Ann Thorac Surg 1995;59:528-530
© 1995 The Society of Thoracic Surgeons
Case Reports
Rupture of Congenital Peripheral Pulmonary Aneurysm
Ichiro Fukai, MD,
Akira Masaoka, MD, PhD,
Yosuke Yamakawa, MD,
Hiroshi Niwa, MD,
Toyohiro Tada, MD,
Mitsuhiro Kamei, MD,
Emiko Muto, MD
Second Department of Surgery and Pathology, Nagoya City University Medical School, and Department of Respiratory Disease, Chubu-Rosai Hospital, Nagoya, Japan
Accepted for publication May 31, 1994.
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Abstract
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A 58-year-old man presenting with solitary aneurysm of a peripheral pulmonary artery was treated by left lower lobectomy. Histologically, the aneurysmal wall showed medial hypertrophy with the loss of smooth muscle fibers, without evidence of a mycotic process or inflammatory exudate. The aneurysm appeared to be congenital in origin.
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Introduction
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See also page 530.
Pulmonary artery aneurysm is rare, and peripheral-type aneurysm of the segmental pulmonary artery is even less common [1, 2]. The etiologic factors of peripheral pulmonary aneurysm are multiple. They include congenital weakness of the arterial wall, trauma, infection (specific or nonspecific), and pulmonary hypertension [1–3]. In this report, we describe a patient with massive hemoptysis who was found to have a solitary peripheral pulmonary aneurysm, probably congenital in origin.
A 58-year-old man was brought to the emergency room by ambulance on May 9, 1993, for massive hemoptysis necessitating intensive treatment. Profound hypoxia (arterial oxygen tension, 44 mm Hg) developed, but the patient was resuscitated. Bronchoscopy performed at that time revealed blood issuing from all lobes of the left lung. Chest roentgenogram showed a round mass, 1.5 cm in diameter, in the left inferior lobe overlying the cardiac silhouette (Fig 1). The mass had been present for 3 years and had remained unchanged in size, although it was unnoticed at the time. Pulmonary arteriogram on the tenth hospital day demonstrated a solitary aneurysm in a branch of the left pulmonary artery to the inferior lobe (subsuperior branch) (Fig 2). Simultanously performed right cardiac catheterization revealed normal pulmonary and right ventricular pressures.
Left posterolateral thoracotomy was performed on July 9, 1993. Dissection was begun in the major fissure, but severe adhesion, probably due to the bleeding from the aneurysm into the lung parenchyma, was encountered. No aneurysm was identified, and there was unfortunate injury to the superior branch of the pulmonary artery (A6). Because repair of A6 could not be accomplished satisfactorily, we could not perform basal segmentectomy and selected full lobectomy.
The aneurysm, 1.5 cm in diameter, was filled with a thrombus and had eroded into the lumen of the adjacent bronchus (suprabasal bronchus). Histologically, there was medial hypertrophy with loss of smooth muscle (Fig 3). The specimen showed no evidence of a mycotic process or inflammatory exudate.
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Comment
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A solitary peripheral pulmonary aneurysm is a rare entity. Etiologic factors are thought to include trauma, infections (specific or nonspecific), pulmonary hypertension, and pulmonary vascular defects (congenital or aquired) [1–3]. In our case, right cardiac catheterization ruled out associated congenital heart disease and pulmonary hypertension, and there was no history of venereal disease or chest injury. Histologic examination revealed no evidence of a mycotic process. It therefore seems more likely that the cause of the aneurysm in our case was weakness of the pulmonary artery wall.
Solitary peripheral pulmonary aneurysms caused by congenital vascular defects are extremely uncommon, and pathologic findings are rarely discussed in detail in the literature. With special reference to pulmonary medial degenerations, cystic medial necrosis, which could be considered a forme furste of Marfan's syndrome, is thought to be the leading cause of true pulmonary arterial aneurysms [4, 5]. Pulmonary hypertension is probably an essential factor that underlies cystic medial necrosis [6]. The histology of our case may be compatible with an early stage of cystic medial necrosis [6], although pulmonary hypertension was not associated.
Concerning the diagnosis of solitary pulmonary aneurysm, physicians should pay more attention to a peripheral coin lesion on chest roentgenograms and obtain a correct diagnosis by angiography. Once the diagnosis is made, surgical resection should be considered because of the possible fatal outcome if the aneurysm is left untreated. In fact, the pulmonary aneurysm in the case under study had been left untreated for at least 3 years retrospectively, and had ruptured. Surgical treatment consists of lobectomy, segmental resection, and aneurysmectomy [1]. Cardiopulmonary bypass may be attempted for proximal lesions and particularly to preserve lung tissue in patients with limited pulmonary reserve [7].
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Fig 1. . Chest roentgenogram showing a round mass in the inferior lobe, overlying cardiac silhouette (arrows). The chest roentgenogram 3 years before admission also revealed the mass, the size of which was the same, although it had gone unnoted at the time.
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Fig 3. . Pulmonary artery aneurysm originating from subsuperior branch (A) and low-magnification photomicrograph of the aneurysm (B) (x3 before 33% reduction). The small arrow points to the wall of the aneurysm, showing medial hypertrophy with loss of smooth muscle. The asterisk points to the bronchial lumen (B6). The aneurysm eroded into the bronchus. The large arrow indicates the spot where the aneurysm eroded into the bronchus.
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Footnotes
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Address reprint requests to Dr Fukai, Second Department of Surgery, Nagoya City University Medical School, Mizuho-ku, Nagoya, Japan.
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References
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- Monchik J, Wilkins EW Jr. Solitary aneurysm of the middle lobe artery: a case report and review of solitary peripheral pulmonary artery aneurysms. Ann Thorac Surg 1974;17:496–503.[Medline]
- Ungaro R, Saab S, Almond CH, Kumar S. Solitary peripheral pulmonary artery aneurysmas: pathogenesis and surgical treatment. J Thorac Cardiovasc Surg 1976;71:566–71.[Abstract]
- Brenner O. Pathology of vessels of the pulmonary circulation. Arch Intern Med 1935;56:1189–1241.[Abstract/Free Full Text]
- Natelson EA, Watts HD, Fred HL. Cystic medionecrosis of the pulmonary artery. Chest 1970;57:333–5.[Abstract/Free Full Text]
- Butto F, Lucas RV, Edwards JE. Pulmonary artery aneurysm. A pathologic study of five cases. Chest 1987;91:237–41.[Abstract/Free Full Text]
- Tredal SM, Carter JB, Edwards JE. Cystic medial necrosis of the pulmonary artery. Association with pulmonary hypertension. Arch Pathol 1974;97:183–5.[Medline]
- Murphy JP, Adyanthaya AV, Adams PR, et al. Peripheral pulmonary artery aneurysm in a patient with limited respiratory reserve: controlled resection using cardiopulmonary bypass. Ann Thorac Surg 1987;43:323–5.[Abstract]
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Abstract
Introduction
