This article has Open Peer Review reports available.
Khat – a new precipitating factor for reversible cerebral vasoconstriction syndrome: a case report
© The Author(s). 2016
Received: 29 March 2016
Accepted: 23 November 2016
Published: 15 December 2016
Postpartum reversible cerebral vasoconstriction syndrome is one of the rare reversible cerebral vasoconstriction syndromes. The clinical presentation is usually characterized by recurrent headache, focal neurological deficit, and reversible cerebral vasoconstriction seen on cerebral angiography.
We report a case of a 35-year-old Yemeni woman who presented with headache and focal neurological deficits that occurred 10 days after delivery, with segmental narrowing of cerebral arteries on angiography. She had significant clinical and radiological improvement on follow-up.
The presentation of our patient’s reversible cerebral vasoconstriction syndrome is unusual as she has two possible precipitating factors. In addition to being in the postpartum state, she also has a long history of chewing khat, a vasoactive substance commonly used by immigrants from Yemen. We hope that this case report will increase awareness among physicians about the use of this plant by immigrants from the horn of Africa and Yemen.
KeywordsReversible cerebral vasoconstriction syndrome Khat Postpartum cerebral angiopathy Call–Fleming syndrome
Postpartum reversible cerebral vasoconstriction syndrome (RCVS) is one of the rare RCVSs, and commonly occurs shortly after pregnancy . The syndromes have been given various names, including Call–Fleming syndrome, postpartum cerebral angiopathy, benign angiopathy of the central nervous system, thunderclap headache with reversible vasospasm, migrainous vasospasm, or migraine angiitis . The syndromes are more common in women than in men and peak close to age 40 . RCVS is clinically characterized by acute onset of severe headaches, with neurologic signs and symptoms, and reversible multifocal vasoconstriction of large and medium-sized cerebral arteries, which spontaneously resolves within 3 months . Although the pathogenesis remains unknown, the leading hypothesis involves a disturbance in cerebral vascular tone that leads to multifocal and segmental arterial constriction . RCVS may occur spontaneously or be provoked by various precipitating factors, most commonly the postpartum state or exposure to various vasoactive substances such as cocaine or serotonin reuptake inhibitors . Diagnosis requires demonstration of the characteristic “string of beads” on cerebral angiography with resolution on follow-up imaging . Calcium channel blockers are the most commonly used treatment .
We report the case of a patient with headache and focal neurological deficit that occurred 1 week after delivery, with segmental narrowing of cerebral arteries on angiography and significant clinical and radiological resolution on follow-up. The patient has a history of chewing khat (which contains various amphetamine-like substances), which we think may have provoked her symptoms.
RCVS is a rare, poorly understood and often underdiagnosed syndrome, but recently disease recognition is increasing [5, 6]. The disorder may be short lived and reversible or neurological deficits may become persistent with poor outcome if severe vasoconstriction leads to brain ischemia or hemorrhage . RCVS may occur spontaneously during the postpartum period or as a reaction to various drugs. Our patient presented 1 week after delivery and despite MRI showing a left frontal stroke, she fortunately recovered completely from a clinical standpoint. In the literature, many RCVS precipitating factors have been reported for patients not in the postpartum period, including cannabis, cocaine, serotonin reuptake inhibitors, catecholamine-secreting tumors, immunosuppressants, and many other vasoactive substances . Only a few cases have been reported on the development of postpartum RCVS after the use of vasoactive drugs [9–12]. In our case, the patient presented in her tenth pregnancy; no similar postpartum complication occurred in her nine previous pregnancies. Most reported cases of postpartum RCVS occur within the first three pregnancies . Therefore, we suspect that she may have an additional inciting factor: use of khat. The khat plant (Catha edulis) is commonly chewed in social settings in Yemen, Somalia, and Ethiopia [13, 14]. It contains cathinone, a stimulant drug similar to amphetamines, which is illegal in many Western countries, including the USA and Canada; despite that, many immigrants to these countries still use khat, as did our patient. We suspect her long history of khat usage may have precipitated her condition. We think khat plant usage needs to be recognized among Yemeni immigrants who currently live in developed countries. To the best of our knowledge, there are only a few reported cases of stroke in patients consuming khat [15, 16] and one case report on khat usage and RCVS .
RCVS is no longer an unrecognized disease. In this case, our patient with nine uncomplicated pregnancies developed postpartum RCVS on her tenth pregnancy, which is unusual. We therefore conclude that her long history of khat usage may be causally associated with her RCVS. We want the medical community to be aware that khat is a common illicit drug used by immigrants from the horn of Africa and Yemen.
The authors thank Dr Jeffrey Benjamin (Center for Neuroscience, Department of Medicine, New York Methodist Hospital) for his insight in reviewing this manuscript.
No funding was received for this manuscript.
HB Drafting/writing the manuscript, interpretation of data, and study design. AZ Critically revised the manuscript for intellectual content, analysis, interpretation of data, and supervised the manuscript. Both authors read and approved the final manuscript.
The authors declare that they have no competing interests.
Consent for publication
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-chief of this journal.
The authors have no conflict of interest or disclosure related to this manuscript. This case report has not been published and is not under consideration for publication elsewhere.
Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
- Call GK, Fleming MC, Sealfon S, Levine H, Kistler JP, Fisher CM. Reversible cerebral segmental vasoconstriction. Stroke. 1988;19:1159–70.View ArticlePubMedGoogle Scholar
- Calabrese LH, Dodick DW, Schwedt TJ, Singhal AB. Narrative review: reversible cerebral vasoconstriction syndromes. Ann Intern Med. 2007;146:34–44.View ArticlePubMedGoogle Scholar
- Ducros A, Boukobza M, Porcher R, Sarov M, Valade D, Bousser MG. The clinical and radiological spectrum of reversible cerebral vasoconstriction syndrome: a prospective series of 67 patients. Brain. 2007;130:3091–101.View ArticlePubMedGoogle Scholar
- Singhal AB, Bernstein RA. Postpartum angiopathy and other cerebral vasoconstriction syndromes. Neurocrit Care. 2005;3:91–7.View ArticlePubMedGoogle Scholar
- Ducros A, Bousser MG. Reversible cerebral vasoconstriction syndrome. Pract Neurol. 2009;9:256–67.View ArticlePubMedGoogle Scholar
- Ducros A, et al. Reversible cerebral vasoconstriction syndrome. Handb Clin Neurol. 2014;121:1725–41.View ArticlePubMedGoogle Scholar
- Sattar A, Manousakis G, Jensen MB. Systematic review of reversible cerebral vasoconstriction syndrome. Expert Rev Cardiovasc Ther. 2010;8(10):1417–21.View ArticlePubMedPubMed CentralGoogle Scholar
- Ducros A, et al. Reversible cerebral vasoconstriction syndrome. Lancet Neurol. 2012;11:906–17.View ArticlePubMedGoogle Scholar
- Barinagarrementeria F, Cantu C, Balderrama J. Postpartum cerebral angiopathy with cerebral infarction due to ergonovine use. Stroke. 1992;23(9):1364–6.View ArticlePubMedGoogle Scholar
- Lucas C, Deplanque D, Salhi A, Hachulla E, Doumith S. Benign angiopathy of the puerperium: a clinicoradiological case associated with ingestion of bromocriptine. Rev Med Interne. 1996;17(10):839–41.View ArticlePubMedGoogle Scholar
- Williams TL, Lukovits TG, Harris BT, Rhodes CH. A fatal case of postpartum cerebral angiopathy with literature review. Arch Gynecol Obstet. 2007;275:66–7.Google Scholar
- Granier I, Garcia E, Geissler A, Boespflug MD, Durand-Gasselin J. Postpartum cerebral angiopathy associated with the administration of sumatriptan and dihydroergotamine – a case report. Intensive Care Med. 1999;25(5):532–4.View ArticlePubMedGoogle Scholar
- Kalix P. The pharmacology of khat. Gen Pharmacol. 1984;15(3):179–87.View ArticlePubMedGoogle Scholar
- Widler P, Mathys K, Brenneisen R, Kalix P, Fisch HU. Pharmacodynamics and pharmacokinetics of khat: a controlled study. Clin Pharmacol Ther. 1994;55(5):556–62.View ArticlePubMedGoogle Scholar
- Kulkarni SV, Mughani Y, Onbol E, Kempegowda P. Khat and stroke. Ann Indian Acad Neurol. 2012;15(2):139–40.View ArticlePubMedPubMed CentralGoogle Scholar
- Vanwalleghem IE, Vanwalleghem PW, De Bleecker JL. Khat chewing can cause stroke. Cerebrovasc Dis. 2006;22(2–3):198–200.View ArticlePubMedGoogle Scholar
- Tuladhar AM, Boogaarts HD, de Leeuw FE, van Dijk E. Reversible cerebral vasoconstriction syndrome after chewing khat leaves. Cerebrovasc Dis. 2013;36:158–9.View ArticlePubMedGoogle Scholar