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This pathway is dependent on multi-ligand receptors: megalin and cubilin. Abnormalities in genes associated with endocytosis in the proximal tubule can lead to tubular proteinuria, where the urine contains albumin and low-molecular\u2013weight proteins. Dent disease is a hereditary X-linked disorder characterized by low-molecular\u2013weight proteinuria, hypercalciuria, nephrocalcinosis, nephrolithiasis, and progressive kidney dysfunction, often leading to CKD stage 5. <jats:italic>CLCN5<\/jats:italic> is the gene responsible for Dent disease-1 and encodes the voltage-gated chloride channel ClC-5. Meanwhile, <jats:italic>OCRL<\/jats:italic> is the causative gene of Dent disease-2 and encodes phosphatidylinositol 4,5-bisphosphate 5-phosphatase, and its variants are also associated with Lowe syndrome. ClC-5 and OCRL are essential to the endocytic machinery, and their loss affects endosomal acidification and trafficking, resulting in disruption of megalin and cubilin recycling. <jats:italic>CUBN<\/jats:italic>, which encodes cubilin, was originally identified as the causative gene of Imerslund\u2013Gr\u00e4sbeck syndrome, a disorder of megaloblastic anemia associated with proteinuria. However, recently, a biallelic C-terminal variant of <jats:italic>CUBN<\/jats:italic> was shown to be responsible for isolated proteinuria without kidney dysfunction. This proteinuria is recognized as a new disease concept called chronic benign proteinuria (proteinuria, chronic benign: PROCHOB), which contradicts the common belief that proteinuria is harmful and ultimately leads to kidney damage. This article deepens the understanding of genetic tubular proteinuria and its origins, focusing on the role of megalin- and cubilin-mediated endocytosis in the proximal tubule.<\/jats:p>\n          <jats:p>\n            <jats:bold>Graphical abstract<\/jats:bold>\n          <\/jats:p>","DOI":"10.1007\/s00467-025-06745-x","type":"journal-article","created":{"date-parts":[[2025,4,1]],"date-time":"2025-04-01T07:56:13Z","timestamp":1743494173000},"page":"3367-3377","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Tubular proteinuria due to hereditary endocytic receptor disorder of the proximal tubule: Dent disease and chronic benign proteinuria"],"prefix":"10.1007","volume":"40","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8387-8008","authenticated-orcid":false,"given":"Nana","family":"Sakakibara","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kandai","family":"Nozu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2025,3,31]]},"reference":[{"key":"6745_CR1","doi-asserted-by":"publisher","first-page":"F1258","DOI":"10.1152\/ajprenal.90638.2008","volume":"296","author":"GA Tanner","year":"2009","unstructured":"Tanner GA (2009) Glomerular sieving coefficient of serum albumin in the rat: a two-photon microscopy study. 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Kandai Nozu has a patent for developing exon skipping therapy for Alport syndrome patients.Kandai Nozu also receives lecture fees from Ono Pharmaceutical Co., Ltd., Astellas Pharma Inc., Novo Nordisk Pharmaceuticals Ltd., Alexion Pharmaceuticals, Inc., Sumitomo Pharma Co., Ltd., Sanofi S.A., Otsuka Pharmaceutical Co., Ltd., Daiichi Sankyo Company, Limited, and Miyarisan Pharmaceutical Co., Ltd. Kandai Nozu receives speaker\u2019s bureaus from Sumitomo Pharma Co., Ltd., Chugai Pharmaceutical Co., Ltd., JCR Pharmaceuticals Co., Ltd., Sanofi S.A., Zenyaku Kogyo Co., Ltd., and Kyowa Kirin Co., Ltd. Finally, Kandai Nozu receives grants from Toa Eiyo Ltd., Zenyaku Kogyo Co., Ltd., and Torii Pharmaceutical Co., Ltd. Nana Sakakibara has nothing to disclose.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing Interests"}}]}}