scholarly journals Late adult presentation of ALCAPA syndrome: need for a new clinical classification? A case report and literature overview

Author(s):  
Eno-Martin Lotman ◽  
Külliki Karu ◽  
Mehis Mikkel ◽  
Märt Elmet

Abstract Background Anomalous origin of the left main coronary artery from the pulmonary artery (ALCAPA) is a very uncommon congenital coronary artery anomaly, most commonly presenting in early infancy. Late adult presentation of ALCAPA syndrome is extremely rare. Case summary We present a case of a 76-year-old patient with first presentation of ALCAPA. The coronary anomaly was first diagnosed during elective coronary angiography. The case was discussed at the Heart Team meeting and as the patient was asymptomatic, had good coronary collateral circulation, a medical treatment strategy was chosen and the patient was discharged in a good physical condition. During 3 years of follow-up, the patient has experienced no cardiovascular complications. Discussion We hereby also discuss briefly the clinical presentation, epidemiology, diagnostics and treatment options for adults with newly diagnosed ALCAPA and discuss the need for a new clinical classification. Only a few cases have been published of septuagenarians or octogenarians with first presentation of ALCAPA. To our knowledge, the patient presented in our case was one of the least symptomatic patients during her eight decades of life.

2015 ◽  
Vol 2015 ◽  
pp. 1-4
Author(s):  
Nathalie Jeanne Magioli Bravo-Valenzuela ◽  
Guilherme Ricardo Nunes Silva

We report a case of anomalous left coronary artery from the pulmonary artery (ALCAPA) or Bland-White-Garland syndrome, present the challenges of performing a differential diagnosis, and discuss the treatment of the syndrome. Although ALCAPA is a rare congenital heart disease, it is one of the most common causes of myocardial ischemia in childhood and presents a diagnostic challenge. A four-year-old girl was referred to a pediatric cardiologist for evaluation of mitral valve regurgitation murmur and heart failure. The transthoracic echocardiogram demonstrated the left coronary artery (LCA) not arising from the aorta, presence of coronary collateral circulation, and moderate mitral valve regurgitation. ALCAPA was confirmed using angiotomography. The LCA was surgically reimplanted into the aorta. After 3 years of postoperative follow-up, the patient developed an LCA aneurysm. Diagnosis of cardiac ischemia in childhood remains a challenge, and careful evaluation of coronary arteries on the echocardiogram is an important tool. In this report, we present a case of ALCAPA with an uncommon postoperative outcome.


2019 ◽  
Vol 15 (3) ◽  
pp. 335-342 ◽  
Author(s):  
S. Yu. Martsevich ◽  
Yu. V. Lukina ◽  
N. P. Kutishenko ◽  
V. P. Voronina ◽  
N. A. Dmitrieva ◽  
...  

Aim. To assess the influence of addition of nicorandil on long-term cardiovascular complications in patients with chronic coronary artery disease (CAD) in the framework of a prospective observational study.Material and methods. Prospective observational multicenter NIKEA study included 590 patients with stable CAD. All patients were recommended to add nicorandil to their baseline therapy. After 21 months, 547 telephone contacts were made, the life status of 524 people was specified, 23 patients were lost to follow-up. The following complications were registered: death from any cause; non-fatal myocardial infarction (MI); non-fatal stroke; urgent myocardial revascularization; hospitalization due to deterioration of CAD, chronic heart failure (CHF) or atrial fibrillation (AF). In 479 patients, a telephone survey was performed to identify adherence (persistence) to nicorandil– i.e. to verify the continuing the treatment for the prescribed duration.Results. During the follow-up period, 15 deaths were recorded, the causes of which were as follows: 3 CAD (2 MI, 1 left ventricular aneurysm), 1 pulmonary embolism, 2 CHF; 1 stroke, 1 oncology, 1 bilateral pneumonia. In the remaining 6 cases, the causes of death were not known. Cardiovascular (CV) complications were MI (8 cases in 7 patients, 1 patient had 2 MI in the follow-up period, 3 patients from this group died), stroke in 6 patients (7 cases, 1 patient had 2 strokes, 1 patient died). Urgent percutaneous coronary intervention was performed in 3 patients (in 2 patients due to acute MI, in 1 patient due to unstable angina). During the observation period, 21 people were urgently hospitalized due to deterioration of clinical condition. According to the results of a medical survey, by the end of the follow-up period, 237 people were not adherent to prescribed nicorandil, and 242 patients took the recommended medication (adherent patients). The main cardiovascular complications and death from any cause (р<0.001) as well as the number of urgent hospitalizations (р=0.017) were significantly more common in non-adherent to nicorandil patients in comparison with adherent patients.Conclusion. The overall rate of CAD complications in NIKEA study was typical for patients with stable CAD receiving contemporary medical therapy. Addition of nicorandil significantly reduced the rate of CV complications in adherent patients in comparison with non-adherent patients. 


2018 ◽  
Vol 2018 ◽  
pp. 1-4
Author(s):  
Shantanu Patil ◽  
Mahek Shah ◽  
Brijesh Patel ◽  
Lohit Garg ◽  
Larry Jacobs ◽  
...  

Anomalous origin of the left main coronary artery from the pulmonary artery (ALCAPA) is a rare congenital coronary anomaly with high mortality. It is associated with cardiovascular complications and is usually diagnosed soon after birth. Those who survive into adulthood can present with signs of myocardial infarction, heart failure, mitral regurgitation, severe pulmonary hypertension, or sudden cardiac death. We present a 53-year-old female presenting with atrial fibrillation and found to have an incidental diagnosis of ALCAPA who refused surgical correction. We also review the epidemiology, diagnosis, age-based clinical presentations, and treatment options for ALCAPA.


Author(s):  
Marija Bjelobrk ◽  
Slobodan Dodic ◽  
Slobodan Dodic ◽  
Tatjana Miljkovic ◽  
Golub Samardzija ◽  
...  

The most common cause of coronary artery aneurysms is atherosclerosis, which is associated with over 50% of all aneurysms diagnosed in adults. Although patients can be asymptomatic throughout their lives, giant coronary artery aneurysms can manifest themselves as myocardial infarction, aneurysmal rupture, and sudden cardiac death as well. Herein, we describe an asymptomatic patient with numerous risk factors and a positive cardiopulmonary exercise test who was admitted to the cardiology clinic for coronary angiography. A giant coronary artery aneurysm (3.0×2.0 cm in diameter) in the left anterior descending coronary artery and significant stenosis in both left and right coronary arteries were found. After discussing possible treatment options, the hospital’s heart team recommended the surgical resection of the aneurysm and double coronary artery bypass graft. Four years after the cardiac surgery, at the time of writing the current manuscript, the patient is still in good condition and with no symptoms.


Author(s):  
Panupong Pota ◽  
Pannipa Suwannasom ◽  
Surin Woragidpoonpol ◽  
Tanop Srisuwan

Abstract Background IgG4-related disease (IgG4-RD) is a chronic fibroinflammatory condition with multiple-organ involvement. Rupture of coronary artery aneurysms (CAAs) in IgG4-RD is rare. Case summary A 65-year-old man with IgG4-RD has suffered from recurrent episodes of arterial aneurysms since 2003. He presented with chest pain and hypotension caused by localized cardiac tamponade at right ventricle free wall due to the rupture of coronary artery aneurysm(CAA) of left anterior descending artery (LAD). An urgent LAD aneurysm repaired with bovine pericardium and obliterated aneurysmal sac with cryo-acrylate glue was done together with coronary artery bypass grafting (CABG) using saphenous vein graft(SVG) to LAD and SVG to posterior descending artery. Three-month after surgery, the follow-up coronary computed tomography angiography (CCTA) revealed a growing in size of LAD and the second obtuse marginal(OM) branch aneurysm. Heart team discussion agreed to schedule the patient for double coil embolization to LAD and second OM aneurysm under intravascular ultrasound guidance. Both aneurysms were successfully obliterated with vascular coils. Two-week follow-up coronary angiogram showed complete occlusion of LAD aneurysm and near occlusion of the second OM branch aneurysm. Discussion CAA rupture is a life-threatening condition that required prompt detection and treatments. In IgG4-RD patients, acute cardiac tamponade suggesting the rupture of CAA. Coil embolization is an alternative treatment in patients who suffered from recurrent CAA after surgical repair. Serial CCTA is important for early detection of aneurysm in IgG4-RD patients who had vascular involvement.


2006 ◽  
Vol 55 (5) ◽  
pp. 451
Author(s):  
Seung Ho Joo ◽  
Byoung Wook Choi ◽  
Jae Seung Seo ◽  
Young Jin Kim ◽  
Tae Hoon Kim ◽  
...  

2012 ◽  
Vol 15 (3) ◽  
pp. 136 ◽  
Author(s):  
Shahzad G. Raja ◽  
Kareem Salhiyyah ◽  
Manoraj Navaratnarajah ◽  
Muhammad Umar Rafiq ◽  
Jeremy Felderhof ◽  
...  

<p><b>Objectives:</b> Despite increasing recognition that off-pump coronary artery bypass surgery and sequential grafting strategy individually are associated with improved outcomes, concerns persist regarding the safety and efficacy of combining these 2 techniques. We compared in-hospital and midterm outcomes for off-pump multivessel sequential and conventional coronary artery bypass grafting.</p><p><b>Methods:</b> From September 1998 to September 2008, 689 consecutive patients received off-pump multivessel sequential coronary artery bypass grafting performed by a single surgeon. These patients were propensity matched to 689 patients who underwent off-pump coronary artery bypass grafting without sequential anastomoses. A retrospective analysis of prospectively collected perioperative data was performed. In addition, medical notes and charts of all the study patients were reviewed. The mean duration of follow-up was 5.1 � 2.0 years.</p><p><b>Results:</b> The major in-hospital clinical outcomes in the sequential and control groups were found to be similar. After adjusting for clinical covariates, sequential grafting was not an independent predictor of in-hospital adverse events (odds ratio [OR], 1.18; 95% confidence interval [CI], 0.86-1.50; <i>P</i> = .31), medium-term mortality (hazard ratio [HR], 1.26; 95% CI, 1.06-1.32; <i>P</i> = .92), and readmission to hospital (HR, 1.12; 95% CI, 0.96-1.20; <i>P</i> = .80). Sequential grafting was an independent predictor of receiving more than 3 distal anastomoses (OR, 7.46; 95% CI, 4.27-11.45; <i>P</i> < .0001). Risk-adjusted survival was 89% for sequential grafting patients and 88% for conventional grafting patients (<i>P</i> = .96) during the medium-term follow-up.</p><p><b>Conclusion:</b> Our analysis confirms the short- and midterm safety and efficacy of off-pump sequential coronary artery bypass grafting.</p>


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