{"title":"在x连锁的低磷佝偻病中,一种新的PHEX突变与近端小管中NaPi-IIa和NaPi-IIc的表达减少:一个病例报告。","authors":"Masahiro Muto, Tomohito Gohda, Miyuki Takagi, Takashi Kobayashi, Masao Kihara, Harumi Saeki, Takayasu Mori, Eisei Sohara, Shinichi Uchida, Yuichiro Yamashiro, Yusuke Suzuki","doi":"10.1111/nep.70104","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":520716,"journal":{"name":"Nephrology (Carlton, Vic.)","volume":"30 8","pages":"e70104"},"PeriodicalIF":1.9000,"publicationDate":"2025-08-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"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.\",\"authors\":\"Masahiro Muto, Tomohito Gohda, Miyuki Takagi, Takashi Kobayashi, Masao Kihara, Harumi Saeki, Takayasu Mori, Eisei Sohara, Shinichi Uchida, Yuichiro Yamashiro, Yusuke Suzuki\",\"doi\":\"10.1111/nep.70104\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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.</p>\",\"PeriodicalId\":520716,\"journal\":{\"name\":\"Nephrology (Carlton, Vic.)\",\"volume\":\"30 8\",\"pages\":\"e70104\"},\"PeriodicalIF\":1.9000,\"publicationDate\":\"2025-08-01\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Nephrology (Carlton, Vic.)\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1111/nep.70104\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Nephrology (Carlton, Vic.)","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1111/nep.70104","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"","JCRName":"","Score":null,"Total":0}
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.
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.