Ostial plication: a rarely reported cause of sudden death

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Abstract

We report a rare case of ostial plication as a potential cause of sudden death. Very few reports and images are available in the specialized literature regarding this anomaly. Ostial plication may be a source of sudden death or cause of death when no other significant autopsy findings are present.

Ostial plication is a congenital severe obstruction/occlusion of the right or left ostium. Plication of the aortic wall leads to a "valve-like" ridge that can act as a door blocking inflow during diastolic filling, with consequent ischemia and a potentially life-threatening arrhythmia. The true incidence of this condition and its relationship to sudden death have not been reported in the literature. We believe that this case will be useful to autopsy pathologists in detecting this infrequent anomaly, thus providing a more accurate estimation of its incidence.

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Most cited references 4

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Coronary anomalies: incidence, pathophysiology, and clinical relevance.

J Velasco, Scott Flamm, Paolo Angelini (2002)

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Anomalous origin of coronary arteries and risk of sudden death: a study based on an autopsy population of congenital heart disease.

C Frescura, L Daliento, G Thiene … (1998)

Coronary arteries anomalies may be part of complex congenital malformations of the heart or be an isolated defect. In our anatomic collection of congenital heart disease, an isolated anomalous origin of coronary arteries was observed in 27 of 1,200 specimens (2.2%): left coronary artery from pulmonary trunk in five, origin from the wrong aortic sinus in 12 (both right and left coronary artery from the right sinus in four and from the left sinus in seven, left coronary artery from the posterior sinus in one), left circumflex branch from right aortic sinus or from very proximal right coronary artery in three, high takeoff of right coronary artery in three, stenosis of the coronary ostia attributable to valvelike ridge in four. In 16 (59%) patients (12 males and 4 females, age ranging from 2 months to 53 years; median, 14), the final outcome was sudden death; it occurred in all cases of left coronary artery origin from right aortic sinus, in 43% of right coronary artery origin from left aortic sinus, and in 40% of the left coronary artery from the pulmonary trunk. Sudden death was precipitated by effort in eight (50%) and was the first manifestation of the disease in eight (50%); previous symptoms consisted of recurrent syncope in four, palpitations in three, and chest pain in one. Five patients who died suddenly during effort were athletes. In conclusion, (1) more than half of our postmortem cases with anomalous origin of coronary arteries died suddenly, (2) all but two patients with sudden death had anomalous coronary artery origin from the aorta itself, (3) the fatal event was frequently precipitated by effort, (4) palpitations, syncope, and ventricular arrhythmias were the only prodromic symptoms and signs. Recognition during life of these coronary anomalies, by the use of noninvasive procedures, is mandatory to prevent the risk of sudden death and to plan surgical correction if clinically indicated.

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Acute takeoffs of the coronary arteries along the aortic wall and congenital coronary ostial valve-like ridges: association with sudden death.

Renu Virmani, Patrick K.C. Chun, Robert E. Goldstein … (1984)

Congenital coronary artery anomalies have been associated with sudden death. Twenty-two patients who were victims of sudden death (mean age 46) and who had no significant anatomic cause of death were examined at autopsy and compared with 19 patients who died of known causes (control group). The hearts of these 41 patients were examined for abnormalities of acute angle takeoff of the coronary artery and presence of ostial valve-like ridges. Of the 22 patients who died suddenly, 13 (59%) had acute angle takeoff of the coronary artery and 9 (41%) had ostial valve-like ridges. Of the 19 control subjects, 4 (21%) had acute angle takeoff and only 2 (11%) had an ostial valve-like ridge. The difference was statistically significant (p = 0.015 and 0.031, respectively). It is suggested that aortic root dilation may compress coronary arteries with acute angle takeoff and that ostial valve-like ridges may act as occlusion valves. Thus, either may cause acute obstruction of the proximal coronary artery and lead to sudden death. A very lethal combination for sudden death would be the presence of severe coronary artery disease, an acute angle takeoff and an ostial valve-like ridge.

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Journal

Journal ID (nlm-ta): Diagn Pathol

Title: Diagnostic Pathology

Publisher: BioMed Central

ISSN (Electronic): 1746-1596

Publication date Collection: 2010

Publication date (Electronic): 25 February 2010

Volume: 5

Page: 15

Affiliations

[1 ]Institute of Legal Medicine, Catholic University, School of Medicine, Rome, Italy

[2 ]Institute of Pathologic Anatomy, Catholic University, School of Medicine Rome, Italy

Article

Publisher ID: 1746-1596-5-15

DOI: 10.1186/1746-1596-5-15

PMC ID: 2841130

PubMed ID: 20184724

SO-VID: d507ac19-8645-4244-a2f0-6a2dc96e4c0d

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This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Ostial plication: a rarely reported cause of sudden death

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Karger: Ophthalmology

Most cited references 4

Coronary anomalies: incidence, pathophysiology, and clinical relevance.

Anomalous origin of coronary arteries and risk of sudden death: a study based on an autopsy population of congenital heart disease.

Acute takeoffs of the coronary arteries along the aortic wall and congenital coronary ostial valve-like ridges: association with sudden death.

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