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Bilateral and multicystic major salivary gland disease: a rare presentation of primary Sjögren's syndrome

Published online by Cambridge University Press:  29 June 2007

Ijaz Ahmad*
Affiliation:
Department of Otolaryngology, Dudley Group of Hospitals, Russells Hall Hospital, Dudley, West Midlands, UK.
J. Ray
Affiliation:
Department of Otolaryngology, Dudley Group of Hospitals, Russells Hall Hospital, Dudley, West Midlands, UK.
R. J. Cullen
Affiliation:
Department of Otolaryngology, Dudley Group of Hospitals, Russells Hall Hospital, Dudley, West Midlands, UK.
R. T. J. Shortridge
Affiliation:
Department of Otolaryngology, Dudley Group of Hospitals, Russells Hall Hospital, Dudley, West Midlands, UK.
*
Address for correspondence: Ijaz Ahmad, F.R.C.S., Department of Otolaryngology, Russells Hall Hospital, Dudley DY1 2HQ. Fax: 01384 244169

Abstract

We present a case of a 15-year-old girl with bilateral parotid and sub-mandibular salivary gland enlargement as the sole presentation of primary Sjögren's syndrome. The clinical, radiological, immunological and pathological features have been discussed. The relevant literature has been reviewed. To our knowledge this is the only reported case of Sjögren's syndrome presenting as multicystic disease with bilateral major salivary gland involvement.

Type
Clinical Records
Copyright
Copyright © JLO (1984) Limited 1998

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References

Bradus, R. J., Hybarger, P., Gooding, G. A. W. (1988) Parotid gland: US findings in Sjögren syndrome. Radiology 169: 749751.CrossRefGoogle ScholarPubMed
Flescher, E., Talal, N. (1991) Do viruses contribute to the development of Sjögren's syndrome? American Journal of Medicine 90: 283285.CrossRefGoogle Scholar
Fox, R. I., Howell, F. V., Bone, R. C., Michelson, P. (1984) Primary Sjogren's syndrome: Clinical and immunopathologic features. Seminars in Arthritis and Rheumatism 14: 77105.CrossRefGoogle ScholarPubMed
Hong, S. S., Ogawa, Y., Yagi, T., Wakasak, K., Sakurai, M., Sato, M., Harada, T. (1990) Benign lymphoepithelial lesion with large cysts: Case report. Journal of Oral Pathology and Medicine 19: 266270.CrossRefGoogle ScholarPubMed
Hudson, N. P. (1986) Manifestation of systemic diseases. In Otolaryngology-Head and Neck Surgery Vol. 2 (Cummings, C. W., Fredrickson, J. M., Harker, L. A., eds.), Mosby Co., St Louis, pp 10071013.Google Scholar
Hurt, D. F., Burroughs, J. (1985) Sjögren's syndrome in a young adult. Journal of Oral and Maxillofacial Surgery 43(10): 819821.CrossRefGoogle ScholarPubMed
Jones, J. E., Mason, D. R. (1980) Oral Manifestations of Systemic Disease. W. B. Saunders, Philadelphia, pp 194.Google Scholar
Kassau, S. S. (1978) Increased risk of lymphoma in sicca syndrome. Annals of Internal Medicine 89: 888.CrossRefGoogle Scholar
Lindvall, A. M., Jonsson, R. (1986) The salivary gland component of Sjögren's syndrome: An evaluation of diagnostic methods. Oral Surgery, Oral Medicine, Oral Pathology 62: 3242.CrossRefGoogle ScholarPubMed
March, D. E., Rao, V. M., Zwillenberg, D. (1989) Computed tomography of salivary glands in Sjögren's syndrome. Archives of Otolaryngology, Head and Neck Surgery 115: 105106.CrossRefGoogle ScholarPubMed
Martinez, L. M., Vaughan, J. H., Tan, E. M. (1979) Autoantibodies and the spectrum of Sjögren's syndrome. Annals of Internal Medicine 912: 185190.CrossRefGoogle Scholar
Saito, T., Fukuda, H., Horikawa, M., Shindoh, M., Amemiya, A. (1994) Sjogren's syndrome in the adolescent. Report of four cases. Oral Surgery, Oral Medicine, Oral Pathology 77(4): 368372.CrossRefGoogle ScholarPubMed
Som, P. M., Brandwein, M. S., Slivers, A. (1995) Nodal inclusion cysts of the parotid gland and parapharyngeal space: a discussion of lymphoepithelial, Aids-related parotid and branchial cysts, cystic Warthin's tumors, and cysts in Sjögren's syndrome. Laryngoscope 105(10): 11221128.CrossRefGoogle ScholarPubMed
Som, P. M., Shugar, J. M. A., Biller, H. T. (1981) Manifestation of parotid gland enlargement: radiographic, pathologic and clinical correlation. Radiology 141: 415419.CrossRefGoogle Scholar
Schmid, U., Helbron, D., Lennert, K. (1982) Development of malignant lymphoma in myoepithelial sialedenitis (Sjögren's syndrome). Virchows Archiv. A, Pathological Anatomy and Histopathology 395(1): 1143.CrossRefGoogle Scholar
Takashima, S., Takeuchi, N., Morimoto, S., Tomiyama, N., Ikezoe, J., Shogen, K., Kozuka, T., Okumura, T. (1991) MRI imaging of Sjögren's syndrome: correlation with sialography and pathology. Journal of Computer Assisted Tomography 15: 393400.CrossRefGoogle ScholarPubMed
Yamagata, H., Akizuki, M., Tojo, T., Homma, M. (1986) Anti-Ro/SSA and -La/SSB Antibodies in patients with connective tissue diseases. Scandanavian Journal of Rheumatology 61: 98101.Google ScholarPubMed