Objective To explore the clinical characteristics of cerebral venous sinus thrombosis （CVST） in patients with antiphospholipid syndrome （APS）， so as to improve the diagnosis and treatment level. Methods The clinical characteristics， laboratory examination， imaging results， treatment and outcome of APS patients with CVST admitted to Xuanwu Hospital， Capital Medical University from January 2012 to April 2019 were analyzed retrospectively， and compared with APS patients without CVST. Results A total of 12 patients with APS and CVST were recruited， including 10 female and 2 male patients. The average age was （35.25±4.61） years old， and median disease duration was 1.9 （0.6， 6.0） months. 7 with acute onset， 2 subacute onset， and 3 chronic onset. The main clinical manifestations were headache in 12 cases， increased intracranial pressure in 10 cases， visual impairment in 7 cases， papilledema in 7 cases， diplopia or eye movement disorder in 4 cases， nausea and vomiting in 4 cases. MR venography （MRV） showed venous involvement in 8 cases of sigmoid sinus， 7 cases of internal jugular vein， 6 cases of transverse sinus and 5 cases of superior sagittal sinus. The incidence of thrombocytopenia in APS patients with CVST （5/12） was significantly higher than that in non CVST patients （11.1%，4/36）， and the difference was statistically significant （P=0.032）. All patients received anticoagulant therapy， some of them received antiplatelet， intravascular recanalization， dehydration， intracranial pressure reduction and hormone therapy. All the 12 patients improved at the time of discharge， and there was no recurrence during the follow-up period of 6-28 months. Conclusions APS patients need to be aware of the possibility of CVST. When the patients with APS have symptoms of intracranial hypertension， it is suggested to improve the imaging examination such as MRV of the head as soon as possible to evaluate whether there is CVST， so as to make early diagnosis and treatment for better prognosis.