Abstract
The aim of this study was to investigate the performance of minor salivary gland biopsy (MSGB), serological and clinical data in diagnosis of primary Sjögren’s syndrome (pSS). Retrospective review of 216 patients who underwent minor labial salivary gland biopsy in last 5 years was performed. Results of the patients with diagnosis of pSS were compared with the patients failing to fill the classification criteria of pSS. Two groups did not differ significantly in terms of clinical symptoms and signs except presence of Raynaud’s phenomenon. Specificity and positive likelihood ratio of clinical signs in diagnosis of pSS were quiet low. A total of 78.7% of pSS patients had a focus score ≥1 (Chiscolm’s score III/IV) while all of the non-SS patients had a focus score <1 (P < 0.001). MSGB has the best predictive value with highest sensitivity and specificity for pSS diagnosis. Serological markers have higher predictive values compared to clinical symptoms and signs. Presence of Raynaud’s phenomenon, lymphopenia and/or hypergammaglobulinemia strengthens the probability of pSS in a patient with sicca symptoms.
Similar content being viewed by others
References
Mavragani CP, Moutsopoulos NM, Moutsopoulos HM (2006) The management of Sjögren’s syndrome. Nat Clin Pract Rheumatol 2:252–261
Vitali C, Bombardieri S, Moutsopoulos HM, Balestrieri G, Bencivelli W, Bernstein RM, Bjerrum KB, Braga S, Coll J, de Vita S et al (1993) Preliminary criteria for the classification of Sjögren’s syndrome. Results of a prospective concerted action supported by the European Community. Arthritis Rheum 36:340–347
Vitali C, Bombardieri S, Moutsopoulos HM et al (1996) Assessment of the European classification criteria for Sjögren’s syndrome in a series of clinically defined cases: results of a prospective multicentre study. The European Study Group on Diagnostic Criteria for Sjögren’s Syndrome. Ann Rheum Dis 55:116–121
Vitali C, Bombardieri S, Jonsson R, Moutsopoulos HM, Alexander EL, Carsons SE, Daniels TE, Fox PC, Fox RI, Kassan SS, Pillemer SR, Talal N, Weisman MH, European Study Group on Classification Criteria for Sjögren’s Syndrome (2002) Classification criteria for Sjögren’s syndrome: a revised version of the European criteria proposed by the American-European Consensus Group. Ann Rheum Dis 61:554–558
Fox RI, Robinson CA, Curd JG, Kozin F, Howell FV (1986) Sjögren’s syndrome. Proposed criteria for classification. Arthritis Rheum 29:577–585
Manthorpe R, Oxholm P, Prause JU, Schiødt M (1986) The Copenhagen criteria for Sjögren’s syndrome. Scand J Rheumatol Suppl 61:19–21
Homma M, Tojo T, Akizuki M, Yamagata H (1986) Criteria for Sjögren’s syndrome in Japan. Scand J Rheumatol Suppl 61:26–27
Guobis Z, Baseviciene N, Paipaliene P, Sabalys G, Kubilius R (2006) Xerostomia: clinic, etiology, diagnosis and treatment. Medicina (Kaunas) 42:171–179
Perry HD, Donnenfeld ED (2004) Dry eye diagnosis and management in 2004. Curr Opin Ophthalmol 15:299–304
Langerman AJ, Blair EA, Sweiss NJ, Taxy JB (2007) Utility of lip biopsy in the diagnosis and treatment of Sjogren’s syndrome. Laryngoscope 117:1004–1008
Chisholm DM, Mason DK (1968) Labial salivary gland biopsy in Sjogren’s disease. J Clin Path 21:656–660
Greenspan JS, Daniels TE, Talal N, Sylvester RA (1974) The histopathology of Sjogren’s syndrome in labial salivary gland biopsies. Oral Surg Oral Med Oral Pathol 37:217–229
van den Berg I, Pijpe J, Vissink A (2007) Salivary gland parameters and clinical data related to the underlying disorder in patients with persisting xerostomia. Eur J Oral Sci 115:97–102
Pleşivenik M, Rozman B, Hocevar A, Grmek M, Kveder T, Tomsic M (2004) Incidence of primary Sjögren syndrome in Slovenia. Ann Rheum Dis 63:874–876
Sreebny LM, Valdini A (1987) Xerostomia, a neglected symptom. Arch Intern Med 147:1333–1337
Kohler P, Winter M (1985) A quantitative test for xerostomia. Arthritis Rheum 28:1128–1132
Rubin P, Holt JF (1957) Secretory sialography in diseases of the major salivary glands. AJR Am J Roentgenol 77:575–598
de Azevedo BB, Bussoloti Filho I (2005) Evaluation of sialometry and minor salivary gland biopsy in classification of Sjögren’s syndrome patients. Rev Bras Otorrinolaringol (Engl Ed) 71:346–354
Yonetsu K, Takagi Y, Sumi M, Nakamura T, Eguchi K (2002) Sonography as a replacement for sialography for the diagnosis of salivary glands affected by Sjögren’s syndrome. Ann Rheum Dis 61:276–277
Papiris SA, Maniati M, Constantopoulos SH, Roussos C, Moutsopoulos HM, Skopouli FN (1999) Lung involvement in primary Sjögren’s syndrome is mainly related to the small airway disease. Ann Rheum Dis 58:61–64
Diaz-Lopez C, Geli C, Corominas H et al (2004) Are there clinical or serological differences between male and female patients with primary Sjögren’s syndrome? J Rheumatol 31:1352–1355
Meyer O (2003) Evaluating inflammatory joint disease: how and when can autoantibodies help? Joint Bone Spine 70:433–447
Gerbracht DD, Steen VD, Ziegler GL, Medsger TA Jr, Rodnan GP (1985) Evolution of primary Raynaud’s phenomenon (Raynaud’s disease) to connective tissue disease. Arthritis Rheum 28:87–92
García-Carrasco M, Sisó A, Ramos-Casals M, Rosas J, de la Red G, Gil V, Lasterra S, Cervera R, Font J, Ingelmo M (2002) Raynaud’s phenomenon in primary Sjögren’s syndrome. Prevalence and clinical characteristics in a series of 320 patients. J Rheumatol 29:726–730
Skopouli FN, Talal A, Galanopoulou V, Tsampoulas CG, Drosos AA, Moutsopoulos HM (1990) Raynaud’s phenomenon in primary Sjögren’s syndrome. J Rheumatol 17:618–620
Kraus A, Caballero-Uribe C, Jakez J, Villa AR, Alarcón-Segovia D (1992) Raynaud’s phenomenon in primary Sjögren’s syndrome. Association with other extraglandular manifestations. J Rheumatol 19:1572–1574
Pennec Y, Youinou P, Mottier D, Jouquan J, Colin F, Le Menn G (1984) Gougerot-Sjögren syndrome and monoclonal immunoglobulin. Rev Med Intern 5:303–307
Brennan MT, Sankar V, Leakan RA, Kleiner D, Atkinson JC, Wilkinson WE, Baum BJ, Pillemer SR (2002) Risk factors for positive minor salivary gland biopsy findings in Sjögren’s syndrome and dry mouth patients. Arthritis Rheum 47:189–195
Aoki A, Ohno S, Ueda A, Ideguchi H, Ohkubo T, Hagiwara E, Shirai A, Oketani M, Nagaoka S, Senuma A, Ohota S, Matsunaga K, Ishigatsubo Y (2000) Hematological abnormalities of primary Sjogren’s syndrome. Nihon Rinsho Meneki Gakkai Kaishi 23:124–128
Alexander EL, Ranzenbach MR, Kumar AJ, Kozachuk WE, Rosenbaum AE, Patronas N, Harley JB, Reichlin M (1994) Anti-Ro(SS-A) autoantibodies in central nervous system disease associated with Sjögren’s syndrome (CNS-SS): clinical, neuroimaging, and angiographic correlates. Neurology 44:899–908
Gökçay F, Celebisoy N, Gökçay A, Kabasakal Y, Oder G (2007) Primary Sjögrens syndrome presenting as neuromyelitis optica. Pediatr Neurol 36(1):58–60
Volter F, Fain O, Mathieu E, Thomas M (2004) Esophageal function and Sjögren’s syndrome. Dig Dis Sci 49:248–253
Acknowledgments
The study was supported by Akdeniz University Scientific Research Projects Unit.
Conflict of interest statement
The authors have declared no conflict of interest.
Author information
Authors and Affiliations
Corresponding author
Rights and permissions
About this article
Cite this article
Yazisiz, V., Avci, A.B., Erbasan, F. et al. Diagnostic performance of minor salivary gland biopsy, serological and clinical data in Sjögren’s syndrome: a retrospective analysis. Rheumatol Int 29, 403–409 (2009). https://doi.org/10.1007/s00296-008-0698-1
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00296-008-0698-1