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Arch Neurol. 2010 Feb;67(2):236-8. doi: 10.1001/archneurol.2009.319.

Intraparenchymal hemorrhage in a patient with osteogenesis imperfecta and plasminogen activator inhibitor-1 deficiency.

Author information

1
Department of Neurology, Dartmouth Hitchcock Medical Center, 1 Medical Center Drive, Lebanon, NH 03756, USA. richard.p.goddeau@hitchcock.org

Abstract

BACKGROUND:

Osteogenesis imperfecta is associated with susceptibility to connective tissue damage, including intracranial but usually extra-axial hemorrhage. Plasminogen activator inhibitor-1 deficiency is a rare fibrinolytic cause of systemic bleeding diathesis.

OBJECTIVE:

To describe a case of a brainstem intraparenchymal hemorrhage associated with connective tissue and coagulation disorders.

DESIGN:

Case report.

SETTING:

Academic medical center.

PATIENT:

A 36-year-old woman with a history of osteogenesis imperfecta presented to the emergency department after an argument, during which she developed left ear pain and right eye esotropia followed by quadriparesis and somnolence. Neuroimaging showed a tegmental mesencephalic hemorrhage.

MAIN OUTCOME MEASURES:

Results of computerized tomography, magnetic resonance angiography, and parenchymal imaging; and serum hematologic markers.

RESULTS:

No underlying vascular abnormality or mass lesion was found. Among coagulopathic serum markers, only plasminogen activator inhibitor-1 activity level was abnormally low.

CONCLUSION:

Intraparenchymal hemorrhage may occur in the setting of a fibrinolytic inhibitory deficiency and osteogenesis imperfecta.

PMID:
20142533
DOI:
10.1001/archneurol.2009.319
[Indexed for MEDLINE]

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