| Literature DB >> 33392118 |
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.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