From Volume 41, Issue 1, January 2014 | Pages 73-76
Recurrent parotitis of childhood is an uncommon condition which affects children of variable age. The condition may be misdiagnosed so delaying treatment. These two case reports highlight the variable signs and symptoms of this unusual condition and the value of ultrasound as an aid to diagnosis.
Recurrent parotitis of childhood is characterized by recurring episodes of swelling and/or pain in the parotid gland during mastication and/or swallowing and is usually accompanied by fever and malaise. There is inflammation of the parotid gland, generally associated with non-obstructive sialectasis.1 The condition tends to be unilateral, but it can occur bilaterally, with a more predominant side. It can affect children from the age of 3 months to 16 years, with the highest incidence between 3 and 6 years of age, but may persist into adulthood.1,2,3 Males are more frequently affected.4
Although it is an uncommon condition, it is the second most common salivary disease in children after mumps4 (Figure 1). The interval between episodes of recurrent parotitis and the number of attacks vary individually, the main criterion for establishing the severity being the frequency of the recurrences.3
The parotid gland is the largest of the salivary glands. The gland is found wrapped around the mandibular ramus, and it secretes saliva through Stensen's duct into the oral cavity, to facilitate mastication and swallowing. Perfect function throughout life is normal. Dry mouth, drooling, swelling and pain are essentially the only symptoms caused by dysfunction of the salivary glands.5
The pathogenesis of juvenile recurrent parotitis remains unclear. Some factors suggested as contributory to the development of the condition are congenital ductal malformation,1,6 genetic factors,7 viral or bacteria infection,2 allergy8 and local manifestation of an autoimmune disease.9 Quenin et al4 suggested that recurrent parotitis in children stems from a discrepancy between the excretion and the secretion capacities of the parotid glands as a result of stenosis. A distinction was made between local stenosis and global stenosis. Local stenosis could be due to inflammation, affecting the main duct, and is usually found in adults. Global stenosis was noticed in the cases they examined, affecting the main duct and secondary branches of the ductal tree and appeared more suggestive of a congenital origin. The treatment success rate of 89% in their study, based on lack of recurrence following the use of sialendoscopy, was inconclusive as it could have resulted from the natural resolution of the disease or the procedure itself.4 Sialendoscopy acted as a diagnostic tool and interventional procedure as the actual diameter of the endoscope allows dilation and exploration of Stensen's duct.4
A 10-year-old female presented with intermittent pain from the left side of the lower jaw of about 6 months' duration. The discomfort was aggravated by eating and touching the side of the face. She also related some discomfort to toothbrushing with mint toothpaste in the mornings. Her mother noticed increasing bad breath (halitosis).
Past medical history revealed that she had staphylococcal scalded skin syndrome (Ritter disease) at 8 years of age and was hospitalized for a week. The patient suffered from seasonal hayfever and was on cetirizine hydrochloride (10 mg once daily) antihistamine medication. She was otherwise fit and well.
There was no family history of parotid gland problems. Further enquiries into any history of eye problems or dry mouth were negative.
On extra-oral examination there was no obvious facial asymmetry, however, there was tenderness on palpating the left parotid region posterior to the mandibular ramus. No tenderness of the submandibular and sublingual region was noted.
Oral hygiene was adequate with minimal plaque deposits. The mucosa was well lubricated and moist. Clear saliva was extracted from the left and right parotid ducts and there was no inflammation or tenderness over the duct openings. There was also no tenderness or thickening of the submandibular ducts on bimanual palpation.
The ultrasound images showed the presence of multiple hypoechoic areas within the parenchyma, suggestive of a chronic sialadenitis, confirming the diagnosis of recurrent parotitis10 (Figure 2).
The diagnosis was discussed with the patient and her mother and they were reassured. As there was no purulent discharge from the ducts, the use of non-steroidal anti-inflammatory analgesia was advised to alleviate symptoms. The patient was also advised to keep a diary on frequency and duration of the symptoms and further review appointments were arranged. At subsequent review appointments, over a period of nine months, the episodes of parotid swelling and tenderness became less frequent and she was discharged to the care of her dentist.
An 11-year-old female patient was referred with an 18-month history of intermittent parotid swelling affecting one or both glands. Previously she had been seen by an ENT department for chronic otitis media and surgical removal of the adenoids and tonsils.
The parotid gland enlargement was painful with restricted mouth opening. No dental cause for the swelling was identified during routine dental reviews. The patient had started on a week's course of antibiotics, prescribed by her general medical practitioner (GMP).
The past medical history was uneventful. She had symptoms suggestive of mumps and had been diagnosed as such by her GMP at the age of 8 years.
On extra-oral examination there was a tender erythematous swelling over the right parotid gland. The left parotid gland appeared normal. The oral hygiene was fair and there was no evidence of caries. A small amount of pus was extruded from the right parotid duct and this was swabbed for microbiology culture and sensitivity test. The rest of the oral mucosa appeared normal. An ultrasound scan of the parotid glands was arranged and a review appointment was made. The scan of the parotid glands showed a similar anomaly of multiple hypoechoic shadows within the gland parenchyma and an appearance in keeping with recurrent parotitis of childhood (Figure 3).
At review, the patient presented with marked, firm and tender swelling of the left parotid gland. The swelling of the right parotid gland had regressed and was not tender to palpation. There was no saliva or pus extruded from the left parotid duct. Normal saliva was expressed from the other salivary ducts.
Microbiology culture and sensitivity test reported Streptococcal pneumoniae isolation which was Amoxicillin sensitive so a course of Amoxicillin (250 mg three times daily for seven days) was prescribed.
The patient failed to attend three subsequent appointments but attended 5 months after the initial visit. She reported occasional swelling of both parotid glands. At six month review, she complained of increased severity and frequency of the parotid swellings, up to two episodes per month. The swellings had been quite painful and disturbed her school attendance. On this occasion, there were diffuse swellings and mild tenderness of the right and left parotid glands. Minimal saliva was expressed from all salivary glands.
A repeat ultrasound scan and blood investigations were arranged. The ultrasound report was consistent with recurrent parotitis of childhood and her blood results were within normal limits. The use of non-steroidal anti-inflammatory analgesia (Ibuprofen) was advised to alleviate pain.
Over three years, the patient was seen in the dental hospital with recurrent episodes of swellings and/or tenderness of the parotid glands of varied severity and frequency. Antibiotics and analgesia were prescribed to alleviate symptoms and advice given to increase fluid intake. A sialogram after an acute phase was consistent with recurrent parotitis of childhood, punctuated with globular sialectasia and retention of contrast post evacuation (Figure 4). Investigative tests to rule out Sjögren's syndrome and sarcoidosis were negative.
At the last review appointment, aged 15 years, the parotid glands were non-tender and good salivary flow was elicited.
This report described two patients with recurrent parotitis of childhood which had followed different pathways. Case 1 had recurrent symptoms which subsequently became infrequent. She was discharged to the care of her general dental practitioner within a year for follow-up, with the agreement of her parents. The condition of Case 2 was protracted and of varying intensity over many years until she was eventually lost to follow-up. The mumps reported in Case 2 at the age of 8 could have been an early presentation of recurrent parotitis of childhood, however, this could not be proven as no investigation was carried out. Both cases were symptomatically managed to alleviate pain following ultrasound scan confirmation of the diagnosis, and no definitive line of management was adopted. A sialogram was carried out in Case 2 to clarify the diagnosis due to the protracted nature of her condition; this would be avoided in the future. Other studies have found ultrasonography beneficial as the primary investigation for diagnosis, though a direct comparison with sialography found ultrasound less sensitive for inflammatory changes and sialolithiasis.1,11,12
The uncertainty about the aetiology of recurrent parotitis of childhood has hampered the development of a universally accepted treatment strategy for this condition. The problem is compounded by its rarity and uncertain natural history.1 A conservative approach has been recommended as the disease has a spontaneous recovery at puberty in most cases.13 Cohen et al3 recommended the use of low-dose antibiotic cover or prophylactic administration early in the attack, when a deficiency in salivary immunoglobulin is observed. The use of antibiotics and analgesia in acute episodes aims to prevent damage to the gland parenchyma and to alleviate the symptoms. The patients in this report were prescribed antibiotics, mainly during the acute phases; advice on the use of non-steroidal anti-inflammatory analgesia, as and when required, and increased fluid intake was also given.
A regime of parotid gland massage, increased fluid intake, warmth, use of chewing gum and sialogogues to increase salivary flow have been found to be helpful in alleviating the symptoms.14 Duct probing and dilation have been found effective and may help by clearing the plugs of mucus and cells that form in the acute phase,2,15 however, no preventive measures are available.14 Other treatment methods described in the literature include more invasive procedures, such as duct ligation, parotidectomy and tympanic neurectomy.1
Some studies found that performing a sialographic study resulted in significant improvement of symptoms and this was attributed to the flushing, dilating and use of antiseptic solution during the procedure.4,14,16
The possibility of recurrent parotitis of childhood as an early manifestation of Sjögren's syndrome has been highlighted in the literature.17,18 Sjögren's syndrome is a common slow progressing autoimmune disease with broad organ specific and systemic manifestations, the most prevalent being lacrimal and salivary gland function, xerostomia, keratoconjunctivitis sica and parotid enlargement. Sjögren's syndrome can occur alone (primary) or in association with systemic autoimmune diseases (secondary). The broad symptoms are not always present at the same time and can result in each symptom being treated individually by different specialties, consequently prolonging the interval between the onset and diagnosis.17 Parotitis appears to be the single most common glandular manifestation in children with Sjögren's syndrome. Symptoms related to dryness of the mouth and eyes are reported in a minority of childhood Sjögren's syndrome sufferers.19 Diagnosis of Sjögren's syndrome must be based on the American and European criteria for diagnosis;20 the patients with recurrent parotitis of childhood do not fulfil these. Other causes of parotid enlargement in children include inflammatory (viral and bacterial origin), congenital cystic lesion (first branchial cleft cyst, retention cyst), leukaemia, sialolithiasis, haemangioma, lymphangioma, HIV parotitis, benign tumours and malignancies. These tend to present as persistent swellings or one-off episodes.
Recurrent parotitis of childhood is an unusual and relatively rare cause of parotid swelling but should be considered in a child complaining of recurrent ‘mumps’ or peri-auricular swelling. Ultrasound scan provides a useful aid in the initial investigation and has replaced sialography as the imaging of choice in suspected juvenile recurrent parotitis. It should be supplemented with serological investigation to exclude Sjögren's syndrome.
