Herbal energy drinks, phenylpropanoid compounds, and cerebral vasculopathy.
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A 41-year-old woman presented with tinnitus in the left ear and headache, followed by diplopia and pain in the left cheek. Angiography showed a left high-flow direct carotid-cavernous fistula (CCF), causing steal of the blood flow from the internal carotid artery into the cavernous sinus. A few days later, she rapidly developed right hemiparesis, dysarthria, and ocular conjugate deviation to the right, and became somnolent. Angiography at that time revealed occlusion of the superior petrosal sinus, causing engorgement of the veins in the surrounding brainstem. The CCF was completely embolized with interlocking detachable coils. Her consciousness disturbance and ophthalmoparesis dramatically improved within a few days, and the right hemiparesis and dysarthria gradually resolved. Magnetic resonance (MR) imaging after the treatment showed small pontine hemorrhage and perifocal edema but no ischemic lesions in the cerebral hemisphere. Re-evaluation of the MR imaging with gadolinium taken on admission demonstrated engorged veins in the brainstem parenchyma, which corresponded to the hemorrhagic lesion in the brainstem. Brainstem congestion caused by direct CCF is very rare, but it can be life-threatening. Good outcome can be expected if the CCF is completely occluded before congestive hemorrhage occurs.
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INTRODUCTION: The mesencephalic alternating syndromes or syndromes of Weber, Benedikt and Claude are uncommon in clinical practice. They are caused by a lesion in the mesencephalus which affects the third cranial nerve bundle, together with the corticospinal pathway, subthalamic nucleus and the dentato-rubric path. CLINICAL CASE: We present the case of a normotensive patient who, as a consequence of a hematoma in the mesencephalic tegmentum, had the association of these three syndromes consecutively. The clinical course was favorable, so that after three months of follow-up only paralysis of the third cranial nerve bundle persisted. DISCUSSION: In the syndromes of Weber, Benedikt and Claude there is the association of ophthalmoplegia with hemiplegia, or a cerebellar hemisyndrome or contralateral abnormal movements compatible with hemiballismus, respectively. Amongst the commonest causes are expansive processes, tumors and arteriovenous malformations. More rarely they are due to cerebrovascular accidents, which are usually ischemic and occasionally hemorrhagic in aetiology. CONCLUSIONS: Spontaneous mesencephalic hematomas are infrequent. They make up approximately 1% of all intracranial hematomas. The commonest site is the tegmentum followed by the peduncle and tectum. They have better prognosis than other hematomas of the brainstem.
This report concerns a 88-year-old diabetic and hypertensive woman with pontine hemorrhage who presented with Foville syndrome and contralateral hyperhidrosis. She was admitted to our hospital for sudden onset of headaches and disturbed consciousness. Neurologic examination revealed bilateral miosis, Foville syndrome and superficial hemianesthesia on the right side of the face and body. No associated Horner syndrome and other autonomic dysfunction were observed. Laboratory data were normal except for diabetic findings. Brain CT and MRI revealed a hematoma in the left side at the lower pons. One month after the onset, hemihyperhidrosis on the face, arm and upper trunk contralateral side of the lesion appeared abruptly, and gradually disappeared a week later. Sweating on the ipsilateral side was normal and no new lesion was seen on the brain CT then. Only a few cases of contralateral hyperhidrosis due to pontine lesion have been reported. We suggest that the contralateral inhibitory sweating pathway was disrupted though the ipsilateral excitatory one was intact. Contralateral hyperhidrosis attributed to imbalance of the perspiratory control can be observed in the subacute or late phase after pontine hemorrhage.
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We report on 2 patients who uncommonly developed isolated limb myorhythmia in association with inferior olive hypertrophy (IOH) after an acute stroke in the brain stem. A slow tremor presented in the proximal upper limbs predominantly when at rest. It was aggravated by outstretched arms and by active hand movements. The surface electromyogram (EMG) recorded simultaneous activities over the agonist and antagonist muscles with a rate of 3.5 Hz and 2.5 Hz in 2 patients respectively. In the first patient, bilateral limb myorhythmia presented 12 months after the brain stem stroke, and both inferior olives were hypertrophic. In the second patient, unilateral limb myorhythmia developed in the left hand 7 months after right pontine hemorrhage, and only the right inferior olive was hypertrophic. These findings indicate that limb myorhythmia commencing after brain stem insult is anatomically and temporally related to hypertrophy of the contralateral inferior olive. Based on our 2 patients and previously reported cases, we propose that a possible causal relationship exists between limb myorhythmia and contralateral IOH, although its pathophysiological mechanisms remain to be established. We suggest that, similar to palatal myoclonus, isolated limb myorhythmia is within the clinical spectrum of IOH.
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