Disease progression in blepharospasm: a 5-year longitudinal study.


Journal

European journal of neurology
ISSN: 1468-1331
Titre abrégé: Eur J Neurol
Pays: England
ID NLM: 9506311

Informations de publication

Date de publication:
02 2019
Historique:
received: 15 05 2018
accepted: 08 10 2018
pubmed: 12 10 2018
medline: 19 6 2019
entrez: 12 10 2018
Statut: ppublish

Résumé

The clinical manifestation of dystonic spasms in blepharospasm (BSP) patients may be heterogeneous. Whether the varying phenomenology of eyelid spasms becomes manifest sequentially during the course of the disease or aggregates in separate clusters according to different disease courses is still unclear. For this purpose, the clinical features in BSP patients were evaluated longitudinally over a 5-year period and also the blink reflex recovery cycle was tested in a subgroup of BSP patients. Sixty BSP patients were videotaped at time 0 and after approximately 5 years of follow-up. Two experts in movement disorders, who were blinded to the video order, reviewed the videotapes and scored the severity of BSP using the Blepharospasm Severity Rating Scale. Changes in the R2 recovery index were also evaluated in 18 patients twice, i.e. upon enrolment and at the follow-up. The severity of BSP worsened significantly over the 5-year follow-up period owing to the appearance or the increased duration and frequency of prolonged spasms. It was also found that the blink reflex recovery cycle worsened at follow-up in comparison with the baseline. This study shows that the disease progression of BSP is characterized by the appearance or worsening of prolonged spasms. Prolonged spasms are accompanied by changes in the excitability of brainstem interneurons. Aging-related effects may exacerbate the pathophysiological mechanisms underlying spasms.

Sections du résumé

BACKGROUND AND PURPOSE
The clinical manifestation of dystonic spasms in blepharospasm (BSP) patients may be heterogeneous. Whether the varying phenomenology of eyelid spasms becomes manifest sequentially during the course of the disease or aggregates in separate clusters according to different disease courses is still unclear. For this purpose, the clinical features in BSP patients were evaluated longitudinally over a 5-year period and also the blink reflex recovery cycle was tested in a subgroup of BSP patients.
METHODS
Sixty BSP patients were videotaped at time 0 and after approximately 5 years of follow-up. Two experts in movement disorders, who were blinded to the video order, reviewed the videotapes and scored the severity of BSP using the Blepharospasm Severity Rating Scale. Changes in the R2 recovery index were also evaluated in 18 patients twice, i.e. upon enrolment and at the follow-up.
RESULTS
The severity of BSP worsened significantly over the 5-year follow-up period owing to the appearance or the increased duration and frequency of prolonged spasms. It was also found that the blink reflex recovery cycle worsened at follow-up in comparison with the baseline.
CONCLUSIONS
This study shows that the disease progression of BSP is characterized by the appearance or worsening of prolonged spasms. Prolonged spasms are accompanied by changes in the excitability of brainstem interneurons. Aging-related effects may exacerbate the pathophysiological mechanisms underlying spasms.

Identifiants

pubmed: 30308706
doi: 10.1111/ene.13832
doi:

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

268-273

Informations de copyright

© 2018 EAN.

Auteurs

G Ferrazzano (G)

IRCCS Neuromed, Pozzilli, Italy.

A Conte (A)

IRCCS Neuromed, Pozzilli, Italy.
Department of Human Neurosciences, Sapienza University of Rome, Rome, Italy.

A Gigante (A)

Department of Basic Medical Sciences, Neurosciences and Sensory Organs, 'Aldo Moro', University of Bari, Bari, Italy.

G Defazio (G)

Department of Basic Medical Sciences, Neurosciences and Sensory Organs, 'Aldo Moro', University of Bari, Bari, Italy.
Department of Medical Sciences and Public Health, University of Cagliari, Monserrato, Italy.

A Berardelli (A)

IRCCS Neuromed, Pozzilli, Italy.
Department of Human Neurosciences, Sapienza University of Rome, Rome, Italy.

G Fabbrini (G)

IRCCS Neuromed, Pozzilli, Italy.
Department of Human Neurosciences, Sapienza University of Rome, Rome, Italy.

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