Double pituitary adenomas in a large surgical series

Zieliński, Grzegorz; Sajjad, Emir; Maksymowicz, Maria; Pękul, Monika; Koziarski, Andrzej

doi:10.1007/s11102-019-00996-2

Cited by 21 publications

(19 citation statements)

References 27 publications

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“…In our study men are affected signi cantly more often than women. In comparable studies, equivalent gender ratios [11] or even a female predominance have been described [12]. On this base, the male predominance in our study may also be due to random chance.…”

Section: Multiple Pitnetssupporting

confidence: 79%

“…Because of the high number of GH-and ACTH-secreting tumors, multiple PitNETs are often associated with the symptoms of acromegaly or Cushing's disease [11]. In the surgically removed specimens in our collection, symptoms of acromegaly were described in 64.7% and symptoms of Cushing's disease in 11.8% of the cases.…”

Section: Multiple Pitnetsmentioning

confidence: 86%

“…In the literature, GH secreting PitNETs have most frequently been diagnosed in pituitaries with multiple PitNETs [10,11].…”

Section: Multiple Pitnetsmentioning

confidence: 99%

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Multiple Tumorous Lesions of The Pituitary Gland

Schöning

Flitsch

Lüdecke

et al. 2021

Preprint

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Multiple tumorous lesions with an adenoma make up 1.4 % (232 cases) in the large collection of the German Registry of Pituitary Tumors. 2.0% of all PitNETs are associated with a second lesion. Within the selected cases synchronous multiple pituitary neuroendocrine tumors (PitNETs) account for 17,3%, PANCH cases for 14.7%, PitNETs and posterior lobe tumors for 2.2%, PitNETs and metastases for 5.2%, PitNETs and mesenchymal tumors for 2.6%, PitNETs and cysts for 52.2% and PitNETs and primary inflammation for 6.0%. The mean patient age was 53.8 years with a standard deviation of 18.5 years. 55.3% of the patients were female and 44.7% were male. From 1990 to 2018 there was a continuous increase in the number of multiple tumorous lesions.

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Section: Multiple Pitnetssupporting

confidence: 79%

Section: Multiple Pitnetsmentioning

confidence: 86%

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Multiple Tumorous Lesions of The Pituitary Gland

Schöning

Flitsch

Lüdecke

et al. 2021

Preprint

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“…Molecular investigation of double PAs as tumour entities sharing the same general genetic and epigenetic background may possibly disclose similarities and differences in tumorigenesis and/or tumour differentiation. For this purpose the adenomas need to differ in their hormone profile; it does not suffice that the tumours appear as separate masses in MRI imaging [11]. In the present study we report 12 double PAs (DPA) with diverse hormone profile collected in the German Registry of Pituitary Tumours between 2012 and 2020, which were investigated in regard to their immunohistochemical expression pattern, and in 4 cases additionally concerning their chromosomal gains and losses and DNA methylation profile.…”

Section: Introductionmentioning

confidence: 99%

Double adenomas of the pituitary reveal distinct lineage markers, copy number alterations, and epigenetic profiles

et al. 2021

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Purpose Pituitary adenoma (PA) constitutes the third most common intracranial neoplasm. The mostly benign endocrine lesions express no hormone (null cell PA) or the pituitary hormone(s) of the cell lineage of origin. In 0.5–1.5% of surgical specimens and in up to 10% of autopsy cases, two or three seemingly separate PA may coincide. These multiple adenomas may express different hormones, but whether or not expression of lineage-restricted transcription factors and molecular features are distinct within multiple lesions remains unknown. Methods Searching the data bank of the German Pituitary Tumor Registry 12 double pituitary adenomas with diverse lineage were identified among 3654 adenomas and 6 hypophyseal carcinomas diagnosed between 2012 and 2020. The double adenomas were investigated immunohistochemically for expression of hormones and lineage markers. In addition, chromosomal gains and losses as well as global DNA methylation profiles were assessed, whenever sufficient material was available (n = 8 PA). Results In accordance with the literature, combinations of GH/prolactin/TSH–FSH/LH adenoma (4/12), GH/prolactin/TSH–ACTH adenoma (3/12), and ACTH–FSH/LH adenoma (3/12) were observed. Further, two out of 12 cases showed a combination of a GH/prolactin/TSH adenoma with a null-cell adenoma. Different expression pattern of hormones were confirmed by different expression of transcription factors in 11/12 patients. Finally, multiple lesions that were molecularly analysed in 4 patients displayed distinct copy number changes and global methylation pattern. Conclusion Our data confirm and extend the knowledge on multiple PA and suggest that such lesions may origin from distinct cell types.

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“…Specifically, in DPAs, each adenoma develops from a different cell type in the pituitary gland. [5][6][7] Here, we describe the case of a patient who was initially diagnosed with a gonadotroph pituitary adenoma (GPA) and underwent surgery. Subsequently, about 7 years later, he was diagnosed with a somatotroph pituitary adenoma (SPA) that was also surgically resected.…”

mentioning

confidence: 99%

Metachronous Double Pituitary Adenoma with Altered Transcriptional Factor Profile: A Case Report and Literature Review

Taguchi

Kinoshita

Tominaga

et al. 2021

NMC Case Report Journal

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Double pituitary adenomas (DPAs), especially metachronous DPAs, are extremely rare and there has been no report about DPAs with altered transcriptional factors. We describe the case of a 25-year-old man who presented with acromegaly 7 years after surgery for a non-functioning pituitary adenoma (NFPA). Before the initial surgery, endocrine evaluation confirmed NFPA or silent somatotroph pituitary adenoma (SPA) because of normal serum levels of insulin-like growth factor-1 (IGF-1) and insufficient suppression of growth hormone (GH) levels in the oral glucose tolerance test (OGTT). Immunohistochemistry of resected tissue obtained from gross total resection (GTR) with transsphenoidal surgery (TSS) was negative for follicle-stimulating hormone, luteinizing hormone, GH, and Pit-1 but positive for GATA3, which confirmed the gonadotroph pituitary adenoma (GPA) diagnosis. Seven years later, follow-up brain MRI revealed a 13.3 × 5.6 × 4.7 mm tumor within the sellar turcica. The endocrine evaluation confirmed acromegaly because of high serum levels of IGF-1 and insufficient suppression of GH levels upon OGTT. GTR with TSS was again performed, and immunohistochemistry was negative for GATA3 but positive for GH and Pit-1. Surprisingly, he showed altered transcription factor expressions between initial and recurrent surgery. Based on the overall clinical course and hormonal secretion findings, we speculated metachronous development of a DPA, i.e., SPA followed by GPA, wherein a few remaining cells of the SPA might have regrown after the initial surgery. We conducted a literature review of cases that documented altered hormone secretion at recurrence and emphasized the necessity of identifying a small adenoma when there is a discrepancy between pathological findings and hormone secretion tests.

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Double pituitary adenomas in a large surgical series

Cited by 21 publications

References 27 publications

Multiple Tumorous Lesions of The Pituitary Gland

Multiple Tumorous Lesions of The Pituitary Gland

Double adenomas of the pituitary reveal distinct lineage markers, copy number alterations, and epigenetic profiles

Metachronous Double Pituitary Adenoma with Altered Transcriptional Factor Profile: A Case Report and Literature Review

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