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Takotsubo Cardiomyopathy Triggered by a Fatal Subarachnoid Hemorrhage

  • Wael MajdoubEmail author
  • Amal Mosbahi
  • Mariem Beji
  • Sarra Gharsalloui
  • Tasnim Masmoudi
  • Elyes Turki
Medicine
  • 39 Downloads
Part of the following topical collections:
  1. Topical Collection on Medicine

Case Report

A 43-year-old man with no history of cardiac disease was transferred, by his relatives, to the Emergency Department after he lost consciousness. Upon admission, the patient appeared disoriented (Glasgow Coma Scale of 6) without any focal neurologic signs. His blood pressure was 95/50 mmHg, the pulse rate was regular and accelerated at around 110 beats per minute (bpm), and the respiratory rate was 36 breaths per minute. The patient was rapidly intubated, and the cranial CT scan revealed a subarachnoid hemorrhage (SAH) of an average abundance (Fig. 1). Cerebral angiography showed no aneurysmal image (Fig. 2). Twenty hours later, the patient was extubated and developed severe chest pain, followed by syncope. Bilateral crackles were heard on auscultation. A 12-lead electrocardiogram showed sinus rhythm, a diffuse planning R wave, and flattening T (Fig. 3). The chest x-ray showed important pulmonary edema without cardiac enlargement. The troponin I was raised at 3.45 μg/L (normally inferior to 0.02 μg/L), and the cardiac ultrasound showed a left ventricular (LV) basal akinesis with a diminished LV ejection fraction of 42% and a preserved apex contractility (Fig. 4). The diagnosis of takotsubo cardiomyopathy was suspected. The patient was treated with a diuretic. He made a successful recovery of the cardiopulmonary function and by 2 days of admission, the chest x-ray and ECG were normal. Follow-up echocardiography, performed after 1 week of admission, showed a normal appearance of the LV and the return of a normal systolic function with an ejection fraction of 70%. Troponin I level dropped to 0.32 μg/L in 4 days of admission and became normal in 12 days. A coronary angiogram could not be performed. However, there were sudden neurological complications and the patient passed away 2 weeks after his admission to the hospital. An autopsy was performed, 10 h after his death, and it revealed cerebral edema and subarachnoid and intraventricular hemorrhage in association with tonsillar herniation. The heart weighed 357 g. There were no obstructive lesions in the coronary arteries or any myocardial bridging on their way. No valvular abnormalities were noted. Apart from a subepicardial pallor, there were no myocardial abnormalities in the heart cross-sections (Fig. 5). The myocardium histological sections did not reveal any necrosis or specific inflammatory infiltrate but noted an interstitial edema (Fig. 6).
Fig. 1

Non-contrast cranial computed tomography showing an acute subarachnoid hemorrhage

Fig. 2

Cerebral angiography did not show any aneurismal image

Fig. 3

Twelve-lead electrocardiogram showing planning R waves and flattening T waves

Fig. 4

Cardiac echography showing a left ventricular basal akinesis with a preserved apex contractility

Fig. 5

Cross-sections of the heart did not show any myocardial abnormalities

Fig. 6

Myocardium histological sections showing interstitial edema without any other abnormalities

The autopsy showed that death was related to a SAH and confirmed the diagnosis of a takotsubo cardiomyopathy.

Discussion

Stress-induced or takotsubo cardiomyopathy (TTC) is a recognized disease that was firstly described in Japan in 1991 in patients presenting signs of an acute coronary syndrome with transient left ventricular (LV) failure [1, 2, 3]. TTC is typically associated with sudden emotional stress, accompanied by a catecholamine surge. Triggering events may also include physical stressors such as major surgery medical procedures and illnesses [1, 2, 3, 4, 5]. This hypersympathetic state can be seen in SAH which is no longer considered as an exclusion criterion for takotsubo diagnosis according to the recent consensus document updating the Mayo Clinic guidelines and the InterTAK Diagnostic Criteria [2, 3, 4, 6].

In fact, intracerebral high pressure usually observed in cases of SAH can lead to an upregulation of the sympathetic system to assure brain perfusion. Therefore, it has been reported that 61.9% of patients with SAH will present with an abnormal ECG and that 20 to 30% of patients will develop secondary cardiomyopathy [7]. Furthermore, transitory adrenergic activation can explain the occurrence of TTC which is a known complication of SAH.

The prevalence of TTC in SAH ranges between 1.2 and 26% [8]. SAH patients with a high Hunt–Hess score (HHS) or low Glasgow Coma Scale (GCS) are more likely to develop TTC than those with low HHS or high GCS like our case [7].

The recent diagnostic criteria for TTC include transient hypokinesis, akinesis, or dyskinesis in the LV mid-wall segments with or without apical involvement; new ECG abnormalities or modest elevation in cardiac troponin; and the absence of myocarditis [2, 3, 6, 9].

TTC affects mainly the mid-wall and apical segments of the LV. However, when TTC is triggered by SAH, the most common abnormal myocardial segments are the basal and midventricular portions [4, 9] as seen in our case.

TTC affects predominantly postmenopausal women with a mean age of 68 years [3, 5]. Nevertheless, similar to our case, cases with an inverted pattern of TTC associated with SAH described in the literature tend to be younger and were most often associated with severe neurological pathologies and related to “neurologic stress” [3, 4, 7, 10].

The diagnosis of TTC is based on ultrasounds. In fact, some reported cases were diagnosed on the basis of echocardiographic signs without performing a cardiac angiogram. In these cases, patients presented cerebral lesions, ECG changes, high cardiac enzyme levels, and sometimes chest pain [4, 11].

In our case, TTC was suspected on the basis of clinical data (ECG changes, elevation of the cardiac enzyme, and akinesis of the basal walls of the LV). In the absence of cardiac angiogram, autopsy findings showed neither a coronary lesion nor any other anatomical abnormalities, and the histological examination did not reveal any myocarditis.

Most myocardial biopsies in TTC have shown interstitial infiltrates consisting primarily of mononuclear lymphocytes and macrophages; myocardial fibrosis; and contraction bands with or without myocardial necrosis [12]. These abnormalities may be reversible [13].

In our case, no cellular lesion was noted in different histological sections. This allowed us to exclude any other myocardial damage as myocarditis, thus retaining the diagnosis of TTC and confirming the complete histological reconstitution.

The outcome of the TTC triggered by SAH is favorable in almost two thirds of the cases but fatal in one third [7, 14].

The original point in this case is that the TTC is inverted, described in a male patient associated with a SAH with a fatal outcome.

Notes

Compliance with Ethical Standards

Ethical Approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.

Conflict of Interest

The authors declare that they have no conflict of interest.

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Copyright information

© Springer Nature Switzerland AG 2019

Authors and Affiliations

  1. 1.Department of Forensic MedicineIbn El Jazzar University HospitalKairouanTunisia
  2. 2.Department of Forensic MedicineFarhat Hached University HospitalSousseTunisia

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