Literature DB >> 33392118

Case Report: Denys-Drash Syndrome With WT1 Causative Variant Presenting as Atypical Hemolytic Uremic Syndrome.

Cheng Cheng1, Lizhi Chen1, Sijia Wen1, Zhilang Lin1, Xiaoyun Jiang1.   

Abstract

The WT1 variant is confirmed to be pathogenic for Denys-Drash syndrome (DDS), a rare disorder characterized by early-onset nephrotic syndrome and renal failure, pseudo-hermaphroditism, and a high risk of Wilms' tumor. Several cases of DDS presenting with atypical hemolytic uremic syndrome (aHUS) have been reported. Here we report the case of a 2-year-old child who was diagnosed with WT1 missense variant, associated with DDS and initial presentation of aHUS. Complement factor H autoantibodies were negative. Complement regulatory system-related gene variants were not found, but a de novo heterozygous c.754G>A missense variant in exon 9 of WT1 gene was detected, resulting in a p. Asp252Asn substitution, by next-generation sequencing. The patient was a female morphologically but proved to be a genetic male because of karyotype 46, XY with normally developed female external genitalia. Bilateral nephrectomy and renal transplantation were performed 1 year later, and there was no recurrence of aHUS at 10 months after transplantation.
Copyright © 2020 Cheng, Chen, Wen, Lin and Jiang.

Entities:  

Keywords:  atypical hemolytic uremic syndrome (aHUS); case report [publication type]; denys-drash syndrome (DDS); pseudohermaphroditism; wilms tumor 1 gene (WT1)

Year:  2020        PMID: 33392118      PMCID: PMC7775732          DOI: 10.3389/fped.2020.605889

Source DB:  PubMed          Journal:  Front Pediatr        ISSN: 2296-2360            Impact factor:   3.418


  2 in total

1.  Case Report: A Pathogenic Missense Variant of WT1 Cosegregates With Proteinuria in a Six-Generation Chinese Family With IgA Nephropathy.

Authors:  Qianqian Li; Li Zhu; Sufang Shi; Damin Xu; Jicheng Lv; Hong Zhang
Journal:  Front Med (Lausanne)       Date:  2022-01-31

Review 2.  WT1 complete gonadal dysgenesis with membranoproliferative glomerulonephritis: case series and literature review.

Authors:  Erin Anderson; Melanie Aldridge; Ross Turner; James Harraway; Sam McManus; Anna Stewart; Peter Borzi; Peter Trnka; John Burke; David Coman
Journal:  Pediatr Nephrol       Date:  2022-02-24       Impact factor: 3.651

  2 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.