juvenile recurrent parotitis by Rocio Mirella Sihuay Gutiérrez

Oral Surgery ISSN 1752-2471

CASE REPORT

ors_1042

91..94

Juvenile recurrent parotitis: a case report G. Gardner1, P. Fowlie2 & M. Macluskey1 1

Unit of Oral Surgery and Medicine, University of Dundee Dental School, Dundee, UK Department of Paediatrics, Ninewells Hospital, Dundee, UK

Key words: juvenile parotitis, parotid salivary gland, recurrent parotitis of childhood Correspondence to: Dr. Michaelina Macluskey Unit of Oral Surgery and Medicine University of Dundee Dental School Park Place Dundee DD1 4HR UK Tel.: 01382635989 Fax: 01382 425783 email: m.macluskey@dundee.ac.uk

Abstract Juvenile recurrent parotitis is a rare inflammatory disease characterised by intermittent sudden onset episodes of parotid swelling unrelated to eating. It is the second most common salivary gland disease in children, next to mumps. A case of this condition affecting a 2-year-old boy is reported and the literature reviewed.

Accepted: 5 May 2009 doi:10.1111/j.1752-248X.2009.01042.x

Introduction Juvenile recurrent parotitis (JRP) is a rare childhood inflammatory disease of the parotid salivary glands1. It is characterised by intermittent sudden onset episodes of parotid swelling unrelated to eating and may be accompanied by pain and fever. It can cause diagnostic difficulty, but the major clinical features that distinguish it from other causes of parotid swelling are the clinical presentation, the lack of an infective component and normal biochemistry2. Although a congenital or autoimmune factor is suspected, the exact aetiology is yet to be determined1,3. We present a case in a toddler and review the available published literature.

Case report A 2-year-old boy who was born and raised in Perthshire, Scotland, was referred to an oral and maxillofacial department by his dentist with acute pain and swelling of the right side of his face. The dentist had examined the child but found no obvious odontogenic explanation for his right-sided facial swelling. The painful Oral Surgery 2 (2009) 91–94. © 2009 The Authors Journal compilation © 2009 Blackwell Munksgaard

swelling of the right side of the face had developed during the previous week. This was the fourth such episode that the child had experienced beginning the previous year and lasting on average 1–2 weeks, resolving spontaneously. On the first occasion the unilateral swelling was assumed to be mumps. However, when this recurred, antibiotics were prescribed, to no effect. On examination there was a diffuse swelling of the right parotid gland that was tender to palpation with erythema of the overlying skin and an associated ipsilateral lymphadenopathy. The patient was apyrexic. On intra-oral examination, there was a wellmaintained dentition without evidence of caries or other pathology. There was no evidence of pus expressed from the right parotid duct on palpation of the gland (Fig. 1). Blood tests were requested including a full blood count, differential white cell count, paediatric antinuclear antibody and rheumatoid IgM levels. In addition, an ultrasound scan of the area was requested. A provisional diagnosis of JRP was made and second opinion sought from a consultant paediatrician. On 91

Juvenile recurrent parotitis

Gardner et al.

Figure 1 An intra-oral clinical photograph of the right parotid duct oriﬁce. The patient presented with pain and swelling of the right parotid gland. On intra-oral examination there was no evidence of a purulent discharge from the parotid duct or an odontogenic cause for the facial swelling.

that occasion the patient was asymptomatic and clinically there was no evidence of the right-sided facial swelling previously described (Fig. 2). There was no known family history of autoimmune disease and no notable past medical history in the patient was identified. There were no signs of systemic disease on examination. The haematological investigations were within normal limits with the exception of an elevated lymphocyte count of 6.0 ¥ 109/L. The patient was negative for anti-nuclear factor and his rheumatoid IgM was within normal limits at 9.5 IU/mL. The ultrasound scan reported that there was a patchy echotecture with multiple small rounded hypoechoic areas measuring up to approximately 2 mm in diameter (Fig. 3). These appearances suggested that there was punctuate, globular sialectasis without evidence of ductal dilation. A similar ultrasound appearance was found on the left parotid gland although this had never been symptomatic. The ultrasound appearances were thought to be in keeping with changes due to recurrent parotitis of childhood. On follow up 2 months later, the mother reported the there had been yet another episode, but on this occasion it was more protracted, lasting 3 weeks and more severe than previously. The symptoms were managed with analgesics and no further action was considered necessary at that time.

Discussion The incidence of JRP is unknown but it is thought to be a rare condition; however, it is the second most 92

Figure 2 Clinical photograph demonstrating the patient’s facial appearance between episodes of right parotid swelling. There is no evidence of facial asymmetry or lymphadenopathy between episodes of pain and swelling of the right parotid gland.

Figure 3 Ultrasound scan of the right parotid gland. The scan shows small round hopoechoic areas suggestive of sialectasis. Comparison views of the left parotid gland also show a similar echo texture. These ﬁndings would be in keeping with changes due to juvenile recurrent parotitis.

common salivary gland disease in children, next to mumps4. JRP is usually unilateral, but bilateral exacerbation can occur, with symptoms usually more prominent on one side1. It is usually associated with sudden onset swelling, pain, fever and redness that spontaneously subsides within 2 weeks2. JPR is usually evident by the age of 6 years, predominantly in male chilOral Surgery 2 (2009) 91–94. © 2009 The Authors Journal compilation © 2009 Blackwell Munksgaard

Juvenile recurrent parotitis

Gardner et al.

dren2,5, with a ratio of approximately 2:12. It has also been reported to have a biphasic age distribution2,5. The swelling can be unilateral or bilateral, and episodes can occur one to five times per year over a period of years and is associated with sialectasis1–3. Neither the submandibular or sublingual salivary glands are involved in the disease process. Progression into adult life has been observed but remission normally occurs with puberty3. The therapeutic approach is conservative as there is usually complete resolution with puberty3. Exacerbations are readily managed with sialogogic agents (e.g. sugarless sour candy/chewing gum), aggressive glandular massage and duct probing in order to promote ductal lavage and break up mucus plugs1. Antibiotics and steroids are of limited value and surgery is rarely necessary6. Since Sjögren’s syndrome can occur in the young, it must be included in the differential diagnosis1. Confusion arises from the similarity of clinical symptoms, the presence of sialectasis in both recurrent parotitis and Sjögren’s syndrome and reports of the occasional progression of recurrent parotitis in children into Sjögren’s syndrome. The absence of serum autoantibodies, however, generally rules out Sjögren’s syndrome. The aetiology of JRP remains an enigma7. JRP is generally associated with reduced salivary flow and non-obstructive sialectasis; however, it is not clear if sialectasis is the cause or rather the effect of the infection ascending from the mouth8. Theories on aetiology include recurrent infection, allergy, congenital/ structural defects, genetic factors and abnormal masseter muscle activity6,8–15. It is now known that the natural history of the condition is benign in the majority of cases and, therefore, conservative treatment is advocated1. Galili and Yitzhak16 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. Regeneration has been favoured as the preponderant mechanism. However, there are persistent cases12,14. The parotid sialogram and ultrasound2,3 are useful diagnostic tools. In addition, sialography also appears to have a therapeutic effect12,14,17,18 and can decrease the frequency of recurrences17. However, given the age of this child, ultrasound was an appropriate initial investigation12. More recently, diagnostic sialendoscopy has been reported to be a new procedure that can be used in children for reliable evaluation of salivary ductal disorders, with low morbidity19. Sialendoscopic dilation of the main parotid ducts has also been shown to be a safe and effective method for treating JRP18,19. More Oral Surgery 2 (2009) 91–94. © 2009 The Authors Journal compilation © 2009 Blackwell Munksgaard

aggressive forms of treatment have been used, including parotidectomy, duct ligation and tympanic neurectomy but with varying results4,14,20,21. More aggressive treatment would only be justified for adults with persistent problems14.

Conclusion In summary, we report a case of a toddler with RJP based on the clinical picture supported by the ultrasound findings report and lack of significant serological findings. This young boy will continue to be followed up, and if his symptoms persist, may benefit from sialography or the sialendoscopic dilation of the main parotid ducts.

References 1. Nahlieli O, Shacham R, Shlesinger M, Eliav E. Juvenile recurrent parotitis: a new method of diagnosis and treatment. Pediatrics 2004;114:9–12. 2. Leerdam CM, Martin HCO, Isaacs D. Recurrent parotitis of childhood. J Paediatr Child Health 2005;41:631–4. 3. Miziara ID, Sousa Campelo VE. Infantile recurrent parotitis: follow up study of five cases and literature review. Part 1. Braz J Otorhinolaryngol 2005;71:570–5. 4. Orvidas LJ, Kasperbauer JL, Lewis JE, Olsen KD, Lesnick TG. Pediatric parotid masses. Arch Otolaryngol Head Neck Surg 2000;126:177–84. 5. Sitheeque M, Sivachandran Y, Varathan V, Ariyawardana A, Ranasinghe A. Juvenile recurrent parotitis: clinical, sialographic and ultrasonographic features. Int J Paediatr Dent 2007;17:98–104. 6. Bernkopf E, Colleselli P, Broia V, de Benedictis FM. Is recurrent parotitis in childhood still an enigma? A pilot experience. Acta Paediatr 2008;97:478–82. 7. Kolho KL, Saarinen R, Paju A, Stenman J, Stenman UH, Pitkaranta A. New insights into juvenile parotitis. Acta Pediatr 2005;94:1566–70. 8. Ericson S, Zetterlund B, Ohman J. Recurrent parotitis and sialectasis in childhood. Clinical, radiologic, immunologic, bacteriologic, and histologic study. Ann Otol Rhinol Laryngol 1991;100:527–35. 9. Reid E, Douglas F, Crow Y, Hollman A, Gibson J. Autosomal dominant juvenile recurrent parotitis. J Med Genet 1998;35:417–19. 10. Hearth-Holmes M, Baethge BA, Abreo F, Wolf RE. Autoimmune exocrinopathy presenting as recurrent parotitis of childhood. Arch Otolaryngol Head Neck Surg 1993;119:347–9. 11. Cohen HA, Gross S, Nussinovitch M, Frydman M, Varsano I. Recurrent parotitis. Arch Dis Child 1992;67:1036–7.

Juvenile recurrent parotitis

12. Chitre VV, Premchandra DJ. Recurrent parotitis. Arch Dis Child 1997;77:359–63. 13. Mulcahy D, Isaacs D. Recurrent parotitis. Arch Dis Child 1993;68:151. 14. Galili D, Marmary Y. Juvenile recurrent parotitis: clinicoradiologic follow-up study and beneficial effect of sialography. Oral Surg Oral Med Oral Pathol 1986;61:550–6. 15. Ingervall B, Thilander B. Activity of temporal and masseter muscles in children with a lateral forced bite. Angle Orthod 1975;45:249–58. 16. Galili D, Yitzhak M. Spontaneous regeneration of parotid salivary gland following juvenile recurrent parotitis. Oral Surg Oral Med Oral Pathol 1985;60:605–7.

Gardner et al.

17. Wang TC, Shyur SD, Kao YH, Huang LH. Juvenile recurrent parotitis. Acta Paediatr Taiwan 2006;47:297–302. 18. Faure F, Froehlich P, Marchal F. Paediatric sialendoscopy. Curr Opin Otolaryngol Head Neck Surg 2008;16:60–3. 19. Quenin S, Plouin-Gaudon I, Marchal F, Froehlich P, Disant F, Faure F. Juvenile recurrent parotitis: sialendoscopic approach. Arch Otolaryngol Head Neck Surg 2008;134:715–19. 20. Gazi MI, Bhutta ZA. Sialectasis pediatrica recurrens. Acta Odontol. Pediatr 1987;8:21–8. 21. Geterud A, Lindvall AM, Nylen O. Follow-up study of recurrent parotitis in children. Ann. Otol. Rhinol. Laryngol. 1988;97:1–6.