Background: The aim of therapy in patients with congenital adrenal hyperplasia (CAH) is to use glucocorticoid doses as low as possible to achieve adrenal suppression. Both chronically increased androgen secretion and increased glucocorticoid exposure may adversely affect adult height and in some patients this therapeutic balance is difficult to achieve. In these particular cases aromatase inhibitors (AI) could be indicated with de aim to reduce glucocorticoid doses avoiding the subsequent increase of bone age. However, potential safety concerns of the inhibition of oestrogen biosynthesis should be taken into account since some publications report negative effects on bone physiology.
Case report: A 6 year-old boy with CAH was diagnosed at 2 years of age: with pubic hair and bone age of 8 years according to Greulich and Pyle. He required high hydrocortisone dosages (20 mg/m2 BSA per day) to suppress excessive androgen secretion with consequent slowing of growth velocity (−6.2 S.D.). At four years of age, treatment with AI (anastrazole) was considered. Two years after the start of AI therapy the diagnosis of isthmic spondylolisthesis was made by X-ray. The patient did not report lumbar pain or movement limitation.
Discussion: Spondylolisthesis has not been previously described with anastrazole treatment and is not pathologically related to changes in vertebral morphology that have been reported with AI. However, the spondylolisthesis was weird of the age of our patient since his height and weight were insufficient to cause mechanical stress. These findings led us to consider the pathological role of anastrazole.
Conclusion: Use of anastrazole therapy, particularly in prepuberty may predispose to vertebral changes. We recommend the use of anastrazole with serial evaluation of vertebral morphology by X-ray and orthopaedic evaluation, before and during treatment.
10 - 12 Sep 2016
European Society for Paediatric Endocrinology