Document Detail


Successful cyclophosphamide therapy with complete resolution of pulmonary artery aneurysm in Hughes-Stovin syndrome patient.
MedLine Citation:
PMID:  18592134     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Hughes-Stovin syndrome is an exceedingly rare disorder of unknown etiology, and it is characterized by multiple pulmonary artery aneurysms and peripheral venous thrombosis. We report a case of Hughes-Stovin syndrome with complete resolution of pulmonary artery aneurysm after treatment with cyclophosphamide. A 48-year-old man presented with massive hemoptysis, fever, and weight loss of 4-month duration. Computed tomography angiography revealed multiple pulmonary arterial aneurysms of variable sizes in both lung fields. Inferior vena caval thrombosis was also noted. Despite multiple surgical procedures including embolectomy and lobectomy combined with steroid therapy, new aneurysms continued to appear with persistent hemoptysis. Thorough history and physical examination did not suggest any evidence of Behcet's disease. Hughes-Stovin syndrome was subsequently diagnosed. High-dose prednisolone combined with oral cyclophosphamide therapy was initiated. Chest radiography follow-up in 3 months revealed complete resolution of pulmonary aneurysm. No new aneurysm was detected at 15 months of follow-up.
Authors:
Jaejoon Lee; Jung-Won Noh; Ji Won Hwang; Hyungjin Kim; Joong Kyong Ahn; Eun-Mi Koh; Hoon-Suk Cha
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Publication Detail:
Type:  Case Reports; Journal Article     Date:  2008-07-01
Journal Detail:
Title:  Clinical rheumatology     Volume:  27     ISSN:  1434-9949     ISO Abbreviation:  Clin. Rheumatol.     Publication Date:  2008 Nov 
Date Detail:
Created Date:  2008-10-07     Completed Date:  2009-04-14     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8211469     Medline TA:  Clin Rheumatol     Country:  Belgium    
Other Details:
Languages:  eng     Pagination:  1455-8     Citation Subset:  IM    
Affiliation:
Department of Medicine, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Republic of Korea.
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MeSH Terms
Descriptor/Qualifier:
Aneurysm / complications,  drug therapy*
Antirheumatic Agents / therapeutic use*
Cyclophosphamide / therapeutic use*
Humans
Male
Middle Aged
Pulmonary Artery / pathology*
Syndrome
Venous Thrombosis / complications*
Chemical
Reg. No./Substance:
0/Antirheumatic Agents; 50-18-0/Cyclophosphamide

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


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