Hormonal and Volumetric Long Term Control of a Growth Hormone-Releasing Hormone-Producing Carcinoid Tumor

Bruel, Ann Van den; Fevery, Johan; Dorpe, Jo Van; Hofland, Leo J.; Bouillon, Roger

doi:10.1210/jcem.84.9.6001

Cited by 18 publications

(5 citation statements)

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“…Since the initial report of the possible association between the neuroendocrine functional tumor and acromegaly, suggested by Altmann et al. in 1959, around 170 cases of suspected ectopic acromegaly have been described in literature to date, mostly as case reports ( 4 , 6 , 9 – 21 ). To our best knowledge, only 19 cases were described as caused by an ectopic source of GH ( 22 – 27 ).…”

Section: Methodsmentioning

confidence: 99%

“…SRL therapy is the preferred treatment option for patients with inoperable tumors or disseminated metastatic disease, yet, it may also serve successfully as an adjuvant therapy for patients who undergo surgical procedures ( 3 ). Therapy with SRL was administered to 37.8% of patients at different stages of treatment, being the main method in 22 cases ( 9 , 40 , 45 , 47 , 50 , 68 , 75 , 99 , 100 ). However, in some cases it was proved to not be sufficiently effective, presumably due to the lack of somatostatin receptor 2 (SSTR-2) in tumor tissue, which is also uncommon, in hyperplastic somatotroph cells ( 61 ).…”

Section: Methodsmentioning

confidence: 99%

“…The efficacy of surgical resection and SRL therapy cannot be righteously compared in this condition, as the second method has usually served as basic treatment in more advanced cases, often with metastatic disease which itself is a factor of worse prognosis ( 36 ). Along with SRL, other supportive methods that may be mentioned are chemotherapy, radiotherapy, immunotherapy, metastases embolization, and other hormonal treatments such as bromocriptine or pegvisomant ( 9 , 18 , 33 , 36 , 40 , 42 , 43 , 46 , 49 , 51 , 73 , 78 , 90 , 96 ). Altogether, 41 patients received one of these adjuvant treatment options, with pituitary radiation being the most frequent modality ( 18 , 19 , 40 , 59 , 64 , 67 , 70 , 77 , 78 , 96 ).…”

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%

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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“…There were 74 cases reported in the literature until 2012 5 . In cases of ectopic GHRH secretion -bronchial origin varying between 33-79% 8,9 Ectopic cases of acromegaly are more common in women (60%), with median age at presentation -39 years and median diameter -55 mm 7 . Bronchial carcinoids causing ectopic acromegaly are usually of the typical type and located centrally 8,10 .…”

Section: Situationmentioning

confidence: 99%

Acromegaly in association with a bronchial carcinoid tumour

Rys¹,

Smith²

2015

EJEA

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Bronchial carcinoid tumours are uncommon pulmonary neoplasms. Manifestation of a bronchial carcinoid with acromegaly secondary to extra-pituitary growth hormone releasing hormone (GHRH) productionis rare, but bronchial carcinoid tumours are the most common cause of ectopic GHRH secretion. We report the case of 60 year old female, ex-smoker with hypertension presenting with cough,dyspnoea and right lower lobe opacity on the chest X-ray. Patient was noted to have features of acromegaly and IGF-1 was requested. Subsequent CT TAP showed a right lower lobe collapse, 4.5 cm mass obstructing the lower lobe bronchus which was visualised but could not be biopsied during bronchoscopy. 18-FDG PETCTscan showed only mildly avid right lower lobe mass, likely to be a slow growing carcinoid tumour, with chronic collapse of the right lower lobe. Pre-operative IGF-1 level came back elevated at 67 nmol/l. Patient underwent right mid and lower lobectomies and the histology showed morphological appearance of grade 1 neuroendocrine carcinoma (classic carcinoid tumour) with no lymph node involvement. Patient underwent the oral glucose tolerance test (OGTT) with growth hormone (GH) levels reaching a nadir value of 0.82 µ/L. Patient's pituitary hormone profile and the MRI of the pituitary gland were both normal. The GHRH assay was not available.The most recent postoperative random GH level was 0.6 µ/L and IGF-1 was 20 nmol/L. Bronchial carcinoid tumours are one of the commonest causes of ectopic GHRH production, with similar cases reported in the literature. In the view of possible insufficient normalisation of the GH levels post OGTT our patient will require further close follow up. Our case represents the classic carcinoid tumour which is reported to have very good prognosis following surgical resection. The results of staining for GHRH on tissue samples are still awaited as the samples are being processed abroad.

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“…These tumors have somatostatin type-2 receptors and they are usually visible by octreoscan and treatable by somatostatin analogues. Treatment induces a reduction in plasma GH and sometimes in GHRH, a reduction in primary tumor volume and in pituitary enlargement [41]. …”

Section: Ghrh-secreting Tumorsmentioning

confidence: 99%

Growth Hormone-Releasing Hormone: Past and Present

Sassolas¹

2000

Horm Res Paediatr

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The discovery of hypothalamic hypophysiotropic factors confirmed the hypothesis of Green and Harris in the late 1940s. These hormones were isolated from their eutopic site of production (the hypothalamus) with the exception of growth hormone (GH)-releasing hormone (GHRH), which was isolated from an ectopic, tumoral site of production and found to be responsible for acromegaly. Following the isolation, characterization and synthesis of human GHRH, clinical studies were performed and are described below. Circulating levels of GHRH can be measured and provide the basis for the diagnosis of acromegaly related to the ectopic, tumoral production of GHRH. At present, GHRH is used as a test of GH secretion mainly as an adjunct to other agents which modify somatostatin status, or to GH-releasing peptides. Its therapeutic potential in children and the elderly is still under investigation. The role of GHRH in the pulsatile secretion of GH is described.

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Hormonal and Volumetric Long Term Control of a Growth Hormone-Releasing Hormone-Producing Carcinoid Tumor

Cited by 18 publications

References 15 publications

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

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

Acromegaly in association with a bronchial carcinoid tumour

Growth Hormone-Releasing Hormone: Past and Present

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