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Expanding the clinical spectrum of Frasier syndrome.
- Source :
-
Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society [Pediatr Dev Pathol] 2008 Mar-Apr; Vol. 11 (2), pp. 122-7. Date of Electronic Publication: 2007 Mar 22. - Publication Year :
- 2008
-
Abstract
- Frasier syndrome is an uncommon genetic disorder featuring progressive glomerulopathy, male pseudohermaphroditism, and gonadal dysgenesis with increased risk of gonadoblastoma and malignant germ cell tumors. It is caused by mutations in the donor splice site in intron 9 of the WT1 gene. However, because of its rarity there is limited literature available on the precise spectrum and recommended treatment modalities of this syndrome. We present the clinicopathological findings in 4 patients: 3 phenotypically female adolescents presenting with proteinuria and primary amenorrhea and a 6-month-old baby girl presenting with nephrotic syndrome in whom this very unusual case of early onset was confirmed by molecular studies. The significance of early recognition of Frasier syndrome and its differentiation from Denys-Drash syndrome is reviewed and discussed. Our observation of a case presenting with early clinical manifestations, in contrast with the classical presentation in adolescence, justifies the expansion of the clinical spectrum of Frasier syndrome and contributes to the understanding and appropriate clinical management of these patients.
- Subjects :
- Adolescent
Amenorrhea genetics
Amenorrhea pathology
Chromosomes, Human, X
Chromosomes, Human, Y
Denys-Drash Syndrome diagnosis
Diagnosis, Differential
Dysgerminoma genetics
Dysgerminoma secondary
Dysgerminoma surgery
Early Diagnosis
Female
Frasier Syndrome physiopathology
Genes, Wilms Tumor
Glomerulosclerosis, Focal Segmental genetics
Glomerulosclerosis, Focal Segmental pathology
Gonadoblastoma genetics
Gonadoblastoma pathology
Gonadoblastoma surgery
Humans
Infant
Kidney Failure, Chronic genetics
Kidney Failure, Chronic pathology
Mutation
Nephrotic Syndrome genetics
Nephrotic Syndrome pathology
Ovarian Neoplasms genetics
Ovarian Neoplasms pathology
Ovarian Neoplasms surgery
Phenotype
Proteinuria genetics
Proteinuria pathology
Frasier Syndrome genetics
Frasier Syndrome pathology
Subjects
Details
- Language :
- English
- ISSN :
- 1093-5266
- Volume :
- 11
- Issue :
- 2
- Database :
- MEDLINE
- Journal :
- Pediatric and developmental pathology : the official journal of the Society for Pediatric Pathology and the Paediatric Pathology Society
- Publication Type :
- Academic Journal
- Accession number :
- 17378674
- Full Text :
- https://doi.org/10.2350/07-01-0209.1