A somatotropin-producing pituitary adenoma with an isolated adrenocorticotropin-producing pituitary adenoma in a female patient with acromegaly, subclinical Cushing’s disease and a left adrenal tumor

Kobayashi, Yuki; Takei, Masahiro; Ohkubo, Yohsuke; Kakizawa, Yukinari; Matoba, Hisanori; Kumagai, Masanobu; Takeda, Teiji; Komatsu, Mitsuhisa

doi:10.1507/endocrj.ej14-0093

Cited by 8 publications

(8 citation statements)

References 24 publications

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“…Prior patients have been mostly female (71%) with a mean age of 46 years (range 22 to 67 years). Hormone-producing tumors were more commonly seen, with somatotroph and corticotroph adenomas being most prevalent (13)(14)(15)(16). Similar to prior case reports, this patient was a female with a somatotroph and corticotroph adenoma.…”

Section: Discussionsupporting

confidence: 82%

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: Discussionsupporting

confidence: 82%

Two Synchronous Pituitary Adenomas Causing Cushing Disease and Acromegaly

Schorr

Zhang

Zhao

et al. 2019

AACE Clinical Case Reports

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“…Preoperative MRI is more important than CT in the diagnosis of multiple adenomas. As indicated in the present case and previous literature reports, contrast-enhanced MRI is able to confirm separated double or multiple pituitary adenomas (1,3,9,13,17,28). I nt raoperative evaluation, including M R I a nd ultrasonography, as well as surgical exploration, may assist the diagnosis of double or multiple pituitary adenomas (11,14,18,20,25,30,31).…”

Section: Postoperative Values ---------------------------------------supporting

confidence: 74%

“…Between 1978 and 2014, only 115 cases of double or multiple pituitary adenomas, including clearly separated and contiguous adenomas, were reported in the literature (1)(2)(3)(4)(5)(6)(7). Furthermore, the diagnostic criteria for double or multiple pituitary adenomas vary between pathologists and clinical doctors.…”

Section: Introductionmentioning

confidence: 99%

Isolated double adrenocorticotropic hormone-secreting pituitary adenomas: A case report and review of the literature

Wang

Zhou

et al. 2016

Oncology Letters

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Abstract. Only a few cases of double or multiple pituitary adenomas have previously been reported in the literature; however, isolated double adrenocorticotropic hormone (ACTH)-secreting pituitary adenomas are even more rare. The present study reports a rare case of a 50-year-old female patient who presented with typical clinical features of Cushing's disease and was diagnosed with isolated double ACTH-secreting pituitary adenomas. Endocrinological examination revealed an ACTH-producing pituitary adenoma, and preoperative magnetic resonance imaging (MRI) demonstrated a microadenoma with a lower intensity on the right side of the pituitary gland. The patient underwent endoscopic endonasal transsphenoidal surgery, which revealed another pituitary tumor in the left side of the pituitary gland. The two, clearly separated, pituitary adenomas identified in the same gland were completely resected. Immunohistochemistry and pathology revealed that the clearly separated double pituitary adenomas were positive for ACTH, thyroid-stimulating, growth and prolactin hormones. Postoperatively, the levels of ACTH and cortisol hormone decreased rapidly. The case reported in the present study is considerably rare, due to the presence of a second pituitary adenoma in the same gland, which was not detected by preoperative MRI scan, but was noticed during surgery. Intraoperative evaluation may be important in the identification of double or multiple pituitary adenomas.

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Section: Dear Sir;mentioning

confidence: 99%

“…The authors reported a rare case of a 67-year-old Japanese woman with double independent pituitary adenomas, and simultaneous evidence of a left adrenal incidentaloma, causing appearance of signs and symptoms of acromegaly, but without Cushingoid features, and discussed literature regarding double pituitary adenomas [1].In a manuscript published in 1999 [2], we described the first case diagnosed in vivo up to that time of simultaneous occurrence of triple non-functioning pituitary adenomas in a 37-year-old woman who was referred to our outpatient clinic reporting oligomenorrhea, weight gain and persistent frontal and retroocular headache for several years. On admission to our Unit, clinical examination revealed moderate hirsutism and "moon face" while no hypothalamic pituitary dysfunction was demonstrated by basal and dynamic endocrine investigation.…”

mentioning

confidence: 99%

Comment on “A somatotropin-producing pituitary adenoma with an isolated adrenocorticotropin-producing pituitary adenoma in a female patient with acromegaly, subclinical Cushing’s disease and a left adrenal tumor” [Letter to the Editor]

Curtò

Trimarchi

2014

Endocr J

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I read with interest the case report by Kobayashi et al. entitled "A somatotropin-producing pituitary adenoma with an isolated adrenocorticotropin-producing pituitary adenoma in a female patient with acromegaly, subclinical Cushing's disease and a left adrenal tumor" published in Endocrine Journal. The authors reported a rare case of a 67-year-old Japanese woman with double independent pituitary adenomas, and simultaneous evidence of a left adrenal incidentaloma, causing appearance of signs and symptoms of acromegaly, but without Cushingoid features, and discussed literature regarding double pituitary adenomas [1].In a manuscript published in 1999 [2], we described the first case diagnosed in vivo up to that time of simultaneous occurrence of triple non-functioning pituitary adenomas in a 37-year-old woman who was referred to our outpatient clinic reporting oligomenorrhea, weight gain and persistent frontal and retroocular headache for several years. On admission to our Unit, clinical examination revealed moderate hirsutism and "moon face" while no hypothalamic pituitary dysfunction was demonstrated by basal and dynamic endocrine investigation. Preoperative MRI revealed three distinct hypointense areas measuring less than 5 mm in diameter in the left, medium and right side of the pituitary gland, respectively.In the case report by Kobayashi et al., basal and dynamic endocrine evaluation confirmed the diagnosis of acromegaly with a concomitant condition of subclinical Cushing's disease. Preoperative MRI showed two separated and well demarcated tumors with a low intensity on both T2-weighted and T1-weighted images. The left adenoma measured 5×7×6 mm and the right one 6×4×8 mm in diameter, and neither invaded the surrounding structures. In addition, a well demarcated left adrenal nodule (23×16 mm in diameter), not invading the surrounding tissues, was detected by abdominal CT and MRI. The patient underwent transsphenoidal neurosurgery and presence of two separated, totally resected, masses was confirmed. Immunostaining was ACTH-positive and GH-negative in the left adenoma and, on the contrary, GH-positive and ACTH-negative in the right one.Also in our case, not cited by Kobayashi, the preoperative MRI identification of three well defined adenomas was supported by transsphenoidal surgery, confirming MRI as the most sensitive neuroimaging technique in the evaluation of pituitary masses.The occurrence of multiple pituitary adenomas supports the role played by abnormal extrahypophiseal regulatory mechanisms in the pathogenesis of pituitary tumors [3]. Genetic and molecular mechanisms of tumorigenesis may be involved in the pathogenesis of concomitant pituitary adenomas, and the coexistence of multiple adenomas in the same pituitary gland strongly emphasizes the pathogenic role of abnormal stimulatory and/or inhibitory extrapituitary factors. These latter could play a role in the recurrence of pituitary tumors by promoting growth and clonal expansion of genetically altered cells [4].In conclusion, although several combi...

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A somatotropin-producing pituitary adenoma with an isolated adrenocorticotropin-producing pituitary adenoma in a female patient with acromegaly, subclinical Cushing’s disease and a left adrenal tumor

Cited by 8 publications

References 24 publications

Two Synchronous Pituitary Adenomas Causing Cushing Disease and Acromegaly

Two Synchronous Pituitary Adenomas Causing Cushing Disease and Acromegaly

Isolated double adrenocorticotropic hormone-secreting pituitary adenomas: A case report and review of the literature

Comment on “A somatotropin-producing pituitary adenoma with an isolated adrenocorticotropin-producing pituitary adenoma in a female patient with acromegaly, subclinical Cushing’s disease and a left adrenal tumor” [Letter to the Editor]

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