{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,10]],"date-time":"2026-09-10T15:52:30Z","timestamp":1789055550706,"version":"build-2803163510"},"reference-count":50,"publisher":"The Company of Biologists","issue":"1","license":[{"start":{"date-parts":[[2022,2,4]],"date-time":"2022-02-04T00:00:00Z","timestamp":1643932800000},"content-version":"vor","delay-in-days":20,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100005879","name":"National Centre for Biological Sciences","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100005879","id-type":"DOI","asserted-by":"crossref"}]},{"DOI":"10.13039\/501100001502","name":"Department of Atomic Energy, Government of India","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100001502","id-type":"DOI","asserted-by":"crossref"}]},{"name":"the Department of Biotechnology, Government of India","award":["BT\/PR17316\/MED\/31\/326\/2015"],"award-info":[{"award-number":["BT\/PR17316\/MED\/31\/326\/2015"]}]},{"name":"Pratiksha Trust"},{"name":"Wellcome-DBT India Alliance","award":["IA\/S\/14\/2\/501540"],"award-info":[{"award-number":["IA\/S\/14\/2\/501540"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,1,15]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>Human brain development is a complex process where multiple cellular and developmental events are coordinated to generate normal structure and function. Alteration in any of these events can impact brain development, manifesting clinically as neurodevelopmental disorders. Human genetic disorders of lipid metabolism often present with features of altered brain function. Lowe syndrome (LS) is an X-linked recessive disease with features of altered brain function. LS results from mutations in OCRL1, which encodes a phosphoinositide 5-phosphatase enzyme. However, the cellular mechanisms by which loss of OCRL1 leads to brain defects remain unknown. Human brain development involves several cellular and developmental features not conserved in other species and understanding such mechanisms remains a challenge. Rodent models of LS have been generated but failed to recapitulate features of the human disease. Here we describe the generation of human stem cell lines from LS patients. Further, we present biochemical characterization of lipid metabolism in patient cell lines and demonstrate their use as a \u2018disease-in-a-dish\u2019 model for understanding the mechanism by which loss of OCRL1 leads to altered cellular and physiological brain development.<\/jats:p>\n                  <jats:p>This article has an associated First Person interview with the first author of the paper.<\/jats:p>","DOI":"10.1242\/bio.059066","type":"journal-article","created":{"date-parts":[[2022,1,13]],"date-time":"2022-01-13T06:42:25Z","timestamp":1642056145000},"source":"Crossref","is-referenced-by-count":12,"title":["A human stem cell resource to decipher the biochemical and cellular basis of neurodevelopmental defects in Lowe syndrome"],"prefix":"10.1242","volume":"11","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-8346-3689","authenticated-orcid":false,"given":"Bilal M.","family":"Akhtar","sequence":"first","affiliation":[{"name":"Cellular Organization and Signalling, National Centre for Biological Sciences, TIFR-GKVK Campus, Bellary Road, Bengaluru 560065, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Priyanka","family":"Bhatia","sequence":"additional","affiliation":[{"name":"Brain Development and Disease Mechanisms, Institute for Stem Cell Science and Regenerative Medicine, Bengaluru 560065, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shubhra","family":"Acharya","sequence":"additional","affiliation":[{"name":"Brain Development and Disease Mechanisms, Institute for Stem Cell Science and Regenerative Medicine, Bengaluru 560065, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sanjeev","family":"Sharma","sequence":"additional","affiliation":[{"name":"Cellular Organization and Signalling, National Centre for Biological Sciences, TIFR-GKVK Campus, Bellary Road, Bengaluru 560065, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yojet","family":"Sharma","sequence":"additional","affiliation":[{"name":"Cellular Organization and Signalling, National Centre for Biological Sciences, TIFR-GKVK Campus, Bellary Road, Bengaluru 560065, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aswathy","family":"Bhuvanendran Nair Suseela Devi","sequence":"additional","affiliation":[{"name":"Brain Development and Disease Mechanisms, Institute for Stem Cell Science and Regenerative Medicine, Bengaluru 560065, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2005-4714","authenticated-orcid":false,"given":"Kavina","family":"Ganapathy","sequence":"additional","affiliation":[{"name":"Cellular Organization and Signalling, National Centre for Biological Sciences, TIFR-GKVK Campus, Bellary Road, Bengaluru 560065, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4338-0325","authenticated-orcid":false,"given":"Anil","family":"Vasudevan","sequence":"additional","affiliation":[{"name":"Department of Pediatric Nephrology, St. John's Medical College Hospital, Bengaluru 560034, India"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3578-6413","authenticated-orcid":false,"given":"Padinjat","family":"Raghu","sequence":"additional","affiliation":[{"name":"Cellular Organization and Signalling, National Centre for Biological Sciences, TIFR-GKVK Campus, Bellary Road, Bengaluru 560065, India"},{"name":"Brain Development and Disease Mechanisms, Institute for Stem Cell Science and Regenerative Medicine, Bengaluru 560065, India"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2022,2,4]]},"reference":[{"key":"2022020410011278500_BIO059066C1","first-page":"2021.06.22.449382","article-title":"Genomic sequencing of Lowe syndrome trios reveal a mechanism for the heterogeneity of neurodevelopmental phenotypes","author":"Ahmed","year":"2021","journal-title":"bioRxiv"},{"key":"2022020410011278500_BIO059066C2","doi-asserted-by":"publisher","first-page":"239","DOI":"10.1038\/358239a0","article-title":"The Lowe's oculocerebrorenal syndrome gene encodes a protein highly homologous to inositol polyphosphate-5-phosphatase","volume":"358","author":"Attree","year":"1992","journal-title":"Nature"},{"key":"2022020410011278500_BIO059066C3","doi-asserted-by":"publisher","first-page":"647","DOI":"10.1016\/j.stemcr.2015.08.015","article-title":"cGMP-manufactured human induced pluripotent stem cells are available for pre-clinical and clinical 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