Pheochromocytoma Found in Takotsubo Cardiomyopathy Patients

ABSTRACT: Increased level of serum catecholamines in the acute phase was reported to be a feature of takotsubo cardiomyopathy (TC). We report a TC case with pheochromocytoma, which caused a stir in the diagnosis of TC and suggests the importance of screening for a catecholamine-producing tumor.

A female patient was referred to our emergency department due to ongoing chest pain. Coronary angiography showed no abnormality; however, subsequent left ventriculography showed basal hyperkinesis and apical ballooning, which completely recovered in 2 weeks. She experienced significant emotional stress on the eve of her admission, to which the diagnosis of TC was attributed. Although serum catecholamine levels on admission in our case were higher than on day 14, the value on day 14 was much higher than the normal range in our patient. The screening abdominal computed tomography scan revealed a left adrenal mass, which was diagnosed as pheochromocytoma by 24-hour urinary excretion of catecholamine and ¹³¹I-MIBG scintigraphy. The mass was successfully resected and pathological findings supported the diagnosis.

In our case, emotional stress was thought to be the direct trigger to develop TC by exceeding the threshold of catecholamine-induced cardiomyopathy. Screening for a catecholamine-producing tumor through careful history-taking and measuring catecholamines at a follow-up stage were important clinical aspects in this case and may well be for others.

J INVASIVE CARDIOL 2014;26(6):E76-E77

Key words: pheochromocytoma, catecholamine-producing tumor

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Increased levels of serum catecholamine in the acute phase is a well-known feature of takotsubo cardiomyopathy (TC),^1,2 which is supposedly triggered by emotional or physical stress.³ We report a TC case with pheochromocytoma, which creates a stir in the diagnosis of TC and suggests the importance of screening for a catecholamine-producing tumor.

Case Report. A middle-aged female patient with an 8-year history of hypertension, refractory diabetes mellitus, and dyslipidemia was referred to our emergency department due to ongoing chest pain. Due to the presence of complete right bundle branch block, her electrocardiogram was not decisive. The serum troponin I level was modestly elevated and transthoracic echocardiography revealed akinesis of the mid to apical left ventricle. Emergent coronary angiography showed no abnormality; however, subsequent left ventriculography showed basal hyperkinesis and apical ballooning (Figure 1A = diastole; Figure 1B = systole). The electrocardiogram showed T-wave inversion in broad leads on the day after her admission and resolved in several months. Follow-up echocardiography showed complete recovery in 2 weeks.

On the eve of the patient’s admission, her son was injured in a traffic accident, which was thought to be an emotional stressor, to which the diagnosis of TC was attributed. However, the screening abdominal computed tomography scan revealed a left adrenal mass, suspected of being a pheochromocytoma (Figure 2A, black arrows). Twenty-four hour urinary excretion of catecholamine was dopamine 1046.9 µg/day (190.0-740.0 µg/day), epinephrine 113.2 µg/day (3.0-15.0 µg/day), norepinephrine 603.1 µg/day (26.0-121.0 µg/day), metanephrine 1.15 mg/day (0.05-0.23 mg/day) and normetanephrine 1.46 mg/day (0.07-0.26 mg/day), respectively. 131I-MIBG scintigraphy demonstrated accumulation on her left adrenal gland with no metastasis (Figure 2B, black arrow), resulting in diagnosis as a benign pheochromocytoma. The left adrenal mass was surgically resected and pathological findings supported the diagnosis.

Discussion. Several cases of takotsubo-like cardiomyopathy secondary to pheochromocytoma, paraganglioma, or injection of intravenous epinephrine have been reported.^4-6 Although serum catecholamine levels on admission in our case were higher than at day 14, the value on day 14 was much higher than the normal range in our patient (dopamine 53 pg/mL [<20 pg/mL], epinephrine 143 pg/mL [<100 pg/mL], and norepinephrine 1352 pg/mL [100-450 pg/mL] on day 14, respectively). Due to the presence of pheochromocytoma, basal serum catecholamine levels were high in this patient, thus predisposing her to catecholamine-induced cardiomyopathy.

Even though this patient was presenting with typical precipitants and diagnosed as TC after cardiac catheterization, screening for a catecholamine-producing tumor through a careful history-taking and measuring catecholamine at a follow-up stage was an important clinical aspect in this case and may well be for others.

References

1. Wittstein IS, Thiemann DR, Lima JA, et al. Neurohumoral features of myocardial stunning due to sudden emotional stress. N Engl J Med. 2005;352(6):539-548.
2. Kume T, Kawamoto T, Okura H, et al. Local release of catecholamines from the hearts of patients with takotsubo-like left ventricular dysfunction. Circ J. 2008;72(1):106-108.
3. Ako J, Sudhir K, Farouque HM, Honda Y, Fitzgerald PJ. Transient left ventricular dysfunction under severe stress: brain-heart relationship revisited. Am J Med. 2006;119(1):10-17.
4. Naderi N, Amin A, Setayesh A, Pouraliakbar H, Mozaffari K, Maleki M. Pheochromocytoma-induced reverse tako-tsubo with rapid recovery of left ventricular function. Cardiol J. 2012;19(5):527-531.
5. Van Spall HG, Roberts JD, Sawka AM, Swallow CJ, Mak S. Not a broken heart. Lancet. 2007;370(9587):628.
6. Khoueiry G, Abi Rafeh N, Azab B, et al. Reverse takotsubo cardiomyopathy in the setting of anaphylaxis treated with high-dose intravenous epinephrine. J Emerg Med. 2013;44(1):96-99. Epub 2012 May 16.

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From the Sakakibara Heart Institute of Okayama, Okayama, Japan.

Disclosure: The authors have completed and returned the ICMJE Form for Disclosure of Potential Conflicts of Interest. The authors report no conflicts of interest regarding the content herein.

Manuscript submitted September 8, 2013, provisional acceptance given September 12, 2013, final version accepted September 23, 2013.

Address for correspondence: Yuhei Kobayashi, MD, Cardiovascular Medicine, The Sakakibara Heart Institute of Okayama, 2-5-1, Nakai-cho, Kita-ku, Okayama, Japan, 700-0804. Email: yuhei_ko2000@yahoo.co.jp