Case reportRegression of Benedikt's syndrome after single-stage removal of mesencephalic cavernoma and temporal meningioma: A case report
Introduction
We herein report a unique association of midbrain cavernoma and Benedikt's syndrome (BS) that was reversed after microsurgical removal of the lesion. BS was originally described by Moritz Benedikt in 1889, characterized by the presence of an oculomotor nerve palsy and cerebellar ataxia including tremor and ascribed clinical constellation of signs to a lesion of the cerebral peduncle [1]. Subsequently, Charcot [2] and de la Tourette and Charcot [2] reported other cases, and in the latter case, involvement of the red nucleus, substantia nigra, third nerve root and sparing the cerebral peduncle were reported [3]. In this report, the author questioned the involvement of the corticospinal tract as a key feature of BS [3]. By that time, midbrain ischemic lesions and tuberculomas appeared to be the most frequent causes of BS, which was never reversible when midbrain structures were disrupted.
Our case helps to elucidate the clinicopathologic characteristics of BS, the potential reversibility of which may indicate surgery even in severely disabled patients with noninfiltrating midbrain mass lesions.
Section snippets
Case report
A 49-year-old woman with a 3-year history of generalized seizures presented with recent onset of diplopia, difficulty swallowing and language problems associated with progressive right-sided weakness and a coarse right upper limb tremor. Neurological examination confirmed a complete left third cranial nerve deficit, oropharyngeal dysphagia, severe mixed aphasia, right-sided hemiparesis with no movement against resistance, right-sided hyperreflexia and dysmetria, and a right arm tremor present
Discussion
To our knowledge, an association between BS and midbrain cavernoma has not been reported to date. The mesencephalic tegmentum is a rarely documented site of expanding mass lesions. Loseke at al. reported a case of red nucleus syndrome featuring only a unilateral involuntary tremor caused by a solitary metastatic lesion from a glandular epithelioma of the prostate [4]. Ono et al. reported a case of a 26-year-old man who presented with an unusual multilobulated cyst in the cerebellopontine angle
Conclusion
BS is a highly localized syndrome rarely caused by expanding mass lesions. In our case, the deficits were regressive after removal of the etiological agent, a midbrain cavernoma. Even highly disabled patients with BS may be improved by surgery if the cause is a noninfiltrating lesion, as in our case.
Acknowledgment
The authors heartily thank Dr. Mariagrazia Signorelli for the artistic drawing.
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