A Perihilar Variant of Focal Segmental Glomerulosclerosis Due to De novo Branchio-oto-renal Syndrome

Intern Med. 2022 Jul 1;61(13):2033-2038. doi: 10.2169/internalmedicine.8508-21. Epub 2021 Dec 4.

Abstract

Branchio-oto-renal syndrome is an autosomal dominant disorder characterized by branchial anomalies, hearing loss, and renal urinary tract malformations. We herein report a 32-year-old Japanese man with a right preauricular pit, bilateral mixed hearing loss, and malposition of the right kidney who presented with proteinuria. The findings of a left kidney biopsy were compatible with a perihilar variant of secondary focal segmental glomerular sclerosis. A trio exome analysis conducted among the patient and his parents failed to identify the causal gene variant, despite a sporadic pattern. His kidney function remained stable for 11 years with an angiotensin II receptor blocker.

Keywords: branchio-oto-renal; de novo; obesity-related nephropathy; proteinuria; trio analysis.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Branchio-Oto-Renal Syndrome* / complications
  • Branchio-Oto-Renal Syndrome* / genetics
  • Deafness* / complications
  • Glomerulosclerosis, Focal Segmental* / complications
  • Glomerulosclerosis, Focal Segmental* / genetics
  • Hearing Loss*
  • Humans
  • Kidney
  • Male