Plurihormonal Pituitary Tumor of Pit-1 and SF-1 Lineages, with Synchronous Collision Corticotroph Tumor: a Possible Stem Cell Phenomenon
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Thyrotropin (TSH)-secreting pituitary tumors are the rarest functioning pituitary tumors. Nonetheless, they are not infrequently plurihormonal, as they may express/secrete hormones made by other pituitary cells derived from the Pit-1 lineage such as growth hormone (GH), prolactin (PRL), and α-subunit (αSU). However, adrenocorticotropin (ACTH) or gonadotropin secretion by such a tumor is exceptional. Although double pituitary tumors are rare, they often combine ACTH and GH secretion. A 41-year-old presented almost 2 years after delivering her 10th child; she had thyrotoxicosis (goiter and palpitations) masquerading as autoimmune postpartum thyroiditis. She was still breastfeeding and amenorrheic. She proved to have TSH, GH, PRL, and ACTH hypersecretion. Imaging revealed an invasive pituitary macrotumor. She had stigmata neither of Cushing’s disease nor of acromegaly. Prior to surgery, hormonal control was achieved for close to 1 year by combined octreotide and cabergoline treatment with significant shrinking of the tumor. Following surgery, pathology revealed a collision tumor; the dominant lesion was positive for GH, βTSH, βFSH, and αSU and expressed both Pit-1 and SF-1.The smaller lesion was a corticotroph tumor. We report an unusual plurihormonal tumor co-expressing Pit-1 and SF-1 along with hormones made by cells of both lineages. Its simultaneous occurrence adjacent to a corticotroph tumor raises questions regarding the pathogenesis of these tumors. We propose the possibility of a stem cell tumor with multiple lineage differentiation. We hypothesize that pregnancy might have played a permissive role in tumorigenesis.
KeywordsPituitary tumor SF-1 Pit-1 Stem cell Collision Double tumor Plurihormonal
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The authors declare that they have no conflict of interest.
The patient gave her consent to have the case reported; there are no details in the manuscript that could allow identification.
- 2.Yamada S, Fukuhara N, Horiguchi K, Yamaguchi-Okada M, Nishioka H, Takeshita A, Takeuchi Y, Ito J, Inoshita N. Clinicopathological characteristics and therapeutic outcomes in thyrotropin-secreting pituitary adenomas: a single-center study of 90 cases. J Neurosurg. 2014:121:1462-1473.CrossRefGoogle Scholar
- 7.Asa SL: Tumors of the Pituitary Gland. Armed Forces Institute of Pathology Atlas of Tumor Pathology, Series 4, Fascicle 15, ARP Press, Washington DC, 2011.Google Scholar
- 8.Astaf'eva LI, Kadashev BA, Shishkina LV, Kalinin PL, Fomichev DV, Kutin MA, Aref'eva IA, Dzeranova LK, Sidneva YG, Klochkova IS, et al. Clinical and morphological characteristics, diagnostic criteria, and outcomes of surgical treatment of TSH-secreting pituitary adenomas. Zhl Vopr Neirokhiri ImiN. N. Burdenko. 2016;80:24-35.CrossRefGoogle Scholar
- 10.Mete O, Kefeli M, Çalişkan S, Asa SL: GATA-3 Immunoreactivity Expands the Transcription Factor Profile of Pituitary Neuroendocrine Tumors. Mod Pathol 2018, in press.Google Scholar
- 12.Karavitaki N, Thanabalasingham G, Shore HC, Trifanescu R, Ansorge O, Meston N, Turner HE, Wass JA. Do the limits of serum prolactin in disconnection hyperprolactinaemia need re-definition? A study of 226 patients with histologically verified non-functioning pituitary macroadenoma. Clin Endocrinol (Oxf) 2006;65:524-529.CrossRefGoogle Scholar
- 18.Ogando-Rivas E, Alalade AF, Boatey J, Schwartz TH. Double pituitary adenomas are most commonly associated with GH-and ACTH-secreting tumors: systematic review of the literature. Pituitary 2017;20:702-708.Google Scholar
- 21.Matsuno A, Sasaki T, Mochizuki T, Fujimaki T, Sanno N, Osamura Y, Teramoto A, Kirino T. A case of pituitary somatotroph adenoma with concomitant secretion of growth hormone, prolactin, and adrenocorticotropic hormone-an adenoma derived from primordial stem cell, studied by immunohistochemistry, in situ hybridization, and cell culture. Acta Neurochir. (Wien) 1996;138:1002-1007.CrossRefGoogle Scholar
- 22.Würth R, Barbieri F, Pattarozzi A, Gaudenzi G, Gatto F, Fiaschi P, Ravetti JL, Zona G, Daga A, Persani L , et al. Phenotypical and Pharmacological Characterization of Stem-Like Cells in Human Pituitary Adenomas. Mol Neurobiol.. 2017;54:4879-4895.Google Scholar