Published online before print June 5, 2008, doi: 10.1161/STROKEAHA.107.510107
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(Stroke. 2008;39:2377.)
© 2008 American Heart Association, Inc.
Research Letters |
Postpartum Cervicocephalic Artery Dissection
From the Department of Neurology (M.A., M.C.-J., C.S., A.D., A.V., K.B., M.G.B.) and the Headache Emergency Centre (A.D.), Assistance Publique, Hôpitaux de Paris, University Hospital Lariboisière, Paris, France; and the Department of Neurology (M.A.), University Hospital, Berne, Switzerland.
Correspondence to Marcel Arnold, MD, Service de neurologie, Hôpital Lariboisière, 2, rue Ambroise Paré 75475, Paris Cedex 10, France. E-mail marcel.arnold{at}insel.ch
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Abstract |
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Background and Purpose— Cervicocephalic artery dissection (CAD) after childbirth is rare. The objective of this study was to determine differences between postpartum and nonpostpartum CAD.
Methods— We compared consecutive patients with postpartum CAD with a control group of women with nonpostpartum CAD.
Results— Of 245 patients with CAD, 102 women <50 years (6 with postpartum CAD and 96 with nonpostpartum CAD) were identified. Vascular risk factors and presenting characteristics did not differ significantly between postpartum CAD and nonpostpartum CAD women. By contrast, patients with postpartum CAD had more often coexisting conditions such as reversible cerebral vasoconstriction syndrome (2 of 6 versus 2 of 96; P=0.017), reversible posterior leukoencephalopathy syndrome (2 of 6 versus one of 96; P=0.009), and subarachnoid hemorrhage without signs of intracranial extension of CAD (2 of 6 versus zero of 96; P=0.003).
Conclusion— CAD and associated conditions should be looked for in women with unusual headache after childbirth.
Key Words: stroke • childbirth • dissection • postpartum angiopathy • reversible cerebral vasoconstriction syndrome
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Introduction |
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Postpartum cervicocephalic artery dissection (ppCAD) is a rare and still poorly understood condition. We add to the 15 cases so far reported1 6 cases prospectively recruited in the last 10 years.
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Methods |
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Patients were recruited from an ongoing monocenter prospective cervicocephalic artery dissection (CAD) registry from January 1997 through October 2005. Inclusion criteria were as follows: female sex; aged <50 years; and diagnosis of spontaneous CAD by catheter angiography or MRI. Risk factors were assessed as reported before.2 ppCAD was defined as CAD occurring within 6 weeks after childbirth.3 Clinical and radiological data were compared between ppCAD and nonpostpartum CAD women. Outcome at 3 months was assessed using the modified Rankin scale.
Statistical Analysis
For differences between postpartum and nonpostpartum women, 
2 test, Fisher exact test, or Mann-Whitney test were used as appropriate. For comparison of outcome, we divided patients into 2 groups: modified Rankin scale 0 to 1 and modified Rankin scale 2 to 6.
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Results |
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Among 245 patients with CAD, 102 women <50 years of age were identified among whom 6 had ppCAD (6%). Four had unilateral vertebral artery dissection (VAD) associated in one with internal carotid artery dissection (ICAD). One had bilateral VAD and one unilateral ICAD.
Baseline characteristics are summarized in Table 1. Headache and/or neck pain were the initial symptoms in all patients and developed between 5 and 18 days after delivery. Headache was bilateral in 4 patients and unilateral in one. It qualified for thunderclap headache in 2. Headache was the only sign of CAD in 2 patients. It was associated with Horner syndrome in one, with transient ischemic attacks in 2, and ischemic stroke in one. Five women had a history of migraine and hypercholesterolemia.
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All migrainous women reported that their headache was different from their previous migraine attacks. None had a history of hypertension but 3 had a transient increase in blood pressure postpartum. The outcome was favorable (modified Rankin scale 0.1) in all 6 women.
The comparison of ppCAD with nonpostpartum CAD is indicated in Table 2. The only significant difference is that women with ppCAD had more often coexisting conditions such as reversible cerebral vasoconstriction syndrome (RCVS; 2 of 6 versus 2 of 96; P=0.017), reversible posterior leukoencephalopathy syndrome (2 of 6 versus one of 96; P=0.009), and subarachnoid hemorrhage (SAH) without signs of intracranial extension of CAD (2 of 96 versus zero of 96; P=0.003).
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Discussion |
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Although women are at a higher risk of stroke postpartum, ppCAD is rare with altogether 15 cases reported so far, 9 with unilateral ICAD, one with bilateral ICAD, one with triple CAD, and one with dissection of all 4 cervical arteries.1,3–7 In addition, 2 patients with basilar artery dissection have been described.8
We report 6 women with 8 ppCAD (6 VAD and 2 ICAD). The mechanism of these ppCADs and their relationship with delivery remain unclear; except for one patient who had chiropractic manipulation, traumatic dissection triggered by a strenuous effort during labor seems unlikely in these women who had uneventful vaginal deliveries and in whom there was an interval of over 5 days between delivery and symptom onset. Furthermore, all women also had uneventful pregnancies without signs of eclampsia. It is however possible that the transient increase in blood pressure observed in 3 women postpartum had played a role.
Among underlying conditions, 5 women had a history of migraine and hypercholesterolemia. Although migraine has been found to be an independent risk factor for CAD in 3 case–control studies,9 there is no evidence so far that hypercholesterolemia is a risk factor for CAD.
We observed no significant difference between ppCAD and nonpostpartum CAD regarding clinical presentation and outcome. Headache and/or neck pain was the initial symptom in all patients. It was followed by ischemic events in 3 (transient ischemic attacks in 2, cerebral infarction in one), associated with a Horner syndrome in one, and completely isolated in 2. Although rare, CAD should thus be added to the long list of causes of headache postpartum, even in the absence of Horner syndrome or of cerebral ischemic events.
The most striking finding in our series is the presence in 4 of 6 patients with ppCAD of associated conditions such as RCVS, reversible posterior leukoencephalopathy syndrome (RPLS; Figure), and SAH (Figure), which, by contrast, were rare in ppCAD affecting only 3 of 96 patients. RCVS occurring postpartum, reported as postpartum angiography, is characterized by prolonged but reversible vasoconstriction of cerebral arteries, usually associated with acute-onset, severe, recurrent headaches with or without associated manifestations such as seizures or ischemic or hemorrhagic stroke.10,11 RCVS may also coexist with RPLS both during and outside postpartum12 and may be associated, in as many as 20% of cases, with a small localized cortical SAH.11,12 We have recently shown that, first, when present in RCVS, both RPLS and cortical SAH were early events occurring in the first few days, and second, that the typical arterial narrowing may not be visible on an initial angiogram.11 In our present series, among the 4 patients with associated conditions, 2 had an angiographically documented RCVS, isolated in one and associated in the other with RPLS. Another patient had RPLS associated with a small localized cortical SAH and the fourth patient had a localized cortical SAH. Both had normal angiograms. Nevertheless, an underlying RCVS is also the most likely explanation in these 2 patients despite the normal angiogram. Thus, among our 6 patients with ppCAD, 2 had definite postpartum angiography and 2 had probable postpartum angiography. Associated risk factors for RCVS were present in only one patient who received bromocriptine to stop lactation and zolmitriptan, a vasoconstrictive antimigraine drug.
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To date, the association of CAD and postpartum angiography has been reported in only one case and was considered coincidental.12 Our series suggests that there is a relationship between these 2 conditions, but the mechanisms underlying this association remain unknown. Furthermore, in our patients as well as in the only other reported one, it is impossible to know which of the 2 conditions occurs first, because both present with acute severe headache. One might speculate about the role of transient arterial wall abnormalities and/or increases in blood pressure induced by postpartum hormonal changes. Migraine might also play a role because all the patients with ppCAD and nonpostpartum CAD with both conditions in our series had migraines.
In conclusion, ppCAD accounts for 6% of spontaneous CAD in women under 50 years of age. There is a significant association of unknown mechanism between ppCAD and postpartum angiography (RCVS). Both conditions should be looked for when women experience unusual severe headaches after childbirth because they require different treatments.
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Acknowledgments |
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We thank Pietro Ballinari, PhD, for statistical advice.
Disclosures
None.
Received November 15, 2007; accepted December 13, 2007.
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References |
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