Case Presentation
A 57-year-old man was referred to the Department of Oral and
Maxillofacial, Kyoto Medical Center with the chief complaint of
recurrent swelling of the right parotid region. Four years before the
first visit, he developed bilateral parotid gland swelling and was
prescribed antibiotics (cefditoren pivoxil) by his otolaryngologist, and
the symptoms disappeared. Three years later, the patient experienced
rapid swelling and pain in the right parotid region during sleep when he
boarded an airplane and landed. He was prescribed antibiotics in the
landed country. The symptoms disappeared gradually and spontaneously,
but 3 weeks before the first visit to our department, severe symptoms
recurred in the same region, and he visited an otolaryngologist. He took
antibiotics (cefditoren pivoxil) for 2 weeks without improvement; he was
advised to visit a dentist, following which the dentist introduced him
to visit our department.
The right parotid gland of the patient was slightly swollen and tender.
He had no fever, and no abnormalities, such as redness, were observed on
the skin of the cheek. Blood tests showed no evidence of inflammation.
Crepitus was generated by light compression of the right parotid gland,
and frothy saliva with fine air bubbles was discharged from the papilla
of the right parotid gland (Fig 1). The crepitus and swelling in the
parotid region were prominent during waking up. Computed tomography
showed air predominantly on the right side of both parotid glands and
ducts, and no sialoliths (Fig 2). He was on oral medication because of
hypertension. The history of mumps remains unclear. For 20 years from
the age of 7 years, he played a traditional Japanese wind instrument,
hichiriki (Fig 3). He did not snore. He has gained 4 kg in the last 5
years. His body mass index was 24.1. Since the patient complained of
daytime sleepiness, sleep apnea syndrome was suspected. A sleep study
was performed on three nights at his home using a pulse oximeter
(Pulsox-300, Konica Minolta, Tokyo, Japan). The examination revealed a
mean apnea-hypopnea index of 10.3 and a mean minimal oxygen saturation
of 81.0%. The patient was diagnosed with mild sleep apnea syndrome. The
minimal oxygen saturation was considerably reduced.
It was postulated that desaturation events could be related to a rapid
increase in intraoral pressure. Therefore, an oral appliance to prevent
obstruction of the upper airway and reduce intraoral pressure (Fig 4)
was fabricated as described previously.29-31 The
mandibular position was 6 mm, protruding from the intercuspal position.
An anterior opening was made between the upper and lower incisors (Fig
4). After insertion of the oral appliance, the sleep variables improved
considerably (mean apnea-hypopnea index, 2.9; mean minimal oxygen
saturation, 90.8%). One month later, the symptoms of pneumoparotid
disappeared completely. The patient was followed up with the appliance.
After a few years, the patient discontinued the appliance as the
symptoms disappeared. After 4 years, the patient had bilateral parotid
gland swelling, and antibiotics were prescribed for 1 week by an
otolaryngologist. After 9 years, the patient had tenderness and swelling
of the left parotid gland. He revisited our department for
re-examination. The left parotid gland was tender. No abnormalities were
observed on the skin of the parotid region. Blood tests showed no
evidence of inflammation. Crepitus was detected by compression of the
left parotid gland, and frothy saliva with fine air bubbles was
discharged from the papilla of the left parotid gland. Computed
tomography showed the presence of air in both ductal systems, with more
prominent findings in the left parotid gland (Fig 2B). A sleep study was
conducted again. The test revealed a mean apnea-hypopnea index of 10.1
and mean minimal oxygen saturation of 79.8%. The results were
remarkably similar to those 9 years ago. Oral appliance was inserted
again, and sleep variables improved (mean apnea-hypopnea index: 3.1,
mean minimal oxygen saturation: 90.3%). The symptoms related to sleep
apnea syndrome and pneumoparotid disappeared completely. The patient was
followed up for 10 years from the first visit. Relapse of the entities
has not been observed until now.