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Bilateral cheiro-oral syndrome

W H Chen1, M Y Lan, Y Y Chang

  • 1Department of Neurology, Kaohsiung Medical College Hospital, Taiwan.

Clinical Neurology and Neurosurgery
|March 10, 1998
PubMed
Summary

Bilateral cheiro-oral syndrome (COS) involves facial and oral sensory issues. Lesions in the pons are the most common cause, even without other brainstem signs.

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Area of Science:

  • Neurology
  • Neuroscience
  • Clinical Neurology

Background:

  • Cheiro-oral syndrome (COS) is a rare neurological condition characterized by sensory disturbances in the hands and perioral region.
  • Bilateral presentations of COS are infrequent, making their etiology and localization challenging to ascertain.
  • Understanding the precise neuroanatomical correlates of bilateral COS is crucial for diagnosis and management.

Purpose of the Study:

  • To report and analyze eight new cases of pure bilateral cheiro-oral syndrome (COS).
  • To investigate the common etiological factors and lesion locations associated with bilateral COS.
  • To determine the most frequent site of brain lesions in bilateral COS, considering existing literature.

Main Methods:

  • Case series reporting eight patients with pure bilateral COS.
  • Detailed neuroimaging (e.g., MRI) and neurophysiological studies were conducted.
  • Etiology and lesion localization were determined for each case.

Main Results:

  • Eight cases of pure bilateral COS were identified and analyzed.
  • Pontine infarct was the most frequent etiology (three cases).
  • Other identified lesions included brainstem hemorrhage, unilateral thalamic infarct, and bilateral subdural hematoma.
  • Two cases had normal neuroimaging and neurophysiological findings.
  • Consolidating with prior reports, the pons remains the most common lesion site for bilateral COS.

Conclusions:

  • The pons is the most frequent location for lesions causing bilateral cheiro-oral syndrome (COS).
  • This holds true even when associated brainstem signs are absent.
  • Further research into the pathogenesis of bilateral COS, particularly in cases with normal imaging, is warranted.

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