PMC:3727638 / 73581-92252 JSONTXT

{"target":"https://pubannotation.org/docs/sourcedb/PMC/sourceid/3727638","sourcedb":"PMC","sourceid":"3727638","source_url":"https://www.ncbi.nlm.nih.gov/pmc/3727638","text":"Causes of MEN1-Related Death\nIn both our 106 NIH MEN1/ZES patients and the 227 MEN1/PET patients from the pooled literature, all of the MEN1-related deaths were due in some way to a NET, either because of its malignancy, complication of its treatment (postoperative death, etc) or due to the hormone excess state associated with it (for example, PET hormone secretion, HPT) (see Table 2). Of the MEN1-related deaths, 79% and 83% in the 2 groups of patients were due to the malignant nature of the NET, 14%–17% due to a NET hormone excess state, and 4%–7% due to a NET-related problem not due to progressive metastatic disease or the hormone excess state. The malignant NET deaths (79%–83% of MEN1 deaths) were primarily due to deaths from malignant PETs (57%–58% total MEN1 deaths) or to deaths from thymic carcinoid tumors (19%–21% of MEN1 deaths).\nTo compare our data to MEN1 series in the literature, all causes of death were expressed as a percentage of the total deaths, as was done in most of these series (see Table 12). When expressed in this form, our data demonstrated the main single cause of death was due to PETs accounting for 38% (NIH) and 44% (pooled literature series) of the total deaths, followed by death from thymic carcinoid tumors (12%–13%). For 15 large series of MEN1 patients in the literature, the average percentage of death due to a PET-related illness was 53.1 ± 7% of all deaths; however, this percentage, as well as the exact causes of the PET-related death, varied widely in the individual series, with the total percentage varying from 19% of all deaths78,251 to 91% of all deaths.254 In the NIH and pooled literature series, the major cause of a PET-related death was the malignant nature of the PET, responsible for 36% of the total 44% PET-related deaths in the pooled literature series and for all 38% of the PET-related deaths in the NIH patients. In 14 of 15 of the large MEN1 literature series (see Table 12), PET-related illnesses were also the major cause of death (mean, 56% ± 7%); however, in 1 series251 the main cause of death was related to malignant thymic carcinoid tumors (24% of all deaths). Similar to our 2 groups of MEN1 patients (the NIH and pooled literature series), in 8 of the 14 literature MEN1 series where PET-related illnesses were the leading cause of death,57,78,88,129,150,217,378,379,448,465 the major cause of the PET-related death was the malignant nature of the PET, averaging 39% ± 9% of all deaths (range, 14%–83% of all deaths).\nIn our 2 series (the NIH and pooled literature series), death due to a malignant PET was most frequently due to a malignant gastrinoma (60% and 100% of cases). This result is similar to that reported in 6 series in the literature54,78,224,226,251,289,367,444 where the percentage of malignant gastrinomas responsible for the PET-related malignant deaths could be assessed; however, it differs from results in 4 series129,150,378,379,448,465 wherein malignant gastrinomas were responsible for only an average of 16% (range, 0–38%) of the malignant PET deaths. It is unclear from the data available in these latter reports whether this low percentage of malignant gastrinomas contributing to the overall number of malignant PETs is clearly reflective of the actual findings in these patients, because in most of these series the specific nature of the malignant PET is poorly described. Therefore, while our data and most recent literature series clearly establish that the malignant nature of the PET is now the leading cause of death in MEN1 patients, it is not clearly established what type of PET is responsible for this, particularly whether the majority of these malignant PETs are nonfunctional PETs or gastrinomas.\nIn 6 of the 14 literature series where a PET-related illness was the leading cause of death15,54,224,226,254,289,367,444 (see Table 12), the hormone excess state was the leading cause of death, and in each case it was primarily due to uncontrolled gastric acid hypersecretion in patients with ZES. These series generally included patients prior to the widespread availability of effective medical management for the gastric acid hypersecretory state of patients with ZES (in the 1980s). Even though a number of studies have reported that the gastric acid hypersecretion in patients with MEN1/ZES, especially those with uncontrolled HPT, may be difficult to control, compared to patients with sporadic ZES,140,189,273,281,317 the data from our NIH patients and from a number of the recent MEN1 literature studies reviewed in Table 12 support the conclusion that current medical gastric antisecretory treatment in these patients is very effective. Especially compared to earlier series prior to the effective medical treatment of the gastric acid in MEN1/ZES patients, there was a low rate of deaths due to gastric acid hypersecretion in 10 of the MEN1 series since 1990 (3.7 ± 1.3).54,57,150,217,251,286,378,379,448,465 This is similar to our results in the 106 NIH MEN1/ZES patients, where 0 patients died due to gastric acid hypersecretion.\nEven though gastric acid hypersecretion is now being controlled in almost every MEN1/ZES patient, in contrast to some older studies,15 other hormone excess states per se are an uncommon cause of death in the current series and in recent series. Specifically, in the NIH patients, only 1/106 (0.9%) patients died from another hormone excess state (insulinoma), and in the 227 MEN1/PET patients from the pooled literature, 2.2% (5/227) died from another non-gastrinoma hormone excess state (8-insulinomas, 1-VIPoma). Even this may be an overestimation because 3 of the 8 insulinoma patients from the pooled literature had malignant insulinomas, and it is unclear whether they died of progression of the malignant disease or of refractory hypoglycemia. Including the 5 patients from the pooled literature series with glucagonomas who died from an MEN1-related cause, all died from progressive metastatic disease. These data support the conclusion that death from the hypersecretion of a PET other than gastrinoma is an uncommon cause of death in MEN1 patients at present. These conclusions are supported by the data from 11 various large literature MEN1 series, where in 8 series, reporting 257 deaths of MEN1 patients,57,129,251,254,277,299,371,444 no patients died from insulinoma, and in 3 other series54,78,448 comprising 161 deaths of MEN1 patients, only 2 deaths (1.2%) were due to an insulinoma. Furthermore in the Groupe d’etude des Tumeurs Endocrines (GTE) MEN1 series comprising 758 patients, the presence of an insulinoma was not associated with increased mortality, in contrast to the presence of other PETs in MEN1 patients.71,150 These data are at some variance with some older studies. Ballard et al,15 in their sentinel 1964 study of MEN1 where they reviewed findings in an extensive MEN1 kindred as well as 74 additional cases previously reported in the literature, concluded that hypoglycemia suggesting a possible insulinoma was frequent in MEN1 patients, occurring in 36% of cases, and that 5 patients (6%) died of insulinomas, suggesting that it was not an infrequent cause of death. Subsequently most studies show that insulinomas occur less frequently than this in MEN1 patients, occurring in 11.8% of 758 MEN1 patients in the GTE study148,150 and in 18% in a recent review of MEN1 cases in a number of series.102,174,189,195,212,261,348,418,436 Both the surgical curability and more effective medical control of the rare MEN1 patient with an insulinoma are contributing to the current low rate of insulinoma as a cause of death in recent MEN1. In recent large series only a small percentage (0–15%) of insulinomas in MEN1 patients are reported to be malignant,22,71,82,153,195,205,247,279,330 and the remaining 80%–100% of the MEN1 patients with insulinomas are cured postresection.21,22,71,75,82,153,195,247,319,330,425 Short-term medical control with diazoxide, diet, or somatostatin analogues prior to surgery is effective in controlling hypoglycemia in most patients,76,81,82,144,196 and therefore uncontrolled hypoglycemia resulting in death is primarily restricted at present to the small percentage with a malignant insulinoma (\u003c1% of all patients) and to an even smaller group comprised of the few patients with malignant insulinomas not controlled by medical therapies (diazoxide, somatostatin analogue, mTOR inhibitors [everolimus, rapamycin])76,144,196,221 or more recently by peptide radio-receptor therapy using radiolabeled somatostatin analogues.76,144,441\nOther functional PET syndromes (not including insulinomas or gastrinomas) were a rare cause of death in our 2 study groups (the NIH group and the patients from the pooled literature series), causing no deaths in the 106 NIH MEN1/ZES patients and 1 death (0.4%) (a VIPoma) in the 227 MEN1/PET patients from the pooled literature. These data are in agreement with the results reported in 13 large literature series54,57,78,129,224,251,254,367,371,444,448,474 which contain data of 463 MEN1 patient deaths, where only 2 deaths (0.4%) were reported due to a glucagonoma and 1 death (0.2%) to a growth hormone-releasing factor-secreting tumor (GRFoma). These rare PETs are frequently malignant when present in MEN1 patients (33%7–50% of cases).51,125,195,233,334 In the large GTE series comprising 758 MEN1 patients150,233 in which glucagonomas/VIPomas/somatostinomas were present in 3.3% of all patients, their presence was associated with decreased overall survival (HR = 4.29; 95% CI, 1.54–11.9) and with a decreased 10-year survival (54%) compared to patients with gastrinomas or insulinomas (82%–92%), and was similar to patients with only nonfunctional PETs (62%). While these latter data show that the presence of these rare PETs can affect survival in MEN1 patients, in the current series as in most of the recent literature series reviewed above, the low frequency of deaths caused by these rare PETs in the MEN1 patients is likely due to their low rate of occurrence in MEN1, reported as 1.6%–3% for glucagonomas, 1%–3% for VIPomas, and 0.65% to \u003c1% for GRFomas or somatostatinomas.102,152,174,189,195,212,233,261,308,348,418,436\nThe second largest single cause of MEN1 death in our 2 groups of patients was thymic carcinoid tumors (12% of the NIH MEN1/ZES patients and 12.5% of the pooled literature series). This finding has both similarities and marked differences from the findings in the 15 large MEN1 literature series reviewed in Table 12 and other recent literature series111,150,371,373 reporting assessment of thymic carcinoids in MEN1 patients. Our results are similar to 2 series129,217 that report thymic carcinoids as the second leading cause of MEN1 death (6%–13% of all deaths) behind PET-related deaths. However, they differ from 1 study reporting thymic carcinoids as the most frequent cause of MEN1-related deaths (24% compared to 10% for malignant PETs), and from 12 series in which thymic carcinoids either caused no deaths15,54,57,78,88,226,254,289,367,394,463 or caused only a small percentage of MEN1-related deaths (5%–6%),150,378,379,465 which was much less than seen with malignant PETs. Almost all of these latter series not reporting thymic carcinoids in MEN1 patients were older series, specifically, they include patients followed before 2000. The likely reason that thymic carcinoids were not reported is because thymic carcinoids were not recognized as part of MEN1 until 1972,363 and were not recognized as a major cause of death until the 1990s;131,378,465 thus they were not specifically sought for or considered as a cause of death in many of these older series. Since the 1990s a number of studies have called attention to the increasing importance of thymic carcinoids as a cause of death in MEN1, particularly in men, because \u003e90% occur in males.45,111,131,151,414,465,477,479 These tumors are particularly aggressive, and are associated with a poor prognosis, with more than one-half of patients in some series having metastases at diagnosis, and even more developing them during follow-up, particularly to the bone.45,111,131,151,363,414,477,479 The aggressive nature of these tumors is supported by the results of the large, retrospective GTE study composed of 758 MEN1 patients, in which the presence of thymic carcinoids was reported to be associated with a higher risk of death compared with unaffected patients (HR = 4.64; 95% CI, 1.7–12.4]. The malignant nature of the thymic carcinoid itself is responsible for almost all deaths in MEN1 patients with these tumors, because MEN1 patients with thymic carcinoids rarely develop a hormone excess state.111,130,131,151,414,470,479 In this sense thymic carcinoids in MEN1 patients differ from those occurring in patients without MEN1 (that is, sporadic thymic carcinoids), because a hormone excess state due to the thymic carcinoid is very infrequent (only 4 cases reported that we know of),111,130,408,470 and the hormone excess state was only Cushing syndrome when it occurred, whereas in patients with sporadic thymic carcinoids, a hormone excess state is more frequent (up to 40%) and more varied, with Cushing syndrome, carcinoid syndrome, and acromegaly being reported.38,111,131,185,342,392 Our results and those from a number of studies in the literature support the increasing importance of thymic carcinoids as a cause of death in MEN1 patients. In our series they occurred in 6% of all NIH patients, and in various retrospective studies they occurred in 2.6%,151 3%,45,57,151 4.2%,305 and 4.9%479 of all MEN1 patients; however, in our series they accounted for 12% (NIH) and 12.5% (pooled literature series), and in some recent series they account for up to 24%251 of all MEN1 deaths, demonstrating their increasingly important role as a cause of death in MEN1 patients.\nIn addition to PETs and thymic carcinoid tumors, other MEN1 features have been reported to be an important cause of MEN1-related death in a number of studies, particular some older series. Complications of uncontrolled HPT including renal failure and hypercalcemic crises are reported to contribute to MEN1-related deaths in 10%–12% of patients in 3 older series of MEN1 patients224,226,378,379,444,465 and in occasional patients in other reports.78,150 In more recent series,54,57,129,217,251,371,448,474 including all of the NIH patients in the current series, death from complications of uncontrolled HPT were not seen, because of the greater understanding of the hyperplastic nature of the parathyroid disease and the need for partial or total parathyroidectomy to control the HPT.42,104,149,168,211,328,330 Pituitary diseases (pituitary apoplexy, tumor invasion) are reported to either be a cause of or contribute to MEN1-related death in 3%–6.5% of patients in a few older series.49,129,150,222,378,379,465 Pituitary tumors in MEN1 patients are more aggressive, more frequently multiple and invasive than in patients with sporadic pituitary tumors.49,147,435,447 However, in the NIH prospective series as well as in most current series, although pituitary adenomas are frequently present (20%–65% of patients), they are recognized and adequately treated, so they rarely cause death.54,57,78,88,195,217,251,448 This latter conclusion is also supported from the findings of the GTE study involving 758 MEN1 patients, where the presence of pituitary disease was not associated with an increased risk of death in MEN1 patients (HR = 1.17; 95% CI, 0.72–1.90; p = 0.54).150\nAdrenal disease is present in 10%–73% of MEN1 patients on imaging studies23,48,128,228,374,388,390,453 and is reported to cause MEN1-related deaths in occasional series (0.4%,150 4%465) and case reports.161,385 Adrenal tumors in MEN1 patients are reported to cause a number of functional hormone excess syndromes including Cushing syndrome, hyperaldosteronism, pheochromocytoma, androgen secretion, or feminization.8,27,48,57,128,157,228,261,276,385,388,390,436,453 Functional hormone excess states caused by the adrenal tumors are rarely a cause of MEN1-related death, because in most series \u003e90% of the adrenal lesions are not associated with a functional syndrome (except 1 series228 where 38% with adrenal lesions had Cushing syndrome).48,388,390 In various series,23,48,128,140,157,374,385,390,453 0–6% of MEN1 patients develop adrenal cortical carcinomas, which can be aggressive, invasive, and associated with a functional syndrome such as Cushing syndrome, and which can contribute to or cause death in these patients. However, in both our series including the 106 NIH MEN1/ZES patients and the 227 MEN1/PET patients from the pooled literature, no patient died of adrenal disease, similar to what was reported in a number of recent large MEN1 series,23,78,129,157,217,448 supporting the conclusion that it is an uncommon cause of death at present in MEN1 patients. This conclusion is also supported by the results from the 758-patient GTE study of MEN1 patients. In that study, those with adrenal tumors had a trend toward an increased risk of death compared to unaffected patients, but it did not reach significance (p = 0.064) (HR = 1.72; 95% CI, 0.97–3.06).150\nIn addition to thymic carcinoid tumors, patients with MEN1 have an increased incidence of pulmonary [0–8%]47,140,190,195,261,368,378 and gastric carcinoid tumors [7%–35%],34,140,187,195,232,326,360 which are reported to be an MEN1-related cause of death in a few series in the literature and in some case reports.39,150,326,378,379,448,465 In the 106 NIH MEN1/ZES patients, neither gastric nor pulmonary carcinoid tumors caused any MEN1 deaths; however, in the 227 pooled literature series, 1 patient died from a gastric carcinoid (0.4% of all deaths) and 1 patient died from a pulmonary carcinoid (0.4% of all deaths). These data suggest that although these tumors can be malignant and also cause a hormone excess syndrome (carcinoid syndrome),40,94,98,190,360,437 they are an uncommon cause of MEN1-related death at present. This conclusion is supported by the results of 15 MEN1 series (see Table 12) and a detailed series on only pulmonary carcinoids in MEN1 patients368 in which pulmonary carcinoids were reported to be a cause of death in a mean of 0.5% ± 0.4% of total deaths (range, 0–5%) and gastric carcinoids in 1.2% ± 0.8% (range, 0–10%), with 11 series reporting no deaths from either gastric or pulmonary carcinoids. This conclusion for bronchial carcinoids was also supported by the GTE study on 758 MEN1 patients, in which the presence of a bronchial carcinoid did not increase the death rate compared to unaffected controls (HR = 1.55; 95% CI, 0.64–3.77; p = 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