Resected invasive thymoma with multiple endocrine neoplasia type 1

Kojima, Yasuyuki; Ito, Hiroyuki; Hasegawa, Shin; Sasaki, Takeshi; Inui, Kentaro

doi:10.1007/bf02662474

Cited by 21 publications

(10 citation statements)

References 4 publications

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“…While the reasons for this difference are unclear, ethnicity could be one explanation. Several additional case reports exist on Japanese female MEN1 patients with Th‐NET who have not been registered in our database . Similarly, there have been some case reports from Asian and Western countries on female MEN1 patients with Th‐NET, indicating that there may be more female Th‐NET patients than previously considered.…”

Section: Discussionmentioning

confidence: 92%

Thymic neuroendocrine tumour in multiple endocrine neoplasia type 1: female patients are not rare exceptions

Sakurai

Imai

Kikumori

et al. 2013

Clinical Endocrinology

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Summary Objective Thymic neuroendocrine tumour (Th‐NET) occurs in 2–5% of patients with MEN1 and has high malignant potency accompanying recurrence and distant metastasis. While Th‐NET is recognized to develop predominantly in men and heavy smokers, a number of female patients have been reported in the literature. The objective of this study is to clarify the clinical features of MEN1 patients with Th‐NET using database analysis. Design/Patients Clinical data of patients with Th‐NET were extracted and analysed from a recently constructed database of Japanese MEN1 patients. Results Among 560 registered cases, Th‐NET was seen in 28 (5·0%) patients. Of note, 36% of patients (10/28) were women; only one patient among those was a smoker and another six patients were non‐smokers. Age at diagnosis of Th‐NET and MEN1, tumour size, prevalence of other MEN1‐related tumours did not differ between male and female patients, and 10‐year survival probability was 0·271 ± 0·106. Conclusions Although the prevalence of Th‐NET in women (3·2%) is significantly lower than that in men (7·6%), a considerable proportion of female patients develop Th‐NET. Given that Th‐NET is a major determinant of life expectancy of patients, our results alert clinicians who treat patients with MEN1 that surveillance of Th‐NET is essential even for female patients without a smoking habit.

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Section: Discussionmentioning

confidence: 92%

Thymic neuroendocrine tumour in multiple endocrine neoplasia type 1: female patients are not rare exceptions

Sakurai

Imai

Kikumori

et al. 2013

Clinical Endocrinology

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“…Recently it is increasingly recognized that MEN1 patients have an increased occurrence of other endocrine and non-endocrine tumors including carcinoid tumors (thymic [0–8%], gastric [7–35%], bronchial [0–8%], rarely intestinal); skin tumors [angiofibromas (88%), collagenomas (72%), lipomas (34%), melanomas]; central nervous system tumors(meningiomas, ependymonas, schwanomas)[0–8%]; and smooth muscle tumors (leiomyomas, leiomyosarcomas) [1–7%] 13,46,48,49,74,131,176,209,228,261,274,354,388,393,413,465 . In other reports small numbers of other tumors are also described, although it is unclear if they are increased in frequency or aggressiveness in MEN1 patients [lymphoma, renal cancer, hematological disorders (thrombotic thrombocytopenic purpura, myeloma), ovarian tumors, gastrointestinal stromal tumors, seminomas, chondrosarcoma, mesothelioma, thymomas] 1,77,84,89,150,214,216,256,312,341,410,432 .…”

Section: Introductionmentioning

confidence: 99%

Causes of Death and Prognostic Factors in Multiple Endocrine Neoplasia Type 1

et al. 2013

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Multiple endocrine neoplasia type 1 (MEN1) is classically characterized by the development of functional or nonfunctional hyperplasia or tumors in endocrine tissues (parathyroid, pancreas, pituitary, adrenal). Because effective treatments have been developed for the hormone excess state, which was a major cause of death in these patients in the past, coupled with the recognition that nonendocrine tumors increasingly develop late in the disease course, the natural history of the disease has changed. An understanding of the current causes of death is important to tailor treatment for these patients and to help identify prognostic factors; however, it is generally lacking.To add to our understanding, we conducted a detailed analysis of the causes of death and prognostic factors from a prospective long-term National Institutes of Health (NIH) study of 106 MEN1 patients with pancreatic endocrine tumors with Zollinger-Ellison syndrome (MEN1/ZES patients) and compared our results to those from the pooled literature data of 227 patients with MEN1 with pancreatic endocrine tumors (MEN1/PET patients) reported in case reports or small series, and to 1386 patients reported in large MEN1 literature series. In the NIH series over a mean follow-up of 24.5 years, 24 (23%) patients died (14 MEN1-related and 10 non-MEN1-related deaths). Comparing the causes of death with the results from the 227 patients in the pooled literature series, we found that no patients died of acute complications due to acid hypersecretion, and 8%–14% died of other hormone excess causes, which is similar to the results in 10 large MEN1 literature series published since 1995. In the 2 series (the NIH and pooled literature series), two-thirds of patients died from an MEN1-related cause and one-third from a non-MEN1-related cause, which agrees with the mean values reported in 10 large MEN1 series in the literature, although in the literature the causes of death varied widely. In the NIH and pooled literature series, the main causes of MEN1-related deaths were due to the malignant nature of the PETs, followed by the malignant nature of thymic carcinoid tumors. These results differ from the results of a number of the literature series, especially those reported before the 1990s. The causes of non-MEN1-related death for the 2 series, in decreasing frequency, were cardiovascular disease, other nonendocrine tumors > lung diseases, cerebrovascular diseases. The most frequent non-MEN1-related tumor deaths were colorectal, renal > lung > breast, oropharyngeal. Although both overall and disease-related survival are better than in the past (30-yr survival of NIH series: 82% overall, 88% disease-related), the mean age at death was 55 years, which is younger than expected for the general population.Detailed analysis of causes of death correlated with clinical, laboratory, and tumor characteristics of patients in the 2 series allowed identification of a number of prognostic factors. Poor prognostic factors included higher fasting gastrin levels, presence of othe...

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“…Kojima et al . [4] reported a mixed-type invasive thymoma (WHO histological type was not available) with MEN 1. De Toma et al .…”

Section: Discussionmentioning

confidence: 99%

Thymoma (World Health Organization type B3) with neuroendocrine differentiation in multiple endocrine neoplasia type 1

Tomita

Ichiki

Ayabe

et al. 2017

Journal of Surgical Case Reports

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Thymic epithelial tumors occur in 1–5% of patients with multiple endocrine neoplasia type 1 (MEN 1). Majority of these thymic epithelial tumors are thymic carcinoids and patients with thymoma in MEN 1 is rare. Furthermore, thymoma with neuroendocrine differentiation was also rarely reported. Herein, we report a 68-year-old man having type B3 thymoma with neuroendocrine differentiation in MEN 1 and to the best of our knowledge this is the first such case ever reported.

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Resected invasive thymoma with multiple endocrine neoplasia type 1

Cited by 21 publications

References 4 publications

Thymic neuroendocrine tumour in multiple endocrine neoplasia type 1: female patients are not rare exceptions

Thymic neuroendocrine tumour in multiple endocrine neoplasia type 1: female patients are not rare exceptions

Causes of Death and Prognostic Factors in Multiple Endocrine Neoplasia Type 1

Thymoma (World Health Organization type B3) with neuroendocrine differentiation in multiple endocrine neoplasia type 1

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