Intensive Care Med (1999) 25: 532±534
Ó Springer-Verlag 1999

I. Granier
E. Garcia
A. Geissler
M. D. Boespflug
J. Durand-Gasselin

Received: 17 August 1998
Final revision received: 25 November 1998
Accepted: 24 February 1999

)

I. Granier ( ) × E. Garcia × A. Geissler ×
J. Durand-Gasselin
Service de RØanimation, Centre
Hospitalier Intercommunal Toulon ±
La Seyne sur Mer, Font-PrØ,
1208 avenue du colonel Picot, BP 1412,
F-83056 Toulon Cedex, France
Tel. + 33 4 94 61 80 98
Fax + 33 4 94 61 60 54
M. D. Boespflug
Service de Radiologie, Centre Hospitalier
Intercommunal Toulon ± La Seyne sur Mer,
Font-PrØ, 1208 avenue du colonel Picot,
BP 1412, F-83056 Toulon Cedex ± France

B R I E F RE PO RT

Postpartum cerebral angiopathy
associated with the administration of
sumatriptan and dihydroergotamine ±
a case report

Abstract Cerebral angiopathy of
the postpartum period is a rare entity, sometimes promoted by vasoconstrictives drug prescription. Its
clinical presentation includes headaches, seizures and focal neurological deficits, which develop shortly
after a normal pregnancy. The diagnosis is based on clinical findings
and angiography, showing multiple
narrowing of the intracranial cerebral arteries. This neurological feature is reversible and the clinical
outcome is good. We report a case of
benign cerebral angiopathy in a 20year-old woman in the postpartum
period, occurring after administration of sumatriptan and ergot derivates.

Key words Postpartum × Cerebral
angiopathy × Vasospasm × Ergot
derivates × Sumatriptan

Introduction

Case report

Numerous studies have demonstrated an association between cerebrovascular disease and pregnancy. Cerebral
angiopathy has been observed following normal pregnancy or in eclampsia [1] and similar features may also
be encountered in cocaine abuse [2], ergot derivate use
[3, 4], sympathomimetic drug intoxication [5] or after
bromocriptine administration [6]. Its clinical presentation consists of severe headache, epileptic seizures and
focal neurological deficits. Thirteen cases of cerebral
postpartum angiopathy have already been reported [1,
3, 6±8]. We report a new case occurring after sumatriptan and ergot alkaloid administration.

A 20-year-old woman, gravida 1, para 1 was admitted to the intensive care unit for a generalized epileptic seizure 2 days after spontaneous delivery. The pregnancy had been uneventful and she
never had signs of preeclampsia. During delivery a peridural anesthesia had been performed without evidence of dural puncture.
Twenty-four hours later, she experienced a severe frontal and occipitonuchal headache, worsened by the upright position. She was
given a 6-mg subcutaneous injection of sumatriptan and the headaches subsided, but they reappeared 1 day later. She received three
tablets of ergonovine, but the symptoms were unchanged. On
day 2, she suffered from a generalized tonico-clonic seizure with
onset in the left arm.
On admission we found a left hemiplegia and Babinski's sign,
without headache. Her blood pressure was 130/80 mmHg and she
was afebrile. Shortly thereafter, she experienced multiple generalized epileptic seizures and she was successively given clonazepam,
phenobarbital and phenytoin. Despite this therapy, a generalized
status epilepticus occurred, requiring continuous infusion of pento-

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Fig. 1 Left, non-contrasted CT
scan shows bilateral hypodensity of both internal capsules and
lenticular nuclei. Right, T2weight MRI shows spontaneous
bilateral hypersignal of capsulo-lenticular area, prominent
on the right side

plane images showed a bilateral area of higher signal in the thalamus (Fig. 1; right). Cerebral angiography, performed 24 h after admission, showed multiple narrowings in the vertebrobasilar and
middle cerebral arteries, predominantly on the right side (Fig. 2).
The venous phase was normal and the diagnosis of postpartum angiopathy was suggested. On day 6, CT scan revealed persistent thalamic hypodensity and disappearance of the cerebral oedema. On
day 10, the patient was awake and the neurological examination
was normal, without headache or epileptic seizure recurring under
oral phenobarbital treatment. Angio-MRI performed 48 h later remained normal. The patient was discharged after 15 days of hospitalization and 3 months later, she remained symptom-free and a repeated angio-MRI was normal.

Discussion

Fig. 2 Angiogram shows segmental narrowing at the level of the
left internal carotid (large arrow) and diffuse vasospasm of right
middle and anterior cerebral arteries (small arrows)
barbital and mechanical ventilation. Laboratory results, including
blood count, coagulation parameters, liver and renal tests, uricemia, urinalysis and antinuclear antibodies were normal. Ophthalmoscopy was also normal. A brain computed tomographic (CT)
scan revealed a moderate cerebral oedema and bilateral hypodensity of both internal capsules and lenticular nuclei, more prominent
on the right side (Fig. 1, left). The cerebrospinal fluid was clear and
showed 3 cells/ml, proteins were 3 g/l, glycorachia: 20 mmol/l, supernatrant was not xantochromic, excluding a subarachnoid haemorrhage. Cultures were sterile. A brain magnetic resonance
imaging (MRI) scan revealed no abnormality in venous sinuses
evocative of cerebral venous thrombosis and T2-weighted axial

Our patient showed a typical feature of benign cerebral
angiopathy of the postpartum period [6]. Its clinical presentation, including headaches, seizures and focal neurological deficits is similar to hypertensive encephalopathy, but she never had hypertension, proteinuria or laboratory criteria for toxaemia during pregnancy, or in
the postpartum period. This presentation may also suggest a subarachnoid haemorrhage, but CT scan and
above normal CSF tests excluded this diagnosis. CT
scan is frequently abnormal in postpartum cerebral angiopathy, showing hypodense areas, revealing cerebral
infarction. Cerebral angiography remains the standard
means of diagnosis, showing widespread segmental and
multifocal vasoconstriction involving the cerebral arteries [3, 6, 8]. Non-invasive methods such as transcranial
Doppler ultrasound shows an increase in the mean velocity of flow, a feature of cerebral vasospasm [1].
The treatment of this case of angiopathy was not specific. Indeed, besides discontinuing the drugs called in

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question, the patient was administered only anti-epileptic drugs (gardenal and thiorenthal). The cerebral oedema was combatted with sedation and by keeping her
average blood pressure high. Nimodipine treatment
might have been considered in this patient. Indeed this
calcium-antagonist, a selective vasodilatator, acts on
cerebral arteries. This justifies its systematic use in order to prevent cerebral vasospasm on the occurrence of
subarachnoid haemorrhages during cerebral aneurysmal rupture.
The mechanism of this angiopathy has not really
been explained. Several drugs have been implicated as
possible causes, especially ergot derivates, due to their
sympathomimetic properties, especially bromocriptine,
a semi-synthetic ergot alkaloid derivate, which is a potent dopaminergic agonist [7]. About ten cases of postpartum cerebral angiopathy reported have involved the
administration of bromocriptine [6, 7, 9]. Then, ergonovine, commonly used during delivery has been implicated in arterial hypertension, stroke, cerebrovascular accident and myocardial infarction [7]. Severe hypertension, as in toxaemia, has also been suggested to explain
the mechanism of vasoconstriction in postpartum angiopathy. Acute hypertension following ergot derivate administration may be responsible for cerebral vasospasm
and subsequent loss of autoregulatory control leading
to ischemia, infarction and oedema [6].
In our patient, sumatriptan and ergonovine had
been administered 24 and 48 h, respectively, after delivery. She had not received bromocriptine therapy.
She had a history of vascular headaches of a similar
nature, but she had never had this treatment before.
The mechanisms of ergot derivates inducing vasoconstriction remain controversial: ergonovine may be an
alpha-adrenorecepteur agonist or may act directly on
the smooth muscle, mediated by serotonergic receptors, or it may enhance the response to biogenic

amines such as catecholamines, histamine or serotonin. Sumatriptan, a serotonin type-1d receptor agonist
is recommended for treating migraine attacks. Usually,
sumatriptan provides relief of the headache in view of
its cerebral vasoconstrictive properties reversing middle cerebral artery dilatation. A recent study reported
that sumatriptan may be used in postpartum to cure
postdural puncture headache, immediately and effectively [10]. The adverse effects reported were chest
pain, coronary spasm and transient arterial hypertension.
This postpartum cerebral angiopathy occurred after
the use of ergonovine and sumatriptan. Only one case
has been reported after bromocriptine therapy and additional use of another sympathomimetic agent [7]. The
authors suggested this association may exacerbate the
adverse effects of each drug. In vivo these two drugs
can induce arterial vasospasm. In postpartum cerebral
angiopathy, they could trigger reinforcement of cerebral
vascular tone. Such mechanisms suggest that vasoconstrictive substances, especially for headaches, should be
given with caution to patients with migraine, Raynaud's
phenomena or other conditions, like pregnancy, which
induce changes responsible for an increase in cerebral
reactivity. These predisposing factors and the administration of vasoconstrictive substances could induce cerebral angiopathy. Indeed, it seems that postpartum patients are truly more vulnerable to the least potent sympathomimetic drugs. Additional use of dihydroergotamine and other drugs with similar actions may also increase the risk of cerebral vasospasm. In the case presented, there is no definite imputability of sumatriptan,
the patient having received ergonovine, also a vasoconstrictive drug. However, we suggest that the association
of ergot derivates with other sympathomimetic medication must be used carefully in patients with headache in
postpartum.

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