Literature DB >> 14977287

Twenty-eight-year-old female with primary amenorrhea and chronic renal failure: a case of Frasier syndrome?

Raphael Onyemekeihia1, Efosa Oviasu.   

Abstract

Frasier syndrome is a very rare developmental disorder of autosomal recessive inheritance. It is characterized by male hermaphroditism, primary amenorrhea, chronic renal failure (CRF), and a number of other abnormalities. A 28-year-old Nigerian female who was considered as a possible case of Frasier syndrome first presented to us in July 2002 with primary amenorrhea, congenital bilateral absence of middle toes, elevated blood pressure, and the uremic syndrome. The management of the case was mainly conservative, including blood pressure control with appropriate antihypertensives. The problems inherent in this index case are discussed while proffering appropriate management approach in a near-ideal situation, which unfortunately is nonexistent in our local environment. The presentation of this case is informed by the need to create awareness about this rare syndrome being a possible cause of CRF in some of our patients.

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Year:  2004        PMID: 14977287      PMCID: PMC2594967     

Source DB:  PubMed          Journal:  J Natl Med Assoc        ISSN: 0027-9684            Impact factor:   1.798


  9 in total

1.  Clinical spectrum of Denys-Drash and Frasier syndrome.

Authors:  S J McTaggart; E Algar; C W Chow; H R Powell; C L Jones
Journal:  Pediatr Nephrol       Date:  2001-04       Impact factor: 3.714

2.  Case report: Teenage girl with proteinuria and amenorrhea.

Authors:  J Hausladen; E Granahan; D Bockenhauer
Journal:  Curr Opin Pediatr       Date:  2001-04       Impact factor: 2.856

Review 3.  Chronic renal failure and XY gonadal dysgenesis: "Frasier" syndrome--a commentary on reported cases.

Authors:  A V Moorthy; R W Chesney; M Lubinsky
Journal:  Am J Med Genet Suppl       Date:  1987

4.  A girl with bilateral ovarian tumours: Frasier syndrome.

Authors:  Hironobu Shimoyama; Mitsuru Nakajima; Hiroyuki Naka; Yong-Dong Park; Kensuke Hori; Hajime Morikawa; Akira Yoshioka
Journal:  Eur J Pediatr       Date:  2002-02       Impact factor: 3.183

5.  Surgical management and genotype/phenotype correlations in WT1 gene-related diseases (Drash, Frasier syndromes).

Authors:  F Auber; S Lortat-Jacob; S Sarnacki; F Jaubert; R Salomon; E Thibaud; C Jeanpierre; C Nihoul-Fékété
Journal:  J Pediatr Surg       Date:  2003-01       Impact factor: 2.545

6.  A mutant form of the Wilms' tumor suppressor gene WT1 observed in Denys-Drash syndrome interferes with glomerular capillary development.

Authors:  Thomas A Natoli; Jing Liu; Vera Eremina; Karen Hodgens; Cong Li; Yuki Hamano; Peter Mundel; Raghu Kalluri; Jeffrey H Miner; Susan E Quaggin; Jordan A Kreidberg
Journal:  J Am Soc Nephrol       Date:  2002-08       Impact factor: 10.121

Review 7.  An unusual phenotype of Frasier syndrome due to IVS9 +4C>T mutation in the WT1 gene: predominantly male ambiguous genitalia and absence of gonadal dysgenesis.

Authors:  Karla F S Melo; Regina M Martin; Elaine M F Costa; Filomena M Carvalho; Alexander A Jorge; Ivo J P Arnhold; Berenice B Mendonca
Journal:  J Clin Endocrinol Metab       Date:  2002-06       Impact factor: 5.958

8.  Gonadal mosaicism of Frasier syndrome in 3 Chinese siblings with donor splice site mutation of Wilms' tumour gene.

Authors:  Wai Leung Chak; Ka Fai To; Yuk Lun Cheng; Kan Ming Tsui; Kwok Lai Lo; Hung Man Tong; Fernand Mac-Moune Lai; Francis Kin Ming Wong; Koon Shing Choi; Ka Foon Chau; Chun Sang Li
Journal:  Nephron       Date:  2002-07       Impact factor: 2.847

9.  Prenatal diagnosis and intrafamilial clinical heterogeneity of Fraser syndrome.

Authors:  Thierry Rousseau; Nicole Laurent; Christel Thauvin-Robinet; Stéphanie Lionnais; Christine Durand; Laurence Faivre; Paul Sagot
Journal:  Prenat Diagn       Date:  2002-08       Impact factor: 3.050

  9 in total

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