Volume 4, Issue 1 , Pages 29-31, January 2009
A rare presentation of recurrent parotid swelling: Self-induced parotitis
Article Outline
Summary
Recurrent swelling of one or both parotid gland is relatively frequent in children. Self-induced pneumoparotitis is a rare cause of recurrent parotitis. Clinicians may misdiagnose self-induced pneumoparotitis which may present with complications and needs specific treatment. We present a case initially suspected as a parotid cellulitis. The diagnosis was based on clinical history and radiological findings.
Keywords: Pneumoparotitis, Juvenile recurrent parotitis, Parotid swelling, Facial crepitus, Juvenile sialedinitis
1. Introduction
Recurrent swellings of parotid glands are relatively frequent in children.
Mumps is a rare cause of parotitis nowadays in developed country due to vaccinations. Parotitis due to other causes is also rare in children, viral parotitis being the most common and recurrent juvenile parotitis the second common. The causative agent of juvenile recurrent parotitis is still of unknown aetiology.
More rarely, recurrent parotid swelling may be caused by insufflations of air via the Stensen's duct. It may be an occupational hazard in trumpet players or glass blowers. Auto insufflation is the most common cause of pneumoparotitis in children.
2. Case report
A 9-year-old boy with suspected recurrent parotitis was referred to the Otolaryngology Department. There had been three previous episodes of parotid swelling. The first one had occurred 3 years earlier and resolved spontaneously. The last episode had been treated with antibiotics and oral steroids but without any clinical effect. Mumps vaccination was up to date. There was no fever, fatigue or any inflammatory sign on the cheek skin. Playing of a wind instrument was denied. Examination found oedema of the Stensen's papilla without pus secretion. In the oral cavity, frothy, air-filled saliva was extruding from the orifice of the left Stensen's duct. Palpation of the cheek, however, revealed a typical emphysematous sensation with crepitus. Complete blood cell count and serum electrolyte levels were normal. Initial ultrasound found diffuse emphysema extending into the left cheek, the parotid gland region and the adjacent lateral cervical region. Stensen's duct appeared dilated, yet without any sign of sialolithiasis.
Facial computed tomography (CT-scan) (Fig. 1) was performed.
Fig. 1.
Facial computed tomography CT-scan: air inside the left parotid duct, as well as extra ductal air within the parotid gland, subcutaneous soft tissues, and even in the parapharyngeal space (Fig. 2).
Non-contrast CT demonstrated air inside the left parotid duct, as well as extra ductal air within the parotid gland, subcutaneous soft tissues, and even in the parapharyngeal space (Fig. 2).
Fig. 2.
Sub-clavicular extension.
On further questioning, the patient admitted puffing his cheeks in stressful situations. He demonstrated the maneuver in the clinic. Psychological evaluation detected conflicts with the child's parents as the most probable cause of chronic stress. Subsequently, the patient was referred for psychiatric counselling.
3. Pathogenesis
Normal intraoral pressure is 2–3
mmHg. Dental instruments driven by compressed air may increase intrabuccal pressure to 60–65mmHg [1]. Glassblowing and trumpet playing can also increase intraoral pressure up to 140 or 150mmHg. Such dramatic and brutal elevations of intraoral pressure may overwhelm normal protective mechanisms and air with saliva can enter the ductal system of salivary glands. This air may be pushed as far up to the acini. At this stage it can become palpable as crepitus. If the pressure continues to increase, the acini may rupture leading to diffusion of air into the parapharyngeal space and neck. Ultimately, a pneumomediastinum may occur [2].
4. Diagnosis
In children, inflammatory swelling of the parotid gland is usually due to acute viral or bacterial infection, juvenile recurrent parotitis, or eventually allergic, autoimmune or systemic disease. Rarely, swelling may result from air being forced through the Stensen's duct, leading to pneumoparotitis. Such an episode may be transient or a recurrent phenomenon [3], [4], [5]. Recurrent parotid insufflation is not without consequence as it may predispose to sialectasy, recurrent parotitis and subcutaneous emphysema. Self-induced pneumoparotitis may be bilateral [4], [5].
The disorder has been reported as an occupational hazard in wind instrument players and glass blowers, and rarely as a non-occupational disease [6]. Such cases have been described in adolescents, and often in association with psychological disorders [7], [8]. At a more important stage, self-induced pneumoparotitis leads to cervical and facial subcutaneous emphysema, with limited pneumomadiastinum.
A review of the English literature found 17 reported cases of self-induced parotitis. Most of these occurred before the age of 17. The cause may be particular maneuver due to unconscious habits or deliberate attempts to obtain secondary gains. In all pediatric cases, medical history review or patient and family questioning found psychological disorders. Typically, physical examination found swelling of the parotid gland with crepitus, and excretion of frothy, air-filled saliva from the Stensen's duct after parotid massage. CT scan confirms the diagnosis by showing extreme widening of the Stensen's duct between the parotid gland, masseter muscle and buccinator muscle. Imaging also reveals total loss of parotid gland tissue signal due to massive air insufflation [9], [10].
Treatment of pediatric self-induced parotitis may be challenging. Patients are most often unaware of causative buccal maneuvers, or reluctant to admit their reasons for and methods of auto-insufflations. These factors combined can delay diagnosis. Acute sialadenitis is usually managed with antibiotics, massage, sialogogues and hydration. Etiological treatment should include counselling in order to counter maladaptive maneuvers, associated with psychotherapy in some cases.
References
- . Pneumoparotitis with orthodontic treatment. Am. J. Orthod. Dentofacial. Orthop. 1993;103(April (4)):377–379
- . Crisis of recurrent parotid swelling in factitious cervico-facial subcutaneous emphysema. Rev. Clin. Esp. 1980;157(May (3)):
- . Pneumoparotiditis. An unusual case of parotid gland swelling. Arch. Otolaryngol. 1973;97(June (6)):468–469
- . Non-occupational recurrent bilateral pneumoparotitis in an adolescent. J. Laryngol. Otol. 1992;106(June (6)):558–560
- . Self-induced pneumoparotitis. Arch. Otolaryngol. Head Neck Surg. 1995;121(December (12)):1426–1429
- . Parotid swelling and school phobia. Arch. Otolaryngol. 1970;92(October (4)):396–398
- . Self-induced pneumoparotitis in an adolescent. Report of a case and review of the literature. Int. J. Pediatr. Otorhinolaryngol. 1987;14(December (2/3)):113–121
- . Subcutaneous emphysema in a borderline patient. Rev. Pneumol. Clin. 2003;59(June (3)):149–153
- . Fischbach R high-resolution magnetic resonance imaging of recurrent pneumoparotitis. Ann. Otol. Rhinol. Laryngol. 1999;108(August (8)):816–818
- . Pneumoparotitis: diagnosis by computed tomography. Am. J. Otolaryngol. 1999;20(January–February (1)):68–71
PII: S1871-4048(08)00040-3
doi:10.1016/j.pedex.2008.05.004
© 2008 Elsevier Ireland Ltd. All rights reserved.
Volume 4, Issue 1 , Pages 29-31, January 2009
