Congenital coronary artery-to-pulmonary fistula with giant aneurysmal dilatation and thrombus formation: a case report and review of literature

Abstract Background Coronary artery-to-pulmonary artery fistula is a rare disorder characterized by abnormal vascular communication between the coronary artery and pulmonary artery. While most patients remain asymptomatic, some might exhibit symptoms of myocardial ischemia, congestive heart failure, or even sudden cardiac death if coronary aneurysm, thrombosis, infective carditis, or other congenital cardiac defects coexist. Case presentation We present a 66-year-old male complaining of angina pectoris with a history of hypertension and active smoking. He was diagnosed with a coronary aneurysm based on coronary computed tomography angiography. We subsequently identified a coronary artery-to-pulmonary artery fistula with giant aneurysmal dilation on coronary angiography. Ultimately we conducted surgery ligation and aneurysmorrhaphy. During surgery, we discovered newly formed thrombus within the aneurysmal cavity. Histological analysis of the aneurysmal wall supported the diagnosis of the congenital disorder. Our patient was successfully discharged and remained asymptomatic at two months of follow-up. Conclusion We presented a rare and complex combination of congenital coronary artery-to pulmonary artery fistula, giant coronary aneurysmal dilatation, and thrombosis through multi-modality evaluations.

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Left main coronary artery-to-pulmonary artery fistula with severe aneurysmal dilatation

The Annals of Thoracic Surgery ◽

10.1016/j.athoracsur.2003.05.006 ◽

2004 ◽

Vol 77 (3) ◽

pp. 1081-1083 ◽

Cited By ~ 22

Author(s):

Kimberly L Gandy ◽

Abdallah G Rebeiz ◽

Andrew Wang ◽

James J Jaggers

Keyword(s):

Coronary Artery ◽

Pulmonary Artery ◽

Left Main Coronary Artery ◽

Left Main ◽

Aneurysmal Dilatation ◽

Pulmonary Artery Fistula

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Post-coronary artery bypass grafting left internal mammary artery to pulmonary artery fistula: a 6 year follow-up following successful surgical division

European Journal of Cardio-Thoracic Surgery ◽

10.1016/s1010-7940(01)00999-x ◽

2001 ◽

Vol 20 (6) ◽

pp. 1258-1260 ◽

Cited By ~ 10

Author(s):

G. Musleh ◽

Anjum Jalal ◽

A.K. Deiraniya

Keyword(s):

Coronary Artery ◽

Coronary Artery Bypass Grafting ◽

Pulmonary Artery ◽

Internal Mammary Artery ◽

Left Internal Mammary Artery ◽

Artery Bypass ◽

Bypass Grafting ◽

Internal Mammary ◽

Pulmonary Artery Fistula

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Abstract 163: Regressed Coronary Aneurysm after Kawasaki Disease: What are they hiding? An Optical Coherence Tomography (OCT) study

Circulation ◽

10.1161/circ.131.suppl_2.163 ◽

2015 ◽

Vol 131 (suppl_2) ◽

Author(s):

Audrey Dionne ◽

Anne Fournier ◽

Ragui Ibrahim ◽

Catherine Gebhard ◽

Nagib Dahdah

Keyword(s):

Coronary Artery ◽

Kawasaki Disease ◽

Intimal Hyperplasia ◽

Imaging Techniques ◽

Coronary Aneurysm ◽

History Of ◽

Oct Imaging ◽

Coronary Events ◽

Smooth Muscle Dysfunction

Background: Coronary artery aneurysms (CAA) are a serious complication of Kawasaki disease (KD). Regression of CAA occurs in 50% of the cases on follow up. Actual imaging techniques often described these segments as normal, whereas studies have shown significant endothelial and smooth muscle dysfunction. Method: KD patients scheduled for angiographic follow-up between June 2013 and August 2014 underwent OCT imaging. We compared coronary intimal changes in coronary artery segments with no history of CAA to segments with regressed CAA, and segments with persistent CAA. The intima was measured in sections adjacent to the CAA for the segments with persistent CAA, at the former CAA and adjacent sections for those who regressed, and at various corresponding sections for segments with no history of CAA. Results: OCT was performed on 18 patients at 12.4 ± 5.5 years. Overall 14/18 (77.7%) had a history of CAA. Of those, 7/14 (50.0%) had regressed CAA at time of OCT. Data was analyzed according to echocardiographic and angiographic progress of CAA segments. Accordingly, all 18/18 persistent CAA segments and 11/11 regressed CAA segments had significant intimal hyperplasia, compared to 1/13 with no history of segmental CAA (P<0.001). The intensity of intimal hyperplasia is displayed in Table 1. Conclusion: Despite normal angiographic features, regressed CAA segments displayed significant intimal hyperplasia, similarly to those with persistent CAA. These features may present a risk of adverse coronary events.

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Surgical Repair of Congenital Coronary Artery Fistula With Giant Aneurysm

The Heart Surgery Forum ◽

10.1532/hsf.2813 ◽

2020 ◽

Vol 23 (2) ◽

pp. E151-E153

Author(s):

Xiaoyong Li ◽

Laichun Song ◽

Huiqiong Guo ◽

Jing Jin ◽

Ming Xu

Keyword(s):

Coronary Artery ◽

Giant Aneurysm ◽

Coronary Aneurysm ◽

Coronary Artery Fistula ◽

Post Operation ◽

History Of ◽

Normal Blood Flow ◽

Flow Through ◽

Congenital Coronary Artery Fistula

A 67-year-old man with a 3-year history of dyspnea on exertion arrived to our institution with discontinuous palpitations and short breath. He was diagnosed with congenital coronary artery fistula (CAF). Angiography revealed 3 giant aneurysmal formations and coronary artery calcification. We report a case of successful repair of CAF with a giant coronary aneurysm by closing the orifice and resecting the aneurysm and reconstructing the left coronary artery. The surgical procedure included closure from within a vessel dilated by aneurysm and excision of the aneurysm. We were able to completely obliterate the fistula and preserve the normal blood flow through the coronary arteries post operation. The postoperative course was eventful, but the patient was discharged home. The patient was doing well at his 28-month follow-up visit.

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Outcomes of surgical repair of anomalous origin of the left coronary artery from the pulmonary artery in infants and children

Cardiology in the Young ◽

10.1017/s104795112100161x ◽

2021 ◽

pp. 1-6

Author(s):

Tong Feng ◽

Guo Zhangke ◽

Bai Song ◽

Fan Fan ◽

Zhen Jia ◽

...

Keyword(s):

Coronary Artery ◽

Mitral Regurgitation ◽

Ejection Fraction ◽

Pulmonary Artery ◽

Left Coronary Artery ◽

Surgical Repair ◽

Left Ventricular ◽

Anomalous Origin ◽

Dimension Index

Abstract Objectives: Anomalous origin of the left coronary artery from the pulmonary artery is associated with high mortality if not timely surgery. We reviewed our experience with anomalous origin of the left coronary artery from the pulmonary artery to assess the preoperative variables predictive of outcome and post-operative recovery of left ventricular function. Methods: A retrospective review was conducted and collected data from patients who underwent anomalous origin of the left coronary artery from the pulmonary artery repair at our institute from April 2005 to December 2019. Left ventricular function was assessed by ejection fraction and the left ventricular end-diastolic dimension index. The outcomes of reimplantation repair were analysed. Results: A total of 30 consecutive patients underwent anomalous origin of the left coronary artery from the pulmonary artery repair, with a median age of 14.7 months (range, 1.5–59.6 months), including 14 females (46.67%). Surgery was performed with direct coronary reimplantation in 12 patients (40%) and the coronary lengthening technique in 18 (60%). Twelve patients had concomitant mitral annuloplasty. There were two in-hospital deaths (6.67%), no patients required mechanical support, and no late deaths occurred. Follow-up echocardiograms demonstrated significant improvement between the post-operative time point and the last follow-up in ejection fraction (49.43%±19.92% vs 60.21%±8.27%, p < 0.01) and in moderate or more severe mitral regurgitation (19/30 vs 5/28, p < 0.01). The left ventricular end-diastolic dimension index decreased from 101.91 ± 23.07 to 65.06 ± 12.82 (p < 0.01). Conclusions: Surgical repair of anomalous origin of the left coronary artery from the pulmonary artery has good mid-term results with low mortality and reintervention rates. The coronary lengthening technique has good operability and leads to excellent cardiac recovery. The decision to concomitantly correct mitral regurgitation should be flexible and be based on the pathological changes of the mitral valve and the degree of mitral regurgitation.

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COILUMICA: coil embolization of a coronary artery to pulmonary artery fistula via novel dual lumen micro catheter technique

CVIR Endovascular ◽

10.1186/s42155-020-00177-4 ◽

2020 ◽

Vol 3 (1) ◽

Author(s):

Mithun Nambiar ◽

Julian Maingard ◽

Kenny Li ◽

Lee-Anne Slater ◽

Ronil V. Chandra ◽

...

Keyword(s):

Coronary Artery ◽

Pulmonary Artery ◽

Coil Embolization ◽

Coronary Artery Fistula ◽

Exertional Dyspnea ◽

Balloon Catheters ◽

Catheter Technique ◽

Patient Reported ◽

Pulmonary Artery Fistula ◽

Micro Catheter

Abstract Background Management of coronary artery fistula (CAF) is based on obliterating the fistula communication between the cardiac arteries and other thoracic vessels. Case presentation We describe the presentation of an 85-year-old female with progressive exertional dyspnea on a background of a long standing left anterior descending diagonal to pulmonary artery fistula. We utilized neuro-interventional techniques to perform coil embolization via use of a Scepter XC dual lumen micro catheter. Conclusions Dual lumen balloon catheters allow for super-selective artery interrogation, stability of balloon positioning, with less trauma to vessel architecture and accurate embolization. There were no complications and the patient reported improvement of symptoms on review.

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A peculiar case of coronary artery to pulmonary artery fistula presenting as ST-elevation myocardial infarction

International Journal of Cardiology ◽

10.1016/j.ijcard.2021.10.130 ◽

2021 ◽

Vol 345 ◽

pp. 45-46

Author(s):

N.A. Malek ◽

I.M. Amran ◽

M.R. Mohamed Rus ◽

N.S. Shahril

Keyword(s):

Myocardial Infarction ◽

Coronary Artery ◽

Pulmonary Artery ◽

St Elevation Myocardial Infarction ◽

St Elevation ◽

Pulmonary Artery Fistula ◽

Peculiar Case

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Left Lung and Pulmonary Artery Hypoplasia: A Rare Case of Hemoptysis

European Journal of Case Reports in Internal Medicine ◽

10.12890/2020_001490 ◽

2020 ◽

Author(s):

Guiomar Pinheiro ◽

Ana Margarida Alves ◽

Isabel Neves ◽

Teresa Sequeira

Keyword(s):

Pulmonary Hypertension ◽

Pulmonary Artery ◽

Pulmonary Hypoplasia ◽

Left Lung ◽

Left Pulmonary Artery ◽

Pulmonary Infections ◽

Rare Congenital Disorder ◽

History Of ◽

Pulmonary Artery Hypoplasia

Pulmonary hypoplasia or agenesis is a rare congenital disorder that results in lung underdevelopment. This disease is usually found in children but rarely encountered in adults. We describe the case of an 84-year-old woman diagnosed with a unilateral pulmonary hypoplasia presenting simultaneously with left pulmonary artery hypoplasia. Due to this condition, the patient had a lifelong history of pulmonary infections that resulted in several bronchiectases in the affected lung. Moreover, the pulmonary artery hypoplasia led to the development of pulmonary hypertension and collateral circulation causing hemoptysis, giving rise to the patient attending the emergency department. Although we were able to medically manage the hemoptysis, it can be fatal and require surgical intervention. Hence, an early diagnosis is essential so that appropriate follow-up and prompt prevention and treatment of complications, such as pulmonary infections, hemoptysis and pulmonary hypertension, are achieved.

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