Multihormonal pituitary adenoma concomitant with Pit-1 and Tpit lineage cells causing acromegaly associated with subclinical Cushing’s disease: a case report

Takiguchi, Tetsuya; Koide, Hisashi; Nagano, Hidekazu; Nakayama, Akihiro; Fujimoto, Masanori; Tamura, Ai; Komai, Eri; Shiga, Akina; Kobayashi, Takashi; Higuchi, Shinji; Sakuma, Ichiro; Hashimoto, Naoko; Suzuki, Shohachi; Miyabayashi, Yui; Ishiwatari, Norio; Horiguchi, Kentaro; Nakatani, Yukio; Yokote, Koutaro; Tanaka, Tomoaki

doi:10.1186/s12902-017-0203-5

Cited by 9 publications

(5 citation statements)

References 23 publications

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“…The rare entity of multiple synchronous pituitary adenomas has been defined as morphologically and immunohistologically distinct adenomas in the same patient. To date, there have been at least 18 case reports, including this one, of patients with multiple synchronous pituitary adenomas, including collision tumors in which 2 histologically distinct tumors occur at the same anatomic site (8)(9)(10)(11). With the increasing availability of higher resolution MRI that increases the sensitivity of detection for pituitary microadenomas, the detection of such multiple synchronous pituitary adenomas may increase over time (12).…”

Section: Discussionmentioning

confidence: 98%

Two Synchronous Pituitary Adenomas Causing Cushing Disease and Acromegaly

Schorr

Zhang

Zhao

et al. 2019

AACE Clinical Case Reports

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Objective:To report the first case of 2 synchronous pituitary adenomas, 1 corticotroph and 1 somatotroph, with distinct molecular lineages confirmed by differential hormone and S-100 protein expression.Methods: A case report followed by a literature review are presented.Results: A 68-year-old woman presented for evaluation of resistant hypertension. Biochemical testing demonstrated adrenocorticotropic hormone (ACTH)-dependent hypercortisolemia and growth hormone (GH) excess. Pituitary magnetic resonance imaging (MRI) revealed a 2 cm left sellar lesion consistent with a pituitary macroadenoma. The patient therefore underwent transsphenoidal surgery for a presumed cosecreting ACTH and GH macroadenoma. Tumor immunohistochemical staining (IHC) was positive for ACTH, but negative for GH. Postoperative biochemical testing confirmed remission from Cushing disease, but the insulin-like growth factor 1 (IGF-1) level remained elevated. Postoperative MRI demonstrated a small right sellar lesion that, in retrospect, had been present on the preoperative MRI. Resection of the right lesion confirmed a GH-secreting adenoma with negative ACTH staining. After the second surgery, the IGF-1 level normalized and blood pressure improved. Further pathologic examination of both surgical specimens demonstrated differential expression of S-100 protein, a folliculostellate cell marker. Reverse transcription polymerase chain reaction of messenger ribonucleic acid from the left sellar lesion was positive for ACTH and negative for GH, confirming the IHC results. Germline mutations in genes known to be associated with pituitary adenoma syndromes (MEN1, CDC73, CDKN1A, CDKN1B, CDKN2B, CDKN2C, and AIP) were not detected.Conclusion: Although the pathogenesis of synchronous pituitary adenomas has not been fully elucidated, this case report suggests that they can have distinct molecular lineages. (AACE Clinical Case Rep. 2019;5:e276-e281) Abbreviations: ACTH = adrenocorticotropic hormone; FSC = folliculostellate cell; FSH = follicle-stimulating hormone; GH = growth hormone; IGF-1 = insulin-like growth factor 1; IHC = immunohistochemical staining; LH = luteinizing hormone; MRI = magnetic resonance imaging; mRNA = messenger ribonucleic acid; RT-PCR = reverse transcription polymerase chain reaction; TSH = thyroid-stimulating hormone 1.0 (1) Borderline 1.1 (1) Borderline Abbreviations: GH = growth hormone; IGF-1 = insulin-like growth factor 1; n = number of samples; OGTT = oral glucose tolerance test.e278 Two Synchronous Pituitary Adenomas, AACE Clinical Case Rep. 2019;5(No. 5)

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Section: Discussionmentioning

confidence: 98%

Two Synchronous Pituitary Adenomas Causing Cushing Disease and Acromegaly

Schorr

Zhang

Zhao

et al. 2019

AACE Clinical Case Reports

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“…This unique tumor entity may be the result of PitNET clone cells undergoing various multidirectional differentiations. 28,32,33 Previously, this mixedlineage PitNET was described using various terms, such as multihormonal adenomas, 9,28,34,35 double (collision) adenomas, 11 synchronous multiple adenomas, 10 or adenomas with lineage infidelity. 36 Prior definitions primarily relied on immunohistochemical analysis of pituitary hormones or the relative positioning of tumor cell clusters observed under a microscope.…”

Section: Discussionmentioning

confidence: 99%

Clinical and Pathological Features of Pit1/SF1 Multilineage Pituitary Neuroendocrine Tumor

Wang,

Tang,

Bie

et al. 2024

Neurosurgery

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BACKGROUND AND OBJECTIVES: Lineage-based classification has critical clinical implications in pituitary neuroendocrine tumor (PitNET). As the most prevalent subtype of multilineage PitNET, PitNET originating from both pituitary-specific positive transcription factor 1 (Pit1) and steroidogenic factor-1 (SF1) lineages (Pit1/SF1-adenoma) is expected to exhibit rich and varied clinical behaviors. A comprehensive understanding of the clinical and pathological characteristics of Pit1/SF1-adenoma will provide mechanistic insight and influence the prognosis and treatment of PitNET. METHODS: A retrospective study was conducted by reviewing 57 cases of Pit1/SF1-adenoma between 2018 and 2022. We also included 88 cases of PitNET arising from Pit1 cell lineage (Pit1-adenoma) and 70 cases of PitNET arising from SF1 cell lineage (SF1-adenoma) as controls. Comprehensive data, including demographic, symptom, endocrinal, radiological, surgical, pathological, and prognostic information, were systematically collected. All specimens were immunostained for pituitary transcription factors (PTFs) and pituitary hormones. RESULTS: The detection rate was 8.0% for Pit1/SF1-adenoma within PitNET surgical specimens. Pit1/SF1-adenoma displayed a male predominance, with the mean diagnosis age falling between Pit1-adenoma and SF1-adenoma. The endocrine activity of Pit1/SF1-adenoma was lower than Pit1-adenoma but higher than SF1-adenoma. Pit1/SF1-adenoma had a higher incidence of cavernous sinus invasion (56.1%) than both Pit1-adenoma (38.6%, P = .039) and SF1-adenoma (27.1%, P = .001). Furthermore, Pit1/SF1-adenoma showed more postoperative complications than Pit1-adenoma (29.8% vs 8.0%, P = .001). Nonfunctional Pit1/SF1-adenoma had a higher radiological tumor recurrence rate than nonfunctional SF1-adenoma (34.8% vs 10.9%, P = .021). Notably, the immunostaining pattern was diverse in Pit1/SF1-adenoma, with various combinations of staining intensity for PTFs and 15 combinations for 6 pituitary hormones. Intriguingly, various PTFs combinations had no different impact on the outcome of Pit1/SF1-adenoma. CONCLUSION: Pit1/SF1-adenoma represents a unique pathological subtype of PitNET, characterized by distinctive clinical behaviors. Identifying Pit1/SF1-adenoma can facilitate more precise management of PitNET by the practical use of Pit1/SF1 immunostaining.

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“…The most frequent associations are with GH and PRL or LH and FSH [11]. GH-producing adenomas with concomitant ACTH production are extremely rare, although they have been reported previously in a few cases [12][13][14][15][16][17][18][19][20]. Clinically, the majority of PHAs are silent, and diagnosis almost always relies on immunohistochemical analysis of the tumor tissue to demonstrate positivity for unrelated hormones [8,9].…”

Section: Discussionmentioning

confidence: 99%

“…Clinically, the majority of PHAs are silent, and diagnosis almost always relies on immunohistochemical analysis of the tumor tissue to demonstrate positivity for unrelated hormones [8,9]. Roca et al [20] recently reviewed the literature on PHAs and reported 21 cases with ACTH-GH plurihormonal pituitary adenoma. Of 3 Postoperative pituitary MRI revealing residual isointense enhancement of the tumor in the sella and along the stalk measuring 9.9 × 9.9 mm in the anteroposterior (AP) and transverse dimensions and 1.3 cm in the craniocaudal (CC) dimension.…”

Section: Discussionmentioning

confidence: 99%

Plurihormonal pituitary macroadenoma: a case report

2021

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Background Plurihormonal pituitary adenomas are a unique type of pituitary adenomas that secrete two or more pituitary hormones normally associated with separate cell types that have different immunocytochemical and ultrastructural features. Although they represent 10–15% of all pituitary tumors, only a small fraction of plurihormonal pituitary adenomas clinically secrete multiple hormones. The most common hormone combinations secreted by plurihormonal pituitary adenomas are growth hormone, prolactin, and one or more glycoprotein hormones. The most common hormonal symptom is acromegaly (50%). The aim of this case report is to bring awareness about this rare type of pituitary adenomas and to describe the unique presentation of our patient, even though plurihormonal pituitary adenomas are known mostly as a clinically silent tumors. Case presentation Herein, we describe an unusual case of plurihormonal pituitary adenoma with triple-positive staining for adrenocorticotropic hormone, growth hormone, and prolactin. The patient is a 65-year-old Egyptian woman who presented with mass effect symptoms of the pituitary tumor, which primarily manifested as severe headache and visual field defects. She also presented with some cushingoid features, and further analysis confirmed Cushing’s disease; slightly high prolactin and normal growth hormone levels were observed. She underwent transsphenoidal surgery and has been in remission thus far. Only a few cases have been reported in the literature, but none has exhibited silent acromegaly or mass effect symptoms as the initial presentation. Conclusion This case highlights an unusual plurihormonal pituitary adenoma case with a rare combination of secreted hormones; mass effect symptoms were dominant, as were uncommon visual field defects. Our case further proves that immunohistochemical analyses of all pituitary hormones are needed to ensure correct diagnosis and to alert clinicians to the need for more rigorous follow-up due to the higher morbidity of these patients. Our case report approval number Federal Wide Assurance NIH, USA is FWA00018774 IRB registration number with OHRP/NIH is IRB00010471.

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Multihormonal pituitary adenoma concomitant with Pit-1 and Tpit lineage cells causing acromegaly associated with subclinical Cushing’s disease: a case report

Cited by 9 publications

References 23 publications

Two Synchronous Pituitary Adenomas Causing Cushing Disease and Acromegaly

Two Synchronous Pituitary Adenomas Causing Cushing Disease and Acromegaly

Clinical and Pathological Features of Pit1/SF1 Multilineage Pituitary Neuroendocrine Tumor

Plurihormonal pituitary macroadenoma: a case report

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