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Thymoma and Thymic Carcinoma

The thymus is a small organ located beneath the breastbone, it is part of the immune system and produces T-lymphocytes - cells which kill viruses and signal B-lymphocytes to make antibodies to fight infection. About 90 percent of tumors that start in the thymus are thymomas. Thyomas begin in the cells which line the thymus (epithelial cells) and look similar to normal thymus cells under the microscope. Thymomas range from slow growing tumors that rarely spread outside the thymus, to more aggressive tumors, which can potentially spread to nearby organs in the chest, such as the lining of the lung (the pleura). Thymic Carcinoma also starts in the cells which line the thymus, but looks different to normal thymus cells under the microscope. These are more likely to spread to other parts of the body. Thymic Carcinoids are a rarer type of tumour which start in hormone-producing cells. Thymic carcinoids are sometimes associated with a rare genetic disorder: multiple endocrine neoplasia type 1 (MEN-1) syndrome. Other rare types include Thymolipoma and primary thymic lymphoma.

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Latest Research Publications

This list of publications is regularly updated (Source: PubMed).

Jordan KS, Mannle SE
An Unusual Case of Chest Pain in an Adolescent Male: Important Cues to Differential Diagnosis.
Adv Emerg Nurs J. 2017 Jan/Mar; 39(1):10-17 [PubMed] Related Publications
Chest pain is a common presenting symptom in the pediatric population, and in contrast to adults, the etiology is rarely cardiac or life-threatening. The majority of chest pain complaints in children and adolescents are benign and can be managed with reassurance and follow-up. The emergency care provider must obtain a comprehensive history and physical examination, as the differential diagnosis of pediatric chest pain is extensive and serious underlying organic pathology may be present. This article describes the case of an adolescent male with an unusual case of chest pain with a serious underlying medical condition. A systematic approach to the clinical decision-making process is discussed to identify those patients who may have a serious underlying organic cause. Information is also included to guide the provider in the identification of red flags for cardiac etiologies of chest pain and indications for a pediatric cardiology consultation.

Bédat B, Scarpa CR, Sadowski SM, et al.
Acute pancreatitis after thoracic duct ligation for iatrogenic chylothorax. A case report.
BMC Surg. 2017; 17(1):9 [PubMed] Free Access to Full Article Related Publications
BACKGROUND: To report the association between thoracic duct ligation and acute pancreatitis. The association between sudden stop of lymphatic flow and pancreatitis has been established in experimental models.
CASE PRESENTATION: A 57-year-old woman operated for thymoma presented a iatrogenic chylothorax. After thoracic duct ligation, she presented an acute pancreatitis which resolved after conservative treatment. The chylothorax disappeared within 4 days of thoracic duct ligation.
CONCLUSIONS: This is the first report of acute pancreatitis following thoracic duct ligation. The pancreas and digestive tract should be assessed in symptomatic patients after thoracic duct ligation.

Yoo SH, Kim HJ, Kim JH, et al.
Nephrotic syndrome associated with metastatic thymoma treated with chemotherapy.
Medicine (Baltimore). 2017; 96(1):e5408 [PubMed] Free Access to Full Article Related Publications
RATIONALE: Nephropathy with concurrent invasive thymoma is a type of paraneoplastic syndrome.
PATIENT CONCERNS AND DIAGNOSES: We report a 32-year-old female with nephrotic syndrome that was first diagnosed along with invasive thymoma and treated by means of cisplatin-based chemotherapy for the thymoma. The patient initially presented with dyspnea and generalized edema. Chest radiography and computed tomography scans revealed right pleural effusion and a mass in the right middle lung field, which were confirmed by a percutaneous lung biopsy as metastatic invasive thymoma. Severe hypoalbuminemia, heavy proteinuria, hyponatremia, and hypercholesterolemia were features of the nephrotic syndrome. A kidney needle biopsy suggested focal segmental glomerulosclerosis.
INTERVENTIONS AND OUTCOMES: All of the symptoms of nephrotic syndrome were resolved simultaneously during the first 2 cycles of chemotherapy. The patient was on regular follow-up with no specific treatment for nephrotic syndrome and underwent successful resection of the left pleura and anterior thymoma. The patient has shown no evidence of recurrence for 2 years.
LESSONS: We conclude that chemotherapy for invasive thymoma is an effective treatment for nephrotic syndrome accompanying the thymoma.

Caronia FP, Fiorelli A, Arrigo E, et al.
Bilateral single-port thoracoscopic extended thymectomy for management of thymoma and myasthenia gravis: case report.
J Cardiothorac Surg. 2016; 11(1):153 [PubMed] Free Access to Full Article Related Publications
BACKGROUND: Video-assisted thoracoscopy is become a widely accepted approach for the resection of anterior mediastinal masses, including thymoma. The current trend is to reduce the number of ports and minimize the length of incisions to further decrease postoperative pain, chest wall paresthesia, and length of hospitalization. Herein, we reported an extended resection of thymoma in a patient with myasthenia gravis through an uniportal bilateral thoracoscopic approach.
CASE PRESENTATION: A 74 years old woman with myasthenia gravis was referred to our attention for management of a 3.5 cm, well capsulate, thymoma. All laboratory and cardio-pulmonary tests were within normal; thus, she was scheduled for thymoma resection through an uniportal bilateral thoracoscopic approach. Under general anaesthesia and selective intubation, the patient was placed in a 60° right lateral decubitus. A 3 cm skin incision was performed in the fourth right intercostal space and, through that a 30° video-camera and working instruments were inserted without rib spreading. After complete dissection of the thymus and mediastinal fat, the contralateral pleura was opened, and, through that the specimen was pushed into the left pleural cavity. Then, the patient was placed in the left lateral decubitus. Similarly to the right side procedure, a 3-cm incision was performed in the fourth left intercostal space to complete thymic dissection and retrieve the specimen. No intraoperative and post-operative complications were found. The patient was discharged four days later. Pathological examination revealed a type A thymoma (Masaoka stage I). No recurrence was found at 18 months of follow-up CONCLUSIONS: Bilateral single-port thoracoscopy is an available procedure for management of thymoma associated with myasthenia gravis. The less post-operative pain, the reduction of hospital stay and the better esthetic results are all potential advantages of this approach over traditional technique. Obviously, our impression should be validated by larger studies in terms of long-term oncological outcomes.

Xin Y, Cai H, Lu T, et al.
miR-20b Inhibits T Cell Proliferation and Activation via NFAT Signaling Pathway in Thymoma-Associated Myasthenia Gravis.
Biomed Res Int. 2016; 2016:9595718 [PubMed] Free Access to Full Article Related Publications
Purpose. We examined the role of miR-20b in development of thymoma-associated myasthenia gravis, especially in T cell proliferation and activation. Materials and Methods. Using qRT-PCR, we assessed expression levels of miR-20b and its target genes in cultured cells and patient samples and examined the proliferation of cultured cells, using MTT cell proliferation assays and flow cytometry based cell cycle analysis. Activation of T cells was determined by both flow cytometry and qRT-PCR of activation-specific marker genes. Results. Expression of miR-20b was downregulated in samples of thymoma tissues and serum from patients with thymoma-associated myasthenia gravis. In addition, T cell proliferation and activation were inhibited by ectopic overexpression of miR-20b, which led to increased T cell proliferation and activation. NFAT5 and CAMTA1 were identified as targets of miR-20b. Expression levels of NFAT5 and CAMTA1 were inhibited by miR-20b expression in cultured cells, and the expression levels of miR-20b and NFAT5/CAMTA1 were inversely correlated in patients with thymoma-associated myasthenia gravis. Conclusion. miR-20b acts as a tumor suppressor in the development of thymoma and thymoma-associated myasthenia gravis. The tumor suppressive function of miR-20b in thymoma could be due to its inhibition of NFAT signaling by repression of NFAT5 and CAMTA1 expression.

Panda PK, Wig N, Kumar S, Arava S
Invasive thymoma presenting as classic superior vena cava syndrome: a case of venous spread metastasis.
BMJ Case Rep. 2016; 2016 [PubMed] Related Publications
The approach to an intrinsic cause of superior vena cava syndrome (SVCS) is usually difficult but rewarding. We report a case of a middle-aged man who presented with progressive oedema of the upper half of the body, dyspnoea, cough and weight loss for a 1-year duration. He was a non-smoker without prior hospitalisation. Chest radiography showed right-sided pleural effusion with an apparent normal superior mediastinum. Contrast-enhanced CT of the chest revealed a right atrial mass extending and completely obliterating to superior vena cava. The differentials were tuberculosis, invasive fungal granuloma, sarcoidosis, primary vasculitis, chronic venous thrombosis, cardiac sarcoma/lymphoma and metastatic thyroid tumour or thymoma. He underwent transvenous (femoral approach) biopsy of the mass and then cardiothoracic surgery after haemodynamic instability. Pathology showed invasive thymoma type B3. This case highlights the approach to an intrinsic cause of SVCS, a complication of the transvenous approach, and importantly a noble finding of venous spread metastasis.

Mendogni P, Rosso L, Tosi D, et al.
Autoimmune hepatitis: an uncommon presentation of thymoma.
Tumori. 2016; 102(Suppl. 2) [PubMed] Related Publications
PURPOSE: In a substantial proportion of patients with thymoma, many different types of paraneoplastic syndromes are observed. The association between thymoma and autoimmune liver diseases, however, has been found in very few cases.
METHODS: We report the case of a 31-year-old man affected by autoimmune hepatitis associated with myasthenia gravis and thymoma, successfully treated with extended thymectomy.
RESULTS: The patient is free from neoplastic and hepatic disease 4 years after surgery. Eighteen months after thymectomy, an exacerbation of hepatitis was successfully treated with steroids.
CONCLUSIONS: To the authors' knowledge, only 7 cases of myasthenia gravis associated with thymoma and autoimmune hepatitis have been reported in the English-language literature. The exact role of thymoma in immune-mediated hepatitis is unclear. It seems likely that thymoma-associated T-cell abnormalities, due to the presence of thymoma, may have a role in the development of this rare clinical triad of autoimmune hepatitis, thymoma and myasthenia gravis.

Yoneda T, Koba H, Tanimura K, et al.
Postoperative Recurrence of Invasive Thymoma with Cold Agglutinin Disease and Autoimmune Hemolytic Anemia.
Intern Med. 2016; 55(18):2685-9 [PubMed] Related Publications
A 50-year-old man presented to our hospital in 1995. Invasive thymoma was diagnosed and extended thymectomy and left upper lobe partial resection were performed. In 2013, he complained of dyspnea. Chest computed tomography showed postoperative recurrence of invasive thymoma. Several chemotherapies were administered. Severe anemia and an increase in the total bilirubin level were observed with chemotherapies. In additional, an examination showed that the direct Coombs test was positive. Cold agglutinin was also high. We herein experienced a rare case of postoperative recurrence of invasive thymoma with cold agglutinin disease and autoimmune hemolytic anemia.

Stefanou MI, Komorowski L, Kade S, et al.
A case of late-onset, thymoma-associated myasthenia gravis with ryanodine receptor and titin antibodies and concomitant granulomatous myositis.
BMC Neurol. 2016; 16(1):172 [PubMed] Free Access to Full Article Related Publications
BACKGROUND: Myasthenia gravis is an autoimmune neuromuscular disorder, which has only rarely been reported to co-manifest with myositis. The diagnosis of concomitant myositis in patients with myasthenia gravis is clinically challenging, and requires targeted investigations for the differential diagnosis, including EMG, autoantibody assays, muscle biopsy and, importantly, imaging of the mediastinum for thymoma screening.
CASE PRESENTATION: This report presents a case-vignette of a 72-year-old woman with progressive proximal muscle weakness and myalgias, diagnosed with thymoma-associated myasthenia and bioptically verified granulomatous myositis, with positive autoantibody status for ryanodine receptor and titin antibodies.
CONCLUSIONS: The diagnosis of concurrent myositis and myasthenia gravis, especially in the presence of ryanodine receptor and titin antibodies, should lead neurologists to adopt different treatment strategies compared to those applied in myasthenia or myositis alone. Moreover, further evidence is warranted that titin and, particularly, ryanodine receptor antibodies may co-occur or be pathophysiologically involved in myasthenia-myositis cases.

Lococo F, Bajocchi G, Caruso A, et al.
Occasional detection of thymic epithelial tumor 4 years after diagnosis of adult onset Still disease: A challenging case report and immuno-oncological considerations coming from pertinent literature review.
Medicine (Baltimore). 2016; 95(36):e4357 [PubMed] Free Access to Full Article Related Publications
BACKGROUND: Thymoma is a T cell neoplasm arising from the thymic epithelium that due to its immunological role, frequently undercover derangements of immunity such a tumors and autoimmune diseases.
METHODS: Herein, we report, to the best of our knowledge, the first description of an association between thymoma and adult onset Still disease (AOSD) in a 47-year-old man. The first one was occasionally detected 4 years later the diagnosis of AOSD, and surgically removed via right lateral thoracotomy. Histology confirmed an encapsulated thymic tumor (type AB sec. WHO-classification).
RESULTS: The AOSD was particularly resistant to the therapy, requiring a combination of immunosuppressant followed by anti-IL1R, that was the only steroids-sparing treatment capable to induce and maintain the remission. The differential diagnosis was particularly challenging because of the severe myasthenic-like symptoms that, with normal laboratory tests, were initially misinterpreted as fibromyalgia. The pathogenic link of this association could be a thymus escape of autoreactive T lymphocytes causing autoimmunity.
CONCLUSION: Clinicians should be always include the possibility of a thymoma in the differential diagnosis of an unusual new onset of weakness and normal laboratories data, in particular once autoimmune disease is present in the medical history.

Luo T, Zhao H, Zhou X
The clinical features, diagnosis and management of recurrent thymoma.
J Cardiothorac Surg. 2016; 11(1):140 [PubMed] Free Access to Full Article Related Publications
Thymoma is a disease with malignant potential, which has a recurrence rate after complete resection ranging from 5 to 50 %. Multiple studies on the risk factors, treatment or prognosis have been reported. Many of them are controversial, however. In this review, we summarized some accepted risk factors, means of diagnosis and different treatments of recurrent thymoma. The risk factors of recurrent thymoma haven't been well-studied, and its management remains controversial. We reviewed the literatures and found some key points which should be noticed during the surgery of initial thymoma. Although reoperation should be taken into account preferentially, multimodal treatments are also available. The prognosis are also been discussed.

Zhao L, Zhou X, Li Z, Liu Y
Bone metastasis of malignant thymomas associated with peripheral T-cell lymphocytosis.
BMC Surg. 2016; 16(1):58 [PubMed] Free Access to Full Article Related Publications
BACKGROUND: Malignant thymomas are rarely associated with bone metastasis and T-cell lymphocytosis.
CASE PRESENTATION: A 47-year-old female patient was admitted to our hospital for ptosis. A diagnosis of malignant thymoma was made based on the thymectomy and pathological result. Peripheral T-cell lymphocytosis and bone metastasis were found later. T-cell lymphocytosis was relived after surgical and radiation therapy to the metastasis.
CONCLUSION: Peripheral T-cell lymphocytosis is a rare paraneoplastic phenomenon associated with thymomas. This report is the first to describe an invasive thymoma with late bone metastasis accompanied with T-cell lymphocytosis. We should be aware of peripheral T-cell lymphocytosis in thymomas and it may contribute to a better understanding of the complex physiology and pathogenesis of thymoma.

Braham E, Kilani T, El Mezni F
Clinicopathologic and prognostic factors of thymoma in Tunisia.
Tunis Med. 2016; 94(2):118-22 [PubMed] Related Publications
BACKGROUND: although rare, thymomas are the most common tumors of the thymus in adults. They represent about 20% of all mediastinal tumors.
AIM: the aim of this study is to present clinicopathological features of thymomas in Tunisia and analyse the prognostic factors.
METHODS: From 1993 to 2004, clinical data of 40 cases of thymomas were compiled retrospectively. Microscopic slides were reviewed and reclassified according to the WHO classification of thymic tumors 2004. Clinical staging adopted was Masaoka system. Analysis of survival was determined by Kaplan-Meier method and log-rank test was used to compare survival curves. These statistical analyses were performed by SPSS.
RESULTS: they were 23 women and 17 males of ages ranging from 14 to 76 years (mean age 51 years). The distribution of histological WHO types was: 1 type A, 7 type AB, 6 type B1, 17 type B2, 6 type B3, 2 cases of micronodular thymoma with lymphoid stroma and 1 case of metaplastic thymoma. According to Masaoka stage, 10 patients were in stage I, 11 stage II, 9 stage IIIa, 4 stage IIIb, 5 stage Iva and 1 stage IVb. The average overall survival was 56 months. Univariate analyses showed that Masaoka stage, completeness surgical resection and age were prognostic factors whereas in multivariate analysis, age was the only prognostic factor. Neither myasthenia gravis nor histological WHO subtypes had effect in survival.
CONCLUSION: masaoka stage, completeness surgical resection and age are the prognostic factors predicting survival in our series.

Hino H, Nishimura T, Seki A, et al.
Microthymoma in elderly-onset myasthenia gravis detected preoperatively.
Asian Cardiovasc Thorac Ann. 2016; 24(8):818-821 [PubMed] Related Publications
A 77-year-old woman with a 3-month history of muscle weakness was diagnosed with elderly-onset generalized myasthenia gravis (Myasthenia Gravis Foundation of America classification IIa) based on a high serum acetylcholine receptor antibody level (25.4 nmol·L(-1)) and neurological findings. Computed tomography detected a small nodule (diameter 15 mm) in the anterior mediastinum, which was suspected to be a thymoma. An extended thymectomy was performed. The pathological examination revealed a 6-mm-diameter thymoma, termed a microthymoma, accompanied with a unilocular thymic cyst without capsule formation (type B2 according to the World Health Organization classification). Some fat tissue was also found within the tumor.

Ahmad U, Huang J
Induction Therapy for Thymoma.
Thorac Surg Clin. 2016; 26(3):325-32 [PubMed] Related Publications
Thymomas are uncommon tumors that can present as locally advanced tumors in approximately 30% of the patients. Stage and complete resection are the strongest prognostic factors. For locally advanced tumors, induction treatment may improve the ability to achieve a complete resection. Combination treatment with cisplatin, doxorubicin, and cyclophosphamide is the most commonly used induction regimen. Similar rates of resectability are noted with the use of induction chemotherapy and chemoradiation therapy; however, more tumor necrosis is noted with the addition of radiation.

Qi K, Wang B, Wang B, et al.
Video-assisted thoracoscopic surgery thymectomy versus open thymectomy in patients with myasthenia gravis: a meta-analysis.
Acta Chir Belg. 2016; 116(5):282-288 [PubMed] Related Publications
BACKGROUND: Video-assisted thoracoscopic surgery (VATS) thymectomy has become a feasible treatment for myasthenia gravis (MG) in recent years. The objective of the present meta-analysis was to evaluate the perioperative characteristics, safety, and completely stable remission rate in patients with MG who received VATS or open thymectomy (OT).
METHODS: We searched PubMed, Embase, ScienceDirect, Web of Science, and CNKI for related articles using combinations of the search terms video-assisted thoracoscopic thymectomy, transsternal thymectomy, and MG. The inter-study heterogeneity was assessed by χ(2)-based Q statistics, and the extent of inconsistency was generated by I(2) statistics.
RESULTS: A total of 12 studies with 1173 patients were included, and there was no difference in the operation time (p = 0.08) and ICU time (p = 0.14) between the two groups, but VATS thymectomy was associated with less intra-operation blood loss and hospital time (p < 0.00001). VATS was also associated with lower rates of total complication (OR =0.59; 95% CI, 0.37-0.94; p = 0.03) and myasthenic crisis (OR = 0.51; 95% CI, 0.28-0.92; p = 0.03), but the rates of pneumonia (OR = 0.59; 95% CI, 0.29-1.32; p = 0.21) and complete remission rate (CSR) (OR = 0.64; 95% CI, 0.38-1.09; p = 0.10) had no obvious differences between the VATS and OT groups.
CONCLUSION: Patients with MG undergoing VATS thymectomy achieved better surgical outcomes and fewer complications than those who received OT.

Karube Y, Kobayashi S, Maeda S, et al.
Tumor-related gene expression levels in thymic carcinoma and Type B3 thymoma.
J Cardiothorac Surg. 2016; 11(1):85 [PubMed] Free Access to Full Article Related Publications
BACKGROUND: Thymic carcinoma (TC) is a rare type of malignant neoplasm that develops in the anterior mediastinum and associated with poor prognosis. Type B3 thymoma (B3) occasionally demonstrates malignant tumor characteristics, especially in the advanced stage. We investigated the expressions of tumor-related genes in resected TC and B3 specimens.
METHODS: TC and B3 specimens resected from 1999 through 2012 were investigated. Tumor segments were collected from the specimens by micro-dissection to extract mRNA, then RT-PCR was performed according to Dannenberg's tumor profile method for semi-quantitation of tumor-related gene mRNA. To compare with other types of cancer, data from lung cancer (LC) cases in our database were also examined.
RESULTS: The gene expression levels of thymidylate synthase were significantly higher in TC and B3 as compared to LC specimens (p < 0.02), while no difference were observed between TC and B3 tumors. The ratio of folypolyglutamyl synthase (FPGS) to gamma-glutamyl hydrolase (GGH) mRNA was significantly lower in TC than in B3 (p < 0.05), with lower FPGS/GGH in those tumors related to overall survival. Also, the gene expression of vascular endothelial growth factor (VEGF) was significantly higher in TC as compared to B3 (p = 0.04), with higher VEGF gene expression in TC and B3 specimens related to overall survival of affected patients. Epidermal growth factor receptor (EGFR) expression was significantly higher in B3 as compared to both TC and LC specimens (p < 0.01). However, there were no EGFR gene mutations detected in any of the specimens.
CONCLUSIONS: These results indicate that elevated expressions of the tumor-related genes FPGS/GGH and VEGF are correlated with malignancy of TC and B3 tumors. Additional examinations will be necessary to investigate their chemosensitivity.

Weissferdt A, Kalhor N, Moran CA
Thymomas With Extensive Clear Cell Component:  A Clinicopathologic and Immunohistochemical Study of Nine Cases.
Am J Clin Pathol. 2016; 146(1):132-6 [PubMed] Related Publications
OBJECTIVES: Nine cases of thymomas with an extensive clear cell component are presented.
METHODS: The patients were six men and three women aged between 45 and 62 years (mean, 52 years). Presenting symptoms included shortness of breath, chest pain, and cough. Diagnostic imaging revealed anterior mediastinal masses in all patients, and all underwent thymectomy.
RESULTS: Grossly, the tumors varied from 3 to 9 cm in the greatest dimension. Four cases were invasive and five encapsulated. Histologically, the tumors were characterized by an epithelial cell component with extensive clear cell change, admixed with lymphocytes in varying proportions. Immunohistochemically, the tumors demonstrated the typical immunophenotype of thymomas characterized by positive staining of the epithelial cells for pancytokeratin and cytokeratin 5/6, absent expression of CD5 and c-kit, and reactivity of the lymphocytes for terminal deoxynucleotidyl transferase. Clinical follow-up available for six patients showed that all were alive and well 12 to 24 months after surgical resection.
CONCLUSIONS: The cases herein presented highlight an unusual feature in thymoma that has more commonly been ascribed to thymic carcinoma. They also emphasize the importance of correct diagnosis to determine the appropriate treatment strategy and to accurately predict prognosis.

Habib AM, Thornton H, Sewell WC, Loubani M
Good's syndrome: Is thymectomy the solution? Case report and literature review.
Asian Cardiovasc Thorac Ann. 2016; 24(7):712-4 [PubMed] Related Publications
Good's syndrome is thymoma accompanied by immunodeficiency. A 69-year-old woman presented with recurrent chest infections, hypogammaglobulinemia, and radiological features of a thymoma. Immunoglobulin replacement therapy was not tolerated prior to surgery. Postoperative recovery was uneventful, and a Masaoka stage II type AB thymoma was confirmed on histology. One-year follow-up revealed no recurrence of the thymoma but the patient remained hypogammaglobulinemic and developed collagenous colitis. She declined immunoglobulin replacement therapy but remains under follow-up. Awareness of Good's syndrome to avoid overwhelming infection is emphasized. The finding of thymoma should prompt the thoracic surgeon to test for immunodeficiency.

Weissferdt A, Moran CA
The spectrum of ectopic thymomas.
Virchows Arch. 2016; 469(3):245-54 [PubMed] Related Publications
Thymomas are rare tumors that usually manifest in the anterosuperior mediastinum. Occasionally, thymomas may also originate from ectopically dispersed thymic tissue and can arise in locations such as the neck, lung, or pleura or other locations in the thoracic cavity. The occurrence of thymomas in these ectopic locations can cause substantial diagnostic difficulty as the entity is almost never included in the differential diagnosis and its biphasic morphology can cause further complications during the diagnostic process. In this review, we summarize the clinical and pathological spectrum of ectopic thymomas and discuss the histogenesis, treatment, and prognosis of these extraordinary tumors.

Fiorelli A, Mazzella A, Cascone R, et al.
Bilateral thoracoscopic extended thymectomy versus sternotomy.
Asian Cardiovasc Thorac Ann. 2016; 24(6):555-61 [PubMed] Related Publications
BACKGROUND: Complete open surgical resection is the standard treatment for thymoma and myasthenia gravis. We evaluated the feasibility of bilateral video-assisted thoracoscopic extended thymectomy, and compared it to surgery via sternotomy.
METHODS: From 2011 to 2014, 43 patients undergoing thymectomy were divided into 2 groups: 23 underwent video-assisted thoracoscopic extended thymectomy, and 20 had thymectomy via sternotomy. The primary outcomes were postoperative pain score (visual analog scale) at 6, 12, 24, 48, and 72 h, and 1-month postoperatively, and morphine consumption in the first 48 h. Secondary outcomes were surgical and clinical results.
RESULTS: There were no significant differences between the 2 groups in terms of demographics and preoperative clinical data. Compared to the sternotomy group, the video-assisted thoracoscopic thymectomy group had lower pain scores and morphine consumption at all time points, significantly less operative blood loss and chest drainage volume, and shorter hospital stay. The rates of improvement in myasthenia gravis were 85% and 86% in the video-assisted thoracoscopic thymectomy and sternotomy groups, respectively. No recurrence of thymoma was found in either group (median follow-up 27 months).
CONCLUSIONS: Our results seem to confirm that in selected cases, video-assisted thoracoscopic thymectomy allows complete resection of thymus and perithymic tissue, similar to sternotomy but with the known advantages of minimally invasive surgery including less pain and a good cosmetic result.

Parihar S, Gohil R, Oparka R, Kennedy C
Benign anlage tumour: a very unusual neck mass.
BMJ Case Rep. 2016; 2016 [PubMed] Related Publications
A 44-year-old woman presented with a slow-growing asymptomatic neck swelling at the left medial clavicle. Haematological and biochemical work up was normal and an ultrasound confirmed the swelling, but needle aspiration was non-diagnostic. As lymphoma was the main differential diagnosis, the swelling was completely excised. Immunohistochemistry yielded a rare lesion, suspected to represent a myoepithelial/mixed cellularity tumour of soft tissue. The extreme rarity of these tumours required a confirmatory secondary opinion, which ultimately led to it being identified as a benign anlage tumour (previously known as an ectopic hamartomatous thymoma) This case highlights the fact that thorough assessment of patients with neck swellings should be undertaken to rule out sinister causes-keeping in mind more rare differentials-helping to guide final management.

Chaudhuri AD, Tapadar SR, Dhua A, et al.
A Case of Good's Syndrome Presenting with Pulmonary Tuberculosis.
Indian J Chest Dis Allied Sci. 2015 Oct-Dec; 57(4):247-50 [PubMed] Related Publications
Adult onset immunodeficiency associated with thymoma is a rare condition. The combination of hypogammaglobulinemia, reduced number of peripheral B and CD4+ T cells, along with thymoma constitutes Good's syndrome (GS). This immunodeficiency condition is often complicated with opportunistic infection with organisms, like bacteria (Haemophilus influenzae, Streptococcus pneumonia etc), viruses (Cytomegalovirus, Herpes simplex etc), fungi and protozoa. We present an unusual case of Good's syndrome with pulmonary tuberculosis (PTB). A 40-year-old man presented with sputum-positive PTB and was started on anti-tuberculosis treatment. Subsequently, he developed symptoms and findings consistent with thymoma and other components of Good's syndrome. Although patients of Good's syndrome are susceptible to various opportunistic infections, infection with Mycobacterium tuberculosis is uncommon. Evidence of recurrent infections or some opportunistic infection in a thymoma patient should trigger a suspicion of Good's syndrome.

Bak V, Spalek P, Rajcok M, et al.
Importance of thymectomy and prognostic factors in the complex treatment of myasthenia gravis.
Bratisl Lek Listy. 2016; 117(4):195-200 [PubMed] Related Publications
BACKGROUND: Thymectomy is indicated in patients with seropositive myasthenia gravis (SPMG) in patients under the age of 50 and in patient with myasthenia gravis (MG) associated with thymoma.
METHODS: 345 patients with MG who underwent an extended thymectomy from April 1990 to December 2010. Patients were separated into the 4 groups: group with a complete stable remission, pharmacological remission group, group of patients with significant improvement and the group with mild improvement of symptoms.
RESULTS: In our study, we observed 345 patients with MG and thymectomy. 137 patients (39.71 %) attained the complete stable remission (CSR) and 92 patients (26.67 %) the pharmacological remission (PR). The significant improvement of MG symptomatology was achieved in 95 patients (27.54 %). The rest of 21 patients from total 345 (6.08 %) have reached only a mild improvement. Patients after thymectomy with CSR were in the clinical stage I and IIA, in accordance to the modified Osserman classification and most benefited from the thymectomy.
CONCLUSIONS: The extended thymectomy combined with immunotherapy is the preferred treatment with an expected satisfactory long-term remission rate. Despite the recent improvements in MG medical therapy, thymectomy plays an important role in the otherwise complex treatment of the disease. Because of early diagnosis with thymectomy performed without a delay, patients can achieve significantly more often favorable outcomes and even stable remissions (Tab. 4, Fig. 7, Ref. 17).

Matsuoka K, Murata Y, Ueda M, Miyamoto Y
Ectopic thymic carcinoma presenting as an intrathoracic mass.
Asian Cardiovasc Thorac Ann. 2016; 24(5):480-3 [PubMed] Related Publications
An asymptomatic 83-year-old man was found to have a right intrathoracic tumor. Computed tomography demonstrated a soft-tissue density mass measuring 55 × 25 × 22 mm adjacent to the right anterior chest wall. At surgery, the tumor was found to adhere to the diaphragm and right lung, contiguous with the mediastinal fat tissue. Histology of the resected specimen demonstrated proliferation of spindle and sarcomatous cells with multinucleated giant cells. Thus the tumor was diagnosed as undifferentiated thymic carcinoma and was considered to have arisen from ectopic thymic tissue. At 2 years postoperatively, the patient had no evidence of recurrence.

Lim YJ, Kim E, Kim HJ, et al.
Survival Impact of Adjuvant Radiation Therapy in Masaoka Stage II to IV Thymomas: A Systematic Review and Meta-analysis.
Int J Radiat Oncol Biol Phys. 2016; 94(5):1129-36 [PubMed] Related Publications
PURPOSE: To evaluate the survival impact of postoperative radiation therapy (PORT) in stage II to IV thymomas, using systematic review and meta-analysis.
METHODS AND MATERIALS: A database search was conducted with EMBASE, PubMed, Web of Science, Cochrane Library, and Ovid from inception to August 2015. Thymic carcinomas were excluded, and studies comparing overall survival (OS) with and without PORT in thymomas were included. The hazard ratios (HRs) of OS were extracted, and a random-effects model was used in the pooled analysis.
RESULTS: Seven retrospective series with a total of 1724 patients were included and analyzed. Almost all of the patients underwent macroscopically complete resection, and thymoma histology was confirmed by the World Health Organization criteria. In the overall analysis of stage II to IV thymomas, OS was not altered with the receipt of PORT (HR 0.79, 95% confidence interval [CI] 0.58-1.08). Although PORT was not associated with survival difference in Masaoka stage II disease (HR 1.45, 95% CI 0.83-2.55), improved OS was observed with the addition of PORT in the discrete pooled analysis of stage III to IV (HR 0.63, 95% CI 0.40-0.99). Significant heterogeneity and publication bias were not found in the analyses.
CONCLUSIONS: From the present meta-analysis of sole primary thymomas, we suggest the potential OS benefit of PORT in locally advanced tumors with macroscopically complete resection, but not in stage II disease. Further investigations with sufficient survival data are needed to establish detailed treatment indications.

Mlika M, Boudaya S, Braham E, et al.
About thymic carcinomas: challenges in diagnosis and management.
Asian Cardiovasc Thorac Ann. 2016; 24(4):350-4 [PubMed] Related Publications
BACKGROUND: Thymic carcinomas are rare tumors with a challenging diagnosis. Our aim was to report our 17-year experience of these tumors and to highlight the challenges encountered and the main differential diagnoses ruled out.
METHODS: We studied 12 (92%) men and 1 (7.7%) woman with a mean age of 37 years (range 15-60 years). All patients were symptomatic, with chest pain representing the most frequent symptom. Radiology revealed anterior mediastinal masses in all cases, with either infiltration of the adjacent organs or pulmonary parenchymal metastases.
RESULTS: The diagnosis was made on surgical biopsies in 12 cases and a lymph node biopsy in one. Microscopic examination revealed squamous carcinoma in 3 cases, synovial sarcoma in 1, mucinous adenocarcinoma in 1, undifferentiated carcinoma in 2, clear cell carcinoma in 1, lymphoepithelioma-like carcinoma in 2, atypical carcinoid tumor in 2, and sarcomatoid carcinoma in 1. Total surgical resection was possible in one patient after neoadjuvant chemotherapy and radiotherapy. Follow-up was possible in only 6 patients, and the mean survival reached 13 months.
CONCLUSION: In spite of the lack of follow-up information, this study demonstrates the poor outcome associated with these tumors and the need for standardized treatment.

Zou J, Su C, Lun X, et al.
Preoperative Anxiety in Patients With Myasthenia Gravis and Risk for Myasthenic Crisis After Extended Transsternal Thymectomy: A CONSORT Study.
Medicine (Baltimore). 2016; 95(10):e2828 [PubMed] Free Access to Full Article Related Publications
A thymectomy can ameliorate the symptoms of myasthenia gravis (MG) and prevent the progression of ocular MG (OMG) to generalized MG (GMG). However, postoperative myasthenic crisis (POMC) is a serious post-thymectomy complication. Preoperative anxiety (POA) is common but typically neglected in MG patients. The association of POA with POMC has not yet been examined.From June 2007 to December 2013, 541 cases of MG were admitted to the First Affiliated Hospital of Sun Yat-sen University (Guangzhou, China). All cases underwent extended transsternal thymectomy (ETT). The clinical and pathological characteristics of these patients, including POA and POMC, were analyzed.A total of 179 patients experienced POA and 67 patients experienced POMC. Patients with POA were more likely to have POMC, a thymoma, and an ectopic thymus. Univariate analysis showed that POMC correlated with POA, presence of an ectopic thymus, dose of pyridostigmine bromide (PYR), presence of a thymoma, MGFA stage, preoperative myasthenic crisis, and postoperative pneumonia. Multivariate logistic regression analysis showed that the independent risk factors for POMC were POA, preoperative myasthenic crisis, higher dose of PYR, and postoperative pneumonia.Our results suggest that clinicians should consider the risk factors for POMC-especially preoperative anxiety-before performing a thymectomy in patients with MG.

Dasgupta S, Bose D, Bhattacharyya NK, et al.
A clinicopathological study of mediastinal masses operated in a tertiary care hospital in Eastern India in 3 years with special reference to thymoma.
Indian J Pathol Microbiol. 2016 Jan-Mar; 59(1):20-4 [PubMed] Related Publications
INTRODUCTION: The mediastinum is the central portion of the thoracic cavity, limited by pleural cavities laterally, thoracic inlet superiorly, and the diaphragm inferiorly. Housing numerous organs, it is a veritable Pandora's box, within which various lesions may develop. This study was conducted to assess the epidemiologic profile, clinicoradiological features, cytological, and histopathological findings in patients presenting with mediastinal masses in a tertiary care hospital over a period of 3 years.
MATERIALS AND METHODS: This is a retrospective study of cases presenting with mediastinal masses attending the Cardiothoracic Surgery Department of Medical College, Kolkata between May 2011 and April 2014. Detailed history, physical, and radiological findings were noted. Fine needle aspiration cytology (FNAC) was performed when feasible. Following surgery, histopathological, and immunohistochemical (IHC) examinations of the specimens were undertaken.
RESULTS: Of the 22 cases included in our study, ten were anterior, seven middle, and five posterior mediastinal masses. Fifteen cases were male and seven were female. Thymic pathology was detected in seven cases, lymphoma in five, extragonadal germ cell tumor (GCT) in three, schwannoma and pericardial cyst in two cases each and neurofibroma, ganglioneuroma, and retrosternal thyroid in one case each. The age group of the patients for each diagnostic category was found to be of significance. FNAC was done in 15 cases. IHC was required for classification of lymphoma cases (CD45, CD15, CD30, CD20, CD3, Tdt, CD34, and Ki-67).
CONCLUSION: This study reflects the incidence of different mediastinal masses in West Bengal with their clinicopathologic correlation.

Okusu T, Sato T, Ogata Y, et al.
Good's Syndrome Accompanied by Agranulocytosis Following a Rapid Clinical Course.
Intern Med. 2016; 55(5):537-40 [PubMed] Related Publications
Good's syndrome is an immunodeficiency disease involving thymoma accompanied by hypogammaglobulinemia. We encountered a case of Good's syndrome accompanied by agranulocytosis that followed a rapid clinical course. A 72-year-old man visited our hospital with a two-week history of a sore throat. Candida albicans was detected in the pharynx, and hypogammaglobulinemia was detected in addition to granulocytopenia. The patient subsequently developed septic shock and followed a rapid clinical course which ended in death. Good's syndrome with agranulocytosis was diagnosed at autopsy. Good's syndrome accompanied by agranulocytosis can follow a rapid clinical course and some cases remain asymptomatic until old age. Its prompt treatment is crucial.

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