            Abnormal cerebellar signaling induces dystonia in mice.

            Carolyn Pizoli, H A Jinnah, Melvin L. BILLINGSLEY … (2002)
            Dystonia is a relatively common neurological syndrome characterized by twisting movements or sustained abnormal postures. Although the basal ganglia have been implicated in the expression of dystonia, recent evidence suggests that abnormal cerebellar function is also involved. In these studies, a novel mouse model was developed to study the role of the cerebellum in dystonia. Microinjection of low doses of kainic acid into the cerebellar vermis of mice elicited reliable and reproducible dystonic postures of the trunk and limbs. The severity of the dystonia increased linearly with kainate dose. Kainate-induced dystonia was blocked by the glutamatergic antagonist 1,2,3,4-tetrahydro-6-nitro-2,3-dioxo-benzo[f]quinoxaline-7-sulfonamide and reproduced by domoic acid microinjection, suggesting that the induction of dystonia is dependent on glutamatergic activation in this model. The abnormal movements were not associated with kainate-induced seizures, because EEG recordings showed no epileptiform activity during the dystonic events. Neuronal activation, as assessed by in situ hybridization for c-fos, revealed c-fos mRNA expression in the cerebellum, locus ceruleus, and red nucleus. In contrast, regions associated with epileptic seizures, such as the hippocampus, did not exhibit increased c-fos expression after cerebellar kainate injection. Furthermore, in transgenic mice lacking Purkinje cells, significantly less dystonia was induced after kainic acid injection, implicating Purkinje cells and the cerebellar cortex in this model of dystonia. Together, these data suggest that abnormal cerebellar signaling produces dystonia and that the cerebellum should be considered along with the basal ganglia in the pathophysiology of this movement disorder.
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              The alien hand: cases, categorizations, and anatomical correlates.

              Lisa Scepkowski, Alice Cronin-Golomb (2003)
              The clinical characteristics and neuroanatomical damage reported in more than 50 published cases of observed "alien-hand" signs are reviewed. The terms alien-hand sign and alien-hand syndrome describe phenomena experienced by patients in which an upper limb performs complex motor activities outside of volitional control. The categories of frontal and callosal subtypes and their relation to behavior and neuropathology are evaluated with reference to the dual premotor system theory, which emphasizes the role of the supplementary motor areas in alien-hand phenomena. Detailed consideration is given to the more recently described posterior subtype, which is purported to result from damage to the parietal lobe or other posterior brain areas. The lack of uniformity in reported assessment methods (behavioral tests, neuroimaging) in published cases contributes to the difficulty in establishing clear subtypes of alien-hand phenomena. Suggestions are made regarding current categorizations and available assessment methods.
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                Author and article information

                Journal
                Journal ID (nlm-ta): J Clin Neurol
                Journal ID (publisher-id): JCN
                Title: Journal of Clinical Neurology (Seoul, Korea)
                Publisher: Korean Neurological Association
                ISSN (Print): 1738-6586
                ISSN (Electronic): 2005-5013
                Publication date (Print): March 2009
                Publication date (Electronic): 31 March 2009
                Volume: 5
                Issue: 1
                Pages: 46-48
                Affiliations
                Department of Neurology, University of California at Irvine Medical Center, California, USA.
                Author notes
                Correspondence: Gregory Youngnam Chang, MD, FAAN Department of Neurology, University of California at Irvine Medical Center, 101 The City Drive, Orange, California, 92868, USA. Tel +1-714-456-5693, Fax +1-714-456-7182, gychang@ 123456uci.edu
                Article
                DOI: 10.3988/jcn.2009.5.1.46
                PMC ID: 2686896
                PubMed ID: 19513334
                SO-VID: 027baa14-60da-40fb-95ac-d06bde3443fc
                Copyright © Copyright © 2009 Korean Neurological Association
                History
                Date received : 16 June 2008
                Date revision received : 23 October 2008
                Date accepted : 23 October 2008
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Neurology
                Keywords: pontine hemorrhage,sensory alien hand,dominant limb
                Data availability:
                ScienceOpen disciplines: Neurology
                Keywords: pontine hemorrhage, sensory alien hand, dominant limb

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