Abstract
In November, 2008, a 37-year-old previously healthy man presented to us with a 6-week history of headache and neck pain occurring while sitting or standing, but not when lying down. CT of the head and cervical spine and one as an outpatient 12 days earlier had been normal. He had no history of trauma, operation, spinal infi ltration, or lumbar puncture. Physical examination was unremarkable. Postural headache caused by intracranial hypotension was diagnosed. He was managed conservatively with bed rest, oral rehydration, and theophylline; he quickly improved and was discharged 3 days later. 1 week later he returned with more severe orthostatic headache and onset of delusional ideas. He believed his illness was a form of “catharsis” of his sins. He exhibited some paranoid ideas, believing that his friend had “polished” his thoughts. Again, physical examination was normal. Laboratory test results and electroencephalography were unremarkable. MRI of the brain (fi gure A) showed bilateral small subdural collections of cerebrospinal fl uid (CSF), also known as subdural hygroma. Contrast images showed diff use, supratentorial and infratentorial enhancement of the dura mater and indicated possible leakage of CSF at the upper cervical spine. The leak was confi rmed by radionuclide cisternography and postmyelography CT (fi gure B and C). Lumbar puncture showed low pressure, normal cell count, and high concentration of protein (1·23 g/L). Conservative therapy was initiated. Psychiatric consultation confi rmed a psychosis, and antipsychotic therapy with quetiapine was started. 14 days later there was no improvement in our patient’s clinical status; he had worsening delusional ideas and had subsequently became stuporous. An epidural blood patch was considered, but MRI showed progression of the subdural hygroma with new subdural haemorrhage; he was transferred to the neurosurgical team and underwent emergency evacuation of the subdural haematoma. 5 days later a laminoforaminotomy was done; a very small CSF leak at the level of the left third cervical pedicle was found and sealed. Postoperatively, our patient fully recovered with no residual headache. He had no recollection of his admission to hospital. Further psychiatric consultation was unremarkable and he had no further delusional ideas. At his fi nal follow-up in May, 2009, he remained well and had resumed work. Postural headache most often arises after lumbar puncture; it rarely occurs as a result of spontaneous intracranial hypotension. Spontaneous intracranial hypotension headache is caused by spontaneous CSF leaks and is an important—but underdiagnosed—cause of headaches of new onset in young and middle-aged patients. Typical MRI fi ndings consist of subdural hygromas and enhanced dura mater; myelography can identify the spinal CSF leak. Therapy includes bed rest, epidural blood patch, and surgical leak repair, although no randomised trials have assessed the outcome of the diff erent strategies. A variety of symptoms can occur, although typically comprise headache, nausea, tinnitus, hypoacusia, or photophobia worsening after sitting and standing. Psychiatric symptoms rarely manifest, and cases of reversible frontotemporal dementia and bipolar disorder have been described. An organic psychosis, as in our case, is possibly caused by diencephalic compression. In our patient, the co-occurrence of a fi rst episode of an underlying primary psychotic disorder cannot be excluded, but is unlikely because the psychotic symptoms developed in parallel to the hygromas, resolved immediately after operation, and have not reappeared.
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