Oral calcium tolerance test in the early diagnosis of primary hyperparathyroidism and multiple endocrine neoplasia type 1 in patients with the Zollinger-Ellison syndrome. Groupe de Recherche et d'Etude du Syndrome de Zollinger-Ellison.

Cadiot, Guillaume; Houillier, Pascal; Allouch, A.; Paillard, Michel; Mignon, M

doi:10.1136/gut.39.2.273

Cited by 27 publications

(27 citation statements)

References 11 publications

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“…This latter result might be explained by the observation that MEN-1-related PHPT can be normocalcemic [18] and show milder biochemical presentation when compared with sporadic PHPT [19]. Given that PHPT is the most frequent and an earlier-onset disease in MEN1 (up to 100% of the cases by the age of 50), the coexistence of PDETs should be looked for in all cases of patients with PHPT, even if asymptomatic, especially in young age.…”

Section: Discussionmentioning

confidence: 98%

Presentation and Outcome of Pancreaticoduodenal Endocrine Tumors in Multiple Endocrine Neoplasia Type 1 Syndrome

Davì¹,

Boninsegna²,

Carbonare³

et al. 2011

Neuroendocrinology

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Aim: To assess presentation and outcome of pancreaticoduodenal endocrine tumors (PDETs) in a single center series of multiple endocrine neoplasia type 1 (MEN1) patients. Methods: Retrospective analysis of prospectively collected data of MEN1 patients observed at the University of Verona. Results: Thirty-one MEN1 patients had PDETs, including 16 nonfunctioning (NF), 6 insulinomas and 9 Zollinger-Ellison syndrome (ZES). In 16 of these patients (52%), PDET was the manifestation which led to the diagnosis of MEN1; among this group, 15 patients (94%) previously had unidentified primary hyperparathyroidism (PHPT), which was asymptomatic in 9 cases (60%). Of the 31 patients, 19 (61%) underwent curativesurgery and 13 (68%, 7 NF-PDETs, 4 insulinomas and 2 ZES) were disease-free after a median follow-up of 3 years (range: 0.5–15). One patient had debulking surgery with stable disease after 2 years of follow-up. Eight patients with NF-PDETs ≤20 mm and 2 with ZES, treated with a conservative approach, showed stable disease. One patient with insulinoma was lost to follow-up. Conclusions: PDET may be the manifestation that leads to MEN1 diagnosis since the almost constant presence of PHPT is very often unrecognized or considered sporadic. Conversely, the presence of PDETs should be looked for in all patients presenting PHPT, even if asymptomatic, particularly before age 50. Surgery may be curative in the majority of insulinomas and can prolong disease-free survival in NF-PDET, but is not proven to be effective in ZES. A conservative approach can be safely reserved for patients with NF-PDETs ≤20 mm.

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Section: Discussionmentioning

confidence: 98%

Presentation and Outcome of Pancreaticoduodenal Endocrine Tumors in Multiple Endocrine Neoplasia Type 1 Syndrome

Davì¹,

Boninsegna²,

Carbonare³

et al. 2011

Neuroendocrinology

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“…both high basal fasting calcium and iPTH levels, were lacking in 5 of 8 patients with primary HPT and ZES. Since primary HPT is most often the only associated endocrinopathy that would suggest the diagnosis of MEN1, an oral calcium tolerance test should be recommended systematically when typical biological features of primary HPT are lacking in any patient with ZES [22]. …”

Section: Factors Affecting the Natural History Of Neeptsmentioning

confidence: 99%

Natural History of Neuroendocrine Enteropancreatic Tumors

Mignon

2000

Digestion

105

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Neuroendocrine enteropancreatic tumors (NEEPTs) include tumors developing from the pancreatic islets and the duodenal loop, and those developing from gut serotonin-secreting cells (mid-gut carcinoids). When functioning, they are rare tumors with frequently inconsistant clinical and biological characteristics. Provocative dynamic tests are very useful. Their natural history, with the exception of insulinomas and mid-gut carcinoids, is still incompletely known due to their rarity, relatively recent description, slow evolution and the absence of definitive reliable markers for prognosis. The following factors affect the natural history of NEEPTs. (1) Inclusion or not within the spectrum of multiple endocrine neoplasia type-1 (MEN1), such an integration often dramatically modifies the management strategy. In patients with MEN1, NEEPTs are often multiple, diffusely spread in the duodenopancreatic area and of small size. Definite cure by surgery is then very scarce. Mid-gut carcinoid tumors are the most protean NEEPTs. In contrast to appendiceal carcinoids, usually with a benign course, mid-gut carcinoids have a more malignant course and are very common indeed: local transmural invasion, early metastasis to lymph nodes and/or liver, and symptoms from hormone oversecretion. Carcinoid syndrome is usually well controlled by long-acting somatostatin analogs. (2) Disease spreading, i.e. liver and bone metastases, are the major causes of death in patients with NEEPTs. Liver metastases (LMs) are synchronous in about 75% of the patients with LMs but may develop during follow-up (about 25% of the patients with LMs were without LMs at the initial evaluation). Development of bone metastases has a very poor prognosis accompanying LM progression.

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“…Due to the existence of slightly different clinical presentations, it is challenging to perform a correct differential diagnosis of PHPT especially in the case of normal calcium levels. In the past, dynamic studies with oral calcium load have been proposed as a completion of diagnosis of PHPT in patients with symptoms who have minimal, intermittent, or no elevation of the levels of total calcium and/or intact PTH (10,11,12,13,14). However, these studies gave contrasting results and, at the present time, no dynamic/stimulative test has been specifically performed in PHPT-N.…”

Section: Introductionmentioning

confidence: 69%

“…Previous works investigating oral calcium load in PHPT addressed the problem of differentiation between adenoma and parathyroid hyperplasia, showing either greater (10) or smaller (11,12,13) decline of serum PTH levels in PHPT patients compared with healthy controls (for a brief summary of these works, see Table 2). After the definition of PHPT-N as a clinical entity, characterized by consistently normal calcium concentrations with persistently abnormal PTH levels (5), (22) only Maruani et al (12) demonstrated in PHPT-N patients a minimal decrease in serum PTH following an oral calcium load (25% change) despite the occurrence of hypercalcemia.…”

Section: Oral Calcium and Pthmentioning

confidence: 99%

Different PTH response to oral peptone load and oral calcium load in patients with normocalcemic primary hyperparathyroidism, primary hyperparathyroidism, and healthy subjects

Invernizzi

Carda

Righini

et al. 2012

European Journal of Endocrinology

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Background: Normocalcemic primary hyperparathyroidism (PHPT-N) is a condition that may have similar long-term implications to primary hyperparathyroidism (PHPT); however, differential diagnosis and treatment for parathyroid disorders are not clearly defined. We investigated the effect of an oral peptone and an oral calcium load on calcium-regulating hormones in PHPT-N compared with PHPT and healthy controls to provide a new potential diagnostic tool. Design: Case-control study. Methods: We evaluated serum gastrin, PTH, ionized calcium, and phosphate responses to oral calcium (1 g) and peptone (10 g) load in 22 PHPT and 20 PHPT-N patients matched for PTH serum values. Moreover, 30 healthy subjects were enrolled as controls. In 12 patients for each group, we also performed the oral peptone test adding aluminum hydroxide (AH) to suppress phosphate absorption. Results: In PHPT patients, PTH increased significantly 30 min after the oral peptone load, while no significant increase was found in PHPT-N and controls. After oral calcium load, PTH remained stable in PHPT while it decreased dramatically in PHPT-N patients, and ionized calcium increased significantly in each of the three groups. Peptones plus AH induced a blunted PTH increase in the three groups. Conclusions: Considering the marked difference in PTH response elicited by peptones in PHPT compared with PHPT-N, we suggest that the oral peptone test could be added to the diagnostic evaluation of PHPT patients. In case of absent response to peptones, patients should have their serum calcium levels assessed twice a year in accordance with recent guidelines.

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Oral calcium tolerance test in the early diagnosis of primary hyperparathyroidism and multiple endocrine neoplasia type 1 in patients with the Zollinger-Ellison syndrome. Groupe de Recherche et d'Etude du Syndrome de Zollinger-Ellison.

Cited by 27 publications

References 11 publications

Presentation and Outcome of Pancreaticoduodenal Endocrine Tumors in Multiple Endocrine Neoplasia Type 1 Syndrome

Presentation and Outcome of Pancreaticoduodenal Endocrine Tumors in Multiple Endocrine Neoplasia Type 1 Syndrome

Natural History of Neuroendocrine Enteropancreatic Tumors

Different PTH response to oral peptone load and oral calcium load in patients with normocalcemic primary hyperparathyroidism, primary hyperparathyroidism, and healthy subjects

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