Resolution of Acromegaly, Amenorrhea-Galactorrhea Syndrome, and Hypergastrinemia after Resection of Jejunal Carcinoid

Spero, Michael; White, Eugene A.

doi:10.1210/jcem-60-2-392

Cited by 21 publications

(7 citation statements)

References 14 publications

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“…8,15 It is noteworthy that persistence of abnormal GH responsiveness to TRH has been described in two patients with ectopic GHRH syndrome, even after complete removal of GHRH-secreting tumors and remission of acromegaly. 5,25 It is likely that hyperplastic somatotrophs do not lose aberrant characteristics immediately after the decline of elevated GHRH levels.…”

Section: Discussionmentioning

confidence: 99%

Acromegaly associated with a granular cell tumor of the neurohypophysis: a clinical and histological study

Losa

Saeger²,

Mortini

et al. 2000

Journal of Neurosurgery

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Acromegaly is usually caused by a growth hormone (GH)-secreting pituitary adenoma, and hypersecretion of GH-releasing hormone (GHRH) from a hypothalamic or neuroendocrine tumor accounts for other cases. The authors report on the unusual association of acromegaly with a granular cell tumor of the neurohypophysis. A 42-year-old woman with a 10-year history of acral enlargement, headache, and menstrual abnormalities was referred to our department for a suspected GH-secreting pituitary adenoma. The patient's basal GH levels were mildly elevated at 4.8 microg/L, were not suppressed in response to an oral glucose tolerance test, and increased paradoxically after administration of thyrotropin-releasing hormone. The patient's insulin-like growth factor-1 (IGF-1) level was elevated at 462 microg/L, whereas a magnetic resonance image of the sella turcica revealed an intra- and suprasellar lesion that was compatible with a diagnosis of pituitary adenoma. A transsphenoidal approach to remove the lesion, which was mainly suprasellar, was successful during a second operative attempt, resulting in the clinical and biochemical regression of the patient's acromegaly. Four months postoperatively, the patient's basal GH level was 0.9 microg/L and her IGF-1 level was 140 microg/L. Histological analysis of the operative specimen demonstrated a granular cell tumor of the neurohypophysis, which when stained proved negative for pituitary hormones and GHRH. This case represents the first reported association between a granular cell tumor of the neurohypophysis and acromegaly. Granular cell tumor of the neurohypophysis could be added to the restricted list of neoplastic causes of acromegaly secondary to hypersecretion of a GH-releasing substance.

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Section: Discussionmentioning

confidence: 99%

Acromegaly associated with a granular cell tumor of the neurohypophysis: a clinical and histological study

Losa

Saeger²,

Mortini

et al. 2000

Journal of Neurosurgery

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“…Resolution of acromegaly was evident in the first weeks after operation, even though the paradoxical GH response to TRH persisted initially and disappeared only after some months (Spero & White, 1985;Boizel et al, 1987), suggesting that basal GH hypersecretion needs to be continuously stimulated by elevated GHRH level, whereas disappearance of the abnormal GH responsiveness to dynamic testing requires a longer period of time, probably because involution of the hyperplastic somatotrophs is not immediate. Reversibility of somatotroph hyperplasia is further demonstrated by normalization of the neuroradiological picture in six patients who had a demonstrable pituitary lesion before surgery and complete resection of the GHRH-secreting tumour (Scheithauer et al, 1984;Spero & White, 1985;Carroll et al, 1987;Ramsay et al, 1987;Hawkins et al, 1985;Barth et al, 1991).…”

Section: Therapymentioning

confidence: 99%

Pathophysiology and clinical aspects of the ectopic GH‐releasing hormone syndrome

Losa¹,

Werder²

1997

Clinical Endocrinology

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“…18 In many cases of GH-producing pituitary tumors, prolactin levels can be moderately elevated, but it is more frequent in ectopic acromegaly. 2,19 A normal or smallsized pituitary gland in addition to acromegaly suggests extrapituitary secretion of GHRH. However, a diffusely enlarged pituitary, which could be difficult to distinguish from a pituitary adenoma, is frequently found on MRI because of somatotrope hyperplasia as the consequence of a prolonged stimulation of these cells by GHRH.…”

Section: Discussionmentioning

confidence: 99%

Acromegaly Secondary to Ectopic Growth Hormone-Releasing Hormone-Secreting Bronchial Carcinoid Cured After Pneumectomy

Reuters¹,

Dias²,

Pupo³

et al. 2003

The Endocrinologist

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Acromegaly is caused by a primary growth hormonesecreting pituitary adenoma in more than 95% of the cases. In a small number of patients, it is the result of hypersecretion of growth hormone-releasing hormone (GHRH). Clinical and laboratory findings are similar in both conditions, and the diagnosis of a GHRHsecreting tumor is possible only by identification of GHRH in the tumor or circulation. We report the case of a 61-year-old woman referred to the Hospital Naval Marcílio Dias to investigate a pulmonary mass. The mass was large and calcified, suggesting a hamartoma. The patient also had acromegalic features, elevated serum insulin-like growth factor-1 (IGF-1) levels, and no GH suppression after an oral glucose tolerance test. Magnetic resonance imaging (MRI) showed a diffusely enlarged pituitary gland, suggestive of hyperplasia. The hypothesis of acromegaly secondary to ectopic GHRH secretion by the pulmonary mass was made. A left pneumectomy was performed and histopathologic analysis revealed a carcinoid tumor. A fragment of the tumor was sent for special stains and GHRH was identified by immunohistochemistry. The clinical and biochemical aspects of acromegaly improved 2 weeks after surgery. One year later, serum GH and IGF-1 levels were normal and MRI revealed a partial empty sella. The differential diagnosis of somatotroph adenoma and ectopic GHRH hypersecretion is difficult but is essential to appropriate therapy.A cromegaly is a rare disease caused by a primary growth hormone-secreting pituitary adenoma (classic acromegaly) in the large majority of the cases. 1 Less than 5% of cases are caused by eutopic tumors (hypothalamic gangliocytomas, hamartomas, and gliomas) or ectopic tumors (carcinoid tumors, pancreatic endocrine tumors, adrenal adenoma, smallcell lung cancer, and pheochromocytoma) secreting growth hormone-releasing hormone (GHRH). This condition is termed ectopic acromegaly. 2 In addition, acromegaly caused by ectopic 6H hypersecretion is extremely rare. 3 We report a patient with acromegaly resulting from ectopic GHRH secretion by a pulmonary carcinoid tumor. CASE REPORTA 61-year-old white woman was referred to the Hospital Naval Marcílio Dias for evaluation of a pulmonary mass in April 2000. The mass was extensively studied by bronchofibroscopy, helicoidal tomography, and magnetic resonance imaging (MRI). The mass was approximately 8 cm in diameter, calcified, and in close contact with mediastinal vessels, suggesting a hamartoma. No metastatic lesions were found. The patient exhibited clinical signs of acromegaly with typical facial features and acral changes. She reported progressive increase in hand and foot size. Hypertension had been present for 10 years. She reported arthralgia, hoarseness, and hyperhidrosis. Physical examination revealed a blood pressure of 140/90 mm Hg (taking ramipril 5 mg per day) and heart rate was 82 beats/min. No visual field defect was found and galactorrhea was not present.Endocrine evaluation confirmed acromegaly. An oral glucose tolerance test (OGTT) was perform...

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Resolution of Acromegaly, Amenorrhea-Galactorrhea Syndrome, and Hypergastrinemia after Resection of Jejunal Carcinoid

Cited by 21 publications

References 14 publications

Acromegaly associated with a granular cell tumor of the neurohypophysis: a clinical and histological study

Acromegaly associated with a granular cell tumor of the neurohypophysis: a clinical and histological study

Pathophysiology and clinical aspects of the ectopic GH‐releasing hormone syndrome

Acromegaly Secondary to Ectopic Growth Hormone-Releasing Hormone-Secreting Bronchial Carcinoid Cured After Pneumectomy

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