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Isolated Vagus Nerve Paralysis Caused by Varicella Zoster Virus Reactivation
Otolaryngology–Head and Neck Surgery (2005) 133, 460-461
Isolated Vagus Nerve Paralysis Caused by Varicella Zoster Virus Reactivation Hideki Nakagawa, MD, Mami Satoh, BA, Toshiyuki Kusuyama, MD, Hiroyuki Fukuda, MD, and Kaoru Ogawa, MD, Shinjuku-Ku, Tokyo
V
agus nerve paralyses by varicella zoster virus (VZV) reactivation have been rare,1,2 and most previously reported cases are associated with other cranial nerve paralysis. We report a case of laryngeal zoster with isolated vagus nerve palsy that was diagnosed by direct immunofluorescence staining for VZV antigens on the samples from laryngeal mucosal lesions.
by fluorescein isothiocyanate (FITC)-labeled mouse monoclonal antibody specific for VZV glycoprotein (Denka Seiken Co. Ltd., Tokyo, Japan) was performed on the sam-
CASE REPORT A 55-year old woman presented with sudden onset dysphagia and hoarseness that had lasted for 3 days. She did not feel sore throat, otalgia, and dysgeusia. The patient’s history revealed hypertension that had been under regular medical control for several years. Her voice was hoarse and asthenic. She was almost unable to swallow saliva. Physical examination showed a left-sided paresis of the soft palate with normal gag reflex. There were no eruptions or vesicles on the skin or the mucosa of the head and neck, including ear, oral cavity, and pharynx. Laryngofiberscopy revealed complete left vocal fold palsy fixed at the intermediate position, saliva pooling in both of the pyriformis sinuses, and small white spots dotted about right of center of the laryngeal surface of the epiglottis (Fig 1). Laboratory studies were almost normal except for a C-reactive protein concentration of 2.0 mg/dL. Neck and chest CT scan and brain MRI and magnetic resonance angiography yielded normal results. Videofluorogram indicated decreased soft palate movement and almost no relaxation of cricopharyngeal muscle. An enzyme immunoassay for serum VZV antibodies revealed slightly increased VZV IgG (1:4.7), whereas VZV IgM was not detected. Direct immunofluorescence staining From the Department of Otolaryngology, Seibo International Catholic Hospital (Dr Nakagawa), Tokyo Voice Center, International University of Health and Welfare (Drs Satoh, Kusuyama, Fukuda), and Department of Otolaryngology, Keio University School of Medicine (Dr Ogawa).
Figure 1 Laryngofiberscopy demonstrating small white spots on the laryngeal surface of the epiglottis.
ples obtained from the white spots on the epiglottis by abrasion with a cotton swab under indirect laryngoscopy, and it revealed VZV antigen (Fig 2). The patient was admitted on February 13, 2004 and was treated by intravenous administration of acyclovir, 5 mg/kg every 8 hours for 7 days. Direct and indirect rehabilitation programs for her dysphagia were started. The white spots of Reprint requests: Hideki Nakagawa, MD, Department of Otolaryngology, Seibo International Catholic Hospital, 2-5-1 Nakaochiai, Shinjuku-ku, Tokyo 161-8521, Japan. E-mail address: [email protected].
0194-5998/$30.00 © 2005 American Academy of Otolaryngology–Head and Neck Surgery Foundation, Inc. All rights reserved. doi:10.1016/j.otohns.2005.01.019
Nakagawa et al
Isolated Vagus Nerve Paralysis Caused by Varicella . . .
Figure 2 The direct immunofluorescence staining for VZV antigens by FITC-labeled monoclonal antibody. VZV-infected cells generate fluorescent green, whereas VZV-noninfected cells are counterstained red by Evans blue.
the epiglottis were increased in the consecutive 4 days after admission, spreading throughout the laryngeal surface of the epiglottis and to the left arytenoid and aryepiglottic fold, then fading away and finally disappearing on February 22. Subsequent concentration of VZV IgG were 1:104 on February 20 and 1:128 on March 8, respectively. Dysphagia and vocal fold paralysis improved gradually, and the patient was discharged on March 9. Four months after the onset of illness, the patient did not complain of hoarseness and dysphagia. Laryngofiberscopy revealed complete left vocal fold movement, and videofluorogram revealed almost normal palatal movement, laryngeal elevation, and passage of bolus through the hypopharynx.
DISCUSSION Serological tests of VZV antibodies can offer laboratory evidence of varicella zoster infection. Although they have been used for the diagnostic tools for laryngeal zoster,1 it is thought that they are not appropriate for early diagnosis, because they would not be positive in the early stage of the infection and often require phase examination for confirmed diagnosis. Early initiation of the antiviral treatment is the key to succeeding in control over herpes zoster, because antiviral therapy with intravenous acyclovir, which acts by incorporation and interruption of viral DNA synthesis, is effective only when the virus is multiplying.3 So that early definitive diagnosis that can be reasonable ground for administration of antiviral agents is undoubtedly important. The detection method of VZV antigen by FITC-labeled monoclonal antibody realized rapid detection of VZV with high reliability. Its turnaround time is only 1 to 2 hours, and both the specificity and sensitivity are ⬎90%.4 This is the
461
first report of laryngeal zoster diagnosed with a monoclonal antibody– based direct immunofluorescence technique. In this case, rapid definite diagnosis by the detection of VZV antigen made possible early initiation of the antiviral treatment and was thought to result in favorable prognosis. The day of the visit, the small white spots were found at the right, the nonparalyzed side, of the epiglottis. The distribution of the lesions might make the diagnosis for laryngeal zoster difficult, because reactivation of latent VZV infection usually presents unilateral distribution of eruptions, according to the innervation, and it has been believed that most areas of laryngeal mucosa, including epiglottis, had unilateral innervation. In recent years, Sanders and Mu5 have investigated the innervation of the branches of internal superior laryngeal nerve by the whole-mount nerve staining method. They concluded that the branches broke up numerous twigs that form a dense network with those from the opposite side, which extends over the laryngeal surface of the epiglottis. The area of the vesicles that were seen in this case has confirmed their findings. Therefore, it should be recognized that the diagnosis of laryngeal zoster can be done irrespective of the laterality and the distribution of the mucosal lesion in the epiglottis, and detection of VZV antigen from the mucosal lesion might be of help in diagnosing such cases. Dysphagia caused by vagus nerve paralysis could be severer than that by simple recurrent laryngeal nerve palsy, because the former includes paresis of pharynx and sensory disturbance of supraglottic mucosa, in addition to vocal fold paralysis and dysfunction of cricopharyngeal muscle. Although laryngeal zoster often causes vagus nerve paralysis, which can cause life-threatening aspiration pneumonia, definite diagnosis is not always easy, so that even biopsies are considered. Rapid detection of VZV antigen from laryngeal mucosa by monoclonal antibody– based direct immunofluorescence technique is extremely useful for early diagnosis, and aggressive antiviral therapy should be initiated as quickly as possible.
REFERENCES 1. Nishizaki K, Onoda K, Akagi H, et al. Laryngeal zoster with unilateral laryngeal paralysis. ORL J Otorhinolaryngol Relat Spec 1997; 59:235–7. 2. Pahor AL. Herpes zoster of the larynx— how common? J Laryngol Otol 1979;93:93– 8. 3. Dickins JRE, Smith JT, Graham SS, et al. Herpes zoster oticus: treatment with intravenous acyclovir. Laryngoscope 1988;98:776 –9. 4. Schirm J, Meulenberg J, Pastoor G, et al. Rapid detection of varicellazoster virus in clinical specimens using monoclonal antibodies on shell and smears. J Med Virol 1989;28:1– 6. 5. Sanders I, Mu L. Anatomy of the human internal superior laryngeal nerve. Anat Rec 1998;252:646 –56.
Isolated Vagus Nerve Paralysis Caused by Varicella Zoster Virus Reactivation Hideki Nakagawa, MD, Mami Satoh, BA, Toshiyuki Kusuyama, MD, Hiroyuki Fukuda, MD, and Kaoru Ogawa, MD, Shinjuku-Ku, Tokyo
V
agus nerve paralyses by varicella zoster virus (VZV) reactivation have been rare,1,2 and most previously reported cases are associated with other cranial nerve paralysis. We report a case of laryngeal zoster with isolated vagus nerve palsy that was diagnosed by direct immunofluorescence staining for VZV antigens on the samples from laryngeal mucosal lesions.
by fluorescein isothiocyanate (FITC)-labeled mouse monoclonal antibody specific for VZV glycoprotein (Denka Seiken Co. Ltd., Tokyo, Japan) was performed on the sam-
CASE REPORT A 55-year old woman presented with sudden onset dysphagia and hoarseness that had lasted for 3 days. She did not feel sore throat, otalgia, and dysgeusia. The patient’s history revealed hypertension that had been under regular medical control for several years. Her voice was hoarse and asthenic. She was almost unable to swallow saliva. Physical examination showed a left-sided paresis of the soft palate with normal gag reflex. There were no eruptions or vesicles on the skin or the mucosa of the head and neck, including ear, oral cavity, and pharynx. Laryngofiberscopy revealed complete left vocal fold palsy fixed at the intermediate position, saliva pooling in both of the pyriformis sinuses, and small white spots dotted about right of center of the laryngeal surface of the epiglottis (Fig 1). Laboratory studies were almost normal except for a C-reactive protein concentration of 2.0 mg/dL. Neck and chest CT scan and brain MRI and magnetic resonance angiography yielded normal results. Videofluorogram indicated decreased soft palate movement and almost no relaxation of cricopharyngeal muscle. An enzyme immunoassay for serum VZV antibodies revealed slightly increased VZV IgG (1:4.7), whereas VZV IgM was not detected. Direct immunofluorescence staining From the Department of Otolaryngology, Seibo International Catholic Hospital (Dr Nakagawa), Tokyo Voice Center, International University of Health and Welfare (Drs Satoh, Kusuyama, Fukuda), and Department of Otolaryngology, Keio University School of Medicine (Dr Ogawa).
Figure 1 Laryngofiberscopy demonstrating small white spots on the laryngeal surface of the epiglottis.
ples obtained from the white spots on the epiglottis by abrasion with a cotton swab under indirect laryngoscopy, and it revealed VZV antigen (Fig 2). The patient was admitted on February 13, 2004 and was treated by intravenous administration of acyclovir, 5 mg/kg every 8 hours for 7 days. Direct and indirect rehabilitation programs for her dysphagia were started. The white spots of Reprint requests: Hideki Nakagawa, MD, Department of Otolaryngology, Seibo International Catholic Hospital, 2-5-1 Nakaochiai, Shinjuku-ku, Tokyo 161-8521, Japan. E-mail address: [email protected].
0194-5998/$30.00 © 2005 American Academy of Otolaryngology–Head and Neck Surgery Foundation, Inc. All rights reserved. doi:10.1016/j.otohns.2005.01.019
Nakagawa et al
Isolated Vagus Nerve Paralysis Caused by Varicella . . .
Figure 2 The direct immunofluorescence staining for VZV antigens by FITC-labeled monoclonal antibody. VZV-infected cells generate fluorescent green, whereas VZV-noninfected cells are counterstained red by Evans blue.
the epiglottis were increased in the consecutive 4 days after admission, spreading throughout the laryngeal surface of the epiglottis and to the left arytenoid and aryepiglottic fold, then fading away and finally disappearing on February 22. Subsequent concentration of VZV IgG were 1:104 on February 20 and 1:128 on March 8, respectively. Dysphagia and vocal fold paralysis improved gradually, and the patient was discharged on March 9. Four months after the onset of illness, the patient did not complain of hoarseness and dysphagia. Laryngofiberscopy revealed complete left vocal fold movement, and videofluorogram revealed almost normal palatal movement, laryngeal elevation, and passage of bolus through the hypopharynx.
DISCUSSION Serological tests of VZV antibodies can offer laboratory evidence of varicella zoster infection. Although they have been used for the diagnostic tools for laryngeal zoster,1 it is thought that they are not appropriate for early diagnosis, because they would not be positive in the early stage of the infection and often require phase examination for confirmed diagnosis. Early initiation of the antiviral treatment is the key to succeeding in control over herpes zoster, because antiviral therapy with intravenous acyclovir, which acts by incorporation and interruption of viral DNA synthesis, is effective only when the virus is multiplying.3 So that early definitive diagnosis that can be reasonable ground for administration of antiviral agents is undoubtedly important. The detection method of VZV antigen by FITC-labeled monoclonal antibody realized rapid detection of VZV with high reliability. Its turnaround time is only 1 to 2 hours, and both the specificity and sensitivity are ⬎90%.4 This is the
461
first report of laryngeal zoster diagnosed with a monoclonal antibody– based direct immunofluorescence technique. In this case, rapid definite diagnosis by the detection of VZV antigen made possible early initiation of the antiviral treatment and was thought to result in favorable prognosis. The day of the visit, the small white spots were found at the right, the nonparalyzed side, of the epiglottis. The distribution of the lesions might make the diagnosis for laryngeal zoster difficult, because reactivation of latent VZV infection usually presents unilateral distribution of eruptions, according to the innervation, and it has been believed that most areas of laryngeal mucosa, including epiglottis, had unilateral innervation. In recent years, Sanders and Mu5 have investigated the innervation of the branches of internal superior laryngeal nerve by the whole-mount nerve staining method. They concluded that the branches broke up numerous twigs that form a dense network with those from the opposite side, which extends over the laryngeal surface of the epiglottis. The area of the vesicles that were seen in this case has confirmed their findings. Therefore, it should be recognized that the diagnosis of laryngeal zoster can be done irrespective of the laterality and the distribution of the mucosal lesion in the epiglottis, and detection of VZV antigen from the mucosal lesion might be of help in diagnosing such cases. Dysphagia caused by vagus nerve paralysis could be severer than that by simple recurrent laryngeal nerve palsy, because the former includes paresis of pharynx and sensory disturbance of supraglottic mucosa, in addition to vocal fold paralysis and dysfunction of cricopharyngeal muscle. Although laryngeal zoster often causes vagus nerve paralysis, which can cause life-threatening aspiration pneumonia, definite diagnosis is not always easy, so that even biopsies are considered. Rapid detection of VZV antigen from laryngeal mucosa by monoclonal antibody– based direct immunofluorescence technique is extremely useful for early diagnosis, and aggressive antiviral therapy should be initiated as quickly as possible.
REFERENCES 1. Nishizaki K, Onoda K, Akagi H, et al. Laryngeal zoster with unilateral laryngeal paralysis. ORL J Otorhinolaryngol Relat Spec 1997; 59:235–7. 2. Pahor AL. Herpes zoster of the larynx— how common? J Laryngol Otol 1979;93:93– 8. 3. Dickins JRE, Smith JT, Graham SS, et al. Herpes zoster oticus: treatment with intravenous acyclovir. Laryngoscope 1988;98:776 –9. 4. Schirm J, Meulenberg J, Pastoor G, et al. Rapid detection of varicellazoster virus in clinical specimens using monoclonal antibodies on shell and smears. J Med Virol 1989;28:1– 6. 5. Sanders I, Mu L. Anatomy of the human internal superior laryngeal nerve. Anat Rec 1998;252:646 –56.