Painless mass in the parotid region

J Or01 Moxilfofoc

Surg

58-316319.2000

Painless

Mass in the Parotid John E. Mullins,

Jr, DMD, * Owett Ogle, DDS, f

and Darki

Case

Region

A. Cottrell,

DMD#

drdining. On palpation. the swelling was noted to be fixed. tluctuant. and was not warm to touch. The submandibular and cervical lymph nodes were palpable and nontender. Cranial nerves II to Xl1 were grossly intact. The rest of the head and neck examination was within normal limits. The intraoral examination showed a mixed dentition in good repair. The oral mucosa. tongue. floor of mouth, and throat appeared normal. Stenson’s duct was patent and produced clear saliva on milking. Results of the rest of the physical examination were normal. The patient did not have an elevated temperature. The panoramic ndiogrdph of the jaws showed a pattern that was normal for a 12-year-old child.

Presentation

A 12-year-old Hispanic girl presented to the oral and maxillofacial surgery clinic at Woodhull Medical Center with a chief complaint of a nontender right facial swelling. The history was obtained from both the father and the patient through a translator. This revealed that the patient first became aware of the swelling about 4 months previously and that it had continued to increase in size. On further questioning about the origin of the swelling, the father gave a history of a mild to moderate upper respirator! infection at about the same time that the swelling first appeared. The medical history indicated that the child had not received the usual childhood vaccines. She was never hospitalized, was not on any medications, and did receive routine pediatric care. Overall, her genenl health had been good. The social history disclosed that the patient moved from Mexico approximately 1 year ago and was living with her father, aunt, uncle, and 2 siblings, ages 5 and 18 years, in a S-bedroom apartment in Brooklyn, New York. The mother was still living in Mexico with her remaining 2 children, ages 14 and 15 years. The patient had achieved the fourth grade while in Mexico. but had never attended school in the United States. She denied having a boyfriend; nor had she hzd any’ close personal or sexual contact with anyone in the United States. There was no histoy of tobacco, alcohol. or illicit drug usage. The review of systems revealed that she had had 3 or 4 upper respiratory infections since coming to the United States; there was no history of chronic cough or hemoptosis. She had not yet reached menarche. Examination showed a 5.0 X S.S-cm swelling located over the center of the right parotid gland (Fig 1). The skin wds erythematous, and there was a sinus tract located at the inferior aspect from which a serosanguinous exudate was

Differential

Diagnosis

This

1Z-year-old nonimmunized immigrant girl with a skin mass and a parallel history of upper respiratory infections (URI) and cervicofacial lymphadenapathy. Normal childhood immunizations usually include vaccines against hepatitis B, varicella, measles, mumps, rubella (MMR), Hcwmpbilus influCVZZLZ~. polio (TOPV), and diptheria, tetanus, pertussus (DTP). Although the lack of immunization could explain the patient’s respiratory symptoms (diptheria, pertussus), the skin lesion is most likely unrelated. Lack of immunization is probably more significant and consistent with a lack of appropriate medical care for the patient and her family. The soft tissue lesion appears consistent with either a primary or secondary infectious process. presents

INFECTIONS Poverty, along with overcrowding and malnutrition, is the most signiticant epidemiologic factor for the development of tuberculosis (TB).’ In most individuals, the infection is subclinical, without sequelae. However, in 5% of cases, an active lung infection develops. A small proportion of these infected cases can progress to involve multiple organ systems (miliary TB) through lymphohematogenous spread. In the United States, most TB infections are from the human strain. Infection is transmitted by tiny (Cl0 pm) aerosol droplets from an infected individual that are inhaled by a noninfected individual. The bovine form is usually transmitted to the unsuspecting victim after ingesting raw milk from a tuberculous cow.? Infection of the lymphatics can also occur, leading to caseation

‘Chief Resident (1997-1998). Woodhull Medical Center. Depanment of Dentistry. Division of Oral and Maxillofacial Surgery. Brooklyn. NY. tDirector, Woodhull Medical Center. Department of Dentistry/ Oral and Maxillofacial Surgery. Brooklyn, NY. *Associate Professor, Director, Oral and Maxillofacial SurRrq Residency Training, and Director of Resident Research. Depanmcnt of Oral and Maxillofacial Surgery, Boston Universiv School of Dental Medicine, Boston, MA. Address correspondence and reprint requests to Dr Mullins: PO Box 839, Scotch Plains. NJ 07076; e-mail: [email protected] o 2000

Amerlcon

Assoc~ot~on of Oral and Mortllnfoclol

0278.2391/00/58030013$3

Surgeons

00/O

316

MULLINS.

FIGURE facial

OGLE.

AND

317

CO’ITRELL

1. Preoperative photogroph of the patient showing mass located in the region of the parotid gland.

the right

and breakdown of lymph glands. Cervical node involvement is known as “scrofula.” Major salivary glands also may be infected, either primarily or secondarily, and the parotid gland is most commonly involved. Pulmonary spread to the trachea and larynx, as well as the gastrointestinal tract, can occur as a secondary infection with an “open lesion.” Symptoms of a URI can be mimicked. General signs and symptoms of a TB infection include night sweats, fever, malaise, weight loss, and productive cough with or without bloodtinged sputum. The diagnosis of TB normally includes not only a positive skin reaction to purified protein derivative (PPD), but also culture of acid-fast mycobacterium from sputum or discharge from other infected sites. Chest radiographs are also necessary to evaluate lung involvement. Bony involvement is not common but does occur. In this case the panoramic radiograph was normal. Granulomatous infections that also need to be considered are leprosy (Hansen’s disease), cat-scratch disease, gnnuloma inguinale, and syphillis. A reported negative sexual history should never rule out “sexually transmitted” diseases from the differential diagnosis. Other infections, including mononucleosis, toxoplasmosis, and human immunodeficiency virus (HIV), also commonly cause cervical-facial lymphadenapathy. Necrosis and subsequent soft tissue breakdown is not common with these processes. Cervicofacial actinomycosis is an infection primarily caused by Actinomyces ismeli. Actinomyces both causes lumpy jaw in cows, but is rarely pathogenic in humans. Trauma is commonly the portal of entry for an actinomycosis infection, although a dental origin is not unusual. In the face and neck, abscess formation followed by the formation of fistulous tracts can occur in salivary glands, bone, or skin. The skin will often become indurated and fluctuance can occur. The so called sulfur granules are colonies of organisms that appear as tiny yellow granules in the suppurative

discharge from the lesion. Although the cervicofacial form of the disease is most common, an abdominal and pulmonary form can also be seen. The pulmonary form can produce fever, chills, cough, and pleuritc pain. The diagnosis is confirmed by biopsy and culture of the infected lesion. Culture is very difZcult because the organism is not very fastidious. Mycotic infections such as blastomycosis, histoplasmosis, coccidioidomycosis, cryptococcosis, and mucormycosis must be suspected because these pathogens can cause both skin and pulmonary infections. Blastomycosis presents in both a North American (Blastornlres dermatitides) and South American (Blastomyces bl-asliensis) variety, although they are no significant clinical difTerences.3 Both forms can affect the skin, bones, liver, lungs, or other organs. The cause of disease transmission is unknown, but people working outdoors appear to have higher infection rates. Skin lesions often begin as small red papules that gradually increase in size, subsequently abscessing and ulcerating. Systemic disease is characterized by fever, weight loss, and a productive cough. Constitutional symptoms are similar to those seen in pulmonary TB. Histoplasmosis is caused by Histoplasma capsulattrm and is endemic in the Mississippi Valley and Northeastern United States. Up to 75% of the population in these areas has had an asymptomatic infection.” Disease transmission is normally due to inhalation of dust containing spores of the fungus. Involvement of the reticuloendothelial system, with a chronic low-grade fever, productive cough, and lymphadenapathy, is often seen, as well as occasional anemia and leukopenia. Skin ulceration is not common. Coccidioidomycosis is a relatively common infection, endemic in the southwest, Mexico, and Central and South America. Most infections are subclinical. The disease is transmitted by the inhalation of dust

FIGURE indurotion

2. Photograph and erythemo

of the patient’s right arm showing on Oreo of resulting from the tuberculosis skin test.

318 contaminated by Coccidioides inmitis. Two forms of the disease are seen, a primary nondisseminated form and a progressive disseminated type. In primary coccidioidomycosis, respiratory symptoms of cough and pleural pain, headache, and anorexia can be seen. Patients can develop skin lesions that are chronic, granulomatous, ulcerated, and tend to scar. Most cases are self-limiting. Cryptococcosis is caused by Cryptococcus ueoformuns. Transmission is most likely through inhalation of airborne microorganisms and seems to more commonly affect immunocompromised persons with diseases such as lymphoma or leukemia. The skin lesions are brown papules that ulcerate. Pulmonary symptoms appear as a nonspecific pneumonitis. Meningeal involvement is not uncommon, and neurologic symptoms consistent with an elevated intracranial pressure can occur. Mucormycosis is almost always seen in immunocompromised individuals with diseases such as diabetes mellitus, lymphoma, or renal failure. Two forms are recognized: superficial and visceral. The superficial form involves the skin, ears, and nails. The visceral forms are more severe and are divided into 3 main types: pulmonary, gastrointestinal, and rhinocerebral. Visceral forms are often fatal, producing vascular invasion and thrombosis, resulting in tissue necrosis. Other secondary infections such as an infected dermoid, epidermoid, or lymphoepithelial cysts should be considered. An infected developmental fistula, foreign body, or parotid gland infection are also possible: but not consistent with the history.

MALIGNANCY

Malignancy must always be suspected in a pathologic process. Any tumor can outgrow its blood supply, leading to tissue necrosis and secondary infection. Rhabdomyosarcoma is the fifth most common tumor in children, and the cheek is one of the more common sites in the facial area.5 The embryonal form of the disease is the most common one in the head and neck region. The tumor is rapidly growing, itiltrative, and associated with pain and bone destruction. Other soft tissue sarcomas are rare, but may be considered. The diagnosis of sarcoma not only involves routine microscopic examination, but also immunohistochemistry and electron microscopy. Leukemia often presents as a painless lymphadenapathy. Acute lymphocytic leukemia is the most common form of the disease in children. Most signs and symptoms of the disease result from bone marrow suppression secondary to overproduction of leukemic cells and infiltration of organ systems. Fever is a common early symptom of the disease, along with

PAINLESS MASS IN THE PAROTID

REGION

infections of the lungs, skin, urinary tract, mouth. and upper respiratory tract. Skin cancer includes basal cell carcinoma, melanoma, and squamous cell carcinoma. Because of the patient’s race, age, and negative familial history, these are probably unlikely. Parotid tumors are uncommon in children but, as with adults, mucoepidermoid carcinoma is the most commonly occurring malignancy. Apparent normal function of the parotid gland does not rule out this tumor. There is no apparent facial nerve involvement in this patient, which makes a late-stage tumor unlikely. LYMPHADENITS

Lymphadenitis with subsequent soft tissue breakdown should also be considered. Kikuchi-Fujimoto disease is a poorly understood necrotizing lymphadenitis found in young girls presenting with cervical lymphadenapathy, which would be consistent with this case.‘* Presenting systemic symptoms vary, with sore throat, fever, abdominal pain, chest pain, diarrhea, chills, nausea, and vomiting being most cornrnon. The disease is often misdiagnosed as a nonHodgkin lymphoma. Although 1 fatality has been reported, spontaneous and complete resolution usually occurs with appropriate supportive care.

Conclusion ESased on the limited information available, an infectious process is most likely. Tuberculosis or another gnnulomatous process is consistent with the presenting history. A mycotic infection is also possible. Although less likely, leukemia or lymphoma should be suspected, along with other malignant processes. Necrotizing lymphadentitis (Kikuchi-Fujimoto disease) is also consistent with the presenting findings. SUBSEQUENT

COURSE

The patient was admitted to Woodhull Medical Center for medical and laboratory workup, incision and drainage of the mass, and intravenous cefazolin. An admitting chest radiograph was done. The cardiomediastinal silhouette was within normal limits. A right upper lobe granuloma was seen, but no focal lung consolidation was evident. Based on these findings. it was decided to administer a tuberculin skin

test (Mantoux test). Illis resulted in an intlammatory reaction with an erythematous area 10 mm in diameter (Fig 2). In addition,

the patient

had an anerbT panel performed

for

mumps, trichophytum, and candida. An erythematous area of approximately 15 mm in diameter appeared on the arm where this test was performed. This was indicative of a positive anergy panel. On the second day after admission, the patient was taken to the oprrdting

room for an extrdordl

incision

and drainage

and a biopsy. After the patient was prepared and draped in the normal fashion for a sterile surgical procedure. aspintion of the lesion with an 1Rgauge needle was performed

319

MLILLINS. OGLE, AND <:OTTRELL material ~3s obtained. and appfoxim:itcly 7 mL piiriilent The spccimcn 1~3s sent to the laboratory for anaerobic and wrohic cultures mntl (;rxm stxin. After the neccllc ;tspirJtion. :I cu134 hemost;it b%s used to further drain the lesion. and 3 biopsy specimen from within the Icsion was taken for microscopic cxamitxition. The surgic;ll site \v;ls then copioiisl~ irrig;ited. hcmostasis wis oht;iined. and ;I quarter-inch Penrose drain w;i5 placed and ligated to the most inferior ;ispect of the incision nith i-0 nylon suture. Additiond gr;ivit;ition;il dr;iinage h2s allowed to occur over the next 3 clays hcfore removal of the drain. The
Discussion of tuberculosis in the United States has increased 20% from 1985 to 1992.” Among the poor of New York. there is estimated to be 235 cases per 100.000 people. I” This sharp increase is attributable to the HIV epidemic in the city. and to the expanding immigr;mt population. Extrapulmonary tuberculosis. with or without pulmonary involvement, now accounts for approximately ~0% of the total c:lses,” with most concentrated among racial ethnic minorities and in the foreign-born population. The parotid is the most frequently involved salivaq gland, with most infections starting in the parotid lymph nodes. These inflamed nodes may become necrotic and develop into a subacute infection or an abscess. Ilsu;~lly there will be no evidence of pulmonary involvement. In patients with cervical TB, concurrent pulmonary lesions are present in less than 15%. The paucity of patients with cervical TB and concurrent pulmonq TB may reflect that tuberculosis has now become a more localized disease. II Therefore, the clinician can The incidence

no longer rely on the presence of pulmonary lesions to suggest ;I mycobacterium infection. and must now include tuberculosis in the differential diagnosis in patients who present with parotid and other neck masses or abscesses and who are at risk for tuberculous infections. Cervical tuberculous lymphadenitis (scrofida) is a tuberculosis infection that involves the submaxillary and cervical lymph nodes. The formation of a granulomatous lesion or abscess can manifest in the head and nrck region unilaterally or bilaterally. The increase in the number of cases of tuberculosis should be of interest to the oral and maxillofacial surgeon because of the large number of patients who will have extrapulmonary involvement and head and neck manifestations. In patients who are referred with ;1 subacute infectious process or a mass in the head and neck, TB infection needs to be included in the differential diagnosis. especially if they are recent immigrants or known HIV patients. In many reported ases. the presumptive diagnosis was ;1 neoplastic process. and the diagnosis of TB was made only after a biopsy.

References 1. Robbin

SL. Angdl .\I. Eumar V. Basic I’:~thology (rd 3). 1’hil;tdclphia. I’.\. Satmdcrs. 19X 1, p 42 2. Lunch ,MA. Brightman VI. Grew-nhcrg ?vlS: Burkrtts Oral Mcdicinc (cd 8). I’hilxlelphia. PA, Lippincott. 198-k. p 655 of Oral 5. Shafer WC;. Hiw MK:. LC\T BM (cds): A Tcstbod Pathology (cd -1). 1’hiladclplli;t. PA. S:mndrrs. 19X3, pp %-I--.%39 4. Shafer WC;. Hint ME. Le\7’ BM (4s): h Testhook of Or.11 I?tthoIogy (c-d -I). PhilxJelph~:t. PA. Saunders. 19X5. p 390 i. Suit HI). et al: Cancc-r Manual (cd 8). Boston. X4. Americ;m C;rncrr Sockty, 1990. p 321 D. et RI: Histiocvtic 6. Mcnascc LP. Bane-rjc-c- SS, Etlmondson nwrotizing Iymphxlcntitis (~;iliuchi-Fujimoto disease): Contkuing diqqostic ditlicultic-s. Histopathology 33:2+KLi+. 1998 Lcrosq Y. Lcclcr-ScarwIld V. Francois A. et al: A pscudtr tumoral form of Kikuchi‘s disc;tsc in children: A case report and rcvicw of the litctxturc. Int .I Pcdiatr Otolatygol ii: 1, 1998 H. O‘Ncill I). O’Grdy J. Vxiend S: Child btality associated with p;tthologic;tl fbturc-s of histiocytic nrcrotizing Iymphadenitis (Kiliuchi-Fttjimoto &xxx). Pcdkttr I’athol L;lh ~Med 18:‘9-88. 1998 9. C;mtwdI MF. Snider DE. Cwther GM. et al: Epidemiology of tuhcrculosis in the Ilnitd St;ltcs. 1985 through 1992. .IXMA L’L:i~i. 1994 diagnostics target TB. Biotechnol10. Yule A: Amplification-lusd ogy 1213.35. 1994 G: Tulxrculosis infections of the head and 11. 1.~; KC. Schuwr neck. Enr Nose ThroatJ ‘-1:?95. 1995