Buitrago, Carlos

Engelter, Stefan

Gobbi, Claudio

Hardmeier, Matrin

Lyrer, Philippe

Steck, Andreas

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Matrin HardmeierClaudio GobbiCarlos BuitragoAndreas SteckPhilippe LyrerStefan EngelterDissection of the internalcarotid artery mimickingepisodic cluster headacheReceived: 4 February 2006Received in revised form: 1 April 2006Accepted: 3 April 2006Published online: 2 March 2007Sirs: Internal carotid artery dis-section (ICAD) is one of the mostfrequent causes of stroke in youngpatients [1,2]. Diagnosis may bedelayed by misinterpreting localsigns of dissection. Typical fea-tures of cluster headache (CH)have rarely been reported as pre-senting symptoms of ICAD [3–7].Our case shows that subtle clinicalfeatures may point to symptom-atic forms of CH, and thereforeboth history taking and clinicalexamination must be particularlyaccurate.Three weeks prior to admis-sion, a 38-year-old man had a firstattack of severe left-sided retro-orbital pain extending into themouth and ear, fluctuating overthree days with pain maximum inthe evening. He noted nasal con-gestion, tearing and hanging of theleft eyelid. The pain subsided;however, mild ear pain persisted.An additional pain attack occurredone week later, in the evening,lasting for several hours. The his-tory was negative for headache. Afirst episode of CH was suspectedby his general practitioner. Oralsumatriptan prompted no relief.Thoracic CT was ordered to ex-clude a mass lesion in the lungapex and ocular pressure wasfound to be normal. On admis-sion, the main complaint was anew attack of severe left-sidedretroorbital and temporal pain. Onexamination the patient had hy-perlacrimation, conjunctivalinjection and Horner‘s syndromeon the left without any furthercranial nerve abnormalities. Oxy-gen inhalation and local nasallidocaine were not effective, nasalsumatriptan resulted in partialrelief. Neurosonography revealeda resistance flow pattern of the leftdistal internal carotid artery (ICA)without signs of atherosclerosis.Magnetic resonance imaging(MRI) and angiography (MRA) ofthe brain and neck demonstratedintramural haematoma and ste-nosis of the left distal ICA (Fig. 1),but no ischaemic lesions.Based on these findings wediagnosed a left ICAD. Treatmentconsisted of acetylsalicylic acid(ASA) 100 mg daily for strokeprevention [8] and verapamil240 mg daily for prophylaxis ofpain attacks. The attacks disap-peared within two weeks, but amoderate continuous pain per-sisted in the left lower face. Clini-cal signs of ICAD are oftendissociated with initial pain andlocal signs followed by retinal orcerebral ischaemic events [1]. In aseries of 65 ICAD patients, painwas the presenting symptom in58.5%, typically unilateral andlocalized, a painful Horner‘s syn-drome was the initial sign in 29%,in two cases, pain mimicked CH[2]. Five similar cases have beenreported [3–7]: in all Horner‘ssyndrome persisted in pain freeintervals, autonomic parasympa-thetic signs variably included lac-rimation, conjunctival injectionand nasal congestion, durationand character of pain was lessstereotyped than in CH.In ICAD, Horner‘s syndrome issecondary to vessel wall injuryaffecting the pericarotid sympa-thetic nerve fibers [1]. The pain ofdissection may trigger a physio-logical trigeminal-autonomic re-flex in susceptible patients,resulting in parasympatheticsymptoms. Experimentally, thisreflex can be activated by painfulstimulation of the first trigeminalbranch with injection of capsaicininducing ipsilateral parasympa-thetic activation as measured byvasodilatation of the ICA [9]. InCH it is hypothesized that in-creased susceptibility to trigemi-nal input or increasedparasympathetic outflow from thebrain stem activate the trigeminal-autonomic reflex during an acuteattack [10].In patients with typical painand autonomic features suggestingCH persistent Horner‘s syndrome,absence of pain free periods, var-iability in both pain attack dura-tion and pain characteristics, as inour case, should raise suspicion ofICAD. Clinical features alone maynot reliably distinguish primaryCH from symptomatic forms ofCH. Neurosonography as ascreening tool [11] and brain MRIwith contrast-enhanced MRA forconfirmation of the diagnosis areuseful in these patients. Treatmentrecommendations of ICAD are notbased on controlled-randomizedDr. M. Hardmeier, MD (&)Prof. C. Gobbi Æ Prof. A. SteckProf. P. Lyrer Æ S. Engelter, MDDept. of NeurologyUniversity Hospital Basel Petersgraben 4,4031 BaselSwitzerlandTel.: +41-61/265-4151Fax: +41-61/265-4100E-Mail: mhardmeier@uhbs.chProf. C. Buitrago, MDDivision of NeuroradiologyDept. of Diagnostic RadiologyUniversity Hospital BaselPetersgraben 4, 4031Basel, SwitzerlandLETTER TO THE EDITORSJ Neurol (2007) 254:253–254DOI 10.1007/s00415-006-0337-2JON2337trials [1,8]. There is still a debate,whether ASA is sufficient or anti-coagulants are needed [12–14].References1. Schievink WI (2001) Spontaneous dis-section of the carotid and vertebralarteries. N Eng J Med 344:898–9062. Biousse V, D’Anglejan-Chatillon J,Massiou H, Bousser MG (1994) Headpain in non-traumatic carotid arterydissection: a series of 65 patients.Cephalalgia 14:33–363. Rosebraugh J, Griebel DJ, DiPette DJ(1997) A case report of carotid arterydissection presenting as cluster head-ache. Am J Med. 102:418–4194. Leira EC, Cruz-Flores S, Leacock RO,Abdulrauf SI (2001) Sumatriptan canalleviate headaches due to carotid ar-tery dissection. Headache 41:590–5915. Mainardi F, Maggioni F, Dainese F,Amista P, Zanchin G (2002) Spontane-ous carotid artery dissection withcluster-like headache. Cephalalgia22:557–5596. Frigerio S, Buhler R, Hess CW, Stur-zenegger M (2003) Symptomatic clusterheadache in internal carotid arterydissection – consider anhidrosis.Headache 43:896–9007. Hannerz J, Arnardottir S, Bro Skejo HP,Lilja JA, Ericson K (2005) Peripheralpostganglionic sympathicoplegia mim-icking cluster headache attacks. Head-ache 45:84–868. Lyrer P, Engelter S (2003) Antithrom-botic drugs for carotid artery dissec-tion. Cochrane database Syst Rev.(3):CD0002559. May A, Buchel C, Turner R, Goadsby PJ(2001) Magnetic resonance angiogra-phy in facial and other pain: neuro-vascular mechanisms of trigeminalsensation. J Cereb Blood Flow Metab21:1171–610. Goadsby PJ (2002) Pathophysiology ofcluster headache: A trigeminal auto-nomic cephalalgia. Lancet Neurol1:251–25711. Arning C (2005) Ultrasonographic cri-teria for diagnosing a dissection of theinternal carotid artery. UltraschallMed. 26:24–812. Norris WN (2005) Extracranial ArterialDissection, anticoagulation is thetreatment of choice: for. Stroke36:2041–204213. Lyrer PA (2005) Extracranial ArterialDissection, anticoagulation is thetreatment of choice: against. Stroke36:2043–204414. Donnan GA, Davis SM (2005) Extra-cranial Arterial Dissection, anticoagu-lation is the treatment of choice. Stroke36:2045Fig. 1 Axial T1 weighted MRI at the level of the skullbase without (A) and with fat suppression combinedwith cranial-caudal saturation (B) show intramuralhaematoma (arrows) in the distal extracranial part ofthe left internal carotid artery. Contrast enhancedMR-angiography (C) depicts marked longitudinalstenosis of the left internal carotid artery (circle)254

Dissection of the internal carotid artery mimicking episodic cluster headache

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Dissection of the internal carotid artery mimicking episodic cluster headache

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