Case report: report of a rare case of juvenile recurrent parotitis and review of literature.
Recurrent parotitis (RP) is defined as recurrent parotid inflammation, generally associated with non-obstructive sialectasis of the parotid gland. It is a rare condition, and its aetiology remains an enigma [Baurmash, 2004; Shkalim et al., 2004; Kolho et al., 2005; Fazekas et al., 2005]. It is also known as juvenile recurrent parotitis (JRP) [Chitre and Premchandra, 1997], infantile recurrent parotitis [Miziara and Campelo, 2005] or recurrent acute parotitis [Nahlieli, 2004].
The disease is characterised by recurring unilateral or bilateral episodes of swelling and/or pain in the parotid gland, usually accompanied by fever and malaise [Chitre and Premchandra, 1997; Orvidas et al., 2000]. There may be discharge of muco-purulent saliva through the duct upon compression of the parotid gland and often the salivary secretion is reduced [Geterud, et al., 1988]. Between the acute episodes of inflammation, the children can have intervals of months to years without symptoms [Huisman et al., 2001]. The age at onset usually ranges between 2 and 7 years of age, but earlier and later occurrence has been observed [Huisman et al., 2001; Motamed et al., 2003]. Leerdam et al  have shown a biphasic age distribution with peaks at 2-5 years and at 10 years of age. Boys are more often affected than girls. At puberty, the frequency of inflammatory episodes usually decreases and eventually subsides completely by adulthood [Motamed et al., 2003].
Presentation: A 14 year-old boy presented with a complaint of swelling associated with pain on the right side of the face 2 days. The swelling was associated with pain, discomfort and salty taste in his mouth.
History: The patient gave a history of recurrent swellings on the right and left side of face alternatively since one year of age, at least twice a year. The symptoms subsided on taking antibiotics and analgesics. A hypertrophic scar was noticed in front of the tragus on left side of face corresponding to the extraoral incision and drainage since 8 years of age for the same problem (Fig. 1b).
[FIGURE 1 OMITTED]
Examination: A right parotid swelling was noticed which was roughly oval in shape measuring 3 x 4 cm in diameter, and the skin over the right cheek was stretched (Fig. 1a). On palpation it was firm in consistency, tender and febrile. The submandibular lymph nodes were palpable bilaterally, soft-to-firm in consistency, tender, and mobile. Intraoral findings include thick ropy saliva and clear yellow fluid was expressed on milking the right parotid gland.
Provisional and Differential Diagnosis: Provisional diagnosis of recurrent parotitis was considered and differential diagnosis included:
* Bacterial sialadenitis,
* Sjogren's Syndrome,
* Tubercular sialadenitis.
Investigations: The complete blood picture revealed Hb% 11.9 gm/dl, WBC count 7,200 /[micro]L, differential count of N34, L61, E3, M2, B0, ESR--22 mm/hr and PCV of 35.8%. Peripheral smear showed normocytic, normochromic blood picture with few macrocytes. Serum IgE and IgM levels were within the normal range. Mantoux test was negative and no abnormality was detected in chest radiograph. Salivary flow rate for unstimulated and stimulated saliva was 0.26 ml/min and 0.45 ml/min respectively suggestive of salivary gland hypofunction. Culture of saliva from the parotid ducts was positive for coagulase negative streptococci and was sensitive to cloxacillin, erythromycin and amikacin.
Ultrasonogram of the right and left parotid gland revealed multiple hypoechoic areas and heterogeneous distribution of internal echoes suggestive of inflammatory changes and dilation of acini in both glands (Fig. 2). Sialography was performed using Iopromide (ultravist-300) as a contrast media for both right and left parotid gland after the clinical symptoms subsided. Sialography revealed dots or blobs of contrast media distributed throughout the gland, an appearance known as 'sialectasis' caused by the inflammation of glandular tissue producing dilation of terminal duct and sac-like acini with normal main duct suggestive of sialadenitis (Figs. 3 and 4).
[FIGURE 2 OMITTED]
[FIGURE 3 OMITTED]
[FIGURE 4 OMITTED]
Based on the history given by the patient, clinical picture, laboratory investigations, ultrasonogram and sialographic appearance, a final diagnosis of Juvenile Recurrent Parotitis (JRP) was given.
Treatment and Follow-up
Antibiotic Dicloxacillin 500 mg tid for 7 days following culture and sensitivity and analgesics, a combination of Diclofenac 50 mg and Paracetamol 500 mg tid was prescribed for 10 days. Sialography was performed twice at an interval of 6 months for glandular lavage that helps to clear the mucous plugs that form during the acute phase. The treatment seems to be effective as there has been no recurrence of parotitis for 18 months.
JRP is a rare disorder of childhood characterized by repeated episodes of non-obstructive parotitis.
Aetiopathogenesis: Although numerous pathogenic theories have been considered, including congenital malformations of the parotid duct, systemic immunologic diseases such as hypogammaglobulinaemia, IgA and IgG deficiency, primary or secondary infection, and allergic or hereditary factors, the aetiology of JRP remains unknown [Huisman et al., 2001; Sitheeque et al., 2007]. Because of its self-limiting nature, gender predilection favouring boys, and absence of autoimmune antibodies indicate that autoimmunity is a less probable cause of JRP. Reid et al.  found a pattern of autosomal dominant inheritance with incomplete penetrance, suggesting that, at least in some cases, genetic factors may be involved. Ericson et al.'  found no evidence of allergy as an aetiological cause for JRP. One of the most probable causes of JRP is retrograde infection, mostly during dehydration of the child, as occurs in upper respiratory tract infection, fever, diarrhoea etc. Dehydration causes a decrease in salivary secretion of the parotid gland resulting in stasis and decreased clearance thus providing a breeding ground for bacterial multiplication and infection [Nahlieli et al., 2004; Miziara and Campelo, 2005].
Clinical Presentation: The most pathognomonic sign is wide opening of the Stensen's papilla with plaque exudates. A firm mass can be felt when the symptomatic gland is palpated [Nahlieli et al., 2004]. JRP has to be differentiated from mumps, caused by a paramyxovirus, accompanied by systemic manifestations such as fever, malaise, headache, and chills, whereas in JRP, the symptoms are usually more local to the parotid gland, with fever developing occasionally. Moreover 90% of the population is provided with mumps vaccine, thus gaining immunity for life. Other conditions such as Sjogren's syndrome, pneumoparotid, lymphoma, and recurrent parotitis associated with acquired immune deficiency syndrome can be also taken into consideration when making the differential diagnosis [Nahlieli et al., 2004].
Several authors have advanced the possibility that JRP is a precursor of adult Sjogren syndrome (SS), with xerostomia as a presenting symptom. This concept appears to contradict the reports by Ericson et al  and Galili and Yitzhak,  who suggested that JRP abates when children enter adulthood [Sitheeque et al., 2007].
Diagnostic Aids: Sialography is considered the most important and most reliable diagnostic test of JRP, however, ultrasound has been increasingly used for diagnosis and follow-up as well [Miziara and Campelo, 2005]. Sialographic studies have shown a combination of sialectasis, strictures, dilations, and kinks. In most of the cases (73%), the sialographic findings were bilateral [Nahlieli et al., 2004].
Ultrasound is superior to sialography in the diagnosis of sialectasis. Ultrasonographic examinations demonstrate multiple small hypoechogenic areas and punctate calcifications, corresponding to the sialectases demonstrated on sialography [Nahlieli et al., 2004]. MRI and sialoendoscopy have proved to be helpful in the diagnosis of JRP [Nahlieli et al., 2004]. Histopathological examination of the diseased gland reveals lymphocyte infiltrate that tends to form lymphoid follicles and small ductal dilations [Nahlieli et al., 2004; Miziara and Campelo, 2005].
Treatment: The aim of the treatment is to stop the recurrent infection of the glands and to prevent irreversible damage in the parotid glands. Various studies have suggested that the symptoms usually subside, and may disappear completely after puberty [Chitre and Premchandra, 1997; Bhattarai and Wakode, 2006]. Geterud et al.  reported that 84% of their patients had recovered by the time they attained puberty and another 8% were cured by the time they reached the age of 22. Galili and Yitzhak  proposed two possible ways by which this spontaneous recovery might occur: total atrophy with consequent lack of symptoms, or regeneration of the gland from surviving ductal system.
Treatment of the acute episode 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. Cohen et al.  recommended low-dose antibiotic cover or prophylactic administration early in an attack to prevent additional damage to the glandular parenchyma. No preventive therapy against JRP has been available to date.
Nahlieli et al.  diagnosed and treated 21 cases of juvenile recurrent parotitis with a combined endoscopic approach. The treatment composed of lavage with 60 ml of normal saline, ductal dilatation with saline pressure or balloon dilation followed by hydrocortisone (100mg) injections via the endoscope into the gland. The treatment used lavage in the ductal system and the sialectases from plaques and to dilate strictures [Nahlieli et al., 2004]. Steroids may reduce swelling, but will not prevent recurrences [Chitre and Premchandra, 1997]. Bailey recommended duct cannulation and lavage with 1% merbromin [Bailey, 1945]. In our case sialography was carried out twice at an interval of 6 months that proved beneficial. Following sialography the patient had no further episodes of parotitis. This could be attributed either to the sialography, which may help in clearing the mucous plugs formed during the acute phase, or it could be because the child has attained puberty.
Other treatment modalities include use of sialogogues to increase salivary flow, encouragement of fluid intake, massage, and duct probing and dilation. Other treatment modalities mentioned in the literature are invasive, such as duct ligation, parotidectomy, and tympanic neurectomy [Chitre and Premchandra, 1997; Nahlieli et al., 2004].
Although JRP is self-limiting and may completely regress at puberty, paediatric dentists should be aware of this rare condition and should be able to diagnose and treat it. Dentists should have proper knowledge of the role of sialography and ultrasonography in the diagnosis and as well as the overall management of patients with JRP.
The authors would like to acknowledge Dr. H. N. Shama Rao, Principal, M. S. Ramaiah Dental College and Hospital, Bangalore for his guidance and support.
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S.Sujatha, N. Namita, N. Raghav, D. Devaraju, G. Shridevi
Dept. of Oral Medicine, Diagnosis and Radiology, M. S. Ramaiah Dental College and Hospital, Bangalore, Karnataka, India.
Postal address: Prof. S. Sujatha Reddy Dept of Oral Medicine, Diagnosis and Radiology, M.S. Ramaiah Dental College & Hospital, msrit post, New Bel Road, Bangalore-560054, India
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|Author:||Sujatha, S.; Namita, N.; Raghav, N.; Devaraju, D.; Shridevi, G.|
|Publication:||European Archives of Paediatric Dentistry|
|Article Type:||Case study|
|Date:||Dec 1, 2009|
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