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Pituitary tumors and hyperplasia in multiple endocrine neoplasia type 1 syndrome (MEN1): a case-control study in a series of 77 patients versus 2509 non-MEN1 patients.
Am J Surg Pathol. 2008 Apr; 32(4):534-43.AJ

Abstract

Patients affected by the multiple endocrine neoplasia type I syndrome (MEN1) display a high incidence of pituitary adenomas, though it is still unknown whether these pituitary tumors have specific pathologic features that would distinguish them from sporadic pituitary adenomas. Pituitary tissue specimens of 77 MEN1 patients from the GTE (Groupe d'étude des Tumeurs Endocrines) register were compared with unselected 2509 non-MEN1 sporadic pituitary tumors and also to a control subgroup of 296 cases, where 1 MEN1 tumor was matched with 4 sporadic tumors of the same hormonal immunoprofile. Sex, age, size, and invasiveness of tumors, and menin gene mutations were documented. Histologic analysis took into account 33 items, including immunocytochemical data, the proliferative marker Ki-67, and an examination of the juxtatumoral pituitary. MEN1 tumors were significantly larger and more often invasive by histology. MEN1 patients with large pituitary tumors (grade IV) were younger than non-MEN1 patients. MEN1 tumors had no other characteristic histologic features and no predominance of any one hormone producing subtype. However, plurihormonal adenomas versus monohormonal and nonimmunoreactive adenomas were more frequent in MEN1 tumors (39%) than in the control non-MEN1 group (P = 0.001). Especially, the growth hormone and prolactin plurihormonality with unusual association with follicle-stimulating hormone, luteinizing hormone, or adrenocorticotropic hormone was more frequent in MEN1 tumors. In addition, multiple adenomas were significantly more frequent (4% vs. 0.1%; P < 0.0001), especially prolactin-adrenocorticotropic hormone. Somatotroph hyperplasia, with or without a microadenoma was found in only 3 MEN1 patients, with growth hormone-releasing hormone hypersecretion by a pancreatic tumor in 2 of them. All types of mutation were observed, including frameshifts, nonsenses, missenses, and 1 case of germline MEN1 encompassing large deletion, strongly suggesting the absence of any phenotype-genotype correlation.

Authors+Show Affiliations

INSERM, U842, Lyon, F-69372, France. jacqueline.trouillas@recherche.univ-lyon1.frNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Journal Article
Research Support, Non-U.S. Gov't

Language

eng

PubMed ID

18300794

Citation

Trouillas, Jacqueline, et al. "Pituitary Tumors and Hyperplasia in Multiple Endocrine Neoplasia Type 1 Syndrome (MEN1): a Case-control Study in a Series of 77 Patients Versus 2509 non-MEN1 Patients." The American Journal of Surgical Pathology, vol. 32, no. 4, 2008, pp. 534-43.
Trouillas J, Labat-Moleur F, Sturm N, et al. Pituitary tumors and hyperplasia in multiple endocrine neoplasia type 1 syndrome (MEN1): a case-control study in a series of 77 patients versus 2509 non-MEN1 patients. Am J Surg Pathol. 2008;32(4):534-43.
Trouillas, J., Labat-Moleur, F., Sturm, N., Kujas, M., Heymann, M. F., Figarella-Branger, D., Patey, M., Mazucca, M., Decullier, E., Vergès, B., Chabre, O., & Calender, A. (2008). Pituitary tumors and hyperplasia in multiple endocrine neoplasia type 1 syndrome (MEN1): a case-control study in a series of 77 patients versus 2509 non-MEN1 patients. The American Journal of Surgical Pathology, 32(4), 534-43. https://doi.org/10.1097/PAS.0b013e31815ade45
Trouillas J, et al. Pituitary Tumors and Hyperplasia in Multiple Endocrine Neoplasia Type 1 Syndrome (MEN1): a Case-control Study in a Series of 77 Patients Versus 2509 non-MEN1 Patients. Am J Surg Pathol. 2008;32(4):534-43. PubMed PMID: 18300794.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Pituitary tumors and hyperplasia in multiple endocrine neoplasia type 1 syndrome (MEN1): a case-control study in a series of 77 patients versus 2509 non-MEN1 patients. AU - Trouillas,Jacqueline, AU - Labat-Moleur,Françoise, AU - Sturm,Nathalie, AU - Kujas,Michèle, AU - Heymann,Marie-Françoise, AU - Figarella-Branger,Dominique, AU - Patey,Martine, AU - Mazucca,Marc, AU - Decullier,Evelyne, AU - Vergès,Bruno, AU - Chabre,Olivier, AU - Calender,Alain, AU - ,, PY - 2008/2/28/pubmed PY - 2008/5/30/medline PY - 2008/2/28/entrez SP - 534 EP - 43 JF - The American journal of surgical pathology JO - Am. J. Surg. Pathol. VL - 32 IS - 4 N2 - Patients affected by the multiple endocrine neoplasia type I syndrome (MEN1) display a high incidence of pituitary adenomas, though it is still unknown whether these pituitary tumors have specific pathologic features that would distinguish them from sporadic pituitary adenomas. Pituitary tissue specimens of 77 MEN1 patients from the GTE (Groupe d'étude des Tumeurs Endocrines) register were compared with unselected 2509 non-MEN1 sporadic pituitary tumors and also to a control subgroup of 296 cases, where 1 MEN1 tumor was matched with 4 sporadic tumors of the same hormonal immunoprofile. Sex, age, size, and invasiveness of tumors, and menin gene mutations were documented. Histologic analysis took into account 33 items, including immunocytochemical data, the proliferative marker Ki-67, and an examination of the juxtatumoral pituitary. MEN1 tumors were significantly larger and more often invasive by histology. MEN1 patients with large pituitary tumors (grade IV) were younger than non-MEN1 patients. MEN1 tumors had no other characteristic histologic features and no predominance of any one hormone producing subtype. However, plurihormonal adenomas versus monohormonal and nonimmunoreactive adenomas were more frequent in MEN1 tumors (39%) than in the control non-MEN1 group (P = 0.001). Especially, the growth hormone and prolactin plurihormonality with unusual association with follicle-stimulating hormone, luteinizing hormone, or adrenocorticotropic hormone was more frequent in MEN1 tumors. In addition, multiple adenomas were significantly more frequent (4% vs. 0.1%; P < 0.0001), especially prolactin-adrenocorticotropic hormone. Somatotroph hyperplasia, with or without a microadenoma was found in only 3 MEN1 patients, with growth hormone-releasing hormone hypersecretion by a pancreatic tumor in 2 of them. All types of mutation were observed, including frameshifts, nonsenses, missenses, and 1 case of germline MEN1 encompassing large deletion, strongly suggesting the absence of any phenotype-genotype correlation. SN - 0147-5185 UR - https://www.unboundmedicine.com/medline/citation/18300794/Pituitary_tumors_and_hyperplasia_in_multiple_endocrine_neoplasia_type_1_syndrome__MEN1_:_a_case_control_study_in_a_series_of_77_patients_versus_2509_non_MEN1_patients_ L2 - http://dx.doi.org/10.1097/PAS.0b013e31815ade45 DB - PRIME DP - Unbound Medicine ER -