1 23

Journal of Nuclear Cardiology ISSN 1071-3581 J. Nucl. Cardiol.DOI 10.1007/s12350-020-02312-z

Multimodality evaluation of Takotsubocardiomyopathy in an isolated singlecoronary artery anomaly

Shiro Miura, Osamu Manabe, MasanaoNaya, Akira Ando, Atsushi Usami,Chihoko Miyazaki, Ohkusa Takanori &Takehiro Yamashita

1 23

Your article is protected by copyright and

all rights are held exclusively by American

Society of Nuclear Cardiology. This e-offprint

is for personal use only and shall not be self-

archived in electronic repositories. If you wish

to self-archive your article, please use the

accepted manuscript version for posting on

your own website. You may further deposit

the accepted manuscript version in any

repository, provided it is only made publicly

available 12 months after official publication

or later and provided acknowledgement is

given to the original source of publication

and a link is inserted to the published article

on Springer's website. The link must be

accompanied by the following text: "The final

publication is available at link.springer.com”.

ORIGINAL ARTICLE

Multimodality evaluation of Takotsubocardiomyopathy in an isolated single coronaryartery anomaly

Shiro Miura, MD, MSc,a Osamu Manabe, MD, PhD,b

Masanao Naya, MD, PhD,c Akira Ando, CNMT,d Atsushi Usami, CNMT,d

Chihoko Miyazaki, MD, PhD,e Ohkusa Takanori, MD, PhD,a and

Takehiro Yamashita, MD, PhD, FACCa

a Department of Cardiology, Hokkaido Ohno Memorial Hospital, Sapporo, Japanb Department of Radiology, Saitama Medical Center, Jichi Medical University, Saitama-shi, Japanc Department of Cardiovascular Medicine, Hokkaido, University Graduate School of Medicine,

Sapporo, Japand Division of Medical Imaging Technology, Hokkaido Ohno Memorial Hospital, Sapporo, Japane Department of Diagnostic Radiology, Hokkaido Ohno Memorial Hospital, Sapporo, Japan

Received Jul 13, 2020; accepted Jul 13, 2020

doi:10.1007/s12350-020-02312-z

CASE SUMMARY

An 85-year-old woman with no remarkable cardio-

vascular risk factor was referred, complaining of sudden

chest pain 3 days before admission. She no longer had

chest pain and had a blood pressure of 142/73 mmHg, a

pulse rate of 76 beats per minute and a temperature of

98.4 �F. Her physical examination was unremarkable

and electrocardiogram (ECG) revealed a normal sinus

rhythm with ST elevation and T-wave inversion (Fig-

ure 1A). Transthoracic echocardiography (TTE)

revealed wall motion abnormalities and severe left

ventricular (LV) dysfunction. Laboratory findings were

as follows: troponin I, 37.6 pg/mL and N-terminal pro-

brain natriuretic peptide, 2,553 pg/mL. She underwent

an urgent coronary angiography wherein the left coro-

nary artery (LCA) was patent with no stenosis; however,

selective cannulation of the right coronary artery was

impossible. Left ventriculography revealed severe LV

dysfunction with apical hypokinesis and basal hyperki-

nesis, suggesting takotsubo cardiomyopathy (TCM)

(Figure 2). A subsequent coronary computed tomogra-

phy angiography revealed a rare anomaly of a congenital

single L-I type coronary artery, but no stenosis (Fig-

ure 3). Cardiovascular magnetic resonance imaging

(Figure 4), four days after admission, revealed wall

motion abnormalities, localized pericardial effusion and

diffuse myocardial edema, compatible with TCM.1 The13N-ammonia positron emission tomography (PET) and18F-fluorodeoxyglucose PET (Figure 5) revealed a clear

inverse metabolic/perfusion mismatch. The quantitative

analysis of stress myocardial blood flow myocardial

flow reserve (MFR) showed a mild reduction in apical to

midventricular segments compared to basal segments.

The patient was discharged uneventfully 15 days after

admission and carefully followed up. At the 6-month

visit, she was asymptomatic and her ECG (Figure 1B)

and TTE were within normal ranges. The repeated PET

examinations showed the improvement of inverse

metabolic/perfusion mismatch and MFR (Figure 5).

Thus, a severe reduction of glucose utility assessed by

FDG PET with mildly microcirculatory dysfunction

(low MFR) was developed in the acute phase of TCM.2

Reportedly, single LCA may be associated with myocar-

dial ischemia directly caused by the abnormal anatomy

of the arteries.3

DISCLOSURE

The authors indicate that they have no financial

conflicts of interest.

Reprint requests: Shiro Miura, MD, MSc, Department of Cardiology,

Hokkaido Ohno Memorial Hospital, 2-1-16-1 Miyanosawa, Nishi-

ku, Sapporo063-0052, Japan; shirotan1027m@yahoo.co.jp

1071-3581/$34.00

Copyright � 2020 American Society of Nuclear Cardiology.

Author's personal copy

Figure 1. Initial 12-lead electrocardiogram (ECG) (A) showing sinus rhythm, ST elevation in leadsVI and VII and T-wave inversion in leads VII–VVI. ECG at the 6-month follow-up visit (B) showingnormalized ST-T changes.

Journal of Nuclear Cardiology�

Author's personal copy

Figure 2. Coronary angiography on hospital day 1 (A, right caudal view; B, left cranial view)showing no significant stenosis in the left coronary artery. Left ventriculography demonstratingapical ballooning hypokinesis with basal hyperkinesis and severe left ventricular (LV) dysfunction.There was no LV outflow tract obstruction (C, systolic phase; D, diastolic phase).

Journal of Nuclear Cardiology�

Author's personal copy

Figure 3. Coronary computed tomographic angiography on hospital day 6 revealing an isolatedsingle L-I type coronary artery anomaly from the left coronary sinus in the absence of rightcoronary artery on the three-dimensional (3D) volume-rendering image (A) and 3D-maximalintensity projection reformation (B).

Journal of Nuclear Cardiology�

Author's personal copy

Figure 4. Cardiovascular magnetic resonance imaging on hospital day 4 revealing moderatehypokinesia of the apical to mid segments of both anterior and anteroseptal walls with cine imagingas apical ballooning (A, systolic phase; B, diastolic phase). Focal pericardial effusion was visiblearound the apex (red arrow). High signal intensity with fat-saturated T2-weighted images (fsT2WI)(yellow heads) due to the myocardial edema (C) and the presence of the late gadoliniumenhancement alongside the epicardial myocardium in the broad anterior wall (D) were shown inconcordance to the area of edema (red arrows).

Journal of Nuclear Cardiology�

Author's personal copy

Figure 5. ATP-induced stress (A) and rest (B) 13N-ammonia positron emission tomography/com-puted tomography (PET/CT) and glucose loading 18F-fluorodeoxyglucose (18F-FDG) PET/CT wereperformed (C) on hospital day 6 and 5, respectively. Myocardial perfusion imaging showed amoderate reduction in tracer uptake after ATP-infusion (A) in the apical and midventricularsegments that almost normalized at rest (B), demonstrating the reversible myocardial ischemia. Incontrast, a severe reduction in 18F-FDG uptake in the apex and progressively less beyond themidventricular segments were observed which culminated in an inverse metabolic/perfusionmismatch. The global myocardial blood flow (MBF) at stress and rest were 1.99 and 1.02 mL g-1

min -1, respectively, and myocardial flow reserve (MFR) was 1.98 with a localized drop in theapical segments, which implies the possibility of localized microvascular dysfunction (D). Thefollow-up images were obtained at 6 months after the normalization of left ventricular kinesis:perfusion images at stress (E) and rest (F) were normal and 18F-FDG uptake almost normalized(G). The stress global MBF and MFR showed a significant increase to 2.35 mL g-1 min-1 and2.22, respectively, with MFR in 17 segments improved in the entire left ventricle (H). SA short axis;VLA vertical long axis; HLA horizontal long axis.

Journal of Nuclear Cardiology�

Author's personal copy

References

1. Manabe O, Naya M, Oyama-Manabe N, Koyanagawa K, Tamaki N.

The role of multimodality imaging in takotsubo cardiomyopathy. J

Nucl Cardiol 2019;26:602-6

2. Feola M, Chauvie S, Rosso GL, Biggi A, Ribichini F, Bobbio M.

Reversible impairment of coronary flow reserve in takotsubo

cardiomyopathy: A myocardial PET study. J Nucl Cardiol

2008;15:811-7

3. Muhyieddeen K, Polsani VR, Chang SM. Single right coronary

artery with apical ischaemia. Eur Heart J Cardiovasc Imaging

2012;13:533

Publisher’s Note Springer Nature remains neutral with regard to

jurisdictional claims in published maps and institutional affiliations.

Journal of Nuclear Cardiology�

Author's personal copy