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Spinal cheiro-oral syndrome: a common neurological entity in an unusual site
Affiliations- 1 Department of Neurology, Institute of Psychiatry and Neurology, Warsaw, Poland
- Acibadem Hospital, Department of Pathology, Istanbul/TURKEY, 34718 Istanbul, Türkiye
- department of neurosurgery
- Graduate Institute of Science and Technology Law, National Kaohsiung First University of Science and Technology, Kaohsiung
- Department of Neuroradiology, Chang Gung Memorial Hospital — Kaohsiung Medical Center and College of Medicine, Chang Gung University, Kaohsiung
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Abstract
Cheiro-oral syndrome (COS) is an established neurological entity characterized by a sensory impairment confined to the mouth angle and ipsilateral finger(s)/ hand. The current understanding of localization is a concomitant involvement of the spinothalamic and trigeminothalamic tract between the cortex and pons. The cervical spinal cord has not been mentioned in this situation yet, and this unusual location may heretofore increase the risk of misdiagnosis.
Material and methodsSix patients who presented with unilateral COS due to cervical cord disorder are reported.
ResultsAll patients were women and their age ranged between 42 and 70 years. Their neurological deficits included unilateral paraesthesiae restricted to cheiro-oral distribution, positive radicular sign, and mild change of tendon reflex. Cervical spinal stenosis at middle/lower cervical spine with variable magnitude of cord compression and intrinsic cord damage was found. A diagnostic dilemma obviously arises from the lack of tangible neurological signs or typical pattern of myelopathy, in addition to the previous concept of cerebral involvement. A benign course ensued in all reported patients.
ConclusionsCheiro-oral syndrome can be an early neurological sign for cervical cord disorder; it further suggests that it is a strong neurological but weak localizing sign. A reciprocal influence of multiple factors is considered to generate COS at the cervical cord. Therefore, an absence of brain pathology should lead to a thorough examination of the cervical cord in case of COS.
Abstract
Cheiro-oral syndrome (COS) is an established neurological entity characterized by a sensory impairment confined to the mouth angle and ipsilateral finger(s)/ hand. The current understanding of localization is a concomitant involvement of the spinothalamic and trigeminothalamic tract between the cortex and pons. The cervical spinal cord has not been mentioned in this situation yet, and this unusual location may heretofore increase the risk of misdiagnosis.
Material and methodsSix patients who presented with unilateral COS due to cervical cord disorder are reported.
ResultsAll patients were women and their age ranged between 42 and 70 years. Their neurological deficits included unilateral paraesthesiae restricted to cheiro-oral distribution, positive radicular sign, and mild change of tendon reflex. Cervical spinal stenosis at middle/lower cervical spine with variable magnitude of cord compression and intrinsic cord damage was found. A diagnostic dilemma obviously arises from the lack of tangible neurological signs or typical pattern of myelopathy, in addition to the previous concept of cerebral involvement. A benign course ensued in all reported patients.
ConclusionsCheiro-oral syndrome can be an early neurological sign for cervical cord disorder; it further suggests that it is a strong neurological but weak localizing sign. A reciprocal influence of multiple factors is considered to generate COS at the cervical cord. Therefore, an absence of brain pathology should lead to a thorough examination of the cervical cord in case of COS.
Keywords
cheiro-oral syndrome, radicular sign, spinal cord, myelopathy, sensory, localization
About this articleTitle
Spinal cheiro-oral syndrome: a common neurological entity in an unusual site
Journal
Neurologia i Neurochirurgia Polska
Issue
Pages
583-589
Page views
223
Article views/downloads
1004
DOI
10.1016/S0028-3843(14)60126-2
Bibliographic record
Neurol Neurochir Pol 2011;45(6):583-589.
Keywords
cheiro-oral syndrome
radicular sign
spinal cord
myelopathy
sensory
localization
Authors
Hung-Sheng Lin
Hsin-Ling Yin
Chi-Chui
Chun-Chung Lui
Wei-Hsi Chen
