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We report a case of a middle-aged female who presented with altered mental status, hypotension, and hypoglycemia and was diagnosed with secondary adrenal insufficiency. She was also found to have elevated troponin I on initial evaluation with diffuse T wave inversions on electrocardiogram. Transthoracic echocardiogram revealed ejection fraction of...
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Citations
... The paradox in the index case is the development of TCM in a patient with adrenal insufficiency, wherein in the absence of steroid hormones, the toxic effects of sympathetic catecholamines were realized. Indeed, there have been only very few case reports documenting this association to date [15][16][17]. Pathophysiological mechanism of this association is largely unknown, but certain theories have been hypothesized. In adrenalectomized animal models, there was a toxic effect of catecholamines on the cardiac tissue when they were exposed to stress. ...
... In the index case, the induction of stress dose glucocorticoid therapy had a favorable effect on the myocardium. The same has been reported earlier although sparingly [15][16][17][18]. This may reflect the role of glucocorticoids in proper functioning of the membrane calcium transport chain in the sarcoplasmic reticulum which is essential for adequate myocardial contractility [4]. ...
Background
Takotsubo cardiomyopathy (TCM) is a rare disease entity characterized by acute, non-ischemic, reversible myocardial dysfunction that mimics acute myocardial infarction. Activation and excessive outflow of sympathetic nervous system are believed to be central to the figure in the disease pathogenesis. Adrenocortical hormones potentiate the systemic actions of sympathetic nervous system and accordingly are essential for regulation of myocardial function. We present an unusual case of a middle-aged woman with primary adrenal insufficiency who presented paradoxically with TCM.
Case presentation
A 50-year-old woman with past history of hypothyroidism presented to emergency department with history of acute chest pain and syncope. There was no significant drug history or history of an emotional or physical stimulus prior to admission. Prominent pigmentation over the tongue and skin creases of hands were noted. On presentation, she was in shock and had ventricular tachycardia which required electrical cardioversion. The subsequent electrocardiogram demonstrated diffuse T-wave inversions with prolonged QT C . There was apical hypokinesia on echocardiogram, and cardiac biomarkers were elevated. There was persistent inotropic requirement. She had marked postural symptoms, and a postural blood pressure drop of 50 mm Hg was present. Initial laboratory parameters were significant for hyperkalemia (7.8 mEq/L) and hyponatremia (128 mEq/L). These findings prompted evaluation for adrenal insufficiency which was confirmed with appropriate tests. Autoimmune polyendocrine syndrome II was thus diagnosed based on the above findings. Coronary angiography revealed normal coronaries. The diagnoses of TCM was established in accordance with the International Takotsubo Diagnostic Criteria. She was started on stress dose steroid replacement therapy and improved dramatically. At one month of follow-up, the patient is asymptomatic, and there was normalization of her left ventricular function.
Conclusions
Intricate relationship and interplay exist between the steroid hormones and catecholamines in the pathogenesis of TCM. Steroid hormones not only potentiate the actions of catecholamines, but they also regulate and channelize catecholaminergic actions preventing their deleterious effects on the cardiac tissue. Hence, both steroid deficiency and exogenous steroid replacement may precipitate TCM. Evidence from more such cases and larger perspective studies in future will further improve our understanding of this complex disease process and its myriad associations.
... The paradox in the index case is the development of TCM in a patient with adrenal insu ciency wherein in the absence of steroid hormones, the toxic effects of sympathetic catecholamines were realized. Indeed, there have been only very few case reports documenting this association to date (13)(14)(15). ...
... In the index case, the induction of stress dose glucocorticoid therapy had a favorable effect on the myocardium. The same has been reported earlier although sparingly (13)(14)(15)(16). This may re ect the role of glucocorticoids in proper functioning of the membrane calcium transport chain in the sarcoplasmic reticulum which is essential for adequate myocardial contractility (4). ...
Background
Takotsubo cardiomyopathy (TCM) is a rare disease entity characterized by acute, non-ischemic, reversible myocardial dysfunction that mimics acute myocardial infarction. Activation and excessive outflow of sympathetic nervous system is believed to be central to the figure in the disease pathogenesis. Adrenocortical hormones potentiate the systemic actions of sympathetic nervous system and accordingly are essential for regulation of myocardial function. We present an unusual case of a middle-aged woman with primary adrenal insufficiency who presented paradoxically with TCM.
Case presentation
A 50-year-old woman with past history of hypothyroidism presented to emergency department with history of acute chest pain and syncope. There was no significant drug history or history of an emotional or physical stimulus prior to admission. Prominent pigmentation over the tongue and skin creases of hands were noted. On presentation she was in shock and had ventricular tachycardia which required electrical cardioversion. The subsequent electrocardiogram demonstrated diffuse T-wave inversions with prolonged QTC. There was apical hypokinesia on echocardiogram and cardiac biomarkers were elevated. There was persistent inotropic requirement. She had marked postural symptoms and a postural blood pressure drop of 50mm Hg was present.
Initial laboratory parameters were significant for hyperkalemia (7.8mEq/L) and hyponatremia (128mEq/L). These findings prompted evaluation for adrenal insufficiency which was confirmed with appropriate tests. Autoimmune polyendocrine syndrome II was thus diagnosed based on the above findings. Coronary angiography revealed normal coronaries. The diagnoses of TCM was established in accordance with the International takotsubo diagnostic criteria. She was started on stress dose steroid replacement therapy and improved dramatically. At one month of follow-up, the patient is asymptomatic and there was normalization of her left ventricular function.
Conclusions
Intricate relationship and interplay exist between the steroid hormones and catecholamines in the pathogenesis of TCM. Steroid hormones not only potentiate the actions of catecholamines, they also regulate and channelize catecholaminergic actions preventing their deleterious effects on the cardiac tissue. Hence, both steroid deficiency and exogenous steroid replacement may precipitate TCM. Evidence from more such cases and larger perspective studies in the future will further improve our understanding of this complex disease process and its myriad associations.
Takotsubo cardiomyopathy (TCM) is a reversible syndrome that resembles a myocardial infarction but without typical coronary stenosis and with an apical “ballooning” image present on an echocardiogram. Multiple triggers have been linked to TCM but rarely, acute severe hypoglycemia.
This is a case of a 39-year-old woman who was brought to the emergency department after being found unresponsive at home. She was severely hypoglycemic with a glucose of 18 mg/dL and suspected to have sulfonylurea intoxication. The patient was intubated and transferred to our ICU from an outside facility for a higher level of care.
The patient was noted to have an elevated troponin and the initial echocardiogram demonstrated TCM. The patient remained persistently hypoglycemic, despite continuous dextrose infusion and glucagon treatment. Stress dose steroids were added with the eventual resolution of hypoglycemia. A repeat echocardiogram demonstrated the resolution of TCM.
We report a case of a 36-year-old male who presented to the emergency department with complaints of weakness. On presentation the patient was hypotensive, hyperkalemic, and hyponatremic. The patient experienced a sudden cardiac arrest in the computed tomography (CT) scanner moments after arrival. Electrocardiogram (EKG) demonstrated PR prolongation and widened QRS. Echocardiogram demonstrated a left ventricular ejection fraction of 26%-30% with evidence of severe hypokinesis of the mid antero-septal and inferior-septal segments of the left ventricle. CT of the chest, abdomen, and pelvis demonstrated hypoplastic/atrophic adrenal glands. Total cortisol level was undetectable by lab measurement. The patient was diagnosed with stress cardiomyopathy secondary to adrenal crisis. He was managed with hydrocortisone and eventually made a full clinical recovery and improvement in left ventricular ejection fraction. This article references the rarity of this phenomenon and its relevance to early clinical detection.
