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Journal of Indian Society of Pedodontics and Preventive Dentistry Official publication of Indian Society of Pedodontics and Preventive Dentistry
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CASE REPORT
Year : 2014  |  Volume : 32  |  Issue : 3  |  Page : 262-265
 

Juvenile recurrent parotitis in children: Diagnosis and treatment using sialography


1 Department of Pedodontics and Preventive Dentistry, GITAM Dental College and Hospital, Rushikonda, Visakhapatnam, Andhra Pradesh, India
2 Reader, Departments of Oral Medicine and Radiology, J.S.S Dental College, S.S. Nager, Mysore, India
3 Department of Pedodontics and Preventive Dentistry, Coorg Institute of Dental Sciences, Virajpet, Karnataka, India

Date of Web Publication2-Jul-2014

Correspondence Address:
Vanga V Narsimha Rao
Department of Pedodontics and Preventive Dentistry, GITAM Dental College and Hospital, Rushikonda, Vishkapatnam - 530 045, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-4388.135846

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   Abstract 

Juvenile recurrent parotitis (JRP) is a nonobstructive, nonsuppurative parotid inflammation in young children. Causative factors are many such as allergy, infection, local autoimmune manifestations, and genetic inheritance have been suggested, but none have been proved. Parotid sialography is a hallmark in the diagnosis of JRP but newer modalities such as ultrasonography, computed tomography and magnetic resonance imaging-sialography are noninvasive investigative techniques. Recurrent attacks are often managed conservatively. Here we report a case of a 5-year-old child with JRP. Sailography can be used as both diagnostic and therapeutic modality.


Keywords: Juvenile recurrent parotitis, juvenile parotitis, sialography


How to cite this article:
Narsimha Rao VV, Putta Buddi JH, Kurthukoti AJ. Juvenile recurrent parotitis in children: Diagnosis and treatment using sialography. J Indian Soc Pedod Prev Dent 2014;32:262-5

How to cite this URL:
Narsimha Rao VV, Putta Buddi JH, Kurthukoti AJ. Juvenile recurrent parotitis in children: Diagnosis and treatment using sialography. J Indian Soc Pedod Prev Dent [serial online] 2014 [cited 2019 Nov 21];32:262-5. Available from: http://www.jisppd.com/text.asp?2014/32/3/262/135846



   Introduction Top


Juvenile recurrent parotitis (JRP) is a rare, recurrent nonobstructive, and nonsuppurative parotid inflammation in young children with a multifactorial etiology. [1]

It is characterized by recurrent episodes of swelling and or pain in the parotid gland usually associated with fever and malaise. It affects commonly children, but may persist into adulthood. [2] It is a rare condition, and its etiology remains obscure and has a tendency to resolve spontaneously after puberty. [2],[3]

Juvenile recurrent parotitis is usually unilateral, but bilateral exacerbation can also occur, with symptoms usually more prominent on one side. Diagnosis of JRP is made mainly through history, parental report of unilateral or bilateral parotid gland recurrent infections. [4]

In general, symptoms begin as early as 3 years of age. The duration of attacks average 3-7 days, but may last up to 2-3 weeks in some individuals. During these attacks, the parotid gland is enlarged, moderately red, and tender. Massaging the salivary glands from back to front produces clear saliva with lots of "snowflakes" or little white curds from the Stensen's duct. [5] Bacterial cultures from saliva of such cases generally produce cultures of Streptococcus viridans or other low-virulence bacterium that are considered as normal oral flora. [5]

The purpose of this report is to discuss a case of recurrent parotitis, its diagnosis using sialography and its clinical management.


   Case Report Top


A 5-year-old male child reported with a history of repeated episodes (8-9 times over the last 1 year) of a painful swelling near the right ear. The swelling was initially reported to be small in size and gradually increased over 2 days which was extremely painful associated with fever and difficulty in opening the mouth. Extra-oral examination showed a diffuse swelling in the right preauricular region extending posteriorly beyond the ramus of the mandible with mild erythema and rise in temperature. The right submandiblar lymph node was enlarged, single in number, tender, mobile, oval and soft in consistency.

Intra-oral examination showed poor oral hygiene with multiple carious teeth. Sialometry of both unstimulated and stimulated salivary flow showed decreased salivary secretion with lots of snowflakes. Differential diagnoses of salivary duct calculi, JRP, and Sjogren's syndrome were considered.

Hematological investigations revealed hemoglobin level of 13 g% and total leucocyte count of 8,600/mm 3 with 52% neutrophils, 43% lymphocytes, and 5% eosinophils. Erythrocyte sedimentation rate was also elevated at 18 mm/h.

To reduce the acute symptoms amoxicillin (200 mg) + clavulanic acid (28.5 mg) was administered. After acute symptoms subsided, salivary assessment test was done to evaluate salivary flow, pH and buffering capacity using GC salivary kit. The unstimulated salivary flow rate for 5 min was <3 ml (low), salivary pH was 6.8 and salivary buffering capacity was 5 which was low (2 blue + 1 red/blue; 2 + 2 + 1 = 5).

Radiographic examination of the skull (anteroposterior and lateral) and orthopantomograph (OPG) did not reveal any abnormalities [Figure 1]. Ultrasound of salivary gland revealed right parotid gland normal in size, with altered ecotexture with reduced ecogenenicity when compared to contra lateral side. Multiple cystic areas were evident with increased blood flow suggestive of chronic inflammation [Figure 2].
Figure 1: Stout orthopantomograph image

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Figure 2: Ultrasound images showing hyperechoic areas with cavitation of parotid gland

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Complete restorative and rehabilitation procedures of all the carious teeth were performed along with thorough oral prophylaxis to rule out any odontogenic source of infection. Simple injection sialography technique was performed using water soluble dye, urografin 76% (meglumine amidotrizoate) under local anesthesia, after confirming the absence of allergy to the dye.

Parotid gland sialography showed uniform normal Stenson duct diameter of 2-3 mm from opening till the periphery of the gland. Accessory diversification was also seen from the duct giving appearance as "tree in winter" appearance [Figure 3]. Secondary branches showed areas of "blobs" and "dots" known as sialectasis, which happens due to repeated chronic inflammatory condition leading to loss of adhesion between the cells lining the duct. Digital OPG repeated after 1 min revealed 50% excretion of the dye. After 5 min, on using secretogogues (lemon juice) complete excretion of the dye from the gland was observed suggesting normal functional state of the gland [Figure 4]. The patient was advised to use secretogogues (limcee chewable tablet) and analgesics for 3 days (ibuprofen + paracetamol). The child was on regular follow-up once every 6 months and followed for 2 years. There was no recurrence of parotitis during this period.
Figure 3: Sialography image of diversifi cation appearing as "tree in winter"

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Figure 4: Orthopantomograph showing complete execration of dye after 5 min

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   Discussion Top


Juvenile recurrent parotitis is characterized by recurrent episodes of swelling and pain in parotid gland. [6] The notable difference between JRP and mumps, is that while the child develops fever, malaise, headache, and chills with mumps, the symptoms in JRP are usually more localized to the parotid gland with occasional bouts of fever. [4] The onset of disease is early in life with a peak during 3-5 years of age [6] with a male predilection. [2],[6],[7] JRP is self-limiting and after puberty, the symptoms usually subside and the disease may resolve completely. [4]

Galili and Marmary [8] proposed two possible mechanisms for spontaneous resolution of the symptoms: Total atrophy with consequent absence of symptoms or regeneration of gland from surviving ductal cells. Geterud et al.[3] have reported drainage of purulent secretion through the duct after compression of the parotid gland. Swelling normally lasts from few days to 2 weeks and is spontaneously resolved, regardless of the treatment.

Sialographic examination is the commonly used diagnostic modality for children with recurrent parotitis. [6] Different contrast agent for sialography are:

  1. Ionic aqueous solution: Diatrizoate, metrizoate.
  2. Nonionic aqueous solution: Iohexol.
  3. Oil based solution: Iodized oil.


Urografin 290 (water soluble) is the better contrast agent for sialography because of better filling of intragland ducts and greater clinical tolerance when compared with lipiodol ultra fluid (lipid soluble).

Sialograph revealed dots or blobs of contrast media distributed throughout the gland, an appearance known as "sialectasis" suggestive of sialadentis caused by the inflammation of glandular tissue producing dilation of terminal duct and sac like acini with normal main duct. [9] Histologically these pools of contrast correspond to the peripheral intralobular ducts. [6]

The most frequently used test for the diagnosis of JRP is still sialography, since ultrasonography is noninvasive has been increasingly used for diagnosis and during follow-up.

In our case, ultrasonography of the parotid gland revealed a normally sized right parotid gland, with altered ecotexture as compared to left with reduced ecogenenicity. Multiple cystic areas and increased blood flow suggestive of chronic inflammation was reported.

Although, parotid sialography aids in the diagnosis of JRP, yet the invasive nature and the difficulties encountered in the conventional sialography has led to the development of newer diagnostic modalities such as ultrasound, computed tomography and magnetic resonance (MR) sialography. MR sialography does not require any contrast medium. It primarily images liquid structures and the flow can be adequately demonstrated after stimulation with ascorbic acid. These special features of MR sialography allow its use also during acute episode of sialadenitis.

Antibiotic treatment prevents additional damage to the glandular parenchyma. In addition to antibiotics, analgesics and attention to good oral hygiene, massage of the parotid gland, warmth, use of chewing gum and sialagogues agents may be helpful in reducing the attack frequency. [2]

Parotid sialography is hallmark in the diagnosis of JRP [10] and also plays an important role as a therapeutic aid in the treatment of patients suffering from obstructive sialadenitis. While in our case, sialography was performed only once and proved beneficial. This treatment option seems to be effective as it helps clear the mucous plugs and cells in case of recurrent infection [2],[9] The reason for the improvement in symptoms and preventing the recurrence of attacks is due to the following reasons:

  1. Creating the patency of the duct during cannulation
  2. Antiseptic property of the dye (iodine based)
  3. Flushing action of the duct and gland which flushes out the debris and epithelial plugging in the duct and gland. In this case, patient followed for 2 years showed no recurrence of symptoms.



   Conclusion Top


Treatment of acute episode of JRP aims to deliver relief of symptoms and to prevent damage to the gland parenchyma. Analgesics and antibiotics have been found to be rapidly effective in relieving the pain and swelling. Treatment is conservative in the first instance, ultrasound is the appropriate initial investigation, and is usually supplemented by sialography.

Despite of the availability of newer advanced imaging techniques, sialography remains the most popular procedure for assessment of salivary gland disorders, due to its ease and affordability. The added therapeutic benefit that it prevents the recurrence of JRP is yet another advantage that warrants sialography to be considered as the principal investigation to be considered in such cases.

 
   References Top

1.Wang TC, Shyur SD, Kao YH, Huang LH. Juvenile recurrent parotitis. Acta Paediatr Taiwan 2006;47:297-302.  Back to cited text no. 1
    
2.Chitre VV, Premchandra DJ. Recurrent parotitis. Arch Dis Child 1997;77:359-63.  Back to cited text no. 2
    
3.Geterud A, Lindvall AM, Nylén O. Follow-up study of recurrent parotitis in children. Ann Otol Rhinol Laryngol 1988;97:341-6.  Back to cited text no. 3
    
4.Nahlieli O, Shacham R, Shlesinger M, Eliav E. Juvenile recurrent parotitis: A new method of diagnosis and treatment. Pediatrics 2004;114:9-12.  Back to cited text no. 4
    
5.Templer JW, Liess BD. Parotitis, E Medicine Specialties. Available from: http://emedicine.medscape.com/article/882461-overview  Back to cited text no. 5
    
6.Jain V, Mani NB, Singh M, Kumar L. Juvenile recurrent parotitis. Indian Pediatr 2000;37:1126-9.  Back to cited text no. 6
    
7.Miziara ID, Campelo VE. Infantile recurrent parotitis: Follow-up study of five cases and literature review. Braz J Otorhinolaryngol 2005;71:570-5.  Back to cited text no. 7
    
8.Galili D, Marmary Y. Spontaneous regeneration of the parotid salivary gland following juvenile recurrent parotitis. Oral Surg Oral Med Oral Pathol 1985;60:605-7.  Back to cited text no. 8
[PUBMED]    
9.Reddy SS, Rakesh N, Raghav N, Devaraju D, Bijjal SG. Sialography: Report of 3 cases. Indian J Dent Res 2009;20:499-502.  Back to cited text no. 9
[PUBMED]  Medknow Journal  
10.Bharti B, Parmar VR. Juvenile recurrent parotitis. Indian Pediatr 2001;38:311-2  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4]



 

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    Abstract
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   Case Report
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