Systemic thromboembolism including multiple cerebral infarctions with middle cerebral artery occlusion caused by the progression of adenomyosis with benign gynecological tumor: a case report

Abstract

Background Adenomyosis, a benign gynecological disease, causes cerebral infarction. Similar to Trousseau’s syndrome, it elevates cancer antigen 125 (CA125) and D-dimer levels; causes hypercoagulability; and results in cerebral infarction. However, no case of adenomyosis causing major cerebral artery occlusion and requiring endovascular thrombectomy has yet been reported. We report on a woman with middle cerebral artery occlusion caused by adenomyosis progression with a benign gynecological tumor and recurrent cerebral infarction. She was successfully treated by endovascular thrombectomy and hysterectomy. Case presentation A 48-year-old woman with heavy uterine bleeding was transported by ambulance to our hospital. Upon arrival, she presented with impaired consciousness. Laboratory test results revealed decreased hemoglobin (8.2 g/dL) and elevated D-dimer (79.3 µg/mL) levels. Radiological imaging revealed adenomyosis, a left ovarian tumor, multiple uterine myomas, and old and new bilateral renal infarctions. She experienced repeated episodes of excessive menstruation caused by adenomyosis and was scheduled for hysterectomy in 2 months at another hospital. After hospital admission, uterine bleeding stopped. However, 5 days after initial bleeding, she had another episode of heavy uterine bleeding and developed left hemiparesis and dysarthria 20 min later. Brain magnetic resonance imaging revealed bilateral multiple cerebral infarctions indicating right middle cerebral artery occlusion. Thus, endovascular thrombectomy was performed, and anticoagulant therapy was administered. Laboratory test results after thrombectomy revealed elevated CA125 (3536 U/mL) and CA19-9 (892 U/mL) levels. She was at a risk of recurrent heavy uterine bleeding leading to repeated cerebral infarction because of anticoagulant treatment. Therefore, we performed hysterectomy and ovariectomy 11 days after initial bleeding. Histopathological assessment revealed no malignancy. Although she developed asymptomatic pulmonary thromboembolism 14 days after initial bleeding, D-dimer and tumor marker levels returned to normal soon after gynecological surgery. At 15 months post-surgery, she had not experienced further ischemic events. Conclusions Adenomyosis with benign gynecological tumors may be associated with elevated D-dimer and tumor marker levels; excessive menstruation; and anemia. It may cause systemic thromboembolism, including cerebral infarction. To our knowledge, no other study has reported that adenomyosis causes major cerebral artery occlusion requiring endovascular thrombectomy. Hysterectomy may be an effective radical treatment of this condition.

Recurrent Cerebral Infarcts Associated with Uterine Adenomyosis: Successful Prevention by Surgical Removal

“…Carcinoma mucins, including CA125 and CA19-9, promote thrombosis in Trousseau's syndrome by adhesion-dependent, bidirectional signaling in neutrophils and platelets ( 14 ). In previous reports, most adenomyosis patients with cerebral infarctions had high CA125 levels ( 1 - 9 ). In addition, several studies reported that CA19-9 levels were elevated in patients with adenomyosis who developed cerebral infarction ( 3 , 4 , 6 , 7 , 9 ).…”

“…In previous reports, most adenomyosis patients with cerebral infarctions had high CA125 levels ( 1 - 9 ). In addition, several studies reported that CA19-9 levels were elevated in patients with adenomyosis who developed cerebral infarction ( 3 , 4 , 6 , 7 , 9 ). In our case, CA125 and CA19-9 levels were slightly elevated, and pathological examination did not prove the overproduction of mucus.…”

“…The occurrence of cerebral infarction in adenomyosis patients has been previously reported (1)(2)(3)(4)(5)(6)(7)(8)(9). Yamashiro et al reported cerebral infarction and adenomyosis in four patients (1).…”

“…Hypercoagulability associated with malignant tumors sometimes causes thrombosis, such as cerebral infarct. Adenomyosis is a benign gynecological tumor; however, there are some reports referring to cerebral infarction that can be caused by adenomyosis (1)(2)(3)(4)(5)(6)(7)(8)(9).…”

Possible association between adenomyosis and disseminated intravascular coagulation and thromboembolism: A systematic review

“…A laboratory examination revealed an elevated serum CA125 level (334.8 U/ml) (26). Aiura et al (27) reported the case of a 48-year-old woman with middle cerebral artery occlusion and recurrent cerebral infarction caused by adenomyosis progression. She was successfully treated with catheter-directed mechanical thrombectomy and by hysterectomy (27).…”