• Xinxing Hu and Meihong Liu.
• Department of Neurology, Loudi Central Hospital, Loudi, China.
• Medicine (Baltimore). 2024 Dec 13; 103 (50): e40829e40829.

RationaleCatastrophic antiphospholipid syndrome (CAPS) is the most serious type of antiphospholipid antibody syndrome (APS) and can be easily confused with other disorders, such as hemolytic uremic syndrome, disseminated intravascular coagulation and thrombocytopenia syndromes. Timely diagnosis of CAPS poses considerable challenges due to its rarity and the fact that clinicians often lack knowledge of the disease.Patient ConcernsA 21-year-old patient was 32 weeks and 5 days pregnant when she presented to the hospital with a 7-hour history of sudden onset of left-sided limb weakness with no apparent cause. Lupus anticoagulant and/or anticardiolipin antibodies were positive. Head magnetic resonance imaging + magnetic resonance angiography + diffusion weighted imaging: right temporo-occipital insula, right basal ganglia and bilateral radial corona-hemispheric center showed multiple acute-phase cerebral infarction changes and right middle cerebral artery occlusion.DiagnosesCatastrophic antiphospholipid syndrome.InterventionsBy intracranial artery thrombectomy and anticoagulation with low-molecular heparin.OutcomesThe patient's left limb muscle strength recovered to grade 5. A healthy baby boy was delivered by cesarean section. Both mother and child are safe.LessonsThe rarity of CAPS is such that misdiagnosis often occurs, culminating in serious complications and even death, emphasizing the need for early recognition, timely diagnosis and immediate treatment. In CAPS that improves with treatment, monitoring and prevention of recurrence is also essential.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.

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