Unusual association of diseases/symptoms

CASE REPORT

Postpartum vertebral artery dissection with posterior
reversible encephalopathy syndrome
Mitsushige Nishimura, Eiji Hiraoka, Kenji Kanazawa, Hozuka Akita
Department of General Internal
Medicine, Kobe University
Hospital, Kobe, Hyogo, Japan
Correspondence to
Dr Mitsushige Nishimura,
kazu9313@hotmail.co.jp
Accepted 23 June 2015

SUMMARY
We diagnosed postpartum eclampsia with posterior
reversible encephalopathy syndrome (PRES) in a 35-yearold woman who began experiencing headaches after
delivery. Cervical MR angiography (MRA) suggested
concomitant vertebral artery (VA) dissection. Antiplatelet
therapy was not indicated. Each episode resolved
spontaneously. The patient subsequently developed
sudden onset of thunderclap headache. She experienced
a tonicoclonic seizure in the outpatient clinic. On the
basis of clinical course and MRI, we diagnosed
postpartum eclampsia with PRES. Antiplatelet therapy
was not indicated, as there were no signs of stroke.
Antiepileptic and antihypertensive medicines were used
for a short term to control seizure and blood pressure.
Subsequent MRI 17 days after discharge showed
resolution of the abnormal signals, but the intramural
heme signals became apparent on the dissecting
segment of VA. Subsequent MRI after 3 months
indicated resolution of arterial dissection. To the best of
our knowledge, this represents the third report of
postpartum cervicocephalic artery dissection with PRES
in the literature.

BACKGROUND
Headache is a common finding in the postpartum
period. Stella et al1 reported causes of postpartum
headache for 95 women >24 h after delivery.
Posterior reversible encephalopathy syndrome
(PRES) is one entity causing postpartum headache.
According to that study, PRES accounts for about
4% of cases. The pathogenesis of PRES remains
unclear, but appears related to disordered cerebral
autoregulation and endothelial dysfunction.
However, cervicocephalic artery dissection
(CAD) after childbirth is rare. No patients with
CAD were found by Stella et al.1 Arnold et al2
reported that 6% of 102 women with CAD
<50 years old were in a postpartum condition. In
that study, patients with postpartum CAD had
coexisting PRES more often than patients with
non-postpartum CAD.2 Concomitant CAD and
PRES after childbirth is rare. We present a case of
postpartum headache with PRES and concomitant
vertebral artery dissection in a 35-year-old woman.

CASE PRESENTATION
To cite: Nishimura M,
Hiraoka E, Kanazawa K,
et al. BMJ Case Rep
Published online: [please
include Day Month Year]
doi:10.1136/bcr-2014207332

A 35-year-old woman had a normal pregnancy and
delivered her first baby without any complications.
Blood pressure (BP) remained within the range of
100–110/70–80 mm Hg during the pregnancy. She
began experiencing episodic headaches on postpartum day 5, with BP 150/90 mm Hg. Initially,

the headaches were located in bioccipital areas and
posterior neck with pressure qualities, resolving
spontaneously after 7 h. On postpartum day 8, she
developed sudden onset of a thunderclap headache,
at which time CT of the head was performed and
read as normal in the emergency room. The headaches persisted, with orthostatic qualities, and were
worse when supine and relieved when upright. At
this stage, she was referred to our medical outpatient clinic on postpartum day 11.
BP was 154/96 mm Hg, much higher than her
usual pregnant outpatient BP. Axillary temperature
was 37°C, heart rate was 72 bpm and respiratory
rate was 15 breaths/min. Neurological examination
showed no abnormalities.

INVESTIGATIONS
Laboratory investigations were all within normal
limits, including blood cell count, liver enzyme levels,
renal function and electrolytes. Urinalysis revealed no
proteinuria. Cerebrospinal fluid examination yielded
normal results. Fluid-attenuated inversion recovery
images of MRI revealed bilateral multiple hyperintense areas, predominantly in the subcortical white
matter (figure 1A). Diffusion-weighted imaging
demonstrated no evidence of ischaemic stroke
(figure 1B). MR angiography (MRA) showed no segmental arterial narrowing or dilation in the cerebral
vessels (figure 1C). However, the right vertebral
artery at the cervical level showed stenosis
(figure 1D). After MRI, the patient experienced a
tonicoclonic seizure lasting for 1 min. Her level of
consciousness gradually normalised postictally,
without treatment. She was admitted to our intensive
care unit.

DIFFERENTIAL DIAGNOSIS
A woman in the puerperal period who presents
with thunderclap headaches and neurological symptoms may have PRES, reversible cerebral vasoconstriction syndrome (RCVS) or CAD. PRES may be
reversible, but CAD is not, so these pathologies
require differentiation. Identification of one entity
causing headaches tends to make us overlook other
causes. Variations in the quality of headaches over
the clinical course may indicate dual entities as
causes of headaches.
We reached a diagnosis of postpartum eclampsia
with PRES, based on the clinical course and previously reported MRI findings.3 RCVS is usually
diagnosed from the presence of arterial narrowing
on angiography. However, MRA of our patient on
the day of admission showed no arterial narrowing.

Nishimura M, et al. BMJ Case Rep 2015. doi:10.1136/bcr-2014-207332

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Unusual association of diseases/symptoms

Figure 1 MRI and MR angiography (MRA) on the day of admission. (A) Fluid-attenuated inversion recovery images showing multiple high-intense
areas in the bilateral occipital area (arrows). (B) Diffusion-weighted images showing no high-intense area, which suggests that there is no ischaemic
stroke. (C) Cranial MRA. There is no segmental arterial narrowing, dilation or occlusion. (D) Right: cervical MRA, left: horizontal image of MRA at
the level of fifth cervical vertebra. Cervical MRA showing stenosis at the right vertebral artery.

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Nishimura M, et al. BMJ Case Rep 2015. doi:10.1136/bcr-2014-207332

Unusual association of diseases/symptoms

Figure 2 Subsequent MRI and MR angiography (MRA) 17 days after discharge. (A) Fluid-attenuated inversion recovery images showing that
multiple high-intense areas in the bilateral occipital area, which were observed on the day of admission, disappeared. (B) Right: cervical MRA, left:
horizontal image of MRA at the level of fifth cervical vertebra. Cervical MRA showing intramural heme signals at the level of fifth cervical vertebra
(arrow and circle).

TREATMENT
The headaches resolved and further seizures were prevented by
intravenous treatment with phenytoin and magnesium sulfate.
Nicardipine was also administered to control BP. The patient
was discharged without any neurological sequela.

OUTCOME AND FOLLOW-UP
The clinical course was good until subsequent MRI 17 days
after discharge was performed. The imaging showed resolution
of the multiple abnormal signals in the white matter (figure 2A).
However, MRA was suggestive of right vertebral artery dissection at the level of the fifth cervical vertebra, based on the presence of an intramural haematoma (figure 2B),4 which showed
stenosis on the day of admission. So, dissection was considered.
Antiplatelet therapy for prevention was not indicated, as there
were no signs of stroke. All intramural heme signals had
resolved by follow-up MRA 3 months later (figure 3).

DISCUSSION
Headaches are a frequent symptom in the postpartum period. In
the majority of cases, such headaches represent either migraines
or tension-type headaches, which are both easily treated.5
Rarely, headaches in the postpartum patient signal a lifeNishimura M, et al. BMJ Case Rep 2015. doi:10.1136/bcr-2014-207332

threatening condition. Our patient initially reported of a bioccipital headache without neurological deficit 5 days after delivery.
This condition was self-limited. However, she developed sudden
onset of a thunderclap headache 8 days after delivery. She also
experienced a tonicoclonic seizure 11 days after delivery. The
quality of the headache, which varied over the clinical course,
suggested dual entities as their causes.
PRES is characterised by headaches, seizures and visual changes.3
Characteristic imaging features often include focal regions of symmetric oedema in the posterior brain parenchyma, as well as in
other brain regions.3 The pathogenesis is hypothesised to involve
disorders of cerebral autoregulation and/or endothelial dysfunction
with increased permeability of the blood–brain barrier.3 PRES generally develops in the setting of preeclampsia/eclampsia, but may
also develop in the puerperium and as a presenting feature of late
eclampsia.3 Most patients with PRES do well if the seizures and
hypertension are appropriately managed. Unfortunately, some
more severe cases can result in lasting neurological morbidity or
even mortality due to ischaemic stroke or haemorrhage.
RCVS and PRES are said to have a shared pathophysiology.5
RCVS should be diagnosed based on the presence of vasoconstriction on MRA. We could not diagnose RCVS in this case due
to the absence of narrowed blood vessels on MRA.
3

Unusual association of diseases/symptoms

Figure 3 MR angiography (MRA) 3 months after discharge. Cervical MRA showing the resolution of dissecting segment (arrow and circle).

CAD is a rare postpartum complication only previously
described in case reports and one small case series involving a
total of six patients.2 6–10 A recent registry database identified
that 2.4% of cases of spontaneous CAD occurred in the postpartum period.2 Postpartum CAD remains poorly understood.
According to the case series of six cases with postpartum CAD,
four had vertebral artery dissection and two had PRES.2
Stella et al1 reported causes of postpartum headache for 95
women >24 h after delivery. According to that study, PRES
accounts for about 4% of cases. However, no patients with
CAD were identified in that study. Arnold et al2 reported that a
postpartum condition accounted for 6% among 102 women
<50 years old with CAD. In that study, patients with postpartum CAD had coexisting PRES more often (2 of 6 patients)
than patients with non-postpartum CAD (1 of 96 patients;
p=0.009).2 We were unable to find any studies showing how
many patients with PRES show concomitant CAD.
Since the dissection segment does not necessarily show the
intramural haematoma at the acute stage, the stenosis on MRA
on admission at the level of the fifth cervical vertebra was considered for dissection in this case. Therefore, careful observation
of clinical symptoms for arterial dissection was made. The prognosis of CAD is related to the severity of associated ischaemic
stroke or subarachnoid haemorrhage. Morbidity and mortality
of acute CAD depend on the specific arteries involved and location of the dissection. Complete or excellent recovery occurs in
70–85% of patients with CAD, with major deficits in 10–25%,

Learning points
▸ Imaging studies are crucial when a patient in the puerperal
period experiences headache with neurological symptoms or
thunderclap characteristics.
▸ The possibility of multiple entities causing headache must be
considered when the quality of the headache varies over the
clinical course.

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and death in 5–10% of cases.11 The optimal strategy for prevention of stroke in patients with CAD is controversial.
We used the key words “postpartum cervicocephalic artery
dissection” and “posterior reversible encephalopathy syndrome”
or “PRES” in the PubMed search. No other case reports of postpartum CAD with concomitant PRES were found. To the best
of our knowledge, the present case represents only the third to
be described in the literature.
Contributors KK identified the patient in the outpatient department. MN, EH and
KK managed the patient in the ward after admission. MN wrote the draft of this
case report. EH, KK and HA reviewed and revised this case report. MN is the
guarantor.
Competing interests None declared.
Patient consent Obtained.
Provenance and peer review Not commissioned; externally peer reviewed.

REFERENCES
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Hosley CM, McCullough LD. Acute neurological issues in pregnancy and the
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posterior cerebral artery. J Clin Neurosci 2007;14:576–81.
Soltanolkotabi M, Ansari SA, Shaibani A, et al. Spontaneous post-partum cervical
carotid artery dissection in a patient with reversible cerebral vasoconstriction
syndrome. Interv Neuroradiol 2011;17:486–9.
Van de Kelft E, Kunnen J, Truyen L, et al. Postpartum dissecting aneurysm of the
basilar artery. Stroke 1992;23:114–16.
Waidelich JM, Bullough AS, Mhyre JM. Internal carotid artery dissection: an unusual
cause of postpartum headache. Int J Obstet Anesth 2008;17:61–5.
Arnold M, Bousser MG, Fahrni G, et al. Vertebral artery dissection: presenting
findings and predictors of outcome. Stroke 2006;37:2499–503.

Nishimura M, et al. BMJ Case Rep 2015. doi:10.1136/bcr-2014-207332

Unusual association of diseases/symptoms
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