Hemoptysis: unilateral pulmonary artery atresia? a case report.

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Citation: Journal Of Cardiothoracic Surgery. 18(1):199, 2023 Jun 29.PMID: 37386643Institution: MedStar Washington Hospital CenterDepartment: Medicine/Pulmonary-Critical Care | Pulmonary/Critical Care Fellowship | Surgery | Surgery/Thoracic SurgeryForm of publication: Journal ArticleMedline article type(s): Case Reports | Journal ArticleSubject headings: *Heart Defects, Congenital | *Hypertension, Pulmonary | *Respiratory Tract Infections | Dyspnea | Hemoptysis/et [Etiology] | Humans | Male | Middle Aged | Pulmonary Artery/dg [Diagnostic Imaging] | Pulmonary Artery/su [Surgery] | Year: 2023Local holdings: Available online through MWHC library: 2006 - 2007ISSN:
  • 1749-8090
Name of journal: Journal of cardiothoracic surgeryAbstract: BACKGROUND: Asymptomatic, isolated cases of unilateral pulmonary artery atresia may present in adulthood with symptoms such as recurrent respiratory infections, dyspnea, hemoptysis, and pulmonary hypertension. Unlike previously reported patients that underwent surgical management for this pathology, the patient in this report had no chronic history of recurrent respiratory infections, dyspnea, or pulmonary hypertension, making a diagnosis prior to extensive imaging difficult.CASE PRESENTATION: A 55-year-old male presented to our emergency department (ED) with a 3-day history of recurrent cough with 2-3 tablespoons of hemoptysis per episode, chills, and occasional wheezing. A computed tomography angiography (CTA) was performed, which identified a congenital absence of the left pulmonary artery and a right-sided aortic arch. Hypertrophied left intercostal and bronchial arteries were noted to be perfusing the left lung. V/Q scan confirmed a heterogeneous distribution of gas throughout both lung fields with 97% perfusion to the right lung, but no visualization of the left lung on the perfusion images. Given extensive collateral blood supply to the left lung, interventional radiology performed a GELFOAM R embolization of the hypertrophied left bronchial artery and two parasitized arteries from the left subclavian artery to minimize intra-operative blood loss. This was immediately followed by a left thoracotomy, pneumonectomy, intercostal muscle flap placement, and bronchoscopy. The procedure was 360 min long with a total of 1500 cc blood loss that was salvaged and re-infused. No additional blood products were administered. The patient remained intubated post-operatively and was transferred to the surgical intensive care unit. His postoperative course was complicated by troponin leak, rhabdomyolysis, delirium, and ileus, all of which resolved over time. He was discharged home on postoperative day seven and continues to do well one-year later.CONCLUSIONS: The patient in this report presented with several episodes of isolated hemoptysis but unlike previously reported cases of unilateral pulmonary artery atresia, he had no history of recurrent respiratory infections, dyspnea, or pulmonary hypertension. Although unilateral pulmonary artery atresia is a rare diagnosis, in patients with unexplained, isolated hemoptysis, further examination of the vasculature may be warranted, and surgical management may be beneficial in appropriate, symptomatic patients. Copyright © 2023. The Author(s).All authors: Cousins S, Holliday T, Khaitan PG, Maini A, Memoli JW, Vasu TSFiscal year: FY2023Digital Object Identifier: Date added to catalog: 2023-07-27
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Item type Current library Collection Call number Status Date due Barcode
Journal Article MedStar Authors Catalog Article 37386643 Available 37386643

Available online through MWHC library: 2006 - 2007

BACKGROUND: Asymptomatic, isolated cases of unilateral pulmonary artery atresia may present in adulthood with symptoms such as recurrent respiratory infections, dyspnea, hemoptysis, and pulmonary hypertension. Unlike previously reported patients that underwent surgical management for this pathology, the patient in this report had no chronic history of recurrent respiratory infections, dyspnea, or pulmonary hypertension, making a diagnosis prior to extensive imaging difficult.

CASE PRESENTATION: A 55-year-old male presented to our emergency department (ED) with a 3-day history of recurrent cough with 2-3 tablespoons of hemoptysis per episode, chills, and occasional wheezing. A computed tomography angiography (CTA) was performed, which identified a congenital absence of the left pulmonary artery and a right-sided aortic arch. Hypertrophied left intercostal and bronchial arteries were noted to be perfusing the left lung. V/Q scan confirmed a heterogeneous distribution of gas throughout both lung fields with 97% perfusion to the right lung, but no visualization of the left lung on the perfusion images. Given extensive collateral blood supply to the left lung, interventional radiology performed a GELFOAM R embolization of the hypertrophied left bronchial artery and two parasitized arteries from the left subclavian artery to minimize intra-operative blood loss. This was immediately followed by a left thoracotomy, pneumonectomy, intercostal muscle flap placement, and bronchoscopy. The procedure was 360 min long with a total of 1500 cc blood loss that was salvaged and re-infused. No additional blood products were administered. The patient remained intubated post-operatively and was transferred to the surgical intensive care unit. His postoperative course was complicated by troponin leak, rhabdomyolysis, delirium, and ileus, all of which resolved over time. He was discharged home on postoperative day seven and continues to do well one-year later.

CONCLUSIONS: The patient in this report presented with several episodes of isolated hemoptysis but unlike previously reported cases of unilateral pulmonary artery atresia, he had no history of recurrent respiratory infections, dyspnea, or pulmonary hypertension. Although unilateral pulmonary artery atresia is a rare diagnosis, in patients with unexplained, isolated hemoptysis, further examination of the vasculature may be warranted, and surgical management may be beneficial in appropriate, symptomatic patients. Copyright © 2023. The Author(s).

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