RECURRENT PAROTITIS JOHN
A.
BIGLER,
M.D. ""
CLASSIFICATION OF PAROTID INFLAMMATlONS
. I.@bLl\fMAll0N. .oLthe....I2g,JQ!!Q,..gI~I!gsmay_ .berSlughly ...~ijvided into four groups: (1) n. mmps, a specific type of infection that is contagious; (2) spppura.1iYe . . par.otitis, a'l.-iflfiam,matOI¥...re.action .Jhltt..usually occurs in tlli:.!!e.biIitated. J!J!!:l JlliL~ak; it may follow surgical procedures, occurs during the course of the infectious diseases, and occurs i~_w..bQrn.iofants; (3) o~structive parotitis O.Gc:;,urs lram.the irrita_~!9!!. of .. ::t...• calqIlps, and (4) r~c_u!'!s!!:lt. parotitis, ~I} jp.flammation of the par~tids of o~~~~r~. t!ti919.gY.. . .which . occurs .in otherwise he.althy inaividu~lumd_.:whicb ruos a benign course. Little discussion is necessary on the subject of mumps. A rise in temperature mayor may not be present and a leukopenia is usually present. It should b~_~'!!!p'~~~iz~g_!hgLJlefru:e. a.[l111I11.ps paJ:"0titis. is diagnosed one should make pressure over the infected glandario' null{ alo~K._SJ;ensen,) g1,1c::f1()·see:~Jlc£p·iis -exu~es·fr~.m· the ductQP~i1Lug. Often no saliva can be seen. If pus is present then it can be assumed that it is not mumps that one is dealing with. Many of the cases of recurrent parotitis have been erroneously diagnosed as mumps from one to three times. Suppurative parotitis fortunately is very rare in children. It is a very serious disease with a reported mortality of 30 to 45 per cent. 1 Surgical incision with drainage is usuall~ n<:.~'!y. As yet there have Been msuffiClent reports on the use 0 sulfonamides and penicillin in these cases. Certainly they should be used and in large dosage. Suppurative parotitis of the newborn is not uncommon and apparently has a low mortality. Sanford2 reported five cases treated with sulfonamides and incision for drainage in four cases with recovery in all. This type of parotitis is usually due to the Staphylococcus aureus although other organisms may be the infecting agent. It is felt that most infections ascend through the duct and are not blood-borne. The condition is usually unilateral but it may be bilateral. A calculus in the parotid gland or duct causing obstructive parotitis is extremely rare in infants and children. There is an acute onset of the swelling with usually severe to excruciating pain. Suppuration of die gland mayor may not occur. Removal of the calculus results in a cure. From Children's Memorial Hospital, Chicago, Illinois . .. Associate Professor of Pediatrics, Northwestern University Medical School, Chicago; Medical Director and Assistant Chief of Staff, Children's Memorial Hospital; Member of the Staff of the Lake Forest and Highland Park Hospitals.
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JOHN A. BIGLER
Recurrent parotitis is apparently more common in infants and children than one would suppose from the few reports in the literature. During the past several years I have observed about sixty cases. Many of these had at the time of the first, second or third occurrence of the swelling been diagnosed as mumps by the family or a physician . . As the infection is a rather benign one and from outward appearances is indistinguishable from mumps it is not uncommon to have a child experience more than one attack before the family seeks medical aid for an explanation. I have never seen any of the salivary glands affected except the parotids. ~ECURRENT PAROTITIS: CLINICAL AND LABORATORY FINDINGS
Although the average age at the time of the first swelling was about three to four years, cases were observed from the age of eight months to the twelfth year. The latter age level is undoubtedly due to a practice limited to pediatrics and cases observed in a clinic where the upper age of admission is the thirteenth birthday. However, four cases have also been observed in adults. Swelling is sudden in onset as it is with mumps and it is usually the first noticeable symptom. The whole gland is involved in nearly all cases. The swelling is rarely as extreme as it often becomes in mumps and it has a nodular, firm feel with little if any edema. Although redness of the skin may occur it is not usual. The swelling may be bilateral or unilateral and recurrences may involve one side and then the other side. Usually, though, recurrences involve the same glilOd each time. Fluctuation is so rare that it was observed in only one patient. The maximum swelling occurs most commonly within the first forty-eight hours. Some change in size may occur during the course of the swelling. The regional. lymph Ilodes ate...llOt..mlarged. While the submaxill~.g!a,1!a~a.r.t fre.QU .. ently:swollen in nlUIIlPs I ?av~ never seen-'l1i.em..JiiYQ}ye..q.iIL.J:~c.uu.~rr.~.P11[Qtitis. 'Siensen's TuCt· opening it...ru:3.C!:ical1.}z:...alw~ moderat.elY.r~4 an..d c:4.ematous. This is in contrast to our usual observation in mumps. The expression of pus from the duct is diagnostic. The pus has a characteristic appearance. I.tJs. grayish or whitish and i~JIQ9culent with flecks and clumps of pus mixed in the saliva. We have never observed blood with the pus or saliva. This purulent material is expressed very easily from the duct opening by pressure over the affected parotid and then milking the duct along the cheek. Ordinarily one does not see the pus coming from the duct without making this pressure. Often with such pressure it will spurt from the duct opening. Usually only a small amount can be expressed at one time and further milking will not produce more for an hour or more. It seems as if there is very little pus pocketed in the duct at anyone time. We have never been able to express any plug of mucus or other material which might have caused a plugging of any of the main ducts. In a
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few instances purulent material was not present until twenty-four hours after the onset of the infection. In only two of the some sixty cases that we have obs~rved have we been unable to obtain pus from the duct during a recurrence. In these cases only normal appearing saliva was obtained. In no instance was there an absence of salivary secretion from the parotid. In mild cases the duration of the swelling may be only one to two days. In the average case it 'Yill last for two to three weeks but it is not unusual to have some swelling present for six to eight weeks or even longer. In the latter instances there will be considerable fluctuation in the size of the gland during that period. When the swelling has been present for more than two weeks the glands become more nodular and firm and resolution is slow. The number of recurrences varies widely in the individual cases. In some cases we have only seen the swelling once while in others it has occurred as often as ten times. Recurrences have occurred after only a few days, affer a week or a month, sometimes two or three times a year and at times after two or three years.. The longer the interval between attacks the less likelihood there is of a recurrence. The severity of the swelling and the length of time it is present has no bearing upon the number of attacks or the interval between them. It has seemed to us, though, as if the younger the age of the first attack the more likelihood there is of frequent recurrences and the shorter the interval between recurrences. Fever mayor may not be present. When present the temperature varies between 100 0 and 1040 F. Fever is usually present at the onset, at which time it is highest, and persists for only a few days. After that there may be no fever or only a very low grade one. Pain in varying degree is nearly always present. At times it comes on a few hours before the onset of the swelling. It is rarely severe and most children do not complain of it after the first few days. The amount of tenderness varies a good· deal but is rarely severe. There is rarely much objection to pressure over the gland and milking of Stensen's duct. Most often some degree of tenderness is present during the whole period that the gland is swollen. Toxicity is rarely present even when the fever is rather high and when the involvement is bilateral. The children do not appear ill and it is difficult to keep them in bed. Most of them have little or no difficulty in eating. In respect to the blood count, there is usually a leukocytosis ranging from 8000 to 18,000 present with an increase in the polymorphonuclear cells. In mild cases the blood count may be normal. As is usual of infections during infancy the lymphocyte ratio may be high in contrast to that normally seen after the infancy period. Secondary anemia when present has no relationship to the parotitis. X-ray examintltion of the parotids failed to reveal the presence of a
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JOHN A.· BIGLER
calculus or foreign body in any of the cases observed. Sialograms, made by injecting opaque material into Stensen's duct, were obtained in a few cases and were negative. Probing of the duct was also performed in a few cases with negative results. ETIOLOGY
The etiology of recurrent parotitis is still obscure. In our experience the condition has been primary in otherwise healthy infants and children. There is apparently no relationship to throat infections, to the presence or absence of the tonsils or the condition of them, or to healthy or diseased gums or teeth. The disease occurs just as frequently in the healthy robust child as in those with varying degrees of malnutrition. There was also no relationship to a history of mumps occurring prior to the onset of the recurrent parotitis. Some of the cases were observed at the time of the first swelling and mumps could be fairly safely excluded because of the presence of pus from the duct, the blood count, and the absence of mumps in other members of the family before or after contact with the patient. In a few cases it seemed fairly safe to conclude that the patient had previously had mumps. In two instances I have seen an apparent mumps develop in patients who had a recurrent parotitis. Pearson3 reported seventeen children with recurrent parotitis, infection of the secretion being present in six cases and no infection in eleven. Sialograms revealed some dilatation of the large duct or terminal ducts in half of the cases but no point of obstruction was mentioned. Sialograms were done in only a few of our cases and no evident dilatation of the ducts was observed. Undoubtedly some degree of dilatation must be present to account for the increased amount of secretion above the normal that can be obtained upon pressure over the gland and duct. The extent of the dilatation must be slight because of the small amount of secretion that can be obtained at anyone time. We have never been able to express anything that looked as if it could plug one of the main ducts. The consensus favors the theory that the infection is an ascending one from the mouth up through Stensen's duct. This is borne out by the work of Berndt, Buck and Von Burton.4 Using a hemolytic Staphylococcus aureus they injected Stensen's duct in ten dogs and produced a parotitis in seven of them while a parotitis developed in only three of fifteen dogs when an artery to the parotid was injected. Whether infection is the primary cause of the parotitis or whether it is secondary to an already swollen parotid with salivary stasis is not known. There is evidence to support the latter view. Cases of recurrent parotitis occur without infection of the secretion; in other cases infected secretion is not obtained for twenty-four hours or more after the swelling occurs.
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Pearson3 also found a history of allergy or some allergic manifestations in eleven of the seventeen cases he reported. After a careful check of our cases we could find no relationship to any form of allergy. In a few reported cases there seemed to be a relationship between the presence of a throat infection and the onset of the parotitis. This was not true in our series of sixty cases. Bac+eriology.-Cultures of the pus from the duct opening was made in all our cases and the Streptococcus viridans was found in every instance. Fairly frequently Staphylococcus aureus and rarely pneumococcus were also found. Sanford2 reported Staphylococcus aureus in the five cases occurring in the newborn he observed. Staphylococcus aureus has been reported also by others in recurrent parotitis. Why our cases were predominantly Streptococcus viridans infections cannot be explained. . TREATMENT
Treatment so far has been very unsatisfactory in shortening the course of the individual attacks or in preventing recurrences. All forms of the sulfonamides have been used with no apparent benefit. Penicillin has not been used but should be given a trial. We have also used potassium chlorate which is excreted through the parotid and potassium iodide without effect. Aspirin is given for fever and for comfort. Bed rest should be insisted upon at least during the period that fever is present. Any food . that is tolerated is given. Heat or cold applied to the swelling may give some comfort but it does not influence the course of the disease. Sedatives such as phenobarbital may be used but are rarely indicated. We have used x-ray therapy in a number of cases with questioqable results. It has seemed, though, that the course of the individual attacks is shortened and that recurrences are decreased in number as the period between recurrences lengthened. It has never been necessary to resort to surgical interference either by slitting the duct opening or incising the gland. In fact, such a procedure should be frowned upon until there is sufficient evidence that frank suppuration has occurred and other methods of therapy have failed. It must be emRhasized that recurrent parotitis is a benign condition that occurs in otherwise healthy infants and children. Surgical intervention is rarely if ever indicated. This is in contrast to suppurative parotitis, whether it occurs in the newborn or is secondary to some other infection or toxins as a complication of surgery, in which cases surgical incision is usually necessary. No instance of contagion from recurrent parotitis has ever been observed
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JOHN A. BIGLER
REPORT OF CASES
The following short case reports are typical of the cases seen. The first one is the only case we have seen in which suppuration occurred. CASE I.-Von W., a girl born July 25, 1935, was first: seen on August 10, 1939, with a history of swelling of both parotids and fever for six days. There was no preceding illness. The rectal temperature was 101.6° F. and there was a marked bilateral parotitis with fluctuation (Fig. 7), pus from both Stensen's ducts and a suppurative otitis media. The patient was' not toxic. She was admitted to the hospital arid heat was applied to both parotid areas: The day after admission both parotids began to suppurate _through the skin. The leukocyte count was 24,500 with 79 per cent polymorphonuclear cells, 18 per cent lymphocytes and 3 per
Fig. 7 (Case- I) .-A, Acute suppurative bilateral parotltls. Spontaneous suppuration through skin seven days after onset of parotitis. Uneventful recovery. H, rear view. cent monocytes. Examination of _the urine was negative as were the serology and Mantoux tests. Cultures of the pus from the Stensen's duct opening, the parotid suppuration and the otitis media revealed Streptococcus viridans and Staphylococcus aureus. The course was an uneventful one -and there was -a complete recovery by September 8, 1939. There have been no recurrences. CASE 11.-0. A., a girl born January 14, 1936, was seen on December 1, 1941, with a left pal,'otitis of three weeks' duration. The swelling involved the whole left parotid and was -firm; no fluctuation was present. The skin over the swelling was slightly reddened. Pus was easily expressed from Stensen's duct which upon -culture showed a ,streptococcus viridans. There had been only slight fever and when the patient was seen the ·temperature was 100.2° rectally. The exam: ination was otherwise negative except for large tonsils and several- carious teeth. The serology was negative and the leukocyte count was 18,900. The swelling
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gradually subsided and was entirely gone by December 19. There have been no recurrences up to the present time. The child had had chickenpox from which she recovered one week before . the present parotitis. This was the third attack of parotitis. There was a bilateral parotitis in February 1941 diagnosed as mumps by another physician. In August 194( she had a right-sided parotitis reported as mumps by still another physician. CASE III.-J. A. was born in February 1931, had her tonsils and adenoids removed in September 1938. There was no history of mumps. In November 1941 she was seen with a left parotitis. There was no fever. Pus was obtained from Stensen's duct and upon culture Streptococcusviridans was found. The swelling remained for two and one-half months. She was again seen in February 1942 with
Fig. 8 (Case V).-A, Third recurrence of a bilateral parotitis. Although only right side is shown in this photograph the left side was similarly swollen. B, Recurrence of left parotitis only, eighteen months later. a recurrent left-sided parotitis which lasted for one month. Cultures of. pus from Stensen's duct again showed Streptococcus viridans. There was no fever. The leukocyte count was 8800. The examination of the ·urine, blood serology and the Mantoux test were negative. Roentgenograms of the parotid were also negative. There have been no further known recurrences. CASE IV.-P. P. was born in August 1940. A left parotitis diagnosed as mumps by. a physician .occurred in April 1942. When seen in September 1942 .there was a left parotid swelling which had been present for one week. Considerable fever had been present at the onset but the temperature was now normal. Pus was expressed from the duct which upon culture showed a Streptococcus viridans. The swelling did not completely subside until October 21, 1942, a period of five weeks from the onset. On October I, after the parotitis had ' been present for two weeks, a severe throat infection with temperature up to 104° F. developed. The
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JOHN A. BIGLER
throat infection subsided in eight days with only a slight increase in the size of the parotitis. There have been two recurrences of the parotitis, each lasting for ten days. The first developed on November 1, 1942 and the last on May 5, 1943. In both, cultures of pus from the duct showed a Streptococcus viridans. Leukocyte counts ranged from 8600 to 12,350 with polymorphonuclears 34 to 43 per cent and lymphocytes 66 to 35 per cent. The tonsils were large and the mouth was clean. CASE V.-G. M. was born April 27, 1935. This boy was seen in January 1938 with a bilateral parotitis (Fig. 8, A). Strep~ococcus viridans and Staphylococcus aureus grew in cultures of the pus expressed from both Stensen's ducts. There was no rise in temperature. The examination was otherwise negative as were the roentgenograms of both parotids. This was the third attack of bilateral parotitis he had experienced during the past year. Each attack lasted from one to three weeks. He was seen again in June 1939 with a left parotitis (Fig. 8, B). At this time no pus could he obtained from the duct but there was mucus. The leukocyte count was 12,750 with polymorphonuclears 45 per cent, lymphocytes 44 per cent, monocytes 4 per cent and eosinophils 7 per cent. Because of these findings and the fact that there was no evident contagion it was felt that this was also a recurrence of the parotitis but without infection and not a mumps parotitis.
c.:
REFERENCES
1. Blair, V. P. and Padgett, E. Pyogenic Infection of the Parotid Glands and Ducts. Arch. Surg., 7:1 (July) 1923. 2. Sanford, H. N. and Shmigelsky, I.: Purulent Parotitis in the Newborn. J. Pediat., 26:149 (Feb.) 1945.
3. Pearson, Bruce, R. S.: Recurrent Swelling of Parotid Glands. Arch. Dis. Child., 10:363 (Oct.) 1935. 4. Berndt, A. L., Buck, R. and Von Buxton, R.: Pathogenesis of Acute Suppurative Parotitis. Am. J. M. Se., 182:639 (Nov.) 1931.