Thymic epithelial tumor complicated by immunological abnormalities: results from a single-center retrospective study in China
Identifieur interne : 000322 ( Main/Curation ); précédent : 000321; suivant : 000323Thymic epithelial tumor complicated by immunological abnormalities: results from a single-center retrospective study in China
Auteurs : Tian-Yan Shi ; Xiao-Hong Wen ; Xu-Hua Shi ; Yue-Wu LuSource :
- Journal of Thoracic Disease [ 2072-1439 ] ; 2019.
Abstract
To describe the clinical manifestations, immunological features, treatments, and outcomes of patients with thymic epithelial tumor (TET) complicated by immunological abnormalities, and to improve knowledge on immunological abnormalities in this rare disease.
Patients with pathologically confirmed TET at Beijing Chaoyang Hospital between January 2013 and May 2018 were included in this study, and clinical data were analyzed retrospectively. Immunological abnormalities were classified into two groups as follows: Good syndrome (GS) and autoimmune disease (AD).
Fifty-nine TET patients were enrolled; twenty-two patients (37.3%) had immune dysfunction. There were no gender, age, or histological type differences between groups with or without immunological abnormalities. Six patients had GS, of whom four patients were diagnosed after thymectomy. Recurrent respiratory infections, particularly opportunistic infections, were the most common manifestation. Three GS patients developed a second cancer (50%; P=0.011). Anti-infective therapy and immunoglobulin supplements effectively treated GS. Seventeen patients developed ADs, including myasthenia gravis (MG) (n=13), Hashimoto’s thyroiditis (n=4), Sjogren’s syndrome (n=1), rheumatoid arthritis (n=1), pemphigus (n=1), and Evans syndrome (n=1). One patient developed both MG and GS and 4 patients presented with two ADs. Three AD cases occurred after thymectomy. Pemphigus and 80% (8/10) of MG cases were resolved following thymectomy.
There is a strong association between immunological abnormalities and TET, which may present at any time point during the disease, even after thymectomy. In addition to infection, GS patients are more likely to develop a second cancer. Thymectomy may produce favorable outcomes for MG in this study, while surgery does not improve immunodeficiency in GS patients.
Url:
DOI: 10.21037/jtd.2019.02.90
PubMed: 31179102
PubMed Central: 6531723
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PMC:6531723Le document en format XML
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<author><name sortKey="Shi, Tian Yan" sort="Shi, Tian Yan" uniqKey="Shi T" first="Tian-Yan" last="Shi">Tian-Yan Shi</name>
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<author><name sortKey="Wen, Xiao Hong" sort="Wen, Xiao Hong" uniqKey="Wen X" first="Xiao-Hong" last="Wen">Xiao-Hong Wen</name>
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<author><name sortKey="Shi, Xu Hua" sort="Shi, Xu Hua" uniqKey="Shi X" first="Xu-Hua" last="Shi">Xu-Hua Shi</name>
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<author><name sortKey="Lu, Yue Wu" sort="Lu, Yue Wu" uniqKey="Lu Y" first="Yue-Wu" last="Lu">Yue-Wu Lu</name>
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<author><name sortKey="Wen, Xiao Hong" sort="Wen, Xiao Hong" uniqKey="Wen X" first="Xiao-Hong" last="Wen">Xiao-Hong Wen</name>
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<author><name sortKey="Shi, Xu Hua" sort="Shi, Xu Hua" uniqKey="Shi X" first="Xu-Hua" last="Shi">Xu-Hua Shi</name>
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<author><name sortKey="Lu, Yue Wu" sort="Lu, Yue Wu" uniqKey="Lu Y" first="Yue-Wu" last="Lu">Yue-Wu Lu</name>
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<series><title level="j">Journal of Thoracic Disease</title>
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<front><div type="abstract" xml:lang="en"><sec><title>Background</title>
<p>To describe the clinical manifestations, immunological features, treatments, and outcomes of patients with thymic epithelial tumor (TET) complicated by immunological abnormalities, and to improve knowledge on immunological abnormalities in this rare disease.</p>
</sec>
<sec><title>Methods</title>
<p>Patients with pathologically confirmed TET at Beijing Chaoyang Hospital between January 2013 and May 2018 were included in this study, and clinical data were analyzed retrospectively. Immunological abnormalities were classified into two groups as follows: Good syndrome (GS) and autoimmune disease (AD).</p>
</sec>
<sec><title>Results</title>
<p>Fifty-nine TET patients were enrolled; twenty-two patients (37.3%) had immune dysfunction. There were no gender, age, or histological type differences between groups with or without immunological abnormalities. Six patients had GS, of whom four patients were diagnosed after thymectomy. Recurrent respiratory infections, particularly opportunistic infections, were the most common manifestation. Three GS patients developed a second cancer (50%; P=0.011). Anti-infective therapy and immunoglobulin supplements effectively treated GS. Seventeen patients developed ADs, including myasthenia gravis (MG) (n=13), Hashimoto’s thyroiditis (n=4), Sjogren’s syndrome (n=1), rheumatoid arthritis (n=1), pemphigus (n=1), and Evans syndrome (n=1). One patient developed both MG and GS and 4 patients presented with two ADs. Three AD cases occurred after thymectomy. Pemphigus and 80% (8/10) of MG cases were resolved following thymectomy.</p>
</sec>
<sec><title>Conclusions</title>
<p>There is a strong association between immunological abnormalities and TET, which may present at any time point during the disease, even after thymectomy. In addition to infection, GS patients are more likely to develop a second cancer. Thymectomy may produce favorable outcomes for MG in this study, while surgery does not improve immunodeficiency in GS patients.</p>
</sec>
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</front>
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