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Meningismus after metaraminol administration in a patient with Familial Mediterranean fever

Russells Hall Hospital, Dudley, West Midlands, United Kingdom, E-mail: drsandeepkapur{at}hotmail.com

To the Editor:

A 38-yr-old male was admitted to our hospital for treatment of Familial Mediterranean fever (FMF)- related severe episodic abdominal pain. Familial Mediterranean fever is a hereditary inflammatory disease characterized by self-limited recurrent attacks of fever and serositis; the recurrent attacks of fever are accompanied by severe abdominal pain, arthritis and/or chest pain along with a marked increase in acute phase reactants.¹ It was decided to implant a spinal cord stimulator (SCS) for pain control because of inadequate pain relief despite high-dose opioid therapy. Following the epidural placement and positioning of the SCS leads under local anesthesia, general anesthesia was administered for sc implantation of the pulse generator. The patient developed hypotension during the course of the anesthetic and this was corrected with fluids and intermittent iv metaraminol boluses (cumulative dose: 10 mg). The procedure was completed uneventfully and the patient awakened. On regaining consciousness, he complained of severe head and neck pain, accompanied by photophobia, nausea and retching. This initially led us to consider a diagnosis of inadvertent dural puncture during epidural placement of the SCS leads. However, on examination, he was found to be pyrexial (38.6°C) and hypertensive (blood pressure 176/104 mmHg); nuchal rigidity and Kernig’s sign were also evident. Intravenous morphine, tramadol and paracetamol were of limited analgesic benefit. Within 24 hr, however, the pain and fever abated and all neurologic symptoms resolved completely. The patient later revealed that he suffered from intermittent headaches of a similar nature, but had always considered them to be ‘migraine attacks’. Case reports have shown recurrent aseptic meningitis, though rare, may occur in FMF.^2,3 Interestingly, the meningitis attacks can be precipitated by injection of metaraminol intravenously; indeed, the metaraminol provocative test has been proposed as a specific diagnostic test for FMF and benign recurrent aseptic meningitis (Mollaret’s meningitis).³ It therefore appears likely that this patient’s meningismus symptoms were triggered by the administration of metaraminol, and we would suggest anesthesiologists remain vigilant to this little-known risk associated with the use of metaraminol in patients with FMF.

Footnotes

Accepted for publication May 17, 2006.

References

1 Ozen S. Familial mediterranean fever: revisiting an ancient disease. Eur J Pediatr 2003; 162: 449–54.[Medline]

2 Collard M, Sellal F, Hirsch E, Mutschler V, Marescaux C. Recurrent aseptic meningitis in periodic disease or Mollaret’s meningitis? (French). Rev Neurol (Paris) 1991; 147: 403–5.[Medline]

3 Barakat MH, Mustafa HT, Shakir RA. Mollaret’s meningitis. A variant of recurrent hereditary polyserositis, both provoked by metaraminol. Arch Neurol 1988; 45: 926–7.[Abstract]

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