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      It’s Not Always Occam's Razor – The Case of a Young Man with Subaortic Membrane with Superimposed Pulmonary Thromboembolism and Left Main Coronary Artery Disease

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          Abstract

          Subaortic membrane (SAM) is a discrete fibromuscular structure which causes left ventricular outflow tract obstruction and leads to the symptomatology of valvular aortic stenosis. It is known to be associated with other congenital cardiac defects in around 30% of cases. However, it has not been associated with chronic pulmonary thromboembolism in the past. We present a case of a middle-aged Pakistani man who presented with dyspnea and hemoptysis. He was found to have a SAM and severe pulmonary hypertension on transthoracic echocardiogram. A coronary angiogram revealed non-obstructive left main coronary artery disease. A computed tomography (CT) scan chest was done to evaluate the cause of severe pulmonary hypertension unexplained by SAM which revealed chronic pulmonary thromboembolism. Surgical resection was deferred due to high risk. Hence, he was kept on anticoagulation for pulmonary thromboembolism, and aspirin and a statin for non-obstructive coronary artery disease. Over the course of two months, his symptoms improved. This case highlights the importance of evaluating different causes of pulmonary hypertension in patients with SAM.

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          Most cited references 7

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          Fixed subaortic stenosis: anatomical spectrum and nature of progression.

          Retrospective echocardiographic review identified 58 consecutive infants and children with fixed subaortic stenosis. Mean (SD) age at diagnosis was 4.8 (3.6) years (range two days to 14.7 years), and diagnosis occurred in infancy in eight. Associated cardiac abnormalities were present in 41 (71%) whereas fixed subaortic stenosis was an isolated lesion in 17 (29%). Four types of fixed subaortic stenosis were identified: short segment (47 (81%)), long segment (7 (12%)), posterior displacement of the infundibular septum with additional discrete narrowing of the left ventricular outflow tract (3 (5%)), and redundant tissue arising from the membranous septum (1 (2%)). Echocardiographic studies had been performed before the diagnosis of fixed subaortic stenosis in nine patients, all with associated abnormalities. These were performed in infancy in each and showed a "normal" left ventricular outflow tract in six and posterior deviation of the infundibular septum in three. In 16 patients serial echocardiographic studies had been performed after the diagnosis of fixed subaortic stenosis but before surgery of the left ventricular outflow tract. Rapid evolution of short segment to long segment narrowing was seen in one patient, and tethering of the aortic valve or mitral valve developed in a further four patients. Aortic valve or mitral valve involvement was not seen before the age of three years but was common thereafter (10/40 patients, 25%). Fixed subaortic stenosis may be an "acquired" lesion with the potential for changes in form as well as progression in severity of left ventricular outflow tract obstruction.
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            Resection of subaortic stenosis; can a more aggressive approach be justified?

             A Parry,  J Kovalchin,  K Suda (1999)
            Discrete subaortic stenosis causes left ventricular outflow tract (LVOT) obstruction and often produces aortic regurgitation (AR) which alone may precipitate surgical intervention. Conventional resection relieves the obstruction, but the recurrence rate is high, and the AR is little changed as the thick fibrous membrane which extends onto the valve leaflets remains. We studied whether an aggressive surgical approach could reduce both the severity of AR and rate of recurrence of obstruction associated with discrete subaortic stenosis, and whether this aggressive approach could be justified.
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              Subaortic membrane: correlation of imaging with pathology

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                Author and article information

                Journal
                Cureus
                Cureus
                2168-8184
                Cureus
                Cureus (Palo Alto (CA) )
                2168-8184
                6 October 2019
                October 2019
                : 11
                : 10
                Affiliations
                [1 ] Cardiology, Aga Khan University Hospital, Karachi, PAK
                [2 ] Medicine, Aga Khan University Hospital, Karachi, PAK
                [3 ] Medicine, Aga Khan University Hospital, Karachi, PAK
                [4 ] Radiology, Aga Khan University Hospital, Karachi, PAK
                Author notes
                Article
                10.7759/cureus.5850
                6830847
                Copyright © 2019, Shahab et al.

                This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

                Categories
                Cardiology
                Cardiac/Thoracic/Vascular Surgery
                Anatomy

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