Meningiomas May Be a Component Tumor of Multiple Endocrine Neoplasia Type 1

Behnam Asgharian, Yuan Jia Chen, Nicholas J. Patronas, Paolo L. Peghini, James C. Reynolds, Alexander Vortmeyer, Zhengping Zhuang, David J. Venzon, Fathia Gibril, Robert T. Jensen

Research output: Contribution to journalArticle

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Abstract

Purpose: Recently, an increased incidence of some non-endocrine tumors are reported in patients with multiple endocrine neoplasia type 1 (MEN 1). There are rare reports of meningiomas and other central nervous system tumors in these patients, but it is unknown if they are more frequent or if allelic loss of the MEN1 gene is important in their pathogenesis. The aim of this study was to address these two latter questions. Experimental Design: Results from a prospective study of 74 MEN 1 patients with suspected/proven pancreatic endocrine tumors (PETs) were analyzed, as well as molecular studies performed on a resected meningioma. All patients had serial brain imaging studies (computed tomography, magnetic resonance imaging, and octreoscanning since 1994) and yearly studies evaluating MEN 1 involvement with a mean follow-up of 7.2 years. Results were compared with 185 patients with sporadic Zollinger-Ellison syndrome. Results: Six patients (8%) had meningiomas. Meningiomas were single and found late in the MEN 1 course (mean age = 51 years). Magnetic resonance imaging/computed tomography were more sensitive than octreoscanning. Their diagnosis averaged 18 years after the onset of hyperparathyroidism, 10-15 years after pituitary disease or PETs. Meningiomas were 11 times more frequent in patients with PETs with MEN 1 than without MEN 1 (P = 0.017). No clinical, laboratory, or MEN 1 feature distinguished patients with meningiomas. Meningiomas were asymptomatic and 60% showed no growth. A resected meningioma showed loss of heterozygosity at 11q13 and 1p, including at p73 and ARHI/NOEY2 locus, but not at the neurofibromatosis 2 gene locus. Conclusions: These results show meningiomas are not an infrequent occurrence in MEN 1, and loss of the function of the MEN1 gene product plays a role in their pathogenesis in these patients.

Original languageEnglish (US)
Pages (from-to)869-880
Number of pages12
JournalClinical Cancer Research
Volume10
Issue number3
DOIs
StatePublished - Feb 1 2004
Externally publishedYes

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Multiple Endocrine Neoplasia Type 1
Meningioma
Neoplasms
Loss of Heterozygosity
Neurofibromatosis 2 Genes
Tomography
Magnetic Resonance Imaging
Pituitary Diseases
Zollinger-Ellison Syndrome
Central Nervous System Neoplasms
Hyperparathyroidism
Neuroimaging
Genes
Research Design
Prospective Studies

ASJC Scopus subject areas

  • Oncology
  • Cancer Research

Cite this

Asgharian, B., Chen, Y. J., Patronas, N. J., Peghini, P. L., Reynolds, J. C., Vortmeyer, A., ... Jensen, R. T. (2004). Meningiomas May Be a Component Tumor of Multiple Endocrine Neoplasia Type 1. Clinical Cancer Research, 10(3), 869-880. https://doi.org/10.1158/1078-0432.CCR-0938-3

Meningiomas May Be a Component Tumor of Multiple Endocrine Neoplasia Type 1. / Asgharian, Behnam; Chen, Yuan Jia; Patronas, Nicholas J.; Peghini, Paolo L.; Reynolds, James C.; Vortmeyer, Alexander; Zhuang, Zhengping; Venzon, David J.; Gibril, Fathia; Jensen, Robert T.

In: Clinical Cancer Research, Vol. 10, No. 3, 01.02.2004, p. 869-880.

Research output: Contribution to journalArticle

Asgharian, B, Chen, YJ, Patronas, NJ, Peghini, PL, Reynolds, JC, Vortmeyer, A, Zhuang, Z, Venzon, DJ, Gibril, F & Jensen, RT 2004, 'Meningiomas May Be a Component Tumor of Multiple Endocrine Neoplasia Type 1', Clinical Cancer Research, vol. 10, no. 3, pp. 869-880. https://doi.org/10.1158/1078-0432.CCR-0938-3
Asgharian, Behnam ; Chen, Yuan Jia ; Patronas, Nicholas J. ; Peghini, Paolo L. ; Reynolds, James C. ; Vortmeyer, Alexander ; Zhuang, Zhengping ; Venzon, David J. ; Gibril, Fathia ; Jensen, Robert T. / Meningiomas May Be a Component Tumor of Multiple Endocrine Neoplasia Type 1. In: Clinical Cancer Research. 2004 ; Vol. 10, No. 3. pp. 869-880.
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abstract = "Purpose: Recently, an increased incidence of some non-endocrine tumors are reported in patients with multiple endocrine neoplasia type 1 (MEN 1). There are rare reports of meningiomas and other central nervous system tumors in these patients, but it is unknown if they are more frequent or if allelic loss of the MEN1 gene is important in their pathogenesis. The aim of this study was to address these two latter questions. Experimental Design: Results from a prospective study of 74 MEN 1 patients with suspected/proven pancreatic endocrine tumors (PETs) were analyzed, as well as molecular studies performed on a resected meningioma. All patients had serial brain imaging studies (computed tomography, magnetic resonance imaging, and octreoscanning since 1994) and yearly studies evaluating MEN 1 involvement with a mean follow-up of 7.2 years. Results were compared with 185 patients with sporadic Zollinger-Ellison syndrome. Results: Six patients (8{\%}) had meningiomas. Meningiomas were single and found late in the MEN 1 course (mean age = 51 years). Magnetic resonance imaging/computed tomography were more sensitive than octreoscanning. Their diagnosis averaged 18 years after the onset of hyperparathyroidism, 10-15 years after pituitary disease or PETs. Meningiomas were 11 times more frequent in patients with PETs with MEN 1 than without MEN 1 (P = 0.017). No clinical, laboratory, or MEN 1 feature distinguished patients with meningiomas. Meningiomas were asymptomatic and 60{\%} showed no growth. A resected meningioma showed loss of heterozygosity at 11q13 and 1p, including at p73 and ARHI/NOEY2 locus, but not at the neurofibromatosis 2 gene locus. Conclusions: These results show meningiomas are not an infrequent occurrence in MEN 1, and loss of the function of the MEN1 gene product plays a role in their pathogenesis in these patients.",
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AU - Asgharian, Behnam

AU - Chen, Yuan Jia

AU - Patronas, Nicholas J.

AU - Peghini, Paolo L.

AU - Reynolds, James C.

AU - Vortmeyer, Alexander

AU - Zhuang, Zhengping

AU - Venzon, David J.

AU - Gibril, Fathia

AU - Jensen, Robert T.

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N2 - Purpose: Recently, an increased incidence of some non-endocrine tumors are reported in patients with multiple endocrine neoplasia type 1 (MEN 1). There are rare reports of meningiomas and other central nervous system tumors in these patients, but it is unknown if they are more frequent or if allelic loss of the MEN1 gene is important in their pathogenesis. The aim of this study was to address these two latter questions. Experimental Design: Results from a prospective study of 74 MEN 1 patients with suspected/proven pancreatic endocrine tumors (PETs) were analyzed, as well as molecular studies performed on a resected meningioma. All patients had serial brain imaging studies (computed tomography, magnetic resonance imaging, and octreoscanning since 1994) and yearly studies evaluating MEN 1 involvement with a mean follow-up of 7.2 years. Results were compared with 185 patients with sporadic Zollinger-Ellison syndrome. Results: Six patients (8%) had meningiomas. Meningiomas were single and found late in the MEN 1 course (mean age = 51 years). Magnetic resonance imaging/computed tomography were more sensitive than octreoscanning. Their diagnosis averaged 18 years after the onset of hyperparathyroidism, 10-15 years after pituitary disease or PETs. Meningiomas were 11 times more frequent in patients with PETs with MEN 1 than without MEN 1 (P = 0.017). No clinical, laboratory, or MEN 1 feature distinguished patients with meningiomas. Meningiomas were asymptomatic and 60% showed no growth. A resected meningioma showed loss of heterozygosity at 11q13 and 1p, including at p73 and ARHI/NOEY2 locus, but not at the neurofibromatosis 2 gene locus. Conclusions: These results show meningiomas are not an infrequent occurrence in MEN 1, and loss of the function of the MEN1 gene product plays a role in their pathogenesis in these patients.

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