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Evidence of delayed nigrostriatal dysfunction in corticobasal syndrome: a SPECT follow-up study.

Abstract：

OBJECTIVE:To demonstrate that degeneration of substantia nigra neurons may occur at later stages of disease in some patients with corticobasal syndrome (CBS) who evidenced preserved nigrostriatal pathway at a baseline FP-CIT SPECT study. BACKGROUND:Current pathological criteria for the definite diagnosis of corticobasal degeneration consider substantia nigra cell loss as a mandatory finding. However, dopamine transporter SPECT imaging performed in a large cohort of CBS patients showed about 10% of normal scans. METHODS:We describe 4 patients with clinical diagnosis of CBS and normal FP-CIT SPECT at baseline whose tracer uptake resulted pathological at 1-year follow-up scan. Clinical assessment has been performed at the time of SPECT scan. A semi-quantitative approach was performed for striatal FP-CIT binding values. RESULTS:Baseline SPECT scans have been performed after 2.3 ± 1.5 years from onset. All CBS patients presented asymmetric rigid-akinetic parkinsonism (mean Hoehn-Yahr stage 2.5; UPDRS motor score 18) with poor levodopa response and ideo-motor limb apraxia. At follow-up, neurological examination revealed some additional features, including limb dystonia, language impairment, postural instability, ocular gaze impairment, alien limb. All patients showed pathological FP-CIT uptake at the SPECT performed 10-15 months apart from the baseline scan. CONCLUSIONS:Our longitudinal FP-CIT SPECT findings support in vivo the hypothesis that substantia nigra neuronal loss may occur at later stages in some patients with CBS, despite early extrapyramidal symptoms.

journal_name

Parkinsonism Relat Disord

journal_title

Parkinsonism & related disorders

authors

Ceravolo R,Rossi C,Cilia R,Tognoni G,Antonini A,Volterrani D,Bonuccelli U

doi

10.1016/j.parkreldis.2013.01.013

subject

Has Abstract

pub_date

2013-05-01 00:00:00

pages

557-9

issue

5

eissn

1353-8020

issn

1873-5126

pii

S1353-8020(13)00048-5

journal_volume

19

pub_type

杂志文章

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