Summary and Background: Spontaneous spinal epidural hematoma (SSEH) is a known occurrence in patients on anticoagulant therapy. There is an increased risk of developing hematoma after the spine surgery if anticoagulation therapy is reinstated. Purpose of Study: The purpose of the study was to find out solution related with perioperative anticoagulant therapy in high-risk cases if patient redevelops hematoma and paraplegia due to continuation of anticoagulant therapy. Case Report: A 30-year-old male presented to us with history of progressive paraparesis. He had history of mitral valve replacement twice followed by cerebrovascular stroke and on regular oral anticoagulant therapy. Magnetic resonance imaging revealed SSEH from C6-T1 level with cord compression. Initial decision was taken to conservatively treat as his coagulation parameters were altered and he was on high-risk for developing thromboembolism related complications if anticoagulant medicines were stopped. However, urgent laminectomy and evacuation of SSEH had to be performed due to rapid worsening of neurology. Postoperatively, patient had significant neurological recovery and anticoagulant therapy reinstated after 12 h of surgery. Patient developed acute paraplegia within 2 hours of anticoagulant therapy due to post-operative hematoma, which was drained out by opening the wound bedside. He regained neurological recovery within 5 min. Anticoagulation therapy was withheld for next 36 hours and reinstated with low-dose intravenous heparin followed by low-molecular weight heparin without any complications. His coagulation parameters and 2-D echo were followed up daily to check cardiac conditions. Patient improved clinically and became self-ambulatory. Conclusion: Post-operative hematoma after spine surgery should be kept in mind in patients who are on anticoagulant treatment. Reinstating anticoagulation treatment in such high-risk patients should be done with lot of caution and initially with low-dose heparin followed by regular anticoagulation therapy. Close observation on neurological status is must to avoid permanent neurological injury. Keywords: Spontaneous spinal epidural hematoma, Anticoagulat treatment, Decision dilemma