Rare recurrence of apical ballooning (takotsubo) syndrome in an elderly man

Abstract

Apical ballooning syndrome (ABS) is an under recognised clinical entity characterised by acute reversible left ventricular systolic dysfunction that mimics acute myocardial infarction in the absence of obstructive coronary artery disease; typically occurring in the setting of profound stress.¹ ABS disproportionately affects older women and recurrences are infrequent. We, hereby, describe a rare phenomenon of recurrent ABS in an elderly male patient, 10 years apart, presenting with the same left ventricular morphological appearance following non-cardiac surgeries. The case illustrates the importance of considering ABS in the differential diagnosis of perioperative acute myocardial infarction in older men undergoing major surgery.

Background

Apical ballooning syndrome (ABS) (also known as takotsubo cardiomyopathy/syndrome after the Japanese octopus trap and stress cardiomyopathy) is an acute reversible clinical entity that was initially described in the early 1990s. The clinical and ECG features of ABS are similar to an acute myocardial infarction (AMI).² Over two-thirds of cases are triggered by either an emotional or physical trigger. For reasons that remain to be established, most cases develop in women, predominantly postmenopausal. However, it is increasingly recognised that ABS may also occur in men, with men accounting for approximately 5%–10% of cases.^{3 4} In-hospital survival is close to 95% with a low recurrence rate of 1%–2% per year,^{5 6} virtually all reported cases being in women.

Case presentation

An 81-year-old man with a history of hypertension, hyperlipidaemia and non-obstructive coronary artery disease was admitted to the hospital with worsening abdominal symptoms and inflammatory changes on imaging concerning for gallbladder necrosis. The patient underwent laparoscopic cholecystectomy which was complicated by an enterotomy, requiring conversion to open cholecystectomy for small bowel resection and anastomosis. He tolerated the procedure well, but developed postoperative atrial fibrillation with haemodynamic compromise; prompting transfer to the intensive care unit for supportive therapies which included intubation for airway protection and vasopressors. Investigations into potential infectious or additional iatrogenic complications from the procedure were unrevealing. The patient’s condition improved over the subsequent 48 hours, and he was weaned off the vasopressors and mechanical ventilation. On postoperative day 5, the patient developed progressive respiratory distress, altered mental state and intermittent runs of supraventricular tachyarrhythmia with haemodynamic instability, requiring reintubation and vasopressor support.

Of note, 10 years previously, the patient was diagnosed with rectal cancer and underwent surgical resection with J-pouch anal anastomosis and loop ileostomy. After completion of chemotherapy, he presented to the hospital for ileostomy take-down. On postoperative day 5, he developed severe dyspnoea and hypoxic respiratory failure, requiring intubation. A 12-lead ECG demonstrated new T-wave inversions in the anterolateral leads and elevated cardiac biomarkers (troponin T 0.68 ng/mL (normal <0.03 ng/mL); creatine kinase-MB 18.3 ng/mL (normal <6.2 ng/mL)). Transthoracic echocardiogram (TTE) revealed reduced systolic function ejection fraction (EF 30%), and anterior, anterolateral, inferior and apical akinesis. Angiogram showed no flow-limiting lesions. The patient was managed medically with beta-blocker and ACE-inhibitor therapy. Seven weeks later, there was resolution of the ECG abnormalities and regional wall motion abnormalities with normal left ventricle function.

Investigations

A 12-lead ECG obtained during the acute episode demonstrated atrial fibrillation with rapid ventricular response and loss R waves in the right precordial leads. Few hours later, there was diffuse T-wave inversions (figure 1A,B). Index troponin T was elevated at 0.14 ng/mL (normal, <0.01 ng/mL). A TTE revealed moderate left ventricular enlargement, reduced EF of 30% and new regional wall motion abnormalities (akinesis of the mid-ventricle and apical segments) that were not present a week prior (video 1). A 12-lead ECG (figure 1C), TTE and myocardial perfusion study obtained a few weeks prior as part of his preoperative evaluation demonstrated no significant abnormality. Emergent coronary angiography showed moderate multivessel atherosclerosis without flow limiting lesions. Left-ventricular angiogram confirmed severe regional wall motion abnormality of the mid and apical segments (figure 2A,B and videos 2 and 3). The distribution of wall motion abnormalities, low EF, relatively mild troponin rise and absence of flow-limiting lesions on coronary angiogram in a patient who had undergone a major non-cardiac surgery were consistent with ABS.

Figure 1.

(A) 12-lead ECG tracing showing atrial fibrillation with rapid ventricular response, loss of R waves in leads V1 to V3, and non-specific ST-T segment changes. (B) 12-lead ECG tracing showing ectopic atrial rhythm and diffuse T wave inversions in multiple leads. (C) Baseline 12-lead ECG obtained prior to admission.

Video 1.

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DOI: 10.1136/bcr-2017-222451.video01

Transthoracic echocardiogram in the apical four-chamber and short-axis views (Mayo format) showing apical and mid-ventricle regional wall motion abnormalities. 2D, two dimensional.

Figure 2.

(A) Left ventriculogram in right anterior oblique view projection shown at end diastole (left) and end systole (right), respectively, showing mid-ventricle and apical akinesis. (B) Left ventriculogram in left anterior oblique view projection shown at end diastole and end systole, respectively, showing mid-ventricle and apical akinesis.

Video 2.

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DOI: 10.1136/bcr-2017-222451.video02

Right anterior oblique view of the left ventricular gram with mid-ventricle and apical akinesis.

Video 3.

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DOI: 10.1136/bcr-2017-222451.video03

Left anterior oblique view of the left ventricular gram with mid-ventricle and apical akinesis.

Differential diagnosis

The differential diagnoses considered in the postoperative setting of new ischaemic ECG changes, abnormal cardiac biomarkers of myonecrosis and new ventricular regional wall motion abnormalities included: AMI and ABS. AMI is much more frequent and therefore the initial presumed diagnosis in most cases with the early treatment pathways directed accordingly.

The patient described in this case report developed atrial fibrillation acutely in the postoperative setting and while tachycardia-mediated cardiomyopathy should be part of the differential diagnosis in patients with new left ventricular dysfunction; it is more likely to occur after a sustained period of tachyarrhythmia which was not seen in this patient.

Treatment

The patient was admitted to the cardiac intensive care unit after an intra-aortic balloon pump was placed for haemodynamic support. Over the next week, he was successfully weaned off the vasopressors, mechanical support and ventilation. Medical therapy with ACE-inhibitor and beta-blocker was initiated and optimised prior to discharge.

Outcome and follow-up

Six weeks after discharge, the ECG had returned to baseline and a repeat TTE demonstrated normal wall motion with an EF of 54% (video 4). The patient’s normalisation of cardiac function confirmed the diagnosis of recurrent ABS.

Video 4.

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DOI: 10.1136/bcr-2017-222451.video04

Transthoracic echocardiogram in the apical four-chamber and short-axis views (Mayo format) illustrating recovered left ventricular function. FPS, frames per second.

Discussion

ABS is a rare cause for a transient acute cardiomyopathy in men. Approximately 90% of patients who develop ABS are women, the vast majority being postmenopausal, and it has been estimated that 5%–10% of women who are initially suspected of an AMI at the time of presentation actually have ABS.^{4 7} The fact that men account for no more than 10% of cases of ABS likely reflects gender differences in cardiovascular pathophysiology; but it has been speculated that other reasons include misdiagnosis given the higher prevalence of coronary artery disease in men, and potentially worse prognosis of ABS and greater risk of sudden death in men. Current evidence suggests that among those admitted to hospital, men and women have similar clinical characteristics. However, physical stressor is more common triggers, and shock or cardiac arrest occurs with greater frequency in men.^{3 8} Recent data suggest that in-hospital mortality may be higher in males suggesting that male gender is a prognostic risk marker in ABS.^{4 9}

Another poorly understood aspect of this unique condition is the low rate of recurrence. In our experience, the estimated rate of recurrence is 11.4% over 4 years.^{6 10} A similar conclusion was reached in a systematic review of the literature that the recurrence rate is 1%–2% per year. The mean age at recurrence was 65.5 years, considerably younger than our patient. Nearly all recurrent cases, according to the review, were in women though no details are available.¹⁰ In our prior publication, virtually all cases of recurrence were in women who were less than 50 years old, underscoring the uniqueness of our case report.³ There is one recently published case report of two recurrent episodes of ABS in a 66-year-old man.¹⁰ Thus, our case illustrates the importance of ABS as a differential diagnosis of acute haemodynamic decompensation in the postoperative setting, and the need for awareness for the possibility of its recurrence during preoperative medical assessment in male and female patients with a history of ABS.

Learning points.

• Apical ballooning syndrome (ABS) is characterised by acute reversible left ventricular systolic dysfunction that mimics an acute coronary syndrome.

• ABS affects postmenopausal women, but infrequently may occur in older men.

• ABS recurrence is infrequent (1%–2%/year), and we report the novel observation that it may rarely occur in men.

• A history of ABS during preoperative medical assessment should lead to the awareness that recurrence is a possibility during major non-cardiac surgery.