Multiple Cerebral Infarctions in a Patient with Adenomyosis on Hormone Replacement Therapy: A Case Report

Hijikata, Nanako; Sakamoto, Yuki; Nito, Chikako; Matsumoto, Noriko; Abe, Arata; Nogami, Akane; Sato, Takahiro; Hokama, Hiroyuki; Okubo, Seiji; Kimura, Kazumi

doi:10.1016/j.jstrokecerebrovasdis.2016.07.024

Cited by 19 publications

(30 citation statements)

References 7 publications

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“…Some patients with adenomyosis develop multiple cerebral infarctions (CIs) [ 3 – 5 ] (Table 1 ). Almost all patients are middle-aged, with severe anemia and elevated serum carbohydrate antigen 125 (CA125).…”

Section: Introductionmentioning

confidence: 99%

“Recurrent multiple cerebral infarctions related to the progression of adenomyosis: a case report”

et al. 2018

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BackgroundBenign gynecologic tumor, such as uterine adenomyosis, has been suggested to develop hypercoagulability. Although some cases of cerebral infarction associated with adenomyosis have been reported, the mechanism of hypercoagulation initiated by adenomyosis is still not clear, and the therapeutic strategy is uncertain.Case presentationA 44-year-old woman was presented to our department with headache, left hand weakness, and gait disturbance during her menstrual phase. She had a history of adenomyosis and infertility treatment for 18 years and heavy menstrual bleeding. Magnetic resonance imaging on admission showed multiple hyperintense lesions in cortical and subcortical areas in the cerebrum and cerebellum on diffusion-weighted imaging. Transesophageal echocardiography showed neither embolic sources nor existence of foramen ovale. Her laboratory data revealed anemia, a high D-dimer level, and elevated levels of a mucinous tumor marker. She had adenomyosis and no malignancy was detected. Anticoagulation therapy with intravenous heparin followed by rivaroxaban did not prevent recurrence of cerebral infarction. We discontinued rivaroxaban, and started warfarin therapy with pseudomenopause treatment, which prevented recurrence for 6 months. Five months after her last pseudomenopause treatment, multiple cerebral infarctions occurred. Total hysterectomy was performed, which prevented recurrence of the multiple cerebral infarctions for 2 years without anticoagulation therapy.ConclusionsOur findings reveal for the first time that anticoagulation therapy, including novel oral anticoagulants, had no preventive effect against cerebral infarctions associated with adenomyosis in a middle-aged woman. Although pseudomenopause treatment temporarily prevented recurrence, resection of the adenomyosis might be the most effective therapy in these cases.

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Section: Introductionmentioning

confidence: 99%

“Recurrent multiple cerebral infarctions related to the progression of adenomyosis: a case report”

et al. 2018

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“…Regarding acute cerebral infarction with adenomyosis, we consulted cases and found 13 patients in the cases [2][3][4][5][6][7][8]. The median age of the 13 patients was 45.4 years.…”

Section: Discussionmentioning

confidence: 99%

“…The pathway can cause systemic thrombin and even can cause DIC in extreme cases. Blood stagnation caused by intramural hemorrhage in adenomyosis during menstruation may activate the tissue factor (TF) coagulation pathway [3]. Although TF plays a key role in the initiation of coagulation, TF also involves in the pathogenesis of adenomyosis and may involve in angiogenesis.…”

Section: Discussionmentioning

confidence: 99%

Acute cerebral infarction with adenomyosis in a young woman: a case report

Zhao

Zhang

Yang

2019

Preprint

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Background Acute cerebral infarction with adenomyosis in a young woman has been rarely reported. Case presentation We describe a 34-year-old young woman who presented headache and fever (38°C) for 4 days and left limb weakness for 1 day during her menstrual phase. Laboratory test data showed: Hemoglobin (HGB) (112g/L, normal: 120-150 g/L), Carcinoembryonic antigen 125 (CA125) (937.70U/ml, normal: 0-35 U/ml), D-Dimer (27.4mg/L, normal: 0-1.5mg/L). Magnetic resonance imaging (MRI) indicated acute cerebral infarction in right basal ganglia and subcortical region of right frontotemporal lobe. Further, brain computed tomography angiography (CTA) showed that the M1 segment of right middle cerebral artery was strictured and the distal branches of right middle cerebral artery were significantly less than those on the opposite side. No obvious abnormality was found in cranial magnetic resonance venogram (MRV). She had a 5-year history of adenomyosis. No tumors were found by whole body positron emission tomography-computed tomography (PET-CT). We treated this patient by using anti-infective therapy for 1 week and using anticoagulant therapy with low molecular weight heparin for 2 weeks. Subsequently, the anticoagulant therapy was discontinued and replaced by antiplatelet therapy with poliovir. We followed up this patient for 4 months, and no recurrence of cerebral infarction was found. Conclusions Acute cerebral infarction with adenomyosis may be related to elevated D-Dimer, elevated CA125, anemia, menstruation and fever. Our report suggests that acute cerebral infarction with adenomyosis can occur not only in middle-aged women but also in young women, and fever during menstrual phase in a woman with adenomyosis may be a factor leading to acute cerebral infarction.

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“…[Ref] Age (y.o) CA125 (U/mL) D-dimer (μg/mL) Secondary prevention Recurrence 1 [ 9 ] 45 159 1.1 Antiplatelet, GnRH agonist (−) 2 [ 9 ] 44 Not mentioned FDP 5.9 μg/mL Warfarin, GnRH agonist (−) 3 [ 9 ] 55 42.6 0.57 (normal) Aspirin, GnRH agonist (−) 4 [ 8 ] a 42 1750 6.0 Antiplatelet (6 m). GnRH agonist (6 m) (+) 5 [ 9 ] a 42 907 4.1 Warfarin, GnRH agonist (−) 6 [ 11 ] 59 334.8 7.0 Discontinuation of hormone replacement therapy (−) 7 b 42 395 1.4 Warfarin (−) 8 b 50 143 3.7 Rivaroxaban (−) a Case Nos. 4 and 5 are the same patient b Case Nos.…”

Section: Discussionmentioning

confidence: 99%

“…Long-term hormone replacement therapy may cause hypercoagulability in patients with adenomyosis, and discontinuation of this therapy in one reported case (Case No. 6, Table 1 ) did not lead to recurrence [ 11 ]. Overall, further studies are needed to clarify the mechanisms of development of cerebral infarction in patients with adenomyosis or other mucin-producing benign.…”

Section: Discussionmentioning

confidence: 99%

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

et al. 2018

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BackgroundCerebral 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 presentationThe 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.ConclusionsFrom 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.

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Multiple Cerebral Infarctions in a Patient with Adenomyosis on Hormone Replacement Therapy: A Case Report

Cited by 19 publications

References 7 publications

“Recurrent multiple cerebral infarctions related to the progression of adenomyosis: a case report”

“Recurrent multiple cerebral infarctions related to the progression of adenomyosis: a case report”

Acute cerebral infarction with adenomyosis in a young woman: a case report

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

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