{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,10]],"date-time":"2026-07-10T13:22:41Z","timestamp":1783689761860,"version":"3.55.0"},"reference-count":15,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2024,7,3]],"date-time":"2024-07-03T00:00:00Z","timestamp":1719964800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,7,3]],"date-time":"2024-07-03T00:00:00Z","timestamp":1719964800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Head and Neck Pathol"],"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n                <jats:title>Purpose<\/jats:title>\n                <jats:p>There are a number of diagnostic criteria that can be used to support a diagnosis of Sj\u00f6gren\u2019s syndrome (SS), a chronic autoimmune condition often characterised by xerostomia and xerophthalmia. Of the available investigations, the most invasive is the labial gland biopsy (LGB) for histopathology, which is associated with a risk of long-term altered sensation to the lip. A positive histological diagnosis is currently considered to be one of the most objective criteria, however there is debate about the interobserver agreement between pathologists, as well as the sensitivity and specificity of this test. We aim to determine if the diagnostic value of the LGB is significant enough to warrant the surgical procedure and its associated risks.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Methods<\/jats:title>\n                <jats:p>This study involved assessing the degree of agreement between members of a pathology team for a cohort of 50 LGBs taken for the purpose of confirming or excluding SS. The Tarpley system was used, which involves the allocation of a \u2018focus score\u2019. Additionally, the histological diagnoses were compared to the relevant serological findings where available.<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Results<\/jats:title>\n                <jats:p>All cases within the cohort had adequate tissue for assessment. 84% agreement (Cohen\u2019s Kappa\u2009=\u20090.585) was seen between the current team\u2019s consensus and the original reporting pathologist on whether the appearance was supportive of SS. However, only 58% agreement was seen for focus scores (Weighted Kappa\u2009=\u20090.496). The agreement between the serology result and whether the histology was supportive of SS was 79% (Cohen\u2019s Kappa\u2009=\u20090.493).<\/jats:p>\n              <\/jats:sec><jats:sec>\n                <jats:title>Conclusion<\/jats:title>\n                <jats:p>The findings raise the possibility that undue emphasis is placed on the value of a histological SS diagnosis. The current system for assessing and grading these biopsies is ambiguous in nature, with a low threshold considered indicative of SS. Due to the risk of complications associated with a LGB, alternative minimally invasive investigations should always be considered. The histological findings in isolation, particularly when a low focus score is seen, may not be predictive of a diagnosis of SS.<\/jats:p>\n              <\/jats:sec>","DOI":"10.1007\/s12105-024-01662-1","type":"journal-article","created":{"date-parts":[[2024,7,3]],"date-time":"2024-07-03T13:03:09Z","timestamp":1720011789000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["The Value of Labial Gland Biopsies as a Diagnostic Test for Sj\u00f6gren\u2019s Syndrome"],"prefix":"10.1007","volume":"18","author":[{"given":"Mollie","family":"Clark","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hannah","family":"Walsh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"India","family":"Stephens-Laborde","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Syed Ali","family":"Khurram","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,7,3]]},"reference":[{"key":"1662_CR1","doi-asserted-by":"publisher","first-page":"1275","DOI":"10.1001\/ARCHINTE.164.12.1275","volume":"164","author":"SS Kassan","year":"2004","unstructured":"Kassan SS, Moutsopoulos HM (2004) Clinical manifestations and early diagnosis of Sj\u00f6gren Syndrome. Arch Intern Med 164:1275\u20131284. https:\/\/doi.org\/10.1001\/ARCHINTE.164.12.1275","journal-title":"Arch Intern Med"},{"key":"1662_CR2","doi-asserted-by":"publisher","unstructured":"Fragkioudaki S, Mavragani CP, Moutsopoulos HM (2016) Predicting the risk for lymphoma development in Sjogren syndrome an easy tool for clinical use. https:\/\/doi.org\/10.1097\/MD.0000000000003766","DOI":"10.1097\/MD.0000000000003766"},{"key":"1662_CR3","doi-asserted-by":"publisher","first-page":"554","DOI":"10.1136\/ard.61.6.554","volume":"61","author":"C Vitali","year":"2002","unstructured":"Vitali C, Bombardieri S, Jonsson R et al (2002) Classification criteria for Sj\u00f6gren\u2019s syndrome: a revised version of the European criteria proposed by the American-European Consensus Group. Ann Rheum Dis 61:554. https:\/\/doi.org\/10.1136\/ard.61.6.554","journal-title":"Ann Rheum Dis"},{"key":"1662_CR4","doi-asserted-by":"publisher","first-page":"64","DOI":"10.1016\/0030-4220(74)90160-1","volume":"37","author":"TM Tarpley","year":"1974","unstructured":"Tarpley TM, Anderson LG, White CL (1974) Minor salivary gland involvement in Sj\u00f6gren\u2019s syndrome. Oral surgery, oral medicine. Oral Pathol 37:64\u201374. https:\/\/doi.org\/10.1016\/0030-4220(74)90160-1","journal-title":"Oral Pathol"},{"key":"1662_CR5","doi-asserted-by":"publisher","first-page":"1161","DOI":"10.1136\/annrheumdis-2016-210448","volume":"76","author":"BA Fisher","year":"2017","unstructured":"Fisher BA, Jonsson R, Daniels T et al (2017) Standardisation of labial salivary gland histopathology in clinical trials in primary Sj\u00f6gren\u2019s syndrome. Ann Rheum Dis 76:1161. https:\/\/doi.org\/10.1136\/annrheumdis-2016-210448","journal-title":"Ann Rheum Dis"},{"key":"1662_CR6","doi-asserted-by":"publisher","first-page":"208","DOI":"10.1080\/03009742.2021.1999671","volume":"52","author":"P Olsson","year":"2023","unstructured":"Olsson P, Ekblad F, Hassler A et al (2023) Complications after minor salivary gland biopsy: a retrospective study of 630 patients from two Swedish centres. Scand J Rheumatol 52:208\u2013216. https:\/\/doi.org\/10.1080\/03009742.2021.1999671","journal-title":"Scand J Rheumatol"},{"key":"1662_CR7","doi-asserted-by":"publisher","first-page":"2589","DOI":"10.1016\/j.jtauto.2021.100138","volume":"5","author":"S Veenbergen","year":"2022","unstructured":"Veenbergen S, Kozmar A, Van Daele PLA, Schreurs MWJ (2022) NC-ND license autoantibodies in Sj\u00f6gren\u2019s syndrome and its classification criteria. J Transl Autoimmun 5:2589\u20139090. https:\/\/doi.org\/10.1016\/j.jtauto.2021.100138","journal-title":"J Transl Autoimmun"},{"key":"1662_CR8","doi-asserted-by":"publisher","first-page":"S19","DOI":"10.1016\/S0140-6736(13)60459-6","volume":"381","author":"S Ali","year":"2013","unstructured":"Ali S, Brown J, Ngu R et al (2013) Correlation between ultrasound imaging of major salivary glands and histopathological findings of labial gland biopsy samples in Sjogren\u2019s syndrome. Lancet 381:S19. https:\/\/doi.org\/10.1016\/S0140-6736(13)60459-6","journal-title":"Lancet"},{"key":"1662_CR9","doi-asserted-by":"publisher","unstructured":"Li N, Ye Y, Hai W, Xie Y, Jiang L (2023) Alterations in histology of the aging salivary gland and correlation with the glandular inflammatory microenvironment. iScience 26(5). https:\/\/doi.org\/10.1016\/j.isci.2023.106571","DOI":"10.1016\/j.isci.2023.106571"},{"key":"1662_CR10","doi-asserted-by":"publisher","first-page":"159","DOI":"10.1093\/ageing\/13.3.159","volume":"13","author":"S Syrj\u00e4nen","year":"1984","unstructured":"Syrj\u00e4nen S (1984) Age-related changes in structure of labial minor salivary glands. Age Ageing 13:159\u2013165. https:\/\/doi.org\/10.1093\/ageing\/13.3.159","journal-title":"Age Ageing"},{"key":"1662_CR11","doi-asserted-by":"publisher","first-page":"247","DOI":"10.2147\/CLEP.S47399","volume":"6","author":"R Patel","year":"2014","unstructured":"Patel R, Shahane A (2014) The epidemiology of Sj\u00f6gren\u2019s syndrome. Clin Epidemiol 6:247\u2013255. https:\/\/doi.org\/10.2147\/CLEP.S47399","journal-title":"Clin Epidemiol"},{"issue":"3","key":"1662_CR12","doi-asserted-by":"publisher","first-page":"416","DOI":"10.1016\/j.autrev.2012.08.001","volume":"12","author":"D Guellec","year":"2013","unstructured":"Guellec D, Cornec D, Jousse-Joulin S, Marhadour T, Marcorelles P, Pers J-O, Saraux A, Devauchelle-Pensec V (2013) Diagnostic value of labial minor salivary gland biopsy for Sj\u00f6gren\u2019s syndrome: a systematic review. Autoimmun rev 12(3):416\u2013420. https:\/\/doi.org\/10.1016\/j.autrev.2012.08.001","journal-title":"Autoimmun rev"},{"key":"1662_CR13","doi-asserted-by":"publisher","first-page":"714","DOI":"10.1002\/art.23579","volume":"59","author":"R Caporali","year":"2008","unstructured":"Caporali R, Bonacci E, Epis O et al (2008) Safety and usefulness of minor salivary gland biopsy: retrospective analysis of 502 procedures performed at a single center. Arthritis Care Res (Hoboken) 59:714\u2013720. https:\/\/doi.org\/10.1002\/art.23579","journal-title":"Arthritis Care Res (Hoboken)"},{"key":"1662_CR14","doi-asserted-by":"publisher","first-page":"450","DOI":"10.1111\/jop.12387","volume":"45","author":"E Astorri","year":"2016","unstructured":"Astorri E, Sutcliffe N, Richards PS et al (2016) Ultrasound of the salivary glands is a strong predictor of labial gland biopsy histopathology in patients with sicca symptoms. J Oral Pathol Med 45:450\u2013454. https:\/\/doi.org\/10.1111\/jop.12387","journal-title":"J Oral Pathol Med"},{"key":"1662_CR15","doi-asserted-by":"publisher","first-page":"12903","DOI":"10.3390\/ijms222312903","volume":"22","author":"J-Y Jung","year":"2021","unstructured":"Jung J-Y, Kim J-W, Kim H-A, Suh C-H (2021) Salivary biomarkers in patients with Sj\u00f6gren\u2019s Syndrome\u2014A systematic review. Int J Mol Sci 22:12903. https:\/\/doi.org\/10.3390\/ijms222312903","journal-title":"Int J Mol Sci"}],"container-title":["Head and Neck Pathology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12105-024-01662-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s12105-024-01662-1\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12105-024-01662-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,1,8]],"date-time":"2025-01-08T15:23:11Z","timestamp":1736349791000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s12105-024-01662-1"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,7,3]]},"references-count":15,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2024,12]]}},"alternative-id":["1662"],"URL":"https:\/\/doi.org\/10.1007\/s12105-024-01662-1","relation":{},"ISSN":["1936-0568"],"issn-type":[{"value":"1936-0568","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,7,3]]},"assertion":[{"value":"17 April 2024","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"1 June 2024","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"3 July 2024","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors declare no competing interests.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of Interest"}},{"value":"This article does not contain any studies with human participants or animals performed by any of the authors.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical Approval"}},{"value":"For this type of study informed consent is not required.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Informed Consent"}},{"value":"For this type of study consent for publication is not required.","order":5,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for Publication"}}],"article-number":"64"}}