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

Gowda ST 1 , Latson L 2 , Sivakumar K 3 , Hiremath G 4 , Crystal M 5 , Law M 6 Show all authors , Shahanavaz S 7 , Asnes J 8 , Veeram Reddy S 9 , Kobayashi D 10 , Alwi M 11 , Ichida F 12 , Hirono K 12 , Tahara M 13 , Takeda A 14 , Minami T 15 , Kutty S 16 , Nugent AW 17 , Forbes T 10 , Prieto LR 18 , Qureshi AM 1

Affiliations 

  • 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.)
Circ Cardiovasc Interv, 2021 12;14(12):e009750.
PMID: 34903033 DOI: 10.1161/CIRCINTERVENTIONS.120.009750

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.

* Title and MeSH Headings from MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine.

MeSH terms
Similar publications