Gut, 1979, 2, 1102-1108

Recurrent unilateral swelling of the parotid glandS. I. SULEIMAN', J. P. S. THOMSON, AND M. HOBSLEY

From the Department of Surgical Studies, The Middlesex Hospital and Medical School, London

SUMMARY The clinical features of 109 patients with recurrent unilateral parotid swelling (24 patientswith Sjogren's disease were excluded) have been analysed to explore the best system of management.The cause was definitely a parotid duct calculus in 36 patients, and evidence is presented that thesame diagnosis probably applied to another 59 patients. Features suggesting a diagnosis of calculusincluded age (greater than 29 years); duration of the attacks of pain (less than 24 hours); cessationof salivation on the affected side; and a spurt of saliva heralding the relief of symptoms. Onlythree patients in the definite calculus group (8.3 %) had no physical signs. However, had physicalexamination not included inspection and palpation of the parotid duct and its orifice from withinthe mouth 75% of the proven calculi would have been missed. The intraoral and anteroposteriorplain radiographs are likely to be helpful, and sialography even more so. A sialographic appearanceof a stricture in the main duct with proximal dilatation is usually due to a calculus. It would appearthat calculi are the cause of recurrent unilateral parotid swelling (after exclusion of Sjogren's disease)in an overwhelming proportion of patients with this symptom.

The first attack of swelling of the parotid glandis customarily managed by the family practitioner.The diagnosis usually made is mumps, especially ina child, and the patient is managed accordingly;should there be no more trouble there is no need toquestion this diagnosis. However, those who suffer arecurrence of the parotid gland enlargement, orthose who have a prolonged attack require referral.At hospital it will be noted whether or not the

symptoms and signs are bilateral or unilateral. Ifbilateral or other salivary glands are involved,particularly if the patient complains of dry mouthand eyes, he should be investigated for the possibilityof Sjogren's syndrome. If unilateral, and providedthat the swelling affects the whole gland and is not alump in the parotid region (Hobsley, 1970), thecondition may be referred to as chronic (or recurrent)unilateral parotitis. The following communication isconcerned with this group of patients; 24 patientswith Sj6gren's disease (benign lymphoepithelialdisease of the parotid gland) are excluded.

Methods

PATIENTSDuring the period 1967-78 133 patients withunilateral recurrent parotid swelling were referred to

Bowater Ralli Foundation Fellow.Received for publication 16 July 1979.

one of us (M. H.). All patients had a full history,examination and radiology, including sialography.When Sj6gren's disease was suspected, serum anti-bodies were studied (including specific salivary glandantibodies) and parotid secretion studies wereperformed by the technique described by Curry andPatey (1964). When parotid tissue had been excised,the histological material was reviewed. The casematerial was collected and analysed and the 24patients who were suffering from Sjogren's disease(benign lymphoepithelial disease) were excluded asthey form the material of another paper. The re-maining 109 patients are presented here.

Results

After reviewing all the evidence, the 109 patientswere grouped into the diagnostic categories shownin Table 1.

GROUP 1: PROVED CALCULI (36 patients)They were patients in whom a parotid duct calculuswas definitely proven by recovering the calculus. Ithad either passed spontaneously into the mouth, orbeen removed by operation.The male to female ratio was 21:15. The right

parotid gland was affected in 28 and the left in eight.The ages ranged from 29 to 69 years with a mean of50 5 years; 92% of the patients were aged between45 and 65 years.

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Recurrent unilateral swelling of the parotid gland

Table 1 Diagnosticgroups of 133 patients managedforrecurrent unilateral swelling oftheparotidgland (24patients with Sjogren's disease excluded)

Diagnosis No. ofpatients

1 Proved calculi 362 Presumed calculi 593 Parotitis secondary to other causes 34 Traumatic parotid duct stricture 25 Pending diagnosis 9Total 109

The presenting symptoms are shown in Table 2.The attacks lasted for less than a day in 69% ofpatients and for less than a week in all. Particularattention is to be paid to the fact that pain andswelling were intermittent rather than continuousand that the patients had frequently noticed a spurtof saliva heralding the relief of the symptoms.By the time they reached hospital only 18 had

parotid enlargement and the whole gland wasaffected; the rest had no swelling, although thefamily doctor had detected an enlargement weeksearlier. Stones were palpable along the parotid ductin 26 patients (72-2 %). In all these cases they werepalpable from within the mouth, and in three fromthe outer aspect of the cheek as well. The stomaof Stensen's duct was deformed in 11, and a turbiddischarge was expressible through the stoma insix. Three patients had no positive physical signs(8.3%).

Plain radiology, including an intraoral view(Hobsley, 1970) (Fig. 1) was performed on 35patients and reported as positive in 25 (71 4%).When the radiographs of 31 patients were reviewed,the success rate of the intraoral view was 22 (71 %),of the anteroposterior view, 17 (54'8 %) (Fig. 2),and of the lateral view only six (19-4%). Sialographyby Pattinson's (1969) technique was done in 34patients; filling defects within the duct system wereshown in 29 (85-3 %), dilatation of the main duct

Fig. 1 Technique ofobtaining theplain intraoral viewradiograph. The dentalfilm is held by thepatient againstthe buccal aspect of the cheek. (Reproduced bypermission from Hobsley, M. (1979), Pathways inSurgical Management. Edward Arnold, London.)

system proximal to a localised region of apparentnarrowing in four (11 8%) (Fig. 3), and a suddencut-off of the duct with no penetration of thecontrast medium beyond that point, in one.

Figure 4 shows the subsequent management of allthe cases. One patient developed a parotid abscessafter sialography; on incising the abscess one stonedischarged itself. Two weeks later a second calculus

Table 2 Symptoms of36 patients with proven parotid calculi

SymptomPatients Pain Swelling Cessation of Rush of Passing Unpleasant Pain In

Intermittent Continuous Intermittent Continuous salivation saliva stones taste ear

No. 33 3 32 6 24 22 6 2 4Percentage 91-7 8-3 88-9 16-7 66-7 61-1 16-7 5-6 111

Table 3 Symptoms of59 patientspresumed to have had calculi

SymptomPatients Pain Swelling Cessation of Rush of Passing Unpleasant

Intermittent Continuous Intermittent Continuous salivation saliva stones taste

No. 52 7 50 12 15 16 3 3Percentage 88-1 119 84-7 20-3 254 27-1 5 5

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S. L Suleiman, J. P. S. Thomson, and M. Hobsley

Fig. 2 Anteroposterior radiograph showing a calculus thatwas not shown on the lateral view.

passed through the fistula, the latter then healed andthe patient had no further trouble. Stomaductoplastywas performed when the symptoms were presistentand the stone was at or near the duct orifice. Re-stomaplasty was carried out if the stoma narroweddown. Superficial parotidectomy (Fig. 5) was per-formed if the symptoms were persistent and thestone was far away from the stoma, or the re-stomaplasty had failed. After superficial paroti-dectomy all patients developed varying degrees offacial paresis, lasting for a few weeks in the majority

of patients and up to nine months in two. One patienthad a minor residual paresis of the upper lip for upto one year after operation: this was not noticeablein repose, and the patient did not complain of it.Another patient had a second intraoral operation toremove the parotid duct remnant containing a tinycalculus.

GROUP 2: PRESUMED CALCULI (59 patients)In this group (Fig. 6) a presumptive diagnosis ofcalculus was made because of evidence that was

F-ig. P3Jarotid sialogram showing a localised area of duct narrowing with dilatation proximalto it. A stone was laterfound in the duct oftheparotidectomy specimen at the site oftheapparent narrowing.

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Recurrent unilateral swelling of the parotid gland

suggestive, but never confirmed by recovery of astone. The evidence was one or more of the followingfeatures: (1) a definite past history of passing parotidcalculi (three patients); (2) palpation of a stone withinthe duct (34 patients); (3) radiographic appearancesdiagnostic of a stone in 44 patients (18 positive onplain radiographs, all 44 on sialography; (4) histo-logy of a parotidectomy specimen reported astypical of the large duct sialectasis produced bya stone (seven patients).

In 22 patients features (2) and (3) coexisted, whileall seven patients with histological evidence had botha stone that was palpable and radiological evidence.All 34 palpable stones were felt from within themouth and three of them were also palpable fromthe external aspect of the cheek. In seven patientshistological material was obtained. The sectionsshowed microcalculi, dilatation of the ducts, andperiductal inflammation. No stones were found bythe surgeon; but radiographs of two out of threeexcised parotid specimens examined showed localisedcalcification.The symptoms of the 59 patients are shown in

Table 3: it is evident that they experienced sharppain in relation to meals, with cessation and rush ofsaliva in many. These features were very suggestiveof calculi being the underlying cause. Moreover, theoutcome of the management (Fig. 6) was in favourof calculi, as the attacks stopped spontaneously inmost of the patients, presumably after a calculushad been pass-.d.

GROUP 3: PAROTITIS SECONDARY TO

ASCENDING INFECTION DUE TO OTHER CAUSES(three patients)In this group of patients there was an oral identifiablecause of recurrent parotitis. In two, the attacksstarted immediately after a tooth extraction, whilethe third patient was in the terminal stages of dissem-inated lymphosarcoma with poor mouth hygiene. Inall three patients the condition was apparentlyprecipitated by ascending infection and progressed toabscess formation in the parotid gland. Surgicaldrainage was required in one patient: in the othertwo, pus was noted to be draining from the orificeof Stensen's duct, and conservative management

Fig. 4 Management of36 patients withparotid calculi.

+ 9

12+ 13

25rl|5uperf icial parotidectomy|

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S. I. Suleiman, J. P. S. Thomson, and M. Hobsley

succeeded. Once the infection was controlled, theyremained symptom free. Subsequent sialographywas normal in two but the patient with lympho-sarcoma died without a sialogram.

GROUP 4: TRAUMATIC PAROTID DUCTSTRICTURE (two patients)These had lacerations of the face. On sialography aparotid duct stricture was shown in both. One hadextravasation of the contrast medium at the site ofthe stricture and therefore required a superficialparotidectomy. The other responded to dilatation ofthe duct.

GROUP 5: DIAGNOSIS PENDING GROUP (ninepatients)In this group no final diagnosis has been made.

Seven of the nine patients had normal radiologyincluding sialography. Of these seven, two had a

history suggestive of calculi with a definite story of agush -of saliva, after which they have remainedsymptom-free for more than four years. It is possiblethat they had passed tiny calculi. Four of theremainder had recurrent attacks of intermittent pain

Fig. 5 Superficialparotidectomy specimen. The duct isfollowedasfarforwards aspossible, it has been split opento reveal the calculus within.

and swelling of the parotid gland in relation tomeals, but no history of cessation or gush of saliva;their parotid gland enlargement was never confirmedin hospital. They have now been symptom-free formore than one year, and it is possible that they, too,had had parotid calculi. The remaining patient was awoman aged 40 years who had positive parietal celland thyroid antibodies and could have been a case ofSjogren's disease. She remained symptom-free fornine months after our investigation and thenemigrated. The last two patients of this group hadmultiple strictures in their parotid ducts on sialo-graphy. Their symptoms were so severe that theyrequired superficial parotidectomy, which curedthem. The histological appearances were of chronicinflammation without the large duct dilatationsuggestive of stone or any features indicative ofSjogren's disease, so the aetiology remains unknown.

Discussion

While there have been some reports of recurrentswellings affecting all the salivary glands-forexample, Rose (1954) and Patey (1965)-many haveconcentrated on recurrent parotid gland enlargement(Payne, 1940; Pearson, 1961; Maynard, 1965).The present series is the only one devoted solely tounilateral recurrent swelling of the parotid gland,and the conclusions drawn from this study of 109patients should be worthwhile.

It is clear that parotid stones are a real rarity inthe group below 30 years, so recurrent enlargementof the parotid gland in that age group is most un-likely to be caused by a calculus.The typical history in patients with a parotid

calculus is of more or less intermittent pain andswelling, usually with meals or when brushing theteeth. The feature of brief attacks, lasting forminutes or hours, while often encountered, is not asdominant as it is in patients with submandibularsalivary gland stones, because the submandibularduct is wider, the stone dislodges more easily, andrelief of pain is faster (Thomson and Hobsley, 1973).Thus the attacks often last a day, rather than hours.Nevertheless, the distinction from the periods ofweeks of dull aching in Sjogren's disease remainsfairly clear cut. The patient may notice that theejection of saliva ceases on the affected side and thata gush or spurt of saliva heralds the relief of hissymptoms.With regard to radiology, the value of the intra-

oral view is clearly shown and was formerly stressedby Hobsley (1970), and the anteroposterior view alsohelps. Sialography in expert hands can be expectedto be positive in most cases.However, the fact that a small stone may not

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Recurrent unilateral swelling of the parotid gland

reveal itself as a filling defect is as true here as in thebiliary and urinary tracts. The contrast mediumshould therefore be as thin as possible. Even so, themedium may overwhelm a tiny stone which, if it hascaused obstruction, expresses itself as an apparentarea of narrowing with a sudden dilatation of theduct system proximal to it. These appearances areoften wrongly interpreted as indicating a stricture,but in all four patients in whom they were present, astone at that site was subsequently proved. A suddencut-off of the duct with no contrast medium goingfurther is a rare expression of the presence of acalculus.A group of patients referred to a surgeon with a

special interest in salivary glands may show biastowards the severe cases, yet when the groups ofproved and presumed calculi are scrutinised (95patients) it is clear that 46 patients (48A4%) curedthemselves by passing their stones. Stomaducto-plasty, a minor operation (Hobsley, 1978), wassuccessful in 10 out of 29 cases (34.5 %). Superficialparotidectomy was necessary in 32 patients (33 7 %).Although the superficial parotidectomy is known to

59Patients withsymptomssuggestiveof stones

be difficult in such cases (Conley, 1975), withpatience during the operation and insistence onfollowing the duct to its termination, very goodresults are to be expected. Certainly we do notadvise this procedure as one for the occasional paro-tidectomist. Results in the group of patients withpresumed calculi reflect the milder nature of themajority of parotid duct calculi and the fact that theymay either pass, or trouble the patient very little.The fact that in seven patients with presumed calculino stones were detected by the surgeon in the paro-tidectomy operation might have been related to thelag of eight months between sialography showingfilling defects and the operation: perhaps the stoneshad passed, leaving the gland a seat of sialadenitis,a phenomenon well known with calculi at other sites.

Judging by the patients referred to us for a secondopinion from other centres, the clinical and radio-logical examination of this problem is often incom-plete. Oral inspection and palpation of the termina-tion of Stensen's duct were frequently omitted, plainradiology often did not include the anteroposteriorand intraoral views, and sialography was rarely

?2)stic

8raphy 22)

Fig. 6 Management of596 patientspresumed to have had

Mild symptoms calculi.

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1108 S. L Suleiman, J. P. S. Thomson, and M. Hobsley

performed. Without these examinations, 75 % of ourpatients with stones would have remained incor-rectly labelled as 'recurrent parotitis-cause ob-scure'.

Turning to the patients with ascending parotitissecondary to some other cause; tooth extractiondepresses mastication and the flow of saliva isreduced. Poor mouth hygiene in debilitated patientsis a well known cause for ascending parotitis, but inthe 109 patients only one (the patient with dissemi-nated lymphosarcoma) fell into this category. Thisreflects improved oral, dental, and nursing care.The parotid duct stricture which improved on

dilatation justifies this approach. The second stricturehad much induration around it, and on sialographyit was tight with extravasation of the contrastmedium in parotid tissue. The idea of a plasticoperation on the parotid duct from the outer aspectof the cheek was not entertained because (1) the twobranches of the facial nerve running along with theparotid duct could have been easily injured amidthe fibrous tissue; (2) even though the proceduremight have been technically feasible, the stricturemight recur. Therefore superficial parotidectomy,with the main nerve trunks followed forward andpreserved, was performed and produced effectivepermanent cure.We feel that parotid follow-up should be pro-

tracted. No patient is discharged from the clinic:even symptomless patients are seen annually.Therefore we have been left with a small group ofpatients who are now symptom-free for more thanone year: while a final diagnosis has yet to be made,our provisional diagnosis, that a calculus hadpassed, is probably correct.

In only two out of the 109 patients was the aetio-logy of the chronic parotitis obscure. We would liketo stress the fact that, if the evidence we have givenfor our diagnosis of 'presumed calculus' is accepted,then causes of recurrent parotid gland swelling were

obscure in only 1 8 %. Therefore, the much largerproportions of patients in other series assigned tosuch obscure causes as congenital abnormality of theparotid gland, stricture at the termination of theparotid duct, hypersensitivity to mumps virus, orallergy (Smith, 1953; Rose, 1954) are unlikely to becorrect: many of these were probably caused bystones.

ReferencesConley, J. (1975). Salivary Glands and the Facial Nerve,

p. 39. Georg Thieme: Stuttgart.Currey, R. C., and Patey, D. H. (1964). A clinical test

of parotid function. British Journal of Surgery, 51,891-892.

Hobsley, M. (1970). Affections of the salivary glands andtheir management. Annals of the Royal College ofSurgeons, 46, 224-236.

Hobsley, M. (1978). In General Surgical Operations, pp.348-350. Edited by R. M. Kirk. Churchill Livingstone:London.

Maynard, J. D. (1965). Recurrent parotid enlargement.British Journal of Surgery, 52, 784-789.

Patey, D. H. (1965). Inflammation of the salivary glandswith particular reference to chronic and recurrentparotitis. Annals of the Royal College of Surgeons, 36,26-44.

Pattinson, J. N. (1969). In Operative Surgery: Head andNeck, 2nd edition, pp. 167-169. Edited by C. Rob andR. Smith. Butterworths: London.

Payne, R. T. (1940). Pneumococcal parotitis. BritishMedical Journal, 1, 287-292.

Pearson, R. S. B. (1961). Recurrent swellings of theparotid gland. Gut, 2, 210-217.

Rose, S. S. (1954). Clinical and radiological survey of192 cases of recurrent swelling of the salivary glands.Annals of the Royal College of Surgeons, 15, 374-401.

Smith, M. (1953). Familial incidence of sialectasis.British MedicalJournal, 2, 1359.

Thomson, J. P. S., and Hobsley, M. (1973). Parotid ductcalculus. Proceedings of the Royal Society of Medicine,66, 352-353.