J Oral Maxillofoc Surg 56:676x580, 1998 Pneumoparotid: Report of a Case Rafael E. Alcalde, DDS, PbD, * Yoshiya Ueyama, DDS, PbD, f DarwinJ Lim, DDM,,f and Tomohiro Matsumura, DDS, PbDJ Parotid gland swellings are usually caused by viral and bacterial infection. They also may result from obstructive, inflammatory, metabolic, neoplastic, and other pathologic conditions1 Among the nonneoplas- tic swellings, pneumoparotid is characterized by the presence of air within the duct and the parenchyma of the gland. This pneumatic enlargement usually results from the forced entry of air into the parotid gland acini through Stensenâs duct due to an increase of intraoral pressure.2 Pneumoparotid can lead to pneumoparoti- tis when pain and persistent swelling occur. This report describes a case of pneumoparotid and discusses its cause, diagnosis, and management. Report of Case A 29-year-old-man came to our outpatient clinic complain- ing of pain on the right side of his face, slight swelling in me parotid region, bilateral clicking of the temporomandibular joint (TMJ), bilateral tinnitus, and headache. All of these signs and symptoms apparently appeared after trauma to the head and maxillofacial region that occurred during a trahic accident 4 years ago. The right parotid swelling had in- creased in size in the last 3 months and became painful. The patient neither played a wind instrument nor was he a glassblower, and he denied any nervous tic or habit of blowing out his cheek, as well as any recent dental treat- ment. Physical examination showed a round swelling of the right parotid region. It was painful to palpation, but the overlying skin had a normal appearance (Fig 1). The TMJ symptoms and the headache were still present. Intraoral Received from the Department of Oral and Maxillofacial Surgery II, Okayama University Dental School, Okayama, Japan. *Graduate Student. $-Assistant Professor. *Graduate Student. §Professor and Chairman. Address correspondence and reprint requests to Dr Alcalde: Department of Oral and Maxillofacial Surgery II, Okayama Univer- sity Dental School, Shikata-cho 2-5-l, Okayama-shi 700, Japan. o 1998 American Association of Oral and Maxillofacial Surgeons 0278.2391/98/5605-0020$3.00/O examination showed crepitation and a slightly dilated Stens- enâs duct opening with bubbly salivary discharge. Com- puted tomography (CT) showed a round, well-circum- scribed radiolucency compatible with air within the parotid gland (Fig 2). Stensenâs duct displayed multiple strictures and dilations along its course in a sialogram taken at the first appointment and in a CT sialogram obtained 1 week after the initial examination (Pig 3). A needle aspiration was performed at the first appoint- ment, extracting 10 mL air. The diagnoses of pneumoparotitis and TMJ dysfunction were made. Symptomatic treatment using nonsteroidal anti- inflammatory drugs and antibiotics led to partial relief of the tenderness in the right parotid region and the headaches. The bilateral TMJ noise was reduced by bite appliance therapy, but the patient refused further treatment or obser- vation. Discussion According to the patientâs clinical history, trauma received during a trathc accident was the only predis- posing factor for the pneumoparotid. The problem seems to have originated from the previous traumatic episode for unknown reasons. Thereafter, a slight increase of pressure produced by a cough, sneeze, speech, or other unconscious act may have been the cause of the increased swelling present at the time of consultation. Pneumoparotid may be intentionally created, and has been associated with habits and abnormal psycho- logic behavior, mainly in children and teenagers (Table 1). Self-induced pneumoparotid differs from the unconscious type related to occupational hazards and previous traumatic episodes (Table 2). Acciden- tally induced pneumatic swellings, such as those caused by the use of air syringes during dental treatment, and by trauma, are frequently unilateral because they are a consequence of high pressure in localized areas. However, swellings generated by high total intraoral pressure, including self-induced cases, may be either unilateral or bilateral. Because of the 676 ALCALDE ET AL 677 FIGURE 1. Appearance of patient. Note the moderate swelling in the right parotid region. multiple causes of these swellings, the clinical history should be considered the most important starting point to obtain the correct diagnosis. Clinical examination may be helpful in diagnosing cases of subcutaneous emphysema in adjacent re- gions.lO Crepitation along the course of the Stensenâs FIGURE 2. Computed tomographic view showing a round, well- circumscribed radiolucent area compatible with the presence of air (approximately 1,000 Hunsfield units) within the right parotid gland. FIGURE 3. Computed tomographic sialogram taken 10 days after aspiration of air still shows multiple ductal dilations and strictures. duct, as well as frothy salivary secretion, indicate the presence of air within the duct or gland.28 Imaging studies also help to confirm the diagnosis because the air in as well as ductal abnormalities are usually detected by standard and CT sialography. A variety of treatments are mentioned in the litera- ture, most of which are directed toward elimination of the causative or predisposing factors that favored the condition. The use of antiinflammatory drugs and antibiotics is a widespread practice because the pneu- moparotid is often associated with pain and superim- posed bacterial infections produced by the retrograde flow of air and contaminated saliva.lO To prevent recurrence, patients with nervous tics or with occupa- tional pneumoparotid should be advised to avoid any activity that may increase intraoral pressure. Psychiat- ric or psychologic therapy has been shown to be necessary in self-induced cases that are usually related to behavioral problems. For cases of chronic and recurrent pneumoparotid, various surgical treatments have been suggested. Among them, transposition of the parotid duct to the tonsillar fossa19,20 or its ligation,* and extirpation of the gland,21 are indicated only after multiple recurrence or repeated episodes of infection indicate an irreversible symptomatic prob- lem. Long-term follow-up should be considered in all cases because repeated air entry may result in perma- nent dilation of the ducts and the subsequent develop- ment of infection, calculi, or the presence of intraglan- dular air. l4 678 PNEUMOPAROTID Author Age/ Chief Complaint/ sex Race (Diagnostic Imaging) Diagnosis Treatment Reason for Treatment 1. Rupp, 1963s 1 l/M W Facial swelling (Sialc- Self-induced pneumo- Not specified Not specified gram) parotid 2. Greisen, 1968* 9/M - Recurrent bilateral Self-induced pneumo- None Not necessary parotid enlargement parotid (X-ray and sialography) 3. Calcaterra and Lowe, 14/M W Painful swelling of the left Self-induced pneumopar- a. Antibiotics a. To treat a misdiag- 19735 parotid gland (X-ray, otid b. Parotid incision nosed bacterial par- fluoroscopy, and sialo c. Psychological therapy OtitiS sram> b. To explore the parotid c. To break the habit 5. Wan, 1977â * -/M - IoRamedrlghtparotid Self-induced pneumc- a. Parotidectomy a. To treat the emphy- gland (Sialogram) parotid b. Psychiatric therapy sema Secondary infection b. To treat the abnormal Emphysema behavior 6. Markowitz-Spence et 12/P - Recurrent bilateral pre- Self-induced pneumo a. Oxacillin a. To treat a misdiag al, 19877 auricular swelling (CT parotid b. Antibiotics and soft nosed acute parotitis scan, CT sialogram, diet b. To avoid secondary and sialogmm) c. Psychological therapy infection c. Not specified 7. David and Kanga, 6.5/F W Crepitant swelling over Pneumoparotid by a ner- a. Percutaneous evacua- a. To remove the air and 1988s the left periauricular vous tic tion of the gland exudate in the duct area that persisted for 9 b. Positive reinforcement and gland for analysis months a-ray) b. To abolish the tic 8. Krief et al, 19929 10/M - Bilateral painful parotid Pneumoparotid by a habit a. Antibiotics a. Not specified gland swelling (CT b. Parotid puncture b. Temporary relief of the scan and sialography) c. Counseling and swelling training c. To break the habit 9. Ferlito et al, 1992tâ 14/M W Recurrent bilateral Pneumoparotid by a ner- a. Antibiotics/antiintlam- a. Symptomatic treat- parotid painful vous tic matory ment of recurrences swelling (X-ray and b. Counseling b. To break the habit ultrasound) 10. Birzgalis et al, 1993iâ 16/M - Painful right parotid a. Pleomorphic adenoma a. Superticial parotidec- a. Excision of the plec- swelling (X-ray and CT b. Self-induced pneumo- tomy morphic adenoma =W parotid b. Counseling b. To break the habit 11. Nassimbeni et al, WM - Recurrent painless epi- Self-induced pneumo- Psychiatric therapy To break the habit 1995â2 sodes of unilateral or parotid bilateral parotid swelling (CT scan) 12. 9/M - Recurrent swelling and Self-induced pneumo a. Antibiotics a. To prevent infection tenderness of the right parotid b. Psychologic therapy b. To break the habit parotid gland &ray Secondary infection c. Lesion puncture c. Remove the air sialogmphy, and ultra- d. Surgical incision d. To drain the abscess sound) 13. Goguen et al, 1995i3 9/M - Recurrent swelling of the Self-induced pneumo- Counseling To break the habit tight parotid gland parotid without pain (CT scan) 14. 9/F - Long history of recurrent Self-induced pneumo- a. Antibiotic/massage a. To treat recurrent bilateral parotitis. parotid b. Counseling swellings Painful swelling of the c. Superficial parotidec- b. To break the habit parotid gland (CT scan) tomy of the left parotid c. Recurrent episodes gland 15. 13/M - Multiple episodes of non- a. Recurrent juvenile par- a. Antibiotics and ste- a. To treat the juvenile tender bilateral otitis roids parotitis swelling of the parotid b. Self-induced pneumc- b. Psychiatric counseling b. To break the habit region @-ray and sialo parotid gram) NOTE. In case 4, Pritchen (1973) reported a case of self-induced pneumoparotid in an adolescent (male). Abbreviations: NSAIDs, nonsteroidal anti-inflammatory drugs; CT, computed tomography; W, white. *wattâs report included only the profession of accidental cases included in Table 2. ALCALDE ET AL 679 Age/ Chief Complaint/ Reason for Author Sex Race (Diagnostic Imaging) Diagnosis Treatment Treatment 1. R&linger, 1965â* 55/M - Intermittent and painful a. Occupational pneumo- a. Conservative treat- a. Not specified swelling of the right partotitis (trumpet ment b. To eliminate the infec- parotid gland (SiaIo- player) b. Duct drainage and cal- tion gram) b. Seecondary obstruc- culi removal tion (calculi) and infec- tion 7. OâHara and Keohane, 19/M W Recurrent sudden a. Tracheobronchitis a. Tetracycline and &or- a. To treat the tracheo- 19731â 8. Watt, 19776 * 11. Byard, 1986râ 12. Garber. 1987is 13. Brodie and Cole, 1988r9 14. Telfer and Irvine, 1989aâ 15. Mandel et al, 1991ai 53/M 16. Piette and WaIker, 199122 17. Takenoshita et al, 199123 18. Curtin et al, 1992* 19. Brown et al, 199224 20. McDuffle et al, 199325 24/M 21. Cook and Layton, 199326 22. Yonetsu et al, 1993aâ 23. Current case, 1996 - 9/M 32/M 14/M 29/M 34/F 24/M 36/M 30/M 64/M 29/M swelling of the left side b. Unconsciously of the face and dii- induced pneumopar- culty in breathing otid e-ray and sialogram) - Recurrent unilateral Pneumoparotid by baro- swelling (Not speci- trauma (diving) fled) - Recurrent acute left Pneumoparotid induced parotid gland swelling by a habit to relieve (Not specified) aphtous ulcer pain while eating - Bilateral parotid swelling Pneumoparotid after an after clearing the nose attack of hay fever during a hay fever attack (Not taken) - Recurrent swelling after Pneumosialadenitis by facial trauma (CT scan barotrauma and sialogram) - Squelching noise and Spontaneous pneumopar- feeling in the right side otid of the face and ear for 2 years (X-ray, sialo- gram, and ultrasound) W Moderate and slightly Unconsciously induced painful swelling of the pneumoparotid right parotid gland (Sialogram) C Painful elastic swelling on Pneumoparotid caused by the right side of the air syringe during face (X-ray) dental treatment - Swelling in the left Pneumoparotid produced parotid region (LIltra- by air syringe during sound) dental treatment - Recurrent bilateral Pneumoparotid caused by parotid swelling (X-ray, a habit to clear his ears sialogram, and ultra- sound) W Unilateral palm%1 swelling Pneumoparotid induced of the left side of the by dental prophylaxis face during a dental prophylaxis (Not speci- fied) B Bilaterally swollen cheeks Pneumoparotitis by habit (Not specified) to relieve irritation from âorthodontic applianceâ - Bilateral painless swelling Pneumoparotid by intra at the parotid @-ray oral pressures needed and sialogram) for respiration in COPD - Swelling in the left bucco- Spontaneous pneumopat- masseteric region otid (X-ray and CT scan) J Pain and swelling on the a. Pneumoparotitis right side of the face (Trauma) (CT scan, CT sialo- b. TMJ dysfunction gram, and sialogram) pheni&ine b. Complete removal of the parotid gland Not specified Counseling Antibiotics and aspirin Transposition of the parotid duct Surgical transposition of the parotid duct counseling Antibiotics Antibiotics Counseling a. NSAIDs b. Antibiotics a. Adjustment of orth- odontic appliance b. Counseling Not specified None a. Antibiotics b. Splint therapy c. NSAIDs bronchitis b. Multiple recurrences Not specified To break the habit Not specified To decrease the risk of autointkation Inefticiency of the bucci- nator sphincter mecha- nism To break the habit To prevent infection To prevent infection To break the habit a. To reduce further edema b. To prevent infection a. To reduce irritation b. To break the habit Not specified The patient refused any treatment a. To avoid secondary infection b. To treat the TMD c. To relieve the symp- toms NOTE. Saunders (1973)r5 reported only the causes of five cases, nos. 2,3,4,5 (wind instrument), and 6 (blowing a balloon). Abbreviations: W, white; C, Chinese; B, black; J, Japanese; CT, computed tomography; NSAIDs, nonsteroidal anti-inflammatory drugs; COPD, chronic obstructive pulmonary disease; TMD, temporomandibular dysfunction. Watt6 reported only case no. 9, a sailor watchkeeping in a high-pressure compartment, and no. 10, a bugler. 680 PNEUMOPAROTID References 15. 16. Saunders HF: Wind parotitis. N Engl J Med 289:698, 1973 OâHara AE, Keohane RB: Sialography in an unusual case of subcutaneous emphysema of the neck. Arch Otolaryngol 98: 354,1973 Byard R: Acute parotid swelling with rapid subsidence in childhood. J Otolaryngol15:67, 1986 Garber Mw: Pneumoparotid: An unusual manifestation of hay fever. Am J Emerg Med 5:40, 1987 Brodie HA, Chole RA: Recurrent pneumosiaiadenitis: A case presentation and new surgical intervention. 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