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      A case of ruptured sinus of Valsalva aneurysm and reversible flow-induced pulmonary hypertension

      case-report
      Author(s): , ,
      Publication date (Electronic):
      Journal: Pulmonary Circulation
      Publisher: SAGE Publications
      Keywords: congenital, echocardiography, right atrium, shunt

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          Abstract

          Pulmonary hypertension (PH) in adults with congenital heart disease (CHD) and significant systemic-to-pulmonary shunting is a significant cause of morbidity and mortality. Its pathophysiology is incompletely understood, but involves a flow-induced pulmonary arteriopathy characterized by endothelial cell dysfunction and vascular remodeling that alters pulmonary arterial vasoreactivity. There is a paucity of literature linking PH with left-to-right shunting due to ruptured sinus of Valsalva aneurysms (SOVA). We present a unique case of reversible, flow-associated PH due to a ruptured congenital right SOVA fistulizing into the right atrium (RA), with emphasis on non-invasive and invasive assessment of pulmonary hemodynamics before and after surgical intervention.

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          The role of increased pulmonary blood flow in pulmonary arterial hypertension.

          Erwin H J M Kemna, R. Van Der Leeuw, R. Schoemaker … (2005)
          Chronic increased pulmonary blood flow is considered a pre-requisite for the induction of advanced vascular lesions in pulmonary arterial hypertension in congenital heart defects. The aim of the present study was to characterise the effects of increased pulmonary flow induced by an aortocaval shunt in the monocrotaline rat model for pulmonary hypertension in terms of survival, haemodynamics, pathology and histology. Male Wistar rats were injected with monocrotaline followed by the creation of an abdominal aortocaval shunt. Animals were sacrificed when displaying symptoms of weight loss or dyspnoea, 4-5 weeks after the creation of the shunt. Echocardiography identified increased ventricular dimensions in shunted rats and right ventricular hypertrophy in monocrotaline-treated rats. At similar pulmonary artery pressures, shunted monocrotaline rats displayed higher morbidity and mortality, increased pulmonary-to-systemic artery pressure ratios and increased right ventricular hypertrophy compared with nonshunted monocrotaline rats. Histological assessment demonstrated increased number and diameter of pre-acinar pulmonary arteries. Intra-acinar vessel remodelling and occlusion occurred to a similar extent in shunted and nonshunted monocrotaline rats. In conclusion, increased pulmonary blood flow in monocrotaline-induced pulmonary hypertension is associated with increased morbidity, mortality, and unfavourable haemodynamic and cardiac effects. These effects could be attributed to more pronounced right heart failure rather than to altered intra-acinar pulmonary vessel remodelling.
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            Gene expression profile in flow-associated pulmonary arterial hypertension with neointimal lesions.

            R. Schoemaker, Frans Gerbens, Peter van Lent … (2010)
            Pulmonary arterial hypertension (PAH) is a pulmonary angioproliferative disease with high morbidity and mortality, characterized by a typical pattern of pulmonary vascular remodeling including neointimal lesions. In congenital heart disease, increased pulmonary blood flow has appeared to be a key mediator in the development of these characteristic lesions, but the molecular mechanisms underlying the pulmonary vascular lesions are largely unknown. We employed a rat model of flow-associated PAH, which induced specific pulmonary neointimal lesions. We identified gene expression profiles in rats specifically related to the addition of increased pulmonary blood flow to monocrotaline and the associated occurrence of neointimal lesions. Increased pulmonary blood flow induced the expression of the transcription factors activating transcription factor-3 (ATF3) and early growth response factor-1 (EGR-1), for which presence was confirmed in neointimal lesions. Monocrotaline alone induced increased numbers of activated mast cells and their products. We further identified molecular pathways that may be involved in treatment with the prostacyclin analog iloprost, a vasoactive compound with clinically beneficial effects in patients with PAH, which were similar to pathways described in samples from patient studies. These pathways, associated with the development of angioproliferative lesions as well as with the response to therapy in PAH, may provide new therapeutic targets.
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              Aneurysm of the sinus of Valsalva

              DA OTT, D Ott, D.A. Ott … (2006)
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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): 26 February 2018
                Publication date Collection: April 2018
                Volume: 8
                Issue: 2
                Electronic Location Identifier: 2045894018760656
                Affiliations
                [1-2045894018760656]Division of Cardiology, Department of Medicine, University of Western Ontario, London, ON, Canada
                Author notes
                [*]Ryan Davey, Division of Cardiology, Department of Medicine, University Hospital, London Health Sciences Centre, University of Western Ontario, 339 Windermere Road, London, ON N6A5A5, Canada. Email: ryan.davey@ 123456lhsc.on.ca
                Article
                Publisher ID: 10.1177_2045894018760656
                DOI: 10.1177/2045894018760656
                PMC ID: 5888825
                PubMed ID: 29480096
                SO-VID: 83d78bdb-5b59-4fcb-b18e-e49dbf4a36a3
                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 : 22 November 2017
                Date accepted : 30 January 2018
                Categories
                Subject: Case Report
                Custom metadata
                cover-date April-June 2018

                ScienceOpen disciplines: Respiratory medicine
                Keywords: congenital,echocardiography,right atrium,shunt
                Data availability:
                ScienceOpen disciplines: Respiratory medicine
                Keywords: congenital, echocardiography, right atrium, shunt

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