The coexistence of autoimmune rheumatic diseases and thymomas

Jiang, Ying; Dai, Xiaodan; Duan, Liping; Zhou, Ye

doi:10.17219/acem/61004

Cited by 5 publications

(4 citation statements)

References 23 publications

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“…Thymomas are commonly associated with parathymic syndromes,[ 7 8 9 ] one of the most common is myasthenia gravis (MG). In general, thymic tumors occur in approximately 20–30% of patients with MG and, in turn, MG occurs in 15–60% of patients with thymoma according to different reports.…”

Section: Introductionmentioning

confidence: 99%

Value of Adjuvant Radiotherapy for Thymoma with Myasthenia Gravis after Extended Thymectomy

Yun

et al. 2018

Chinese Medical Journal

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Background:The co-existence of myasthenia gravis (MG) and thymoma makes the surgical treatment more complicated and adjuvant radiation more controversial. The aim of this study was to investigate adjuvant radiotherapy for thymoma with MG after extended thymectomy.Methods:A total of 181 patients with both MG and thymoma were recruited between 2003 and 2014 at Tongren Hospital, China. Among all the patients, 157 patients received radiation therapy after surgery (Group A); whereas the other 24 patients did not receive radiation therapy (Group B). According to the time that patients started mediastinal radiation therapy, we subdivided the 157 patients in Group A into subgroups (1-month subgroup, n = 98; 2-month subgroup, n = 7; and 3-month subgroup, n = 52). We then compared the effect of the mediastinal radiation therapy across these different groups using the survival rate, the rate of postoperative myasthenic crisis, and the complete stable remission (CSR) rate as the primary endpoints.Results:There was a significant difference in the occurrence of postoperative myasthenic crisis between 1-month subgroup and Group B (χ2 = 4.631, P = 0.031). The rates of reaching CSR were 32.6% in 1-month subgroup, 25% in 3-month subgroup, and 22.7% in Group B, respectively. The overall survival rates of 1-month subgroup, 3-month subgroup, and Group B were 88.8%, 83.3%, and 77.3%, respectively. Analysis on the Kaplan-Meier survival curves demonstrated that within 8 years after surgery, there was no significant difference in aspects of overall survival and disease-free survival between 1-month subgroup and Group B, and between 3-month subgroup and Group B; over 8 years after surgery, the disease-free survival rates in 1-month subgroup, 3-month subgroup and Group B were 79.4%, 70.6%, and 55.3%, respectively.Conclusions:Adjuvant radiation within 1 month after extended thymectomy may be helpful in controlling postoperative MG, such as decreasing the possibility of postoperative myasthenic crisis, and raising cumulative probabilities of reaching CSR.

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Section: Introductionmentioning

confidence: 99%

Value of Adjuvant Radiotherapy for Thymoma with Myasthenia Gravis after Extended Thymectomy

Yun

et al. 2018

Chinese Medical Journal

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“…Thymoma is associated with other autoimmune disorders, such as systemic lupus erythematosus and Sjogren's syndrome. [6]…”

Section: Discussionmentioning

confidence: 99%

Diffuse idiopathic pulmonary neuroendocrine cell hyperplasia: Unusual presentation

et al. 2019

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Diffuse idiopathic pulmonary neuroendocrine cell hyperplasia (DIPNEC) is an abnormal proliferation of pulmonary neuroendocrine cells that occur without underlying etiology. Here, we report a unique case of 55-year-old female with unusual presentation of DIPNECH and thymoma and on the background history of Crohn's disease that might point toward an autoimmune phenomenon. To the best of our knowledge, there were no previous reports of DIPNECH with either thymoma or Crohn's disease. DIPNECH has premalignant potential and reported in association with carcinoid and non-small cell lung cancer; however, its autoimmune association never reported. The presence of multiple lung nodules along with evidence of small airway disease should alert the physician to include DIPNECH as part of the differential diagnosis given its malignant potential. The prognosis is variable and depends on the presence or absence of underlying malignancy as well as the severity of airflow obstruction.

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“…1 The few cases reported in the literature that describe the coexistence of autoimmune disease and thymic masses mainly concern systemic lupus erythematosus or myasthenia gravis. [2][3][4] The role of thymectomy remains unknown, and the available data on clinical outcomes of thymectomy are contradictory. 1,3,5 Elucidating the physiological connections could guide therapeutic options and improve our understanding of autoimmunity.…”

mentioning

confidence: 99%

“…Thymic hyperplasia's association with Sjögren syndrome is rare, and the mechanism behind this association remains unknown 1 . The few cases reported in the literature that describe the coexistence of autoimmune disease and thymic masses mainly concern systemic lupus erythematosus or myasthenia gravis 2–4 . The role of thymectomy remains unknown, and the available data on clinical outcomes of thymectomy are contradictory 1,3,5 .…”

mentioning

confidence: 99%

Sjögren Syndrome and Thymic Lymphoid Hyperplasia

et al. 2020

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A 51-year-old man was admitted to the emergency department with complaints of weight loss, anorexia, lack of strength in the shoulder and hip joints, vespertine fever, and sweats. On physical examination, he presented erythematous, descamative, and pruriginous cutaneous lesions on the chest and fingers (Figs. 1, 2). A cutaneous biopsy was performed, indicating a pattern of dermatomyositis. Electromyography showed no evidence of myopathic/ neuropathic involvement. Immunological investigation detected antinuclear (1/160), anti-Ro (3+), and anti-La (2+) autoantibodies in the serum. Antiacetylcholine receptor antibodies were not detected. Steroid therapy was started with clinical improvement.Thoracoabdominopelvic computed tomography scan revealed a slightly lobulated structure, possibly representing a rebound thymic enlargement. The patient underwent total body positron emission tomography scan, which showed tissue densification suggesting hypermetabolic pathology of the thymus and suspicion of malignancy.Thymectomy was performed. On histologic examination, the lesion had solid and cystic areas, composed of lymphoid tissue, with numerous lymphoid follicles with germinal centers, and Hassall corpuscles, some of which showed cystic dilation. The cystic component had a squamous epithelium lining and was filled with amorphous eosinophilic material and abundant cholesterol clefts (Fig. 3). Lymphoepithelial lesions were a striking feature, highlighted by the immunohistochemical staining with keratins (Fig. 4); these results are consistent with thymic hyperplasia with lymphoepithelial sialadenitis-like features.After a 2-year follow-up, patient developed xerostomia and xerophthalmia, and in association with the previous, a Sjögren syndrome diagnosis was considered. Corticotherapy was progressively replaced by azathioprine with an excellent outcome.Thymic hyperplasia's association with Sjögren syndrome is rare, and the mechanism behind this association remains unknown. 1 The few cases reported in the literature that describe the coexistence of autoimmune disease and thymic masses mainly concern systemic lupus erythematosus or myasthenia gravis. [2][3][4] The role of thymectomy remains unknown, and the available data on clinical outcomes of thymectomy are contradictory. 1,3,5 Elucidating the physiological connections could guide therapeutic options and improve our understanding of autoimmunity.

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The coexistence of autoimmune rheumatic diseases and thymomas

Cited by 5 publications

References 23 publications

Value of Adjuvant Radiotherapy for Thymoma with Myasthenia Gravis after Extended Thymectomy

Value of Adjuvant Radiotherapy for Thymoma with Myasthenia Gravis after Extended Thymectomy

Diffuse idiopathic pulmonary neuroendocrine cell hyperplasia: Unusual presentation

Sjögren Syndrome and Thymic Lymphoid Hyperplasia

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