A Case of Acromegaly Associated with Subclinical Cushing's Disease

Tsuchiya, Kyoichiro; Ohta, Kazuki; Yoshimoto, Takanobu; Doi, Masaru; Izumiyama, Hajime; Hirata, Yukio

doi:10.1507/endocrj.k06-031

Cited by 9 publications

(6 citation statements)

References 26 publications

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“…Recently, a case of a GH-producing eosinophilic adenoma causing typical acromegalic features was described, with a concomitant lack of circadian rhythm of both ACTH and cortisol [20]. This is the first report to describe the impact of tumorcaused optic chiasm compression on melatonin secretion.…”

Section: Discussionmentioning

confidence: 75%

Melatonin and cortisol profiles in patients with pituitary tumors

Zielonka

Sowiński

Nowak

et al. 2015

Neurologia i Neurochirurgia Polska

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The optic tract section at the optic chiasm is expected to disturb the suprachiasmatic nucleus (SCN) rhythm, circadian rhythm and melatonin secretion rhythms in humans, although detailed studies have never been conducted. The aim of this paper was to describe melatonin and cortisol profiles in patients with a pituitary tumor exerting optic chiasm compression. Six patients with pituitary tumors of different size, four of whom had significant optic chiasm compression, were examined. In each brain, MRI, an ophthalmological examination including the vision field and laboratory tests were performed. Melatonin and cortisol concentrations were measured at 22:00 h, 02:00 h, 06:00 h, and 10:00 h in patients lying in a dark, isolated room. One of the four cases with significant optic chiasm compression presented a flattened melatonin rhythm. The melatonin rhythm was also disturbed in one patient without optic chiasm compression. Larger tumors may play a role in the destruction of neurons connecting the retina with the suprachiasmatic nucleus (SCN) and breaking of basic way for inhibiting effect to the SCN from the retina.

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Section: Discussionmentioning

confidence: 75%

Melatonin and cortisol profiles in patients with pituitary tumors

Zielonka

Sowiński

Nowak

et al. 2015

Neurologia i Neurochirurgia Polska

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“…Plurihormonal adenomas could be either monomorphous (one cell type secreting multiple hormones) or plurimorphous (multiple cell types, each secreting one hormone) (7). We surmise that our case was a plurihormonal pituitary adenoma rather than a double adenoma based on his first post-operative histological and immunohistochemical examination, although clonal analysis and electron microscopic study was not performed.…”

Section: Discussionmentioning

confidence: 88%

“…Among the 15 possible cases of coexistent acromegaly and Cushing's disease described in the literature (2) (3) (4) (5) (6) (7) (8), at least nine cases had predominant acromegalic features and only two cases had predominant Cushingoid features, and it was not ascertainable what features predominated in the remaining four cases. There are several theories explaining this, such as production of low-activity high-molecular weight ACTH (8) or biologically inactive ACTH molecules due to impaired PC 1/3 expression in these tumours or impaired intracellular secretory process of ACTH compared with GH (7). Finally, hypercortisolaemia can be masked by GH excess.…”

Section: Discussionmentioning

confidence: 99%

Pasireotide therapy in a rare and unusual case of plurihormonal pituitary macroadenoma

Rajendran

Naik

Sandeman

et al. 2013

Endocrinology, Diabetes &Amp; Metabolism Case Reports

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SummaryWe report the use of pasireotide in a rare and unusual case of pituitary macroadenoma co-secreting GH, prolactin and ACTH. A 62-year-old Caucasian man presented with impotence. Clinically, he appeared acromegalic and subsequent investigations confirmed GH excess and hyperprolactinaemia. Magnetic resonance imaging (MRI) of pituitary revealed a large pituitary macroadenoma. He underwent trans-sphenoidal surgery and histology confirmed an adenoma with immunohistochemistry positive for ACTH, GH and prolactin. Acromegaly was not cured following surgery and inadequately controlled despite subsequent octreotide therapy. He underwent further debulking pituitary surgery, following which IGF1 levels improved but still high. This time adenoma cells showed immunohistochemistry positivity for ACTH only, following which subsequent investigations confirmed intermittent hypercortisolaemia compatible with pituitary Cushing's disease. We recommended radiotherapy, but in view of the pluripotential nature of the tumour, we proceeded with a trial of s.c. pasireotide therapy on the basis that it may control both his acromegaly and Cushing's disease. After 3 months of pasireotide therapy, his mean GH and IGF1 levels improved significantly, with improvement in his symptoms but intermittent hypercortisolaemia persists. His glycaemic control deteriorated requiring addition of new anti-diabetic medication. MRI imaging showed loss of contrast uptake within the tumour following pasireotide therapy but no change in size. We conclude that our patient has had a partial response to pasireotide therapy. Long-term follow-up studies are needed to establish its safety and efficacy in patients with acromegaly and/or Cushing's disease.Learning points Plurihormonal pituitary adenomas are rare and unusual.Patients with pituitary adenomas co-secreting ACTH and GH are more likely to present with acromegaly because GH excess can mask hypercortisolaemia.Pasireotide holds potential where conventional somatostatin analogues are not effective in acromegaly due to higher affinity for somatostatin receptor subtypes 1, 2, 3 and 5.Significant deterioration in glycaemic control remains a concern in the use of pasireotide.Currently, long-term safety and efficacy of pasireotide in patients with acromegaly and/or Cushing's disease are not fully clear.

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“…female GHoma CD IHC Bimorphous tumor (heterogeneous) 2002 Tahara et al [ 7 ] 53 y.o. female Subclinical GHoma CD IHC ISH The same cell (double producer) 2006 Tsuchiya et al [ 16 ] 54 y.o. male GHoma SCD IHC N.D. 2009 Oki et al [ 17 ] 36 y.o.…”

Section: Discussionmentioning

confidence: 99%

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

et al. 2017

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BackgroundA functional pituitary adenoma can produce multiple anterior-pituitary hormones, such as growth hormone (GH) -producing adenomas (GHoma) with prolactin or thyrotropin stimulating hormone production in the same lineage. However, it is very rare that acromegaly shows subclinical Cushing’s disease (SCD) beyond the lineage. Here we describe the involvement of intratumoral coexistence with 2 types of hormone-producing cells associated with different lineage in acromegaly concomitant with SCD.Case presentationIn our study, we performed clinical evaluation of the patient showing acromegaly with SCD. To elucidate the mechanisms of this pathology, we analyzed immunohistochemistry and gene expression of anterior-pituitary hormones and transcriptional factors in the resected pituitary tumor. On immunohistochemical staining, most of the tumor cells were strongly stained for GH antibody, while some cells were strongly positive for adrenocorticotropic hormone (ACTH). Gene expression analysis of a transsphenoidal surgery sample of the pituitary gland revealed that ACTH-related genes, such as POMC, Tpit, and NeuroD1 mRNA, had higher expression in the tumor tissue than the nonfunctional adenoma but lower expression compared to an adenoma of typical Cushing’s disease. Further, double-labeling detection methods with a fluorescent stain for ACTH and GH demonstrated the coexistence of ACTH-positive cells (GH-negative) among the GH-positive cells in the tumor. Additionally, Pit-1 expression was reduced in the ACTH-positive cells from tumor tissue primary culture.ConclusionHere we described a case of a pituitary tumor diagnosed with acromegaly associated with SCD. We performed quantitative-expression analyses of transcriptional factors of the tumor tissue and immunohistochemistry analysis of tumor-derived primary culture cells, which suggested that the multihormonal pituitary adenoma concomitant with Pit-1 and Tpit lineage cells caused acromegaly associated with SCD.

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A Case of Acromegaly Associated with Subclinical Cushing's Disease

Cited by 9 publications

References 26 publications

Melatonin and cortisol profiles in patients with pituitary tumors

Melatonin and cortisol profiles in patients with pituitary tumors

Pasireotide therapy in a rare and unusual case of plurihormonal pituitary macroadenoma

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

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