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Primary and reversible Pisa syndrome in juvenile normal pressure hydrocephalus

Published online by Cambridge University Press:  22 February 2013

Fidias E. Leon-Sarmiento ,
Gustavo Pradilla  and
Maria del Rosario Zambrano
Fidias E. Leon-Sarmiento*
Affiliation:
Smell and Taste Center, University of Pennsylvania, Philadelphia, PA, USA Department of Otorhinolaryngology – Head and Neck Surgery, University of Pennsylvania, Philadelphia, PA, USA Unit of Parkinson and Movement Disorders, Mediciencias Research Group, Unit of Aerospace Medicine, Universidad Nacional, Bogota, Colombia Human Aerospace Laboratory, Mount Sinai School of Medicine, New York, NY, USA
Gustavo Pradilla
Affiliation:
Department of Internal Medicine, UIS, Unit of Neurology, Bucaramanga, Colombia
Maria del Rosario Zambrano
Affiliation:
Neurorehabilitation Unit-NeuroSalud, “Ramon & Cajal” Panamerican Health Foundation, Bogota, Colombia
*
Dr Fidias E. Leon-Sarmiento, Smell and Taste Center, University of Pennsylvania, 5 Ravdin Pavilion 3400, Spruce Street, Philadelphia, PA 19104, USA. Tel: +13152458183; Fax: +13152458183; E-mail: [email protected]
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Abstract

Objective

To report a case of Pisa syndrome in a patient with idiopathic normal pressure hydrocephalus, who had never been exposed to psychotropic medications.

Methods

A 26-year-old, Colombian, male patient, was referred because he had cognitive abnormalities, gait disturbances and urinary incontinence. This patient also displayed pleurothotonos. Neurofunctional evaluations of sensory and motor integration at peripheral and central nervous system levels were done. Idiopathic normal pressure hydrocephalus (NPH) was diagnosed.

Results

Pisa syndrome disappeared after spinal tap drainage with further gait, balance and behavioural improvement. A brainstem-thalamocortical deregulation of the central sensory and motor programming, due to the chaotic enlargement of brain ventricles was thought to be the pathophysiological mechanism underlying this case.

Conclusion

NPH must not be longer considered as an exclusive geriatric disorder. Further, uncommon movement disorders may appear with this disorder, which should be carefully approached to avoid iatrogenic and deleterious pharmacological interventions.

Keywords

central nervous systemcerebral cortexdementiapsychiatric disorders
Type
Case Report
Information
Acta Neuropsychiatrica , Volume 25 , Issue 1 , February 2013 , pp. 57 - 60
Copyright
Scandinavian College of Neuropsychopharmacology 2013

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References

1Suzuki, T, Matsuzaka, H.Drug-induced Pisa syndrome (pleurothotonus): epidemiology and management. CNS Drugs 2002;16:165–174.CrossRefGoogle ScholarPubMed
2Villarejo, A, Camacho, A, García-Ramos, Ret al.Cholinergic-dopaminergic imbalance in Pisa syndrome. Clin Neuropharmacol 2003;26:119–121.CrossRefGoogle ScholarPubMed
3Hakim, S, Adams, RD.The special clinical problem of symptomatic hydrocephalus with normal cerebrospinal fluid pressure. Observations on cerebrospinal fluid hydrodynamics. J Neurol Sci 1965;2:307–332.CrossRefGoogle ScholarPubMed
4Pereira Damasceno, B.Normal pressure hydrocephalus. Diagnostic and predictive evaluation. Dememt Neuropsychol 2009;3:8–11.CrossRefGoogle Scholar
5Leon-S, FE, Pradilla, G, Villamil, L, Vesga, B, Dallos, M.Análisis crítico del minimental en el mundo: 1975–1995. Acta Neurol Col 1999;15:135–145.Google Scholar
6Leon-S, FE, Gomez, A, Kimura, J.Neurofisiologia clinica. In: Toro, J, Yepes, M, Palacios, E, eds. Neurologia, 7th edn. Mexico, DF: Manual Moderno, 2010: 683–701.Google Scholar
7Leon-S, FE, Arimura, K, Sonoda, Y, Arimura, Y, Osame, M.Instability of R3 of the blink reflex in patients with HAM/TSP. Funct Neurol 1994;8:199–202.Google Scholar
8Leon-S, FE, Pradilla, G, Calderon, A, Duque, JR.Dilación, dilatación e inclinación en hidrocefalia de presión normal. Rev Colomb Psiquiat 2008;37:280–286.Google Scholar
9Relkin, N, Marmarou, A, Klinge, P.Diagnosing idiopathic normal-pressure hydrocephalus. Neurosurgery 2005; 57(Suppl. 3):4–16.CrossRefGoogle ScholarPubMed
10Leon-S, FE, Pradilla, G, Calderon, A.Pleurototonus. Acta Med Colomb 2008;33:153.CrossRefGoogle Scholar
11Bret, P, Chazal, J.Chronic (“normal pressure”) hydrocephalus in childhood and adolescence. A review of 16 cases and reappraisal of the syndrome. Childs Nerv Syst 1995;11:687–691.CrossRefGoogle ScholarPubMed
12Colosimo, C.Pisa syndrome in a patient with multiple system atrophy. Mov Disord 1998;13:607–609.CrossRefGoogle Scholar
13Patel, S, Tariot, PN, Hamill, RW.Pisa syndrome without neuroleptic exposure in a patient with dementia of the Alzheimer type. J Geriatr Psychiatry Neurol 1991;4:48–51.CrossRefGoogle Scholar
14Bhattacharva, KF, Giannakikou, I, Munroe, N, Chaudhuri, KR.Primary anticholinergic-responsive Pisa syndrome. Mov Disord 2000;15:1285–1287.3.0.CO;2-U>CrossRefGoogle Scholar
15Lee, WK, Shiah, IS, Chen, CY, Lin, TL, Shen, LJ.Electroconvulsive therapy-associated Pisa syndrome in clozapine treatment. Psychiatry Clin Neurosci 2008;62:753.CrossRefGoogle ScholarPubMed
16Kan, AE.Striatonigral degeneration. Pathology 1978;10: 45–52.CrossRefGoogle ScholarPubMed
17Ouchi, Y, Nakayama, T, Kanno, T, Yoshikawa, E, Shinke, T, Torizuka, T.In vivo presynaptic and postsynaptic striatal dopamine functions in idiopathic normal pressure hydrocephalus. J Cereb Blood Flow Metab 2007;27: 803–810.CrossRefGoogle ScholarPubMed
18Kondziella, D, Sonnewald, U, Tullberg, M, Wikkelso, C.Brain metabolism in adult chronic hydrocephalus. J Neurochem 2008;106:1515–1524.CrossRefGoogle ScholarPubMed
19Krauss, JK, Regel, JP, Droste, DW, Orszagh, M, Borremans, JJ, Vach, W.Movement disorders in adult hydrocephalus. Mov Disord 1997;12:53–60.CrossRefGoogle ScholarPubMed
20Peiser, C, Fisher, A.Expression of dopamine receptors in sensory neurons of the rat. Am J Respir Crit Care Med 2001;163:A905.Google Scholar
21Morishita, T, Foote, KD, Okun, MS.INPH and Parkinson disease: differentiation by levodopa response. Nat Rev Neurol 2010;6:52–56.CrossRefGoogle ScholarPubMed
22Doménech, J, Tormos, JM, Barrios, C, Pascual-Leone, A.Motor cortical hyperexcitability in idiopathic scoliosis: could focal dystonia be a subclinical etiological factor? Eur Spine J 2010;19:223–230.CrossRefGoogle ScholarPubMed
23Bohlhalter, S, Leon-S, FE, Hallett, M.Abnormality of motor cortex excitability in peripherally induced dystonia. Mov Disord 2007;22:1186–1189.CrossRefGoogle ScholarPubMed
24Bhatia, KP, Quinn, NP, Marsden, CD.Clinical features and natural history of axial predominant adult onset primary dystonia. J Neurol Neurosurg Psychiatry 1997;63:788–791.CrossRefGoogle ScholarPubMed
25Hawkes, CH, Doty, RL.The neurology of olfaction. Cambridge: Cambridge University Press, 2009.CrossRefGoogle Scholar
26Kapoor, KG.Do patients with idiopathic intracranial hypertension suffer from hyposmia? Med Hypotheses 2008;71:816–817.CrossRefGoogle ScholarPubMed
27Crowson, AN, Magro, C.Insterstital granulomalotous dermatitis with arthritis. Hum Pathol 2002;35:779–780.CrossRefGoogle Scholar