Skip to main content
SpringerLink
Log in

Heart Rate Induced Dynamic Coronary Ischemia Secondary to Aortic Valve Insufficiency from a Diminutive Aortic Valve Leaflet: Diagnosis and Repair

  • Case Report
  • Published:
Pediatric Cardiology Aims and scope Submit manuscript

Abstract

Hemodynamically significant congenital obstruction of the coronary arteries is a rare phenomenon. The potential etiology of these obstructions is variable including coronary os stenosis, occlusive fibrous webs, or a dysplastic aortic valve leaflet resulting in obstruction. In infants, diagnosis of these coronary lesions is often difficult due to the small anatomic size and limitations of imaging modalities in the setting of the underlying rapid heart rates. We present a case of an infant presenting in heart failure secondary to dynamic coronary artery insufficiency from a diminutive aortic valve leaflet and aortic sinus and variable levels of aortic valve insufficiency based on heart rate.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4

Similar content being viewed by others

References

  1. Shen TC, Chou HH, Tsai KT (2012) Ectopic and atretic ostium of left main coronary artery: from interleaflet triangle between left and noncoronary cusps. Texas Hear Inst J 39:60–62

    Google Scholar 

  2. Angelini P, Velasco JA, Flamm S (2002) Coronary anomalies: incidence, pathophysiology, and clinical relevance. Circulation 105:2449–2454. https://doi.org/10.1161/01.CIR.0000016175.49835.57

    Article  PubMed  Google Scholar 

  3. Shriki JE, Shinbane JS, Rashid MA et al (2012) Identifying, characterizing, and classifying congenital anomalies of the coronary arteries. Radiographics 32:453–468. https://doi.org/10.1148/rg.322115097

    Article  PubMed  Google Scholar 

  4. Angelini P (2012) Congenital coronary artery ostial disease: a spectrum of anatomic variants with different pathophysiologies and prognoses. Texas Hear Inst J 39:55–59

    Google Scholar 

  5. Likes ML, Silverman NH, Albers EL et al (2015) Near-fatal neonatal coronary ischaemia associated with intermittent aortic regurgitation: successful surgical treatment. Cardiol Young 25:1531–1535. https://doi.org/10.1017/S1047951115002024

    Article  PubMed  Google Scholar 

  6. Martin MH, Perry SB, Prochazka JV et al (2010) Surgical management of life threatening events caused by intermittent aortic insufficiency in a native valve: case report. J Cardiothorac Surg 5:94. https://doi.org/10.1186/1749-8090-5-94

    Article  PubMed  PubMed Central  Google Scholar 

  7. Masaki N, Onoki T, Ozawa A et al (2020) Rare case of neonatal cardiac ischemia due to intermittent aortic insufficiency. Ann Thorac Surg 110:e295–e297. https://doi.org/10.1016/j.athoracsur.2020.01.080

    Article  PubMed  Google Scholar 

  8. Harada T, Fukae K, Ando Y (2017) Surgical repair of ostial obstruction of the coronary artery for quadricuspid aortic valve in an infant. Interact Cardiovasc Thorac Surg 24:634–635. https://doi.org/10.1093/icvts/ivw438

    Article  PubMed  Google Scholar 

  9. Line DE, Babb JD, Pierce WS (1979) Congenital aortic valve anomaly. Aortic regurgitation with left coronary artery isolation. J Thorac Cardiovasc Surg 77:533–535. https://doi.org/10.1016/s0022-5223(19)38257-1

    Article  CAS  PubMed  Google Scholar 

  10. Gibson R, Nihill M, Mullins C, Cooley D, Sandiford FMD (1981) Congenital coronary artery anomalies. Circulation 64:857–861. https://doi.org/10.1007/978-1-84800-064-3_14

    Article  CAS  PubMed  Google Scholar 

  11. Benassi F, DiBenedetto G, Labia C, Stefanelli G (2012) Left coronary ostium isolation and aortic valve dysplasia in a young boy: a case report. J Heart Valve Dis 21:135–137

    PubMed  Google Scholar 

  12. Aksoy S, Cam N, Guney MR et al (2012) Myocardial ischemia in severe aortic regurgitation despite angiographically normal coronary arteries. Tohoku J Exp Med 226:69–73. https://doi.org/10.1620/tjem.226.69

    Article  PubMed  Google Scholar 

Download references

Funding

No financial assistance was received to support this study.

Author information

Authors and Affiliations

  1. Division of Cardiology, Department of Pediatrics, Oregon Health & Science University, 700 SW Campus Dr, Portland, OR, 97239, USA

    Mohammad Alnoor & Erin Madriago

  2. Division of Cardiothoracic Surgery, Department of Surgery, Oregon Health & Science University, Portland, OR, USA

    Ashok Muralidaran

Authors
  1. Mohammad Alnoor
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Ashok Muralidaran
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Erin Madriago
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Mohammad Alnoor.

Ethics declarations

Conflict of interest

All authors declare that he/she has no conflict of interest.

Ethical Approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.

Informed Consent

Informed consent was obtained from all individual participants included in the study.

Additional information

Publisher's Note

Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Alnoor, M., Muralidaran, A. & Madriago, E. Heart Rate Induced Dynamic Coronary Ischemia Secondary to Aortic Valve Insufficiency from a Diminutive Aortic Valve Leaflet: Diagnosis and Repair. Pediatr Cardiol 43, 691–694 (2022). https://doi.org/10.1007/s00246-021-02772-1

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00246-021-02772-1

Keywords

Search

Navigation