{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,17]],"date-time":"2026-05-17T04:21:30Z","timestamp":1778991690109,"version":"3.51.4"},"reference-count":10,"publisher":"The Endocrine Society","issue":"5","license":[{"start":{"date-parts":[[2024,5,3]],"date-time":"2024-05-03T00:00:00Z","timestamp":1714694400000},"content-version":"vor","delay-in-days":10,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,4,23]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>In infants, hypercalcemia from elevated parathyroid hormone-related protein (PTHrP) is rare, often signaling neoplasm or renal or urinary anomalies. We report an infant who presented with failure to thrive and hypercalcemia at 10 months old, with initial evaluation showing elevated PTHrP of unclear etiology with imaging negative for neoplasm and no structural anomalies of the kidneys or ureters on ultrasound. Within 6 months of presentation, the patient developed nephrotic syndrome and by 2 years had progressed to end-stage kidney disease, necessitating kidney transplantation. Genetic testing was inconclusive but suggested congenital nephrotic syndrome. While reports of hypercalcemia secondary to elevated PTHrP exist in children with known structural renal anomalies, this is the first to demonstrate hypercalcemia and PTHrP elevation before detection of renal abnormalities. Experimental models have suggested a role for increased PTHrP expression in renal cells following acute kidney injury from nephrotic syndrome, and clinically detectable PTHrP levels may indicate progression of renal injury. We suggest monitoring of renal function for early detection of nephrotic syndrome in infants and children with elevated PTHrP who otherwise lack anatomical renal anomalies or detectable malignancies.<\/jats:p>","DOI":"10.1210\/jcemcr\/luae074","type":"journal-article","created":{"date-parts":[[2024,5,3]],"date-time":"2024-05-03T10:08:44Z","timestamp":1714730924000},"source":"Crossref","is-referenced-by-count":1,"title":["Hypercalcemia Secondary to Elevated PTHrP in an Infant Followed by Progression to Nephrotic Syndrome"],"prefix":"10.1210","volume":"2","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7098-914X","authenticated-orcid":false,"given":"Alex F","family":"Gimeno","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Vanderbilt University School of Medicine , Nashville, TN 37232 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8766-551X","authenticated-orcid":false,"given":"Tracy E","family":"Hunley","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Pediatric Nephrology, Vanderbilt University Medical Center , Nashville, TN 37232 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9015-4737","authenticated-orcid":false,"given":"Jennifer C","family":"Kelley","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Pediatric Endocrinology and Diabetes, Vanderbilt University Medical Center , Nashville, TN 37232 , USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"80","published-online":{"date-parts":[[2024,5,3]]},"reference":[{"key":"2024052407042684800_luae074-B1","first-page":"728","volume-title":"Sperling Pediatric Endocrinology","author":"Root","year":"2021","edition":"5th"},{"issue":"11","key":"2024052407042684800_luae074-B2","doi-asserted-by":"crossref","first-page":"2157","DOI":"10.1002\/jbmr.3296","article-title":"Hypercalcemic disorders in children","volume":"32","author":"Stokes","year":"2017","journal-title":"J Bone Min Res"},{"issue":"4","key":"2024052407042684800_luae074-B3","first-page":"510","article-title":"Physiological roles of parathyroid hormone-related protein","volume":"90","author":"Lai","year":"2019","journal-title":"Acta Bio Medica: Atenei Parmensis"},{"issue":"1","key":"2024052407042684800_luae074-B4","doi-asserted-by":"crossref","first-page":"52","DOI":"10.1186\/s40697-015-0052-y","article-title":"PTHrP-related hypercalcaemia in infancy and congenital anomalies of the kidney and urinary tract (CAKUT)","volume":"2","author":"Kodous","year":"2015","journal-title":"Can J Kidney Health Dis"},{"issue":"3","key":"2024052407042684800_luae074-B5","doi-asserted-by":"crossref","first-page":"179","DOI":"10.1159\/000046244","article-title":"Parathyroid hormone-related protein as a renal regulating factor","volume":"21","author":"Esbrit","year":"2001","journal-title":"Am J Nephrol"},{"key":"2024052407042684800_luae074-B6","first-page":"1375","article-title":"Pediatric reference ranges for glomerular filtration rate determined by a single injection of Tc-99m DTPA","volume":"50","author":"Du","year":"2009","journal-title":"J Nucl Med"},{"issue":"5","key":"2024052407042684800_luae074-B7","doi-asserted-by":"crossref","first-page":"777","DOI":"10.1038\/ki.2012.469","article-title":"Parathyroid hormone\u2013related protein and regulation of cell survival in the kidney","volume":"83","author":"Kramann","year":"2013","journal-title":"Kidney Int"},{"issue":"4","key":"2024052407042684800_luae074-B8","doi-asserted-by":"crossref","first-page":"863","DOI":"10.1016\/j.ajpath.2017.12.012","article-title":"Parathyroid hormone\u2013related protein contributes to early healing of Habu snake venom\u2013induced glomerulonephritis in mice","volume":"188","author":"Hochane","year":"2018","journal-title":"Am J Pathol"},{"issue":"4","key":"2024052407042684800_luae074-B9","doi-asserted-by":"crossref","first-page":"457","DOI":"10.1038\/ng.3512","article-title":"Mutations in nuclear pore genes NUP93, NUP205, and XPO5 cause steroid resistant nephrotic syndrome","volume":"48","author":"Braun","year":"2016","journal-title":"Nat Genet"},{"issue":"7","key":"2024052407042684800_luae074-B10","doi-asserted-by":"crossref","first-page":"2538","DOI":"10.1073\/pnas.0707730105","article-title":"Mutations in FN1 cause glomerulopathy with fibronectin deposits","volume":"105","author":"Castelletti","year":"2008","journal-title":"Proc Natl Acad Sci U S A"}],"container-title":["JCEM Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jcemcr\/article-pdf\/2\/5\/luae074\/57595438\/luae074.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jcemcr\/article-pdf\/2\/5\/luae074\/57595438\/luae074.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,5,24]],"date-time":"2024-05-24T07:40:26Z","timestamp":1716536426000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jcemcr\/article\/doi\/10.1210\/jcemcr\/luae074\/7663639"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,4,23]]},"references-count":10,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2024,4,23]]}},"URL":"https:\/\/doi.org\/10.1210\/jcemcr\/luae074","relation":{},"ISSN":["2755-1520"],"issn-type":[{"value":"2755-1520","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2024,5]]},"published":{"date-parts":[[2024,4,23]]},"article-number":"luae074"}}