Gamble, David T.

Leslie, Stephen J.

Scally, Caroline

Shuttleworth, Kara J.

BMJ Case Reports

PubMed

Aberdeen University Research

CASE REPORTTakotsubo cardiomyopathy with severebradyarrhythmia following epidural insertionDavid T Gamble,1 Kara J Shuttleworth,1 Caroline Scally,2 Stephen J Leslie1,31Cardiac Unit, RaigmoreHospital, Inverness, UK2University of Aberdeen,Aberdeen, UK3Department of Diabetes &Cardiovascular Science,University of the Highlands andIslands, The Centre for HealthScience, Inverness, UKCorrespondence toProfessor Stephen J Leslie,stephen.leslie@nhs.netAccepted 11 October 2016To cite: Gamble DT,Shuttleworth KJ, Scally C,et al. BMJ Case RepPublished online: [pleaseinclude Day Month Year]doi:10.1136/bcr-2016-216946SUMMARYWe present a case of takotsubo cardiomyopathy (TTC)with ventricular stand still and atrioventricular block. TTCcan mimic ST elevation myocardial infarction and heartfailure, but in this case resulted in a severe cardiacconduction disorder and ventricular standstill. This is arecognised but unusual presentation and serves as alesson to those undertaking anaesthetics to be vigilantfor TTC.BACKGROUNDTakotsubo cardiomyopathy (TTC) or ‘apical bal-looning syndrome’ is a relatively new clinicalcondition first described in 1990.1 It is a form ofnon-ischaemic cardiomyopathy, typically, but notexclusively, characterised by transient left ventricu-lar (LV) dysfunction of the mid-apical segment,associated with compensatory excessive contractionat the cardiac base and apical ballooning. Mostpatients present with chest pain and dyspnoeaaccompanied by ECG changes, consistent withmyocardial infarction, cardiac enzyme release andnormal coronary arteries.2 Other complicationshave also been described, including arrhythmias,3–5cardiogenic shock,6 intraventricular thrombi7 andarterial embolism.8 TTC has a female predilectionand is thought to occur due to a catecholaminesurge following stressful physical or emotionalevents leading to coronary spasm, autonomic dis-turbance and microvascular impairment, althoughthe precise pathophysiology remains unclear.4CASE PRESENTATIONA 66-year-old woman with a history of melanomawas admitted for an elective left-sided open adrena-lectomy to manage an adrenal mass. Shortly afteran uncomplicated epidural insertion and before anysurgical intervention, she developed profoundbradycardia with asystole requiring cardiopulmon-ary resuscitation (CPR) including intravenous epi-nephrine (0.5 mg) and atropine (600 mg). Therewas prompt return of spontaneous circulation withspontaneous breathing shortly after CPR (<1 min)was started. The patient reported no precedingchest pain.INVESTIGATIONSAn ECG at the time showed widespread Twave inver-sion and prolonged QT interval (figure 1A). Theseare well-recognised ECG changes in TTC but usuallyoccur after 24–48 hours. Initial plasma troponin wasundetectable (lower limit of detection ≤40 ng/L) butwas raised after 12 hours at 1275 ng/L, other bloodtests including haemoglobin, urea and electrolyteswere within normal limits. An echocardiogram per-formed shortly after this episode showed apical akin-esia resulting in moderate LV dysfunction with nosignificant valvular abnormality. The patient was sub-sequently transferred to the coronary care unit forfurther management and continuous ECG monitor.At this time, the ECG intermittently demonstratedcomplete heart block (CHB), with a ventricularescape rhythm of 30 bpm (figure 1B). There wereperiods of asystole (up to 10 s; figure 1C).An invasive coronary angiogram showed normalcoronary arteries. A repeat echocardiogram at4 days showed complete recovery of LV function.The diagnosis of TTC was made.Despite recovery of LV function, CHB waspersistent.TREATMENTA dual-chamber rate responsive pacemaker wasinserted without complication. The patient was dis-charged on day 5.OUTCOME AND FOLLOW-UPBecause of the pressing need for surgery, this wasrescheduled in the subsequent 3 weeks and wasuncomplicated other than a brief episode of non-sustained ventricular tachycardia postoperatively onthe surgical high dependency ward. The cause of theadrenal mass was metastatic melanoma. Continuedfollow-up of the pacemaker (at 3 months) was unre-markable, with atrial pacing 1.3% and ventricularpacing <1%.DISCUSSIONArrhythmias are an increasingly recognised complica-tion of TTC and include atrial fibrillation, ventriculartachycardia and conduction defects.3–6 Completeatrioventricular (AV) block is less common (<5%)and the involvement of prolonged asystole rarer.The underlying pathophysiology of bradyar-rhythmias in TTC remains unclear; proposedmechanisms including reduced coronary blood flowto conduction pathways secondary to ventriculardyskinesia, catecholamine-induced coronary vaso-spasm (especially to right coronary artery) and con-duction pathway fibrosis secondary to prolongedischaemia.5 The pathophysiology of tachyarrhyth-mia is most likely a result of long QT as is com-monly seen in TTC and catecholamine surge. Inour case, it is possible that the administration ofepinephrine during the brief cardiac arrest mayhave contributed to the development of TTC asthis mechanism has been previously suggested.9Gamble DT, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-216946 1Unusual presentation of more common disease/injuryThe correct management of TTC with complete AV block isuncertain and can be challenging. Literature on this subject islimited but recent studies have suggested that AV block persistsdespite resolution of the cardiomyopathy, and that permanentpacemaker implantation improves mortality in these patients. Inour case, there was some time pressure due to the need forsurgery and therefore early permanent pacemaker implantationwas undertaken.4 10 11This case of ventricular asystole and cardiac arrest in thecontext of a TTC is unusual, and in this case mimicked a pro-found vasovagal episode triggered by an epidural insertion. Thecorrect diagnosis was made with the use of earlyechocardiography and exclusion of coronary artery disease, inour case by conventional coronary angiography although onecould argue that CT coronary angiography could be useful insuch cases. Less invasive modalities such as CT coronary angiog-raphy would be appropriate in a clinical setting where ST eleva-tion was not suspected. Cardiac MRI (CMR) would also be veryimportant where clinical uncertainty or poor echo images result-ing in diagnostic doubt.12 CMR is a useful imaging modality insuspected TTC, as the absence of late gadolinium enhancementrules out a myocardial infarction and the presence of myocardialoedema can point to the diagnosis. If performed in the acutephase, CMR can also help differentiate between myocarditisFigure 1 (A) Initial ECG showing widespread T wave inversion and QT interval prolongation. (B) ECG showing complete heart block with escaperhythm at 30 bpm. (C) Two lead rhythm strip and plethysmography trace showing ventricular asystole.2 Gamble DT, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-216946Unusual presentation of more common disease/injuryand TTC. In this case, the profound bradycardia would havemade MRI difficult and the need for permanent pacing wouldpreclude MRI in the recovery phase.The persistence of AV block and need for surgery mandatedpermanent pacemaker insertion. This case illustrates that anaes-thetist should be vigilant for this increasing recognised cause ofcardiovascular compromise and request an early echocardiogramin suspected cases.12Learning points▸ Takotsubo cardiomyopathy (TTC) is an increasinglyrecognised clinical condition.▸ TTC can mimic other cardiac conditions and earlyinvestigation with echo and coronary imaging is importantto avoid unnecessary therapy, for example, thrombolysis.▸ Treatment of TTC is supportive.▸ Recovery is usually good although recurrence can occur.Contributors DTG was involved in the clinical care of this patient, and preparationof the first draft of this paper and subsequent review. KJS was involved in theclinical care of this patient, preparation of the first draft of this paper andsubsequent review, and had major role in preparing the images. CS was involved inthe review of the first draft and literature review. SJL was involved in the clinical careof this patient and had the idea for the case report and was involved at all stages ofwriting.Competing interests None declared.Provenance and peer review Not commissioned; externally peer reviewed.Open Access This is an Open Access article distributed in accordance with theCreative Commons Attribution Non Commercial (CC BY-NC 4.0) license, whichpermits others to distribute, remix, adapt, build upon this work non-commercially,and license their derivative works on different terms, provided the original work isproperly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/REFERENCES1 Sato H, Tateishi H, Uchida T. Takotsubo type cardiomyopathy due to multivesselspasm. In: Kodama KH, Hon H. eds. Clinical aspect of myocardial injury fromischemia to heart failure. Ka- gakuhyouronsya Tokyo, 1990:56–64.2 Kurisu S, Sato H, Kawagoe T, et al. Tako-tsubo-like left ventricular dysfunction withST-segment elevation: a novel cardiac syndrome mimicking acute myocardialinfarction. Am Heart J 2002;143:448–55.3 Yoshida T, Hibino T, Fujimaki T, et al. Transient mid-ventricular ballooning syndromecomplicated by syncope: a variant of tako-tsubo cardiomyopathy. Int J Cardiol2009;135:e20–3.4 Korantzopoulos P, Nikas DN, Letsas K, et al. High-grade atrioventricular block andtakotsubo cardiomyopathy: case report and review of the literature. Int CardiovascRes J 2015;9:119–24.5 Limm BN, Hoo AC, Azuma SS. Variable conduction system disorders in takotsubocardiomyopathy: a case series. Hawai J Med Public Health 2014;73:148–51.6 Dib C, Prasad A, Friedman PA, et al. Malignant arrhythmia in apical ballooningsyndrome: risk factors and outcomes. Indian Pacing Electrophysiol J2008;8:182–92.7 Haghi D, Papavassiliu T, Heggemann F, et al. Incidence and clinical significance ofleft ventricular thrombus in tako-tsubo cardiomyopathy assessed withechocardiography. QJM 2008;101:381–6.8 Andò G, Saporito F, Trio O, et al. Systemic embolism in tako-tsubo syndrome. IntJ Cardiol 2009;134:e42–43.9 Calvo-Taracido M, Almendro-Delia M, Garcia-Rubira J. Tako-tsubo syndrome afteradministration of intravenous adrenaline during atrioventricular block. Open J EmergMed 2013;1:8–10.10 Le MT, Grimard C, Varenne O, et al. Tako-tsubo cardiomyopathy and high-degreeatrio-ventricular block: do we need to wait left ventricular function recovery toimplant a pacemaker? Int J Cardiol 2014;176:e56–9.11 Stiermaier T, Rommel KP, Eitel C, et al. Management of arrhythmias in patientswith takotsubo cardiomyopathy: is the implantation of permanent devicesnecessary? Heart Rhythm 2016;13:1979–86.12 Dastidar AG, Frontera A, Palazzuoli A, et al. TakoTsubo cardiomyopathy: unravellingthe malignant consequences of a benign disease with cardiac magnetic resonance.Heart Fail Rev 2015;20:415–21.Copyright 2016 BMJ Publishing Group. All rights reserved. For permission to reuse any of this content visithttp://group.bmj.com/group/rights-licensing/permissions.BMJ Case Report Fellows may re-use this article for personal use and teaching without any further permission.Become a Fellow of BMJ Case Reports today and you can:▸ Submit as many cases as you like▸ Enjoy fast sympathetic peer review and rapid publication of accepted articles▸ Access all the published articles▸ Re-use any of the published material for personal use and teaching without further permissionFor information on Institutional Fellowships contact consortiasales@bmjgroup.comVisit casereports.bmj.com for more articles like this and to become a FellowGamble DT, et al. BMJ Case Rep 2016. doi:10.1136/bcr-2016-216946 3Unusual presentation of more common disease/injury

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Takotsubo cardiomyopathy with severe bradyarrhythmia following epidural insertion

David T Gamble

Kara J Shuttleworth

Caroline Scally

Stephen J Leslie

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http://dx.doi.org/10.1136/bcr-2016-216946

Takotsubo cardiomyopathy with severe bradyarrhythmia following epidural insertion

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