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SS-A52 antigen expression in thymic carcinoma accompanied with Sjögren syndrome: A case report

RATIONALE: The relationship between thymic tumors and Sjögren syndrome (SjS) is unknown, and surgical resection has not been optimized. Especially, thymic carcinoma with autoimmune disease is rare. Analysis of SS-A52, germinal centers, plasma cells, and Foxp3+ Treg in thymic carcinoma has never been...

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Autores principales: Isono, Tomomi, Wakasa, Tomoko, Kusumoto, Hidenori, Shimada, Keiji, Ogawa, Takafumi, Shiono, Hiroyuki
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7870235/
https://www.ncbi.nlm.nih.gov/pubmed/33592902
http://dx.doi.org/10.1097/MD.0000000000024491
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author Isono, Tomomi
Wakasa, Tomoko
Kusumoto, Hidenori
Shimada, Keiji
Ogawa, Takafumi
Shiono, Hiroyuki
author_facet Isono, Tomomi
Wakasa, Tomoko
Kusumoto, Hidenori
Shimada, Keiji
Ogawa, Takafumi
Shiono, Hiroyuki
author_sort Isono, Tomomi
collection PubMed
description RATIONALE: The relationship between thymic tumors and Sjögren syndrome (SjS) is unknown, and surgical resection has not been optimized. Especially, thymic carcinoma with autoimmune disease is rare. Analysis of SS-A52, germinal centers, plasma cells, and Foxp3+ Treg in thymic carcinoma has never been reported, and their pathological roles in causing SjS have not been studied. PATIENT CONCERNS: A 78-year-old man presented with sputum production and xerostomia while asleep. Chest computed tomography showed a homogeneous and hypodense mass in the anterosuperior mediastinum. Serum levels of the antinuclear antibody, antibody to SS-A, and antibody to SS-B were positive. DIAGNOSES: Thymic carcinoma (squamous cell carcinoma) and SjS. INTERVENTIONS: Video-assisted thoracoscopic resection of the mediastinal tumor and postoperative radiation therapy was performed. OUTCOMES: The histological diagnosis was thymic squamous cell carcinoma. Histologically, the squamous carcinomatous cells were arranged in nests and cords in the fibrohyaline stroma with capsular invasion. In the stroma, dense lymphoid tissues containing large reactive germinal centers and many plasma cells were also noted. In the involuted thymus, CD20-positive mature lymphocytes infiltrated, and germinal centers were noted. Double immunohistochemical staining revealed that SS-A52 antigen was positive in both the carcinoma component and CD20-positive mature B cells. Postoperatively, the xerostomia persisted, and serum SS-A and SS-B remained positive. No evidence of carcinoma recurrence with chest computed tomography scan was observed at 18-months follow-up. LESSONS: In the surgical treatment of thymic tumors with SjS, extended thymectomy might be worth considering to stop the progressive destruction of the targets of SjS-specific autoantibodies. However, the postoperative symptoms may not dramatically improve because the target organs might have changed irreversibly, and memory B cells might persist. This is the first report that demonstrated the SS-A52 antigen presentation in a thymic tumor to the best of our knowledge.
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spelling pubmed-78702352021-02-10 SS-A52 antigen expression in thymic carcinoma accompanied with Sjögren syndrome: A case report Isono, Tomomi Wakasa, Tomoko Kusumoto, Hidenori Shimada, Keiji Ogawa, Takafumi Shiono, Hiroyuki Medicine (Baltimore) 7100 RATIONALE: The relationship between thymic tumors and Sjögren syndrome (SjS) is unknown, and surgical resection has not been optimized. Especially, thymic carcinoma with autoimmune disease is rare. Analysis of SS-A52, germinal centers, plasma cells, and Foxp3+ Treg in thymic carcinoma has never been reported, and their pathological roles in causing SjS have not been studied. PATIENT CONCERNS: A 78-year-old man presented with sputum production and xerostomia while asleep. Chest computed tomography showed a homogeneous and hypodense mass in the anterosuperior mediastinum. Serum levels of the antinuclear antibody, antibody to SS-A, and antibody to SS-B were positive. DIAGNOSES: Thymic carcinoma (squamous cell carcinoma) and SjS. INTERVENTIONS: Video-assisted thoracoscopic resection of the mediastinal tumor and postoperative radiation therapy was performed. OUTCOMES: The histological diagnosis was thymic squamous cell carcinoma. Histologically, the squamous carcinomatous cells were arranged in nests and cords in the fibrohyaline stroma with capsular invasion. In the stroma, dense lymphoid tissues containing large reactive germinal centers and many plasma cells were also noted. In the involuted thymus, CD20-positive mature lymphocytes infiltrated, and germinal centers were noted. Double immunohistochemical staining revealed that SS-A52 antigen was positive in both the carcinoma component and CD20-positive mature B cells. Postoperatively, the xerostomia persisted, and serum SS-A and SS-B remained positive. No evidence of carcinoma recurrence with chest computed tomography scan was observed at 18-months follow-up. LESSONS: In the surgical treatment of thymic tumors with SjS, extended thymectomy might be worth considering to stop the progressive destruction of the targets of SjS-specific autoantibodies. However, the postoperative symptoms may not dramatically improve because the target organs might have changed irreversibly, and memory B cells might persist. This is the first report that demonstrated the SS-A52 antigen presentation in a thymic tumor to the best of our knowledge. Lippincott Williams & Wilkins 2021-02-05 /pmc/articles/PMC7870235/ /pubmed/33592902 http://dx.doi.org/10.1097/MD.0000000000024491 Text en Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc. http://creativecommons.org/licenses/by/4.0 This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0
spellingShingle 7100
Isono, Tomomi
Wakasa, Tomoko
Kusumoto, Hidenori
Shimada, Keiji
Ogawa, Takafumi
Shiono, Hiroyuki
SS-A52 antigen expression in thymic carcinoma accompanied with Sjögren syndrome: A case report
title SS-A52 antigen expression in thymic carcinoma accompanied with Sjögren syndrome: A case report
title_full SS-A52 antigen expression in thymic carcinoma accompanied with Sjögren syndrome: A case report
title_fullStr SS-A52 antigen expression in thymic carcinoma accompanied with Sjögren syndrome: A case report
title_full_unstemmed SS-A52 antigen expression in thymic carcinoma accompanied with Sjögren syndrome: A case report
title_short SS-A52 antigen expression in thymic carcinoma accompanied with Sjögren syndrome: A case report
title_sort ss-a52 antigen expression in thymic carcinoma accompanied with sjögren syndrome: a case report
topic 7100
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7870235/
https://www.ncbi.nlm.nih.gov/pubmed/33592902
http://dx.doi.org/10.1097/MD.0000000000024491
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