Karolina Raczkowska-Łabuda, Monika Jabłońska-Jesionowska, Jolanta Jadczyszyn, Magdalena Frąckiewicz, Maciej Pilch, *Lidia Zawadzka-Głos
Self-induced pneumoparotitis – a rare case report
Pneumoparotitis wywołany autogennie ? opis przypadku rzadkiego schorzenia
Department of Paediatric Otolaryngology, Medical University of Warsaw
Head of Department: Associate Professor Lidia Zawadzka-Głos, MD, PhD
Pneumoparotitis (pneumoparotiditis, pneumosialoadenitis) to schorzenie rzadkie i często błędnie diagnozowane. Cechy odmy podskórnej w rzucie przyusznicy, szyi czy śródpiersia należą do niepokojących objawów, które bezzwłocznie należy wyjaśnić. Badanie TK stanowiące złoty standard diagnostyczny można w populacji dziecięcej z powodzeniem zastąpić badaniem USG. Sialografia stanowi dopełnienie obrazowania gruczołu i daje szansę na irygację przewodów ślinianki z zalegających złogów ? w prewencji nawracających stanów zapalnych. W artykule przedstawiono przypadek 12-latka, który świadomie, przez około pół roku, wypełniał obie przyusznice powietrzem, wytwarzając pneumocele prawego gruczołu o średnicy ok. 25 mm. Diagnostykę obrazową rozszerzono o badania z grupy chorób autoimmunologicznych. Przeprowadzono szczegółowy wywiad rodzinny. Zastosowano leczenie zachowawcze, w tym irygację przewodu Stenona deksametazonem, uzyskując pełne wycofanie objawów. W 3-miesięcznej obserwacji nie stwierdzono powiększenia pneumocele ani epizodu parotiditis.
Pneumoparotitis (pneumoparotiditis, pneumosialoadenitis) is a rare and frequently misdiagnosed condition. Signs of subcutaneous emphysema in parotid gland, the neck or the mediastinum are alarming symptoms that should be promptly addressed. Computed tomography, which is the gold diagnostic standard, may be successfully replaced with ultrasonography in the paediatric population. Sialography has a complementary role in the imaging of the gland, and allows for parotid duct irrigation to remove deposits and prevent recurrent inflammation. The paper presents a case of a 12-year-old boy who deliberately inflated his both parotid glands for about 6 months, and thus developed parotid pneumatocele with a diameter of about 25 mm. Diagnostic imaging was extended to include autoimmune diseases. Detailed family history was obtained. Conservative treatment, including dexamethasone irrigation of the Stensen’s duct, was used and the symptoms fully resolved. A 3-month follow-up showed no increase in the size of pneumatocele or episodes of parotiditis.
The parotid glands should be impalpable in children. Noticeable facial asymmetry and unilateral or bilateral oedema are usually associated with parotid pathology. The most common causes include infection, autoimmune or lymphoproliferative diseases, duct obstruction, as well as benign or malignant tumours. Rapidly growing oedema is a typical outcome of viral infection or chronic, recurrent parotitis.
The term “pneumoparotid” refers to the presence of air in either the parotid gland or the Stensen’s duct (1, 2). Pneumoparotiditis is a parotitis secondary to a retrograde pathogen invasion through the duct orifice, which is dilated as a result of barotrauma (3). The presence of pneumoparotid in the absence of pneumoparotiditis is unlikely due to the diversity of oral flora. The co-existence of secondary inflammatory oedema often leads to a misdiagnosis of simple parotitis.
Patients at risk of pneumoparotitis include wind instrument players and adults with mental disorders (4, 5). Most patients are aware of their ability to inflate the gland. Patients with unintentional parotid gland inflation pose a greater diagnostic challenge.
A 12.5-year-old boy was admitted in an emergency to the Department of Paediatric Otolaryngology, Medical University of Warsaw, due to a swelling in the right cheek. About 1.5 days earlier, he completed a 7-day course of oral clindamycin for parotitis. The diagnosis and the treatment were suggested in the Department of Otolaryngology of another centre, where the patient had stayed for 5 days prior to outpatient treatment. At that time, salivary gland ultrasound showed inflammation of the gland and a cyst in the mandibular angle. Head CT was performed (fig. 1, 2).
Fig. 1. CT in a patient with pneumoparotid. A dilated Stensen’s duct and a pneumatocele in its distal part may be seen on the right
Fig. 2. CT in a patient with pneumoparotid. A dilated Stensen’s duct on the left
The findings were as follows: “A 23 mm space filled with air in the right parotid region, below the external auditory canal, in the projection of the salivary gland; air is also present in the salivary ducts of the right parotid gland and the parotid duct. On the left, air is present in the parotid duct and salivary ducts within the gland. Cervical lymphadenopathy up to 21 mm. Positive intra-oral pressure causes dilation of both the salivary ducts and the right-sided gas-filled space. The CT image most likely corresponds to a cyst drained through a fistula to salivary ducts, with a leaking orifice and air reflux into the salivary glands”. Buccal oedema located directly under the right ear lobe again increased rapidly, causing moderate pain in the child. Inflammatory markers on admission were normal: CRP < 0.5, WBC 8.59 thous., LYMF 34.3% (N: 24.7-56.0%), NEUT 56.3% (N: 43-65%), MONO 6.6% (N: 4.4-12.3), EOSINO 2.4% (N: 0.0-4.4%). Body temperature was 36.6°C. Otolaryngological examination showed no evacuation of pathological content from the dilated orifice of the gland, but only normal secretion containing air bubbles. Emergency ultrasound findings were as follows: “Both parotid glands are not enlarged, but show inhomogeneous parenchymal echostructure, without any focal lesions. A slight amount of air in the left parotid duct; a large amount of air and a small amount of fluid (about 6-5 cm) in the right parotid gland. Slightly increased vascularity in the right parotid gland. Mild reactive lymphadenopathy up to approx. 24 x 7 mm at both submandibular areas. No oedema of the surrounding tissue”. Based on the clinical picture and medical history, IV Amoxiclav was included by the doctor on duty. The boy admitted that during the past months he “played” by inflating both parotid glands. Initially, the dilation of the distal end of the duct was unnoticeable, and the resistance during gland insufflation was high. With time, however, the boy was able to develop a 27-mm-long pneumatocele. He also reported that he derived much joy from the “bubbling sensation” during air evacuation.
According to the patient’s mother, the boy had a history of 2 episodes of right-sided parotitis. Purulent discharge from the Stensen’s duct was observed each time. Both infections were developed in the last 6 months.
At the time of admission, the patient was under outpatient immunological, gastroenterological and cardiological care. He complained of a few-minute episodes of intra-abdominal pain unrelated to food ingestion for several years. His mother confirmed the lack of appetite, extended chewing time and difficulties swallowing solid foods. Percentile charts were difficult to develop due to the lack of measurements. At examination, the boy’s body weight of 31 kg corresponded to the third percentile (25th percentile at 5 years old), and his height of 149 cm corresponded to the 25th percentile (27th at 5 years old). The mother reported recurrent URTIs, allergy (no allergen test or treatment), gastroesophageal reflux disease (no medical records or treatment) and excluded celiac disease (again no records). Family history additionally revealed that the boy’s father was diagnosed with and treated for Barrett’s oesophagus.
Laboratory tests were run at the Department to evaluate total IgE, serum and urine amylase, anti-TPO, TSH, ANA, ANCA, Ig A, Ig M, Ig G, EBV, CMV, RF, CRP, and complete blood cell count. Abnormal findings were reported for total IgE ? 163 kU/L (N: 0-100). The above mentioned ultrasound was performed.
The patient was qualified for bilateral sialography. The procedure was performed under local anaesthesia: “Contrast agent (Imeron) was administered through a catheter inserted into the right parotid duct ? the dilated parotid duct was filled; several smaller spaces in the distal part of the duct and one significantly larger space were also filled. Contrast agent evacuation from these spaces was spontaneous (fig. 3). This was followed by contrast agent (Imeron) administration through a catheter inserted into the left parotid duct ? the parotid duct, which gave off branches in its distal dilated part, was filled with contrast agent (fig. 4)”.
Fig. 3. Right parotid fistulography. Dilated duct and a contrast-enhanced cistern at its end are seen
Fig. 4. Left parotid fistulography. A dilated duct, mainly in its distal portion, is seen
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