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.

Keywords: Adenomyosis; Benign gynecological tumor; Endovascular thrombectomy; Hypercoagulability; Hysterectomy; Middle cerebral artery occlusion; Multiple cerebral infarction; Systemic thromboembolism.

Fig. 1

Pelvic imaging on the day of initial bleeding a, d Pelvic contrast-enhanced computed tomography (CT) and magnetic resonance images show adenomyosis (arrowheads), multiple uterine myomas (arrows), and a left ovarian tumor (stars). b-c Pelvic contrast-enhanced CT shows old and new bilateral renal infarctions (arrows)

Fig. 2

Brain imaging and thrombus collected on the day of the development of cerebral infarctions a-c Brain magnetic resonance imaging shows bilateral multiple cerebral infarctions indicating right middle cerebral artery occlusion. d, e Right internal carotid artery angiography shows right cerebral artery occlusion, and thus, we performed endovascular thrombectomy. f The collected thrombus

Fig. 3

The resected uterus

Fig. 4

Chest contrast-enhanced computed tomography shows a giant thrombus in the pulmonary artery (arrow)

Fig. 5

Changes in D-dimer, CA125, and CA19-9 levels on the day after the initial bleeding. Abbreviations: CI, cerebral infarction; OP, operation day; PTE, pulmonary thromboembolism.

Fig. 6

Brain magnetic resonance image at 11 months postoperatively (a-b) No cerebral infarction can be observed, and the major cerebral arteries are well visualized