Spontaneous spinal epidural hematoma without discernable underlying conditions is a rare complication during pregnancy. We report a pregnant woman who developed the hematoma at 16 weeks of pregnancy; the earliest occurrence ever reported. A 36-year-old Japanese primipara with twins felt shoulder, neck and back pain at 16(+1) weeks with progressive quadriplegia. Magnetic resonance imaging revealed a mass in the C3-7 epidural space, compatible with a spinal epidural hematoma, and decompression was performed 9 h after symptom onset. Rehabilitation started and she gave birth abdominally to healthy twin infants at the 29th week. Motor, sensory and sphincter disturbances remained. This case demonstrates that spinal epidural hematoma can occur even without known underlying conditions and even at an early gestational age.

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