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Case Reports Indian Pediatrics 2000;37: 1126-1129 |
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Juvenile Recurrent Parotitis |
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Vivek Jain N.B.S. Mani* Meenu Singh Lata Kumar
Juvenile recurrent parotitis is a rare disease characterized by recurrent parotid inflammation, associated with non-obstructive sialectesis(1). Here we report a case of juvenile recurrent parotitis, suspected clinically and diagnosed by sialography. To the best of our knowledge, this condition has never been reported previously from India.
A four-year-old male child presented with a history of repeated episodes (thrice over last one year) of painful swelling below the right ear. Each time the swelling subsided over a period of 7-10 days. This time also he complained of increasing swelling accom-panied by pain and fever. Except for bad oral hygiene his physical examination was unremarkable. His height (96 cm) and weight (13 kg) were normal for age. Initially possibilities of salivary duct calculi, Sjogren’s syndrome and juvenile recurrent parotitis were considered. The laboratory investigations revealed hemoglobin of 10.1 g/dl and total leukocyte count of 8,600/mm3 with 68% neutrophils, 30% lymphocytes and 2% eosinophils. Erythrocyte sedimentation rate was elevated (25 mm/h; normal: 1-20). Gram stain and culture of saliva did not reveal any micro-organism. As juvenile recurrent parotitis is associated with immunological abnormal-ities, a detailed immunological workup including NBT test, ELISA for HIV, Rebuck skin window test and serum immune electrophoresis was done, which was normal. Schirmers’ test and slit lamp examination done for Sjogren’s syndrome were non-contributory. Investigations for auto-immune markers were not done. Plain X-rays of the skull (AP and lateral) did not reveal any abnormalities. Ultrasound of right parotid gland revealed enlarged gland with hypoechoeic areas. Other salivary glands were normal. Sialography performed with water soluble contrast medium revealed multiple ectatic ducts in the periphery which were uniform in size measuring 2-3 mm in diameter (Figs. 1 & 2). The main parotid duct was normal, there was no filling defect within it to suggest calculus. The child was treated with oral penicillin V (250 mg bd) and naproxen for seven days. The swelling subsided within three days. The child is on regular follow up.
Juvenile recurrent parotitis is characterized by recurrent episodes of swelling and pain in parotid gland(1). This condition is usually misdiagnosed as mumps but in contrast, the swelling is recurrent and affects the parotid gland unilaterally and when bilateral, one gland is affected less than the other(1). The onset of disease is early in life with a peak during 3-5 years of age. It is usually accompanied by pain, fever and malaise and the frequency of exacerbations can be quite variable, though the disease disappears completely in adult life(2). The disease is more common in males(3). The histological features of recurrent parotitis in childhood include sialectasis of peripheral ducts in the parotid gland with periductal lymphocytic infiltration. The ectatic ducts are usually 1-2 mm in diameter. Congenital ectasia of portions of salivary gland ducts and infection ascending from mouth have been postulated to explain the parenchymal changes and pathogenesis of juvenile recurrent parotitis(2). An extensive search of the medical literature did not reveal involvement of other salivary glands in this clinical entity. The higher rate of secretion in the submandibular gland compared to partoid gland may protect it from infections and hence from recurrent enlargement. Also, the submandibular secretion has been found to have antiseptic properties(2). Sialographic examination is the commonly used diagnostic modality for children with recurrent parotitis. It reveals numerous scattered punctate/globular pools of contrast medium which usually measure 1-2 mm in diameter(4). Histologically these pools of contrast correspond to the peripheral intralobular ducts(2). Ultrasonography of the parotid gland using 7.5 MHz high frequency transducer reveals enlarged parotid gland in majority of patients with multiple small hypoechoic areas measuring 2-3 mm in diameter(4,5). The hypoechoic areas represent both sialectasis of peripheral ducts and surrounding lymphocytic infiltration. The ultrasonographic findings are characteristic of recurrent parotitis but is not specific for this disease. The differential diagnosis includes mumps, Sjogren’s syndrome and other chronic inflammatory diseases(5). Juvenile recurrent parotitis has been occasionally associated with immunological abnormalities, especially phagocytic dysfunctions(6). The recurrent attacks are treated conser-vatively with oral penicillin and analgesics. However, no prophylactic therapy is available. In addition to antibiotics, analgesics and attention to good oral hygiene, massage of the parotid gland, warmth, use of chewing gum and sialogogic agents may be helpful in reducing the attack frequency(3). More aggressive treatment is justified only for those patients with persistent problems. This may include parotid duct ligation, paroti-dectomy or tympanic neurectomy depending upon preference and experience of the treating physician(3). Intraductal tetracycline produced acinar atrophy in rabbits(7). No studies have been conducted on human subjects.
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