pNET co-secreting GHRH and calcitonin: ex vivo hormonal studies in human pituitary cells

Zornitzki, Taiba; Rubinfeld, Hadara; Lysyy, Lyudmila; Schiller, Tal; Raverot, Véronique; Shimon, Ilan; Knobler, Hilla

doi:10.1530/edm-15-0134

Cited by 5 publications

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“…Before GHRH isolation in 1982, its possible secretion was tested mostly with bioassay, which is an indirect examination based on GH production by rat pituitary cells triggered by the substance obtained from tumor extracts ( 88 ). This method may still serve as an additional test and was recently performed on cultured human pituitary cells ( 16 , 60 ). As shown in Table 4 , GHRH expression was documented histopathologically in 99 tumors, mostly by means of immunostaining which was performed in 83 cases.…”

Section: Methodsmentioning

confidence: 99%

“…Somatostatin expression has been found in both intra- and extracranial tumor tissues (6 and 9 documented cases, respectively). Other marked substances included ACTH, calcitonin, insulin, gastrin, glucagon, PP, serotonin, and VIP, produced one or more at a time by several tumors ( 16 , 33 , 40 , 77 , 90 ). As above mentioned, tumoral hormone expression has not always translated to elevated plasmatic levels, let alone the symptoms.…”

Section: Methodsmentioning

confidence: 99%

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Acromegaly Caused by Ectopic Growth Hormone Releasing Hormone Secretion: A Review

et al. 2022

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IntroductionEctopic acromegaly is a rare condition caused most frequently by growth hormone releasing hormone (GHRH) secretion from neuroendocrine tumors. The diagnosis is often difficult to establish as its main symptoms do not differ from those of acromegaly of pituitary origin.ObjectivesTo determine most common clinical features and diagnostic challenges in ectopic acromegaly.Patients and MethodsA search for ectopic acromegaly cases available in literature was performed using PubMed, Cochrane, and MEDline database. In this article, 127 cases of ectopic acromegaly described after GHRH isolation in 1982 are comprehensively reviewed, along with a summary of current state of knowledge on its clinical features, diagnostic methods, and treatment modalities. The most important data were compiled and compared in the tables.ResultsNeuroendocrine tumors were confirmed in 119 out of 121 patients with histopathological evaluation, mostly of lung and pancreatic origin. Clinical manifestation comprise symptoms associated with pituitary hyperplasia, such as headache or visual field disturbances, as well as typical signs of acromegaly. Other endocrinopathies may also be present depending on the tumor type. Definitive diagnosis of ectopic acromegaly requires confirmation of GHRH secretion from a tumor using either histopathological methods or GHRH plasma concentration assessment. Hormonal evaluation was available for 84 patients (66%) and histopathological confirmation for 99 cases (78%). Complete tumor resection was the main treatment method for most patients as it is a treatment of choice due to its highest effectiveness. When not feasible, somatostatin receptor ligands (SRL) therapy is the preferred treatment option. Prognosis is relatively favorable for neuroendocrine GHRH-secreting tumors with high survival rate.ConclusionAlthough ectopic acromegaly remains a rare disease, one should be aware of it as a possible differential diagnosis in patients presenting with additional symptoms or those not responding to classic treatment of acromegaly.

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Section: Methodsmentioning

confidence: 99%

Section: Methodsmentioning

confidence: 99%

Acromegaly Caused by Ectopic Growth Hormone Releasing Hormone Secretion: A Review

et al. 2022

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GHRH-producing tumors and other neuroendocrine neoplasms associated with acromegaly and/or gigantism

Pakbaz

Hodgson

Mete

2021

Gigantism and Acromegaly

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Pituitary MRI Features in Acromegaly Resulting From Ectopic GHRH Secretion From a Neuroendocrine Tumor: Analysis of 30 Cases

Potorac

Bonneville

Daly

et al. 2022

The Journal of Clinical Endocrinology &Amp; Metabolism

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Context Ectopic acromegaly is a consequence of rare neuroendocrine tumors (NET) that secrete growth hormone releasing hormone (GHRH). This abnormal GHRH secretion drives growth hormone (GH) and insulin-like growth factor 1 (IGF-1) excess, with a clinical presentation similar to classical pituitary acromegaly. Identifying the underlying cause for the GH hypersecretion in the setting of ectopic GHRH excess is, however, essential for proper management both of acromegaly and the NET. Owing to its rarity, the imaging characteristics of the pituitary in ectopic acromegaly have not been analyzed in depth in large series. Objective Characterize pituitary magnetic resonance imaging (MRI) features at baseline and after NET treatment in patients with ectopic acromegaly. Design Multicenter, international, retrospective Setting Tertiary referral pituitary centers Patients 30 ectopic acromegaly patients due to GHRH hypersecretion Intervention None Main outcome measure MRI characteristics of pituitary gland, particularly T2-weighted signal Results In 30 patients with ectopic GHRH-induced acromegaly, we found that most patients had hyperplastic pituitaries. Hyperplasia was usually moderate but was occasionally subtle, with only small volume increases compared to normal ranges for age and sex. T2-weighted signal was hypointense in most patients, especially in those with hyperplastic pituitaries. After treatment of the NET, pituitary size diminished and T2-weighted signal tended to normalize. Conclusions This comprehensive study of pituitary MRI characteristics in ectopic acromegaly underlines the utility of performing T2-weighted sequences in the MRI evaluation of patients with acromegaly as an additional tool that can help to establish the correct diagnosis.

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pNET co-secreting GHRH and calcitonin: ex vivo hormonal studies in human pituitary cells

Cited by 5 publications

References 10 publications

Acromegaly Caused by Ectopic Growth Hormone Releasing Hormone Secretion: A Review

Acromegaly Caused by Ectopic Growth Hormone Releasing Hormone Secretion: A Review

GHRH-producing tumors and other neuroendocrine neoplasms associated with acromegaly and/or gigantism

Pituitary MRI Features in Acromegaly Resulting From Ectopic GHRH Secretion From a Neuroendocrine Tumor: Analysis of 30 Cases

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