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Article ID: WMC00881 2046-1690

Post Hemorrhagic In A Patient With Orgasmic Paroxysmal Hemicrania

Corresponding Author: Dr. Daniel Jacome, Adjunct Associate Professor, Dartmouth Hitchcock Medical Center. Department of , One Burnham Street, Suite 2, Turners Falls, MA , 01376 - United States of America

Submitting Author: Dr. Daniel E Jacome, MD, Dartmouth Hitchcock Medical Center Department of Neurology, One Burnham Street, Suite 2, 01376 - United States of America

Article ID: WMC00881 Article Type: Case Report Submitted on:07-Oct-2010, 03:38:02 AM GMT Published on: 07-Oct-2010, 07:57:56 PM GMT Article URL: http://www.webmedcentral.com/article_view/881 Subject Categories:NEUROLOGY Keywords:Paroxysma Hemicrania, Hemicrania Continua, Sexual , Sentinel Headache, Hemorraghic , How to cite the article:Jacome D . Post Hemorrhagic Hemicrania Continua In A Patient With Orgasmic Paroxysmal Hemicrania . WebmedCentral NEUROLOGY 2010;1(10):WMC00881 Source(s) of Funding: No funding

Competing Interests: No Conflict of Interest

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Post Hemorrhagic Hemicrania Continua In A Patient With Orgasmic Paroxysmal Hemicrania

Author(s): Jacome D

Abstract lacrimation and conjunctival irritation are common but around one third of the patients report no associated cranial dysautonomic symptoms. Although PH is a primary headache disorder, exceptionally hemorrhagic BACKGROUND: In its classic form sentinel headache stroke may present as PH. Bazan, et al, recently heralds an underlying intracranial aneurysm near described a patient with PH secondary to a contra rupture. Headache related to sexual activity is lateral upper brain stem hemorrhage [3]. In chronic PH normally benign and does not represent a sentinel the pain is consistently recurrent with a variable headache to ulterior intracranial bleeding. Hemicrania frequency, ranging from one to ten attacks a day, to continua is a primary headache only exceptionally one attack every few days in very rare cases {1, 2]. precipitated by acute central nervous system (CNS) Hemicrania continua is not as infrequent as first pathology. believed. In the overwhelming number of cases it METHODS: Clinical case history, repeated constitutes a primary headache. Exceptional post neurological examinations, ancillary imaging traumatic cases, and secondary cases due to brain procedures and review of literature. tumor and to HIV infection, have been described [4]. A RESULTS: A patient with recurrent orgasmic patient with HC associated with an ipsilateral headache manifested as severe left hemicrania of unruptured internal carotid aneurysm was reported by abrupt onset lasting for several hours, developed in Vikelis, et al [5]. Hemicrania continua may evolve from the course of her illness a very severe left side episodic PH and falls within the category of chronic headache while performing physiological valsalva daily headache, along with chronic tension type during defecation, associated with an opposite side headache, newly daily persistent headache and right anterior capsule intracranial bleed. Subsequently chronic . Hemicrania continua is she transitioned into hemicrania continua without characterized by unilateral continuous headache, dysautonomia, ameliorated by valproic acid and by waxing and waning in intensity. The baseline diet-induced weight loss. Her imaging studies showed headache may be accompanied of autonomic signs, no evidence of arteriovenous malformation or stabbing head pain or jabs and jolts, nausea and intracranial aneurysms. sound and light intolerance. In the review of HC CONCLUSION: On this unusual patient, orgasmic provided by Peres, et al, on their own cases and those hemicrania was premonitory of a contralateral collected from the literature, concurrent autonomic hemorrhagic stroke, representing a variant of sentinel symptoms occurred in between 63% to 73% of the headache later evolving into hemicrania continua. patients [4]. Around 25% of those patients with Introduction autonomic symptoms reported only a single autonomic symptom. Normally PH and HC are not triggered by sexual Paroxysmal hemicrania (PH) is a variant of trigeminal activity, and orgasmic headache does not transition autonomic cephalagia (TAC) a family of headache into HC, although one unusual patient previously disorders also encompassing and reported in the literature, developed short lasting unilateral neuralgiform headache with triggered by emotion, following three episodes of coital conjunctival injection and tearing, or SUNCT cephalalgia [6]. A patient with HC evolving from syndrome [1, 2]. Three types of PH are identified: ipsilateral cluster headache responded to the episodic, chronic and hemicrania continua (HC). In administration of valproic acid [7]. episodic PH the patient reports unilateral, paroxysmal, Case Report severe, sharp, throbbing or stabbing pain, with no universal precipitants and no aura [1]. Most attacks have no circadian periodicity. The duration of the A 48 year old female was referred for neurological attacks fluctuates between minutes to hours with a consultation because intense left side continuous mean duration of fifty eight minutes [1, 2]. Autonomic throbbing headache with superimposed stabbing pain, features in the form of ipsilateral rhinorrhea,

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interfering with her sleep and not responding to Orgasmic, coital or (SH) refers to a common analgesics. Her headache followed a small type of exertional headache precipitated by coital or right capsular hemorrhage, triggered by straining hard masturbatory sexual activity [8, 9]. Pre-orgasmic- and during defecation because of constipation. The initial orgasmic headache are recognized by the head pain concurrent with the bleeding was extreme International Classification of Headache disorders, but (10/10 in VAS). She gave history of preceding according to Nick and Bakouche, there is an additional transient episodes of similar left side headaches form of post coital headache lasting from hours to qualified as 8/10 in the VAS, coinciding with days, as a sequel of orgasmic headache [9]. The during sexual intercourse for the last several weeks. orgasmic “explosive” type of headache is acute in Orgasmic headaches lasted for a few hours. During an onset and very intense, to the extreme that the patient early emergency department visit while experiencing feels compelled to seek emergency care, fearing a the original sexual headache, she was found bleeding into the brain. Most SH are benign; hypertensive. Her neurological examination and her exceptionally or embolic head computerized tomography (CT) were found are complications of SH. Of specific relevance normal at the time (illustration 1). Her subsequent to this case, Finelli reported a patient with an head CT following her extreme hemicrania with intracranial basal ganglia hemorrhage triggered by defecation revealed a right anterior capsular bleed on sexual intercourse, secondary to the rupture of a the opposite side to the headache (illustration 2). vascular malformation [10]. Finnelli’s clinical example There was no mass effect, subarachnoid hemorrhage underscores the indication for performing imaging or hydrocephalus. Her head computerized tomography procedures including CT angiogram (CTA), in patients angiography (CTA) showed no aneurysms, beaded or with SH, especially since other intracranial vascular stenotic vessels and no arteriovenous anomalies. pathologies may coexist, such as basilar artery There were no signs of proliferating angiopathy and basilar artery stenosis [11]. Although (“Moyamoya disease”). When symptomatic, and later post stroke pain may complicate up to two thirds of on when asymptomatic, there was no signs of ocular patients suffering strokes, usually the pain takes the ptosis, miosis, weakness of eye movements, ipsilateral form of lateralized complex regional pain syndrome to rhinorrhea, ocular injection or facial flushing on the involved (paretic) side, instead than presenting as repeated neurological examinations. The remaining of PH or HC, as in the current patient. Valsalva her repeated general physical and neurological maneuver precipitates intracranial bleed and cluster examinations was normal including her blood pressure headache in exceptional individuals [12, 13]. Valsalva readings. Her past medical history was significant for is one of the precipitating mechanisms adduced in the hypertension, insulin dependent diabetes mellitus, genesis of exertional, , laughing and crying hyperlipidemia and nephrolithiasis. She had no history headache in general, and of SH in particular. Valsalva of migraine. Medications included simvastatin, mediated headache seemingly results from transient lisinopril, metformin, fluoxetine and insulin at variable intracranial hypertension and pulsing maximal doses. She had no . She had family history of distention of the intracranial vessels, on those multiple sclerosis but no family history of intracranial individuals with a migraine diathesis, perhaps aneurysms, hemorrhagic stroke or migraine. facilitated by incremental central pain sensitization. Significant clinical information obtained on her review Central sensitization can explain why the persistent of symptoms was limited to occasional tingling of the headache in some patients with exertional migraine, toes, interpreted as early signs of diabetic i.e, brought by weight lifting, on whom valsalva is only polyneuropathy. She was prescribed valproic acid at a the initial trigger. As in the specific examples of SH dose of 500 milligrams a day along with tramadol 50 discussed above, imaging of the brain is justified on milligrams four times a day, as needed. Although she patients with exertional headache, because the was not morbidly obese she also embarked on a occasional occurence of secondary valsalva-induced formal weight loss program. In six weeks her headache, on individuals with occult Arnold Chiari continuous headache subsided. In follow up visit malformation and cerebellar tonsillar ectopia, fifteen months later she reports milder episodes of originating from abnormal orgasmic headache; blood pressure readings and hydrodynamics [14]. neurological examination continue normal. Additional pathophysiological explanations advanced for the specific appearance of SH are systemic Discussion hypertension during sexual activity, loss of normal cerebral vascular autoregulation, and painless, segmental or diffuse, persistent cerebral arterial

WebmedCentral > Case Report Page 3 of 8 WMC00881 Downloaded from http://www.webmedcentral.com on 28-Dec-2011, 04:30:46 AM vasospasm [15]. When vasospasm is present in a References patient with SH however, it needs to be distinguished from Call Fleming syndrome or benign cerebral vasospasm, documented in some patients on whom 1. Boes CJ, Dodick DW. Refining the spectrum of spontaneous is the initial chronic paroxysmal hemicranias. Headache 2002; presentation, and on whom, in rare occasions, a 42(8) :699-708 non-aneurysmal subarachnoid bleed is diagnosed [16]. 2. Cohen AS, Matharu MS, Goadsby PJ. Trigeminal Underlying stenosis of the intracranial venous sinus, autonomic cephalalgias: current and future treatments. migraine predisposition and central sensitization with Headache 2007; 47 (6):969-980 repeated intercourse also are potential triggers of SH, 3. Bazan R, Azevedo de Almeida L, Coronetti Gomes as a variant of exertional headache [17]. da Rocha F, Noronha Raffin C, Guimaraes Fonseca R. According to Goadsby and Lipton prevailing Headache secondary to hemorrhagic stroke hypothesis, the pathophysiology of PH and HC as a resembling paroxysmal hemicrania. Arq. subclass of TAC, is based on the over activation of the Neuro-Psiquiatr 2008; 66 (3): 761-762 brain stem trigeminoautonomic reflex, in where pain is 4. Peres MFP, Silberstein SD, Nahmias S, Schecter mediated by the trigeminal ganglion afferents to the AL, Youssef I, Rozen TD, Young WB. Hemicrania meninges, and its autonomic manifestations are continua is not that rare. Neurology 2001; 57: 948-951 mediated by the facial nerve parasympathetic 5. Vikelis M, Xifaras M, Magoufis G, Gekas G, pathways [18]. Mitsikostas DD. Headache attributed to unruptured Sentinel headache, albeit a controversial diagnostic saccular aneurysm, mimicking hemicrania continua. J category, refers to acute headaches, usually Headache Pain 2005; 6 (3):156-158 spontaneous and taking the form of thunderclap 6. Kim HJ, Seo SY. Recurrent emotion-triggered headache, developing on individual harboring an headache following primary headache associated with intracranial aneurysm near full rupture, days or weeks sexual activity. J Neurol Sci 2008; 273 (1-2):142-143 before the actual major bleed [19]. Although an 7. Lambru G, Castellini P, Bini A, Evangelista A, aneurysm micro-bleed or “leak” is commonly Manzoni GC, Torelli P. Hemicrania continua evolving suspected as its cause, because the initial head from cluster headache responsive to valproic acid. computerized tomography (CT) , and the subsequent Headache 2008; 48 (9): 1374-1376 are often negative for subarachnoid 8. Frese A, Rahmann A, Gregor N, Biehl K, Husstedt hemorrhage, the diagnosis is established in retrospect, IW, Evers S. Headache associated with sexual when is maybe too late. It is obvious than identifying activity: prognosis and treatment options. Cephalalgia these patient are critical to proceed to coiling or 2007; 27 (11): 1265-1270 clipping of the aneurysm before a potentially 9. Nick J, Bakouche P. Headache related to sexual catastrophic intracranial bleed occurs. Uterga, et al, intercourse. Sem Hop 1980; 56 (13-14): 621-628 described a patient with recurrent SH of bilateral 10. Finelli PF. Coital cerebral hemorrhage. Neurology temporal localization and an un-ruptured right carotid 1993; 43 (12): 2683-2685 saccular aneurysm, on whom the headaches 11. Delasobera BE, Osborn SR, Davis JE. disappeared following intracranial aneurysm Thunderclap headache with : a case of basilar embolization. Uterga, et al, case highlights the fact artery dissection associated with sexual intercourse. J that the discovery of an “asymptomatic” or un-ruptured Emerge Med 2009; 20 (10): 1-5 aneurysm in similar patients, is not necessary an 12. Carlson AP, Pappu S, Keep MF, Turner P. Large incidental finding [20]. Other than CSF subarachnoid cerebellar hemorrhage during trumpet playing: micro-bleed, alternative hypothesis offered for the importance of blood pressure elevation during the development of sentinel headache include bleeding valsalva maneuver: case report. Neurosurgery 2008; within the wall of the aneurysm, sudden expansion of 62 (6) E-1377 its arterial wall brought by sudden increased in blood 13. Ko J, Rozen TD. Valsalva-induced cluster: a new flow and pulsation transmission, and peri-aneurysmal subtype of cluster headache. Headache 2002; 42 (4): focal [5,19 ] The patient herein described 301-302 represents an atypical example of sentinel orgasmic 14. Van den Bergh R. Headache caused by headaches for an ulterior contralateral capsular craniospinal pressure dissociation in the Arnold hemorrhage , ultimately precipitated by physiological Chiari-syringomyelia syndrome. J Neurol 1992; 239 ( valsalva, in the absence of an underlying cerebral 5):263-266 aneurysm, arterial dissection, or arteriovenous 15. Valenca MM, Valenca LPAA, Bordini CA, Farias malformation.

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da Silva W, Leite JP, Antunes-Rodriguez J, Speciali JG. Cerebral vasospasm and headache during sexual intercourse and masturbatory orgasm. Headache 2004; 44 (3): 244-248 16. Moustafa RR, Allen CMC, Baron J-C. Call-Fleming syndrome associated with subarachnoid hemorrhage: three new cases. J Neurol Neurosurg 2008; 79 (5): 602-605 17. Donnet A, Dufour H, Levrier O, Metellus P. Exertional headache: a new venous disease. Cephalalgia 2008; 28 (11): 1201-1203 18. Goadsby PJ, Lipton RB. A review of paroxysmal hemicranias, SUNCT syndrome and other short-lasting headaches with autonomic feature, including new cases. Brain 1997; 120 (1): 193-209 19. Verweij RD, Wijdicks EFM, van Gijn J. Warning headache in aneurysmal subarachnoid hemorrhage. A case-control study. Arch Neurol 1988; 45 (9): 1019-1020 20. Uterga JM, Gonzalez de Garay MA, Ortiz de Luna I, Uribarri JB. Recurrent coital headache associated with an unruptured carotid saccular aneurysm. Headache 2009; 49 (8): 1232-1239

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Illustrations Illustration 1

PRE-HEMORRHAGIC POST-ORGASMIC NORMAL HEAD CT

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Illustration 2

HEAD CT SHOWS ACUTE RIGHT ANTERIOR INTERNAL CAPSULE HEMORRHAGE

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