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Letters to the Editor

5 Yalug I, Tufan AE, Kayaalp L. Quetiapine may be associated
with new-onset seizures in patients with seizurogenic conditions. J Neuropsychiatry Clin Neurosci 2007; 19: 341–342.
6 Fauci AS, Braunwald E, Kasper DL et al. Harrison’s Principles
of Internal Medicine, 17th edn. New York: McGraw-Hill,
2009.

7 Rao SC, Dove G, Cascino GD. Recurrent seizures in
patients with dementia: frequency, seizure types, and treatment outcome. Epilepsy Behav 2009; 14: 118–120.
8 Naranjo CA, Busto U, Sellers EM et al. A method for estimating the probability of adverse drug reactions. Clin
Pharmacol Ther 1981; 30: 239–245.

Non-bacterial thrombotic endocarditis in a Trousseau
syndrome patient with stomach cancer: A case report
Dear Editor,
Older adults often experience stroke or cancer, which
can have important influences on their activities of daily
living and life. Unfortunately, stroke and cancer are
sometimes complicated and related to each other.
Since 1865, many studies have confirmed the association between cancer and cerebral infarction (CI).1,2
Trousseau syndrome and non-bacterial thrombotic
endocarditis (NBTE) are widely recognized as the
causes of stroke in patients with cancer. However, the
therapeutic strategy for patients with both Trousseau
syndrome and NBTE remains controversial excluding
treatment for cancer.3–7
A 78-year-old woman with gastric cancer developed
paresthesia of the right hand 10 months after gastrectomy with adjuvant chemotherapy. In addition, she
experienced paresthesia of the right side of her lips and
dysarthria after waking up from sleep; thus, she visited
our hospital, Tokyo Metropolitan Geriatric Hospital,
Tokyo, Japan. She had a history of transient ischemic
attack. The patient had neither fever nor cardiac
murmur. Neurological findings included mild right
hemiparesis, paresthesia of the right hand and instability during walking.
The serum values of carcinoembryonic antigen,
d-dimer, thrombin–antithrombin complex, plasmin-α2plasmin inhibitor complex, and protein C and S activity
were elevated. Serum tests of lupus anticoagulant and
anticardiolipin antibodies were within normal limits.
There was no evidence of collagen disease, atrial fibrillation, pulmonary thromboembolism, deep vein thrombosis or stenosis of the carotid artery. Repeated blood
cultures were negative. Transthoracic echocardiography
showed only mitral annular calcification.

Correspondence: Dr Kazumasa Harada MD PhD, Department
of Cardiology, Tokyo Metropolitan Geriatric Hospital and
Institute of Gerontology, 35-2, Sakae-cho, Itabashi-ku, Tokyo,
173-0015, Japan. Email: kharada@tmghig.jp

© 2016 Japan Geriatrics Society

Head magnetic resonance imaging showed a scattered
high intensity of diffusion-weighted images in the bilateral cerebral cortex, white matter and cerebellum,
including the border zone (Fig. 1a).
She was diagnosed with Trousseau syndrome; thus,
treatment with anticoagulants was initiated. However,
the patient’s response to the treatment was poor, and
her neurological symptoms deteriorated. A further
cardiac assessment using transesophageal echocardiography showed an abnormal mobile structure
attached to the bicuspid of the mitral valve without
leaflet destruction, suggesting infectious endocarditis
(Fig. 1b; supplemental data). We suspected a cardiac
tumor or NBTE, and carried out a mitral valve replacement with a biological valve to avoid fatal embolism.
The abnormal structures on the mitral valve were
diagnosed as NBTE by pathological findings
(Fig. 1c, d).
Her postoperative course was good, and she underwent rehabilitation; however, recurrent small CI could
not be prevented. The patient’s stomach cancer became
terminal; thus, she was discharged to home care on her
family’s request.
Here we report a Trousseau syndrome patient with
pathologically confirmed NBTE who presented with
recurrent ischemic strokes. The patient underwent
mitral valve replacement to avoid lethal CI and preserve activities of daily living. Before the present
report, little evidence existed about successful treatments for CI patients with both NBTE and Trousseau
syndrome.8 Also, in older adults, invasive therapy,
including cardiac surgery, represents a valuable therapeutic alternative in patients with large mobile vegetations, valvular dysfunctions or recurrent embolic
events that are unresponsive to treatment with
anticoagulants.9

Disclosure statement
The authors declare no conflict of interest.
doi: 10.1111/ggi.12694

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S Morimoto et al.

Figure 1 Head magnetic resonance
imaging, cardiac echography and
histopathological findings of the resected
mitral valve. (a) Diffusion-weighted images
show hyperintense lesions in multiple
vascular territories on admission. (b) Twoand three-dimensional transesophageal
echocardiography show large abnormal
mobile structures attached on A1–A2 lesions
of the anterior leaflet and P3 lesions of the
posterior leaflet of the mitral valve. Arrows
show abnormal structures on the mitral
valve. (c) Macroscopic finding of surgical
materials shows several white vegetations on
the anterior (left; 4 mm in diameter) and
posterior (right; 3 mm in diameter, 2 in
number) leaflet of the mitral valve (arrows).
Bar, 1 cm. (d) Histology of multiple fibrin
thrombi with neutrophilic infiltration and
myxoid degeneration of the mitral valve. Bar,
100 μm. Hematoxylin–eosin and Elastica van
Gieson staining.

Satoru Morimoto,1 Jun Tanaka,2 Yoko Saito,3
Naoko Tsuyama,4 Takashi Nishimura,5 Tadashi Komiya,1,6
Shunei Kyo,5 Tomio Arai,4 Akiko Kanemaru,3
Kazutomi Kanemaru1 and Kazumasa Harada2
Departments of 1Neurology, 2Cardiology, 3Rehabilitation,
4
Pathology, and 5Cardiovascular Surgery, Tokyo Metropolitan
Geriatric Hospital, and 6Department of Neurology, National
Hospital Organization Tokyo National Hospital, Tokyo, Japan

References
1 Trousseau A. “Phlegmasia alba dolens,” Clinique Medicale
de l’Hotel Dieu de Paris, 1865, 3, p. 654–712, translated by
the New Sydenham Society, London as ‘Lectures on Clinical Medicine delivered at the Hotel Dieu de Paris’, 1872, p.
281–95.
2 El-Shami K, Griffiths E, Streiff M. Nonbacterial thrombotic endocarditis in cancer patients: pathogenesis,
diagnosis, and treatment. Oncologist 2007; 12: 518–
523.

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3 Kossmann CE. Nonbacterial thrombotic endocarditis: a
specific lesion of Trousseau’s syndrome. J Tenn Med Assoc
1980; 73: 14–16.
4 Bessis D, Sotto A, Viard JP, Bérard M, Ciurana AJ, Boffa
MC. Trousseau’s syndrome with nonbacterial thrombotic
endocarditis: pathogenic role of antiphospholipid syndrome. Am J Med 1995; 98: 511–513.
5 Aryana A, Esterbrooks DJ, Morris PC. Nonbacterial thrombotic endocarditis with recurrent embolic events as manifestation of ovarian neoplasm. J Gen Intern Med 2006; 21:
C12–C15.
6 Ito S, Yoshitomi H, Pak M et al. Trousseau syndrome with
nonbacterial thrombotic endocarditis in a patient with
uterine cancer. Intern Med 2013; 52: 1353–1358.
7 Yoshida K, Kimura T, Aburakawa Y, Suzuki Y, Kuroda K,
Yahara O. Recurrent ischemic stroke in a patient with the
Trousseau syndrome treated with dabigatran. J Stroke
Cerebrovasc Dis 2014; 23: 1724–1726.
8 Hisashi S, Hitoshi O, Kozo N, Junji Y. Trousseau Syndrome
Caused by Ovarian Cancer and Nonbacterial Thrombotic
Endocarditis in Aortic Valves. Jpn J Cardiovasc Surg 2006; 35:
102–105.
9 Rabinstein AA, Giovanelli C, Romano JG, Koch S, Forteza
AM, Ricci M. Surgical treatment of nonbacterial thrombotic
endocarditis presenting with stroke. J Neurol 2005; 252: 352–
355.

© 2016 Japan Geriatrics Society