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Reversible Pisa syndrome in patients with Parkinson’s disease on dopaminergic therapy

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Abstract

Background

The wide variability of dystonic postures manifested in the clinical course of Parkinson’s disease (PD) represents a complicated on-going issue. Several recently published reports of Pisa syndrome (PS) in parkinsonian patients on dopaminergic therapy have described a variable means of onset and clinical course of this truncal dystonia.

Objective

To describe PD patients with PS, with the aim of stressing the frequent iatrogenic origin and potential reversibility of this syndrome during the initial stages of its appearance.

Subjects and methods

Eight consecutive PD patients who developed a PS after modifications of antiparkinson therapy were studied. All patients underwent detailed clinical assessment, [123I]FP-CIT-SPECT being performed in three cases. Four patients were videotaped.

Results

All patients developed PS within a variable time-span ranging from 15 days to 3 months after adjustment of treatment. Seven cases of PS were manifested following an increase and one a decrease of dopaminergic treatment. A marked reversal of dystonia was produced in the first seven patients by the withdrawal or dose decrease of dopaminergic PS priming drug, and in the eighth patient an increase of dopaminergic therapy was necessary.

Conclusions

In our opinion, the recognition of reversibility of PS during the initial stages of its appearance may be of considerable clinical importance. Indeed, it may facilitate the rapid withdrawal or reintroduction of dopaminergic treatment, thus avoiding an initial veering towards the subchronic variant and, subsequently into a chronic irreversible variant.

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References

  1. Ekbom K, Lindholm H, Ljungberg L (1972) New dystonic syndrome associated with butyrophenone therapy. Z Neurol 202:94–103

    Article  PubMed  CAS  Google Scholar 

  2. Suzuki T, Matsuzaka H (2002) Druginduced Pisa syndrome (Pleurothotonus) epidemiology and management. CNS Drugs 16(3):165–174

    Article  PubMed  CAS  Google Scholar 

  3. Patel S, Tariot PN, Hamill RW (1991) Pisa syndrome without neuroleptic exposure in a patient with dementia of the Alzheimer Type. J Geriatr Psychiatry Neurol 4:48–51

    Article  PubMed  CAS  Google Scholar 

  4. Colosimo C (1998) Pisa syndrome in a patient with multiple system atrophy. Mov Disord 13:607–609

    Article  PubMed  CAS  Google Scholar 

  5. Stubner S, Padberg F, Grohmann R, et al. (2000) Pisa syndrome (pleurothotonus): report of a multicenter drug safety surveillance project. J Clin Psychiatry 61(8):569–574

    PubMed  CAS  Google Scholar 

  6. Bhattacharya KF, Giannakikou I, Munroe N, et al. (2000) Primary anticholinergic- responsive Pisa syndrome. Mov Disord 15:1285–1286

    Article  PubMed  CAS  Google Scholar 

  7. Villarejo A, Camacho A, Garcia-Ramos R, Moreno T, Penas M, Juntas R, Ruiz J (2003) Cholinergic-dopaminergic imbalance in Pisa syndrome. Clinical Neuropharmacology 26:119–121

    Article  PubMed  Google Scholar 

  8. Cannas A, Solla P, Floris GL, et al. (2005) Reversible Pisa syndrome in Parkinson’s disease during dopaminergic treatment with Pergolide: a case report. Clin Neuropharm 28(5):252

    Article  Google Scholar 

  9. Garbarin M, Antonini A, Moretto G, et al. (2006) Pisa syndrome without neuroleptic exposure in a patient with Parkinson’s disease: case report. Mov Disord 21(2):270–273

    Article  Google Scholar 

  10. Yokochi F (2006) Lateral flexion in Parkinson’s disease and Pisa syndrome. J Neurol 253(Suppl 7):VII17–VII20

    Article  PubMed  Google Scholar 

  11. Kim JS, Park JW, Chung SW, Kim YI, Kim HT, Lee KS (2007) Pisa syndrome as a motor complication of Parkinson’s disease. Parkinsonism Relat Disord 13(2):126–128

    Article  PubMed  Google Scholar 

  12. Duvoisin RC, Marsden CD (1976) Note on the scoliosis of Parkinsonism. J Neurol Neurosurg Psychiatry 38(8):787–793

    Article  Google Scholar 

  13. Herrera-Marschitz M, Utsumi H, Ungerstedt U (1990) Scoliosis in rats with experimentally-induced hemiparkinsonism ¸dipendence upon striatal dopamine denervation. J. Neurol Neurosurg Psychiatry 53:39–43

    Article  PubMed  CAS  Google Scholar 

  14. Jankovic J, Tintner R (2001) Dystonia and parkinsonism. Parkinsonism Relat Disord 8:109–121

    Article  PubMed  CAS  Google Scholar 

  15. Djaldetti R, Hellmann M, Malamed E (2004) Bent knees and tiptoeing: late manifestations of end-stage Parkinson’s disease. Mov Disord 19:1325–1328

    Article  PubMed  Google Scholar 

  16. Azher SN, Jankovic J (2005) Camptocormia, pathogenesis, classification, and response to therapy. Neurology 65:355–359

    Article  PubMed  Google Scholar 

  17. Jancovic J (2005) Motor fluctuations and dyskinesias in Parkinson’s disease: clinical manifestations. Mov Disorders 20(Suppl 11):S11–S16

    Article  Google Scholar 

  18. Harada K (2006) Pisa syndrome without neuroleptic exposure in a patient with Parkinson’s disease: A case report. Mov Disord 21(12):2264

    Article  PubMed  Google Scholar 

  19. Bruneau MA, Stip E (1999) Metronome or alternating Pisa sindrome: a form of tardive distonia under clozapine treatment. Int Clin Psychopharmacol 13:229–232

    Article  Google Scholar 

  20. Schwarting RK, Huston JP (1996) The unilateral 6-hydroxydopamine lesion model in behavioral brain research. Analysis of functional deficits, recovery and treatments. Prog Neurobiol 50:275–331

    Article  PubMed  CAS  Google Scholar 

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Correspondence to A. Cannas.

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Cannas, A., Solla, P., Floris, G. et al. Reversible Pisa syndrome in patients with Parkinson’s disease on dopaminergic therapy. J Neurol 256, 390–395 (2009). https://doi.org/10.1007/s00415-009-0072-6

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  • DOI: https://doi.org/10.1007/s00415-009-0072-6

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