ABSTRACT:Objectives:Meige syndrome is a movement disorder that includes blepharospasm and oromandibular dystonias. Its etiology may be idiopathic (primary) or it may arise secondary to focal brain injury. Acute respiratory distress as a feature of such dystonias occurs infrequently. A review of the literature on Meige syndrome and the relationship between dystonias and respiratory compromise is presented.Methods:A 60-year-old woman suffered a cerebral anoxic event secondary to manual strangulation. She developed progressive blepharospasm combined with oromandibular and cervical dystonias. Neuroimaging demonstrated bilateral damage localized to the globus pallidus. Years later, she presented to the emergency department in intermittent respiratory distress associated with facial and cervical muscle spasms.Results and conclusions:Increasing frequency and severity of the disorder was noted over years. The acute onset of respiratory involvement required intubation and eventual tracheotomy. A partial therapeutic benefit of tetrabenazine was demonstrated.
ABSTRACT:We report a case of primary malignant schwannoma of the trigeminal nerve, and review the 5 previously reported cases. Clinical presentation, along with radiological and pathological features are discussed. RESUME: Des tumeurs primaires malins du nerf trijumeau Un sixieme cas de neurinome primaire malin du trijumeau est presente avec une revue de la litterature. La presentation clinique et les manifestations radiologiques et pathologiques sont discutees.Can. J. Neurol. Sci. 1986; 13:146-148 Schwannomas of the trigeminal nerve are rare; 120 cases had been described up to 1972.12 Malignant schwannoma is very rare, with only five cases having been reported.3,5 These malignant tumours, which are believed to arise from schwannoma cells, 6,7 have been termed malignant schwannoma, neurogenic sarcoma, malignant neurilemmoma, neurofibrosarcoma.8 As experience with these tumours is limited, their management is a subject of controversy.In this paper, we present a new case of malignant schwannoma of the trigeminal nerve and review the five previously reported cases. CASE REPORTA 49 year old woman was admitted in April 1984 with a one month history of left sided facial and periorbital pain, and a two week history of diplopia. The only abnormality on examination was left lateral rectus paresis. Investigations which included an edrophonium test to rule out myasthenia gravis, and a CT head scan, were negative. It was postulated that the sixth nerve palsy may have resulted from a small hemorrhage into the abducens nerve as a complication of long term anticoagulant therapy which she had been taking for lower limb thrombophlebitis. Anticoagulant therapy was discontinued.At follow-up in August 1984 she reported that the facial pain had increased in severity and there was no improvement in diplopia. There was loss of sensation in the territory of the first and second divisions of the left trigeminal nerve, and absent left corneal reflex, decreased taste in the anterior two-thirds of the left side of the tongue, left masseter weakness, and a persistent left sixth nerve palsy.A CT head scan (Figure 1), performed in the coronal plane, showed destructive bone change involving the superior aspect of the clivus on the left side, and the left side of the dorsum sella. A mass lesion demonstrated in this region extended into the sphenoid sinus on the left side, and had also breeched the posterior aspect of the floor of the sphenoid sinus to extend to the right of the sphenoid septum. There was lateral extension to involve the carotid canal, as well as involvement of the cavernous sinus (Figure 2).At surgery, through a transnasal-transphenoidal approach, part of the tumour, which was in the posterior aspect of the sphenoid sinus, was removed. It was hard, avascular, and whitish. Detailed histologic examination using multiple stains including hematoxylin and eosin. PAS, PTAH. Masson's, Holmes and reticulin revealed a highly cellular spindle-celled tumour (Figure 3). The cells showed bipolar fibrillary components and were arranged i...
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