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A Novel PHEX Mutation in X-Linked Hypophosphataemic Rickets With Reduced Expression of NaPi-IIa and NaPi-IIc in the Proximal Tubules: A Case Report.

作者信息

Muto Masahiro, Gohda Tomohito, Takagi Miyuki, Kobayashi Takashi, Kihara Masao, Saeki Harumi, Mori Takayasu, Sohara Eisei, Uchida Shinichi, Yamashiro Yuichiro, Suzuki Yusuke

机构信息

Department of Nephrology, Juntendo University Faculty of Medicine, Tokyo, Japan.

Department of Human Pathology, Faculty of Medicine, Juntendo University, Tokyo, Japan.

出版信息

Nephrology (Carlton). 2025 Aug;30(8):e70104. doi: 10.1111/nep.70104.

Abstract

X-linked hypophosphataemia (XLH, MIM#307800) is an inherited form of rickets resulting from mutations in the phosphate-regulating neutral endopeptidase (PHEX) gene on the X chromosome. These mutations lead to elevated circulating fibroblast growth factor 23 (FGF23), which disrupts phosphate homeostasis and contributes to XLH pathogenesis. We present a sporadic case of a 41-year-old woman diagnosed with rickets in childhood who later developed persistent proteinuria. Kidney biopsy revealed segmental sclerosis with a perihilar lesion in one of 19 glomeruli, along with dilated proximal tubules, reduced expression of the sodium-dependent phosphate transporters (NaPi-IIa and NaPi-IIc) and lysosomal particle accumulation in proximal tubule epithelial cells. Next-generation sequencing identified a novel heterozygous missense mutation in PHEX (c.2179T>A; p.Phe727Ile), which, to our knowledge, has not been previously reported. Detailed kidney biopsy findings in XLH are rare. This case report provides novel insights into XLH pathophysiology, highlighting kidney-specific pathological changes and reinforcing the importance of genetic testing for precise diagnosis and management.

摘要

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