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Background Patients with connective tissue disorders such as Marfan syndrome confer an increased risk of spontaneous intracranial hypotension as they are predisposed to structural weaknesses causing dura ectasia. Rebound intracranial hypertension can develop as an acute complication of epidural blood patches used to treat CSF leaks but has not been previously described in patients with Marfan.Case Study A 17-year-old male patient with Marfan Syndrome who presented with postural headaches was initially diagnosed with intracranial hypotension and successfully treated with an epidural blood patch targeting the spinal CSF leak identified on myelogram with complete clinical and radiological resolution. Approximately 11 months after this, he re-presented with a reversal of his postural headaches, new papilledema, and an opening pressure of 35cmH2O with features of raised intracranial pressure on a brain MRI. Rebound intracranial hypertension with this latency is infrequently reported particularly in a patient with Marfan syndrome.Conclusion This case highlights the importance in recognising this potential delayed complication of CSF leak treatment, and distinguishing between rebound intracranial hypertension and recurrence of intracranial hypotension as there are differing diagnostic and management implications.