Anatomical Classification and Posttreatment Remodeling Characteristics to Guide Management and Follow-Up of Neonates and Infants With Coronary Artery Fistula: A Multicenter Study From the Coronary Artery Fistula Registry

Author:

Gowda Srinath T.1ORCID,Latson Larry2ORCID,Sivakumar Kothandam3ORCID,Hiremath Gurumurthy4ORCID,Crystal Matthew5,Law Mark6ORCID,Shahanavaz Shabana7ORCID,Asnes Jeremy8ORCID,Veeram Reddy Surendranath9,Kobayashi Daisuke10ORCID,Alwi Mazeni11,Ichida Fukiko12ORCID,Hirono Keiichi12,Tahara Masahiro13ORCID,Takeda Atsuhito14ORCID,Minami Takaomi15,Kutty Shelby16ORCID,Nugent Alan W.17ORCID,Forbes Thomas10,Prieto Lourdes R.18ORCID,Qureshi Athar M.1ORCID

Affiliation:

1. Department of Pediatrics, Lillie Frank Abercrombie Section of Cardiology, Texas Children’s Hospital, Baylor College of Medicine, Houston (S.T.G., A.M.Q.).

2. Pediatric Cardiology, Joe DiMaggio Children’s Hospital, Hollywood, FL (L.L.).

3. Pediatric Cardiology, The Madras Medical Mission, Chennai, India (K.S.).

4. Department of Pediatrics, University of Minnesota, Masonic Children’s Hospital, Minneapolis (G.H.).

5. Pediatric Cardiology, Irving Medical Center, Columbia University, New York, NY (M.C.).

6. Pediatric Cardiology, Children’s of Alabama, University of Alabama at Birmingham (M.L.).

7. Pediatric Cardiology, Cincinnati Children’s Hospital, OH (S.S.).

8. Pediatric Cardiology, Yale New Haven Children’s Hospital, CT (J.A.).

9. Pediatric Cardiology, UT Southwestern Medical Center, Dallas, TX (S.V.R.).

10. Pediatric Cardiology, Children’s Hospital of Michigan, Detroit, MI (D.K., T.F.).

11. Pediatric Cardiology, Institut Jantung Negara, Kuala Lumpur, Malaysia (M.A.).

12. Department of Pediatrics, University of Toyoma, Japan (F.I., K.H.).

13. Department of Pediatrics, Tsuchiya General Hospital, Hiroshima, Japan (M.T.).

14. Department of Pediatrics, Hokkaido University Hospital, Japan (A.T.).

15. Department of Pediatrics, Jichi Medical University, Tochigi, Japan (T.M.).

16. Pediatric Cardiology, Helen B. Taussig Heart Center, The Johns Hopkins Hospital, Baltimore, MD (S.K.).

17. Pediatric Cardiology, Ann & Robert H. Lurie Children’s Hospital of Chicago, IL (A.W.N.).

18. Pediatric Cardiology, Nicklaus Children’s Hospital, Miami, FL (L.R.P.).

Abstract

Background: Coronary artery fistulas (CAFs) presenting in infancy are rare, and data regarding postclosure sequelae and follow-up are limited. Methods: A retrospective review of all the neonates and infants (<1 year) was conducted from the CAF registry for CAF treatment. The CAF type (proximal or distal), size, treatment method, and follow-up angiography were reviewed to assess outcomes and coronary remodeling. Results: Forty-eight patients were included from 20 centers. Of these, 30 were proximal and 18 had distal CAF; 39 were large, 7 medium, and 2 had small CAF. The median age and weight was 0.16 years (0.01–1) and 4.2 kg (1.7–10.6). Heart failure was noted in 28 of 48 (58%) patients. Transcatheter closure was performed in 24, surgical closure in 18, and 6 were observed medically. Procedural success was 92% and 94 % for transcatheter closure and surgical closure, respectively. Follow-up data were obtained in 34 of 48 (70%) at a median of 2.9 (0.1–18) years. Angiography to assess remodeling was available in 20 of 48 (41%). I. Optimal remodeling (n=10, 7 proximal and 3 distal CAF). II. Suboptimal remodeling (n=7) included (A) symptomatic coronary thrombosis (n=2, distal CAF), (B) asymptomatic coronary thrombosis (n=3, 1 proximal and 2 distal CAF), and (C) partial thrombosis with residual cul-de-sac (n=1, proximal CAF) and vessel irregularity with stenosis (n=1, distal CAF). Finally, (III) persistent coronary artery dilation (n=4). Antiplatelets and anticoagulation were used in 31 and 7 patients post-closure, respectively. Overall, 7 of 10 (70%) with proximal CAF had optimal remodeling, but 5 of 11 (45%) with distal CAF had suboptimal remodeling. Only 1 of 7 patients with suboptimal remodeling were on anticoagulation. Conclusions: Neonates/infants with hemodynamically significant CAF can be treated by transcatheter or surgical closure with excellent procedural success. Patients with distal CAF are at higher risk for suboptimal remodeling. Postclosure anticoagulation and follow-up coronary anatomic evaluation are warranted.

Publisher

Ovid Technologies (Wolters Kluwer Health)

Subject

Cardiology and Cardiovascular Medicine

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1. Surgical Management of Coronary Artery Fistulas in Children;Journal of Chest Surgery;2024-01-05

2. Diagnosis and Management of Congenital Coronary Artery Fistulas in Infants and Children;Current Cardiology Reports;2023-12

3. Coronary Artery Fistula: A Diagnostic Dilemma;Interventional Cardiology: Reviews, Research, Resources;2023-11-23

4. Surgical Repair of a Left Main Coronary Artery to Right Ventricle Fistula in a Neonate;World Journal for Pediatric and Congenital Heart Surgery;2023-10-26

5. Transcatheter Closure of a Coronary Artery Fistula in a Neonate with Tetralogy of Fallot;Pediatric Cardiology;2023-03-13

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