Literature DB >> 17378674

Expanding the clinical spectrum of Frasier syndrome.

Katja Gwin1, Mariana M Cajaiba, Alejandra Caminoa-Lizarralde, Maria L Picazo, Manuel Nistal, Miguel Reyes-Múgica.   

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.

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Year:  2007        PMID: 17378674     DOI: 10.2350/07-01-0209.1

Source DB:  PubMed          Journal:  Pediatr Dev Pathol        ISSN: 1093-5266


  6 in total

1.  Frasier syndrome, a potential cause of end-stage renal failure in childhood.

Authors:  Manon Bache; Céline Dheu; Bérénice Doray; Hélène Fothergill; Sylvie Soskin; Françoise Paris; Charles Sultan; Michel Fischbach
Journal:  Pediatr Nephrol       Date:  2010-03       Impact factor: 3.714

Review 2.  The Genetics of Nephrotic Syndrome.

Authors:  Michelle N Rheault; Rasheed A Gbadegesin
Journal:  J Pediatr Genet       Date:  2015-08-13

Review 3.  Malformation syndromes associated with disorders of sex development.

Authors:  John M Hutson; Sonia R Grover; Michele O'Connell; Samuel D Pennell
Journal:  Nat Rev Endocrinol       Date:  2014-06-10       Impact factor: 43.330

4.  Evolutive study of children with diffuse mesangial sclerosis.

Authors:  Ana Pilar Nso Roca; Antonia Peña Carrión; Marta Benito Gutiérrez; Carmen García Meseguer; Araceli García Pose; Mercedes Navarro
Journal:  Pediatr Nephrol       Date:  2008-12-10       Impact factor: 3.714

5.  Spectrum of Clinical Manifestations in Children With WT1 Mutation: Case Series and Literature Review.

Authors:  Patricia Arroyo-Parejo Drayer; Wacharee Seeherunvong; Chryso P Katsoufis; Marissa J DeFreitas; Tossaporn Seeherunvong; Jayanthi Chandar; Carolyn L Abitbol
Journal:  Front Pediatr       Date:  2022-04-15       Impact factor: 3.418

6.  Clinical characteristics and outcomes of children with WAGR syndrome and Wilms tumor and/or nephroblastomatosis: The 30-year SIOP-RTSG experience.

Authors:  Janna A Hol; Marjolijn C J Jongmans; Hélène Sudour-Bonnange; Gema L Ramírez-Villar; Tanzina Chowdhury; Catherine Rechnitzer; Niklas Pal; Gudrun Schleiermacher; Axel Karow; Roland P Kuiper; Beatriz de Camargo; Simona Avcin; Danka Redzic; Antonio Wachtel; Heidi Segers; Gordan M Vujanic; Harm van Tinteren; Christophe Bergeron; Kathy Pritchard-Jones; Norbert Graf; Marry M van den Heuvel-Eibrink
Journal:  Cancer       Date:  2020-11-04       Impact factor: 6.860

  6 in total

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