OBJECTIVE AND IMPORTANCE: We describe an atypical case of transient Foix-Chavany-Marie syndrome, or faciopharyngoglossomasticatory diplegia with automatic voluntary dissociation, occurring after surgical resection of a right insulo-opercular glioma. CLINICAL PRESENTATION: A 26-year-old right-handed man experienced partial seizures that were poorly controlled by antiepileptic drugs during a 2-year period as a result of a right insulo-opercular low-grade glioma, leading to the proposal of surgical resection. In addition, 1 year before the operation, the patient experienced a severe brain injury that resulted in a coma. A computed tomographic scan revealed left opercular contusion. The patient recovered completely within 6 months. INTERVENTION: Intraoperative corticosubcortical electrical functional mapping was performed along the resection, allowing the identification and preservation of the facial and upper limb motor structures. A subtotal removal of the glioma was achieved. The patient had postoperative anarthria, with loss of voluntary muscular functions of the face and tongue, and he had trouble chewing and swallowing. All of these symptoms resolved within 3 months. CONCLUSION: These findings provide insight into the use of surgery to treat a right insulo-opercular tumor. First, surgeons must be particularly cautious in cases with a potential contralateral lesion (e.g., history of head injury), even if such a lesion is not visible on magnetic resonance imaging scans; preoperative metabolic imaging and electrophysiological investigations should be considered before an operative decision is made. Second, surgeons must perform intraoperative functional mapping to identify and to attempt to preserve the corticosubcortical facial motor structures. A procedure performed while the patient is awake should be discussed to detect the structures involved in chewing and swallowing in cases of suspected bilateral lesions. Third, the patient must be informed of this particular risk before surgery is performed.
OBJECTIVE AND IMPORTANCE: We describe an atypical case of transient Foix-Chavany-Marie syndrome, or faciopharyngoglossomasticatory diplegia with automatic voluntary dissociation, occurring after surgical resection of a right insulo-opercular glioma. CLINICAL PRESENTATION: A 26-year-old right-handed man experienced partial seizures that were poorly controlled by antiepileptic drugs during a 2-year period as a result of a right insulo-opercular low-grade glioma, leading to the proposal of surgical resection. In addition, 1 year before the operation, the patient experienced a severe brain injury that resulted in a coma. A computed tomographic scan revealed left opercular contusion. The patient recovered completely within 6 months. INTERVENTION: Intraoperative corticosubcortical electrical functional mapping was performed along the resection, allowing the identification and preservation of the facial and upper limb motor structures. A subtotal removal of the glioma was achieved. The patient had postoperative anarthria, with loss of voluntary muscular functions of the face and tongue, and he had trouble chewing and swallowing. All of these symptoms resolved within 3 months. CONCLUSION: These findings provide insight into the use of surgery to treat a right insulo-opercular tumor. First, surgeons must be particularly cautious in cases with a potential contralateral lesion (e.g., history of head injury), even if such a lesion is not visible on magnetic resonance imaging scans; preoperative metabolic imaging and electrophysiological investigations should be considered before an operative decision is made. Second, surgeons must perform intraoperative functional mapping to identify and to attempt to preserve the corticosubcortical facial motor structures. A procedure performed while the patient is awake should be discussed to detect the structures involved in chewing and swallowing in cases of suspected bilateral lesions. Third, the patient must be informed of this particular risk before surgery is performed.
Authors: Tom Theys; Sofie Van Cauter; Kuan H Kho; Anne-Catherine Vijverman; Ronald R Peeters; Stefan Sunaert; Johannes van Loon Journal: J Neurol Date: 2012-08-15 Impact factor: 4.849