Seronegative Sicca Syndrome: Diagnostic Considerations and Management Strategies

Seronegative sicca syndrome encompasses patients who present with xerostomia and/or keratoconjunctivitis sicca but lack anti-SSA/SSB antibodies and do not fulfill current classification criteria for primary Sjögren’s syndrome (pSS). Despite symptom overlap with pSS, these individuals remain diagnost...

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Main Authors: Yordanka M. Basheva-Kraeva, Krasimir I. Kraev, Petar A. Uchikov, Maria I. Kraeva, Bozhidar K. Hristov, Nina St. Stoyanova, Vesela T. Mitkova-Hristova, Borislav Ivanov, Stanislav S. Karamitev, Nina Koleva, Aleksandar Marinkov, Veselin A. Vassilev
Format: Article
Language:English
Published: MDPI AG 2025-06-01
Series:Life
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Online Access:https://www.mdpi.com/2075-1729/15/6/966
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author Yordanka M. Basheva-Kraeva
Krasimir I. Kraev
Petar A. Uchikov
Maria I. Kraeva
Bozhidar K. Hristov
Nina St. Stoyanova
Vesela T. Mitkova-Hristova
Borislav Ivanov
Stanislav S. Karamitev
Nina Koleva
Aleksandar Marinkov
Veselin A. Vassilev
author_facet Yordanka M. Basheva-Kraeva
Krasimir I. Kraev
Petar A. Uchikov
Maria I. Kraeva
Bozhidar K. Hristov
Nina St. Stoyanova
Vesela T. Mitkova-Hristova
Borislav Ivanov
Stanislav S. Karamitev
Nina Koleva
Aleksandar Marinkov
Veselin A. Vassilev
author_sort Yordanka M. Basheva-Kraeva
collection DOAJ
description Seronegative sicca syndrome encompasses patients who present with xerostomia and/or keratoconjunctivitis sicca but lack anti-SSA/SSB antibodies and do not fulfill current classification criteria for primary Sjögren’s syndrome (pSS). Despite symptom overlap with pSS, these individuals remain diagnostically and therapeutically unclassified. This review studies the clinical, immunological, and pathological spectrum of seronegative sicca, highlighting its heterogeneity and the limitations of antibody-centric diagnostic frameworks. Histopathologic findings in some seronegative patients—including focal lymphocytic sialadenitis—mirror those seen in pSS, suggesting underlying immune-mediated glandular damage. In others, nonspecific or normal biopsy findings suggest non-immune mechanisms. New evidence of immune activity, such as elevated cytokines (BAFF, IFN-α), and novel autoantibodies (SP-1, CA-VI), further supports the concept of subclinical autoimmunity in a subset of these patients. Clinically, they often face significant burden, including dryness, fatigue, and pain, yet remain excluded from most research cohorts, therapeutic trials, and clinical guidelines. Their management is often individualized, relying on symptomatic therapies rather than immunomodulatory agents. The lack of validated diagnostic criteria and prognostic markers compounds the uncertainty surrounding disease evolution, as some patients may later seroconvert or develop systemic features. To address these gaps, a paradigm shift is needed—one that embraces the spectrum of sicca syndromes, incorporates advanced immunophenotyping, and allows inclusion of seronegative patients in research and care algorithms.
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spelling doaj-art-c27e05e4c8ac43ffb2ba9cb7ed7a425b2025-08-20T03:16:33ZengMDPI AGLife2075-17292025-06-0115696610.3390/life15060966Seronegative Sicca Syndrome: Diagnostic Considerations and Management StrategiesYordanka M. Basheva-Kraeva0Krasimir I. Kraev1Petar A. Uchikov2Maria I. Kraeva3Bozhidar K. Hristov4Nina St. Stoyanova5Vesela T. Mitkova-Hristova6Borislav Ivanov7Stanislav S. Karamitev8Nina Koleva9Aleksandar Marinkov10Veselin A. Vassilev11Department of Ophthalmology, Faculty of Medicine, Medical University of Plovdiv, 4002 Plovdiv, BulgariaDepartment of Propedeutics of Internal Diseases, Medical Faculty, Medical University of Plovdiv, 4002 Plovdiv, BulgariaDepartment of Special Surgery, Medical Faculty, Medical University of Plovdiv, 4002 Plovdiv, BulgariaDepartment of Otorhinolaryngology, Medical Faculty, Medical University of Plovdiv, 4002 Plovdiv, BulgariaSecond Department of Internal Diseases, Section “Gastroenterology”, Medical Faculty, Medical University of Plovdiv, 4002 Plovdiv, BulgariaDepartment of Ophthalmology, Faculty of Medicine, Medical University of Plovdiv, 4002 Plovdiv, BulgariaDepartment of Ophthalmology, Faculty of Medicine, Medical University of Plovdiv, 4002 Plovdiv, BulgariaFaculty of Medicine, Medical University of Plovdiv, 4002 Plovdiv, BulgariaDepartment of Orthopedics and Traumatology, Faculty of Medicine, Medical University of Plovdiv, 4002 Plovdiv, BulgariaSpeciality “Assistant Pharmacist”, Medical College, Medical University of Plovdiv, 4002 Plovdiv, BulgariaDepartment of Propedeutics of Internal Diseases, Medical Faculty, Medical University of Plovdiv, 4002 Plovdiv, BulgariaDepartment of Physiology, Medical Faculty, Medical University of Plovdiv, 4002 Plovdiv, BulgariaSeronegative sicca syndrome encompasses patients who present with xerostomia and/or keratoconjunctivitis sicca but lack anti-SSA/SSB antibodies and do not fulfill current classification criteria for primary Sjögren’s syndrome (pSS). Despite symptom overlap with pSS, these individuals remain diagnostically and therapeutically unclassified. This review studies the clinical, immunological, and pathological spectrum of seronegative sicca, highlighting its heterogeneity and the limitations of antibody-centric diagnostic frameworks. Histopathologic findings in some seronegative patients—including focal lymphocytic sialadenitis—mirror those seen in pSS, suggesting underlying immune-mediated glandular damage. In others, nonspecific or normal biopsy findings suggest non-immune mechanisms. New evidence of immune activity, such as elevated cytokines (BAFF, IFN-α), and novel autoantibodies (SP-1, CA-VI), further supports the concept of subclinical autoimmunity in a subset of these patients. Clinically, they often face significant burden, including dryness, fatigue, and pain, yet remain excluded from most research cohorts, therapeutic trials, and clinical guidelines. Their management is often individualized, relying on symptomatic therapies rather than immunomodulatory agents. The lack of validated diagnostic criteria and prognostic markers compounds the uncertainty surrounding disease evolution, as some patients may later seroconvert or develop systemic features. To address these gaps, a paradigm shift is needed—one that embraces the spectrum of sicca syndromes, incorporates advanced immunophenotyping, and allows inclusion of seronegative patients in research and care algorithms.https://www.mdpi.com/2075-1729/15/6/966seronegative sicca syndromeprimary Sjögren’s syndromeautoimmune exocrinopathy
spellingShingle Yordanka M. Basheva-Kraeva
Krasimir I. Kraev
Petar A. Uchikov
Maria I. Kraeva
Bozhidar K. Hristov
Nina St. Stoyanova
Vesela T. Mitkova-Hristova
Borislav Ivanov
Stanislav S. Karamitev
Nina Koleva
Aleksandar Marinkov
Veselin A. Vassilev
Seronegative Sicca Syndrome: Diagnostic Considerations and Management Strategies
Life
seronegative sicca syndrome
primary Sjögren’s syndrome
autoimmune exocrinopathy
title Seronegative Sicca Syndrome: Diagnostic Considerations and Management Strategies
title_full Seronegative Sicca Syndrome: Diagnostic Considerations and Management Strategies
title_fullStr Seronegative Sicca Syndrome: Diagnostic Considerations and Management Strategies
title_full_unstemmed Seronegative Sicca Syndrome: Diagnostic Considerations and Management Strategies
title_short Seronegative Sicca Syndrome: Diagnostic Considerations and Management Strategies
title_sort seronegative sicca syndrome diagnostic considerations and management strategies
topic seronegative sicca syndrome
primary Sjögren’s syndrome
autoimmune exocrinopathy
url https://www.mdpi.com/2075-1729/15/6/966
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