An unusual case of Cushing's syndrome due to bihormonal ACTH–prolactin secreting pituitary macroadenoma with rapid response to cabergoline

Kunasegaran, Shalini; Croxson, Michael; Holdaway, I. M.; Murphy, Rinki

doi:10.1136/bcr-2017-219921

Cited by 4 publications

(4 citation statements)

References 18 publications

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“…There are very few cases in the literature with a clinical phenotype of multiple pituitary hormone secretions 1 3–10. Our case has some important features, which need further elaboration.…”

Section: Discussionmentioning

confidence: 73%

Dopamine agonist-responsive Cushing’s disease

et al. 2019

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A 47-year-old Caucasian man was referred to our clinic with a severe clinical and biochemical phenotype of endogenous hypercortisolism for further evaluation and treatment. In addition to confirming adrenocorticotropic hormone (ACTH)-dependent Cushing’s syndrome, we found left temporal hemianopsia, massively increased prolactin, increased growth hormone/insulin-like growth factor 1 values, hypogonadotropic hypogonadism and central hypothyroidism. As the cause of these abnormalities we revealed an invasive macroadenoma of the pituitary secreting ACTH, prolactin and growth hormone, resulting not only in a clinically predominant picture of Cushing’s syndrome but also causing hypogonadotropic hypogonadism and central hypothyroidism. The patient responded surprisingly well to dopamine agonist treatment leading not only to normalisation of prolactin levels but also to clinical and biochemical remission of Cushing’s syndrome. Tumour size decreased successively in follow-up MRI scans. Despite lacking immunohistochemical analysis of tumour tissue, we assume plurihormonal secretion of ACTH, prolactin and growth hormone from pituitary macroadenoma, which fortunately responded well to dopamine agonist treatment.

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Section: Discussionmentioning

confidence: 73%

Dopamine agonist-responsive Cushing’s disease

et al. 2019

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“…Dopamine receptor (DR) expression in ACTH-secreting PAs has been described [ 17 ]. While several case reports have demonstrated that ACTH-secreting PAs may respond to treatment with Cabergoline [ 18 , 19 , 20 , 21 , 22 , 23 ], the results in larger cohorts of patients bearing such tumors still remain controversial [ 24 , 25 ]. However, administration of dopamine agonists is worth considering as an option to treat these tumors [ 13 , 14 ], and a clinical trial on Cabergoline treatment in corticotroph PAs (Mumbai, India ( (accessed on 6 November 2021)) [ 26 ]; trial identifier: NCT00889525; Table 1 ) has been initiated.…”

Section: Emerging Targeted Treatment Strategies At the Molecular Levelmentioning

confidence: 99%

Targeting Aggressive Pituitary Adenomas at the Molecular Level—A Review

Voellger

Zhang

Benzel

et al. 2021

JCM

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Pituitary adenomas (PAs) are mostly benign endocrine tumors that can be treated by resection or medication. However, up to 10% of PAs show an aggressive behavior with invasion of adjacent tissue, rapid proliferation, or recurrence. Here, we provide an overview of target structures in aggressive PAs and summarize current clinical trials including, but not limited to, PAs. Mainly, drug targets in PAs are based on general features of tumor cells such as immune checkpoints, so that programmed cell death 1 (ligand 1) (PD-1/PD-L1) targeting may bear potential to cure aggressive PAs. In addition, epidermal growth factor receptor (EGFR), mammalian target of rapamycin (mTOR), vascular endothelial growth factor (VEGF), fibroblast growth factor (FGF) and their downstream pathways are triggered in PAs, thereby modulating tumor cell proliferation, migration and/or tumor angiogenesis. Temozolomide (TMZ) can be an effective treatment of aggressive PAs. Combination of TMZ with 5-Fluorouracil (5-FU) or with radiotherapy could strengthen the therapeutic effects as compared to TMZ alone. Dopamine agonists (DAs) are the first line treatment for prolactinomas. Dopamine receptors are also expressed in other subtypes of PAs which renders Das potentially suitable to treat other subtypes of PAs. Furthermore, targeting the invasive behavior of PAs could improve therapy. In this regard, human matrix metalloproteinase (MMP) family members and estrogens receptors (ERs) are highly expressed in aggressive PAs, and numerous studies demonstrated the role of these proteins to modulate invasiveness of PAs. This leaves a number of treatment options for aggressive PAs as reviewed here.

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“…• PRL and LH; PRL and TSH [7] • GH and ACTH [9][10][11] • PRL and ACTH [5,7,12,13] • ACTH, LH, and TSH [7] • PRL, LH, FSH, and TSH [7] • GH, PRL, TSH, and a-SU [5] • GH, PRL, and ACTH [7,14] • GH, PRL, TSH, FSH, and aSU [15] Other less common combinations are also published [5]. Original examples of plurihormonal adenomas in the author series [7] are shown in…”

Section: Plurihormonal Pituitary Adenomasmentioning

confidence: 99%

“…Pituitary adenomas with the combination of different hormone groups from the same cell lineage (GH and prolactin or FSH and LH) are relatively common, but true plurihormonal adenomas with immunoreactivities that cross the cytogenetic lineage are rare in most series as "in Refs. [5,6,13]. "…”

Section: Pituitary Diseasesmentioning

confidence: 99%

Hormone Secretion in Pituitary Adenomas: Immunohistochemical Studies

Khani¹

2019

Pituitary Diseases

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Classification of pituitary adenomas is still changing and not completely satisfying. Various types of hormones, prehormonal substances, and transcription factors are detected in pituitary adenomas. Monohormonal secretion in pituitary adenomas is frequent, notably prolactin secretion. Secretion of more than one hormone normally originating from the same adenohypophyseal cell lineage is well known, classically GH-PRL and FSH-LH. Other combinations of hormonal secretion are reported; they are sometimes underestimated. Plurihormonal secretion in pituitary adenomas, which is secretion of hormones that are normally originating from different adenohypophyseal cell lineages, is usually underestimated and in most cases remains subclinical. An immunohistochemical study of all pituitary hormones and prehormonal substances, as demonstrating transcription factors, is not always available; it is frequently not performed. This chapter aims to show the underestimated and vague areas of pituitary adenomas and to emphasize the importance of immunohistochemical studies in the diagnosis and prediction of clinical outcomes of these adenomas.

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An unusual case of Cushing's syndrome due to bihormonal ACTH–prolactin secreting pituitary macroadenoma with rapid response to cabergoline

Cited by 4 publications

References 18 publications

Dopamine agonist-responsive Cushing’s disease

Dopamine agonist-responsive Cushing’s disease

Targeting Aggressive Pituitary Adenomas at the Molecular Level—A Review

Hormone Secretion in Pituitary Adenomas: Immunohistochemical Studies

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