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International Headache Society Diagnostic criteria for hemicrania continua Description: persistent strictly unilateral headache responsive to indomethacin
Source publication
Hemicrania continua (HC) is a rare primary headache disorder. It presents some autonomic features (including conjunctival injection, ptosis, eyelid edema, lacrimation, nasal congestion, and rhinorrhea). Response to indomethacin treatment is the mandatory criteria for the diagnosis of HC. However, previously reported literature indicates that there...
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Citations
... Case 1 had an unsatisfying response to indomethacin and was thus switched to a specific cyclooxygenase inhibitor, rofecoxib, as the literature had shown the effect of this in children with Bartter syndrome (12,24,25). ...
Aim:
Bartter syndrome is an autosomal recessive inherited disease in which patients present with hypokalaemia and metabolic alkalosis. We present two apparently non-related cases with antenatal Bartter syndrome type I, due to a novel variant in the SLC12A1 gene encoding the bumetanide-sensitive sodium-(potassium)-chloride cotransporter 2 in the thick ascending limb of the loop of Henle.
Methods:
Blood samples were received from the two cases and 19 of their relatives and deoxyribonucleic acid was extracted. The coding regions of the SLC12A1 gene were amplified using polymerase chain reaction, followed by bi-directional direct deoxyribonucleic acid sequencing.
Results:
Each affected child in the two families were homozygous for a novel inherited variant in the SLC12A1gene, c.1614T>A. The variant predicts a change from a tyrosine codon to a stop codon (p.Tyr538Ter). The two cases presented antenatally and at six months of age, respectively.
Conclusion:
The two cases were homozygous for the same variant in the SLC12A1 gene, but presented clinically at different ages. This could eventually be explained by the presence of other gene variants or environmental factors modifying the phenotypes. The phenotypes of the patients were similar to other patients with antenatal Bartter syndrome. This article is protected by copyright. All rights reserved.
Indomethacin has been used for the treatment of headache disorders since the 1960's, shortly after it was introduced as a treatment for pain and joint swelling in rheumatologic conditions. A subgroup of primary headache disorders, often refractory to other pharmacologic treatment such as triptans and the usual non-steroidal anti-inflammatories, was noted to be exquisitely and absolutely responsive to the analgesic effects of indomethacin. These disorders have been better characterized over the past decade and classified into primary headache disorders of paroxysmal hemicrania (PH) and hemicrania continua (HC). Since the current ICHD-3 beta requires response to indomethacin as a diagnostic criterion, studies on alternative treatments in HC and PH generally occur in patients with intolerance to its gastro-intestinal side effects rather than loss of analgesia effectiveness. More rarely, the development of new headaches have been reported in chronic indomethacin use. In these settings, other classes of medications such as selective cyclooxygenase-2 inhibitors (celecoxib), anti-epileptic agents (topiramate), calcium channel blockers (verapamil, flunarizine), melatonin, and local nerve blocks with anesthetic and steroids have been shown to be effective in case reports and series. We review the literature and provide our clinical recommendations on alternative therapies for the "indomethacin-responsive headaches".
