Ann Thorac Surg 2001;71:1023-1025
© 2001 The Society of Thoracic Surgeons
Case report
Pulmonary artery aneurysm in a pregnant woman
Peter J. Gruber, MD, PhDa,
Frederic B. Askin, MDb,
Richard F. Heitmiller, MDa
a Department of Surgery, The Johns Hopkins Medical Institutions, Baltimore, Maryland, USA
b Department of Pathology, The Johns Hopkins Medical Institutions, Baltimore, Maryland, USA
Accepted for publication April 15, 2000.
Address reprint requests to Dr Heitmiller, Osler 624, Johns Hopkins Hospital, Baltimore, MD 21136
e-mail: rheitmil{at}jhu.edu
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Abstract
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We present a patient with a pulmonary artery (PA) aneurysm who has none of the documented causes of PA aneurysm but who is pregnant. We believe that this patient represents a case of primary pregnancy–associated PA aneurysm.
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Introduction
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Pulmonary artery (PA) aneurysms are rare and most commonly are associated with cardiac defects, pulmonary hypertension, intravascular infection, tuberculosis, and chest trauma. We present a pregnant patient with a PA aneurysm who had none of these associated factors. A wide range of nonpulmonary systemic aneurysms have been documented during pregnancy. We believe that our patient represents a case of primary pregnancy–associated PA aneurysm.
A 26-year-old woman presented in her 19th week of pregnancy with a 3-month history of cough, hemoptysis, and left pleuritic chest pain. The patient had previously enjoyed good health. All of her children were healthy and the products of full-term vaginal deliveries. She denied a history of intravenous drug use, chest trauma tuberculosis, or heart disease. Physical examination revealed a well-appearing pregnant woman. She was afebrile with a regular pulse and no cardiac or chest murmurs were identified. The chest was clear to auscultation and no bruits were noted. There was no clubbing. An electrocardiogram and white blood cell count were normal. She was admitted to the hospital for evaluation.
Chest MRI showed a 4.6 x 3.5 x 3.0–cm mass in the left upper lobe anterior segment. Gradient echo imaging demonstrated a vascular mass originating from a nondistended left pulmonary arterial (PA) branch. Pulmonary venous drainage from the mass was not identified (Figs 1 and 2). Both PA aneurysm and arteriovenous malformation were initially considered in the differential diagnosis. On the basis of these radiographic findings and patient symptoms, the diagnosis of PA aneurysm was made. Surgery was recommended.
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Fig 1. Sagittal magnetic resonance imaging view using gradient echo imaging demonstrating a vascular mass originating from a nondistended pulmonary arterial branch.
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Fig 2. Transverse magnetic resonance imaging view demonstrating a peripheral aneurysm originating from a nonenlarged pulmonary arterial branch. No venous drainage is identified.
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A left posterolateral fifth interspace thoracotomy was employed. Examination revealed a 4-cm peripheral mass in the anterior upper lobe segment. The mass was soft and ballotable without a palpable thrill. No other lung or vascular lesions were identified. A left upper lobectomy was performed. The postoperative recovery was uncomplicated and the patient was discharged home on postoperative day 4. Pathology demonstrated aneurysmal dilatation of the involved artery lumen with intimal thickening. High-power views demonstrated mucoid degeneration in the media of the aneurysmal artery (Fig 3). All cultures and stains for infectious organisms were negative. The patient continues to recover uneventfully. The cough, hemoptysis, and pleuritic chest pain have resolved. There have been no problems with her pregnancy, which is progressing to term.
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Fig 3. Photomicrograph demonstrating the lumen and wall of the aneurysm. Dilatation of the artery and intimal thickening are demonstrated by Verhoeff-van Gieson elastic stain. (Magnification x2.)
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Comment
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Pulmonary artery aneurysms are rare. They account for less than 1% of all thoracic aneurysms and autopsy data estimate the incidence to be 1:14,000 patients. The majority of PA aneurysms are seen in patients with structural cardiac defects in which pulmonary hypertension is a prominent factor. These include patent ductus arteriosus, tetrology of Fallot, truncus arteriosus, tricuspid atresia, and less frequently mitral stenosis. Other reported causes of PA aneurysms include extravascular pulmonary infection such as tuberculosis, intravascular infection (most commonly seen in intravenous drug abusers), Marfan’s disease with cystic medial necrosis, catheter-induced trauma, Behçet’s disease, hereditary hemorrhagic telangiectasia, and blunt or penetrating trauma [1–3]. Patients with PA aneurysms present with dyspnea, cough, hemoptysis, chest pain, and cyanosis [1].
In the differential diagnosis of this pulmonary vascular lesion arteriovenous (AV) malformation was initially considered. Aside from embolic complications patients with pulmonary AV malformations present with minimal and chronic dyspnea. On examination, evidence of cyanosis and clubbing are identified. A murmur is often identified over the ipsilateral hemithorax. Arteriovenous malformations are most commonly located in the lower lung fields. On contrast chest CT scan, both the feeding arterial and the exiting venous vessels are characteristically identified. Our patient presented with recent-onset symptoms of cough, hemoptysis, and chest pain, as well as an upper pulmonary vascular lesion with a nonenlarged "feeding" artery and without an exiting vein. On the basis of these clinical and radiographic findings the diagnosis of PA aneurysm was made.
We believe that our patient represents a case of primary pregnancy-associated PA aneurysm. To our knowledge, a description of PA aneurysm in the absence of patent ductus, cardiac abnormalities, infection, or trauma during pregnancy has not been reported. Our patient had no evidence for structural cardiac defects, pulmonary hypertension, intravascular infection, cystic medial necrosis, or trauma. On the other hand, aneurysms of a wide range of systemic arteries have been documented during pregnancy, including ovarian, endometrial, splenic, renal, subchorionic, brachial, spinal, and pancreaticoduodenal [4–8]. Clearly pregnancy results in a physiologic environment that facilitates arterial aneurysm formation. There is no reason to expect that the pulmonary artery would be exempt from this risk. In our patient the pathology findings demonstrating mucoid degeneration in the media of the aneurysm would be consistent with a pregnancy-related etiology. The specific mechanism for aneurysm formation in these patients is not known.
The natural history of these aneurysms is conjectural given the small number of reported cases. As spontaneous rupture rates of up to 50% have been reported [1], aggressive surgical treatment seems warranted, especially in young healthy patients. On the other hand, successful conservative management has been reported in a 78-year-old patient who has remained stable over an 18-month follow-up [9].
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References
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Abstract
Introduction
