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BMC Pulm Med. 2019 Feb 8;19(1):34. doi: 10.1186/s12890-019-0797-7.

Pulmonary artery pseudoaneurysm causing massive hemoptysis in hyperimmunoglobulin E syndrome: a case report.

Author information

1
Yale University School of Medicine, New Haven, CT, USA.
2
Section of Pulmonary, Critical Care, and Sleep Medicine, Department of Internal Medicine, Yale University School of Medicine, New Haven, CT, USA.
3
Section of Vascular and Interventional Radiology, Department of Radiology and Biomedical Imaging, Yale University School of Medicine, New Haven, CT, USA.
4
Section of Pulmonary, Critical Care, and Sleep Medicine, Department of Internal Medicine, Yale University School of Medicine, New Haven, CT, USA. geoffrey.chupp@yale.edu.

Abstract

BACKGROUND:

Hyperimmunoglobulin E syndrome (HIES) is a rare primary immunodeficiency disorder defined by high serum immunoglobulin E titers that is associated with recurrent respiratory infections, formation of pneumoatoceles, recurrent skin abscesses, and characteristic dental and skeletal abnormalities.

CASE PRESENTATION:

We report a case of a 56-year-old male with a history of HIES, cavitary mycetomas, and allergic bronchopulmonary aspergillosis who presented with recurrent massive hemoptysis. Bronchial artery angiography and bronchoscopy failed to identify active hemorrhage, and two embolizations of the bronchial artery did not resolve the bleeding. Subsequently, selective pulmonary artery angiography was conducted that demonstrated a subsegmental pulmonary artery branch pseudoaneurysm with extravasation into an adjacent lung cavity. This was treated successfully with transcatheter embolization.

CONCLUSIONS:

To our knowledge, this is the first case reported of pulmonary artery pseudoaneurysm in HIES in the medical literature. Pulmonary artery pseudoaneurysm should be considered in the differential diagnosis in patients with HIES and massive hemoptysis.

KEYWORDS:

Hyperimmunoglobulin E syndrome; Massive hemoptysis; Pulmonary artery pseudoaneurysm

PMID:
30736787
PMCID:
PMC6368789
DOI:
10.1186/s12890-019-0797-7
[Indexed for MEDLINE]
Free PMC Article

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