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This version published online on May 20, 2008
Journal of Clinical Endocrinology & Metabolism, doi:10.1210/jc.2007-2283
A more recent version of this article appeared on August 1, 2008
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Submitted on October 11, 2007
Accepted on May 14, 2008

Treatment of Pituitary Gigantism with the Growth Hormone Receptor Antagonist Pegvisomant

Naila Goldenberg MD, Michael S. Racine MD, Pamela Thomas MD, Bernard Degnan MD, William Chandler MD, and Ariel Barkan MD*

From the Division of Metabolism, Endocrinology and Diabetes, Department of Internal Medicine (NG, MR, WC, AB) and the Department of Neurosurgery (WC and AB), University of Michigan Medical Center, Ann Arbor, MI 48109; Pediatric Endocrinology, Lutheran Children's Hospital (PT), Fort Wayne, IN 46804, Pediatric Endocrinology, St. John's Hospital, Detroit, MI 48236

* To whom correspondence should be addressed. E-mail: abarkan{at}umich.edu.

Context. Treatment of pituitary gigantism is complex and the results are usually unsatisfactory.

Objective. To describe the results of therapy of 3 children with pituitary gigantism by a GH receptor antagonist, pegvisomant.

Design. Descriptive Case Series of up to 3 1/2 years duration.

Setting. University hospital

Patients. Three children (1 female, 2 males) with pituitary gigantism whose GH hypersecretion was incompletely controlled by surgery, somatostatin analog and dopamine agonist.

Intervention. Administration of pegvisomant

Main outcome measures. Plasma IGF-1, growth velocity

Results. In all 3 children, pegvisomant rapidly decreased plasma IGF-1 concentrations. Growth velocity declined to subnormal or normal values. Statural growth fell into lower growth percentiles and acromegalic features resolved. Pituitary tumor size did not change in 2 children but increased in 1 boy despite concomitant therapy with a somatostatin analog.

Conclusions. Pegvisomant may be an effective modality for the therapy of pituitary gigantism in children. Titration of the dose is necessary for optimal efficacy, and regular surveillance of tumor size is mandatory.
Key words: pegvisomant • gigantism • acromegaly • growth hormone • antagonist • IGF-I • growth






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