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Case Reports
. 2018 Oct 4;18(1):166.
doi: 10.1186/s12883-018-1169-2.

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

Koki Okazaki  1 , Fumiaki Oka  2 , Hideyuki Ishihara  2 , Michiyasu Suzuki  2
Affiliations
Case Reports

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

Koki Okazaki et al. BMC Neurol. .

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.

Keywords: Adenomyosis; Benign tumor; Case report; Cerebral infarction; Trousseau’s syndrome.

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Conflict of interest statement

Ethics approval and consent to participate

The authors declare that ethics approval was not required for this case report.

Consent for publication

Written informed consents were obtained from both patients for publication of this case report and accompanying images.

Competing interests

The authors declare that they have no competing interests.

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Figures

Fig. 1
Fig. 1
a Diffusion-weighted magnetic resonance imaging (MRI) revealed an infarct in the left middle cerebral artery territory. b Magnetic resonance angiography showed occlusion at left M2 (arrow). c T2-weighted pelvic MRI revealed enlargement of the uterus and obscure junctional zone, suggesting adenomyosis
Fig. 2
Fig. 2
a Diffusion-weighted MRI revealed an infarct in the left middle cerebral artery territory. b Magnetic resonance angiography at a previous hospital showed left M1 occlusion. c Angiography revealed partial recanalization of the left middle cerebral artery. d Pelvic MRI revealed adenomyosis
See this image and copyright information in PMC

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