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Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome: a case report and review of the literature

Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome: a case... CASE REPORT European Heart Journal - Case Reports (2017) 1, 1–5 doi:10.1093/ehjcr/ytx005 Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome: a case report and review of the literature 1 2 2 Jan J. J. Aalberts *, Theo J. Klinkenberg , Massimo A. Mariani , and Pim van der Harst 1 2 Department of Cardiology, University of Groningen, University Medical Center Groningen, Hanzeplein 1, 9700 RB Groningen, The Netherlands; and Department of Cardiothoracic Surgery, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands Received 24 June 2017; accepted 20 August 2017; online publish-ahead-of-print 13 October 2017 Abstract Takotsubo syndrome (TTS) complicated by refractory cardiogenic shock is a challenging clinical problem, as treat- ment with inotropic agents and/or vasopressors is contraindicated. We illustrate this by a patient presenting with chest pain and shortness of breath caused by TTS complicated by cardiogenic shock requiring mechanical circula- tory support (MCS). The patient received central extracorporeal life support with a cannula in the left atrium (pre- load reduction of left ventricle) and the return cannula in the ascending aorta (neutral on afterload). Treatment with MCS was complicated by a cardiac tamponade. Left ventricular function recovered after 24 h, and the patient was doing well at the outpatient clinic 7 weeks after discharge. In addition, we reviewed the literature (PubMed search) reporting on MCS in patients with TTS. Including our patient, 17 cases of TTS induced cardiogenic shock receiving MCS have been reported. Age of the patients ranged from 16 years to 74 years, and 71% of the patients were female. Extracorporeal life support was the most used type of MCS (82% of the cases). Two patients died, and complications of MCS were rare (one case of leg ischaemia). Theoretically, MCS devices that reduce pre-load and are neutral on afterload are preferable. However, no specific type of MCS can be recommended as random- ized trials are lacking. In conclusion, our case and the available literature suggests that MCS in TTS induced refrac- tory cardiogenic shock is an immediate and feasible lifesaving treatment. Keywords Takotsubo syndrome Cardiogenic shock Mechanical circulatory support Extracorporeal life • • • support Case report . Introduction Learning points . . Takotsubo syndrome (TTS) is an acute and usually reversible heart Takotsubo syndrome (TTS) complicated by cardiogenic shock is . failure syndrome with an estimated prevalence of 1–2% in patients a challenging clinical problem, as treatment with inotropic . suspected of an acute coronary syndrome. Several diagnostic crite- agents and/or vasopressors is contraindicated. ria have been proposed, and we support the criteria suggested by the Mechanical circulatory support for TTS-induced cardiogenic . Heart Failure Association of the European Society of Cardiology. shock is a reasonable treatment option and has acceptable Takotsubo syndrome is characterized by regional wall abnormalities outcomes. of the myocardium (no culprit of atherosclerotic coronary artery disease), frequently preceded by a stressful trigger (emotional of physical). The classical pattern of regional wall abnormalities is apical * Corresponding author. Tel: þ31 503 6112355, Fax: þ31 503 611347, Email: j.j.j.aalberts@umcg.nl. This case report was reviewed by Christian Fielder Camm and Matteo Cameli. V The Author 2017. Published by Oxford University Press on behalf of the European Society of Cardiology. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 2 J.J.J. Aalberts et al. and circumferential mid-ventricular hypokinesia and basal hypercon- Timeline 1,2 tractility; however, anatomical variants exist. In the classical pattern, end-systolic the left ventricle resembles a Takotsubo, which is a . Japanese word for an octopus trap with a narrow neck and globular . lower portion. Excessive catecholamine release caused by the stress- . Time Events ful trigger are believed to play a crucial role in the pathophysiology of . ................................................................................................. 2 . TTS. Complications that can occur include mitral regurgitation, . Start of episode 67-year old woman presenting with chest pain and arrhythmias, thrombus formation, pericardial effusion, ventricular wall . shortness of breath. rupture, cardiogenic shock, and death. Treatment of refractory car- Prehospital ECG suggests ST-elevation myocardial diogenic shock in TTS is particularly challenging, as catecholamines are infarction contraindicated and mechanical circulatory support (MCS) may be • Accepted for STEMI-protocol and straight trans- the only option left. We present here such a patient and review the port to catheterization laboratory literature reporting on the treatment of TTS with MCS. Physical examination: severe distress, heartrate 107 . bpm, RR 88/63 mmHg, no signs of decompensa- Patient information . tion: Shock . Fluid challenge: 2 L NaCl 0.9% in 45 minutes A 67-year-old woman discovered that there had been a burglary in . Phenylephrine (0.05 mg/minute) started as her daughter’s house. . vasopressor Immediately she developed severe chest pain combined with LV-angiogram: Takotsubo syndrome shortness of breath and her husband called the emergency services. Cardiogenic shock Her medical history consisted of type 2 diabetes mellitus controlled Central ECLS implanted by oral drugs and chronic obstructive pulmonary disease (COPD). 24 hours later Cardiac tamponade During transport to the hospital, an electrocardiogram (ECG) was Tamponade surgically relieved taken, and ST-elevation myocardial infarction was suspected. Echocardiography: complete recovery of left ven- tricular function Physical examination . • ECLS weaned and removed On arrival at the catheterization laboratory, the patient was in severe 7 weeks later Fully recovered at the outpatient clinic distress, heart rate was 107 b.p.m., and blood pressure was 88/ 64 mmHg. Central capillary refill time was 6 s. Cardiac and pulmonary auscultation revealed no abnormalities. . Follow-up and outcomes Diagnostic assessment . About 24 h later, the patient deteriorated. A high central venous An ECG (Figure 1) was obtained showing sinus rhythm of 108 b.p.m.; inter- . pressure together with collapse of the right atrium on transoeso- mediate heart axis and normal conduction times; 1 mm concave ST- . phageal echocardiography (TOE) led to the diagnosis of cardiac segment elevations in leads V4 through V6; 1 mm PT-a segment depres- . tamponade as a bleeding complication of ECLS. A repeat sternot- sion in lead II, to a lesser extent also in leads aVF, V5, and V6; and PT-a seg- omy was performed, and once the tamponade was relieved, direct ment elevation in lead aVR. The differential diagnosis included pericarditis, visual inspection as well as TOE (see Supplementary material acute coronary syndrome, or TTS. It was decided to continue with a coro- online, Video S2) demonstrated that the left ventricular function nary angiogram to exclude or treat significant obstructive coronary artery was completely recovered and no regional wall abnormalities disease. Coronary angiography did not show obstructive coronary artery were present. The patient was successfully weaned from the ECLS disease, and a left ventriculogram (Figure 2A and B,see Supplementary mate in the same session. The further course of the patient on the ICU rial online, Video S1) showed apical and mid-ventricular akinesia and basal . was complicated by atrial fibrillation, prolonged mechanical venti- hypercontractility supporting a diagnosis of TTS. . lation due to COPD exacerbation and a delirium. The patient was discharged from the hospital 30 days after admission. Seven weeks Interventions after discharge the patient was seen in the outpatient clinic and Already shortly after arrival at the catheterization laboratory, it was was doing well. concluded that the patient was in shock and a fluid challenge was initi- . ated. During diagnostic evaluation at the catheterization laboratory, . the patient received 2 L of sodium chloride (0.9%) for 45 min. In addi- . Discussion tion, phenylephrine (0.05 mg/min) was added; however, the patient . continued to deteriorate due to cardiogenic shock. The cardiothora- . Cardiogenic shock caused by TTS, as was the case in the patient we cic surgeon was consulted to perform emergency implantation of . reported here, is a difficult entity to treat. In retrospect, the initial central extracorporeal life support (ECLS). To relieve the left ven- treatment with an aggressive fluid challenge and phenylephrine seems tricle, a cannula was inserted in the left atrium, and a return cannula inappropriate. It is, however, important to keep in mind that a final was inserted in the ascending aorta (output 3.8 L/min). The haemody- diagnosis had not yet been made, and the patient was in shock (‘dry namic parameters stabilized, and the patient was transferred to the and cold’). At first, after 1 L of sodium chloride (0.9%), the patient intensive care unit (ICU). was responsive to this treatment. Therefore, it was decided to Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome 3 Figure 1 ECG taken in the catheterization laboratory: sinus rhythm 108 b.p.m., intermediate heart axis, and normal conduction times; 1 mm con- cave ST-segment elevations in leads V4 through V6; 1 mm PT-a segment depression in lead II, to a lesser extent also in leads aVF, V5, and V6; and PT- a segment elevation in aVR. Figure 2 (A) Left ventriculogram in diastole. (B) Left ventriculogram in systole demonstrating apical and mid-ventricular akinesia and basal hyper- contractility (arrows). continue fluid infusion until a final diagnosis had been made. contraindicated in TTS as they might worsen the clinical situation and Phenylephrine was added when the shock was progressive, which we prognosis by further activation of catecholamine-related pathways. believe was a reasonable choice as it is a vasopressor primarily affect- Mechanical circulatory support might be the only remaining viable ing the peripheral vasculature. In hindsight, it would have been more treatment option, which led to the decision of treatment by ECLS in appropriate to discontinue the fluid challenge after 0.5–1 L of sodium our patient. chloride (0.9%) and start an inotropic agent and/or vasopressor . when the patient further deteriorated in shock. Yet, given the final . Mechanical circulatory support diagnosis of TTS, there was no perfect treatment strategy to follow. . The choice for the type of MCS in cardiogenic shock depends on sev- Inotropic agents and/or vasopressors (e.g. dobutamine, dopamine, . eral factors, including institutional availability and experience. norepinephrine, epinephrine, and milrinone) are considered . Evidence is lacking to recommend a specific type. An intra-aortic Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 4 J.J.J. Aalberts et al. balloon pump (IABP) has been used during cardiogenic shock in TTS. and femoral venoarterial ECLS may, in the contrary, increase after- However, haemodynamic support by an IABP is usually inadequate, load. In theory, central MCS would therefore be preferable, being and the results of the IABP-SHOCK II trial, although a different neutral on afterload. However, there are no randomized trials com- patient category than patients with TTS, do not provide evidence for . paring the outcome of these devices in cardiogenic shock, let alone in 3 . the use of an IABP. Femoral venoarterial ECLS has several theoreti- . TTS-induced cardiogenic shock. We chose central ECLS with venous cal advantages: quick and relatively easy insertion, circulatory support . return from the left atrium and a return cannula in the ascending up to >4.5 L/min (depending on the cannula size) and extracorporeal . aorta, because this creates reduction of preload, is neutral on after- membrane oxygenation improves tissue oxygenation in case of pul- . load, and provides adequate haemodynamic support. monary oedema. However, afterload reduction in TTS is desirable . Systematic review of the literature To gain insight in the use of MCS for TTS-induced cardiogenic shock, Arcles idenfied by PubMed we performed a PubMed search (Figure 3) using the following search search (n=35) terms: (Takotsubo) AND (Extracorporeal Circulation or Heart- Assist Devices). Including our patient, 17 cases of TTS-induced car- Arcle with Arcles not Arcle duplicate . diogenic shock requiring MCS have been reported in the literature incomplete concerning MCS (n=1) informaon (n=1) in TTS (n=20) (Table 1). The age of the patients ranged from 16 years to 74 years, and 71% of the patients were female. Arcles included . ECLS was the most used type of MCS (82% of the cases). In- (n=13) . hospital survival was 88%, and the two deaths were not to related to . the device or TTS but were considered to result from traumatic Figure 3 Flow diagram of systematic search of literature . 7 brain injury. Although there might be a publication bias, complica- (performed 18 June 2017). Search terms: (Takotsubo) AND . tions were rare in the reported cases. In one patient, femoral venoar- (Extracorporeal Circulation or Heart-Assist Devices). . terial ECLS was complicated by leg ischaemia, which resolved without permanent injury. In our case, MCS was complicated by Table 1 List of all case reports of mechanical circulatory support in TTS-induced cardiogenic shock Study Age Sex Cause of TTS Inotropic agents/vasopressors MCS type Survival Follow-up (year) (years) before MCS .................................................................................................................................................................................................................... A (2017) 67 F Stress Phenylephrine Central ECLS left Yes 7 weeks atrium–aorta B (2016) 27 F Pheochromocytoma (Nor)epinephrine ECLS V–A Yes 1 month V R C (2016) 56 M Post-liver transplantation Not Reported TandemHeart Yes 25 days V R B (2015) 65 F Stress Norepinephrine, dobutamine, Impella 2.5 Yes 6 days dopamine C (2015) 16 M Poly-trauma Not reported ECLS V–A Yes 22 days 30 M Poly-trauma Not reported ECLS V–A Yes 46 M Poly-trauma Not reported ECLS V–A No 19 F Poly-trauma Not reported ECLS V–A No D (2015) 46 F Pheochromocytoma Norepinephrine, dobutamine ECLS V–A Yes 77 days E (2015) 31 F Medication Norepinephrine, epinephrine ECLS V–A Yes 8 days F (2014) 37 F Post-cardiac surgery Epinephrine, vasopressin ECLS V–A Yes 2 months G (2013) 74 F Unknown Not reported ECLS V–A Yes In-hospital H (2012) 45 F Stress Dobutamine, noradrenaline ECLS V–A Yes 16 days I (2012) 19 M Stress and B-cell lymphoma Not reported Extracorporeal Yes 6 months temporally central LVAD J (2011) 37 F Post-Caesarean section Norepinephrine, dobutamine, ECLS V–A Yes 6 months dopamine K (2009) 74 F Stress Norepinephrine, epinephrine ECLS V–A Yes 6 months L (2008) 51 F Pheochromocytoma Dobutamine, epinephrine ECLS V–A Yes 8 days ECLS, extracorporeal life support; F, female; LVAD, left ventricular assist device; M, male; MCS, mechanical circulatory support; V–A, venoarterial; TTS, Takotsubo syndrome. Patient described in this article. Cause or circumstances in which TTS occurred. Death not related to ECLS (traumatic brain injury). Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome 5 Fuhrmann J, Bo¨hm M, Ebelt H, Schneider S, Schuler G, Werdan K. Intraaortic cardiac tamponade. Bleeding complications occur frequently in . . balloon support for myocardial infarction with cardiogenic shock. N Engl J Med patients with MCS and is related to the use of anticoagulation. . 2012;367:1287–1296. 4. van Zwet CJ, Rist A, Haeussler A, Graves K, Zollinger A, Blumenthal S. Extracorporeal membrane oxygenation for treatment of acute inverted . takotsubo-like cardiomyopathy from hemorrhagic pheochromocytoma in late Conclusion pregnancy. A A Case Resp 2016;7:196–199. 5. Vachiat A, McCutcheon K, Mahomed A, Schleicher G, Brand L, Botha J, Sussman Inotropic agents and/or vasopressors are contraindicated in TTS- M, Manga P. Takotsubo cardiomyopathy post liver transplantation. Cardiovasc J induced refractory cardiogenic shock, making MCS a reasonable . Afr 2016;27:e1–e3. 6. Rashed A, Won S, Saad M, Schreiber T. Use of the Impella 2.5 left ventricular treatment option in this situation, and this has acceptable outcomes. . assist device in a patient with cardiogenic shock secondary to takotsubo cardio- Devices that reduce pre-load and do not increase afterload are pref- . . myopathy. BMJ Case Rep 2015; doi:10.1135/bcr-2014-208354. erable on theoretical grounds. Prospectively designed studies with . 7. Bonacchi M, Vannini A, Harmelin G, Batacchi S, Bugetti M, Sani G, Peris A. Inverted-Takotsubo cardiomyopathy: severe refractory heart failure in poly- sufficient number of patients are needed to further investigate the . trauma patients saved by emergency extracorporeal life support. Interact role of MCS in TTS-induced refractory cardiogenic shock. . . Cardiovasc Thorac Surg 2015;20:365–371. 8. Flam B. Broome´ M, Frenckner B, Bra¨nstro¨m R, Bell M. Pheochromocytome- induced inverted Takotsubo-like cardiomyopathy leading to cardiogenic shock . successfully treated with extracorporeal membrane oxygenation. J Intensive Care Supplementary material Med 2015;30:365–372. 9. Rojas-Marte G, John J, Sadiq A, Moskovits N, Saunders P, Shani J. Medication- Supplementary material is available at European Heart Journal - Case induced Takotsubo cardiomyopathy presenting with cardiogenic shock-utility of . extracorporeal membrane oxygenation (ECMO): case report and review of the Reports online. literature. Cardiovasc Revasc Med 2015;16:47–51. 10. Li S, Koerner MM, El-Banayosy A, Soleimani B, Pae WE, Leuenberger UA. Consent: Informed consent was obtained from this patient for publi- . . Takotsubo’s syndrome after mitral valve repair and rescue with extracorporeal cation of this case history and associated images in line with COPE . membrane oxygenation. Ann Thorac Surg 2014;97:1777–1778. 11. Lazzeri C, Sori A, Bernardo P, Picariello C, Gensini GF, Valente S. In-hospital recommendations. . refractory cardiac arrest treated with extracorporeal membrane oxygenation: a . tertiary single center experience. Acute Card Care 2013;15:47–51. Conflict of interest: none declared. 12. Donker DW, Pragt E, Weerwind PW, Holtkamp JW, Vainer J, Mochtar B, Maessen JG. Rescue extracorporeal life support as a bridge to reflection in fulmi- References . nant stress-induced cardiomyopathy. Int J Cardiol 2012;154:54–56. 1. Kurowski V, Kaiser A, von Hof K, Killermann DP, Mayer B, Hartmann F, 13. Zeballos C, Moraca RJ, Bailey SH, Magovern GJ Jr. Temporary mechanical circula- Schunkert H, Radke PW. Apical and midventricular transient left ventricular dys- tory support for Takotsubo cardiomyopathy secondary to primary mediastinal function syndrome (tako-tsubo cardiomyopathy): frequency, mechanisms, and B-cell lymphoma. J Card Surg 2012;27:119–121. 14. Jo YY, Park S, Choi YS. Extracorporeal membrane oxygenation in a patient with prognosis. Chest 2007;132:809–816. . . stress-induced cardiomyopathy after caesarean section. Anaesth Intensive Care 2. Lyon AR, Bossone E, Schneider B, Sechtem U, Citro R, Underwood SR, Sheppard 2011;39:954–957. MN, Figtree GA, Parodi G, Akashi YJ, Ruschitzka F, Filippatos G, Mebazaa A, 15. Bonacchi M, Maiani M, Harmelin G, Sani G. Intractable cardiogenic shock in Omerovic E. Current state of knowledge on Takotsubo syndrome: a . . stress cardiomyopathy with left ventricular outflow tract obstruction: is extra- position statement from the task force on Takotsubo syndrome of the Heart corporeal life support the best treatment? Eur J Heart Fail 2009;7:721–727. Failure Association of the European Society of Cardiology. Eur J Heart Fail 2016;18: 16. Zegdi R, Parisot C, Sleilaty G, Deloche A, Fabiani JN. Pheochromocytoma- 8–27. induced inverted Takotsubo cardiomyopathy: a case of patient resuscitation with 3. Thiele H, Zeymer U, Neumann F-J, Ferenc M, Olbrich H-G, Hausleiter J, . . extracorporeal life support. J Thorac Cardiovasc Surg 2008;135:434–435. Richardt G, Hennersdorf M, Empen K, Fuernau G, Desch S, Eitel I, Hambrecht R, Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png European Heart Journal - Case Reports Oxford University Press

Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome: a case report and review of the literature

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CASE REPORT European Heart Journal - Case Reports (2017) 1, 1–5 doi:10.1093/ehjcr/ytx005 Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome: a case report and review of the literature 1 2 2 Jan J. J. Aalberts *, Theo J. Klinkenberg , Massimo A. Mariani , and Pim van der Harst 1 2 Department of Cardiology, University of Groningen, University Medical Center Groningen, Hanzeplein 1, 9700 RB Groningen, The Netherlands; and Department of Cardiothoracic Surgery, University of Groningen, University Medical Center Groningen, Groningen, The Netherlands Received 24 June 2017; accepted 20 August 2017; online publish-ahead-of-print 13 October 2017 Abstract Takotsubo syndrome (TTS) complicated by refractory cardiogenic shock is a challenging clinical problem, as treat- ment with inotropic agents and/or vasopressors is contraindicated. We illustrate this by a patient presenting with chest pain and shortness of breath caused by TTS complicated by cardiogenic shock requiring mechanical circula- tory support (MCS). The patient received central extracorporeal life support with a cannula in the left atrium (pre- load reduction of left ventricle) and the return cannula in the ascending aorta (neutral on afterload). Treatment with MCS was complicated by a cardiac tamponade. Left ventricular function recovered after 24 h, and the patient was doing well at the outpatient clinic 7 weeks after discharge. In addition, we reviewed the literature (PubMed search) reporting on MCS in patients with TTS. Including our patient, 17 cases of TTS induced cardiogenic shock receiving MCS have been reported. Age of the patients ranged from 16 years to 74 years, and 71% of the patients were female. Extracorporeal life support was the most used type of MCS (82% of the cases). Two patients died, and complications of MCS were rare (one case of leg ischaemia). Theoretically, MCS devices that reduce pre-load and are neutral on afterload are preferable. However, no specific type of MCS can be recommended as random- ized trials are lacking. In conclusion, our case and the available literature suggests that MCS in TTS induced refrac- tory cardiogenic shock is an immediate and feasible lifesaving treatment. Keywords Takotsubo syndrome Cardiogenic shock Mechanical circulatory support Extracorporeal life • • • support Case report . Introduction Learning points . . Takotsubo syndrome (TTS) is an acute and usually reversible heart Takotsubo syndrome (TTS) complicated by cardiogenic shock is . failure syndrome with an estimated prevalence of 1–2% in patients a challenging clinical problem, as treatment with inotropic . suspected of an acute coronary syndrome. Several diagnostic crite- agents and/or vasopressors is contraindicated. ria have been proposed, and we support the criteria suggested by the Mechanical circulatory support for TTS-induced cardiogenic . Heart Failure Association of the European Society of Cardiology. shock is a reasonable treatment option and has acceptable Takotsubo syndrome is characterized by regional wall abnormalities outcomes. of the myocardium (no culprit of atherosclerotic coronary artery disease), frequently preceded by a stressful trigger (emotional of physical). The classical pattern of regional wall abnormalities is apical * Corresponding author. Tel: þ31 503 6112355, Fax: þ31 503 611347, Email: j.j.j.aalberts@umcg.nl. This case report was reviewed by Christian Fielder Camm and Matteo Cameli. V The Author 2017. Published by Oxford University Press on behalf of the European Society of Cardiology. This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 2 J.J.J. Aalberts et al. and circumferential mid-ventricular hypokinesia and basal hypercon- Timeline 1,2 tractility; however, anatomical variants exist. In the classical pattern, end-systolic the left ventricle resembles a Takotsubo, which is a . Japanese word for an octopus trap with a narrow neck and globular . lower portion. Excessive catecholamine release caused by the stress- . Time Events ful trigger are believed to play a crucial role in the pathophysiology of . ................................................................................................. 2 . TTS. Complications that can occur include mitral regurgitation, . Start of episode 67-year old woman presenting with chest pain and arrhythmias, thrombus formation, pericardial effusion, ventricular wall . shortness of breath. rupture, cardiogenic shock, and death. Treatment of refractory car- Prehospital ECG suggests ST-elevation myocardial diogenic shock in TTS is particularly challenging, as catecholamines are infarction contraindicated and mechanical circulatory support (MCS) may be • Accepted for STEMI-protocol and straight trans- the only option left. We present here such a patient and review the port to catheterization laboratory literature reporting on the treatment of TTS with MCS. Physical examination: severe distress, heartrate 107 . bpm, RR 88/63 mmHg, no signs of decompensa- Patient information . tion: Shock . Fluid challenge: 2 L NaCl 0.9% in 45 minutes A 67-year-old woman discovered that there had been a burglary in . Phenylephrine (0.05 mg/minute) started as her daughter’s house. . vasopressor Immediately she developed severe chest pain combined with LV-angiogram: Takotsubo syndrome shortness of breath and her husband called the emergency services. Cardiogenic shock Her medical history consisted of type 2 diabetes mellitus controlled Central ECLS implanted by oral drugs and chronic obstructive pulmonary disease (COPD). 24 hours later Cardiac tamponade During transport to the hospital, an electrocardiogram (ECG) was Tamponade surgically relieved taken, and ST-elevation myocardial infarction was suspected. Echocardiography: complete recovery of left ven- tricular function Physical examination . • ECLS weaned and removed On arrival at the catheterization laboratory, the patient was in severe 7 weeks later Fully recovered at the outpatient clinic distress, heart rate was 107 b.p.m., and blood pressure was 88/ 64 mmHg. Central capillary refill time was 6 s. Cardiac and pulmonary auscultation revealed no abnormalities. . Follow-up and outcomes Diagnostic assessment . About 24 h later, the patient deteriorated. A high central venous An ECG (Figure 1) was obtained showing sinus rhythm of 108 b.p.m.; inter- . pressure together with collapse of the right atrium on transoeso- mediate heart axis and normal conduction times; 1 mm concave ST- . phageal echocardiography (TOE) led to the diagnosis of cardiac segment elevations in leads V4 through V6; 1 mm PT-a segment depres- . tamponade as a bleeding complication of ECLS. A repeat sternot- sion in lead II, to a lesser extent also in leads aVF, V5, and V6; and PT-a seg- omy was performed, and once the tamponade was relieved, direct ment elevation in lead aVR. The differential diagnosis included pericarditis, visual inspection as well as TOE (see Supplementary material acute coronary syndrome, or TTS. It was decided to continue with a coro- online, Video S2) demonstrated that the left ventricular function nary angiogram to exclude or treat significant obstructive coronary artery was completely recovered and no regional wall abnormalities disease. Coronary angiography did not show obstructive coronary artery were present. The patient was successfully weaned from the ECLS disease, and a left ventriculogram (Figure 2A and B,see Supplementary mate in the same session. The further course of the patient on the ICU rial online, Video S1) showed apical and mid-ventricular akinesia and basal . was complicated by atrial fibrillation, prolonged mechanical venti- hypercontractility supporting a diagnosis of TTS. . lation due to COPD exacerbation and a delirium. The patient was discharged from the hospital 30 days after admission. Seven weeks Interventions after discharge the patient was seen in the outpatient clinic and Already shortly after arrival at the catheterization laboratory, it was was doing well. concluded that the patient was in shock and a fluid challenge was initi- . ated. During diagnostic evaluation at the catheterization laboratory, . the patient received 2 L of sodium chloride (0.9%) for 45 min. In addi- . Discussion tion, phenylephrine (0.05 mg/min) was added; however, the patient . continued to deteriorate due to cardiogenic shock. The cardiothora- . Cardiogenic shock caused by TTS, as was the case in the patient we cic surgeon was consulted to perform emergency implantation of . reported here, is a difficult entity to treat. In retrospect, the initial central extracorporeal life support (ECLS). To relieve the left ven- treatment with an aggressive fluid challenge and phenylephrine seems tricle, a cannula was inserted in the left atrium, and a return cannula inappropriate. It is, however, important to keep in mind that a final was inserted in the ascending aorta (output 3.8 L/min). The haemody- diagnosis had not yet been made, and the patient was in shock (‘dry namic parameters stabilized, and the patient was transferred to the and cold’). At first, after 1 L of sodium chloride (0.9%), the patient intensive care unit (ICU). was responsive to this treatment. Therefore, it was decided to Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome 3 Figure 1 ECG taken in the catheterization laboratory: sinus rhythm 108 b.p.m., intermediate heart axis, and normal conduction times; 1 mm con- cave ST-segment elevations in leads V4 through V6; 1 mm PT-a segment depression in lead II, to a lesser extent also in leads aVF, V5, and V6; and PT- a segment elevation in aVR. Figure 2 (A) Left ventriculogram in diastole. (B) Left ventriculogram in systole demonstrating apical and mid-ventricular akinesia and basal hyper- contractility (arrows). continue fluid infusion until a final diagnosis had been made. contraindicated in TTS as they might worsen the clinical situation and Phenylephrine was added when the shock was progressive, which we prognosis by further activation of catecholamine-related pathways. believe was a reasonable choice as it is a vasopressor primarily affect- Mechanical circulatory support might be the only remaining viable ing the peripheral vasculature. In hindsight, it would have been more treatment option, which led to the decision of treatment by ECLS in appropriate to discontinue the fluid challenge after 0.5–1 L of sodium our patient. chloride (0.9%) and start an inotropic agent and/or vasopressor . when the patient further deteriorated in shock. Yet, given the final . Mechanical circulatory support diagnosis of TTS, there was no perfect treatment strategy to follow. . The choice for the type of MCS in cardiogenic shock depends on sev- Inotropic agents and/or vasopressors (e.g. dobutamine, dopamine, . eral factors, including institutional availability and experience. norepinephrine, epinephrine, and milrinone) are considered . Evidence is lacking to recommend a specific type. An intra-aortic Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 4 J.J.J. Aalberts et al. balloon pump (IABP) has been used during cardiogenic shock in TTS. and femoral venoarterial ECLS may, in the contrary, increase after- However, haemodynamic support by an IABP is usually inadequate, load. In theory, central MCS would therefore be preferable, being and the results of the IABP-SHOCK II trial, although a different neutral on afterload. However, there are no randomized trials com- patient category than patients with TTS, do not provide evidence for . paring the outcome of these devices in cardiogenic shock, let alone in 3 . the use of an IABP. Femoral venoarterial ECLS has several theoreti- . TTS-induced cardiogenic shock. We chose central ECLS with venous cal advantages: quick and relatively easy insertion, circulatory support . return from the left atrium and a return cannula in the ascending up to >4.5 L/min (depending on the cannula size) and extracorporeal . aorta, because this creates reduction of preload, is neutral on after- membrane oxygenation improves tissue oxygenation in case of pul- . load, and provides adequate haemodynamic support. monary oedema. However, afterload reduction in TTS is desirable . Systematic review of the literature To gain insight in the use of MCS for TTS-induced cardiogenic shock, Arcles idenfied by PubMed we performed a PubMed search (Figure 3) using the following search search (n=35) terms: (Takotsubo) AND (Extracorporeal Circulation or Heart- Assist Devices). Including our patient, 17 cases of TTS-induced car- Arcle with Arcles not Arcle duplicate . diogenic shock requiring MCS have been reported in the literature incomplete concerning MCS (n=1) informaon (n=1) in TTS (n=20) (Table 1). The age of the patients ranged from 16 years to 74 years, and 71% of the patients were female. Arcles included . ECLS was the most used type of MCS (82% of the cases). In- (n=13) . hospital survival was 88%, and the two deaths were not to related to . the device or TTS but were considered to result from traumatic Figure 3 Flow diagram of systematic search of literature . 7 brain injury. Although there might be a publication bias, complica- (performed 18 June 2017). Search terms: (Takotsubo) AND . tions were rare in the reported cases. In one patient, femoral venoar- (Extracorporeal Circulation or Heart-Assist Devices). . terial ECLS was complicated by leg ischaemia, which resolved without permanent injury. In our case, MCS was complicated by Table 1 List of all case reports of mechanical circulatory support in TTS-induced cardiogenic shock Study Age Sex Cause of TTS Inotropic agents/vasopressors MCS type Survival Follow-up (year) (years) before MCS .................................................................................................................................................................................................................... A (2017) 67 F Stress Phenylephrine Central ECLS left Yes 7 weeks atrium–aorta B (2016) 27 F Pheochromocytoma (Nor)epinephrine ECLS V–A Yes 1 month V R C (2016) 56 M Post-liver transplantation Not Reported TandemHeart Yes 25 days V R B (2015) 65 F Stress Norepinephrine, dobutamine, Impella 2.5 Yes 6 days dopamine C (2015) 16 M Poly-trauma Not reported ECLS V–A Yes 22 days 30 M Poly-trauma Not reported ECLS V–A Yes 46 M Poly-trauma Not reported ECLS V–A No 19 F Poly-trauma Not reported ECLS V–A No D (2015) 46 F Pheochromocytoma Norepinephrine, dobutamine ECLS V–A Yes 77 days E (2015) 31 F Medication Norepinephrine, epinephrine ECLS V–A Yes 8 days F (2014) 37 F Post-cardiac surgery Epinephrine, vasopressin ECLS V–A Yes 2 months G (2013) 74 F Unknown Not reported ECLS V–A Yes In-hospital H (2012) 45 F Stress Dobutamine, noradrenaline ECLS V–A Yes 16 days I (2012) 19 M Stress and B-cell lymphoma Not reported Extracorporeal Yes 6 months temporally central LVAD J (2011) 37 F Post-Caesarean section Norepinephrine, dobutamine, ECLS V–A Yes 6 months dopamine K (2009) 74 F Stress Norepinephrine, epinephrine ECLS V–A Yes 6 months L (2008) 51 F Pheochromocytoma Dobutamine, epinephrine ECLS V–A Yes 8 days ECLS, extracorporeal life support; F, female; LVAD, left ventricular assist device; M, male; MCS, mechanical circulatory support; V–A, venoarterial; TTS, Takotsubo syndrome. Patient described in this article. Cause or circumstances in which TTS occurred. Death not related to ECLS (traumatic brain injury). Downloaded from https://academic.oup.com/ehjcr/article-abstract/1/2/1/4552946 by Ed 'DeepDyve' Gillespie user on 10 April 2018 Mechanical circulatory support for refractory cardiogenic shock in Takotsubo syndrome 5 Fuhrmann J, Bo¨hm M, Ebelt H, Schneider S, Schuler G, Werdan K. Intraaortic cardiac tamponade. Bleeding complications occur frequently in . . balloon support for myocardial infarction with cardiogenic shock. N Engl J Med patients with MCS and is related to the use of anticoagulation. . 2012;367:1287–1296. 4. van Zwet CJ, Rist A, Haeussler A, Graves K, Zollinger A, Blumenthal S. Extracorporeal membrane oxygenation for treatment of acute inverted . takotsubo-like cardiomyopathy from hemorrhagic pheochromocytoma in late Conclusion pregnancy. A A Case Resp 2016;7:196–199. 5. Vachiat A, McCutcheon K, Mahomed A, Schleicher G, Brand L, Botha J, Sussman Inotropic agents and/or vasopressors are contraindicated in TTS- M, Manga P. Takotsubo cardiomyopathy post liver transplantation. Cardiovasc J induced refractory cardiogenic shock, making MCS a reasonable . Afr 2016;27:e1–e3. 6. Rashed A, Won S, Saad M, Schreiber T. Use of the Impella 2.5 left ventricular treatment option in this situation, and this has acceptable outcomes. . assist device in a patient with cardiogenic shock secondary to takotsubo cardio- Devices that reduce pre-load and do not increase afterload are pref- . . myopathy. BMJ Case Rep 2015; doi:10.1135/bcr-2014-208354. erable on theoretical grounds. Prospectively designed studies with . 7. Bonacchi M, Vannini A, Harmelin G, Batacchi S, Bugetti M, Sani G, Peris A. Inverted-Takotsubo cardiomyopathy: severe refractory heart failure in poly- sufficient number of patients are needed to further investigate the . trauma patients saved by emergency extracorporeal life support. Interact role of MCS in TTS-induced refractory cardiogenic shock. . . Cardiovasc Thorac Surg 2015;20:365–371. 8. Flam B. Broome´ M, Frenckner B, Bra¨nstro¨m R, Bell M. Pheochromocytome- induced inverted Takotsubo-like cardiomyopathy leading to cardiogenic shock . successfully treated with extracorporeal membrane oxygenation. J Intensive Care Supplementary material Med 2015;30:365–372. 9. Rojas-Marte G, John J, Sadiq A, Moskovits N, Saunders P, Shani J. Medication- Supplementary material is available at European Heart Journal - Case induced Takotsubo cardiomyopathy presenting with cardiogenic shock-utility of . extracorporeal membrane oxygenation (ECMO): case report and review of the Reports online. literature. Cardiovasc Revasc Med 2015;16:47–51. 10. 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Journal

European Heart Journal - Case ReportsOxford University Press

Published: Dec 1, 2017

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