Thymic hyperplasia associated with primary Sjogren’s syndrome cured by thymectomy

Yu, Xin; Cai, Hongfei; Li, Yang; Cui, Youbin

doi:10.21037/jtd.2017.02.24

Cited by 9 publications

(10 citation statements)

References 7 publications

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“…In cases of thymoma-induced autoimmune disease, the mainstay of treatment is surgical resection [2] . Surgical resection of early thymic carcinoma has been reported to offer control of autoimmune phenomena [11] . However, this relapsed with postoperative metastases of the tumour [12] , whereas in metastatic thymic carcinoma, surgery is not an option.…”

Section: Discussionmentioning

confidence: 99%

Two Very Rare Cases of Metastatic Thymic Carcinoma with Sjogren’s Syndrome: A Case Series

et al. 2023

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Thymic tumours are rare thoracic malignancies with thymic carcinoma accounting for approximately 12% of all thymic tumours compared to thymomas which account for approximately 86%. Unlike thymomas, it is very rare for thymic carcinomas to be associated with autoimmune disorders or paraneoplastic syndromes. When these phenomena do occur, the vast majority are myasthenia gravis, pure red cell aplasia, or systemic lupus erythematous. Paraneoplastic Sjogren’s syndrome is a rare complication of thymic carcinoma, with only two cases previously reported. Here we present 2 cases of patients with metastatic thymic carcinoma who developed autoimmune phenomena consistent with Sjogren’s syndrome without classical symptoms prior to treatment. One patient opted for surveillance of their malignancy, while the other underwent chemoimmunotherapy with favourable results. These case reports describe two distinctive clinical presentations of a rare paraneoplastic phenomenon.

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Section: Discussionmentioning

confidence: 99%

Two Very Rare Cases of Metastatic Thymic Carcinoma with Sjogren’s Syndrome: A Case Series

et al. 2023

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“…Previous reports on thymoma complicated with autoimmune disease mostly exist as case reports. Various autoimmune diseases, including aplastic anemia (10), Sjogren's syndrome (11), and total encephalitis (12), have been satisfactorily controlled through the removal of thymoma or thymectomy. This study encompassed a variety of autoimmune diseases, including pure erythrocyte aplastic anemia, GS, anti-AMPA2-R autoimmune meningitis, and nephrotic syndrome.…”

Section: Discussionmentioning

confidence: 99%

Surgical treatment is effective in treating thymoma accompanied with non-myasthenia gravis autoimmune diseases: a single-center clinical experience

et al. 2020

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Background and Aims: Although thymoma is inextricably linked to autoimmune disease, its best treatment method remains unclear. In this study, we sought to evaluate therapeutic effect of surgical resection of thymoma on non-myasthenia gravis autoimmune diseases. Materials and Methods: This was a retrospective study covering 32 patients with thymoma accompanied with non-myasthenia gravis autoimmune disease. The relationships between surgical treatment, thymoma pathological type, and prognosis of autoimmune diseases were analyzed from postoperative follow-up data. Results: In total, 32 patients in this study underwent surgical treatment. The mean age of the patients was 51.7 years. By the last follow-up, 2 patients had died, while the other 30 patients showed no sign of tumor recurrence and metastasis. According to the postoperative follow-up data, 22 patients (68.75%) showed improvement or even complete recovery of autoimmune disease symptoms, 9 patients (28.13%) showed no significant change, and only 1 patient’s (3.12%) postoperative symptom was aggravated. Female patients and patients aged 50 and older were more likely to combine with non-organ-specific autoimmune diseases (p = 0.036, p = 0.017). Conclusion: In conclusion, this study presents that surgical treatment achieves a satisfactory prognosis for thymoma combined with non-myasthenia gravis autoimmune disease.

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“…SS is usually related to thymic lesion. Thymic hyperplasia, multilocular thymic cyst, thymoma associated with SS has been reported in the literature [4][5][6]. However, to our knowledge, the case of thymic NEN with SS has not been reported.…”

Section: Thymic Neuroendocrine Tumor With Sjogren's Syndrome Cured Bymentioning

confidence: 99%

Thymic Neuroendocrine Tumor With Sjogren’s Syndrome Cured by Neoadjuvant Chemotherapy and Thymectomy：A Case Report

Cai¹,

Qin²,

Wang³

et al. 2021

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Background: Thymic neuroendocrine tumors are rare neoplasms usually accompanied with excessive secretion functional hormones of endocrine glands, such as multiple endocrine neoplasia 1(MEN1) and ectopic ACTH syndrome(EAS). As a kind of autoimmune disease, Sjogren’s syndrome(SS) is characterized by destruction of exocrine glands resulting in oral and ocular dryness and is usually related to thymic lesion. However, we present herein a rare case of thymic neuroendocrine tumor with primary Sjogren’s syndrome cured by neoadjuvant chemotherapy and thymectomy.Case presentation: A 70-year-old woman was diagnosed as neuroendocrine carcinoma by puncture with Sjogren’s syndrome(SS). Considering that the boundary between the tumor and the surrounding tissue is not clear，we chose two cycles of neoadjuvant chemotherapy which regimen was etoposide and cisplatin. After treatment, the symptoms of Sjogren’s syndrome were partial relieved and therefore the patient underwent R0 resection. After one year of follow-up, apparent remission of the symptoms was achieved.Conclusions: We believe neoadjuvant chemotherapy combined with radical surgery is an alternative treatment for locoregional resectable thymic neuroendocrine tumors. Because chemotherapy can lead to necrosis, it is a question whether the pathological grade of neuroendocrine tumors will be changed after neoadjuvant chemotherapy. Meanwhile, despite the fact that the association between thymic neuroendocrine tumors and Sjogren’s syndrome remains unclear, the latter may be co-treated by oncologic therapy of the former，thus it also provides a new treatment for Sjogren's syndrome.

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Thymic hyperplasia associated with primary Sjogren’s syndrome cured by thymectomy

Cited by 9 publications

References 7 publications

Two Very Rare Cases of Metastatic Thymic Carcinoma with Sjogren’s Syndrome: A Case Series

Two Very Rare Cases of Metastatic Thymic Carcinoma with Sjogren’s Syndrome: A Case Series

Surgical treatment is effective in treating thymoma accompanied with non-myasthenia gravis autoimmune diseases: a single-center clinical experience

Thymic Neuroendocrine Tumor With Sjogren’s Syndrome Cured by Neoadjuvant Chemotherapy and Thymectomy：A Case Report

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