Multimodality evaluation of Takotsubo
cardiomyopathy in an isolated single
coronary artery anomaly
Shiro Miura, Osamu Manabe, Masanao
Naya, Akira Ando, Atsushi Usami,
Chihoko Miyazaki, Ohkusa Takanori &Takehiro Yamashita
Journal of Nuclear Cardiology
ISSN 1071-3581
J. Nucl. Cardiol.
DOI 10.1007/s12350-020-02312-z

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Author's personal copy
ORIGINAL ARTICLE
Multimodality evaluation of Takotsubo
cardiomyopathy in an isolated single coronary
artery anomaly
Shiro Miura, MD, MSc,a Osamu Manabe, MD, PhD,bMasanao Naya, MD, PhD,c Akira Ando, CNMT,d Atsushi Usami, CNMT,dChihoko Miyazaki, MD, PhD,e Ohkusa Takanori, MD, PhD,a andTakehiro Yamashita, MD, PhD, FACCaa

Department of Cardiology, Hokkaido Ohno Memorial Hospital, Sapporo, Japan
Department of Radiology, Saitama Medical Center, Jichi Medical University, Saitama-shi, Japan
cDepartment 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, Japanb

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 cardiovascular 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, apulse rate of 76 beats per minute and a temperature of
98.4 °F. Her physical examination was unremarkableand electrocardiogram (ECG) revealed a normal sinus
rhythm with ST elevation and T-wave inversion (Figure 1A). Transthoracic echocardiography (TTE)
revealed wall motion abnormalities and severe left
ventricular (LV) dysfunction. Laboratory findings wereas follows: troponin I, 37.6 pg/mL and N-terminal probrain natriuretic peptide, 2,553 pg/mL. She underwent
an urgent coronary angiography wherein the left coronary artery (LCA) was patent with no stenosis; however,selective cannulation of the right coronary artery wasimpossible. Left ventriculography revealed severe LV
dysfunction with apical hypokinesis and basal hyperkinesis, suggesting takotsubo cardiomyopathy (TCM)(Figure 2). A subsequent coronary computed tomography angiography revealed a rare anomaly of a congenital
single L-I type coronary artery, but no stenosis (Figure 3). Cardiovascular magnetic resonance imaging

Reprint requests: Shiro Miura, MD, MSc, Department of Cardiology,
Hokkaido Ohno Memorial Hospital, 2-1-16-1 Miyanosawa, Nishiku, Sapporo063-0052, Japan; shirotan1027m@yahoo.co.jp
1071-3581/$34.00
Copyright Ó 2020 American Society of Nuclear Cardiology.

(Figure 4), four days after admission, revealed wallmotion abnormalities, localized pericardial effusion anddiffuse myocardial edema, compatible with TCM.1 The
13
N-ammonia positron emission tomography (PET) and18
F-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 afteradmission and carefully followed up. At the 6-monthvisit, 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 myocardial ischemia directly caused by the abnormal anatomyof the arteries.3
DISCLOSURE
The authors indicate that they have no financial
conflicts of interest.

Author's personal copy
Journal of Nuclear CardiologyÒFigure 1. Initial 12-lead electrocardiogram (ECG) (A) showing sinus rhythm, ST elevation in leads
VI and VII and T-wave inversion in leads VII–VVI. ECG at the 6-month follow-up visit (B) showing
normalized ST-T changes.

Author's personal copy
Journal of Nuclear CardiologyÒFigure 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 demonstrating
apical ballooning hypokinesis with basal hyperkinesis and severe left ventricular (LV) dysfunction.
There was no LV outflow tract obstruction (C, systolic phase; D, diastolic phase).

Author's personal copy
Journal of Nuclear CardiologyÒFigure 3. Coronary computed tomographic angiography on hospital day 6 revealing an isolated
single 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-maximal
intensity projection reformation (B).

Author's personal copy
Journal of Nuclear CardiologyÒFigure 4. Cardiovascular magnetic resonance imaging on hospital day 4 revealing moderate
hypokinesia of the apical to mid segments of both anterior and anteroseptal walls with cine imaging
as 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 gadolinium
enhancement alongside the epicardial myocardium in the broad anterior wall (D) were shown inconcordance to the area of edema (red arrows).

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Journal of Nuclear CardiologyÒFigure 5. ATP-induced stress (A) and rest (B) 13N-ammonia positron emission tomography/computed 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/perfusion
mismatch. 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). The
follow-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 and
2.22, respectively, with MFR in 17 segments improved in the entire left ventricle (H). SA short axis;
VLA vertical long axis; HLA horizontal long axis.

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Journal of Nuclear CardiologyÒReferences
1. Manabe O, Naya M, Oyama-Manabe N, Koyanagawa K, Tamaki N.
The role of multimodality imaging in takotsubo cardiomyopathy. JNucl 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
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