sialadenitis in childhood 2

Sialadenitis in Childhood

Leonard B. Kaban, DMD, MD, Boston, Massachusetts

John B. Mulliken, MD, Boston, Massachusetts

Joseph E. Murray, MD, Boston, Massachusetts

Inflammatory salivary disease comprises more than one third of all salivary gland pathology in infancy and childhood [I]. The following is a study of 30 years’ experience with sialadenitis at the Children’s Hospital Medical Center. This analysis suggests that salivary gland inflammation in children can be di- vided into four distinct clinical entities, each re- quiring a different therapeutic approach.

Material and Methods

The records were reviewed of all patients up to age sixteen years who had been treated for sialadenitis, from January 1,1947 through December 31,1976. This study included only those patients in whom the diagnosis was documented by clinical, radiologic, or pathologic findings. Radiographs were reviewed to confirm the presence of sialangiectasis (acinar dilatation) and/or calculi. Long-term follow-up of patients with recurrent parotitis was obtained by telephone interview.

Results

There were forty-nine patients (28 male, 21 fe- male) with a total of sixty-seven hospital admissions. Thirteen were admitted two or more times. Four distinct types of salivary gland inflammation were noted: (1) acute submaxillary sialadenitis (8 patients); (2) acute suppurative parotitis (18 patients); (3) chronic parotitis (9 patients); and (4) recurrent acute parotitis (14 patients).

Acute Submaxillary Slaladenltls

Obstruction of Wharton’s duct was the basis of acute submaxillary sialadenitis in all eight patients: seven had sialolithiasis and one had congenital ductal stenosis. The age range was three months to fifteen years. There were no underlying metabolic or chronic illnesses. A distinct area of ductal narrowing was seen

From the Harvard Medical School and School of Dental Medicine, Division of Plastic and Oral Surgery. Children’s Hospital Medical Center, Boston, Massachusetts.

Reprint requests should be addressed to L. B. Kaban, DMD, MD, Harvard MedIcal School and School of Dental Medicine, Children’s Hospital Medical Center, Boston, Massachusetts 02115.

Presented at the Fifty-Eighth Annual Meeting of the New England Surgical Society, Portsmouth, New Hampshire, September 30-October 2, 1977.

on the one sialographic study in this group. (Figure 1A.) In the other patients the diagnosis of sialolithiasis was made by either physical examination or soft tissue x-ray of Wharton’s duct. (Figure 1B.)

Submaxillary gland excision was necessary for all patients with sialolithiasis, and they all remained symptom-free during the six month to two year follow-up period. The one patient with congenital stenosis underwent a duct marsupialization procedure and also did well.

Acute Suppuratlve Parotltls

Acute suppurative parotitis was the most common form of pediatric sialadenitis. The eighteen patients in this group ranged from seven days to eleven years of age (mean, 4.4 years). Local or systemic illness preceded the development of parotitis in most patients (14 of 18). The underlying conditions includea: (1) fever and dehydration secondary to upper respiratory infection, systemic viral illness, or ac@e glo- merulonephritis; (2) immunosuppression re&lting from congenital immunoglobulin deficiency or chemotherapy for acute leukemia; (3) failure to thrive, or respiratory distress syndrome in newborn or premature infants; and (4) local alterations in the gland from trauma or mumps parotitis.

The characteristic physical finding was a tender, indurated swelling in the preauricular region and/or the angle of the jaw. Intraoral examination was helpful in that twelve of eighteen patients had an edematous, erythematous Stenson’s papilla, with or without purulent drainage from the duct.

Cultures of the duct secretions in thirteen of eighteen patients showed coagulase-positive staphylococci in six patients, Escherichia coli, acid-fast bacteria, or mixed oral organisms in three patients, and “no growth” in four patients. Nine of the fifteen patients treated with antibiotics received oxacillin or erythromycin. The newborn patients received, in addition, a broad spectrum antibiotic, either ampicillin, kanamycin, or chloramphenicol. The remaining patients were given penicillin or ampicillin.

One patient with acute parotitis died due to systemic sepsis in association with acute mye!ogenous

570 The Amarlcan Journal of Surgery


Figure 1A. Acute submaxillary sialadenttis. S/a&ram demonstrates Wharton’s duct stricture, probable site of previous obstructing calculus.

Figure 78. Teenage g/r/ with acute obstructive submaxillary slaladenitls. intraoral view Illustrates swollen duct and corre- spondhtg soft tlssue x-ray f//m reveals a stone.

leukemia. Four patients developed parotid abscesses, and drainage was necessary for total resolution of the infection. Two patients had parotid biopsies, and one required superficial parotidectomy because of persistent infection and a cutaneous fistula.

Once the acute infection had resolved, sialograms were done in four patients: two delineated a mass

lesion which corresponded to an abscess and two showed sialangiectasis.

Chronic Parotltls

Nine patients had chronic parotitis, and of these, three had associated conditions: Sjogren’s syndrome,

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Kaban, Mulliken, and Murray

systemic lupus erythematosis, or Raynaud’s disease. Diffuse hypergammaglobulinemia, elevated antithyroid globulin, or antinuclear antibody titers were seen in the patients with autoimmune disorders. The majority of patients (5 of 9) with chronic parotitis had bilateral involvement. (Figure 2.) Sialograms were performed on six patients and demonstrated generalized sialangiectasis without anatomic obstruction. Soft tissue x-ray examination in one patient showed a stone in the parotid gland.

Six patients were treated successfully with heat, massage, and sialogogues. The remaining three had specific therapy, namely, bilateral irradiation (300 r) in one patient and total parotidectomy in two patients. All patients remained symptom-free after treatment.

Recurrent Acute Parotttls

Recurrent acute parotitis occurred in fourteen patients and was characterized by multiple attacks of pain, swelling, and fever. There were no local eti- ologic factors or underlying systemic illnesses. The age of onset ranged from eight months to sixteen years with a mean of 9.6 years.

In eight patients, whose histories were well documented, there were sixty-two acute episodes of parotitis over a period of eight weeks to eleven years. One patient had more than eighteen acute attacks. Six other patients reported “multiple episodes.” Purulent drainage from Stenson’s duct occurred less frequently (2 of 14 patients) than in patients with acute suppurative parotitis. The exudate grew

Diplococcus pneumoniae in one patient, and (Y- streptococci in the other. Five patients had nonpu- rulent saliva; however, cultures showed coagulase- negative staphylococci (2 patients), a-streptococci (1 patient), and “no growth” (2 patients).

Sialograms were performed on thirteen patients; three had bilateral studies. These studies showed normal duct systems; however, the majority (11 of 16) demonstrated sialangiectasis without significant delay in emptying after administration of a sialo- gogue. (Figure 3.) One patient had bilateral acinar dilatation in the presence of unilateral parotitis. Four patients had completely normal sialograms.

A variety of antibiotics were used for the treatment of acute attacks; the most common was oxacillin alone or in combination with penicillin or ampicillin. The acute episodes of parotitis resolved on a variety of antibiotic regimens.

Five patients were treated with dilatation of Stenson’s duct and/or a sialogram. Insertion of a drainage tube in another patient caused a flare-up of his parotitis, which resolved after removal of the tube. The patients who had duct dilatation and sialography had no further episodes of parotitis during a one year follow-up. Three patients required superficial parotidectomy for multiple episodes of acute infection. Histologic examination of the three excised specimens showed “chronic sialadenitis.” Only one of the three continued to have recurrent episodes of infection in the remaining deep portion of the gland. One patient underwent tympanic neurectomy, and remained symptom-free for two years. Later, he de-

572 The American Journal of Surgery


veloped recurrent acute parotitis on the opposite side and was again successfully treated with this procedure. Ten patients treated with ductal dilatation or antibiotics responded to therapy during the acute episodes and by their late teens required no further treatment. The patients who underwent superficial parotidectomy or bilateral neurectomy also remained asymptomatic in long-term follow-up.

Comments

Childhood salivary inflammation is an infrequent clinical entity which is frequently reported in the literature [I-5].

(1) Acute submaxillary sialadenitis is usually associated with sialolithiasis, in contrast to acute parotitis in which obstructing calculi are rare [6,7]. Precipitation of calcium salts in submaxillary saliva is potentiated by its alkalinity and high glycopro- tein-mucin content [6]. Local anatomic abnormali- ties, such as duct strictures, also contribute to submaxillary stone formation. (Figure 1A.) Furthermore, these patients do not exhibit generalized abnor- malities of calcium and phosphorous metabolism, nor do they have an increased incidence of calculi in other salivary glands or other organ systems. Stone removal facilitates drainage and allows for resolution of the acute infection. However, the recurrence rate is high because of persistence of the original anatomic obstruction or the development of a new stricture after sialolithotomy. For this reason, most authors agree that excision of the gland should be the definitive treatment of obstructive submaxillary sialadenitis

]1,61. (2) Acute suppuratiue parotitis is usually seen in

newborns, particularly premature infants, and in children with an underlying systemic illness, often in the presence of fever, dehydration, or immunosuppression [3,5,8-111. Postoperative parotitis, so characteristic of the adult population [12], is rare in childhood [5]. Retrograde infection from the mouth [6,13,14] or congenital sialangiectasis with pooling of saliva and infection [5] have both been proposed as the underlying mechanism of acute parotitis. The theory of retrograde infection is inconsistent with the high percentage of Staphylococcus aureus found in culture. It is unclear whether sialangiectasis is the cause or result of the inflammation. There have been several reports of sialangiectasis documented prior to infection [3,15].

Sialography is contraindicated in the acute stage of this disease because the instillation of dye under pressure may result in rupture of the terminal ducts and acini, and spread of the infection. Soft tissue

Figure 2B. Bllaterai slalangiectask seen on a stakgram ot an e@ht year 0M chki with dKonic bttaterat patvthi swetttng and Raynaud’s disease.

films may visualize radiopaque stones, but there is a high false-negative rate because parotid calculi are usually very small [7]. The majority of patients with acute parotitis recover from a single attack and do not go on to develop a chronic or recurrent form of the disease [5].

(3) Chronic parotitis, in contrast, is present in a group of patients who have low grade, persistent, or intermittent swelling and pain in the absence of signs of acute sepsis. To detect underlying autoimmune disease, these patients should be investigated with protein and immunoelectrophoresis, latex fixation test, antinuclear antibody/antithyroid globulin titers, and labial salivary gland biopsy [16,17]. Other etio- logic factors postulated for chronic parotitis are allergy [5,18,19], heredity [20], and duct obstruction by stones [7] or mucus plugs [21,22]. Chronic paro-

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titis has been noted in only two of 900 patients with diseases [25,26]. Radiotherapy is therefore not rec- cystic fibrosis. [I]. ommended for chronic parotitis.

Diminished salivary secretion and failure of the bacteriostatic action of saliva may be the basis for low grade swelling and infection in patients with chronic parotitis [23,24]. This theory is consistent with the fact that many of these patients improve with local heat therapy, massage, and sialogogues.

One patient in this series was successfully treated with radiotherapy. However, there is a significant risk of late neoplasm developing in irradiated glands, especially in patients with associated autoimmune

(4) Recurrent acute parotitis occurs in patients with a mean age of onset of 9.8 years, in contrast to acute suppurative parotitis, for which the mean age of onset is 4.4 years. Although the patients have no underlying systemic illnesses, they suffer significant morbidity from their multiple attacks of salivary inflammation.

Alpha streptococcus is commonly cultured in the recurrent acute parotitis group, whereas Staphylo- coccus aureus is usually found in acute suppurative

TABLE I Sialadenitis in Childhood

Associated Illness Pathophysiology Bacteriology Sialography Therapy Prognosis

Acute Submaxillary Sialadenltis None Obstruction:

stones, Duct stenosis

Acute Suppurative Parotitls Fever, dehydration,

immunosuppression, systemic illness in the newborn

Chronic Parotitis Autoimmune dlsorders

I Viscosity Secretion

2” Bacterial infection

1 Secretion on autoimmune or allergic basis

“Pooling” of saliva

Recurrent Acute Parotltis None ? J Secretion

? “Pooling” of saliva

Mouth organisms

Purulent drainage usually S aureus

Minimal secretion for culture

Purulent drainage Sialangiectasis Antibiotics, (infrequent), mixed (study may drainage, culture with be thera- possible

Stone often seen on soft tissue x-ray

Contraindicated in acute phase

Antibiotics, stone removal, gland excision

Antibiotics, fluids, heat

Heat, massage, sialogogues

No recurrence

Usually 1 episode

Control with symptomatic treatment

Multiple attacks, high morbidity, “burns out”

cu-streotococci oeutic) oarotidectomv late teens

574 The American Journal ol Surgery


with a normal duct system, and no delay in emptying. (Figure 3.) The examination, therefore, has limited diagnostic value. However, sialography may provide curative drainage as observed in this series and reported by others [6,15,28].

Patients treated medically or by manipulation of the parotid duct eventually become symptom-free in the middle to late teens. Other authors have also noted the tendency of recurrent acute episodes to cease near the age of puberty [3,27] or in late ado- lescence [5]. Because this disease has a self-limited course, superficial parotidectomy is usually unnec- essary. The three patients in our series who were treated surgically had experienced an extremely debilitating course over a period of 1.5 to 4 years. Beahrs, Devine, and Wollner [29] reported twenty- nine cases of parotidectomy for recurrent parotitis, yet also cautioned that the operation is not recommended for all patients. Transmeatal avulsion of the

/ glossopharyngeal nerve fibers in the tympanic plexus (“tympanic neurectomy”) has been noted to produce

t atrophy of the parotid gland and relieve recurrent attacks of acute parotitis [30,31]. One of our patients

! , was successfully treated by this method. The long- term recurrence rate after tympanic neurectomy in a large number of patients is unknown; therefore, superficial parotidectomy is the procedure of choice. /

/ Penicillin is the drug recommended for recurrent

I acute parotitis; oxacillin is added to the regimen if the patient does not improve within 24 hours. Saliva or purulent material from Stenson’s duct should be cultured, and if necessary, antibiotic therapy should be adjusted according to the bacteriology. A diagnostic and possibly therapeutic sialogram should be done three or four weeks after the acute episode re- solves.

Figure 38. Specfrum of sialangiecfasis in recurrenf acufe paroflflo. Top, m/M bilateral. Mlddfe, moderate diffuse. Boffom, large saccular dllafafion of ducfs with pooling of dye.

parotitis. The underlying etiology of recurrent acute parotitis is unknown, although factors similar to those discussed for chronic parotitis have been postulated [4,7,14,19,22,23].

Sialography usually demonstrates sialangiectasis,

Conclusion

There are four distinct types of pediatric salivary gland inflammation (Table I): (1) acute submaxillary sialadenitis; (2) acute suppurative parotitis; (3) chronic parotitis; and (4) recurrent acute parotitis. The four conditions differ in etiology, clinical man- agement, and prognosis. Acute submaxillary sialadenitis occurs in the presence of obstruction and requires excision of the gland for curative treatment. Acute suppurative parotitis occurs most frequently in a “compromised host.” Once the acute episode is treated, these patients usually do not go on to develop a chronic form of the disease. Chronic parotitis is usually low grade, often associated with autoimmune disorders, and responds to symptomatic treatment. Recurrent acute parotitis is characterized by fre-


quent acute attacks which require antibiotic therapy, with eventual termination of the disease process in the second decade of life.

References

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Pearson RSB: Recurrent swelling of the parotid gland. Arch Dis Child 10: 363, 1935.

Blatt I: Chronic and recurrent inflammations about the salivary glands with special reference to children. Laryngcscope 76: 917.1966.

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Brook AH: Recurrent parotitis in childhood. 8rDentJ 127: 271, 1969.

David RB, O’Connell EJ: Suppurative parotitis in children. Am J Dis Child 119: 332, 1970.

Rose SS: A clinical and radiological survey of 192 cases of recurrent swellings of the salivary glands. Ann R Coil Sufg Engl15: 374, 1954.

7. Patey DH: Inflammation of the salivary glands with particular reference to chronic and recurrent parotitis. Ann R Cc/i Swg Engi 36: 26, 1965.

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Shulman BH: Acute suppurative infections of the salivary glands in the newborn. Am J Dis Child 80: 413, 1950.

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Plewka W: Zur Pathogenese der eitrigen Parotitis der Neuge- borenen. Arch Kinderh 69: 279, 1921.

Carlson RG, Glass WW: Acute suppurative parotitis: twenty- eight cases in a county hospital. Arch Surg 86: 659, 1963.

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Payne RT: Recurrent pyogenic parotitis: its pathology, diagnosis, and treatment. Lancet 1: 348, 1933.

Jones HE: Recurrent parotitis in children. Arch Dis Child 28: 182,1953.

Bailey H: Congenital parotid sialectasis. J Int Co/i Surg 8: 109, 1945.

Chisholm DM, Mason DK: Labial salivary gland biopsy in Sj& gren’s disease. J C/in Pathol21: 656, 1968.

Greenspan JS, Daniels TE, Talal N, Sylvester RA: The histo- pathology of Sjmren’s syndrome in labial salivary gland bioosies. Oral Sum 37: 217, 1974.

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Pear&n RSB: Recurrent swelling of the parotid gland. Gut 2: 210, 1961.

Waldbott GL, Shea JJ: Allergic parotitis. J Allergy 18: 51, 1974.

Smith M: Familial incidence of sialectasis. Br Med J 2: 1359, 1953.

Maynard JD: Parotid enlargement. Hasp h&d 1: 620, 1967. Banks P: Non-neoplastic parotid swellings: a review. Oral Surg

25: 732, 1968. Maynard JD: Recwent parotid enlargement. &J Surg 52: 784,

1965. Katzen M. DuPlessis DJ: Recurrent parotitis in children. S Afr

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Hempelmann LH. Hall WJ, Phillips M, Cooper RA, Ames WR: Neoplasms in persons treated with x-ray in infancy: fourth survev in 20 vears. J NaN Cancer lnst 55: 519, 1975.

Carroll RG: The ielationship of head and neck irradiation to the subsequent development of thryoid neoplasms. Sem Nut/ Med41: 411, 1976.

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576 The American Journal of Surgery

treatment of chronic sialadenitis. Am J Surg 102: 760. 1961.

30. Friedman WH, Swerdlow RS, Pomarico JM: Tympanic neurectomy: a review and an additional indication for this procedure. Laryngcscope 84: 508, 1974.

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Discussion

Frederic Rueckert (Hanover, NH): I am particularly impressed by the large number of cases presented. After superficially reviewing the literature, reviewing our cases in Hanover on the surgical service as well as talking to our pediatricians and otolaryngologists, I came to the conclusion that the incidence of sialadenitis in children is extremely low, at least up in our area. Why were there so many cases at the Children’s Hospital? Is the incidence that much greater in Boston or is it the nature of the re- ferral practice? The few cases we did have were satisfac- torily managed conservatively without resort to surgery. Surgery of the chronically inflamed parotid gland in adults is extremely difficult. I wonder if the authors also found this to be true in the children they operated on.

In adults salivary gland infections are often secondary to other complicated medical or surgical problems. I would be interested in knowing how many of the patients reported here were admitted primarily for the care of th6 sialadenitis and how many were in the hospital for other reasons and developed the salivary infections as postoperative com- plications.

John H. Fisher (Boston, MA): I rise to speak from personal experience. The fourth group, not seen in adult practice, is the one that I have seen. Concerning manage- ment, I use Gantrisinm in a long-term course. I begin with full doses and lower the dose later. I have not seen patients that have gone on to recurrent disease. The swelling has subsided without surgical. intervention.

Leonard B. Kaban (closing): This was a retrospective study of patients who were admitted to the hospital for sialadenitis. The disadvantage of this type of study is that mild forms of the disease, without severe sepsis, are ex- cluded. This would account for the absence of a group of patients with urologic problems and mild parotitis re- sponsive to Gantrisin. We did find several patients with a past history of such mild forms of the disease who were later admitted on one or more occasions with a more severe episode of acute parotitis. These patients were classified in the recurrent acute parotitis group.

The fact that Children’s Hospital Medical Center is a tertiary level hospital would account for the larger number of patients we saw with sialadenitis. The patients in the acute suppurative parotitis group were often admitted on the medical service first with acute systemic illness, ma- lignant disease, or a complication of chemotherapy. The patients with recurrent acute parotitis were seen by us after multiple episodes of parotitis treated first in the commu- nity.

sialadenitis in childhood 2

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