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(Stroke. 1995;26:702-704.)
© 1995 American Heart Association, Inc.

Articles

Hemiplegia Vegetativa Alterna (Ipsilateral Horner's Syndrome and Contralateral Hemihyperhidrosis) Following Proximal Posterior Cerebral Artery Occlusion

Claudio Bassetti, MD Ivan N. Staikov, MD

From the Department of Neurology, University Hospital, Inselspital, Bern, Switzerland.

Correspondence to Claudio Bassetti, MD, Department of Neurology, University Hospital, Inselspital, 3010 Bern, Switzerland.

	Abstract

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Background Posterior cerebral artery (PCA) occlusive disease usually produces homonymous visual field defects, hemisensory loss, and neuropsychological deficits. Conversely, the combination of hemiparesis, Horner's syndrome, and contralateral hemihyperhidrosis has never been reported before.

Case Description A patient with infarction in the superficial and deep territories of the right PCA presented with a unique clinical picture, which included contralateral hemiparesis, hemihyperhidrosis, and ipsilateral Horner's syndrome. Magnetic resonance imaging disclosed infarcts in the right anterolateral midbrain, ventroposterolateral thalamic-subthalamic area, and temporo-occipital lobes.

Conclusions The alternating vegetative syndrome (hemiplegia vegetativa alterna) observed in this patient supports the hypothesis of the existence of an uncrossed excitatory and a crossed inhibitory hypothalamospinal sympathetic pathway.

Key Words: cerebral arteries • Horner's syndrome • sympathetic nervous system

	Introduction

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Infarction in the territory of the posterior cerebral artery (PCA) usually presents with homonymous visual field defects, hemisensory loss, abnormal visual perception, and neuropsychological deficits.^{1 2 3} Horner's syndrome is frequent in laterotegmental brain stem or medullary strokes,⁴ as well as in hemispheric strokes due to carotid artery dissection, but has only rarely been reported following PCA infarcts.⁵ Similarly, hemihyperhidrosis has been reported in middle cerebral artery strokes^{6 7 8} and brain stem strokes^{9 10} but not in the course of PCA stroke. We therefore find it interesting to report the observation of a patient with magnetic resonance imaging (MRI)–proven infarction in the superficial and deep territories of the right PCA in whom right-sided Horner's syndrome was associated with a left-sided hemihyperhidrosis. Based on the clinicoradiological correlation in this patient and review of the literature, an explanation for the rare syndrome of alternating sympathetic deficits following proximal PCA occlusion is proposed.

	Case Report

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An obese 36-year-old right-handed man with a history of cigarette smoking first noted reading difficulties. Four days later, he experienced paresthesias and sensory loss of his left hand and of the left side of his lips and tongue. On admission, there was severe left hemihyperesthesia for all modalities of sensation involving the entire hemibody, mild left hemiparesis affecting the face, arm, and leg (with Babinski's sign), and mild left brachiocrural ataxia. Furthermore, left homonymous hemianopsia, left motor and sensory neglect, left hyperhidrosis, right Horner's syndrome (Fig 1), and visuospatial apraxia were noted. Excessive sweating was particularly marked on the face, thorax, and hand. Pupillary reflexes and accommodation were normal. MRI revealed a right posterior cerebral artery territory infarction with ischemic areas in the occipital lobe, inferomedial temporal lobe, and anterolateral midbrain (crus cerebri, Fig 2, top) as well as the ventroposterolateral thalamo-subthalamic area (Fig 2, bottom). Cerebral angiography showed a posterior cerebral artery occlusion at the P2A segment (Fig 3). Results of routine blood tests, vasculitis screening, electrocardiogram, transesophageal echocardiography, and Doppler ultrasound examination were unremarkable; the origin of the stroke was therefore undetermined. With anticoagulant therapy, Horner's syndrome, hemihyperhidrosis, and motor paresis fully disappeared within 1 week. One month after stroke onset, the patient still presented with visuoconstructive apraxia, left superior quadrantanopsia, mild left hemihyperesthesia, and hyperreflexia.

View larger version (102K): [in this window] [in a new window]   Figure 1. Photograph showing 36-year-old man with ptosis and miosis on the right side (Horner's syndrome) following right-sided posterior cerebral artery infarction.

View larger version (96K): [in this window] [in a new window]   Figure 2. T2-weighted magnetic resonance imaging of the brain showing ischemic areas (top) in the occipital lobe, inferomedial temporal lobe, and anteromedial-lateral midbrain (arrow) as well as in the ventroposterolateral thalamic-subthalamic area (bottom).

View larger version (113K): [in this window] [in a new window]   Figure 3. Vertebrobasilar angiography showing occlusion of the right posterior cerebral artery at the P2A segment (arrow).

	Discussion

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In this young patient, the association of contralateral hemiparesis, hemisensory loss, hemianopsia, neglect, and hemihyperhidrosis with ipsilateral Horner's syndrome first suggested MCA stroke following extracranial carotid dissection. Unexpectedly, brain MRI showed a proximal PCA stroke, which stimulated the search for alternative explanations of this clinical syndrome.

Hemiparesis in PCA stroke has been recently reviewed by Chambers et al⁵ and Hommel et al.¹¹ It results from infarction of the corticobulbar and corticospinal pathways in the anterolateral midbrain, as shown also in our patient. The association of occipital infarction with midbrain and posterolateral thalamic infarctions suggests a simultaneous occlusion of peduncular perforating arteries and thalamogeniculate arteries at their origin at the P2A level of the PCA, as demonstrated angiographically in our patient as well as in the case of Hommel et al.¹²

Central Horner's syndrome in patients with stroke is most commonly observed ipsilaterally to laterotegmental medullary and pontine infarctions.⁴ It is usually associated with ipsilateral hypohidrosis of the face¹³ or the entire hemibody¹⁴ and is explained by the interruption of an excitatory hypothalamospinal sympathetic pathway. The latter is thought to begin in the hypothalamic oculosympathetic center (of Karplus and Kreidl) localized rostrodorsally to the red nucleus and ventrally to the ventroposterolateral thalamic nucleus.^{15 16} Sympathetic fibers then descend, mainly uncrossed, through the dorsolateral mesencephalon and pons to the dorsolateral medulla (pupillodilatatory center of Babinski-Nageotte) and spinal cord (ciliospinal center of Budge).^{6 15}

Central Horner's syndrome due to supratentorial strokes is conversely rare and usually associated, as in our patient, with ipsilateral thalamic-hypothalamic lesions.^{7 17 18 19 20} Few cases have been reported following ipsilateral as well as contralateral hemispheric lesions,^{6 8 15} suggesting the existence of crossed and uncrossed corticohypothalamic fibers modulating oculosympathetic function.

Contralateral hemihyperhidrosis involving the face and upper extremities (as in the present case) or the whole hemibody has been reported following hemispheric,^{8 21} thalamic,^{19 20} brain stem,⁹ or spinal cord²² lesions. Its occurrence has been explained by the existence of one or more²³ sympathetic cortico-hypothalamo-spinal pathways crossing at the spinal level and inhibiting contralateral sweating.^{8 21} Very rarely, as in our patient, contralateral (segmental) hemihyperhidrosis can be associated with ipsilateral Horner's syndrome (the so-called hemiplegia vegetativa alterna). This syndrome has been reported following thalamic^{19 20} and dorsolateral pontomedullary strokes (syndrome of Babinski and Nageotte²⁴ ), suggesting a close proximity of the above-mentioned antagonistic sympathetic pathways in these areas. In our patient, contralateral hemihyperhidrosis could be alternatively attributed to a disruption of the pathway, inhibiting sweating at the temporo-occipital or anterolateral midbrain level. The latter hypothesis would be supported by the correlation between excessive sweating and hemiparesis, found in our patient as well as in the literature,^{8 21 25} and by reports of isolated hyperhidrosis after anterior brain stem strokes.^{10 26}

In conclusion, the analysis of an unusual clinical presentation of proximal PCA occlusion supports the hypothesis of the existence of at least two antagonistic sympathetic pathways, descending ipsilaterally through the brain stem to project to the ipsilateral and contralateral spinal cord. Further studies are needed to define the exact anatomy and physiology of central sympathetic control.

	Acknowledgments

 
We thank Professor C.W. Hess for helpful comments, Professor G. Schroth for performing the neuroradiological studies, and E. Wilder-Smith, MD, for correction of the English text.

Received November 2, 1994; revision received January 9, 1995; accepted January 9, 1995.

	References

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