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Sinha SK, Khanra D, Jha MJ, et al. Unusual Survival of Anomalous Left Coronary Artery From the Pulmonary Artery With Severe Rheumatic Mitral Stenosis in Septuagenarian Women: Foes Becoming Friends?. J Clin Med Res. 2016;8(10):760-3doi: 10.14740/jocmr2674w.
Sinha, S. K., Khanra, D., Jha, M. J., Singh, K., Razi, M., Goel, A., Mishra, V., Asif, M., Sachan, M., Afdaali, N., Kumar, A., Thakur, R., Krishna, V., Pandey, U., & Varma, C. M. (2016). Unusual Survival of Anomalous Left Coronary Artery From the Pulmonary Artery With Severe Rheumatic Mitral Stenosis in Septuagenarian Women: Foes Becoming Friends?. Journal of clinical medicine research, 8(10), 760-3. https://doi.org/10.14740/jocmr2674w
Sinha, Santosh Kumar, et al. "Unusual Survival of Anomalous Left Coronary Artery From the Pulmonary Artery With Severe Rheumatic Mitral Stenosis in Septuagenarian Women: Foes Becoming Friends?." Journal of clinical medicine research vol. 8,10 (2016): 760-3. doi: https://doi.org/10.14740/jocmr2674w
Sinha SK, Khanra D, Jha MJ, Singh K, Razi M, Goel A, Mishra V, Asif M, Sachan M, Afdaali N, Kumar A, Thakur R, Krishna V, Pandey U, Varma CM. Unusual Survival of Anomalous Left Coronary Artery From the Pulmonary Artery With Severe Rheumatic Mitral Stenosis in Septuagenarian Women: Foes Becoming Friends?. J Clin Med Res. 2016 Oct;8(10):760-3. doi: 10.14740/jocmr2674w. Epub 2016 Aug 30. PMID: 27635184; PMCID: PMC5012248.
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J Clin Med Res. 2016 Oct;8(10):760-3. doi: 10.14740/jocmr2674w. Epub 2016 Aug 30.

Unusual Survival of Anomalous Left Coronary Artery From the Pulmonary Artery With Severe Rheumatic Mitral Stenosis in Septuagenarian Women: Foes Becoming Friends?.

Journal of clinical medicine research

Santosh Kumar Sinha, Dibbendhu Khanra, Mukesh Jitendra Jha, Karandeep Singh, Mahamdulla Razi, Amit Goel, Vikas Mishra, Mohammad Asif, Mohit Sachan, Nasar Afdaali, Ashutosh Kumar, Ramesh Thakur, Vinay Krishna, Umeshwar Pandey, Chandra Mohan Varma

Affiliations

  1. Department of Cardiology, LPS Institute of Cardiology, G.S.V.M. Medical College, Kanpur 208002, Uttar Pradesh, India.

PMID: 27635184 PMCID: PMC5012248 DOI: 10.14740/jocmr2674w

Abstract

ALCAPA syndrome (anomalous origin of the left coronary artery from the pulmonary artery) is a rare disease but lethal with clinical expression from myocardial infarction, congestive heart failure to death during early infancy and unusual survival to adulthood. We report a 73-year-old woman with ALCAPA who presented with exertional dyspnea (NYHA functional class II) over past 2 years. Physical examination revealed soft S, long mid diastolic rumbling murmur and apical pan-systolic murmur. Electrocardiography displayed biatrial enlargement and poor R progression and normal sinus rhythm. Echocardiography established calcified severe mitral stenosis (MS), presence of continuous flow entering the pulmonary trunk, turbulent continuous flow in inter-ventricular septum with left to right shunt in contrast echocardiography and normal systolic function. Coronary angiogram showed absence of left coronary artery (LCA) originating from aorta, dilated and tortuous right coronary artery (RCA) and abundant Rentrop grade 3 intercoronary collateral communicating with LCA originating from pulmonary trunk which was also confirmed on coronary CT angiogram thus establishing diagnosis of ALCAPA. It is exceedingly rare to be associated with severe MS. However, such a long survival in our patient can be explained by the severe pulmonary arterial hypertension which may be contributing to lesser coronary steal.

Keywords: ALCAPA syndrome; Contrast echocardiography; Coronary CT; Mitral stenosis; NYHA functional class; Rentrop collateral

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