Dystonia in progressive supranuclear palsy.

Barclay, Catherine; Lang, Anthony E.

doi:10.1136/jnnp.62.4.352

Cited by 117 publications

(74 citation statements)

References 27 publications

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“…However, the differential diagnosis of parkinsonism continues to be challenging with a high misdiagnosis rate, particularly in the early stage [6], because parkinsonian patients show similar symptoms and specific symptoms do not appear in early stage [7]. It is of importance to make an accurate differential diagnosis of parkinsonism to decide on treatment regimens [8], provide a prognosis [9], investigate etiology and pathogenesis, and to develop new therapeutic strategies [10].…”

Section: Introductionmentioning

confidence: 99%

Differential Diagnosis of Parkinsonism Using Dual-Phase F-18 FP-CIT PET Imaging

et al. 2012

Nucl Med Mol Imaging

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Purpose Dopamine transporter (DAT) imaging can demonstrate presynaptic dopaminergic neuronal loss in Parkinson's disease (PD). However, differentiating atypical parkinsonism (APD) from PD is often difficult. We investigated the usefulness of dual-phase F-18 FP-CIT positron emission tomography (PET) imaging in the differential diagnosis of parkinsonism. Methods Ninety-eight subjects [five normal, seven druginduced parkinsonism (DIP), five essential tremor (ET), 24 PD, 20 multiple system atrophy-parkinson type (MSA-P), 13 multiple system atrophy-cerebellar type (MSA-C), 13 progressive supranuclear palsy (PSP), and 11 dementia with Lewy bodies (DLB)] underwent F-18 FP-CIT PET. PET images were acquired at 5 min (early phase) and 3 h (late phase) after F-18 FP-CIT administration (185 MBq). Regional uptake pattern of cerebral and cerebellar hemispheres was assessed on early phase images and striatal DAT binding pattern was assessed on late phase images, using visual, quantitative, and statistical parametric mapping (SPM) analyses. Results Striatal DAT binding was normal in normal, ET, DIP, and MSA-C groups, but abnormal in PD, MSA-P, PSP, and DLB groups. No difference was found in regional uptake on early phase images among normal DAT binding groups, except in the MSA-C group. Abnormal DAT binding groups showed different regional uptake pattern on early phase images compared with PD in SPM analysis (FDR< 0.05). When discriminating APD from PD, visual interpretation of the early phase image showed high diagnostic sensitivity and specificity (75.4 % and 100 %, respectively). Regarding the ability to distinguish specific APD, sensitivities were 81 % for MSA-P, 77 % for MSA-C, 23 % for PSP, and 54.5 % for DLB. Conclusions Dual-phase F-18 FP-CIT PET imaging is useful in demonstrating striatal DAT loss in neurodegenerative parkinsonism, and also in differentiating APD, particularly MSA, from PD.

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Section: Introductionmentioning

confidence: 99%

Differential Diagnosis of Parkinsonism Using Dual-Phase F-18 FP-CIT PET Imaging

et al. 2012

Nucl Med Mol Imaging

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“…It is possible that limb dystonia in PSP is an indicator of concomitant cortical and basal ganglia neuropathological changes. It is becoming increasingly recognized that such cortical pathology does occur in PSP 17 . Interestingly, oromandibular dystonia was observed in one of our patients and it was not related to levodopa therapy.…”

Section: Discussionmentioning

confidence: 99%

“…The neck positioning in PSP tends to be fixed and does not alter with posture or activity as idiopathic dystonia does. Patients fail to show sensory tricks for improving the posturing, diurnal variation is absent ("morning benefit"), and painful neck spasms and hypertrophic muscles are not seen 17 . Two of our patients (patients 3 and 5) presented retrocollis and MrI abnormalities in brainstem.…”

Section: Discussionmentioning

confidence: 99%

“…Interestingly, oromandibular dystonia was observed in one of our patients and it was not related to levodopa therapy. The potential role of medication, especially levodopa, must be considered when dealing with dystonia in patients with PSP 17 . A Brazilian series of PSP patients observed that 13% had transiently good response to levodopa, but it did not point out whether these patients also presented dystonia 21 .…”

Section: Discussionmentioning

confidence: 99%

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Clinical features of dystonia in atypical parkinsonism

Godeiro-Júnior

Felı́cio

Barsottini

et al. 2008

Arq. Neuro-Psiquiatr.

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-Background:The association between Dystonia and Parkinson's disease (PD) has been well described especially for foot and hand dystonia. There is however few data on dystonic postures in patients with atypical parkinsonism. Objective: To evaluate the frequency and pattern of dystonia in a group of patients with atypical parkinsonism (multiple system atrophy -MSA, progressive supranuclear palsy -PSP, and corticobasal degeneration -CBD) and to investigate whether dystonia could be the first presenting symptom at disease onset in those patients. Method: A total of 38 medical charts were reviewed (n=23/MSA group; n=7/CBD group; n=8/PSP group) and data values were described as means/standard deviations. The variables evaluated were sex, age at onset, disease duration, first symptom, clinical features of dystonia and other neurological signs, response to levodopatherapy, Hoehn&Yahr scale >3 after three years of disease, and magnetic resonance imaging findings. Results: The overall frequency of dystonia in our sample was 50% with 30.4% (n=7) in the MSA group, 62.5% (n=5) in the PSP group, and 100% (n=8) in the CBD group. In none of these patients, dystonia was the first complaint. Several types of dystonia were found: camptocormia, retrocollis, anterocollis, blepharoespasm, oromandibular, and foot/hand dystonia. Conclusion: In our series, dystonia was a common feature in atypical parkinsonism (overall frequency of 50%) and it was part of the natural history although not the first symptom at disease onset. Neuroimaging abnormalities are not necessarily related to focal dystonia, and levodopa therapy did not influence the pattern of dystonia in our group of patients.KeY worDS: dystonia, atypical parkinsonism, multiple system atrophy, progressive supranuclear palsy, corticobasal degeneration, Parkinson's disease. Características clínicas da distonia no parkinsonismo atípicoResumo -Introdução: A associação de distonia e doença de Parkinson (DP) já foi bem estabelecida, principalmente para distonia focal em pé ou mão. entretanto, há poucos dados quanto a distonia em pacientes com parkinsonismo atípico. Objetivo: Avaliar a freqüência e o padrão da distonia em um grupo de pacientes com parkisnonismo atípico (atrofia de múltiplos sistemas -AMS; paralisia supranuclear progressiva -PSP; degeneração corticobasal -DCB) e investigar se a distonia pode ser a manifestação inicial neste grupo. Método: Um total de 38 prontuários médicos foi revisado (n=23/grupo AMS; n=8/grupo PSP; n=7/grupo PSP) e os dados foram apresentados em médias/desvios padrões. As variaveis avaliadas foram: sexo, idade de início, duração da doença, primeiro sintoma, características clínicas da distonia e outros sinais neurológicos, resposta ao tratamento com levodopa, escala de Hoehn & Yahr >3 em 3 anos de doença, e achados de ressonância magnética. Resultados: A frequência total de distonia em nosso grupo foi 50%, sendo 30,4% (n=7) no grupo AMS, 62.5% (n=5) no grupo PSP e 100% (n=8) no grupo DCB. em nenhum dos pacientes, distonia foi o primeiro sintoma...

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“…For these reasons the term dystonia should be avoided in these cases. Instead, "nuchal rigidity in extension" should be adopted (21).…”

Section: Progressive Supranuclear Palsymentioning

confidence: 99%