Acute recurrent cerebral infarction caused by moyamoya disease complicated with adenomyosis:A case report

BACKGROUND Moyamoya disease is essentially an ischemic cerebrovascular disease.Here,we describe a case of acute recurrent cerebral infarction caused by moyamoya disease with concurrent adenomyosis which,to our knowledge,is the first in the literature.A literature review is also presented.CASE SUMMARY A 38-year-old female presented to the Research and Treatment Center of Moyamoya Disease in our hospital with"left limb weakness"as the main symptom.She was diagnosed with acute cerebral infarction and moyamoya disease through magnetic resonance imaging and digital subtraction angiography.Prior to this,she had experienced a prolonged menstrual period of one-month duration.This was investigated and adenomyosis was diagnosed.After passing the acute cerebral infarction phase,the patient underwent surgery for adenomyosis followed by combined cerebral revascularization.During the postoperative follow-up,improvements of the perfusion imaging stage and modified Rankin Scale were observed.A review of the literature showed only 16 reported cases of gynecological diseases complicated with stroke.The clinical characteristics,pathogenesis,therapeutic effects,and long-term prognosis of these cases have been studied and discussed.CONCLUSION In patients with moyamoya disease,early management of gynecological-related bleeding disorders is essential to prevent the complications of cerebral events.

Risk factors of postoperative cerebral hyperperfusion syndrome and its relationship with clinical prognosis in adult patients with moyamoya disease

Background To investigate the incidence,risk factors,and clinical prognosis of cerebral hyperperfusion syndrome(CHS)after superficial temporal artery-middle cerebral artery anastomosis combined with encephalo-duro-arterio-synangiosis(STA-MCA/EDAS)in adult patients with moyamoya disease(MMD).Methods The clinical data of 160 adult patients with MMD treated by STA-MCA/EDAS from January 2016 to January 2017 were retrospectively analyzed.According to CHS diagnosis,MMD patients were divided into CHS and non-CHS group.Univariate and multivariate analysis of risk factors and Kaplan-Meier curve of stroke-free survival for CHS were performed.Results A total of 12 patients(7.5%)developed postoperative CHS,of which 4 patients(2.5%)presented with cerebral hemorrhage.Univariate and multivariate analysis showed moyamoya vessel on the surgical hemisphere(OR=3.04,95%CI=1.02-9.03,P=0.046)and left operated hemisphere(OR=5.16,95%CI=1.09-21.34,P=0.041)were independent risk factors for CHS.The other variables,such as age,gender,presentation,hypertension,diabetes,smoking,mean mRS score on admission,modified Suzuki stage and pre-infarction stage on surgical hemisphere,and bypass patency,had no association with postoperative CHS(P>0.05).At final follow-up with average 38 months,there were 18 out of 133 patients(13.5%,4.91%per person year)presented with newly developed complications.There was no significant difference between newly developed complications,mean mRS scores,and Kaplan-Meier curve of stroke-free survival in patients with and without CHS(P>0.05).Conclusion The concentration of moyamoya vessels and left operated hemisphere was independent risk factors for CHS,which could not affect the clinical prognosis if treated timely and properly.The current study offers a new perspective of moyamoya vessels and supporting data for choosing MMD candidates on cerebral revascularization.

Superficial temporal artery-middle cerebral artery bypass combined with encephalo-duro-myo-synangiosis in treating moyamoya disease： surgical techniques, indications and midterm follow-up results

Background Surgical interventions for moyamoya disease include direct and indirect revascularizations. This study aimed to evaluate the therapeutic effect of superficial temporal artery-middle cerebral artery bypass combined with an indirect revascularization procedure, encephalo-duro-myo-synangiosis, in the treatment of moyamoya disease. Methods From October 2005 to November 2009, we performed this combined revascularization procedure in 111 patients with different types and stages of moyamoya disease. The superficial temporal artery, middle meningeal artery and the deep temporal artery were evaluated for individualized surgical planning in these cases. The integrity of the deep temporal artery and the middle meningeal artery network, and the pre-existing spontaneous anastomoses of the distal branches of the external carotid artery with the cortical arteries were well preserved. The mean follow-up time was 72.5 months, all clinical and radiological data were retrospectively reviewed. Results A total of 198 stomas were performed in 122 hemispheres, all remaining patent until the last follow-up. The encephalo-duro-myo-synangiosis resulted in extensive anastomoses of the deep temporal artery （100%）, the middle meningeal artery （90.9%）, and the sphenopalatine artery （39.8%） with the cortical arteries, respectitvely. The superficial temporal artery, deep temporal artery, and the middle meningeal artery were significantly thickened in 88 patients as determined by digital subtraction angiography at follow-up. The relative cerebral blood flow increased significantly within one week after the operation. At 6 months post the operation, the relative cerebral blood flow was further increased by 15.5% from the gradual formation of anastomoses as a result of indirect revascularization. Transient ischemic attacks were effectively reduced or totally arrested. The neurological deficits significantly improved in 37 patients, with the National Institutes of Health Stroke Scale scores lowered by 2-8. There was no rehemorrhage in hemorrhagic moyamoya disease patients. Conclusion This study showed that the superficial temporal artery-middle cerebral artery bypass combined with encephalo-duro-myo-synangiosis can achieve good therapeutic effect in the treatment of moyamoya disease.

Factors associated with delayed intracerebral hemorrhage after superficial temporal artery-middle cerebral artery bypass in stenoocclusive cerebrovascular diseases

Background Intra-cerebral hemorrhage （ICH） is a devastating complication that can result from superficial temporal artery-middle cerebral artery （STA-MCA） bypass in patients undergoing treatment for steno-occlusive cerebrovascular disease （CVD）.There is a clinical need to find the possible risk factors to prevent ICH,as it is a significant cause of mortality and morbidity.The aim of the study was to investigate the factors associated with delayed ICH after STA-MCA bypass in patients with steno-occlusive CVDs.Methods We retrospectively analyzed the records of 163 patients seen from 2002 to 2011 with STA-MCA bypass for steno-occlusive cerebrovascular diseases at the Department of Neurosurgery,Xuan Wu Hospital,Beijing.Demographic and clinical data,including age,gender,vascular risk factors,preoperative syndrome,preoperative National Institutes of Health Stroke Scale （NIHSS）,ipsilateral ischemic lesions,classification of steno-occlusive CVDs,donor branches of STA,graft patency,postoperative hypertension,and postoperative-increased MCA velocity were recorded and analyzed.Binary Logistic regression served to identify factors associated with delayed ICH after STA-MCA bypass.Results We identified 8 （4.9%） patients with delayed ICH after STA-MCA bypass.Patients with hypertension,preoperative stroke,ipsilateral ischemic lesions,postoperative hypertension and postoperative-increased MCA velocity were significantly more prone to experiencing delayed ICH after STA-MCA bypass.Logistic regression analysis shows ipsilateral ischemic lesions,postoperative hypertension,and postoperative-increased MCA velocity remained independent predictors for delayed ICH after STA-MCA bypass.Conclusion Despite the varied associated factors in patients with steno-occlusive CVDs,ipsilateral ischemic lesions,postoperative hypertension,and postoperative-increased MCA velocity could be associated with delayed ICH after STAMCA bypass.