Pneumoparotitis: A Rare Case Report

Abstract

Pneumoparotitis, a rare condition characterized by the presence of air within the parotid gland, typically arises due to an accumulation of air in the salivary ducts. Pneumoparotitis is a rare entity which affects children and young adolescents. This case report presents a rare instance of pneumoparotitis occurring in a 14-year-old female. The patient exhibited symptoms of localized facial swelling, discomfort, and tenderness, which initially led to diagnostic challenges. Through a combination of clinical evaluation, imaging studies, and meticulous history-taking, the condition was accurately diagnosed. Treatment involved conservative measures including hydration, warm compresses, and analgesics. This case emphasizes the importance of considering pneumoparotitis in the differential diagnosis of pediatric patients presenting with parotid gland swelling.

Introduction

The term “pneumoparotid” is defined as the presence of air within parotid acini and ducts. Pneumoparotitis is pneumoparotid with associated infection, a rare pathology of the parotid gland. It is caused by the retrograde introduction of microbes into the parotid gland via pneumatic trauma to the gland [1]. It is unusual to have pneumoparotid without pneumoparotitis due to the presence of abundant oral flora. Increase in intraoral pressure by Valsalva maneuver, or an incompetent Stenson’s duct leads to pneumoparotitis [2]. This entity presents as a diagnostic dilemma for clinicians as it clinically mimics parotitis.

Pneumoparotitis generally occur in children with psychosocial problems who induce parotid insufflation to avoid school or gain attention. This maybe self-induced or related to wind instrument blowing. It can pose as a diagnostic dilemma for young clinicians especially in patients who cannot voluntarily insufflate the parotid gland. Treatment is dependent upon the severity and duration of the disease which ranges from supportive measures to surgery [3]. In recurrent pneumoparotid associated with parotitis Stensen’s duct ligation, transposition of the Stensen’s duct to the tonsillar fossa, and parotidectomy can be considered as possible surgical options [4]. We wish to report a rare case of pneumoparotitis in order to create awareness about this rare but well-known cause of parotid swelling in children and adolescents. Pneumoparotitis should be also considered as a differential diagnosis in pediatric patients presenting with parotid gland swelling.

Case Report

A 14-year-old girl came to ENT emergency, with a history of pain and swelling over the right parotid region for 6 days. No history of fever, malaise, any wind instrument playing or past dental treatment. Past medical and psychological histories were unremarkable. On examination of the right parotid region, a 4 × 5 cm swelling which was localized, firm to soft, and tender with local rise of temperature and crepitus was present on palpation (Fig. 1). On oral cavity examination, no trismus was seen. On pressing the right parotid gland, frothy secretions coming out from the Stenson’s duct were visualized (Fig. 2). The rest of the ENT examination was unremarkable. All routine investigations were done which revealed an elevated total leucocyte count. Ultrasound of the parotid region was inconclusive due to an air shadow obscuring the local site. There was no collection noted in the peripheral parts of the right parotid region. CECT Neck was done which demonstrated the presence of a rounded air-filled right parotid mass (Fig. 3). Clinically, a diagnosis of pneumoparotitis was made. Analgesics, hydration, gland massage, warm compresses and a course of broad-spectrum antibiotics were given throughout the hospital stay. The swelling resolved in a week (Fig. 4). The child is doing well even after 1 year of follow-up.

Fig. 1.

Clinical photograph showing a right sided parotid swelling

Fig. 2.

Clinical photograph showing frothy secretions coming out from the Stenson’s duct

Fig. 3.

CECT Neck (axial view) showing a rounded air-filled right parotid mass

Fig. 4.

Clinical photograph showing resolution of parotid swelling post-treatment

Discussion

Pneumoparotitis was first described by Hyrtel in 1865, and since then, sporadic cases were reported in the literature under various synonyms like pneumoparotitis, wind parotitis, pneumosialadenitis, and anesthetic or surgical mumps. Over the years, the total number of documented cases has increased, with more than 170 cases now reported globally [1, 2, 5]. Various synonyms for the condition, such as wind parotitis, pneumosialadenitis, and anesthetic mumps, reflect the diverse circumstances under which pneumoparotitis can manifest. Early literature primarily focused on case reports and small series, often highlighting the association between pneumoparotitis and activities that lead to increased intraoral pressure, such as wind instrument playing, glassblowing, or certain occupational hazards. More recently, psychosocial factors have gained attention, especially in pediatric cases, with self-induced pneumoparotitis being observed in children and adolescents with underlying psychological concerns. Cases have been reported across a wide age spectrum, from young children to elderly adults. The condition has been observed bilaterally in many patients, but unilateral presentations are not uncommon. A significant portion of the literature has also focused on iatrogenic causes, particularly in patients undergoing positive pressure ventilation or dental procedures, which further underscores the condition's multifactorial etiology [2]. This case report of a 14-year-old girl presents a unique clinical scenario compared to the systematic analysis of pneumoparotid cases from the literature.

Clinical presentation commonly involved bilateral parotid swelling and was most frequently associated with self-induction, especially in children with psychosocial factors. Other etiologies included idiopathic causes and iatrogenic factors. In contrast to the systematic analysis that primarily focused on chronic cases and their etiologies, this case report highlights an acute presentation in a young patient with no associated psychological or medical history [3].

Diagnosing pneumoparotitis typically begins with a thorough medical history and physical examination by an ENT specialist. Patients often present with symptoms such as swelling, pain, and crepitus around the parotid gland, especially during or after activities that increase intraoral pressure. The characteristic popping or crackling noise during these activities can be a key diagnostic clue. The pathophysiology of pneumoparotitis arises when air enters the parotid duct. The Stensen's duct usually has safeguards to prevent air from going backward into the parotid gland. But in some people, a large increase in the intraoral pressure overcome these normal protections, and air and saliva can get into the duct. Anatomic abnormalities, including a patulous Stensen’s duct, masseter muscle hypertrophy and buccinator muscle weakness are thought to make individuals more prone to develop pneumoparotitis [6]. Diagnostic methods have evolved from clinical assessments to more sophisticated imaging modalities such as ultrasound and computed tomography (CT) scans, which can effectively demonstrate air within the parotid gland and surrounding tissues. In several reports, ultrasound findings, including echogenic foci with acoustic shadowing within the gland, have been key to confirming the diagnosis. CT imaging remains a gold standard for assessing the extent of pneumoparotitis and ruling out complications like pneumomediastinum, which has been highlighted in several cases. Imaging can also help rule out other potential causes of parotid gland swelling, such as infections or stones within the duct [7]. Once a preliminary diagnosis is made, further investigations may be necessary to determine the underlying cause of pneumoparotitis and to guide appropriate treatment.

The management of pneumoparotitis primarily depends on the underlying cause and severity of the condition. In mild cases, conservative measures such as avoiding activities that increase intraoral pressure, warm compresses, analgesics and occasionally antibiotics may be sufficient to alleviate symptoms. In instances where infection is present, appropriate antibiotic therapy is essential. In children and adolescents with psychosocial problems, behavioural therapy can also be useful [8]. However, in cases with recurrent or severe pneumoparotitis, surgical interventions in the form of Stensen’s duct ligation, transposition of the Stensen’s duct to the tonsillar fossa, and parotidectomy may be considered [4].

Recurrence remains a common theme in the literature, with up to 42.6% of cases experiencing relapse, particularly among patients with underlying psychiatric disorders. Complications like subcutaneous emphysema, pneumomediastinum, and parotitis have also been well-documented, emphasizing the importance of prompt diagnosis and appropriate management to prevent potentially life-threatening outcomes. Despite these challenges, the prognosis for pneumoparotitis remains favourable, with most cases resolving without significant long-term sequelae when managed appropriately [7, 8]. The specific treatment approach should be individualized to determine the most appropriate course of action.

Conclusion

This rare case of pneumoparotitis highlights the need for thorough history-taking and imaging for accurate diagnosis. Air in the parotid gland is uncommon in children which can lead to misdiagnosis. By raising awareness, we can ensure quicker treatment and improve patient outcomes.

Funding

None.

Declarations

Conflict of interest

There are no potential conflicts of interest.

Ethical Approval

Yes.

Informed Consent

A written informed consent was taken from the patient's parents in the study.