LETTER TO THE EDITOR
LETTER TO THE EDITOR Reversible cerebral vasoconstriction syndrome triggered by an electronic cigarette: case report S. Vanniera, T. Ronzierea, J. C. Ferreb, V. Lassallea and M. Verina
EUROPEAN JOURNAL OF NEUROLOGY
a Department of Neurology, Rennes University Hospital, Rennes, and bDepartment of Neuroradiology, Rennes University Hospital, Rennes, France
Case report
Correspondence: S. Vannier, Department of Neurology, Rennes University Hospital, 2 rue Henri le Guilloux, 35000 Rennes, France (tel.: +33(0)299284293; fax: +33(0)299284123; e-mail: stephane. [email protected]).
A 39-year-old man presented with a 7day history of headaches and two seizures; he was admitted to the neurology unit. He had no medical history or regular medication and had smoked 60 cigarettes a day for 20 years. There was no use of other drugs or alcohol. Two days after beginning the use of an electronic cigarette in association with 20 cigarettes per day, because of a smoking cessation wish, he had sudden onset headaches. He experienced daily severe thunderclap headaches, three to four per day, lasting for a few minutes. After 7 days associating cigarettes with electronic cigarettes, he had two seizures and suffered from recurrent headaches. The physical and neurological examinations showed moderately high blood pressure at 150/ 90 mmHg. Brain computed tomography scan was normal. Magnetic resonance imaging (MRI) of the brain (Fig. 1a, b),
Keywords: electronic cigarette, reversible cerebral vasoconstriction syndrome, stroke doi:10.1111/ene.12657 Received: 24 August 2014 Accepted: 28 November 2014 Reversible cerebral vasoconstriction syndrome (RCVS) is a rare disorder characterized by transient reversible multifocal arterial constrictions [1]. Severe headaches with or without seizures and focal neurological deficits are the characteristic
(a)
performed 8 days after the first signs, showed splenium corpus callosum vasogenic edema on T2-weighted fluid attenuated inversion recovery (FLAIR) sequence compatible with a posterior reversible encephalopathy syndrome (PRES). On time-of-flight cerebral MR angiography, there were multiple cerebral artery irregularities with alternations of segmental multifocal constrictions and dilatations. There was no abnormality on diffusion-weighted images or after contrast sequences. Blood tests with normal or negative findings included complete hematology counts, erythrocyte sedimentation rate, liver enzymes, blood chemistry, antinuclear antibody panel, anticardiolipin antibodies, blood coagulation studies including anti-phospholipid, angiotensin-converting enzyme, cultures, numerous serology tests for virus, and toxicological urinalysis (cannabis, cocaine, amphetamine and opiates). Cerebrospinal fluid protein concentration was 55 mg/dl (normal 15–40) and there were no leucocytes and no erythrocyte cells. Electronic cigarette liquid toxicological analysis did not find any illicit substances such as amphetamine or cannabis; nicotine concentration was 12 mg/ml. Cerebral vasoconstriction syndrome associated with PRES was the first hypothesis. The patient was started on
presenting feature. RCVS may occur spontaneously or be caused by a precipitating factor such as vasoactive substances. We report here a patient with RCVS triggered by an electronic cigarette. Written informed patient consent was obtained to perform the study. This study provides class IV evidence.
(b)
(c)
Figure 1 MRI of a case of reversible cerebral vasoconstriction syndrome triggered by an electronic cigarette. (a) Initial MRI T2weighted FLAIR sequence demonstrates a posterior reversible encephalopathy syndrome of the splenium corpus callosum. (b) Initial time-of-flight cerebral MR angiography (TOF) demonstrates multiple cerebral artery irregularities with alternations of segmental multifocal constrictions and dilatations (white heads of arrows). (c) One month later, the TOF has normalized and confirms reversible cerebral vasoconstriction syndrome.
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© 2015 EAN
LETTER TO THE EDITOR
oral calcium-channel antagonist (30 mg nimodipine every 6 h) and electronic cigarette cessation. He continued to smoke 10–15 cigarettes per day and with nicotine patches contraindicated. Evolution was good with headache disappearance on the third day and no seizure recurrence. Nimodipine was progressively stopped within 5 weeks. One month later, a follow-up MRI (Fig. 1c) confirmed RCVS with spontaneously resolving stenosis, and there was an improvement of the corpus callosum PRES. Physical and neurological examination results were normal. The patient had no headache and did not use electronic cigarettes again. A complete cessation of smoking was suggested. Discussion To the best of our knowledge, this report represents the first case of RCVS triggered by an electronic cigarette. RCVS is sometimes spontaneous, and often due to precipitating conditions such as
© 2015 EAN
postpartum or exposure to various vasoactive substances. In the prospective study of Ducros et al. [2] RCVS was secondary in 63% and appeared mainly after exposure to vasoactive substances (cannabis, serotonin recapture inhibitors, nasal decongestant, alcohol etc.). A few cases have been described with nicotine patches alone or associated with cigarette smoking [2,3]. The exact role of nicotine in RCSV remains unknown but is probably due to the vasoactive properties of nicotine. We believe that there is an analogy between nicotine patches and electronic cigarettes in the triggering of RCVS. Electronic cigarettes are a new smoking cessation therapy but there is no background. Electronic cigarette aerosols may contain propylene glycol, glycerol, flavorings, other chemicals and nicotine [4] in variable concentrations (0– 20 mg/ml) with a medium level in our situation. The toxicological analyses realized in our case did not identify amphetamines, cocaine or cannabis and there has been no such case reported to date.
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In this particular case, we believe that this association might have led to RCVS. In cases of RCVS we suggest the research of this dual use. Disclosure of conflicts of interest The authors declare no financial or other conflicts of interest. References 1. Calabrese LH, Dodick DW, Schwedt TJ, Singhal AB. Narrative review: reversible cerebral vasoconstriction syndromes. Ann Intern Med 2007; 146: 34–44. 2. Ducros A, Boukobza M, Porcher R, Sarov M, Valade D, Bousser M-G. The clinical and radiological spectrum of reversible cerebral vasoconstriction syndrome. A prospective series of 67 patients. Brain 2007; 130: 3091–3101. 3. Ducros A. Reversible cerebral vasoconstriction syndrome. Lancet Neurol 2012; 11: 906–917. 4. Callahan-Lyon P. Electronic cigarettes: human health effects. Tob Control 2014; 23 (Suppl. 2): ii36–ii40.
Copyright of European Journal of Neurology is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
LETTER TO THE EDITOR Reversible cerebral vasoconstriction syndrome triggered by an electronic cigarette: case report S. Vanniera, T. Ronzierea, J. C. Ferreb, V. Lassallea and M. Verina
EUROPEAN JOURNAL OF NEUROLOGY
a Department of Neurology, Rennes University Hospital, Rennes, and bDepartment of Neuroradiology, Rennes University Hospital, Rennes, France
Case report
Correspondence: S. Vannier, Department of Neurology, Rennes University Hospital, 2 rue Henri le Guilloux, 35000 Rennes, France (tel.: +33(0)299284293; fax: +33(0)299284123; e-mail: stephane. [email protected]).
A 39-year-old man presented with a 7day history of headaches and two seizures; he was admitted to the neurology unit. He had no medical history or regular medication and had smoked 60 cigarettes a day for 20 years. There was no use of other drugs or alcohol. Two days after beginning the use of an electronic cigarette in association with 20 cigarettes per day, because of a smoking cessation wish, he had sudden onset headaches. He experienced daily severe thunderclap headaches, three to four per day, lasting for a few minutes. After 7 days associating cigarettes with electronic cigarettes, he had two seizures and suffered from recurrent headaches. The physical and neurological examinations showed moderately high blood pressure at 150/ 90 mmHg. Brain computed tomography scan was normal. Magnetic resonance imaging (MRI) of the brain (Fig. 1a, b),
Keywords: electronic cigarette, reversible cerebral vasoconstriction syndrome, stroke doi:10.1111/ene.12657 Received: 24 August 2014 Accepted: 28 November 2014 Reversible cerebral vasoconstriction syndrome (RCVS) is a rare disorder characterized by transient reversible multifocal arterial constrictions [1]. Severe headaches with or without seizures and focal neurological deficits are the characteristic
(a)
performed 8 days after the first signs, showed splenium corpus callosum vasogenic edema on T2-weighted fluid attenuated inversion recovery (FLAIR) sequence compatible with a posterior reversible encephalopathy syndrome (PRES). On time-of-flight cerebral MR angiography, there were multiple cerebral artery irregularities with alternations of segmental multifocal constrictions and dilatations. There was no abnormality on diffusion-weighted images or after contrast sequences. Blood tests with normal or negative findings included complete hematology counts, erythrocyte sedimentation rate, liver enzymes, blood chemistry, antinuclear antibody panel, anticardiolipin antibodies, blood coagulation studies including anti-phospholipid, angiotensin-converting enzyme, cultures, numerous serology tests for virus, and toxicological urinalysis (cannabis, cocaine, amphetamine and opiates). Cerebrospinal fluid protein concentration was 55 mg/dl (normal 15–40) and there were no leucocytes and no erythrocyte cells. Electronic cigarette liquid toxicological analysis did not find any illicit substances such as amphetamine or cannabis; nicotine concentration was 12 mg/ml. Cerebral vasoconstriction syndrome associated with PRES was the first hypothesis. The patient was started on
presenting feature. RCVS may occur spontaneously or be caused by a precipitating factor such as vasoactive substances. We report here a patient with RCVS triggered by an electronic cigarette. Written informed patient consent was obtained to perform the study. This study provides class IV evidence.
(b)
(c)
Figure 1 MRI of a case of reversible cerebral vasoconstriction syndrome triggered by an electronic cigarette. (a) Initial MRI T2weighted FLAIR sequence demonstrates a posterior reversible encephalopathy syndrome of the splenium corpus callosum. (b) Initial time-of-flight cerebral MR angiography (TOF) demonstrates multiple cerebral artery irregularities with alternations of segmental multifocal constrictions and dilatations (white heads of arrows). (c) One month later, the TOF has normalized and confirms reversible cerebral vasoconstriction syndrome.
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© 2015 EAN
LETTER TO THE EDITOR
oral calcium-channel antagonist (30 mg nimodipine every 6 h) and electronic cigarette cessation. He continued to smoke 10–15 cigarettes per day and with nicotine patches contraindicated. Evolution was good with headache disappearance on the third day and no seizure recurrence. Nimodipine was progressively stopped within 5 weeks. One month later, a follow-up MRI (Fig. 1c) confirmed RCVS with spontaneously resolving stenosis, and there was an improvement of the corpus callosum PRES. Physical and neurological examination results were normal. The patient had no headache and did not use electronic cigarettes again. A complete cessation of smoking was suggested. Discussion To the best of our knowledge, this report represents the first case of RCVS triggered by an electronic cigarette. RCVS is sometimes spontaneous, and often due to precipitating conditions such as
© 2015 EAN
postpartum or exposure to various vasoactive substances. In the prospective study of Ducros et al. [2] RCVS was secondary in 63% and appeared mainly after exposure to vasoactive substances (cannabis, serotonin recapture inhibitors, nasal decongestant, alcohol etc.). A few cases have been described with nicotine patches alone or associated with cigarette smoking [2,3]. The exact role of nicotine in RCSV remains unknown but is probably due to the vasoactive properties of nicotine. We believe that there is an analogy between nicotine patches and electronic cigarettes in the triggering of RCVS. Electronic cigarettes are a new smoking cessation therapy but there is no background. Electronic cigarette aerosols may contain propylene glycol, glycerol, flavorings, other chemicals and nicotine [4] in variable concentrations (0– 20 mg/ml) with a medium level in our situation. The toxicological analyses realized in our case did not identify amphetamines, cocaine or cannabis and there has been no such case reported to date.
e65
In this particular case, we believe that this association might have led to RCVS. In cases of RCVS we suggest the research of this dual use. Disclosure of conflicts of interest The authors declare no financial or other conflicts of interest. References 1. Calabrese LH, Dodick DW, Schwedt TJ, Singhal AB. Narrative review: reversible cerebral vasoconstriction syndromes. Ann Intern Med 2007; 146: 34–44. 2. Ducros A, Boukobza M, Porcher R, Sarov M, Valade D, Bousser M-G. The clinical and radiological spectrum of reversible cerebral vasoconstriction syndrome. A prospective series of 67 patients. Brain 2007; 130: 3091–3101. 3. Ducros A. Reversible cerebral vasoconstriction syndrome. Lancet Neurol 2012; 11: 906–917. 4. Callahan-Lyon P. Electronic cigarettes: human health effects. Tob Control 2014; 23 (Suppl. 2): ii36–ii40.
Copyright of European Journal of Neurology is the property of Wiley-Blackwell and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use.
