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Long-term efficacy of sialendoscopy in treating childhood Sjögren’s disease with chronological monitoring by salivary gland ultrasonography: A novel approach

BACKGROUND: Childhood Sjögren’s Disease (cSjD) is an underdiagnosed phenomenon with clinical and pathophysiological nuances in contrast to Sjögren’s Disease (SjD) in the adult population. While adults typically experience sicca symptoms, children with cSjD often present with recurrent parotitis, div...

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Autores principales: Drew, Kristin, Kronlage, René, Cha, Seunghee, Thatayatikom, Akaluck, Schrepfer, Thomas
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10424334/
https://www.ncbi.nlm.nih.gov/pubmed/37574559
http://dx.doi.org/10.1186/s12969-023-00870-3
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author Drew, Kristin
Kronlage, René
Cha, Seunghee
Thatayatikom, Akaluck
Schrepfer, Thomas
author_facet Drew, Kristin
Kronlage, René
Cha, Seunghee
Thatayatikom, Akaluck
Schrepfer, Thomas
author_sort Drew, Kristin
collection PubMed
description BACKGROUND: Childhood Sjögren’s Disease (cSjD) is an underdiagnosed phenomenon with clinical and pathophysiological nuances in contrast to Sjögren’s Disease (SjD) in the adult population. While adults typically experience sicca symptoms, children with cSjD often present with recurrent parotitis, diverse autoantibody profiles, and renal and neurological manifestations. Diagnosis and classification in pediatric rheumatology remain controversial due to the reliance on adult-focused diagnostic criteria and the lack of standardized treatment and understanding of outcomes. The purpose of the paper is to propose a multimodal treatment plan and demonstrate the effectiveness of sialendoscopy in the management of cSjD. CASE PRESENTATION: We present the case of a twelve-year-old female diagnosed with cSjD using the 2016 American College of Rheumatology (ACR) and the European League Against Rheumatism (EULAR) diagnostic criteria for SjD. In addition to medical management, she underwent sialendoscopy with triamcinolone irrigation under sedation and was monitored for progress via salivary gland ultrasonography (SGUS). Over the course of one year, she demonstrated significant improvement in symptoms, with serial SGUS scores gradually decreasing by five points. CONCLUSIONS: This paper proposes a multimodal treatment plan involving sialendoscopy and medical management as a non-invasive and potentially more effective approach for cSjD. Standardized monitoring through SGUS scoring allows objective and quantifiable measurement of treatment progress, enabling better assessment of glandular tissue status. Recurrence is possible, and each cSjD patient may present differently. Nevertheless, our year-long observation of a patient with cSjD demonstrates that sialendoscopy, as seen in adults, can promote remission of recurrent parotitis in children as well.
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spelling pubmed-104243342023-08-15 Long-term efficacy of sialendoscopy in treating childhood Sjögren’s disease with chronological monitoring by salivary gland ultrasonography: A novel approach Drew, Kristin Kronlage, René Cha, Seunghee Thatayatikom, Akaluck Schrepfer, Thomas Pediatr Rheumatol Online J Case Report BACKGROUND: Childhood Sjögren’s Disease (cSjD) is an underdiagnosed phenomenon with clinical and pathophysiological nuances in contrast to Sjögren’s Disease (SjD) in the adult population. While adults typically experience sicca symptoms, children with cSjD often present with recurrent parotitis, diverse autoantibody profiles, and renal and neurological manifestations. Diagnosis and classification in pediatric rheumatology remain controversial due to the reliance on adult-focused diagnostic criteria and the lack of standardized treatment and understanding of outcomes. The purpose of the paper is to propose a multimodal treatment plan and demonstrate the effectiveness of sialendoscopy in the management of cSjD. CASE PRESENTATION: We present the case of a twelve-year-old female diagnosed with cSjD using the 2016 American College of Rheumatology (ACR) and the European League Against Rheumatism (EULAR) diagnostic criteria for SjD. In addition to medical management, she underwent sialendoscopy with triamcinolone irrigation under sedation and was monitored for progress via salivary gland ultrasonography (SGUS). Over the course of one year, she demonstrated significant improvement in symptoms, with serial SGUS scores gradually decreasing by five points. CONCLUSIONS: This paper proposes a multimodal treatment plan involving sialendoscopy and medical management as a non-invasive and potentially more effective approach for cSjD. Standardized monitoring through SGUS scoring allows objective and quantifiable measurement of treatment progress, enabling better assessment of glandular tissue status. Recurrence is possible, and each cSjD patient may present differently. Nevertheless, our year-long observation of a patient with cSjD demonstrates that sialendoscopy, as seen in adults, can promote remission of recurrent parotitis in children as well. BioMed Central 2023-08-13 /pmc/articles/PMC10424334/ /pubmed/37574559 http://dx.doi.org/10.1186/s12969-023-00870-3 Text en © The Author(s) 2023 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Drew, Kristin
Kronlage, René
Cha, Seunghee
Thatayatikom, Akaluck
Schrepfer, Thomas
Long-term efficacy of sialendoscopy in treating childhood Sjögren’s disease with chronological monitoring by salivary gland ultrasonography: A novel approach
title Long-term efficacy of sialendoscopy in treating childhood Sjögren’s disease with chronological monitoring by salivary gland ultrasonography: A novel approach
title_full Long-term efficacy of sialendoscopy in treating childhood Sjögren’s disease with chronological monitoring by salivary gland ultrasonography: A novel approach
title_fullStr Long-term efficacy of sialendoscopy in treating childhood Sjögren’s disease with chronological monitoring by salivary gland ultrasonography: A novel approach
title_full_unstemmed Long-term efficacy of sialendoscopy in treating childhood Sjögren’s disease with chronological monitoring by salivary gland ultrasonography: A novel approach
title_short Long-term efficacy of sialendoscopy in treating childhood Sjögren’s disease with chronological monitoring by salivary gland ultrasonography: A novel approach
title_sort long-term efficacy of sialendoscopy in treating childhood sjögren’s disease with chronological monitoring by salivary gland ultrasonography: a novel approach
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10424334/
https://www.ncbi.nlm.nih.gov/pubmed/37574559
http://dx.doi.org/10.1186/s12969-023-00870-3
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