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Recurrent Parotitis Archives of Disease in Childhood 1997;77:359–363 359 Arch Dis Child: first published as 10.1136/adc.77.4.359 on 1 October 1997. Downloaded from REGULAR REVIEW Recurrent parotitis V V Chitre, D J Premchandra Recurrent parotitis is defined as recurrent glands in patients with unilateral disease.4 This parotid inflammation, generally associated with suggests that those with low salivary flow rates non-obstructive sialectasis of the parotid might be predisposed to suVer from repeated gland.1 Also known as juvenile recurrent ascending infections. This relation to salivary parotitis,23 this disease is characterised by flow rates could also explain the familial recurring episodes of swelling and/or pain in tendency that has been reported.389 the parotid gland, usually accompanied by The histological picture includes lym- fever and malaise. It usually aVects children, phocytic infiltration around the intralobular but may persist into adulthood.(Arch Dis Child ducts, and Patey and Thakray proposed that 1997;77:359–363) It is a rare condition, and its aetiology this lymphocytic infiltration damages the duct remains an enigma. Its natural history is wall reticulum, allowing extravasation of secre- tions into the gland parenchyma, and thus variable, and in adults more aggressive inter- 10 vention is often needed. In addition, there is no exacerbating the inflammation. This was sup- 41112 satisfactory explanation for its usual tendency ported by Hemenway and others. to resolve spontaneously after puberty. All this The fragmentation of connective tissue sup- has resulted in considerable uncertainty con- porting the intralobular ducts was also impli- cerning its appropriate management. This cated by these authors in the production of the review was undertaken to collate all the characteristic punctate sialectasis. They pro- information available on this uncommon and posed that the dye used for sialography distressing condition. Diagnosis and manage- ruptured the already weakened duct walls, pro- ment of this condition is also discussed. ducing the appearance of punctate sialectasis. This theory therefore neatly explained the presence of sialectasis in the absence of http://adc.bmj.com/ Aetiology demonstrable distal obstruction. Its cause remains unknown despite several The situation, however, is not as simple. studies. Though the aVected glands demon- Punctate sialectasis is seen in totally asympto- strate sialectasis of the distal ducts, there seems matic glands of aVected individuals in up to to be no element of obstruction in most cases. 70% of cases.13 Further, detailed histopatho- Several theories of causation have been put logical studies have confirmed the presence of forward over the years. duct dilatation and cystic cavities associated on September 23, 2021 by guest. Protected copyright. Traditionally, ascending infection from the with a chronic inflammatory process.214 And oral cavity has been considered the primary more recently, ultrasonography consistently event, with sialectasis being a secondary revealed hypoechoic areas that corresponded change. Maynard proposed that the recurrent to the punctate sialectases demonstrated by episodes of parotid swelling was the end result 15 of a sequence of events4: sialography. It therefore would appear that x There is first a low grade inflammation of the sialectases are actually present, and are not the gland and duct epithelium, possibly caused merely artefacts produced by the radio-opaque by a low salivary flow rate due to dehydration dye. and debility. As long ago as 1945, Hamilton Bailey x This results in distortion and stricturing of proposed the presence of a congenital abnor- the distal ducts, and metaplasia of the duct mality of the ductal system, and drew a parallel 16 epithelium. with bronchiectasis. He pointed out that x The metaplasia results in excessive mucus bronchiectasis could be congenital as well as Department of secretion. acquired, and in both cases, the end result was Otolaryngology, James These changes, along with possibly a further secondary infection of the bronchioles and Paget Hospital, Great reduction in salivary flow rate, then predispose alveoli. He has been subsequently supported by Yarmouth, Norfolk to recurrent parotid inflammations. several others.1–3 14 According to this argument, V V Chitre D J Premchandra A reduced salivary flow rate may result from punctate dilatation of the small distal ducts glandular damage caused by the primary infec- results in stasis and ascending infection, giving Correspondence to: tion. However, it may be a primary factor as rise to the recurrent acute attacks. Though no Mr V V Chitre, Flat 6, well. Several workers showed low salivary flow evidence has so far emerged in favour of a con- Rosemont, 80/81 Mount 4–7 Ephraim, Tunbridge Wells, rates, and the significant finding was that the genital abnormality, it is still possible that Kent TN4 8BS. flow rate was reduced in even the unaVected genetic factors may prove important. 360 Chitre, Premchandra Given this state of incomplete knowledge, The number of attacks vary individually, Arch Dis Child: first published as 10.1136/adc.77.4.359 on 1 October 1997. Downloaded from the present consensus is towards a multifacto- with attacks every three to four months being rial approach. Thus Kono and Ito concluded the commonest pattern.13Mandel and Kaynar that the sialectasis is both the cause and the state that attacks tend to occur one to five times result of recurrent parotitis.14 Their histological a year.23 The frequency rate peaks during the studies detected dilated cavities consistent with first year at school, but otherwise remains fairly true sialectasis, as well as a few areas of constant for each individual until puberty. extravasated dye which mimicked sialectasis on After puberty, the symptoms usually subside, the sialogram. Similarly, a detailed study of and may disappear completely.1–4 14 17 25 clinical, radiological, immunological, bacterio- Geterud et al reported that 84% of their logical, and histological findings in 20 aVected patients had recovered by the time they children concluded that the cause was probably attained puberty.1 Further, another 8% of their a combination of a congenital malformation of patients were considered cured by the time portions of the salivary ducts and infections they reached the age of 22. Galili and Yitzhak ascending from the mouth after dehydration of proposed two possible ways by which this the aVected children.2 However, it must also be spontaneous recovery might occur: total atro- acknowledged that juvenile recurrent parotitis phy with consequent lack of symptoms, or can occasionally occur without sialectasis.11417 regeneration of the gland from surviving ductal One child has suVered repeated attacks of system.29 The authors favoured regeneration as parotitis secondary to repeated chewing of the the likely mechanism. However, there are also Stenson’s duct orifice.18 persistent cases.1–3 13 20 23 29 30 The actual Many associations have been proposed in the proportion of ‘persisters’ is debatable, though past; these include immunodeficiency, allergy, most researchers agree that the numbers are upper respiratory infections, mumps, etc. None small.1341114 of these, however, has been conclusively shown The painful swelling is usually associated to have any bearing on this disease.2 Friis et al19 with fever.2 There is typically an absence of pus and others14 17 proposed an autoimmune ori- despite the pyrexia and malaise,3 though gin, but the self limiting nature of recurrent Geterud et al noticed a few drops of mucopu- parotitis and the absence of detectable auto- rulent secretion on palpating the parotid antibodies makes this unlikely.2131420 There gland.1 The swelling lasts from several days to have been reports of sensitivity to upper respi- two weeks,23 and resolves spontaneously, inde- ratory tract infections21421; these infections pendent of any treatment. may set oV attacks of sialadenitis merely by causing dehydration in a child with sialectasis.2 Investigations The higher rate of secretion in the sub- (1) SIALOGRAPHY mandibular gland compared with the parotid The mainstay of diagnosis is sialography, gland may protect it from infections2; also, the though its role is now becoming secondary to submandibular gland secretion is relatively ultrasonography. In 1971, Hemenway classi- richer in mucus, which has antiseptic proper- fied sialectasis into (a) large duct sialectasis, due http://adc.bmj.com/ ties. In fact, no report was found in the to obstruction of the main duct, and (b) small literature implicating the submandibular duct/punctate/terminal sialectasis, in which there gland. are multiple small round opacities at the termi- nation of the smaller ducts.11 Later, Gates31 and Clinical features Noyek et al32 reviewed the classification of Recurrent parotitis presents as a recurrent punctate sialectasis, and further classified this painful swelling during mastication and/or into (a) pruned tree appearance—in the early swallowing.22 The disease usually starts in a stages, the ducts are stretched, tapered, and on September 23, 2021 by guest. Protected copyright. child between 3 and 6 years of age,131323 but decreased in number; (b) punctate sialectasis— earlier and later occurrence has been the peripheral ducts demonstrate punctate observed.214172425Ericson et al followed up 20 dilatation <1 mm diameter, and the intraglan- children and the age of onset in their series dular ductal system is stretched and tapered; ranged from 3 months to 16 years.2 (c) globular
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