Document Detail


Prophylactic bilateral salpingo-oopherectomy in a 17-year-old with Frasier syndrome reveals gonadoblastoma and seminoma: a case report.
MedLine Citation:
PMID:  17101338     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Mutations in the Wilms' tumor gene are present in children with Frasier syndrome, Denys-Drash syndrome, WAGR syndrome, and some cases of Wilms' tumor. The Wilms' tumor gene product, WT1, is necessary for normal urogenital development. Frasier syndrome is an association between focal segmental glomerulosclerosis, beginning in the second and third decade, male to female sex reversal, and dysgenetic gonads. We report a case of Frasier syndrome in a 17-year-old adolescent girl with renal failure, kidney transplant, and dysgenetic gonads, with development of gonadoblastoma and dysgerminoma (seminoma). The diagnosis of Frasier syndrome was based on nephrotic syndrome with diffuse mesangial sclerosis leading to chronic renal failure, dysgenetic gonads, 46 XY karyotype in a phenotypic female, and a mutation in the Wilms' tumor gene. Prophylactic laparoscopic bilateral salpingo-oopherectomy revealed gonadoblastoma and seminoma in opposite atrophic ovaries as well as a hypoplastic uterus. Early prophylactic resection of dysgenetic gonads is indicated in children with Frasier syndrome to prevent the development of germ cell malignancy.
Authors:
Joseph D Love; Steven D DeMartini; Christopher P Coppola
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Publication Detail:
Type:  Case Reports; Journal Article    
Journal Detail:
Title:  Journal of pediatric surgery     Volume:  41     ISSN:  1531-5037     ISO Abbreviation:  J. Pediatr. Surg.     Publication Date:  2006 Nov 
Date Detail:
Created Date:  2006-11-14     Completed Date:  2006-12-05     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  0052631     Medline TA:  J Pediatr Surg     Country:  United States    
Other Details:
Languages:  eng     Pagination:  e1-4     Citation Subset:  IM    
Affiliation:
Department of Surgery, Wilford Hall Medical Center, San Antonio, Lackland AFB, TX 78236, USA.
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MeSH Terms
Descriptor/Qualifier:
Adolescent
Fallopian Tubes / surgery
Female
Frasier Syndrome / complications*
Gonadoblastoma / diagnosis*,  etiology,  surgery*
Gynecologic Surgical Procedures
Humans
Ovariectomy
Seminoma / diagnosis*,  etiology,  surgery*

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