Fatal coronary artery spasm triggered before transseptal puncture for mitral transcatheter edge-to-edge-repair: a case report

Abstract

Background

Transient ST-segment elevation or coronary artery spasm during transseptal catheterization has been previously described. Most cases were either reversible or asymptomatic.

Case summary

We present a case of severe multiple coronary artery spasms with advanced atrioventricular block and ventricular fibrillation during compression of the fossa ovalis by a sheath catheter, before the performance of the transseptal puncture procedure for mitral transcatheter edge-to-edge-repair.

Discussion

The mechanical effects of forward tension from transseptal puncture on the interatrial vagal network could be the most possible explanation for the occurrence of this phenomenon.

Learning points.

• Coronary artery spasm is a rare and fatal complication during transseptal catheterization and could occur even before successful atrial septal crossing.

• Careful monitoring and timely coronary angiography, administration of nitrates, and haemodynamic support are necessary for bringing about a quick and proper reversal of this complication.

Introduction

Mitral transcatheter edge-to-edge-repair (TEER) has shown to effectively improve symptoms and functional capacity in patients with severe mitral valve regurgitation. The clip accesses the left atrium (LA) from the femoral vein approach, transseptal techniques, and equipment. Also, atrial septal puncture is essential for clip delivery. Transient ST-segment elevation (STE) or coronary artery spasm is a rare complication that occurs during transseptal catheterization.¹ But, persistent STE and multiple coronary artery spasms could prove fatal.² We present a case of life-threatening multiple coronary artery spasms with advanced atrioventricular block and ventricular fibrillation during compression of the fossa ovalis by a sheath catheter, before the performance of the transseptal puncture procedure for mitral TEER.

Timeline

Case presentation

An 85-year-old woman complaining of dyspnoea on exertion was referred to our hospital for the treatment of severe mitral regurgitation. In addition to her current condition, she had a history of systemic arterial hypertension, chronic renal dysfunction (CKD) IV stage, and hip surgery, treated with amlodipine and hydrochlorothiazide. Cardiopulmonary auscultation revealed a grade 4 systolic murmur at apex and bilateral coarse crackles. Transthoracic echocardiography showed a severe eccentric mitral regurgitation jet related to a prolapse of the posterior leaflet (P2) with incomplete systolic coaptation (leaflets systolic separation: 0.6 cm; jet area: 11.4 cm², vena contracta: 0.7 cm, effective regurgitant orifice area at proximal isovelocity surface area method: 0.63 cm², R volume 81 mL). Biventricular function was normal, with a mild dilated left ventricle (left ventricular end-diastolic diameter 57 mm) and left atrial (40 mm). These data were subsequently confirmed with transoesophageal echocardiography (TEE) (Figure 1A, B; Supplementary material online, files S1 and S2). Coronary computed tomography angiography, which was performed before hospitalization, showed a mild stenosis of the middle left anterior descending (LAD) artery and the left circumflex (LCX) artery. N-terminal pro-B-type natriuretic peptide was 3241 pg/mL (normal range 0–150 pg/mL). Low-density lipoprotein (LDL) cholesterol was 2.48 mmol/L (normal range 0–4.14 mmol/L), and high-sensitivity C-reactive protein (hsCRP) (normal range 0–5 mg/L) was 2.58 mg/L. Considering the high risk of open-heart surgery (The Society of Thoracic Surgeons risk score was 11.538%), the Heart Team opted for mitral TEER.

Figure 1.

(A) Transoesophageal echocardiography showing a severe eccentric mitral regurgitation jet; (B) transoesophageal echocardiography showing a prolapse of the posterior leaflet (P2) with an incomplete systolic coaptation.

The patient was on general anaesthesia. Brockenbrough’s procedure was conducted with a standard technique with real-time TEE guidance. Mullins sheath (8F) was inserted from the right femoral vein to the superior vena cava, then slowly pulled down in the right atrium, and gently compressed onto the fossa ovalis (Figure 2; Supplementary material online, file S3). Sinus bradycardia and hypotension were observed before the tip of puncture needle penertrated into the left atrium. The puncture procedure was temporary stopped. Cardiac tamponade was excluded, and the tip of the sheath was still in the right atrium, which was confirmed by TEE. With the injection of 0.5 mg atropine, the patient’s heart rate and blood pressure returned to baseline. The procedure was continued. In a few minutes during catheter compressing before needle puncture, profound sinus bradycardia and hypotension were noted again, and STE in leads II, III, and augmented Vector Foot (avF) was observed. The procedure was interrupted again. Following another shot of atropine, a fast saline drip, and even withdrawal of the Mullins sheath, ventricular fibrillation, and cardiac arrest progressively happened within minutes. Continuous cardiopulmonary resuscitation, defibrillation, and intravenous epinephrine were routinely undertaken, and immediately, coronary angiogram from the left femoral artery showed a severe vasospasm in the proximal right coronary artery (RCA) with 99% stenosis and with the LCX 100% occluded, although the LAD artery was still patent (Figure 3A). Intracoronary injection of nitroglycerine was administered but without any improvement. Balloon angioplasty was also performed in the RCA and LCX, respectively, to exclude coronary thrombus embolism, without favourable blood flow. Extracorporeal membrane oxygenation (ECMO) was simultaneously inserted, and repeated intracoronary nitroglycerine injection finally restored coronary flow. Coronary angiogram was re-checked with ECMO support after minutes, and no stenosis was found in the RCA and LCX (Figure 3B). Blood pressure and electrocardiograph returned to normal, and the patient was sent to the intensive care unit (ICU) for continuous monitoring. Following 6 days of monitoring treatment in the ICU, the subsequent multi-organ dysfunction (liver, kidney, and coagulation system) led to the death of this elderly patient.

Figure 2.

Mullins sheath (8F) is seen gently ‘tenting’ on the fossa in the right atrium, while a sinus bradycardia (43 beats per minute) is noted.

Figure 3.

(A) Electrocardiogram shows a high degree atrioventricular block and ST-segment elevation in inferior leads. Coronary angiography reveals multiple coronary spasms; (B) coronary angiogram was rechecked with extracorporeal membrane oxygenation support and no stenosis was found in the right coronary artery and left circumflex artery. Electrocardiogram showed an improvement of ST-segment elevation.

Discussion

The transseptal (Brockenbrough’s) puncture is widely used to access the LA during the procedure of TEER. Known complications of the transseptal procedure include aortic root and atrial perforation, stroke, and transient ischaemic attacks.³ In our laboratory, this procedure has been conducted since the 1970s as a routine technique for accessing the LA for performing percutaneous balloon mitral valvuloplasty, ablation of atrial fibrillation, and TEER. Our case presents an unexpected and hazardous complication that manifested as persistent STE, followed by life-threatening ventricular arrhythmia, which was caused by multiple severe coronary spasms (CASs), during a transseptal procedure. To our knowledge, however, refractory CAS, which occurs only during sheath ‘tenting’ but before puncture or catheter passage, has been seldom reported before.

Transient STE during transseptal catheterization is a well-recognized phenomenon, as reported in several case studies.^4,5 However, different studies have reported varied incidence rates of STE. Tang et al.⁶ reported that the incidence rate of STE during transseptal catheterization for atrial fibrillation ablation was about 0.38%. However, Vale et al.¹ showed that the rate was 1.3% in a retrospective cohort and 5.2% in a prospective cohort. Another study found that the phenomenon of STE occurred mostly after transseptal puncture, even after ablation.⁷ There was only one case of a patient who exhibited STE and sinus bradycardia during repeated compression of the fossa ovalis by a sheath catheter, but they resolved soon after atropine administration.⁴ This phenomenon is believed to be a short-term one and benign. The cases of most patients were either self-relieving or their condition resolved in a few minutes with atropine and fast saline drip.⁷

The mechanism and underlying pathophysiology of STE have not been well established. There are two possible mechanisms that can explain the occurrence of STE during or after transseptal catheterization: (i) A Bezold–Jarisch-like reflex causes CAS. The atrial septum is concentrated with a high density of parasympathetic fibres, which preferentially innervates the RCA, leaving it vulnerable to cholinergic vasospasms.⁴ The passage of the catheter through these high-density nerve complexes of the atrial septum during transseptal catheterization might irritate or damage the plexus and then induce a Bezold–Jarisch-like reflex. Most cases occur after transseptal puncture, while some occur after the ablation procedure.^1,6 In our case, the patient experienced bradycardia and hypotension before the catheter entered the LA during the process of manipulation in the atrial septum, and ‘tenting’. The forward tension by the sheath on the foramen ovale for ‘tenting’ that even before crossing the septum can trigger the nerve complexes and cause a Bezold–Jarisch-like reflex. The induced hypervagotonia would result in the release of acetylcholine, which causes spasm in the coronary circulation.⁸ The RCA is more susceptible to cholinergic constriction and vasospasm.⁵ Transient STE would occur. Nakamura et al.⁹ reported a 0.19% (42 of 22 232) incidence rate of CAS related to the AF ablation procedure. Also, in their study, they found a CAS rate of 21% before ablation. A previous report showed that patients with CAS tended to have a smaller LA than those without.¹⁰ The LA in our case was mildly dilated. The nerve fibre distribution in the smaller atria might be vulnerable to a reflex activation during the transseptal puncture. On the other hand, age, smoking, hypercholesterinaemia, diabetes, and hsCRP are significant risk factors for CAS.¹¹ In this case, advanced age, LDL-cholesterol level, and atherosclerotic coronary artery disease could be potential factors contributing to CAS. (ii) Coronary artery embolism (air or thrombus) is a different potential cause of this phenomenon. Coronary air embolism occurring as a complication of transseptal RF ablation of the left free wall accessory pathways has been described in previous reports.^12,13 Air emboli or thrombus can be induced by the transseptal sheath, which migrates into the RCA due to of the RCA ostium placed in the superior position in a supine patient, which can partly explain the occurrence of STE in inferior leads. In our case, both the needle and the catheter had not been transduced into the LA. Pre-operation TEE and simultaneous TEE excluded a patent foramen ovale. Air embolism could not be the reason of STE in our case.

Conclusion

Multiple CASs could be a severe complication of transseptal puncture during TEER. A neutrally mediated pathway, activated by the mechanical effects of the forward tension from transseptal puncture on the interatrial vagal network and leading to CAS, appears to be a possible explanation for the occurrence of this phenomenon, even before the catheter enters the LA. As the number of TEER and radiofrequency ablation procedures increases, CAS patients during the perioperative period may also rise. Careful monitoring, timely coronary angiography, administration of nitrates, and haemodynamic support are necessary for bringing about a quick and proper reversal of this rare but potentially life-threatening complication.

Lead author biography

Yujing Mo, MD, PhD, is a cardiologist at the Guangdong Cardiovascular Institute in Guangzhou, China. She is board-certified in Internal Medicine and Cardiology. She is interested in cardiac intensive care, valvular heart disease, and echocardiography.

Supplementary material

Supplementary material is available at European Heart Journal – Case Reports.

Data availability

The authors confirm that written consent for submission and publication of this case report including images and associated text has been obtained from the patient in line with COPE guidance.