The effects of long-term raloxifene and estradiol treatments on bone in a patient with congenital aromatase deficiency.

[aromatase deficiency]

In adult aromatase-deficient men , estrogen treatment has always resulted in a rapid skeletal maturation with epiphyseal closure and improved BMD . Raloxifene is a SERM with proven estrogen agonist action on bone that leads to an improvement in BMD and a reduction in bone turnover . The present study reports the effects of raloxifene and transdermal estradiol treatment , respectively , on epiphyseal closure and BMD in an aromatase-deficient man , over a 24 -month follow-up , with the aim of obtaining further insight into the role of estrogens in the male skeletal homeostasis .A 25 -year -old Caucasian man with aromatase deficiency , a bone age of 15 .3 years , unfused epiphyses and an impaired BMD was initially administered raloxifene (60 mg /day per os ) for 12 months , while transdermal estradiol (25 microg twice weekly ) was administered for the subsequent 12 months . During the follow-up , the effects of the two treatments on epiphyseal closure , BMD and bone turnover markers were investigated . An iliac crest bone biopsy was performed only before and after the raloxifene treatment , but it was not repeated after transdermal estradiol treatment .No changes in bone age were observed after raloxifene therapy , whereas a complete epiphyseal closure was achieved with transdermal estradiol treatment . Compared with baseline values , raloxifene treatment led to improved BMD both at the ultradistal forearm and 33 % radius ; the transdermal estradiol treatment resulted in a further slight increase in BMD at the 33 % radius , but not at the ultradistal forearm . The baseline bone biopsy showed elevated bone remodelling in trabecular bone , while the second biopsy following raloxifene treatment revealed a decrease in remodelling .This study shows that the management of aromatase deficiency in the male can not consider raloxifene as a first choice treatment , but should be still based on estrogen replacement treatment since in this patient the completion of bone maturation has only been obtained once estradiol substitution was performed . The present case also demonstrates that raloxifene is able to improve BMD in aromatase-deficient men .

Diseases
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Diseases presenting "deficient men" symptom

aromatase deficiency

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