Acromegaly caused by growth hormone-releasing hormone-producing tumors: long-term observational studies in three patients

Pituitary. 2007;10(3):237-49. doi: 10.1007/s11102-007-0045-7.

Abstract

We report on three newly diagnosed patients with extracranial ectopic GHRH-associated acromegaly with long-term follow-up after surgery of the primary tumor. One patient with a pancreatic tumor and two parathyroid adenomas was the index case of a large kindred of MEN-I syndrome. The other two patients had a large bronchial carcinoid. The first patient is still in remission now almost 22 years after surgery. In the two other patients GHRH did not normalize completely after surgery and they are now treated with slow-release octreotide. IGF-I normalized in all patients. During medical treatment basal GH secretion remained (slightly) elevated and secretory regularity was decreased in 24 h blood sampling studies. We did not observe development of tachyphylaxis towards the drug or radiological evidence of (growing) metastases. We propose life-long suppressive therapy with somatostatin analogs in cases with persisting elevated serum GHRH concentrations after removal of the primary tumor. Independent parameters of residual disease are elevated basal (nonpulsatile) GH secretion and decreased GH secretory regularity.

Publication types

  • Case Reports

MeSH terms

  • Acromegaly / etiology*
  • Acromegaly / pathology
  • Adenoma / metabolism*
  • Adenoma / pathology
  • Adenoma / surgery
  • Adult
  • Carcinoid Tumor / diagnostic imaging
  • Carcinoid Tumor / metabolism*
  • Carcinoid Tumor / surgery
  • Entropy
  • Female
  • Hormones / blood
  • Human Growth Hormone / metabolism*
  • Humans
  • Longitudinal Studies
  • Lung Neoplasms / diagnostic imaging
  • Lung Neoplasms / metabolism*
  • Lung Neoplasms / surgery
  • Magnetic Resonance Imaging
  • Male
  • Middle Aged
  • Octreotide
  • Pancreatic Neoplasms / metabolism*
  • Pancreatic Neoplasms / pathology
  • Pancreatic Neoplasms / surgery
  • Paraneoplastic Endocrine Syndromes / metabolism*
  • Paraneoplastic Endocrine Syndromes / pathology
  • Paraneoplastic Endocrine Syndromes / surgery
  • Parathyroid Neoplasms / metabolism*
  • Parathyroid Neoplasms / pathology
  • Parathyroid Neoplasms / surgery
  • Pituitary Gland / pathology
  • Positron-Emission Tomography
  • Tomography, X-Ray Computed
  • Treatment Outcome

Substances

  • Hormones
  • Human Growth Hormone
  • Octreotide