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Case Reports
. 2010 Sep;126(3):e728-33.
doi: 10.1542/peds.2010-0596. Epub 2010 Aug 16.

Bicalutamide and third-generation aromatase inhibitors in testotoxicosis

Anne M Lenz  1 , Dorothy Shulman, Erica A Eugster, Samar Rahhal, John S Fuqua, Ora H Pescovitz, Katherine A Lewis
Affiliations
Case Reports

Bicalutamide and third-generation aromatase inhibitors in testotoxicosis

Anne M Lenz et al. Pediatrics. 2010 Sep.

Abstract

Testotoxicosis, a form of gonadotropin-independent precocious puberty, results from an activating mutation of the luteinizing hormone receptor expressed in testicular Leydig cells. Affected males experience early testosterone secretion, virilization, advancing bone age, and resultant short stature. Recently, the use of combination therapy with a potent antiandrogen agent (bicalutamide) and a third-generation aromatase inhibitor (anastrozole or letrozole) was reported to yield encouraging short-term results. We present here the results of longer-term treatment (4.5 and 5 years) with this combination therapy in 2 boys who demonstrated that it is well tolerated, slows bone-age advancement in the face of continued linear growth, and prevents progression of virilization.

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Figures

FIGURE 1
FIGURE 1
Growth chart for patient 1. A, Start of testolactone and spironolactone therapy; B, start of bicalutamide and letrozole (depot leuprolide was added 1 month later). ▲ indicates BA radiograph results at corresponding heights.
FIGURE 2
FIGURE 2
Growth chart for patient 2. A, Start of bicalutamide and anastrozole; B, depot leuprolide reinitiated. ▲ indicates BA radiograph results at corresponding heights. CASE REPORT
See this image and copyright information in PMC

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