Relative risk of spread of symptoms among the focal onset primary dystonias

Weiss, Elliott Mark; Hershey, Tamara; Karimi, Morvarid; Racette, Brad A.; Tabbal, Samer D.; Mink, Jonathan W.; Paniello, Randal C.; Perlmutter, Joel S.

doi:10.1002/mds.20919

Cited by 99 publications

(133 citation statements)

References 25 publications

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“…The pattern of CD spread in our group of patients was noteworthy and somewhat different from previous reports that showed a predilection for spread to the cranial segment or to the upper limb 6,7,12 . In fact, most of patients spreaded to cranial segment, but we found a high prevalence of generalized spreading (28% of patients).…”

Section: Discussioncontrasting

confidence: 99%

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Retrocollis, anterocollis or head tremor may predict the spreading of dystonic movements in primary cervical dystonia

Godeiro-Júnior

Felı́cio

Aguiar

et al. 2009

Arq. Neuro-Psiquiatr.

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-Background and Purpose: Few studies have attempted to develop clinical predictors for cervical dystonia (CD) aiming at progression of the dystonic movement. Method: We retrospectively evaluated 73 patients with primary CD who underwent treatment with Botulinum toxin type-A (BTX-A). The patients were assembled in two groups according to the spread of dystonia during follow-up: spreading and non-spreading CD. We performed a binary logistic regression model using spreading of cervical dystonia as dependent variable aiming to find covariates which increase the risk of spreading. Results: Our logistic regression model found the following covariates and their respective risk ratios: time of disease >18.5 months=2.4, retrocollis=1.9, anterocollis=1.8, head tremor=1.6. Conclusion: Time of disease >18.5 months, retrocollis, anterocollis and head tremor may predict spreading of dystonic movement to other regions of the body in CD patients.Key WOrDs: cervical dystonia, progression, retrocollis, anterocollis, head tremor. Retrocolis, anterocolis ou tremor cefálico podem predizer a progressão dos movimentos distônicos em pacientes com distonia cervical primáriaResumo -Introdução: Poucos estudos avaliam preditores clínicos de progressão dos movimentos distônicos, para além da região cervical, em pacientes com distonia cervical (DC) primária. Método: Avaliamos, retrospectivamente, 73 pacientes com DC primária, que tinham sido submetidos ao tratamento com a toxina botulínica tipo A (BTX-A). estes pacientes foram divididos em dois grupos de acordo com a progressão ou não da DC para outras áreas do corpo. Aplicamos um modelo de regressão logística binária usando a progressão da distonia como variável dependente com o objetivo de identificar co-variáveis que aumentassem o risco de progressão. Resultados: O modelo de regressão logístico identificou as seguintes co-variáveis como preditoras de progressão e seus respectivos índices de risco: tempo de doença >18,5 meses=2,4, retrocolis=1,9, anterocolis=1,8, tremor cefálico=1,6. Conclusão: Tempo de doença >18,5 meses, retrocolis, anterocolis, e tremor cefálico podem predizer a progressão do movimento distônico para outras regiões do corpo em pacientes com DC primária.PAlAvrAs-ChAve: distonia cervical, progressão, retrocolis, anterocolis, tremor cefálico.

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Section: Discussioncontrasting

confidence: 99%

“…Therefore, an important concern is the spread of CD to other body parts. Previous studies that evaluated clinical and demographic characteristics of patients with CD were not able to demonstrate predictable variables for the risk of CD progression [4][5][6][7][8] .…”

mentioning

confidence: 90%

Retrocollis, anterocollis or head tremor may predict the spreading of dystonic movements in primary cervical dystonia

Godeiro-Júnior

Felı́cio

Aguiar

et al. 2009

Arq. Neuro-Psiquiatr.

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“…Dystonia spreads in a significant portion of patients [16][17][18] suggesting that slowing or halting clinical progression may be of interest; however, developing such a disease-modifying therapy will probably require a better understanding of the disease mechanism. A trial aimed at slowing disease progression would require long-term evaluations and outcomes, or a validated, short-term surrogate outcome, which is currently lacking for dystonia.…”

Section: Clinical Characteristics Of Dystonia and Associated Challengesmentioning

confidence: 99%

Designing Clinical Trials for Dystonia

et al. 2014

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With advances in the understanding of the pathophysiology of dystonia, novel therapeutics are being developed. Such therapies will require clinical investigation ranging from exploratory studies to examine safety, tolerability, dosage selection, and preliminary efficacy to confirmatory studies to evaluate efficacy definitively. As dystonia is a rare and complex disorder with clinical and etiological heterogeneity, clinical trials will require careful consideration of the trial design, including enrollment criteria, concomitant medication use, and outcome measures. Given the complexities of designing and implementing efficient clinical trials, it is important for clinicians and statisticians to collaborate closely throughout the clinical development process and that each has a basic understanding of both the clinical and statistical issues that must be addressed. To facilitate designing appropriate clinical trials in this field, we review important general clinical trial and regulatory principles, and discuss the critical components of trials with an emphasis on considerations specific to dystonia. Additionally, we discuss designs used in early exploratory, late exploratory, and confirmatory phases, including adaptive designs.

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“…Patients with BPS are more likely to experience dystonias in other parts of the body than patients with other focal dystonias 7 . Complaints of dry eye and photophobia are usually reported, but the exact nature of this association remains unestablished 5 .…”

mentioning

confidence: 97%

Clinical features and treatment with botulinum toxin in blepharospasm: a 17-year experience

Aquino

Felı́cio

Castro

et al. 2012

Arq. Neuro-Psiquiatr.

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OBJECTIVE: It was to analyze clinical aspects of patients with blepharospasm, including outcomes of botulinum toxin treatment. Additionally, clinical characteristics of isolated blepharospasm were compared to those of blepharospasm plus other movement disorders. METHODS: Clinical data recorded during 17 years were reviewed. The variables included age, gender, age of onset, past medical history, head trauma, smoking history, family history of dystonia, severity, duration of botulinum toxin relief and adverse effects. RESULTS: A total of 125 patients were included and 75.2% were female. The mean age of onset was 54.3 years; 89.6% of the individuals started with contractions in eye region, and 39.2% of them spread to lower face or neck. Isolated blepharospasm group was compared with blepharospasm-plus group for demographic and clinical features, and therapeutic outcomes, without significant differences. Botulinum toxin treatment improved the severity of contractions (p=0.01) with low rate of side effects (14%). CONCLUSIONS: Both groups - isolated blepharospasm and blepharospasm-plus - shared similar results concerning epidemiology, clinical features and therapeutic response to botulinum toxin.

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Relative risk of spread of symptoms among the focal onset primary dystonias

Cited by 99 publications

References 25 publications

Retrocollis, anterocollis or head tremor may predict the spreading of dystonic movements in primary cervical dystonia

Retrocollis, anterocollis or head tremor may predict the spreading of dystonic movements in primary cervical dystonia

Designing Clinical Trials for Dystonia

Clinical features and treatment with botulinum toxin in blepharospasm: a 17-year experience

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