Document Detail


Tamoxifen treatment in a patient with blepharophimosis-ptosis-epicanthus inversus syndrome and peripheral precocious puberty.
MedLine Citation:
PMID:  17451085     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Patients with blepharophimosis-ptosis-epicanthus inversus syndrome (BPES) can be classified as types 1 or 2, according to the presence or not of ovarian failure. We report a 5 year-old girl with BPES and large multicystic ovaries who developed peripheral precocious puberty with thelarche (Tanner stage III) and pubarche (Tanner stage II). Pelvic ultrasound revealed pubertal uterus and enlarged multicystic ovaries. Fibrous dysplasia and McCune-Albright syndrome were ruled out. Treatment with an estrogen antagonist was started (tamoxifen, 10 mg/day), achieving regression of thelarche. Tamoxifen treatment was stopped at 10-(7/12) years, and growth velocity and skeletal maturation rate returned to normal. No treatment-associated adverse effects were observed.
Authors:
Cristina González-Niño; Diego Yeste; Antonio Carrascosa
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of pediatric endocrinology & metabolism : JPEM     Volume:  20     ISSN:  0334-018X     ISO Abbreviation:  J. Pediatr. Endocrinol. Metab.     Publication Date:  2007 Mar 
Date Detail:
Created Date:  2007-04-24     Completed Date:  2007-05-16     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  9508900     Medline TA:  J Pediatr Endocrinol Metab     Country:  England    
Other Details:
Languages:  eng     Pagination:  445-8     Citation Subset:  IM    
Affiliation:
Pediatric Endocrinology Department, Vall d'Hebron Hospital, Barcelona, Spain.
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MeSH Terms
Descriptor/Qualifier:
Blepharophimosis / complications*
Blepharoptosis / complications*
Child, Preschool
Estrogen Antagonists / therapeutic use*
Eyelids / abnormalities
Female
Humans
Magnetic Resonance Imaging
Ovarian Cysts / complications,  drug therapy*,  pathology
Puberty, Precocious / complications,  drug therapy*,  pathology
Syndrome
Tamoxifen / therapeutic use*
Chemical
Reg. No./Substance:
0/Estrogen Antagonists; 10540-29-1/Tamoxifen

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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