Isolated unilateral absence of left pulmonary artery with peribronchial arteriovenous malformation showing recurrent hemoptysis

Taguchi, Tomoaki; Ikeda, Kazuyuki; Kume, Kazuhiro; Nagasaki, Akira; Goto, Shunsuke; Matsuo, Susumu; Kanehiro, Hiromichi; Tamura, S

doi:10.1007/bf02388246

Cited by 21 publications

(7 citation statements)

References 8 publications

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“…In only three cases, the aortic arch was on the same side as the absent pulmonary artery. 16,30 Symptoms were chest pain, 35,36 pleural effusion, 35 and recurrent pulmonary infections in 37% of patients (35 of 94 patients), dyspnea or exercise limitations in 40% of patients (36 of 92 patients), and hemoptysis in 20% of patients (18 of 92 patients). Pulmonary hypertension was found in 44% of patients (25 of 57 patients), while high-altitude pulmonary edema (HAPE) was present in 12% of patients (7 of 57 patients) [ Table 1].…”

Section: Resultsmentioning

confidence: 99%

Isolated Unilateral Absence of a Pulmonary Artery

Harkel

Blom

Ottenkamp

2002

Chest

277

137

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Section: Resultsmentioning

confidence: 99%

Isolated Unilateral Absence of a Pulmonary Artery

Harkel

Blom

Ottenkamp

2002

Chest

277

137

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“…If there are recurrent hemoptyses in the patient, embolization may be risky and pneumonectomy or lobectomy may be necessary. In the literature, cases that required resectional surgery at a young age, especially because of massive hemoptysis, were reported (15)(16)(17). In two of our cases, massive hemoptysis was observed; pneumonectomy was performed for one case and blood transfusion for the other.…”

Section: Discussionmentioning

confidence: 65%

Pulmonary Artery Agenesis: A Case Series

Ağca¹,

Bulum²,

Günen³

et al. 2015

Eurasian J Pulmonol

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Pulmonary artery agenesis is a rare congenital abnormality in which atresia was encountered in the short segment of the right or left pulmonary arteries. It can be isolated or associated with cardiac abnormalities such as tetralogy of Fallot, septal defects or pulmonary stenosis. The majority of cases are diagnosed in childhood whereas some cases yield no symptoms until adulthood. We evaluated retrospectively 5 pulmonary artery agenesis cases diagnosed in our clinics between 1998-2010 with respect to the literature. Keywords: Angiography, echocardiography, pulmonary artery agenesis, vascular abnormalities INTRODUCTIONIsolated one-sided pulmonary artery agenesis is a rare congenital anomaly in the proximal short segment of the right or left pulmonary artery. Generally, the distal parts of the artery at the hilus are protected (1). It can occur with Fallot tetralogy, septal defects, or pulmonary stenosis as well as by itself. It can often be identified during childhood years, but it can hide symptoms until the late adult ages. When it presents, the symptoms are recurrent infections, hemoptysis, shortness of breath, restricted exercise, and chest pain are observed. The diagnosis is hard because of the lack of specific symptoms and rarity of the disease. It is important to diagnose this disease at early ages because it causes serious complications such as massive hemoptysis, pulmonary hypertension, respiratory failure, and bronchiectasis (2).The mortality rate of this rare anomaly is high because of its complications. We retrospectively analyzed five cases of pulmonary artery agenesis at older ages in accordance with the literature knowledge. CASE PRESENTATIONS Case1A 37-year-old male patient was admitted to our emergency department with complaints of fever, shortness of breath, yellow phlegm, and hemoptysis occurring occasionally. It was learned from his history that he had been treated for bronchiectasis. Because a left pulmonary artery shadow was not seen on posteroanterior (PA) chest X-ray (Figure 1a), echocardiography (ECO) was performed for vein malformation. A mild expansion in the right atrium and right ventricle and a systolic pulmonary artery pressure (PAPs) of 50 mmHg were observed. Chest computerized tomography (CT) (Figure 1b) revealed varicose and cystic bronchiectasis in the left lung, nearly total volume loss, rising in the left diaphragm, and replacement toward the left in mediastinal structures. In the spirometric examination, mild restrictive and moderate obstructive [FVC: 3.29 L (72% of expected), FEV 1 : 1.90 L (51% of expected), FEV 1 /FVC: 57.7% (not expected)] respiratory dysfunction was detected. In the bronchoscopic analysis of the patient, the mouth of the left upper lobe was found to be narrowed in the shape of a lip. The left lung was totally hypoplasic and hypoperfused on ventilation perfusion (V/Q) scintigraphy Figure 1c). Invasive pulmonary angiography of the patient ( Figure 1d) revealed that the left pulmonary artery was closed starting from the ostium level and the patien...

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“…Methods have included pneumonectomy in an adult, reported by Mehta et al [14]. Taguchi reported pneumonectomy and embolectomy in a 10-year-old child [15]. A staged extra-anatomic conduit to deliver low-pressure blood to the lung, and interruption of the high-pressure vessels were reported in a 14-year-old child by Rankin [16].…”

Section: Discussionmentioning

confidence: 99%

Evaluation and Management of Massive Endotracheal Hemorrhage in Pediatric Patients: A Case Report and Review of Literature

Vaughn¹,

Shete²,

Stocks³

et al. 2013

IJOHNS

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Massive hemoptysis is a frightening and life-threatening event in children. Prompt, aggressive evaluation and management are necessary. The most common cause of hemoptysis in a pediatric patient is infectious, but other various etiologies including tracheotomy related problems, cystic fibrosis, bronchiectasis, congenital anomalies of the cardiopulmonary vasculature and suction trauma must be considered as well. Presented is a report of a case of acute, massive endotracheal hemorrhage with multiple methods of management including balloon tamponade, highly selective embolization, and mainstem occlusion. This case is an addition to our previously reported case series of nine pediatric patients with massive pulmonary hemorrhage. Various diagnostic and management techniques of hemoptyis in pediatric patients are discussed with an extensive review of the literature.

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Isolated unilateral absence of left pulmonary artery with peribronchial arteriovenous malformation showing recurrent hemoptysis

Cited by 21 publications

References 8 publications

Isolated Unilateral Absence of a Pulmonary Artery

Isolated Unilateral Absence of a Pulmonary Artery

Pulmonary Artery Agenesis: A Case Series

Evaluation and Management of Massive Endotracheal Hemorrhage in Pediatric Patients: A Case Report and Review of Literature

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