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Background <p>Cyanotic nephropathy (CN) is a known complication of cyanotic congenital heart disease (CCHD). However, many aspects of its pathophysiology remain unclear.</p> Case presentation <p>We report the case of a 29-year-old male with a history of tetralogy of Fallot. Renal biopsy revealed glomerular hypertrophy and focal segmental glomerulosclerosis. Electron microscopy revealed extensive endothelial cell damage. To investigate the etiology of endothelial cell damage, PAL-E staining was conducted, revealing staining along the glomerular capillary wall.</p> Conclusion <p>This is the first report of PAL-E staining in CN, suggesting potential overexpression of PV-1. The association of PV-1 expression with endothelial cell damage indicates its role in the pathogenesis of CN.</p>

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Overexpression of plasmalemmal vesicle-associated protein-1 in patient with cyanotic nephropathy: a case report

  • 期刊论文

  • 出版模式：

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  • 发表日期：

    2025年3月3日

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  • Yusuke Ushio,
  • So Hirata,
  • Shun Manabe,
  • Mayuko Suyama,
  • Ayano Tanaka,
  • Momoko Seki,
  • Haruka Kato,
  • Kana Nomura,
  • Anna Nakai,
  • Hitoko Sumori,
  • Yuki Kawaguchi,
  • Shizuka Kobayashi,
  • Shiho Makabe,
  • Hiroshi Kataoka,
  • Naoko Itoh,
  • Sekiko Taneda,
  • Kazuho Honda,
  • Junichi Hoshino

摘要

Background

Cyanotic nephropathy (CN) is a known complication of cyanotic congenital heart disease (CCHD). However, many aspects of its pathophysiology remain unclear.

Case presentation

We report the case of a 29-year-old male with a history of tetralogy of Fallot. Renal biopsy revealed glomerular hypertrophy and focal segmental glomerulosclerosis. Electron microscopy revealed extensive endothelial cell damage. To investigate the etiology of endothelial cell damage, PAL-E staining was conducted, revealing staining along the glomerular capillary wall.

Conclusion

This is the first report of PAL-E staining in CN, suggesting potential overexpression of PV-1. The association of PV-1 expression with endothelial cell damage indicates its role in the pathogenesis of CN.