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      Isolated large vessel pulmonary vasculitis leading to pulmonary artery aneurysm formation: a case report and literature review

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          Abstract

          Pulmonary artery (PA) vasculitis occurs in association with primary vasculitides—in particular, giant cell arteritis, Takayasu’s arteritis, or Behçet’s disease—or secondary vasculitis as a result of infections or malignancy. However, PA vasculitis in isolation and with concomitant aneurysmal dilation is an unusual finding. We present a rare case of PA aneurysm secondary to isolated PA vasculitis in an asymptomatic patient with no features of systemic vasculitis. This case highlights one of the first cases of PA vasculitis managed with surgical resection alone.

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          Pulmonary artery involvement and associated lung disease in Behçet disease: a series of 47 patients.

          Gulen Hatemi, Melike Melikoglu, Harun Ozer … (2011)
          Pulmonary artery aneurysms (PAAs) are well known causes of mortality and morbidity in Behçet disease (BD). However, pulmonary artery involvement in BD is not limited to PAA; the other main type of pulmonary artery involvement is pulmonary artery thrombus (PAT), with or without associated PAA. In addition, other types of lung disease like nodules and cavities in the lung parenchyma are frequently associated with pulmonary artery involvement, and can be misinterpreted as being due to infection. We surveyed the clinical, radiologic, and laboratory characteristics and outcome of 47 BD patients with pulmonary artery involvement and the associated findings, all seen and followed at a single dedicated tertiary care center.We identified 47 (41 male, 6 female) patients in whom pulmonary artery involvement was diagnosed, who were registered in the multidisciplinary clinic at Cerrahpasa Medical Faculty between January 2000 and December 2007. Mean age at diagnosis was 29 ± 8 years, and mean disease duration to the onset of pulmonary artery involvement was 3.6 ± 4.8 years. Hemoptysis was the most common presenting symptom (79%) followed by cough, fever, dyspnea, and pleuritic chest pain. Thirty-four of 47 patients (72%) presented with PAA, including 8 with associated PAT. The remaining 13 patients (28%) had isolated PAT. Patients with isolated PAT in general have clinical features similar to patients with PAA. However, hemoptysis was less frequent and voluminous in patients with isolated PAT. Most (91%) of the patients had active disease outside the lungs when they presented with pulmonary artery involvement.Forty (85%) patients had nodules and 6 (13%) had cavities when first seen. Peripheral venous thrombosis was present in 36 of 47 (77%) patients, and intracardiac thrombi in 12 of the 36 (33%) patients. Nodules, cavities, and intracardiac thrombi were mainly present in the acute stages of pulmonary artery involvement.Pulmonary artery involvement is usually multiple, and involves mostly descending branches of the pulmonary artery. Pulmonary artery involvement may disappear, but arterial stenosis or occlusions usually develop at the same location. After a mean follow-up of 7 years, 12 of 47 (26%) patients were dead; patients with larger aneurysms were more likely to die. Sixteen of 47 (34%) patients were symptom free, and the remaining 40% had mild dyspnea (13/47) and/or small bouts of hemoptysis (8/47).Pulmonary artery pressure may be elevated, and may indicate a poor prognosis. Mediastinal lymphadenopathy and mild pleural and pericardial effusions may also be observed. Corticosteroids and immunosuppressive agents are the mainstays of treatment; however, refractory cases may require embolization, lobectomy, cavitectomy, and decortication.
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            Rupture and dissection in pulmonary artery aneurysms: incidence, cause, and treatment--review and case report.

            W Klepetko, O Senbaklavaci, A Bartunek … (2001)
            Article 0 comments Cited 24 times – based on 0 reviews     Review now
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              Pulmonary artery aneurysms: four case reports and literature review.

              P Theodoropoulos, A. Ziganshin, Maryann Tranquilli … (2013)
              Aneurysms of the pulmonary artery are proven to be a very rare entity. Association with structural cardiac anomalies, structural vascular anomalies, pulmonary hypertension, vasculitis, and infection has been noted. Surgical intervention of symptomatic aneurysms is recommended. A more detailed study of the natural history of these aneurysms is needed. Here, we report four cases of pulmonary artery aneurysms as well as a brief review of the literature existing on this subject. The first case is of a 41-year-old woman with the aneurysm located 1 cm distal to the pulmonary valve extending to the bifurcation of the main pulmonary artery. The second case is of a 76-year-old woman with a large aneurysm of the main pulmonary artery and the left pulmonary artery. The third case is of a 61-year-old woman with an aneurysm of the common pulmonary artery and right pulmonary artery. The fourth case is of a 28-year-old woman with a 5-cm symptomatic aneurysm extending from the valve up to the pulmonary bifurcation. Surgical excision and reconstruction was ordered for cases 1, 2, and 4.
              Article 0 comments Cited 23 times – based on 0 reviews     Review now
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                Author and article information

                Journal
                Journal ID (nlm-ta): Pulm Circ
                Journal ID (iso-abbrev): Pulm Circ
                Journal ID (publisher-id): PUL
                Journal ID (hwp): sppul
                Title: Pulmonary Circulation
                Publisher: SAGE Publications (Sage UK: London, England )
                ISSN (Print): 2045-8932
                ISSN (Electronic): 2045-8940
                Publication date (Electronic): 28 February 2018
                Publication date Collection: April 2018
                Volume: 8
                Issue: 2
                Electronic Location Identifier: 2045894018765346
                Affiliations
                [1 ]Department of Medicine, University of British Columbia, Vancouver, BC, Canada
                [2 ]Division of Rheumatology, University of British Columbia, Vancouver, BC, Canada
                [3 ]Divison of Cardiology, University of British Columbia, Vancouver, BC, Canada
                Author notes
                [*]Nathan W. Brunner, Gordon and Leslie Diamond Health Care Center, Pulmonary Hypertension Clinic, 7th floor, 2775 Laurel Street, Vancouver, BC, Canada V5Z 1M9. Email: nathan.brunner@ 123456vch.ca
                Article
                Publisher ID: 10.1177_2045894018765346
                DOI: 10.1177/2045894018765346
                PMC ID: 5871219
                PubMed ID: 29488418
                SO-VID: 018d611c-bf39-4156-89ef-1c440d5b63d4
                Copyright © © The Author(s) 2018
                License:

                Creative Commons Non Commercial CC BY-NC: This article is distributed under the terms of the Creative Commons Attribution-NonCommercial 4.0 License ( http://www.creativecommons.org/licenses/by-nc/4.0/) which permits non-commercial use, reproduction and distribution of the work without further permission provided the original work is attributed as specified on the SAGE and Open Access pages ( https://us.sagepub.com/en-us/nam/open-access-at-sage).

                History
                Date received : 26 December 2017
                Date accepted : 24 February 2018
                Categories
                Subject: Case Report
                Custom metadata
                cover-date April-June 2018

                ScienceOpen disciplines: Respiratory medicine
                Keywords: pulmonary artery aneurysm,large vessel vasculitis,isolated pulmonary vasculitis
                Data availability:
                ScienceOpen disciplines: Respiratory medicine
                Keywords: pulmonary artery aneurysm, large vessel vasculitis, isolated pulmonary vasculitis

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