Case Report and Brief Review

Inverted Takotsubo Cardiomyopathy

Amit S. Dande, MD, Lawrence I. Fisher, MD, Mark K. Warshofsky, MD
Amit S. Dande, MD, Lawrence I. Fisher, MD, Mark K. Warshofsky, MD
ABSTRACT: Takotsubo cardiomyopathy is a transient acute left ventricular dysfunction characterized by left ventricular apical akinesis and ballooning without obstructive coronary disease described predominantly in post-menopausal women in the setting of acute emotional or physical stress. Recent reports have described isolated transient basal akinesis (inverted takotsubo cardiomyopathy) in mostly female patients with acute neurologic disorders or pheochromocytoma. We describe a rare case of a 78-year-old male with inverted takotsubo cardiomyopathy in the setting of acute abdominal pain attributed to biliary colic. A review of published literature reveals that inverted takotsubo cardiomyopathy precipitated by acute stress rather than an acute neurologic disorder appears to be an extremely rare presentation in a male patient. We discuss the relevant literature regarding incidence and reported gender distribution of inverted takotsubo cardiomyopathy.
J INVASIVE CARDIOL 2011;23:E76–E78
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Case Report. A 78-year-old male ex-smoker, with a history of hypertension, hypercholesterolemia, peripheral vascular disease and carotid artery stenosis, presented with the chief complaint of sharp, aching left-sided chest pain of a few hours duration. The chest pain was acute in onset, continuous and progressive, accompanied with mild shortness of breath but no diaphoresis, dyspnea or palpitations. He denied fever, chills, nausea, vomiting, diarrhea, constipation or dysuria. Over the preceding days, he had had some mild right upper quadrant abdominal pain that had resolved spontaneously. A screening nuclear stress test performed 8 months prior had shown small fixed apical and lateral wall defects. On initial examination, pulse was 120 bpm, blood pressure was 176/88 mmHg and he was afebrile. HEENT, neck, cardiovascular and lung exams were unremarkable. The abdomen was soft and non-distended, with mild tenderness in the right upper quadrant without guarding or rebound tenderness and normoactive bowel sounds. Murphy's sign was not present. Abnormal laboratory results were: white blood cells, 10,900/mm3 (reference range, 4–10,000/mm3), 13% monocytes (reference range, 4–12%), hemoglobin, 11.2 g/dl (reference range, 13.5–17 g/dl) and hematocrit, 33.5% (reference range, 37–50%). Serum bilirubin, alkaline phosphatase, transaminases, amylase and lipase were normal. An electrocardiogram (ECG) revealed sinus rhythm with diffuse ST depression and mild ST elevation in aVR and V1. The chest x-ray did not reveal an acute cardiopulmonary process and abdominal x-ray showed nonspecific bowel gas pattern. Initial troponin-I level was 0.18 ng/ml (reference range, ≤ 0.04 ng/ml) with normal CK. Serum D-dimer was 976 ng/ml (reference range, ≤ 500 ng/ml) and BNP was 803 pg/ml (reference range, ≤ 100 pg/ml). Abdominal computed tomography (CT) scan showed gallbladder wall thickening and biliary dilation with a large gallstone in the lumen of the gallbladder. Because of the new ECG abnormality suggestive of left main or proximal left anterior descending artery stenosis, abnormal troponin and multiple coronary artery disease risk factors, he was treated for acute coronary syndrome and underwent urgent cardiac catheterization that revealed nonobstructive coronary artery disease. Left ventriculography showed an akinetic base with hypercontractile apex. His anticoagulation was discontinued while he remained on an oral beta-blocker and ACE-inhibitor. Cardiac enzymes showed further rise with peak CK of 329 (reference range, 0–170 U/L), CK-MB of 7.6 (reference range, ≤ 5 ng/ml), and TnI of 1.08 ng/ml (reference range, ≤ 0.04 ng/ml). His chest pain and dyspnea gradually improved. His ECG also improved, although it remained mildly abnormal at discharge. A V/Q scan found low probability for pulmonary embolism and abdominal ultrasound confirmed the CT scan findings. His abdominal pain and leukocytosis resolved spontaneously and he was discharged home in stable condition with a diagnosis of "inverted" takotsubo cardiomyopathy in the setting of acute abdominal pain presumed to be biliary colic. At a follow-up visit at 1 month, ECG had reverted to baseline and an echocardiogram revealed complete resolution of wall motion abnormalities. Discussion. Stress cardiomyopathy or takotsubo cardiomyopathy was initially described in 1991 by Satoh et al1 as a transient cardiomyopathy precipitated by acute emotional or physical stress where angiography revealed normal coronary arteries and left ventriculography revealed apical akinesis and/or ballooning with a hypercontractile base. The overall incidence is reported to be around 1–2% of troponin-positive acute coronary syndrome patients,2 and postmenopausal women make up an overwhelming majority of cases. A variant was subsequently described with mid-ventricular akinesis and hypercontractile apex and base.3,4 Thereafter, reports5,6 of patients admitted with acute coronary syndrome and antecedent stressful events described patients with basal and midventricular akinesis. In one series, 40% of all takotsubo cases (n = 35) had mid-ventricular dysfunction,2 with only 2 male patients (1 with apical and 1 with mid-ventricular variant). This report also compared demographic, clinical, laboratory and angiographic parameters between the 2 groups, and found no significant differences. In 2005, a case series7 described a pattern of isolated left ventricular basal hypokinesis with normal apical contractility (labeled 'inverted tako-tsubo') in 4 patients with acute cerebral disorders, all of whom had normal coronary arteries or non-obstructive coronary disease. In a prospective study of cardiac wall motion abnormalities in 173 patients with subarachnoid hemorrhage,8 echocardiography revealed basal and midventricular akinesis to be more common than apical akinesis. In a review of stress-related cardiomyopathy syndromes,9 isolated left ventricular basal dysfunction was recognized as a morphologic variant of both stress-induced cardiomyopathy as well as neurogenic stress cardiomyopathy. Pathology findings10 from a case of "neurogenic" inverted cardiomyopathy in a patient with acute subarachnoid hemorrhage described sparse foci of myocyte necrosis with contraction bands in the akinetic areas of the myocardium. Contraction band necrosis is caused by massive catecholamine release and this finding is similar to autopsy findings from stress cardiomyopathy patients,11 suggesting a similar pathogenesis for the 2 presentations. Differential expression of beta-adrenergic receptors in the apex compared to the base has been postulated to explain the wall motion abnormality in stress cardiomyopathy. However, patients have been reported to develop a different morphologic variant in rare recurrences.12 In our review of recent literature, the inverted tako-tsubo pattern has been reported predominantly in women with pheochromocytoma13–21 or acute neurologic disorders, such as cerebral hemorrhage or head injury.7,22–24 Sporadic reports of non-neurogenic presentations (presumably in the absence of antecedent stress), such as during infusion of catecholamines,6,25,26 amphetamine abuse,27 alcoholic pancreatitis,28 sepsis, post-partum29 and with anagrelide infusion,30 have been published in the last few years; all patients reported were women. In a large review (n = 107) of stress cardiomyopathy,31 a 1% incidence of basal/midventricular akinesis was reported, but the gender distribution was not specified. A recently published review comparing clinical characteristics of apical and non-apical variants of non-neurogenic stress cardiomyopathy32 reported a higher rate of heart failure and complications in patients with the apical variant. Notably, in a total sample size of 38 patients, all 9 patients with the non-apical variant were females. A recently published case report described a 30-year-old male with pheochromocytoma with inverted takotsubo pattern on left ventriculography.33 The patient we describe presented with inverted takotsubo cardiomyopathy in the absence of an acute neurologic disorder or pheochromocytoma. Although neurogenic/pheochromocytoma-related stress cardiomyopathy has been reported in males (also rare), we could find only 1 prior report34 that described a 21-year-old male with acute appendicitis who developed inverted takotsubo cardiomyopathy. Thus, this appears to be an extremely rare presentation in a male patient. Interestingly, the only identifiable antecedent stressor in this patient was mild abdominal discomfort prior to presentation presumed to be biliary colic.

References

  1. Dote K, Sato H, Tateishi H, et al. Myocardial stunning due to simultaneous multivessel coronary spasms: A review of 5 cases. J Cardiol 1991;21:203–214.
  2. Kurowski V, Kaiser A, von Hof K, et al. Apical and midventricular transient left ventricular dysfunction syndrome (tako-tsubo cardiomyopathy): Frequency, mechanisms, and prognosis. Chest 2007;132:809–816.
  3. Bonnemeier H, Schafer U, Schunkert H. Apical ballooning without apical ballooning. Eur Heart J 2006;27:2246.
  4. Hurst RT, Askew JW, Reuss CS, et al. Transient midventricular ballooning syndrome: A new variant. J Am Coll Cardiol 2006;48:579–583.
  5. Haghi D, Papavassiliu T, Fluchter S, et al. Variant form of the acute apical ballooning syndrome (takotsubo cardiomyopathy): Observations on a novel entity. Heart 2006;92:392–394.
  6. Reuss CS, Lester SJ, Hurst RT, et al. Isolated left ventricular basal ballooning phenotype of transient cardiomyopathy in young women. Am J Cardiol 2007;99:1451–1453.
  7. Ennezat PV, Pesenti-Rossi D, Aubert JM, et al. Transient left ventricular basal dysfunction without coronary stenosis in acute cerebral disorders: A novel heart syndrome (inverted takotsubo). Echocardiography 2005;22:599–602.
  8. Banki N, Kopelnik A, Tung P, et al. Prospective analysis of prevalence, distribution, and rate of recovery of left ventricular systolic dysfunction in patients with subarachnoid hemorrhage. J Neurosurg 2006;105:15–20.
  9. Bybee KA, Prasad A. Stress-related cardiomyopathy syndromes. Circulation 2008;118:397–409.
  10. Marechaux S, Fornes P, Petit S, et al. Pathology of inverted takotsubo cardiomyopathy. Cardiovasc Pathol 2008;17:241–243.
  11. Nef HM, Mollmann H, Kostin S, et al. Tako-tsubo cardiomyopathy: Intraindividual structural analysis in the acute phase and after functional recovery. Eur Heart J 2007;28:2456–2464.
  12. Ikeda E, Hisamatsu K, Kijima Y, et al. Morphologically unique feature of recurrent ampulla (takotsubo) cardiomyopathy. Circ J 2009;73:371–375.
  13. Sanchez-Recalde A, Costero O, Oliver JM, et al. Images in cardiovascular medicine. Pheochromocytoma-related cardiomyopathy: Inverted takotsubo contractile pattern. Circulation 2006;113:E738–E739.
  14. Di Valentino M, Balestra GM, Christ M, et al. Inverted takotsubo cardiomyopathy due to pheochromocytoma. Eur Heart J 2008;29:830.
  15. Sanchez-Recalde A, Iborra C, Costero O, et al. Takotsubo cardiomyopathy — A new variant and widening disease spectrum. "Inverted takotsubo" pattern related to catecholamine-toxicity. Int J Cardiol 2009;132:437–438; Author reply, pp. 438–439.

  1. Zegdi R, Parisot C, Sleilaty G, et al. Pheochromocytoma-induced inverted takotsubo cardiomyopathy: A case of patient resuscitation with extracorporeal life support. J Thorac Cardiovasc Surg 2008;135:434–435.
  2. Kimura S, Mitsuma W, Ito M, et al. Inverted takotsubo contractile pattern caused by pheochromocytoma with tall upright T-waves, but not typical deep T-wave inversion. Int J Cardiol 2010;139:E15–E17.
  3. Jindal V, Baker ML, Aryangat A, et al. Pheochromocytoma: Presenting with regular cyclic blood pressure and inverted takotsubo cardiomyopathy. J Clin Hypertens (Greenwich) 2009;11:81–86.
  4. Gervais MK, Gagnon A, Henri M, Bendavid Y. Pheochromocytoma presenting as inverted takotsubo cardiomyopathy: A case report and review of the literature. J Cardiovasc Med (Hagerstown) 2010 Feb 11.
  5. Kim S, Yu A, Filippone LA, et al. Inverted-takotsubo pattern cardiomyopathy secondary to pheochromocytoma: A clinical case and literature review. Clin Cardiol 2010;33:200–205.
  6. Di Palma G, Daniele GP, Antonini-Canterin F, et al. Cardiogenic shock with basal transient left ventricular ballooning (takotsubo-like cardiomyopathy) as first presentation of pheochromocytoma. J Cardiovasc Med (Hagerstown) 2010;11:507–510.
  7. Kim HS, Chang WI, Kim YC, et al. Catecholamine cardiomyopathy associated with paraganglioma rescued by percutaneous cardiopulmonary support: Inverted takotsubo contractile pattern. Circ J 2007;71:1993–1995.
  8. Marechaux S, Goldstein P, Girardie P, Ennezat PV. Contractile pattern of inverted takotsubo cardiomyopathy: Illustration by two-dimensional strain. Eur J Echocardiogr 2009;10:332–333.
  9. Riera M, Llompart-Pou JA, Carrillo A, Blanco C. Head injury and inverted takotsubo cardiomyopathy. J Trauma 2010;68:E13–E15.
  10. Collen J, Bimson W, Devine P. A variant of takotsubo cardiomyopathy: A rare complication in the electrophysiology lab. J Invasive Cardiol 2008;20:E310–E313.
  11. Subramaniam A, Cooke JC, Ernest D. "Inverted" tako-tsubo cardiomyopathy due to exogenous catecholamines. Crit Care Resusc 2010;12:104–108.
  12. Movahed MR, Mostafizi K. Reverse or inverted left ventricular apical ballooning syndrome (reverse takotsubo cardiomyopathy) in a young woman in the setting of amphetamine use. Echocardiography 2008;25:429–432.
  13. Van de Walle SO, Gevaert SA, Gheeraert PJ, et al. Transient stress-induced cardiomyopathy with an "inverted takotsubo" contractile pattern. Mayo Clin Proc 2006;81:1499–1502.
  14. Lee S, Lee KJ, Yoon HS, et al. Atypical transient stress-induced cardiomyopathies with an inverted takotsubo pattern in sepsis and in the postpartal state. Tex Heart Inst J 2010;37:88–91.
  15. Proietti R, Rognoni A, Ardizzone F, et al. Atypical takotsubo syndrome during anagrelide therapy. J Cardiovasc Med (Hagerstown) 2009;10:546–549.

  1. Singh NK, Rumman S, Mikell FL, et al. Stress cardiomyopathy: Clinical and ventriculographic characteristics in 107 North American subjects. Int J Cardiol 2009 Jan 18.
  2. Jabara R, Gadesam R, Pendyala L, et al. Comparison of the clinical characteristics of apical and non-apical variants of "broken heart" (takotsubo) syndrome in the United States. J Invasive Cardiol 2009;21:216–222.
  3. Kim S, Yu A, Filippone LA, et al. Inverted-takotsubo pattern cardiomyopathy secondary to pheochromocytoma: A clinical case and literature review. Clin Cardiol 2010;33:200–205.
  4. Marti V, Carreras F, Pujadas S, De Rozas JM. Transient left ventricular basal ballooning-"inverted" tako-tsubo. Clin Cardiol 2009;32:E20–E21.
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From the Division of Cardiology, Danbury Hospital, Danbury, Connecticut. The authors report no conflicts of interest regarding the content herein. Manuscript submitted June 16, 2010, provisional acceptance given July 26, 2010, final version accepted August 30, 2010. Address for correspondence: Mark K. Warshofsky, MD, Danbury Hospital, 24 Hospital Ave., Danbury, CT 06810. Email: mark.warshofsky@danhosp.org