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Pisa syndrome in a patient with progressive supranuclear palsy

  • Paolo Solla
    Correspondence
    Corresponding author. Tel.: +39 070 51096336; fax: +39 070 51096336.
    Affiliations
    Movement Disorders Center, Department of Cardiovascular and Neurological Sciences, Institute of Neurology, University of Cagliari, SS 554 Bivio Sestu, Monserrato (Cagliari) 09042, Italy
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  • Antonino Cannas
    Affiliations
    Movement Disorders Center, Department of Cardiovascular and Neurological Sciences, Institute of Neurology, University of Cagliari, SS 554 Bivio Sestu, Monserrato (Cagliari) 09042, Italy
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  • Emanuela Costantino
    Affiliations
    Movement Disorders Center, Department of Cardiovascular and Neurological Sciences, Institute of Neurology, University of Cagliari, SS 554 Bivio Sestu, Monserrato (Cagliari) 09042, Italy
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  • Gianni Orofino
    Affiliations
    Movement Disorders Center, Department of Cardiovascular and Neurological Sciences, Institute of Neurology, University of Cagliari, SS 554 Bivio Sestu, Monserrato (Cagliari) 09042, Italy
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  • Loredana Lavra
    Affiliations
    Movement Disorders Center, Department of Cardiovascular and Neurological Sciences, Institute of Neurology, University of Cagliari, SS 554 Bivio Sestu, Monserrato (Cagliari) 09042, Italy
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  • Francesco Marrosu
    Affiliations
    Movement Disorders Center, Department of Cardiovascular and Neurological Sciences, Institute of Neurology, University of Cagliari, SS 554 Bivio Sestu, Monserrato (Cagliari) 09042, Italy
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      Pisa syndrome (PS) is an abnormal posture characterized by a tonic flexion of the trunk to one side accompanied by a slight rotation in the sagittal plane, originally described as the consequence of acute axial dystonia related to neuroleptic administration.
      • Ekbom K.
      • Lindholm H.
      • Ljungberg L.
      New dystonic syndrome associated with butyrophenone therapy.
      Subsequently PS has been reported in patients treated with antiemetics, antidepressants, cholinesterase inhibitors and dopamine agonists.
      • Suzuki T.
      • Matsuzaka H.
      Drug-induced Pisa syndrome (pleurothotonus): epidemiology and management.
      • Cannas A.
      • Solla P.
      • Floris G.
      • et al.
      Reversible Pisa syndrome in patients with Parkinson’s disease on dopaminergic therapy.
      A similar picture may also occur as an idiopathic phenomenon and in neurodegenerative diseases, including parkinsonism.
      • Suzuki T.
      • Matsuzaka H.
      Drug-induced Pisa syndrome (pleurothotonus): epidemiology and management.
      • Cannas A.
      • Solla P.
      • Floris G.
      • et al.
      Reversible Pisa syndrome in patients with Parkinson’s disease on dopaminergic therapy.
      With regard to parkinsonism, although PS has been previously indicated as a red flag for multiple system atrophy (MSA),
      • Köllensperger M.
      • Geser F.
      • Seppi K.
      • et al.
      European MSA study group. Red flags for multiple system atrophy.
      this abnormal posture is not infrequently observed in patients with typical Parkinson’s disease (PD)
      • Cannas A.
      • Solla P.
      • Floris G.
      • et al.
      Reversible Pisa syndrome in patients with Parkinson’s disease on dopaminergic therapy.
      and other atypical parkinsonism disorders such as dementia with Lewy bodies (DLB).
      • Shinfuku M.
      • Nakajima S.
      • Uchida H.
      • et al.
      Pisa syndrome caused by an acetylcholinesterase inhibitor in a patient with dementia with Lewy bodies.
      However, PS has not yet been described in patients with progressive supranuclear palsy (PSP). We describe a patient affected by PSP who developed a dystonic lateral flexion of the trunk compatible with the diagnosis of PS.
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