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Neurology India, Vol. 58, No. 4, July-August, 2010, pp. 642-644 Case Report Hyper insulinemia in an hemicrania continua patient with sexual headache: Hypothesizing the basis for this unusual association Prakash Sanjay Department of Neurology, Medical College, Baroda, Gujarat-390 001 Correspondence Address:O-19, Doctor's Quarter, Jail Road, Baroda, Gujarat-390 001, drprakashs@yahoo.co.in Date of Acceptance: 02-Nov-2009 Code Number: ni10169 PMID: 20739813 DOI: 10.4103/0028-3886.68689 Abstract The coexistence of hemicrania continua with another primary headache disorder is a very rare event. We report a male patient with both hemicrania continua of 16- year duration and pre-orgasmic headache of three-year duration. Both headache disorders responded to indomethacin. The patient had also in addition persistent elevation of fasting serum insulin.Keywords: Hemicrania continua, hyperinsulinemia, sexual headache Introduction The coexistence of different types of primary headache disorders in the patient is rare, but not uncommon. "Other primary headache disorders" are included in Group 4 of International Headache Society Classification (HIS). Some of the common other primary headache disorders include cluster headache, paroxysmal hemicrania, short-lasting unilateral neuralgiform headache with conjunctival injection and tearing (SUNCT), and headache associated with sexual activity (HSA). [1] The prevalence of these headache disorders is low in the general population and the coexistence of these headache disorders in the same individual is a very unusual phenomenon. We report a patient with hemicrania continua who, later developed pre-orgasmic headache. Case Report A 48-year-old man presented with a 16-year history of continuous right-sided headache. The pain was a constant ache of mild to moderate severity with episodic exacerbations of severe pulsating pain lasting for 10-60 minutes in supra-orbital, retro-orbital and temporo-parietal areas. The frequency of exacerbation was two to seven times a week. The exacerbations were associated with lacrimation and rhinorrhea. The patient denied the presence of aura, nausea, vomiting, photophobia, phonophobia or agitation during the exacerbations. He developed another type of headache since two to three years. The headache, exclusively felt during sexual activity, was band-like and holocranial, especially localized to occipital and neck areas. The headache was noted in about 50% of sexual activities and used to start gradually after a few minutes of foreplay. The severity of the headache used to increase gradually, reaching maximum at the time of orgasm. The stopping of the sexual activity (on achieving orgasm or voluntarily because of the unbearable headache) used to cease the progression of headache. However, he used to get complete relief of headache 5-30 minutes after stopping the sexual activity. He was a known case of hypertension. Physical and neurological examinations were normal Magnetic resonance image (MRI) of the brain and MR angiography were essentially normal. Other laboratory workup was normal, except for high fasting serum insulin levels (120 to 148 m U/l). The oral glucose tolerance test (OGTT) done on a few occasions was within normal limit. The reasons for serum insulin testing (by previous treating physician) was not clear from the history. His father has hypertension and diabetes mellitus.. A diagnosis of hemicrania continua (HC) and headache associated with sexual activity (HSA) (pre orgasmic headache) was made. Treatment with indomethacin (50 mg q8) resulted in complete remission of HC and he had decrease in the frequency and intensity of pre orgasmic headaches. He could prevent pre orgasmic headache by taking additional 50 mg of indomethacin, one to two hours before the sexual activity. In the 15-month follow up he was headache free except for the days of drug default. On each occasion, it resulted in recurrence of both types of headache. Repeat serum insulin testing on two more occasions during the remission phase remained elevated (126 and 132 m U/l). Discussion Clinical characteristics of headaches in our patient fulfilled the diagnostic criteria of HC and HSA. [1] The first report of two primary headache disorders occurring in the same patient was HC with cluster headache (CH). [2] Subsequently cases of HC in association with other primary headache disorders have been reported [Table - 1]. [2],[3],[4],[5],[6],[7],[8],[9] In most of these cases, HC evolved from another primary headache disorder. There are only two case reports in whom HC and another primary headache disorder occurred concurrently, similar to our patient. [3],[4] Migraine and benign exertional headache are the two most common headache disorders associated with HSA. A few reports described HSA in association with another primary headache disorder: HSA in association with CH [10] and HSA in association with hypnic headache. [11] It is difficult to determine, on the basis of case reports, whether the occurrence of this rare association (HC with HSA) in the same patient is just a coincidence or whether they have a common pathophysiology. The clinical characteristics of HC and HSA are totally different. This suggests that the association is more a coincidence. However, a common pathogenic relationship between HC and HSA has been speculated. Response of both headache disorders to indomethacin may suggest a common pathophysiology. Mateo et al.[12] reported a patient with chronic paroxysmal hemicrania (CPH) in association with benign cough headache. Both the headaches responded to indomethacin. Evers et al. [13] demonstrated impaired cerebrovascular autoregulation in patients with HSA and suggested a possibility of stretching of small intracranial arteries to induce the activation of pain- processing trigemino- vascular system. They also considered the possibility of primary dysbalance of the trigemino-vascular system and impaired cerebrovascular autoregulation as a secondary effect. [13] This may be an explanation for HSA in a patient with HC. Activation of the hypothalamus has been observed in patients with HC. In the same way, activation of the hypothalamus has been demonstrated during sexual activity and orgasm. [14] Therefore, the possibility of HSA can be speculated in a patient with HC. Another unique observation in our patient was persistent hyper insulinemia. Insulin levels are tested in the presence of risk factors for metabolic syndrome or cardiovascular syndrome. [15] There was no risk factor for metabolic syndrome (or hyper insulinemia) in our patient. The family history of diabetes mellitus and hypertension may be a reason for testing insulin level. Hyper insulinemia may be related to both normal and abnormal glucose levels. In patients with insulin resistant state, abnormally high insulin level may have either normal or low glucose level. Normal OGTT supports the presence of insulin resistant state. The presence of hyper insulinemia in our patient is largely unexplained. However, impaired insulin sensitivity has been demonstrated in patients with migraine. [16] McCarthy et al. [17] have demonstrated an association between insulin receptor gene and migraine, and suggested a role of insulin receptor in migraine pathogenesis. The clinical feature of the exacerbation of HC is similar to that of migraine. [18] Therefore, a similar type of association is possible in patients with HC. However, a single case report is not sufficient to say that hyper insulinemia predisposes such a type of headache, and at this point of time in clinical practice, a routine estimation of serum insulin level cannot be suggested. However, because of the marked similarity in clinical profiles of HC to that of migraine, we suggest further observational studies to confirm it. If it is confirmed in other patients with HC, it may help in understanding the pathophysiolgy of this uncommon primary headache disorder. References
Copyright 2010 - Neurology India The following images related to this document are available:Photo images[ni10169t1.jpg] |
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