Advertisement

Journal of Neurology

, Volume 265, Issue 10, pp 2442–2444 | Cite as

Management of Pisa syndrome with lateralized subthalamic stimulation

  • Karlo J. LizarragaEmail author
  • Maryam Naghibzadeh
  • Alexandre Boutet
  • Gavin J. B. Elias
  • Alfonso Fasano
Letter to the Editors

Dear Sirs,

Pisa syndrome (PS) describes a postural abnormality characterized by lateral trunk flexion greater than 10° in the upright position that reverts in the supine position or with passive mobilization [1]. PS was initially described as truncal dystonia in patients using typical neuroleptics [2], but it can be triggered by other drugs such as cholinergic agents or dopamine agonists. PS can also occur in neurodegenerative disorders such as Parkinson’s disease (PD) [3]. Even though basal ganglia abnormalities associated with asymmetric paraspinal dystonia appear to be necessary for PS, other factors such as vestibular imbalance, proprioceptive disintegration, and myopathy could also play a role [4, 5].

Pisa syndrome can be reversible in early phases when a triggering factor is identified and discontinued. Otherwise, PS can become chronic, severe, and treatment resistant. In these cases, botulinum neurotoxin (BoNT) injections and deep brain stimulation (DBS) of subthalamic (STN) or...

Notes

Acknowledgements

The authors thank the patient and his family. KJL thanks the Dystonia Medical Research Foundation Canada for their 2018 Clinical Fellowship Grant.

Compliance with ethical standards

Conflicts of interest

On behalf of all authors, the corresponding author states that there are no relevant conflicts of interest.

Ethical standard

The authors declare that the work documented in this manuscript has been carried out in accordance with ethical standards.

Supplementary material

Video 1. Pisa syndrome improvement with lateralized subthalamic neuromodulation. Bilateral subthalamic stimulation was maintained at a pulse width of 60 microseconds and a frequency of 80 hertz. Amplitude of right subthalamic stimulation was maintained at 4.8 volts (V) (monopolar contact three). Right lateral truncal deviation worsened when left subthalamic voltage was increased to 4.8 V (Segment 1) and progressively improved when it was reduced to 4.0 V (Segment 2) and 3.8 V (Segment 3) (monopolar contact six). (MP4 18508 KB)

References

  1. 1.
    Tinazzi M, Geroin C, Gandolfi M, Smania N, Tamburin S, Morgante F, Fasano A (2016) Pisa syndrome in Parkinson’s disease: an integrated approach from pathophysiology to management. Mov Disord 31(12):1785–1795.  https://doi.org/10.1002/mds.26829 CrossRefPubMedGoogle Scholar
  2. 2.
    Ekbom K, Lindholm H, Ljungberg L (1972) New dystonic syndrome associated with butyrophenone therapy. Z Neurol 202(2):94–103PubMedGoogle Scholar
  3. 3.
    Tinazzi M, Fasano A, Geroin C et al (2015) Pisa syndrome in Parkinson disease: an observational multicenter Italian study. Neurology 85(20):1769–1779.  https://doi.org/10.1212/WNL.0000000000002122 CrossRefPubMedGoogle Scholar
  4. 4.
    Castrioto A, Piscicelli C, Perennou D, Krack P, Debu B (2014) The pathogenesis of Pisa syndrome in Parkinson’s disease. Mov Disord 29(9):1100–1107.  https://doi.org/10.1002/mds.25925 CrossRefPubMedGoogle Scholar
  5. 5.
    Tinazzi M, Juergenson I, Squintani G, Vattemi G, Montemezzi S, Censi D et al (2013) Pisa syndrome in Parkinson’s disease: an electrophysiological and imaging study. J Neurol 260(8):2138–2148.  https://doi.org/10.1007/s00415-013-6945-8 CrossRefPubMedGoogle Scholar
  6. 6.
    Umemura A, Oka Y, Ohkita K, Yamawaki T, Yamada K (2010) Effect of subthalamic deep brain stimulation on postural abnormality in Parkinson disease. J Neurosurg 112(6):1283–1288.  https://doi.org/10.3171/2009.10.JNS09917 CrossRefPubMedGoogle Scholar
  7. 7.
    Ricciardi L, Piano C, Bentivoglio AR, Fasano A (2014) Long-term effects of pedunculopontine nucleus stimulation for Pisa syndrome. Parkinsonism Relat Disord 20(12):1445–1446.  https://doi.org/10.1016/j.parkreldis.2014.10.006 CrossRefPubMedGoogle Scholar
  8. 8.
    Horn A, Reich M, Vorwerk J, Li N, Wenzel G, Fang Q et al (2017) Connectivity predicts deep brain stimulation outcome in Parkinson disease. Ann Neurol 82(1):67–78.  https://doi.org/10.1002/ana.24974 CrossRefPubMedPubMedCentralGoogle Scholar
  9. 9.
    Ewert S, Plettig P, Li N, Chakravarty MM, Collins DL, Herrington TM et al (2018) Toward defining deep brain stimulation targets in MNI space: a subcortical atlas based on multimodal MRI, histology and structural connectivity. Neuroimage 170:271–282CrossRefGoogle Scholar
  10. 10.
    van de Warrenburg BP, Bhatia KP, Quinn NP (2007) Pisa syndrome after unilateral pallidotomy in Parkinson’s disease: an unrecognised, delayed adverse event? J Neurol Neurosurg Psychiatry 78(3):329–330.  https://doi.org/10.1136/jnnp.2006.103358 CrossRefPubMedPubMedCentralGoogle Scholar
  11. 11.
    Spanaki C, Zafeiris S, Plaitakis A (2010) Levodopa-aggravated lateral flexion of the neck and trunk as a delayed phenomenon of unilateral pallidotomy. Mov Disord 25(5):655–656.  https://doi.org/10.1002/mds.22988 CrossRefPubMedGoogle Scholar
  12. 12.
    Fasano A, Aquino CC, Krauss JK, Honey CR, Bloem BR (2015) Axial disability and deep brain stimulation in patients with Parkinson disease. Nat Rev Neurol 11(2):98–110.  https://doi.org/10.1038/nrneurol.2014.252 CrossRefPubMedGoogle Scholar
  13. 13.
    Lizarraga KJ, Jagid JR, Luca CC (2016) Comparative effects of unilateral and bilateral subthalamic nucleus deep brain stimulation on gait kinematics in Parkinson’s disease: a randomized, blinded study. J Neurol 263(8):1652–1656.  https://doi.org/10.1007/s00415-016-8191-3 CrossRefPubMedGoogle Scholar
  14. 14.
    Lizarraga KJ, Luca CC, De Salles A, Gorgulho A, Lang AE, Fasano A (2017) Asymmetric neuromodulation of motor circuits in Parkinson’s disease: the role of subthalamic deep brain stimulation. Surg Neurol Int 8:261.  https://doi.org/10.4103/sni.sni_292_17 CrossRefPubMedPubMedCentralGoogle Scholar

Copyright information

© Springer-Verlag GmbH Germany, part of Springer Nature 2018

Authors and Affiliations

  • Karlo J. Lizarraga
    • 1
    • 2
    Email author
  • Maryam Naghibzadeh
    • 1
    • 2
  • Alexandre Boutet
    • 2
  • Gavin J. B. Elias
    • 2
  • Alfonso Fasano
    • 1
    • 2
    • 3
  1. 1.Edmond J. Safra Program in Parkinson’s Disease, Morton and Gloria Shulman Movement Disorders Clinic, Toronto Western Hospital and Division of Neurology, Department of MedicineUniversity of TorontoTorontoCanada
  2. 2.University Health NetworkTorontoCanada
  3. 3.Krembil Brain InstituteTorontoCanada

Personalised recommendations