An Uncommon Cause of Intracerebral Hemorrhage in a Healthy Truck Driver

OBSERVATION An Uncommon Cause of Intracerebral Hemorrhage in a Healthy Truck Driver

Sankalp Gokhale, MD; Shivani Ghoshal, MD; Sourabh A. Lahoti, MD; Louis R. Caplan, MD

Objectives: To describe a case and review literature for having for more than 20 years. Brain imaging revealed intracerebral hemorrhage caused by migraine. left parieto-occipital lobar hemorrhage. Further investi- gations ruled out arteriovenous malformations. He did Design: Case report. not have any vascular risk factors, including hyperten- sion. Migraine-associated intracerebral hemorrhage was Setting: Beth Israel Deaconess Medical Center, Har- considered to be the most likely diagnosis. vard Medical School, Boston, Massachusetts. Conclusions: Intracerebral hemorrhage associated with Patient: A 54-year-old truck driver with a 2-year his- migraine is believed to result from vasoconstriction lead- tory of atypical headaches. ing to ischemia of the walls of blood vessels, making them leaky and porous. It is important to be aware of this phe- Results: A 54-year-old right-handed truck driver was seen nomenon because vasoactive medications used to treat in consultation with a 2-year history of atypical head- migraine can further aggravate the vasoconstriction and aches. The headaches were dull, throbbing, gradually pro- hence the intracerebral bleed. gressive, and limited over the left occipital area. They were accompanied by right visual field deficit, diplopia, and, at times, confusion. These headaches were notably dif- Arch Neurol. 2012;69(11):1500-1503. Published online ferent from the usual migraine headaches he had been August 27, 2012. doi:10.1001/archneurol.2011.3753

54-YEAR- OLD RIGHT- the individual. At times, if he heard some- handed truck driver was thing, he would continue to hear it after- seen in consultation in ward. These episodes were brief, lasting May 2009. Two years ear- less than a minute. Other episodes were lier, in November 2007, he more prolonged. During some attacks, he hadA an unusual attack of headache. On that would become confused. In one, he saw day, he went to a movie. Shortly after eat- a black dot on the right and then lost some ing ice cream, he developed a dull throb- vision within the right visual field; when bing headache. The headache was lo- he tried to read, he had difficulty reading cated in the left occipital area and gradually to the right. In one episode, he had vi- became severe. Shortly afterward, he started sion loss, tremulousness of his right hand, having difficulty with vision and thought and some difficulty speaking. With other he was seeing double, with the objects seen episodes, he had difficulty reading and one beside and above the other in an writing and some difficulty understand- oblique relationship, most apparent in the ing spoken language. In a few of the epi- right visual field. He remembers a tran- sodes, his right arm and hand felt strange. sient episode of difficulty in speaking He also had frequent headaches, some of shortly thereafter. His headache eventu- which were behind the right eye. Head- ally eased. He was evaluated at a nearby aches would often precede or follow the hospital, where he was found to have nor- prolonged attacks, which typically lasted mal blood pressure and imaging was ob- from 15 minutes to an hour or longer. tained (Figure 1). His medical history was significant for Since then, he has had 2 different types frequent headaches 2 to 3 times a week for Author Affiliations: Author Affil Department of Neurology, of attacks. In 1 type of recurring episode, at least 20 to 25 years with occasional se- Department Beth Israel Deaconess Medical he would see an object that would drift off vere headache once every 3 weeks or so. Beth Israel D Center, Harvard Medical to the right; if he saw someone and he or He quit consuming alcohol a few years ago Center, Harv School, Boston, Massachusetts. she moved, he would still continue to see but was a heavy alcohol user in the past. School, Bost

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R L

160 mm

Figure 1. Computed tomographic scan showing left parieto-occipital hemorrhage. L indicates left; R, right.

P He denied any other major medical problems or drug abuse. He has never had high blood pressure and was not Figure 2. Follow-up axial magnetic resonance image (T2*, gradient-recalled aware of any excess bleeding or clotting disorder. After echo, susceptibility-weighted imaging sequence) showing reduction in the the first episode 2 years ago, he began receiving phe- size of the bleed. A indicates anterior; L, left; P, posterior; and R, right. nytoin sodium and levetiracetam. Examination during the consultation revealed a normal pulse at 72 beats/min, systolic blood pressure of 125 The most likely diagnosis in this case is migraine lead- mm Hg, and diastolic blood pressure of 80 mm Hg. On ing to ICH. Migraines have been associated with and neurological examination, his visual fields were normal known to predispose to vasospasm followed by reperfu- to confrontation but he was slow in reading and made sion edema and intracerebral bleeding. No comprehen- spelling and grammatical errors in writing. He had no sive review of this important neurological entity can be motor, sensory, or reflex abnormalities. Follow-up found in the literature in recent years. To date, 7 in- imaging was performed (Figure 2). stances have been described in the literature and all are in women. To our knowledge, our patient is the first man MIGRAINE-RELATED INTRACEREBRAL with ICH as a complication of migraine. HEMORRHAGE Migraine is a form of vascular headache; various po- tential biological mechanisms link migraine to ischemic The patient has had 2 very different types of paroxysmal stroke as outlined by Kurth,1 such as direct vasoconstric- attacks. One type of attack preceded and followed the tion of cerebral vessels. A number of extensive population- imaging finding of a left posterior intracerebral hemor- based studies have shown an association of migraine with rhage (ICH). This type of attack was characterized by ischemic brain lesions. These studies also showed that right visual field loss, abnormalities of spoken and writ- these so-called silent infarcts are more common in women ten language, and occasionally right limb numbness. than men, are more frequent in smokers, seem to be pre- The content of the attacks was variable. The duration dominant in patients with migraine with aura rather than was 15 minutes to an hour or longer. Many were related common migraine, and are predominant in posterior cir- to severe headache. In some attacks, the abnormal func- culation structures.2-4 tioning gradually progressed rather than beginning sud- denly, and one sensory modality was affected before oth- PREVIOUS REPORTS ers. Except for the 1 attack that was followed by an ICH, the other spells left no damage either clinically or by There have been few cases in the literature about asso- repeated brain imaging. These attacks are consistent ciation of migraine with ICH as opposed to ischemic with migraine with aura, although the almost consistent strokes, for which are there are many studies and obser- involvement of the same brain region is atypical. A vas- vations as detailed before. cular malformation–arteriovenous malformation and an Hemorrhage into the brain parenchyma has also been arterial-dural fistula were considered in the differential reported after migraine attacks. The first report of this diagnosis. A digital-catheter angiogram showed no vas- occurrence was by Dunning,5 who described a young nor- cular malformation, leaving migraine with aura over- motensive woman who developed a right ICH on the whelmingly as the most likely diagnosis for the repeated fourth day of a severe migraine attack. Cole and Aube´ 6 attacks. described 3 patients who developed lobar ICHs after at-

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Angiogram With Interval From Intracranial History of Type of History of Headache to Vessel High Blood Source Age/Sex Migraine Migraines Hemorrhage Location Abnormality Pressure Surgery Outcome Dunning,5 1942 35/F Common Years 3-4 d Right frontal No No No Improved Shuaib et al,8 1989 41/F Classic 25 y 36 h Left basal ganglia No No No Improved Saper,9 1989 54/F NA NA Several Right frontal No No NA NA hours Saper,9 1989 43/F NA NA 5 h Right frontoparietal No No NA NA Cole and Aube´,6 1990 61/F Classic 4 y 6 d Right frontoparietal No No Yes Improved Cole and Aube´,6 1990 45/F Common Years 24 h Right frontoparietal No No Yes NA Cole and Aube´,6 1990 46/F Common 20 y 4 d Left frontoparietal No No No Improved Gautier et al,7 1993 62/F Classic Years 2 h Left frontal No No No Improved Furui and Iwata,10 1993 47/F Common 7 y 10 d Left frontal No No No Improved Current study 54/M Common 25 y Several Left parieto-occipital No No No Improved hours

Abbreviation: NA, not available.

tacks of migraine. These individuals were all young vessel walls. The mechanism is the same as that found women who started having headaches only during adult- in hemorrhage after carotid endarterectomy and in re- hood. The migraine headache responsible was atypical perfusion after brain embolization.11,12 and was very severe in all patients. Examination of 2 of It is interesting to consider the role of vasoactive an- the patients during the headache included no neurologi- timigraine medications in the ICH associated with mi- cal symptoms or abnormalities. Findings on an initial com- graine. Nighoghossian et al13 and Pa´ez de la Torre et al14 puted tomographic scan (with and without contrast en- described cases of multiple intracerebral bleeds in pa- hancement) taken at the emergency department visit tients receiving large doses of vasoactive medications for during the headache of 1 of the patients were normal. treatment of migraine. These medications, if given in large Intracranial bleeding was delayed after the onset of head- amounts, could augment vasoconstriction in acute mi- ache by at least several hours. The ICH occurred when graine, thereby increasing the risk of ICH. the headache had improved markedly or was nearly gone. The episodes of brief visual and auditory persevera- Angiography in all 3 patients showed vasoconstriction tion likely represent focal seizures emanating from the in arteries that supplied the region of hemorrhage and region adjacent to the ICH. A prolonged electroencepha- no vascular malformations. lographic recording confirmed the clinical suspicion of Gautier et al7 described an ICH that occurred during an epileptic focus in the left temporal area. However, sei- migraine in a 62-year-old normotensive woman with nor- zures are a very unlikely explanation of the prolonged mal angiographic findings. Shuaib et al8 described a young multifaceted episodes. The attacks of visual and audi- woman with classic migraine who had delayed ICH (36 tory perseverations stopped after prescription of anti- hours after the onset of headache) without any other de- convulsants but the prolonged attacks were not af- monstrable cause for intracranial bleeding. This obser- fected, furnishing more evidence of the disparate nature vation is similar to that made by Saper9 in 2 patients and of the 2 types of attacks. Furui and Iwata10 in another case report. In all of these patients, including ours, migraine headache was followed by a single large lobar hemorrhage. The Table rec- ords the major data from these case reports. Accepted for Publication: December 13, 2011. The association of migraine and ICH is probably un- Published Online: August 27, 2012. doi:10.1001 derrecognized and hence underreported. The patho- /archneurol.2011.3753 physiological mechanism involves vascular changes dur- Correspondence: Sankalp Gokhale, MD, Department of ing migraine attacks. Vasoconstriction is known to occur Neurology, Beth Israel Deaconess Medical Center, 330 during migraine attacks, which was shown by angiogra- Brookline Ave, Boston, MA 02215 (sankalpsgokhale phy in the patients described by Cole and Aube´.6 This @gmail.com). vasoconstriction can cause ischemia to brain tissue but Author Contributions: Study concept and design: Gokhale also leads to ischemic changes in the small blood vessels and Caplan. Acquisition of data: Gokhale and Caplan. supplied by the constricted arteries. The vascular ische- Analysis and interpretation of data: Gokhale, Ghoshal, La- mia can affect the continence of these vessels, making hoti, and Caplan. Drafting of the manuscript: Gokhale, them leakier for fluid and blood. When vasoconstric- Ghoshal, Lahoti, and Caplan. Critical revision of the manu- tion abates, blood flow to the region is augmented and script for important intellectual content: Gokhale, Gho- reperfusion can cause hemorrhage from the damaged ar- shal, and Caplan. Administrative, technical, and material teries and arterioles. This is consistent with the clinical support: Gokhale and Lahoti. Study supervision: Gokhale observations made earlier that the bleed is usually de- and Caplan. layed, suggesting the role of reperfusion and ischemia of Conflict of Interest Disclosures: None reported.

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