complicated coital cephalalgia : three cases with benign evolution

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Complicated coital cephalalgia Three cases with benign evolution JM Martinez, C Roig, A Arboix CEPHALALGIA Martinez JM, Roig C, Arboix A. Complicated coital cephalalgia. Three cases with benign evolution. Cephalalgia 1988;8:265-268. Oslo. ISSN 0333-1024 Three patients with a history of migraine and type-II coital cephalalgia with signs of vertebrobasilar deficiency are presented. Extensive studies including angiography did not show any vascular malformation. Together with the few reports in the lite rature our cases outline a benign form of complicated coital cephalalgia, possibly resulting from ischaemic disturbances triggered by haemodynamic changes occurring in orgasm. • Coital cephalalgia, complicated migraine, headache, vertebrobasilar isch aemia JM Martinez, C Roig, A Arboix, Neurology Service, Hospital Sta Creu i Sant Pau, Autonomous University, Avda. San Antonio Ma Claret 167, 08025 Barcelona, Spain; Correspondence to JM Martinez; Accepted 16 July 1988 The sudden onset of severe headache during sexual activity could be related to serious intracranial events such as subarachnoidal haemorrhage (1, 2). In 1970, Kritz (3) reported 24 patients with headache related to coitus and without abnormalities in the neuroradiologic studies. This entity is better known under the designation "benign coital cephalalgia". It has also been described under other na mes. This condition is still only infrequently reported (4). The concurrence of coital cephalalgia with focal neurologic signs is rarely described (5-7). Intracerebral or subarachnoidal bleeding should always be considered in such cases. However, the possibility also exists that cases of complicated coital hea dache without intracranial haemorrhage have a good prognosis. We report here on three patients with coital headache associated with brain ischaemia and with a favourable evolution. Case reports Case 1 A 50-year-old woman reported a history of common migraine until menopause 2 years earlier and a family history of migraine. She came to our hospital after an explosive occipital headache with dizziness, nausea, and disorientation beginning at the mom ent of orgasm. On admission, she was disoriented with regard to time and space and repeatedly asked the same questions. Tests for recent memory showed severe impairment. Results of neurologic examination were otherwise normal. Although steadily less intense, the he adache persisted for 24 h. The amnesia cleared in 3 days, but the patient did not remember this period. On the 2nd day a right Babinski sign was obtained which lasted 20 days. General examination results, blood pressure, and ECG were normal. Blood tests, urinalysis including catechol-amines, and a cerebrospinal (CSF) study showed normal results. Computed tomography (CT) study on admission and on day 12 and a magnetic resonance study on day 28 did not show any abnormalities. A four-vessel angiographic study on day 7 also gave normal results. The patient has been symptom-free for the past 18 months with 120 mg of propranolol per day. Case 2 A 40-year-old man who smoked 40 cigarettes per day habitually had throbbing hemi-

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Page 1: Complicated coital cephalalgia : Three cases with benign evolution

Complicated coital cephalalgia

Three cases with benign evolution

JM Martinez, C Roig, A Arboix

CEPHALALGIA Martinez JM, Roig C, Arboix A. Complicated coital cephalalgia. Three cases with benignevolution. Cephalalgia 1988;8:265-268. Oslo. ISSN 0333-1024

Three patients with a history of migraine and type-II coital cephalalgia with signs ofvertebrobasilar deficiency are presented. Extensive studies including angiography did notshow any vascular malformation. Together with the few reports in the literature our casesoutline a benign form of complicated coital cephalalgia, possibly resulting from ischaemicdisturbances triggered by haemodynamic changes occurring in orgasm. • Coital cephalalgia,complicated migraine, headache, vertebrobasilar ischaemia

JM Martinez, C Roig, A Arboix, Neurology Service, Hospital Sta Creu i Sant Pau, AutonomousUniversity, Avda. San Antonio Ma Claret 167, 08025 Barcelona, Spain; Correspondence toJM Martinez; Accepted 16 July 1988

The sudden onset of severe headache during sexual activity could be related to serious intracranial eventssuch as subarachnoidal haemorrhage (1, 2).

In 1970, Kritz (3) reported 24 patients with headache related to coitus and without abnormalities in theneuroradiologic studies. This entity is better known under the designation "benign coital cephalalgia". It hasalso been described under other names. This condition is still only infrequently reported (4).

The concurrence of coital cephalalgia with focal neurologic signs is rarely described (5-7). Intracerebral orsubarachnoidal bleeding should always be considered in such cases. However, the possibility also exists thatcases of complicated coital headache without intracranial haemorrhage have a good prognosis.

We report here on three patients with coital headache associated with brain ischaemia and with afavourable evolution.

Case reports

Case 1

A 50-year-old woman reported a history of common migraine until menopause 2 years earlier and afamily history of migraine. She came to our hospital after an explosive occipital headache with dizziness,nausea, and disorientation beginning at the moment of orgasm.

On admission, she was disoriented with regard to time and space and repeatedly asked the samequestions. Tests for recent memory showed severe impairment. Results of neurologic examination wereotherwise normal. Although steadily less intense, the headache persisted for 24 h. The amnesia cleared in 3days, but the patient did not remember this period. On the 2nd day a right Babinski sign was obtained whichlasted 20 days. General examination results, blood pressure, and ECG were normal.

Blood tests, urinalysis including catechol-amines, and a cerebrospinal (CSF) study showed normalresults. Computed tomography (CT) study on admission and on day 12 and a magnetic resonance study onday 28 did not show any abnormalities. A four-vessel angiographic study on day 7 also gave normal results.

The patient has been symptom-free for the past 18 months with 120 mg of propranolol per day.

Case 2

A 40-year-old man who smoked 40 cigarettes per day habitually had throbbing hemi-

Page 2: Complicated coital cephalalgia : Three cases with benign evolution

cranial headache at weekends as a result of heavy drinking.

He came to our hospital after a severe, explosive occipital headache coinciding with orgasm during sexualintercourse. After onset, the headache became throbbing and was combined with nausea; it lasted 10 min,after which he noted weakness and numbness of the right side of face and arm and unsteadiness.

Neurologic examination showed a right hemisensory defect that disappeared within 24h.Electrocardiogram, blood pressure, and cardiologic evaluation findings were normal.

Laboratory studies, including blood coagulation, urine catecholamines, and CSF, showed normal results.CT scan and carotid and vertebrobasilar angiograms were also normal.

During the following 8 months he had only the usual headache after alcohol intake.

Case 3

A 36-year-old man had a 7-year history of classic migraine preceded by scintillating scotoma, successfullytreated with ergotamine.

On one occasion 4 h after a left hemi-cranial headache had disappeared after the usual treatment, thepatient had a holocranial, explosive headache together with vertigo and numbness of the left limbs at the timeof orgasm.

On admission, general examination, including blood pressure and ECG, was unremarkable. Neurologicexamination showed a left homonymous hemianopia and mild left hemihypoaesthesia. CT scan on the 1stday was normal but on the 7th day showed a deep right temporo-occipital lucent area with contrastenhancement (Fig. 1). An angiographic study showed poor filling of terminal branches of the right posteriorcerebral artery. Results of routine laboratory. clotting, immunology, and urine catechol-amine studies werenormal.

Symptoms gradually disappeared over 2 weeks. The patient has since been under treatment with 1.5 mgof pizotifen per day, and in a 2-year follow-up period there have been no recurrences.

Discussion

The term benign coital cephalalgia (BCC) designates acute headache time-related to sexual intercourse ormasturbation, with normal neurodiagnostic studies.

Page 3: Complicated coital cephalalgia : Three cases with benign evolution

This entity has been classified into three varieties on the basis of clinical data and presumedpathophysiology (5). The first is a dull occipital headache progressing with mounting sexual excitement beforeorgasm. The second type is a severe, throbbing, or explosive headache beginning immediately before or atthe moment of orgasm. The third type starts after orgasm and is probably due to low spinal fluid pressure (8).

The second type (type II), probably the most frequent, is referred to as occurring in the occipital or frontalregion or the whole head and may last minutes or hours, sometimes followed by a moderate headache onensuing days. Lance (5) hypothesizes that this form of headache is secondary to a rise in blood pressure anda hyperdynamic circulatory state on the basis of the similarities between haemodynamic changes andheadache occurring at orgasm (9,10), and paroxysm caused by phaeochromocytoma (11). In addition, one ofhis patients with coital cephalalgia presented with a headache of this type after intake of pseudophedrine.

Since BCC is associated with a family or personal history of migraine in some 28% of patients, and afamilial occurrence has also been reported in some cases, several authors believe it to be a variant ofmigraine (4, 6, 12).

Association of coital cephalalgia and symptoms of neurologic deficit is rare. A 25-year-old man withsymptoms of vertebrobasilar insufficiency disappearing 2 days later was reported by Lance (5). Carotidangiography was normal, and a vertebral study was not done.

Levy described a 24-year-old man with a history of vascular headaches related to alcohol intake, a familyhistory of migraine, and a coital episode associated with right-sided motor and sensory deficits (6). CSF, CT,and complete angiographic studies were normal. The only abnormality observed was left temporal slowwaves in the EEG. Nick & Bakouche (7) described three patients, 40, 60, and 61 years old, who presentedwith coital cephalalgia together with Wallenberg syndrome, hemiplegia, and transient global amnesia (TGA),respectively. None had a history of migraine, and two were hypertensive. Angiographic studies were notperformed.

Our three patients all presented with a history of migraine, vertebrobasilar deficit, and type-II coitalcephalalgia. None had hypertension, diabetes, hyperlipidaemia, or cardiopathy. One was a habitual smokerand heavy drinker. Angiographic studies ruled out atheromatosis and vascular malformation.

As in several previously published cases of coital cephalalgia with ischaemic neurologic deficit (5, 6), ourthree patients were young, had antecedents of migraine, and did not have vascular risk factors. We believethat the mechanisms of this condition may be similar to those of complicated migraine (13, 14) and were insome way triggered by the haemodynamic changes occurring during orgasm.

TGA, as presented in our first case, may be related to posterior cerebral artery spasm or ischaemia andhas been associated with both migraine and coitus (15-18). In the third case the occurrence of coitalcephalalgia and ischaemic damage a few hours after a migraine attack may be due to the circulatoryhyperdynamic state during coitus, acting on vascular territory with reduced adaptive capacity. Such casesmight be designated "complicated coital cephalalgia", in contrast to BCC, even though the symptoms evolvefavourably over a short period.

The differential diagnosis of complicated coital cephalalgia must include subarachnoid haemorrhage dueto ruptured aneurysm or arteriovenous malformation, and pheochromocytoma.

Intercourse has been reported as the precipitating cause of subarachnoid hemorrhage in 3.8% to 12% ofthose patients whose bleeding was due to aneurysm (1, 19) and in 4.1% of patients whose bleeding wasbrought about by arteriovenous malformation (19).

Phaeochromocytoma should be ruled out by measuring urinary catecholamine excretion, since symptomsreferable to phaeochromocytoma could be very similar clinically to those of coital headache.

The negative results of angiographic studies and the favourable evolution in

Page 4: Complicated coital cephalalgia : Three cases with benign evolution

our patients question the need for invasive techniques in such cases once intracranial haemorrhage has beenruled out by CT and CSF studies.

In conclusion, we believe that a history of migraine together with coital cephalalgia associated with focalneurologic symptoms might be classified as a complicated form of migraine, triggered by the haemodynamicchanges occurring during orgasm.

References

1. Lundberg PO, Osterman PO. The benign and malignant forms of orgasmic cephalalgia. Headache1974;13:164-5

2. Fisher CM. Headache in cerebrovascular disease. In: Vinken PJ, Bruyn GW eds Handbook of clinicalneurology. Vol. 5. New York: North-Holland Publishing 1968:124-56

3. Kritz K. Coitus as a factor in the pathogenesis of neurological complications. Cesk Neurol Neurochir1970;33:162-7

4. Johns DR. Benign sexual headache within a family. Arch Neurol 1986;43:1158-60

5. Lance JW. Headaches related to sexual activity. J Neurol Neurosurg Psychiatry 1976;39:1226-30

6. Levy RL. Stroke and orgasmic cephalalgia. Headache 1981;21:12-3

7. Nick J, Bakouche P. Les céphalées declenchées par l'acte sexuel. Sem Hop Paris 1980;56:621-8

8. Paulson GW, Klawans HL. Benign orgasmic cephalalgia. Headache 1974;13:181-7

9. Littler WA, Honour AJ, Sleight P. Direct arterial pressure, heart rate and electrocardiogram duringhuman coitus. J Reprod Fertil 1974;40:321-31

10. Mann S, Millar Craig MW, Gould B, Melville D, Raftery ER. Coital blood pressure in hypertensives. BrHeart J 1982;47:84-9

11. Lance JW, Hinterberger H. Symptoms of phaeochromocytoma, with particular reference to headache,correlated with catecholamine production. Arch Neurol 1976;33:281-8

12. Porter M, Jankovic J. Benign coital cephalalgia. Differential diagnosis and treatment. Arch Neurol1981;38:710-2

13. Bartleson JD. Transient and persistent neurological manifestations of migraine. Stroke 1984;15:383-6

14. Broderick JP, Swanson JW. Migraine-related strokes clinical profile and prognosis in 20 patients. ArchNeurol 1987;44:868-71

15. Fisher CM. Transient global amnesia. Precipitating activities and other observations. Arch Neurol1982;39:605-8

16. Growell GF, Stump DA, Biller J, McHenry LC. Toole JF. The transient global amnesia-migraineconnection. Arch Neurol 1984;41:75-9

17. Mayeux R. Sexual intercourse and transient global amnesia. N Engl J Med 1979;12:864

18. Caplan L, Chedru F, Lhermitte F, Mayman C Transient global amnesia and migraine. Neurology (NY)1981;31:1167-70

19. Locksley HB. Natural history of subarachnoid haemorrhage, intracranial aneurysms, andarterio-venous malformations: based on 6368 cases in the cooperative study. J Neurosurg1966;25:219-39