Takotsubo cardiomyopathy triggered by frequent psychogenic nonepileptic seizures: A case report

Abstract

Background

Coexistence of Takotsubo cardiomyopathy and psychogenic nonepileptic seizures has rarely been reported. Herein, we report a case of Takotsubo cardiomyopathy triggered by psychogenic nonepileptic seizures.

Case Presentation

A 50‐year‐old woman with a 22‐year history of dissociative and panic disorders and a 7‐year history of seizures increasing in frequency was admitted due to cardiogenic shock. Based on the left ventriculography and electroencephalography findings, she was diagnosed with Takotsubo cardiomyopathy and psychogenic nonepileptic seizures. Seizures were controlled using antipsychotic agents, resulting in improved cardiac function, and she was discharged. However, she died of cardiopulmonary arrest 9 days after discharge.

Conclusion

Since controlling psychogenic epileptic seizures is difficult, Takotsubo cardiomyopathy triggered by psychogenic nonepileptic seizures may have poor prognosis, requiring careful management and close monitoring.

Takotsubo cardiomyopathy is a transient systolic dysfunction caused by emotional or physical stress. Psychogenic nonepileptic seizures mimic epileptic seizures without abnormal neuronal activity. Coexistence of these two conditions has rarely been reported. Takotsubo cardiomyopathy triggered by psychogenic nonepileptic seizures may have poor prognosis, requiring careful management and close monitoring.

INTRODUCTION

Takotsubo cardiomyopathy (TCMP) is a syndrome characterized by transient regional systolic dysfunction triggered by emotional or physical stress. ¹ Psychogenic nonepileptic seizures (PNES) that mimic epileptic seizures and syncope without abnormal neuronal activity. ² Although neurological dysfunction, such as epileptic seizures, is a risk factor for TCMP, ³ cases of TCMP triggered by PNES have rarely been reported.

CASE REPORT

A 50‐year‐old Japanese woman was brought to our hospital for chest discomfort. She had a 22‐year history of dissociative and panic disorders, and a 7‐year history of recurrent seizures with an unknown cause despite multiple evaluations. The frequency of seizures had increased recently. At the time of admission, she was taking tizanidine.

Her body height was 150.5 cm and her body weight was 43.0 kg. On physical examination, her body temperature was 35.1°C, blood pressure was 62/43 mmHg with marked peripheral coldness, heart rate was 108 bpm, and oxygen saturation was 85% at room air. She had elevated troponin‐T (0.751 ng/mL) and lactate levels (3.5 mmol/L). Chest roentgenography showed generally normal findings. Electrocardiography showed no significant ST‐T changes (Figure 1A). Emergency coronary angiography revealed no coronary artery abnormalities. Left ventriculography showed midventricular type TCMP, with preserved contraction of the apex wall and severe hypokinesis of the mid‐ventricle wall (Figure 2).

FIGURE 1.

Electrocardiography findings on admission. No significant abnormalities are observed.

FIGURE 2.

Left ventriculography findings on admission. Images obtained in the systolic and diastolic phases show global hypokinesia with involvement from the left ventricular septum to the apex.

The patient was diagnosed with TCMP and admitted to the intensive care unit (ICU). She recovered from cardiogenic shock with dobutamine administration but experienced recurrent seizure episodes. As there were no epileptiform waves on electroencephalography, the seizures were considered to be PNES. Following flunitrazepam and risperidone initiation, the seizure frequency decreased and cardiac function improved. She was weaned off dobutamine on hospital day 4 and discharged from the ICU on day 5. Serial electrocardiographic images are shown in Figure 1B–D.

Although the seizures had not completely resolved, hospitalization‐related psychological stress was considered an exacerbating factor, and the patient was discharged on hospital day 9. She was scheduled for follow‐up with the cardiologist 2 weeks after discharge. However, 9 days after discharge, she was transported to a nearby hospital due to cardiopulmonary arrest with an initial waveform of pulseless electrical activity. She underwent extracorporeal cardiopulmonary resuscitation, and no evidence of coronary artery lesions was noted on subsequent coronary angiography. Echocardiography showed diffuse severe wall motion hypokinesis with preserved contraction of the apex wall. Based on these findings and the disease course, TCMP relapse was considered the cause of cardiopulmonary arrest. She died of hypoxic encephalopathy.

DISCUSSION

We presented a rare case of TCMP triggered by PNES. Our patient developed cardiogenic shock due to TCMP, which was considered to be induced by the frequent PNES.

TCMP, also called stress‐induced cardiomyopathy, is a syndrome characterized by transient regional systolic dysfunction of the left ventricle without angiographic evidence of obstructive coronary artery disease or acute plaque rupture. The disease's pathogenesis is not well understood; however, catecholamine excess and microvascular dysfunction might be contributing factors. ¹ TCMP is triggered by emotional or physical stress. According to the International Takotsubo Registry study, 36% of patients experienced physical stress, such as acute respiratory failure, a central nervous system event, or infection. ⁴ Clinically, TCMP often resembles acute myocardial infarction (AMI) based on electrocardiographic and wall motion abnormalities and elevated troponin levels. In fact, 1%–2% of patients with suspected AMI are considered to have TCMP. Our patient was admitted with cardiogenic shock and is suspected to have AMI due to elevated troponin‐T levels and markedly decreased left ventricular wall motion. However, as no coronary artery abnormalities were detected, we diagnosed TCMP.

PNES clinically mimic epileptic seizures or syncope but are not associated with pathological changes in brain physiology. Although the pathogenesis of PNES remains unclear, it may be an adaptive response to intolerable events. ² PNES are more prevalent in women than in men and are common in patients with psychiatric diseases. ² Several clinical characteristics differentiate PNES from tonic–clonic epileptic seizures, including onset in witnessed situations, no onset during sleep, relatively long duration, and rapid recovery of consciousness. PNES is diagnosed based on these characteristics and the absence of epileptiform activity on electroencephalography. Our patient's seizures were clinically consistent with PNES, and she had a history of dissociative and panic disorders. However, the cause of the increased frequency of seizures was unknown.

Cases of PNES combined with TCMP have rarely been reported. Our PubMed search using the terms “psychogenic nonepileptic seizures” and “takotsubo cardiomyopathy” yielded only one study published in English. ⁵ Conversely, several studies have reported an association between TCMP and neurological dysfunction, such as ischemic stroke, subarachnoid hemorrhage, and epilepsy. The reported prevalence of TCMP was 15%–20% among patients with subarachnoid hemorrhage and approximately 1% among those with ischemic stroke. ⁶ The prevalence of TCMP in patients with seizures is approximately 0.1%. ⁶ Although the pathophysiology of seizure‐associated TCMP is less understood, patients with prolonged seizures have increased levels of noradrenaline, which may contribute to TCMP development. ⁷ In our case, seizures were recurring within a short period before admission, suggesting persistently high blood catecholamine levels, which could have led to TCMP onset.

PNES control is difficult, with many patients experiencing recurring seizures despite treatment. In our case, seizures were partially controlled by medications, leading to TCMP recurrence. The yearly risk of TCMP recurrence is 1%–2%. Common risk factors associated with recurrence have not been established. However, the prognosis may be poorer in situations where the disease that caused TCMP is a comorbidity. The other factors for poorer prognosis of TCMP include age >70 years, presence of physical stressor, and left ventricular ejection fraction <40%. ⁸ In a series of 1613 cases, the highest mortality rate was observed in patients with a physical trigger, including neurological disorders. ⁹ Although TCMP recurrence is uncommon, when the causative factors persist, there is a risk of recurrence and poor prognosis. On the other hand, cardiovascular disease is one of the leading causes of death in patients with PNES. In a study of 674 patients, circulatory system diseases accounted for 22.9% of all deaths in patients >50 years old, and sudden unexpected death accounted for 12.7%. ¹⁰ Furthermore, recent studies have reported significantly higher mortality rates in patients with PNES than in the general population, even if suicide and injury are excluded as causes. ¹⁰ Thus, clinicians should be aware that PNES prognosis is unpredictable, with an unfortunate course in some cases, as in our case. Appropriate systemic evaluation and close follow‐up are important in patients with PNES, as well as ensuring a multidisciplinary approach including emergency physicians, cardiologists, and psychiatrists.

CONCLUSION

Since controlling PNES is difficult even with adequate treatment, the coexistence of both conditions may pose a high risk of TCMP recurrence and poor prognosis. Therefore, careful management and close monitoring are crucial for PNES.

AUTHOR CONTRIBUTIONS

D.Y., H.K., S.H., and Y.K. were involved in patient management. D.Y. drafted the manuscript. H.K., T.M., S.M., S.H., and Y.K. revised the manuscript for important intellectual content. All authors have read and approved the final version of the manuscript.

CONFLICT OF INTEREST STATEMENT

The authors declare no conflicts of interest.

ETHICS STATEMENT

Approval of the research protocol: N/A.

Informed consent: Written informed consent was obtained from the patient's family for the publication of this case report and accompanying data.

Registry and the registration no. of the study/trial: N/A.

Animal studies: N/A.

Yomogida D, Kuwano H, Miyakoshi T, Mizuta S, Horikawa S, Koshida Y. Takotsubo cardiomyopathy triggered by frequent psychogenic nonepileptic seizures: A case report. Acute Med Surg. 2024;11:e941. 10.1002/ams2.941

DATA AVAILABILITY STATEMENT

No new data were created or analyzed in this study.