Printer Friendly

Potentially lethal pharyngolaryngeal edema with dyspnea in adult patients with mumps: a series of 5 cases.

Abstract

In this article we describe 5 rare cases of mumps-associated pharyngolaryngeal edema. To the best of our knowledge, this report includes the first case of mumps-associated pharyngolaryngeal edema in a patient who had previously received mumps vaccination, and these cases represent the sixth report of mumps-associated pharyngolaryngeal edema in the English literature. All 5 of our patients with mumps infection were adults and manifested airway stenosis due to pharyngolaryngeal edema. This edema responded favorably to steroid treatment without tracheotomy. We conclude that a pharyngolaryngeal examination is recommended for patients with mumps infection. Steroid treatment is usually effective against pharyngolaryngeal edema; however, in certain cases tracheotomy may be inevitable.

Introduction

Mumps is a viral infection that primarily affects the salivary glands. (1) It can cause various complications such as aseptic meningitis, encephalitis, orchitis, oophoritis, pancreatitis, and acquired sensorineural hearing loss. (1) We encountered 5 rare cases of pharyngolaryngeal edema with dyspnea associated with mumps infection. To our knowledge, this report includes the first case of mumpsassociated pharyngolaryngeal edema in a patient who had previously received mumps vaccination. To date, there are only 5 previous reports of mumps-associated pharyngolaryngeal edema in the English-language literature. (2,6) In this report, we discuss our 5 cases and the characteristics of this potentially lethal complication.

Case reports

Patient 1. A 31-year-old woman presented with dyspnea, dysphasia, and a 4-day history of bilateral parotid swelling. She had no history of mumps infection or vaccination. Physical examination showed bilateral parotid and submandibular swelling, and fiberoptic laryngoscopy showed bilateral swelling of the arytenoid region (figure 1, A). Laboratory examination showed a leukocyte count of 4.1 x [10.sup.3]/[micro]l and a serum C-reactive protein (CRP) level of 0.2 mg/dl. Enzyme-linked immunosorbent assay showed seropositivity for mumps virus immunoglobulin M (IgM) antibody. Intravenous administration of hydrocortisone (300 mg) for 4 days resulted in a rapid reduction of the laryngeal edema.

Patient 2. A 16-year-old boy presented with dyspnea and a 2-day history of bilateral parotid and submandibular swelling. He had no history of mumps infection, but he had received a mumps vaccination. Physical examination showed bilateral parotid and submandibular swelling, and fiberoptic laryngoscopy showed pharyngolaryngeal edema and edema of the epiglottis, vallecula, and the right arytenoid region (figure 1, B).

Contrast-enhanced computed tomography (CT) showed a swelling of the bilateral parotid and submandibular glands. Laboratory examination showed a leukocyte count of 3.5 x [10.sup.3]/[micro]l and a serum CRP level of 1.5 mg/dl. The patient tested positive for mumps IgM antibody. Hydrocortisone (300 mg) was administered intravenously for 4 days, and the pharyngolaryngeal edema was reduced rapidly.

Patient 3. A 31 -year-old woman presented with dyspnea and a 2-day history of bilateral parotid swelling. She had no history of mumps infection or vaccination. Physical examination showed bilateral parotid and submandibular swelling, and fiberoptic laryngoscopy showed marked bilateral supraglottic edema (figure 1, C). CT showed a swollen submandibular gland and arytenoid region on the right side (figure 2), as well as swollen bilateral parotid glands. Laboratory examination showed a leukocyte count of 3.9 x [10.sup.3]/[micro]l and a serum CRP level of 2.6 mg/dl. The patient tested positive for mumps IgM antibody. Hydrocortisone (300 mg) was administered intravenously for 1 day, which resulted in a rapid reduction of the laryngeal edema.

Patient 4. A 40-year-old woman presented with dyspnea, severe pain during swallowing, and a 7-day history of swelling in the left parotid gland. She had no history of mumps infection or vaccination. On admission, physical examination showed a swelling of the left parotid and submandibular glands, and fiberoptic laryngoscopy showed supraglottic swelling (figure 3, A). CT showed a swelling of the left parotid and submandibular glands. Laboratory examination showed a leukocyte count of 6.8 x [10.sup.3]/[micro]l and a serum CRP level of 1.1 mg/dl. The patient tested positive for mumps IgM antibody. Hydrocortisone (300 mg) was administered intravenously for 4 days, which rapidly reduced the laryngeal edema.

Patient 5. A 25-year-old man presented with dyspnea, severe pain during swallowing, and a 1 -day history of bilateral parotid swelling. He had no history of mumps infection or vaccination. Physical examination showed bilateral parotid and submandibular swelling and a marked swelling with tenderness in the left testis. Fiberoptic laryngoscopy showed severe edema of the left arytenoid region (figure 3, B). Laboratory examination showed a leukocyte count of 5.9 x [10.sup.3]/[micro]l and a serum CRP level of 1.0 mg/dl.

A complement fixation reaction test showed a 16-fold increase in this patient's mumps antibody titers. According to the criteria of the Centers for Disease Control and Prevention (i.e., an illness with an acute onset of unilateral or bilateral tenderness, self-limited swelling of the parotid or other salivary glands that lasts for [greater than or equal to] 2 days, not associated with any other apparent cause (7)), the patient was suspected to have a mumps infection. Hydrocortisone (600 mg) was administered intravenously for 3 days, which rapidly reduced the laryngeal edema.

Discussion

The previously reported cases of mumps-associated pharyngolaryngeal edema (2-6) and our cases all involved adult patients, except for 1 pediatric case. (6) Furthermore, they all showed marked unilateral or bilateral submandibular gland swelling, as well as parotid gland swelling. Submandibular gland swelling is considered to occur at a considerably high rate in patients with mumps-associated pharyngolaryngeal edema versus approximately only 10% of patients with common mumps with parotid gland swelling. (8)

Viral replication in the salivary glands, including the ductal epithelium, can lead to periductal interstitial edema and local inflammation, with infiltration of lymphocytes and macrophages. (9) The edema of the upper airway is thought to be caused by local inflammation and circulatory disturbance of the lymphatic flow (e.g., lymphatic obstruction caused by the swollen parotid or submandibular glands). (2)

Steroid treatment without tracheotomy was effective in resolving pharyngolaryngeal edema in all of our cases. However, tracheotomy has been used for the resolution of pharyngolaryngeal edema in 2 cases, (2) whereas other cases have been treated successfully with administration of steroids.

Because mumps infection may be potentially complicated by pharyngolaryngeal edema, clinicians should examine the upper airway of patients with mumps by fiberoptic laryngoscopy. If pharyngolaryngeal edema is detected, steroid administration should be recommended and tracheotomy performed, if necessary.

Our cases include the first case involving a patient with mumps-associated pharyngolaryngeal edema who had previously received the mumps vaccine. This was probably due to the patients failure to respond to the vaccination or to waning immunity. A single dose and 2 doses of the mumps vaccine confer long-term protection in only 64% and 88%, respectively, of those vaccinated. (10) Although mumps infection usually confers lifelong immunity, recurrent mumps infection also has been reported. (11) In fact, in the literature, 2 patients with mumps-associated pharyngolaryngeal edema were diagnosed as having recurrent mumps infection.2 Therefore, patients, especially adults, regardless of a previous history of mumps infection or vaccination, may be at a risk of developing mumps-associated pharyngolaryngeal edema.

References

(1.) Mason WH. Mumps. In: Kliegman RM, Behrman RE (eds). Nelson Textbook of Pediatrics. 18th ed. Philadelphia: Saunders;2007:1341-4.

(2.) Ishida M, Fushiki H, Morijiri M, et al. Mumps virus infection in adults: Three casesofsupraglotticedema. Laryngoscope2006;116(12):2221-3.

(3.) Sasaki T, Tsunoda K. Time to revisit mumps vaccination in Japan? Lancet 2009;374(9702):1722.

(4.) Nakao Y, Tanigawa T, Shibata R. Dyspnea: A rare complication of mumps virus infection. Intern Med 2012;51(23):3311.

(5.) Iizuka T, Kusunoki T, Ono N, Ikeda K. Mumps virus infection with laryngeal oedema and thoracic wall phlegmonous inflammation in an adult. BMJ Case Rep 2013:2013. pii: bcr2012007829. doi: 10.1136/ bcr-2012-007829.

(6.) Hattori Y, Oi Y, Matsuoka R, et al. Pediatric mumps with laryngeal edema. Pediatr Emerg Care 2013;29(10):1104-6.

(7.) Wharton M, Chorba TL, Vogt RL, et al. Case definitions for public health surveillance. MMWR Recomm Rep 1990;39(RR-13):l-43.

(8.) WHO. Weekly epidemiological record2007;82:49-60. www.who.int/ wer/2007/wer8207/en/. Accessed March 6, 2015.

(9.) Weller TH, Craig JM. The isolation of mumps at autopsy. Am J Pathol 1949;25(5):1105-15.

(10.) Hanna-Wakim R, Yasukawa LL, Sung P, et al. Immune responses to mumps vaccine in adults who were vaccinated in childhood. J Infect Dis 2008;197(12):1669-75.

(11.) Yoshida N, Fujino M, Miyata A, et al. Mumps virus reinfection is not a rare event confirmed by reverse transcription loop-mediated isothermal amplification. J Med Virol 2008;80(3):517-23.

From the Department of Otolaryngology, Saitama Medical Center, Saitama, Japan.

Corresponding author: Masafumi Ohki, MD, Department of Otolaryngology, Saitama Medical Center, Saitama Medical University, 1981 Kamoda, Kawagoe-shi, Saitama 350-8550, Japan. Email: masaohkif[R] saitama-med.ac.jp
COPYRIGHT 2015 Vendome Group LLC
No portion of this article can be reproduced without the express written permission from the copyright holder.
Copyright 2015 Gale, Cengage Learning. All rights reserved.

Article Details
Printer friendly Cite/link Email Feedback
Title Annotation:ORIGINAL ARTICLE
Author:Ohki, Masafumi; Baba, Yuka; Kikuchi, Shigeru; Ohata, Atsushi; Tsutsumi, Takeshi; Tanaka, Sunao; Taha
Publication:Ear, Nose and Throat Journal
Article Type:Case study
Geographic Code:9JAPA
Date:Apr 1, 2015
Words:1466
Previous Article:Sinonasal undifferentiated carcinoma as a third primary neoplasm: a case report and review of the literature.
Next Article:Advanced airway management teaching in otolaryngology residency programs in Canada: a survey of residents.
Topics:

Terms of use | Privacy policy | Copyright © 2022 Farlex, Inc. | Feedback | For webmasters |