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Abstract

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Article in Japanese

Case Report

A case of diffuse pulmonary arteriovenous malformations associated with eosinophilia and peripheral neuropathy

Shuichi Yoshidaa,b  Koichi Sayamaa  Takahisa Takiharaa  Subaru Hashimotoc  Hiroaki Sugiurac  Kenjiro Kosakid  Yuichiro Hayashie  Koichirou Asanoa 

aDivision of Pulmonary Medicine, Department of Medicine, Keio University School of Medicine
bDepartment of Pulmonary Medicine, National Hospital Organization Tokyo Medical Center
cDepartment of Radiology, Keio University School of Medicine
dDepartment of Pediatrics, Keio University School of Medicine
eDepartment of Pathology, Keio University School of Medicine

ABSTRACT

A 40-year-old man admitted to our hospital of brain abscess exhibited moderate arterial hypoxemia. Chest CT scan showed diffuse multiple arteriovenous malformations (AVMs) in the bilateral lungs and arterioportal shunts in the liver. He had also been treated for multiple epithelioid hemangiomas in the skin. There was no mutation detected in endoglin (ENG) or activin A receptor type II-like kinase 1 (ACVRL1) gene, and the diagnosis of hereditary hemorrhagic telangiectasia could not be confirmed. A coil embolization of major AVMs (6 AVMs in the right lung and 5 AVMs in the left) was performed without significant decrease in the shunt fraction. He felt numbness on his lower legs, and peripheral blood eosinophilia, elevated serum IgE levels, and positive MPO-ANCA were observed. Systemic treatment with corticosteroid improved the numbness, eosinophilia, and MPO-ANCA. This is the first report of a case with multiple vascular anomaly including pulmonary AVMs, accompanied by peripheral blood eosinophilia and ANCA-positive peripheral neuropathy.

KEYWORDS

Arteriovenous malformation  Eosinophilia  Epithelioid hemangioma  MPO-ANCA 

Received 31 Mar 2011 / Accepted 16 Dec 2011

AJRS, 1(4): 322-326, 2012

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