Labial salivary gland biopsy: a crucial method for confirming seronegative Sjogren’s syndrome—a case report

Abstract Background Sjogren’s syndrome is an autoimmune disease that affects exocrine glands like sweat glands, salivary glands, and lacrimal glands. Dentists frequently encounter these patients and have an important role in establishing the diagnosis. It is mainly diagnosed by clinical features lik...

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Main Authors: Satyasai S. G. Romala, Kishore Moturi, Ravikanth Manyam, Meghana Vadupu, P. Divya Naga Lakshmi, Anil Budumuru, U. Shivajiraju, J. S. D. N. Sai Hyma, R. V. S. N. S. Ramachandrudu
Format: Article
Language:English
Published: SpringerOpen 2024-12-01
Series:Bulletin of the National Research Centre
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Online Access:https://doi.org/10.1186/s42269-024-01279-y
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author Satyasai S. G. Romala
Kishore Moturi
Ravikanth Manyam
Meghana Vadupu
P. Divya Naga Lakshmi
Anil Budumuru
U. Shivajiraju
J. S. D. N. Sai Hyma
R. V. S. N. S. Ramachandrudu
author_facet Satyasai S. G. Romala
Kishore Moturi
Ravikanth Manyam
Meghana Vadupu
P. Divya Naga Lakshmi
Anil Budumuru
U. Shivajiraju
J. S. D. N. Sai Hyma
R. V. S. N. S. Ramachandrudu
author_sort Satyasai S. G. Romala
collection DOAJ
description Abstract Background Sjogren’s syndrome is an autoimmune disease that affects exocrine glands like sweat glands, salivary glands, and lacrimal glands. Dentists frequently encounter these patients and have an important role in establishing the diagnosis. It is mainly diagnosed by clinical features like xerostomia and xerophthalmia, along with anti-Sjögren’s syndrome-related antigen A (anti-SSA/also called Ro antigen) and anti-Sjögren’s syndrome-related antigen B (anti-SSB/ also called La antigen) antibodies in the patients. However, there is a subset of the population where autonuclear antibodies cannot be detected but are still present with all clinical features of Sjogren’s syndrome. Such patients usually remain undiagnosed, and only symptomatic treatment is given in spite of repeated consultation with various medical specialists. Only after establishing a proper diagnosis can we initiate definitive treatment. Case presentation Here, we report a case of seronegative Sjogren’s syndrome in a patient with severe xerostomia without anti-SSA and anti-SSB antibodies. The focus score from the labial biopsy, co-relating with clinical presentation, helped us in establishing the diagnosis. After a definitive diagnosis, our patient is put on cholinergic therapy, which greatly reduced the symptoms of xerostomia. Conclusions Physicians and dentists should refer such patients to an oral and maxillofacial surgeon for a labial biopsy to consider “seronegative Sjogren’s syndrome." The “focus score” evaluation by the pathologist helps in establishing the diagnosis of “seronegative Sjogren’s syndrome”.
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spelling doaj-art-b8d3c7121f9a4344ab7f0a6fd41f04aa2025-08-20T02:35:45ZengSpringerOpenBulletin of the National Research Centre2522-83072024-12-014811510.1186/s42269-024-01279-yLabial salivary gland biopsy: a crucial method for confirming seronegative Sjogren’s syndrome—a case reportSatyasai S. G. Romala0Kishore Moturi1Ravikanth Manyam2Meghana Vadupu3P. Divya Naga Lakshmi4Anil Budumuru5U. Shivajiraju6J. S. D. N. Sai Hyma7R. V. S. N. S. Ramachandrudu8Department of Oral and Maxillofacial Surgery, Vishnu Dental CollegeDepartment of Oral and Maxillofacial Surgery, Vishnu Dental College Department of Oral and Maxillofacial Pathology, Vishnu Dental CollegeDepartment of Oral and Maxillofacial Surgery, Vishnu Dental CollegeDepartment of Oral and Maxillofacial Surgery, Vishnu Dental CollegeDepartment of Oral and Maxillofacial Surgery, Vishnu Dental CollegeDepartment of Oral and Maxillofacial Surgery, Vishnu Dental CollegeDepartment of Anesthesiology, G.S.L. Medical CollegeDepartment of Pharmacology, ASRAM Medical CollegeAbstract Background Sjogren’s syndrome is an autoimmune disease that affects exocrine glands like sweat glands, salivary glands, and lacrimal glands. Dentists frequently encounter these patients and have an important role in establishing the diagnosis. It is mainly diagnosed by clinical features like xerostomia and xerophthalmia, along with anti-Sjögren’s syndrome-related antigen A (anti-SSA/also called Ro antigen) and anti-Sjögren’s syndrome-related antigen B (anti-SSB/ also called La antigen) antibodies in the patients. However, there is a subset of the population where autonuclear antibodies cannot be detected but are still present with all clinical features of Sjogren’s syndrome. Such patients usually remain undiagnosed, and only symptomatic treatment is given in spite of repeated consultation with various medical specialists. Only after establishing a proper diagnosis can we initiate definitive treatment. Case presentation Here, we report a case of seronegative Sjogren’s syndrome in a patient with severe xerostomia without anti-SSA and anti-SSB antibodies. The focus score from the labial biopsy, co-relating with clinical presentation, helped us in establishing the diagnosis. After a definitive diagnosis, our patient is put on cholinergic therapy, which greatly reduced the symptoms of xerostomia. Conclusions Physicians and dentists should refer such patients to an oral and maxillofacial surgeon for a labial biopsy to consider “seronegative Sjogren’s syndrome." The “focus score” evaluation by the pathologist helps in establishing the diagnosis of “seronegative Sjogren’s syndrome”.https://doi.org/10.1186/s42269-024-01279-yXerostomiaSjogren syndromeCholinomimeticsPilocarpine
spellingShingle Satyasai S. G. Romala
Kishore Moturi
Ravikanth Manyam
Meghana Vadupu
P. Divya Naga Lakshmi
Anil Budumuru
U. Shivajiraju
J. S. D. N. Sai Hyma
R. V. S. N. S. Ramachandrudu
Labial salivary gland biopsy: a crucial method for confirming seronegative Sjogren’s syndrome—a case report
Bulletin of the National Research Centre
Xerostomia
Sjogren syndrome
Cholinomimetics
Pilocarpine
title Labial salivary gland biopsy: a crucial method for confirming seronegative Sjogren’s syndrome—a case report
title_full Labial salivary gland biopsy: a crucial method for confirming seronegative Sjogren’s syndrome—a case report
title_fullStr Labial salivary gland biopsy: a crucial method for confirming seronegative Sjogren’s syndrome—a case report
title_full_unstemmed Labial salivary gland biopsy: a crucial method for confirming seronegative Sjogren’s syndrome—a case report
title_short Labial salivary gland biopsy: a crucial method for confirming seronegative Sjogren’s syndrome—a case report
title_sort labial salivary gland biopsy a crucial method for confirming seronegative sjogren s syndrome a case report
topic Xerostomia
Sjogren syndrome
Cholinomimetics
Pilocarpine
url https://doi.org/10.1186/s42269-024-01279-y
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