Display Settings:

Format

Send to:

Choose Destination
See comment in PubMed Commons below
Arch Neurol. 2008 Aug;65(8):1117-21. doi: 10.1001/archneur.65.8.1117.

Response of motor complications in Cockayne syndrome to carbidopa-levodopa.

Author information

  • 1Division ofGenetics, Children's Hospital Boston, Boston, MA 02115, USA.

Abstract

BACKGROUND:

Gait difficulties, tremors, and coordination difficulties are common features of Cockayne syndrome that are consequences of leukodystrophy, cerebellar atrophy, and demyelinating neuropathy, but no pharmacotherapy for these disabling symptoms is available.

OBJECTIVE:

To determine whether carbidopa-levodopa relieves tremors and other motor complications of Cockayne syndrome.

DESIGN:

Mutation analysis and case report study.

SETTING:

Hospital clinic and genetics research laboratory. Patients We studied 3 patients with Cockayne syndrome, a rare autosomal recessive neurodegenerative disorder for which no known treatments are available. Intervention Carbidopa-levodopa therapy.

MAIN OUTCOME MEASURES:

Status of tremors, ability to perform daily tasks, serial physical examinations, and results of handwriting samples.

RESULTS:

All 3 patients had a clear reduction in tremors and improvements in handwriting and manipulation of utensils and cups.

CONCLUSIONS:

Patients with Cockayne syndrome should be evaluated carefully for movement disorders. A clinical trial should be considered to evaluate this therapy further.

PMID:
18695064
[PubMed - indexed for MEDLINE]
PubMed Commons home

PubMed Commons

0 comments
How to join PubMed Commons

    Supplemental Content

    Icon for Silverchair Information Systems
    Loading ...
    Write to the Help Desk