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Ramsay Hunt syndrome affecting the vagus nerve with epiglottic ulcers as the first manifestation: a case report.

ABSTRACT: Ramsay Hunt syndrome involving the vagus nerve is very rare. We herein describe a 53-year-old man who developed severe pharyngeal pain after alcohol intoxication. Antibiotic treatment was ineffective. Laryngoscopy showed an ulcer on the right side of the epiglottis. As the condition progressed, the patient developed hoarseness. He then gradually developed multiple herpes lesions on the right side of the head, face, and neck along with right peripheral facial paralysis. Corticosteroid, analgesic, antiviral, and nutritional nerve therapy resulted in disappearance of the herpes lesions, epiglottis ulcer, pharyngeal pain, and right head and face pain. The facial paralysis slightly improved, but the hoarseness did not improve. The patient was discharged with an unsatisfactory outcome, and he attempted treatment with acupuncture. After 6 months, the right facial paralysis and hoarseness disappeared. Our case emphasizes the importance of early diagnosis and treatment of atypical Ramsay Hunt syndrome as well as timely communication, enhancement of trust, and reduction of disputes between doctors and patients.

SUBMITTER: Zhang J

PROVIDER: S-EPMC7509732 | biostudies-literature | 2020 Sep

REPOSITORIES: biostudies-literature

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Ramsay Hunt syndrome affecting the vagus nerve with epiglottic ulcers as the first manifestation: a case report.

Zhang Jie J Wei Xuemei X

The Journal of international medical research 20200901 9

Ramsay Hunt syndrome involving the vagus nerve is very rare. We herein describe a 53-year-old man who developed severe pharyngeal pain after alcohol intoxication. Antibiotic treatment was ineffective. Laryngoscopy showed an ulcer on the right side of the epiglottis. As the condition progressed, the patient developed hoarseness. He then gradually developed multiple herpes lesions on the right side of the head, face, and neck along with right peripheral facial paralysis. Corticosteroid, analgesic, ...[more]

PMID: 32954886

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Project description:Introduction: VNS is an adjunctive neuromodulation therapy for patients with drug-refractory epilepsy. The antiseizure effect of VNS is thought to be related to a diffuse modulation of functional connectivity but remains to be confirmed. Aim: To investigate electroencephalographic (EEG) metrics of functional connectivity in patients with drug-refractory epilepsy treated by vagus nerve stimulation (VNS), between VNS-stimulated "ON" and nonstimulated "OFF" periods and between responder (R) and nonresponder (NR) patients. Methods: Scalp-EEG was performed for 35 patients treated by VNS, using 21 channels and 2 additional electrodes on the neck to detect the VNS stimulation. Patients were defined as VNS responders if a reduction of seizure frequency of ∼50% was documented. We analyzed the synchronization in EEG time series during "ON" and "OFF" periods of stimulation, using average phase lag index (PLI) in signal space and phase-locking value (PLV) between 10 sources. Based on graph theory, we computed brain network models and analyzed minimum spanning tree (MST) for responder and nonresponder patients. Results: Among 35 patients treated by VNS for a median time of 7 years (range 4 months to 22 years), 20 were R and 15 were NR. For responder patients, PLI during ON periods was significantly lower than that during OFF periods in delta (p = 0.009), theta (p = 0.02), and beta (p = 0.04) frequency bands. For nonresponder patients, there were no significant differences between ON and OFF periods. Moreover, variations of seizure frequency with VNS correlated with the PLI OFF/ON ratio in delta (p = 0.02), theta (p = 0.04), and beta (p = 0.03) frequency bands. Our results were confirmed using PLV in theta band (p < 0.05). No significant differences in MST were observed between R and NR patients. Conclusion: The correlation between VNS-induced interictal EEG time-series desynchronization and decrease in seizure frequency suggested that VNS therapeutic impact might be related to changes in interictal functional connectivity. Impact statement Electroencephalography (EEG) desynchronization has been proposed to be a mechanism for antiepileptic effect of vagus nerve stimulation (VNS). We measured interictal EEG time-series synchronization during stimulated (ON) and nonstimulated (OFF) periods in epileptic patients treated by VNS. Phase lag index differences between ON and OFF periods were measured in delta, theta, and beta bands only in responder patients. To our knowledge, our study is the first to statistically correlate interictal cortical desynchronization during ON periods with reduction in seizure frequency. Our result supports the hypothesis that the antiseizure effect of VNS is mediated by cortical desynchronization.

Dataset Information

Ramsay Hunt syndrome affecting the vagus nerve with epiglottic ulcers as the first manifestation: a case report.

Publications

Ramsay Hunt syndrome affecting the vagus nerve with epiglottic ulcers as the first manifestation: a case report.

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