Literature DB >> 19921279

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

Manon Bache1, Céline Dheu, Bérénice Doray, Hélène Fothergill, Sylvie Soskin, Françoise Paris, Charles Sultan, Michel Fischbach.   

Abstract

The diagnosis of Frasier syndrome is based on the association of male pseudohermaphroditism (as a result of gonadal dysgenesis), with steroid-resistant nephrotic syndrome due to focal and segmental glomerular sclerosis (FSGS), which progresses to end-stage renal failure (ESRF) during adolescence or adulthood. Frasier syndrome results from mutations in the Wilms' tumour suppressor gene WT1, which is responsible for alterations in male genital development and podocyte dysfunction. We describe the case of a 7-year-old girl who was referred to the paediatric emergency department with ESRF. Haemodialysis was started immediately because of severe hypertension and hyperkalaemia. In view of the fact that our patient had a past medical history of pseudohermaphroditism, we suspected that the acute presentation in ESRF may be related to a new diagnosis of Frasier syndrome. Our hypothesis was confirmed on examination of the medical records. There had been no medical follow-up for several years and, in particular, no renal imaging or functional assessment had ever been performed. This lack of surveillance explains why our patient presented with ESRF much earlier in this disease than expected and subsequently had to undergo kidney transplantation at a very young age.

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Year:  2010        PMID: 19921279     DOI: 10.1007/s00467-009-1343-2

Source DB:  PubMed          Journal:  Pediatr Nephrol        ISSN: 0931-041X            Impact factor:   3.714


  23 in total

1.  WT1 splice site mutation in a 46,XX female with minimal-change nephrotic syndrome and Wilms' tumour.

Authors:  Chantal Loirat; Jean Luc André; Jacqueline Champigneulle; Cécile Acquaviva; Dominique Chantereau; Rosine Bourquard; Jacques Elion; Erick Denamur
Journal:  Nephrol Dial Transplant       Date:  2003-04       Impact factor: 5.992

2.  Frasier syndrome: a cause of focal segmental glomerulosclerosis in a 46,XX female.

Authors:  L Demmer; W Primack; V Loik; R Brown; N Therville; K McElreavey
Journal:  J Am Soc Nephrol       Date:  1999-10       Impact factor: 10.121

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.  The same mutation affecting the splicing of WT1 gene is present on Frasier syndrome patients with or without Wilms' tumor.

Authors:  A S Barbosa; C G Hadjiathanasiou; C Theodoridis; A Papathanasiou; A Tar; M Merksz; B Györvári; C Sultan; R Dumas; F Jaubert; P Niaudet; C A Moreira-Filho; C Cotinot; M Fellous
Journal:  Hum Mutat       Date:  1999       Impact factor: 4.878

5.  Further evidence that imbalance of WT1 isoforms may be involved in Denys-Drash syndrome.

Authors:  A König; S Jakubiczka; P Wieacker; H W Schlösser; M Gessler
Journal:  Hum Mol Genet       Date:  1993-11       Impact factor: 6.150

6.  WT-1 is required for early kidney development.

Authors:  J A Kreidberg; H Sariola; J M Loring; M Maeda; J Pelletier; D Housman; R Jaenisch
Journal:  Cell       Date:  1993-08-27       Impact factor: 41.582

7.  Do intronic mutations affecting splicing of WT1 exon 9 cause Frasier syndrome?

Authors:  H Kikuchi; A Takata; Y Akasaka; R Fukuzawa; H Yoneyama; Y Kurosawa; M Honda; Y Kamiyama; J Hata
Journal:  J Med Genet       Date:  1998-01       Impact factor: 6.318

8.  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

9.  Management of Wilms tumors in Drash and Frasier syndromes.

Authors:  F Auber; C Jeanpierre; E Denamur; F Jaubert; G Schleiermacher; C Patte; S Cabrol; G Leverger; C Nihoul-Fékété; S Sarnacki
Journal:  Pediatr Blood Cancer       Date:  2009-01       Impact factor: 3.167

10.  Gonadoblastoma and dysgerminoma associated with XY gonadal dysgenesis in an adolescent with chronic renal failure: a case of Frasier syndrome.

Authors:  Minna M Joki-Erkkilä; Riitta Karikoski; Immo Rantala; Hanna-Liisa Lenko; Tapio Visakorpi; Pentti K Heinonen
Journal:  J Pediatr Adolesc Gynecol       Date:  2002-06       Impact factor: 1.814

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  3 in total

1.  Tumors of bilateral streak gonads in patients with disorders of sex development containing y chromosome material.

Authors:  Fumi Matsumoto; Kenji Shimada; Shinobu Ida
Journal:  Clin Pediatr Endocrinol       Date:  2014-08-06

2.  Systematic Review of Genotype-Phenotype Correlations in Frasier Syndrome.

Authors:  Yurika Tsuji; Tomohiko Yamamura; China Nagano; Tomoko Horinouchi; Nana Sakakibara; Shinya Ishiko; Yuya Aoto; Rini Rossanti; Eri Okada; Eriko Tanaka; Koji Tsugawa; Takayuki Okamoto; Toshihiro Sawai; Yoshinori Araki; Yuko Shima; Koichi Nakanishi; Hiroaki Nagase; Masafumi Matsuo; Kazumoto Iijima; Kandai Nozu
Journal:  Kidney Int Rep       Date:  2021-07-16

3.  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

  3 in total

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