Mumps virus is one of the major causative pathogens of sialadenitis. Some cases of laryngopharyngeal edema developing in association with mumps virus infection have been reported in Japan. We report three cases of suspected mumps virus infection who presented with laryngopharyngeal edema.
(Case 1) A 27-year-old female patient presented to us with the chief complaint of swelling and pain of the parotid gland region and breathlessness. Endoscopic examination showed significant laryngopharyngeal edema. Intravenous administration of a steroid proved ineffective against the edema. Then, we decided to secure the airway by oral intubation. The laryngopharyngeal edema gradually resolved and the patient was extubated on day 3 of hospitalization. Although the patient gave a history of having received vaccination against mumps infection, the serum test for IgM antibody against mumps virus was positive. Therefore, she was diagnosed as a case of mumps with primary vaccine failure.
(Case 2) A 44-year-old male patient presented to us with the chief complaint of swelling of the upper neck. Endoscopic examination revealed laryngopharyngeal edema, which resolved promptly with intravenous administration of a steroid. Mumps was ruled out by a negative serum test for mumps virus antibodies and a negative virus isolation test of the saliva. We suspected the cause to be a viral infection other than mumps.
(Case 3) A 29-year-old female patient presented to us with the chief complaint of swelling and pain of the parotid region and sore throat. Endoscopic examination revealed significant laryngopharyngeal edema, which resolved gradually with intravenous steroid administration. The causative pathogen in this case also remained unknown, as in Case 2.
Among the three cases, mumps virus infection was confirmed in one case, whereas the cause in the other two cases was considered to be a virus other than mumps virus. Patients with sialadenitis presumed to be caused by viral infection who present with the complication of laryngopharyngeal edema should be treated with intravenous steroid and be examined frequently by endoscopy.
While in most cases, viral parotitis is caused by mumps virus, other viruses have also been known to cause the disease. Over 100 viruses have been documented as causing viral parotitis, including parainfluenza, coxsackie and Epstein-Barr virus. Dozens of cases of acute parotitis with laryngeal edema have been reported domestically.
We report a case of parotitis and submandibular inflammation associated with significant laryngeal edema that was caused by a non-mumps virus.
The patient was a 34-year-old female who presented with a 3-day history of bilateral swelling of the parotid and submandibular regions. The swelling had become steadily worse and she complained of difficulty in swallowing. Physical examination revealed bilateral swelling of the parotid and submandibular regions. Laryngeal endoscopic examination showed significant edema of the epiglottis, valleculae and arytenoid cartilages of both sides. The patient was begun on intravenous steroid treatment, and the laryngeal edema immediately improved. Mumps was ruled out as the cause by the negative serum test for mumps virus antibodies, as also other viruses such as parainfluenza, coxsackie and EB viruses, by the negative serological tests for the respective viruses. We suspected some other virus as the cause.
Some cases of parotitis with laryngeal edema have been reported previously in Japan. It is presumed that parotitis with laryngeal edema is caused by circulatory disturbance because of bilateral swelling of the submandibular glands. Some reported cases of parotitis with laryngeal edema required tracheostomy.
In order to avoid unexpected or critical airway obstruction, laryngeal endoscopic examination and careful observation are important and essential.
抗myelin oligodendrocyte glycoprotein (MOG) 抗体は, 小児の急性散在性脳脊髄炎や視神経脊髄炎など脱髄疾患で陽性例が報告されている. 今回, 我々はムンプスウイルス感染症が先行し視神経炎 (optic neuritis; ON) で発症した抗MOG抗体陽性関連脱髄疾患の7歳女児例を経験した. 右眼痛, 右視力低下が出現し視神経炎の診断で当院へ紹介受診した. 右視力は10cm手動弁まで低下しており, 頭部MRIのT2強調画像とfluid attenuated inversion recovery (FLAIR) で右視神経の腫大, 両側大脳皮質下白質に高信号病変を認めた. ステロイドパルス療法3クール, ガンマグロブリン大量療法を施行したところ治療に反応し視力は右眼1.5まで改善した. その後, 抗MOG抗体の陰性を確認してステロイド内服を中止したが, 再発なく経過している. 抗MOG抗体についてはその病的意義や感染症との因果関係は明らかにされていないが, 本症例ではムンプスウイルス感染症が先行した. 抗MOG抗体とムンプスウイルスとの関連を指摘した報告はなく, 今後さらなる症例の蓄積による検討が必要である.