Ultrasound Findings in Juvenile Recurrent Parotitis - Case Series of TypicalPresentation and Sonographic Observations

Introduction

Juvenile recurrent parotitis is a recurrent inflammatory parotitis seen in children. It is a benign condition that is idiopathic and rare in occurrence. Recurrent glandular swelling and mild systemic symptoms are its characteristic findings. Typical ultrasound findings in cases suggestive of recurrent juvenile parotitis are described. Juvenile recurrent parotitis is the second most common cause of parotitis after mumps. This condition is however under recognized and mistaken for other causes of parotitis leading to multiple investigations.

Observations of typical ultrasound findings described in these case series would help in earlier diagnosis and management.

Case Presentation

Three children, two boys (aged 2.0 & 2.5 years) and a girl (aged 5 years) presented with recurrent attacks of parotitis. There was a history of recurrent parotid region swelling over a period of one year (Figure 1). 

The symptoms included asymmetric parotid glandular swelling towards the ear lobule with pain and mild fever. Both the right and left sides were involved. The symptoms were however lateralized to one side during the attack of painful swelling. The children were immunized with MMR vaccine and had good dental hygiene without caries.

They were usually playful after the episodes as described by their mothers; these episodes lasted for a few days and subsided with the medications. No seasonal variation or any predisposing conditions could be elicited. There was no history of fever or significant systemic complaints.

Mild leukocytosis and elevated C-reactive protein (CRP) were observed during the inflammatory episodes. Mumps serology was negative.

There was no history of consanguinity in the family tree.

There was a history of rheumatoid arthritis in the paternal grandfather of one child. His serologic testing was weakly positive for ant-ds DNA. No other immunologic markers were positive. The interpretation for the Antinuclear antibody (ANA) panel was negative for observation of Sjogren, Systemic Lupus Erythematosus (SLE) and rheumatoid arthritis.

There was no history of rheumatologic or connective tissue disorders in the family tree of the other two children. Their serologic tests were negative for any immunological disorder.

None of the children had any immunologic deficiencies or symptoms of the same.

The children were given symptomatic treatment with syrup Ibugesic (Paracetamol/Ibrufen) during the episodes of parotid swelling and were being followed up.

High resolution ultrasound of the parotid glands in all the cases revealed altered parenchymal echo with multiple small hypoechoic nodules (Figure 2). The nodulations and altered echo were observed in both the parotid glands.

Ultrasound system Logiq s8,GE : Matrix ,ML 6-15 linear probe was used.

A correlative Magnetic resonance imaging (MRI) was done for one of the patient. It showed multiple tiny hyperintense foci in both the parotid glands (Figure 3). 

No significant ductal dilatation, sialoliths were seen. There was a mild increase in vascularity on Doppler ultrasound.

Discussion

Juvenile recurrent parotitis is an ailment defined by recurrent attacks of parotitis with a male predilection. The onset is usually noted during 3-5 years of life.1 It is a self-limiting disease with the attacks subsiding usually by puberty.

Multiple theories of aetiopathogenesis have been proposed, but the exact causative factor remains elusive. Congenital, immunological as well as infective causes are noted in the literature.² Familial cases and clustering point towards genetic inheritance.³

Parotid gland is unique due to persistence of lymphatic tissue thereby predisposing it to various immunological and inflammatory disorders.

The disease manifests with multiple attacks of parotitis involving one or both the glands. The glands are usually swollen and tender. The systemic symptoms include fever and malaise. The swelling usually persists for 1-2 days. Cases having mucopurulent discharge have been reported in the literature.³

Sialography usually demonstrates duct ectasia in cases of recurrent parotitis. These changes are usually bilateral. High resolution ultrasound reveals altered glandular parenchyma with small hypoechoic to anechoic areas which represent sialectasis and lymphoid hyperplasia.⁴

A similar observation was noted on B mode high resolution ultrasound in our patients. The high resolution ultrasound revealed multiple small hypoechoic nodules.  

Similar changes have also been described in the salivary glands of patients having Sjogren syndrome and are suggestive of chronicity⁵ (Figure 4). These changes in Sjogren syndrome are also bilateral and serology can further aid in diagnosis.

Juvenile recurrent parotitis can be unilateral or bilateral and observations of such features can help in narrowing the diagnosis.⁶ There are, however, not many ultrasound reports of such observation in children with recurrent parotitis. The characteristic hypoechoic nodulation in these glands is likely due to lymphoid hyperplasia rather than being solely attributed to ductal ectasia.⁷

As parotitis in children could be due to many other causes like mumps, suppurative sialadenitis and sialoliths, ultrasound can be helpful to rule out sialoliths and other causes of sialadenitis. Also ultrasound is routinely available. It is non-invasive and free of radiation.

Magnetic resonance imaging (MRI) can be used for evaluation of the salivary glands and the ductal system. MRI can help in differentiating acute vs chronic changes by noting the difference in the signal intensity. Fibrosis usually appears as a decreased signal on T2 weighted sequence.⁸ However, MRI investigation for this condition, especially in children can be difficult, time consuming and may entail the need of sedation. Ultrasound can be well utilized as it is more readily available, is faster and safer. The characteristic pattern of observation of hypoechoic nodules can be used as a diagnostic marker for this condition. Various treatment modalities are described for this condition.

However, conservative management with anti-inflammatory medications and antibiotics are most commonly employed due to the self-limiting nature of the disease.⁹ Atypical and severe cases have been managed with more aggressive modes of treatment such as sialoscopy, corticosteroid instillation, ductal irrigation, parotidectomy etc.¹⁰

Conflict of interest

Nil.