Document Detail

Safe treatment of pulmonary hypertension with bosentan in a patient with moyamoya disease and cerebral ischemia.
MedLine Citation:
PMID:  19808994     Owner:  NLM     Status:  MEDLINE    
A 7-year-old girl with Down syndrome and moderately severe pulmonary hypertension experienced a stroke while being treated with a calcium channel blocker. Angiography identified bilateral stenosis of the supraclinoid internal carotid arteries, stenosis or occlusion of the proximal anterior and middle cerebral arteries, and occlusion of the left posterior cerebral artery. She underwent surgery to enhance collateral blood flow to vulnerable areas of the brain. Her pulmonary hypertension therapy was changed to an oral endothelin receptor antagonist. She developed excellent collateral blood flow through external carotid arteries to each cerebral hemisphere and an improvement in blood flow through the right internal carotid artery. This case suggests that bosentan can be used safely in children with moyamoya disease. Additional studies are needed to determine whether endothelin receptor antagonists may influence the progression of moyamoya disease or the development of collateral cerebral blood flow following surgery.
Ronald W Day; Douglas L Brockmeyer; G Peter Feola
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't     Date:  2009-10-06
Journal Detail:
Title:  Journal of child neurology     Volume:  25     ISSN:  1708-8283     ISO Abbreviation:  J. Child Neurol.     Publication Date:  2010 Apr 
Date Detail:
Created Date:  2010-04-12     Completed Date:  2010-06-29     Revised Date:  2013-06-18    
Medline Journal Info:
Nlm Unique ID:  8606714     Medline TA:  J Child Neurol     Country:  United States    
Other Details:
Languages:  eng     Pagination:  504-7     Citation Subset:  IM    
Pediatric Cardiology, Primary Children's Medical Center, University of Utah, Salt Lake City, Utah 84113, USA.
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MeSH Terms
Antihypertensive Agents / pharmacology,  therapeutic use
Brain / blood supply,  pathology
Brain Ischemia / chemically induced,  etiology,  physiopathology
Calcium Channel Blockers / adverse effects*
Cerebral Angiography
Cerebral Arteries / drug effects,  pathology,  radiography
Cerebral Revascularization
Cerebrovascular Circulation / drug effects,  genetics
Hypertension, Pulmonary / drug therapy*,  etiology,  physiopathology
Magnetic Resonance Imaging
Moyamoya Disease / complications*,  genetics,  pathology
Receptors, Endothelin / antagonists & inhibitors,  metabolism
Risk Assessment
Stroke / chemically induced*,  etiology,  physiopathology
Sulfonamides / pharmacology*,  therapeutic use
Treatment Outcome
Reg. No./Substance:
0/Antihypertensive Agents; 0/Calcium Channel Blockers; 0/Receptors, Endothelin; 0/Sulfonamides; Q326023R30/bosentan

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine

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