Indian Journal of Surgery Medknow Publications on behalf of Association of Surgeons of India ISSN: 0972-2068 Vol. 65, Num. 2, 2003, pp. 168-171

Indian Journal of Surgery, Vol. 65, No. 2, March-April, 2003, pp. 168-171

Non-neoplastic salivary gland disease with reference to minor salivary gland tuberculosis

Mallika Tewari, H. S. Shukla, Mohan Kumar,* O. P. Sharma**

Division of Surgical Oncology, Departments of *Pathology and **Radiology, Institute of Medical Sciences, Banaras Hindu University, Varanasi 221005
Address for correspondence Prof. H. S. Shukla 7, SPG Colony, Lanka, Varanasi 221005. E-mail: harishukla@usa.net

Paper Received: July 2002, Paper Accepted: November 2002. Source of Support: Nil

Code Number: is03029

ABSTRACT

Objective: To evaluate incidence, type and treatment of non-neoplastic salivary lesions. Design: Retrospective case record review, follow-up review and literature review. Setting: University Hospital, Banaras Hindu University a tertiary care centre. Patients: All patients with non-neoplastic salivary lesions presenting to the Surgical Oncology Unit from July 1997 to Dec 2001. Results: There were 31 non-neoplastic salivary gland lesions. Age of the patients varied from 22-62 years, median age 42 years, and male female ratio 1.2:1.0. The parotid gland had 17, the submandibular gland had 13 and minor salivary glands had 1 lesion. 7 abscesses, 5 cysts, 3 stones and 1 vascular malformation were seen in the parotid gland. 8 cases of adenitis and 5 Wharton's duct stone were observed in the submandibular gland. Tuberculosis was found in a single minor salivary gland lesion. A combination of clinical examination and USG was diagnostic for abscess, adenitis and calculus disease. FNAC did not detect minor salivary gland tuberculosis. The most common operative procedure for the submandibular salivary gland was the excision of the gland for recurrent adenitis. For Wharton's duct stone it was marsupialization. In the parotid gland, incision and drainage for abscess, and superficial parotidectomy for cysts were the common operative procedures. Conclusion: A variety of non-neoplastic lesions occur in the salivary glands. Clinical examination, assisted by USG and FNAC is diagnostic. A complete excision in case of the sumandibular gland and a conservation excision in case of the parotid gland, with careful preservation of the facial nerve, constitute the modality of treatment in these cases.

KEYWORDS: Salivary gland, non-neoplastic, tuberculosis

How to cite this article: Tewari M, Shukla HS, Kumar M, Sharma OP. Non-neoplastic salivary gland disease with reference to minor salivary gland tuberculosis. Indian J Surg 2003;65:168-71.

INTRODUCTION

There are a variety of non-neoplastic disorders affecting the salivary gland. These include chronic sialadenitis, tuberculosis, sarcoidosis, animal scratch disease, actinomycosis and Sjogren's syndrome¹ and require differential diagnosis and management. The non-neoplastic parotid salivary gland disease (NNPD) makes up for about 25% of cases for which parotidectomy is indicated.² The NNPD is of two types, type I is characterised by asymptomatic soft diffuse enlargement of the entire gland or a circumscribed firm nodular enlargement and type II is characterized by an inflammatory lesion with recurrent pain and swelling which may be obstructive or non-obstructive² in nature. The possibility of a neoplastic lesion is the highest in type I nodular lesions. Indication for operation of NNPD is to exclude a tumour, relief of recurrent pain and swelling and to relieve anxiety of the patient. In a series of 62 patients of NNPD, superficial parotidectomy was performed² with complete relief to the patient. This is a review of the non-neoplastic salivary gland lesions including NNPD presenting to one surgeon.

MATERIAL AND METHODS

All patients with salivary gland disease presenting to the Surgical Oncology Unit, SS Hospital, Banaras Hindu University (BHU), Varanasi, between July 1987 to Dec 2001 were included in this study of retrospective clinical and pathological data. The clinical findings, results of investigations, USG of the parotid salivary gland, FNAC and histology findings, and treatment and follow-up observations were entered on a computer database and analyzed for age, sex, site of lesion, treatment, postoperative complications and follow-up results. Frequency tables and percentage inferences were calculated to ascertain the characteristics of type, site, pathology and distribution of non-neoplastic salivary gland diseases presenting to the surgeon.

RESULTS

During the period between July 1987 to Dec 2001, 150 patients with salivary gland disease were seen; out of these 31 patients (20.6%) presented with non-neoplastic salivary disease, 72 (48%) had mixed parotid tumours and 47 (31.3%) had malignant salivary lesions. This report is about the 31 patients with non-neoplastic salivary diseases. The parotid salivary gland had 17 non-neoplastic lesions, most frequent being parotid abscess. The submandibular salivary gland had 13 non-neoplastic lesions submandibular adenitis (in 8 patients) was the commonest. Tuberculosis of a minor salivary gland in the hard palate was found in one patient (Table 1).

The parotid salivary gland abscess presented with a painful swelling in 3 patients and painless gradual enlargement with recurrent pain in 4 patients. Attempt at FNAC revealed pus in all the 7 patients. The parotid salivary gland cysts varied from 1-3 cm in size, of a painless lump of 1-7 years duration. FNAC revealed a clear fluid in all patients. In one patient a deep sinus in the parotid gland, 2 cm in front of the left tragus without any discharge was seen. The sinus tract was lined by squamous epithelium. Parotid duct stone was found in 3 patients. The diagnosis was confirmed on parotid sialography and X-ray examination of the parotid duct. A diffuse large enlargement of the parotid was revealed to be due to vascular malformation in the parotid gland. Large venous channels were found inside the parotid gland requiring multiple ligations.

Recurrent submandibular salivary gland adenitis was present in 8 patients. No specific inflammatory lesion of the submandibular salivary gland was found. In 5 additional patients, a stone in the Wharton's duct was found; recurrent pain during mastication was a dominant feature of submandibular salivary gland swellings. The diagnosis was confirmed on bimanual palpation and X- ray of the Wharton's duct.

In one patient a 2-cm smooth non-tender lump of 1-year duration was present on the hard palate. Wide excision was done. Histopathology revealed it to be tuberculosis in a minor salivary gland. The patient did not have any other focus of tuberculosis. Anti-tubercular treatment was given for 6 months. Treatment details are shown in Table 2.

DISCUSSION

Non-neoplastic lesions of the parotid are not uncommon. Zbaren et al³ in 228 parotidectomies found 13 non-neoplastic lesions, 65 malignant tumours and 150 benign neoplasms. Arshad⁴ found 13 non-neoplastic lesions, 24 malignancies and 73 pleomorphic adenomas of the parotid, in 110 parotid swellings. In our study, 31 non-neoplastic salivary lesions were found in 150 cases. Differential diagnosis of non-neoplastic salivary lesions is required to exclude benign and malignant salivary tumours. Differentiation on clinical grounds alone is not accurate.

Fine needle aspiration cytology (FNAC) of a parotid mass, to distinguish neoplastic lesion from inflammatory mass is very useful.⁵ In a study of 160 salivary gland "tumours", 84 parotid, 70 submandibular and 6 accessory (submucosal) glands, in 155 lesions (97%) adequate material for histological evaluation was obtained. 12 of these patients had lymphoma, 10 patients tuberculosis and 30 benign lympho-epithelial lesion of the parotid with 3 false positive and 3 false negative results. No complications such as haematoma nerve injury or infection occurred following the FNAC.⁵ In this series of patients, fine needle aspiration was also diagnostic for parotid abscess and could exclude malignancy in parotid cysts. FNAC was not done when clinical diagnosis was obvious such as in recurrent adenitis and stone disease. In patients of minor salivary gland tuberculosis, FNAC revealed necrosed tissue. FNAC diagnosis of the parotid salivary gland tuberculosis followed by anti-tubercular therapy can avoid the need for surgery in this rather rare site for tuberculosis.⁵

Tuberculosis (TB) of the parotid gland is an uncommon form of infection reported either as a single case or as a series of 4-6 cases in literature from all over the world.^6-14 Reports of salivary gland TB from high incidence areas of the world are only a few. The proportion of parotid salivary gland tuberculosis, primary or secondary, was 14.3% in 42 case reports of oro-facial tuberculosis.¹⁴ The parotid gland TB presents as a neoplasm and most patients undergo parotidectomy. In rare instances, diffuse tubercular infection of the whole gland is found.^8,13 TB of the parotid gland has no characteristic feature hence confirmation of diagnosis by computerized tomography (CT) is required. The presence of a thick, walled rim enhancing lesions with a central lucency on contrast enhanced CT (CECT) is characteristic of TB of the parotid gland.¹⁵ TB of the parotid gland is treated by parotidectomy with preservation of the facial nerve, either a superficial parotidectomy or an enucleation.^7,10,11 Pathogenesis of parotid salivary gland TB appears to be tuberculous infection occurring in a deep parotid lymph node and involving the surrounding parotid tissue.

No report of TB of the accessory salivary gland is available in the current indexed medical literature. Submandibular salivary gland TB is also not reported though there is a rare report of submandibular tuberculous lymphadenitis.¹⁶

Stones present in the Stensen's duct in 3 patients were removed by opening the duct. Those from the Wharton's duct seen in 5 patients were treated by open surgical removal of the stone by marsupialization. There were no complications. A combination of imaging methods including plain X-ray films, ultrasonography, CT scan and sialography and endoscopy are required and helpful in location of sialolithiasis.¹⁷ Endoscopic laser lithotripsy of proximal parotid duct stone¹⁸ is effective. The modern technique of extra corporeal lithotripsy (ECL) was used by Schlegel et al¹⁹ in 27 cases (10 Stensen's, 17 Wharton's duct stone) of salivary calculus. The lithiasis was more than 2 mm. After single ECL treatment the stone disappeared in 9 patients (33.3%), the stone was fragmented and partially removed in 10 cases (37.0%). In 8 cases (29.6%) there was complete failure to fragment the stone.

Conventional surgical treatment comprising incision of the parotid parenchyma in the direction of the facial nerve until the abscess is located and evacuated, was employed in 7 patients of parotid abscess. Intraoperative ultrasound is helpful in localizing the abscess and in ensuring its complete drainage.²⁰ Ultrasound-guided percutaneous catheter drainage of parotid abscess is an effective alternative method of treatment.²¹ Sump catheter drainage of parotid abscess is an alternative to surgery.²² The accessory ectopic parotid gland is occasionally the seat of a parotid abscess.²³ An abscess was not found in any of the patients with submandibular adenitis. Submandibular salivary gland adenectomy is usually carried out in all such cases.

ACKNOWLEDGEMENT

Help of Mr. Anil K Pandey, MSc, Computer programmer, Division of Surgical Oncology, IMS, BHU in carrying out the computer database analysis, is acknowledged.

REFERENCES

1. Rice DH. Chronic inflammatory disorders of the salivary glands. Otolaryngol Clin North Am 1999;32:813-8.
2. Rosen IB, McHenry CR, Bedard YC. Surgical treatment in non-neoplastic parotid disease: indications and results. Can J Surg 1992;35:560-4.
3. Zbaren P, Schar C, Hotz MA, Loosli H. Value of fine needle aspiration cytology of parotid gland masses. Laryngoscope 2001;111:1989-92.
4. Arshad AR. Parotid swellings: report of 110 consecutive cases. Med J Malaysia 1998;53:417-22.
5. Shaha AR, Webber C, DiMaio T, et al. Needle aspiration biopsy in salivary gland lesions. Am J Surg 1990;160:373-6.
6. Bundgaard N, Hansen IM. Parotid Tuberculosis. Ugeskr Laeger 1990;152:747-8.
7. Ataman M, Sozeri B, Ozcelik T, Gedikoglu G. Tuberculosis of the parotid salivary gland. Auris Nasus Larynx 1992;19:271-3.
8. Rowe-Jones JM, Vowles R, Lighton SE, Freedman AR. Diffuse tuberculous parotitis. J Laryngol Otol 1992;106:1094-5.
9. Esteban Sanchez T, Redondo Lucianez ER, Perez Martinez F, Rodriguez Herrero D, Galindo Leon A, Calero del Castillo
   JB. Tuberculosis of the parotid gland. An Otorrinolaringol Ibero Am 1993;20:361-7.
10. O' Connell JE, George MK, Speculand B, Pahor AL. Mycobacterial infection of the parotid gland: an unusual cause of parotid swelling. J Laryngol Otol 1993;107:561-4.
11. Comeche Cerveron C, Barona de Guzman R, Martinez Leandro E. Parotid Tuberculosis. Acta Otorrinolaringol Esp 1995;46:387-90.
12. Weiner GM, Pahor AL. Tuberculous parotitis: limiting the role of surgery. J Laryngol Otol 1996;110:96-7.
13. Franzen A, Franzen CK, Koegel K. Tuberculosis of parotid gland: a rare differential diagnosis of parotid tumor. Laryngorhinootologie 1997;76:308-11.
14. Mignogna MD, Muzio LL, Favia G, Ruoppo E, Sammartino G, Zarrelli C, et al. Oral tuberculosis: a clinical evaluation of 42 cases. Oral Dis 2000;6:25-30.
15. Bhargava S, Watmough DJ, Chisti FA, Sathar SA. Case report: tuberculosis of the parotid gland-diagnosis by CT. Br J Radiol 1996;69:1181-3.
16. Bodner L, Lewin-Epstein J, Shteyer A. Submandibular tuberculous lymphadenitis (scrofula): report of two cases. J Oral Maxillofac Surg 1990;48:192-6.
17. Karas ND. Surgery of the salivary ducts. Atlas Oral Maxillofac Surg Clin North Am 1998;6:99-116.
18. Kerr PD, Krahn H, Brodovsky D. Endoscopic laser lithotripsy of a proximal parotid duct calculus. J Otolaryngol 2001;30:129-30.
19. Schlegel N, Brette MD, Cussenot I, Monteil JP. Extracorporeal lithotripsy in the treatment of salivary lithiasis. A prospective study apropos of 27 cases. Ann Otolaryngol Chir Cervicofac 2001;118:373-7.
20. Graham SM, Hoffman HT, McCulloch TM, Funk GF. Intra-operative ultrasound-guided drainage of parotid abscess. J Laryngol Otol 1998;112:1098-100.
21. Yeow KM, Hao SP, Liao CT. Ultrasound-guided percutaneous catheter drainage of parotid abscesses. J Vasc Interv Radiol 2000;11:473-6.
22. Breman J, Myssiorek D, Reppucci A, Zito J. Sump catheter drainage of parotid abscess: an alternative to surgery. Ear Nose Throat J 1991;70:393-5.
23. Lee DH. A case of buccal abscess; originating from an ectopic accessory parotid gland? J Laryngol Otol 2002;116:312-3.

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