Multiple microvessels extending from the coronary arteries to the left ventricle in a middle aged female presenting with ischaemic chest pain: a case report

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Abstract

Possible ischaemic chest pain presentations are exceedingly common. Angiographic triage of clinical, electrocardiographic or biomarker positive presentations is increasingly feasible with the expansion of cardiac catheterization facilities. This management pattern often extends to problem patients with negative biomarker screens whose symptoms appear unstable. With invasive triage even very rare congenital or developmental coronary anomalies will be more frequently recognized although their relationship to ischaemia can be confounded by association. In this a case we report a woman with widespread direct coro-ventricular micro-channel formation across the heart and an ischaemic presentation, despite angiographically normal epicoronary vessels. This pattern, while very rare, needs to be recognized as one possible phenotype in this very common clinical presentation.

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Congenital variants in the structure or positioning of the native coronary arteries, or acquired coronary-cameral fistulas in the adult, are rare but well documented [1]. They are often defined in routine diagnostic and/or therapeutic coronary angiographic procedures following related or unrelated symptomatic presentation. Some may be linked to symptomatic ischaemia, with or without conventional atherosclerotic coronary arterial stenoses. In contrast to single arterio-venous or arterio-arterial fistulas, direct microfistulas between individual coronary arteries and the left ventricle are exceedingly rare [2]. We report a patient with multiple micro-channels extending from both coronary arteries to the left ventricle, who presented with acute chest pain typical of myocardial ischaemia.

Case presentation
The patient (Caucasian; female; 58 yr; 83 kg; BMI 32; para 2+0) initially presented to a district hospital with an abrupt history of typically ischaemic exertional chest pain. The pain occurred with a stable frequency, but had increased in severity over several months. Her general and cardiac examination was unremarkable, with the exception of community-based treatment for hypertension. Her presenting 12 lead ECG, and repeated measurements of cardiac troponin I, showed no abnormality. She had been discharged from the admitting hospital for elective cardiac investigation, but was re-admitted within 48 hours because of symptoms of recurrent pain, along with the concerns of both the patient and her general medical practitioner. On the repeat admission, there were again no changes in her repeat ECG and cardiac biomarkers, including troponin I, which were persistently negative. She had had no symptomatic response to additional oral anti-ischaemic therapy (Bisoprolol). Due to persistent symptomatic problems, and a suspicion of reversible ischaemia despite negative biomarkers and normal ECG and chest x-ray, she was transferred for emergent angiographic triage and/or percutaneous coronary intervention. No functional testing had been completed prior to transfer due to the patient's age and gender and given that non invasive tests have well documented poor specificity and sensitivity in female hypertensive patients in midlife [3].

The patient was noted to be asymptomatic on transfer, and her chronic therapies had been adjusted to treatment with Bisoprolol 5 mg od, Aspirin 75 mg od; Simvastatin 40 mg on and Amlodipine 5 mg od. Her accompanying chest x-ray showed a normal cardiac silhouette later confirmed by transthoracic echocardiography showing no abnormality of valvular structure or contractile function. The following day, she was taken to diagnostic cardiac catheterization, with a view to proceeding to percutaneous intervention if required.

Routine selective coronary angiography was completed uneventfully from the right femoral artery, using lignocaine local anaesthesia. Initial injections into the left coronary artery revealed diffuse direct shunting of contrast from all branches into the left ventricular cavity throughout the length of the vessel (Figures 1 and 2). The epicardial coronary arterial tree was otherwise normal, showing no trace of atheroma, and no evidence of isolated arterial stenosis. Spontaneous coronary spasm was not documented. Conventional imaging failed to visualize the entire length of the communications joining the coronary arteries to the cavity of the left ventricle. The right coronary artery was also affected by the same phenomenon, but to a more minor degree, with the micro-shunting occurring predominantly distally and towards the apex of the heart (Figure 3). The contrast left ventriculogram was normal.

Published online 2007 December 10. doi: 10.1186/1752-1947-1-177.
Copyright ├é┬® 2007 MacFadyen et al; licensee BioMed Central Ltd.]\">
  Figure 1
Late intra-coronary injection of contrast in the straight antero-posterior view shows multiple transmural micro-channels emptying directly into the left ventricle.

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References
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