Takotsubo Syndrome Following Emergency Decompression for Acute Cervical Spinal Epidural Hematoma: A Case Report

Introduction

A spinal epidural hematoma is a hemorrhage occurring in the spinal epidural space that can result from traumatic, nontraumatic, or iatrogenic causes [1]. Approximately 40% to 50% of cases have unknown causes and are categorized as spontaneous [2]. The incidence of spontaneous spinal epidural hematoma is approximately 0.1 per 100 000 individuals [1]. Patients typically present with sudden neck or back pain followed by rapidly progressive neurological deficits. Urgent surgical decompression is generally recommended for patients with severe or progressive neurological deficits, whereas conservative management may be appropriate for mild or improving cases [1,2].

Takotsubo syndrome (TTS), also referred to as stress cardiomyopathy, is characterized by transient regional left ventricular systolic dysfunction that typically extends beyond a single coronary artery territory and is often triggered by emotional or physical stress [3–5]. Proposed mechanisms include sympathetic overactivation with catecholamine excess, microvascular dysfunction, and myocardial stunning [4,5].

Acute neurologic disorders have been recognized as potential triggers of TTS through neurocardiac interactions, including autonomic imbalance and sympathetic overactivation. In patients with cervical and upper thoracic spinal pathology, the proximity to sympathetic outflow pathways may provide a biologically plausible substrate for stress cardiomyopathy in addition to nonspecific perioperative stress [4,5]. However, detailed perioperative descriptions of TTS following decompressive surgery for cervical spinal epidural hematoma remain limited, and postoperative myocardial injury after noncardiac surgery is an important competing diagnosis in this setting [6,7].

We report a rare case of postoperative TTS identified on postoperative day (POD) 1 following emergency decompression for acute cervical spinal epidural hematoma triggered by minor neck rotation. We further discuss the potential contributions of combined perioperative and neurogenic stress and the clinical approach to differentiating TTS from myocardial injury after noncardiac surgery in the early postoperative period.

Case Report

A 79-year-old Japanese woman presented to our hospital with quadriparesis. A few hours before admission, she experienced mild neck pain while rotating her neck. Although she initially remained ambulatory, she developed progressive weakness after lying down and was brought to the emergency department. Her medical history included type 2 diabetes mellitus and hypertension. Her medications included antihypertensives and oral antidiabetic drugs, but no anticoagulant or antiplatelet therapy. Preoperative evaluation revealed no prior cardiac symptoms, and a preoperative electrocardiogram (ECG) showed normal results. Cardiac biomarkers were not routinely measured preoperatively because the patient had no cardiac symptoms and a normal ECG.

On arrival, her vital signs were as follows: blood pressure, 100/52 mmHg; heart rate, 60 beats per minute (regular); respiratory rate, 20 breaths per minute; oxygen saturation, 92% on 2 L/min nasal cannula; and body temperature, 36.4°C. Neurological examination revealed marked weakness in all 4 limbs (manual muscle testing: upper extremities, 1/2; lower extremities, 1/1), with preserved sensory function, anal sphincter relaxation, and hyperreflexia, including bilateral Babinski signs. Initial head computed tomography (CT) showed no abnormalities, and cervical CT revealed no evidence of fracture. Cervical spine magnetic resonance imaging (MRI) demonstrated a dorsal epidural hematoma extending from C2 to T3 without intramedullary signal changes (Figure 1). Laboratory findings showed no abnormalities: hemoglobin 12.9 g/dL, platelet count 210 000/μL, prothrombin time 89%, and D-dimer 0.9 μg/mL, indicating no bleeding tendency or coagulopathy.

An acute cervical spinal epidural hematoma was diagnosed, and an emergency right-sided hemilaminectomy at the C2–C5 levels with hematoma evacuation was performed. The operative time was 69 minutes, and estimated blood loss was 50 mL. Anesthesia was maintained with propofol and remifentanil. Transient intraoperative hypotension occurred and was treated with intermittent phenylephrine boluses and a norepinephrine infusion. The hemodynamic parameters subsequently stabilized, and the norepinephrine infusion was discontinued by the end of surgery. No sustained hypoxemia was documented. Postoperatively, the patient’s manual muscle testing grades improved to grades 3–4 in all extremities. Neurologic function improved immediately after surgery, whereas new electrocardiographic abnormalities were first documented on the morning of POD 1.

At that time, new T-wave inversions were observed in leads V3 to V6 despite the absence of chest pain (Figure 2). The troponin I level was elevated (1826 pg/mL), and no electrolyte abnormalities were identified. Transthoracic echocardiography (TTE) demonstrated apical akinesis to severe hypokinesis of the left ventricle with basal hyperkinesis (Figure 3, Video 1). Urgent coronary angiography revealed stenoses in segments 2, 6, and 8; however, no flow-limiting or culprit lesion was identified, and revascularization was not performed (Figure 4). Alternative causes – including acute coronary syndrome, myocarditis, pulmonary embolism, and acute aortic syndrome – were carefully evaluated and considered unlikely based on angiographic findings, the non-territorial wall-motion pattern, and subsequent improvement in the patient’s condition. These findings fulfilled the diagnostic criteria for TTS. Anticoagulation therapy was withheld owing to the potential risk of bleeding. Follow-up TTE showed no evidence of left ventricular thrombus.

On the day following coronary angiography, the troponin I level declined to 1134 pg/mL. During hospitalization, serial TTE revealed gradual improvement of wall-motion abnormalities, whereas T-wave inversions persisted on ECG in the precordial leads. Despite these persistent cardiac abnormalities, her neurologic function progressively improved. The patient exhibited no signs of heart failure, and no further cardiac interventions were required. Her limb strength progressively improved; however, fine motor dysfunction persisted but gradually improved. She was transferred to a rehabilitation facility on POD 16 and discharged on POD 42. A clinical timeline is provided in Table 1.

Discussion

LIMITATIONS:

This report describes a single case; therefore, causality between the spinal pathology, surgical intervention, and TTS cannot be established. Unmeasured perioperative factors, including anesthetic depth, catecholamine levels, or transient hypotension, may also have contributed. Additionally, the literature search was limited to PubMed and may not have captured all relevant reports. Nevertheless, the temporal association and anatomical considerations offer clinically relevant insight into potential neurogenic and perioperative triggers of TTS.

Conclusions

Postoperative TTS following emergency decompressive surgery for acute cervical spinal epidural hematoma appears to be uncommon but clinically important. This case highlights the potential contribution of combined neurogenic and perioperative stress to stress-induced cardiomyopathy. Clinicians should remain vigilant for possible myocardial dysfunction in high-risk patients and consider early cardiac evaluation to facilitate timely diagnosis and appropriate management.