Skip to main content
Top
Published in: BMC Neurology 1/2018

Open Access 01-12-2018 | Case report

Cerebral infarction associated with benign mucin-producing adenomyosis: report of two cases

Authors: Koki Okazaki, Fumiaki Oka, Hideyuki Ishihara, Michiyasu Suzuki

Published in: BMC Neurology | Issue 1/2018

Login to get access

Abstract

Background

Cerebral infarction associated with a malignant tumor is widely recognized as Trousseau syndrome. In contrast, few cases of cerebral infarction associated with benign tumors have been reported. We present two cases of embolic stroke that seemed to be caused by mucin-producing adenomyosis.

Case presentation

The patients were women aged 42 and 50 years old. Both patients developed right hemiparesis and aphasia, and cerebral infarctions were detected in the left cerebral hemisphere. There were no other abnormal findings, except for elevation of CA125 and D-dimer. Trousseau syndrome was suspected in both cases, but whole body examinations did not reveal any malignant tumors. However, uterine adenomyosis was detected in both patients.

Conclusions

From our findings and a review of the literature, both mucin-producing malignant tumors and mucin-producing benign tumors such as adenomyosis may cause hypercoagulability and cerebral infarction. This mechanism should be considered in a case of a young to middle-aged woman with embolic stroke of an undetermined origin.
Literature
1.
go back to reference Babacan A, Kizilaslan C, Gun I, Muhcu M, Mungen E, Atay V. CA 125 and other tumor markers in uterine leiomyomas and their association with lesion characteristics. Int J Clin Exp Med. 2014;7:1078–83.PubMedPubMedCentral Babacan A, Kizilaslan C, Gun I, Muhcu M, Mungen E, Atay V. CA 125 and other tumor markers in uterine leiomyomas and their association with lesion characteristics. Int J Clin Exp Med. 2014;7:1078–83.PubMedPubMedCentral
2.
go back to reference Liu X, Nie J, Guo SW. Elevated immunoreactivity to tissue factor and its association with dysmenorrhea severity and the amount of menses in adenomyosis. Hum Reprod. 2011;26:337–45.CrossRef Liu X, Nie J, Guo SW. Elevated immunoreactivity to tissue factor and its association with dysmenorrhea severity and the amount of menses in adenomyosis. Hum Reprod. 2011;26:337–45.CrossRef
3.
go back to reference Evans TR, Mansi JL, Bevan DH. Trousseau’s syndrome in association with ovarian carcinoma. Cancer. 1996;77:2544–9.CrossRef Evans TR, Mansi JL, Bevan DH. Trousseau’s syndrome in association with ovarian carcinoma. Cancer. 1996;77:2544–9.CrossRef
4.
go back to reference Varki A. Trousseau’s syndrome: multiple definitions and multiple mechanisms. Blood. 2007;110:1723–9.CrossRef Varki A. Trousseau’s syndrome: multiple definitions and multiple mechanisms. Blood. 2007;110:1723–9.CrossRef
5.
go back to reference Wahrenbrock M, Borsig L, Le D, Varki N, Varki A. Selectin-mucin interactions as a probable molecular explanation for the association of trousseau syndrome with mucinous adenocarcinomas. J Clin Invest. 2003;112:853–62.CrossRef Wahrenbrock M, Borsig L, Le D, Varki N, Varki A. Selectin-mucin interactions as a probable molecular explanation for the association of trousseau syndrome with mucinous adenocarcinomas. J Clin Invest. 2003;112:853–62.CrossRef
6.
go back to reference Felder M, Kapur A, Gonzalez-Bosquet J, Horibata S, Heintz J, Albrecht R, et al. MUC16 (CA125): tumor biomarker to cancer therapy, a work in progress. Mol Cancer. 2014;13:129.CrossRef Felder M, Kapur A, Gonzalez-Bosquet J, Horibata S, Heintz J, Albrecht R, et al. MUC16 (CA125): tumor biomarker to cancer therapy, a work in progress. Mol Cancer. 2014;13:129.CrossRef
7.
go back to reference Jovin TG, Boosupalli V, Zivkovic SA, Wechsler LR, Gebel JM, et al. High titers of CA-125 may be associated with recurrent ischemic strokes in patients with cancer. Neurology. 2005;64:1944–5.CrossRef Jovin TG, Boosupalli V, Zivkovic SA, Wechsler LR, Gebel JM, et al. High titers of CA-125 may be associated with recurrent ischemic strokes in patients with cancer. Neurology. 2005;64:1944–5.CrossRef
8.
go back to reference Yamashiro K, Furuya T, Noda K, Urabe T, Hattori N, Okuma Y. Cerebral infarction developing in a patient without cancer with a markedly elevated level of mucinous tumor marker. J Stroke Cerebrovasc Dis. 2012;21:619.e1–2. Yamashiro K, Furuya T, Noda K, Urabe T, Hattori N, Okuma Y. Cerebral infarction developing in a patient without cancer with a markedly elevated level of mucinous tumor marker. J Stroke Cerebrovasc Dis. 2012;21:619.e1–2.
9.
go back to reference Yamashiro K, Tanaka R, Nishioka K, Ueno Y, Shimura H, Okuma Y, et al. Cerebral infarcts associated with adenomyosis among middle-aged women. J Stroke Cerebrovasc Dis. 2012;21:910.e1–5. Yamashiro K, Tanaka R, Nishioka K, Ueno Y, Shimura H, Okuma Y, et al. Cerebral infarcts associated with adenomyosis among middle-aged women. J Stroke Cerebrovasc Dis. 2012;21:910.e1–5.
10.
go back to reference Ikushima S, Ono R, Fukuda K, Sakayori M, Awano N, Kondo K. Trousseau’s syndrome: cancer-associated thrombosis. Jpn J Clin Oncol. 2016;46:204–8.CrossRef Ikushima S, Ono R, Fukuda K, Sakayori M, Awano N, Kondo K. Trousseau’s syndrome: cancer-associated thrombosis. Jpn J Clin Oncol. 2016;46:204–8.CrossRef
11.
go back to reference Hijikata N, Sakamoto Y, Nito C, Matsumoto N, Abe A, Nogami A, et al. Multiple cerebral infarctions in a patient with adenomyosis on hormone replacement therapy: a case report. J Stroke Cerebrovasc Dis. 2016;25:183–4.CrossRef Hijikata N, Sakamoto Y, Nito C, Matsumoto N, Abe A, Nogami A, et al. Multiple cerebral infarctions in a patient with adenomyosis on hormone replacement therapy: a case report. J Stroke Cerebrovasc Dis. 2016;25:183–4.CrossRef
Metadata
Title
Cerebral infarction associated with benign mucin-producing adenomyosis: report of two cases
Authors
Koki Okazaki
Fumiaki Oka
Hideyuki Ishihara
Michiyasu Suzuki
Publication date
01-12-2018
Publisher
BioMed Central
Published in
BMC Neurology / Issue 1/2018
Electronic ISSN: 1471-2377
DOI
https://doi.org/10.1186/s12883-018-1169-2

Other articles of this Issue 1/2018

BMC Neurology 1/2018 Go to the issue