{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,19]],"date-time":"2026-08-19T03:32:57Z","timestamp":1787110377848,"version":"build-2736575974"},"reference-count":15,"publisher":"The Endocrine Society","issue":"4","license":[{"start":{"date-parts":[[2024,4,23]],"date-time":"2024-04-23T00:00:00Z","timestamp":1713830400000},"content-version":"vor","delay-in-days":32,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,3,22]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Hypoparathyroidism is the most common endocrinological feature in children with Sanjad-Sakati syndrome. Treatment includes active vitamin D and calcium supplementation. Here, we report a case of a newborn with Sanjad-Sakati syndrome who had severe hypocalcemia since birth who responded to PTH subcutaneous pump infusion. The child was born at 35 weeks with hypocalcemia since the first day of life. The standard medical treatment proved ineffective for the newborn, necessitating the administration of unusually high doses of oral and IV calcium and vitamin D analogue for a 2 months. As a result, intermittent subcutaneous injections of PTH were commenced, resulting in an initial improvement in calcium levels, although this proved to be short-lived. Subsequently, a switch to continuous infusion via a Medtronic Vio pump was made, which unfortunately resulted in iatrogenic hypercalcemia, requiring management of hypercalcemia. Later, calcium carbonate and alfacalcidol were resumed at a lower dosage and continued to have average requirements for patients with hypoparathyroidism. PTH subcutaneous infusion can be highly effective in refractory hypocalcemia cases and can significantly impact the treatment course and facilitate hospital discharge as seen in our case. Careful dosage and monitoring are required to avoid iatrogenic hypercalcemia.<\/jats:p>","DOI":"10.1210\/jcemcr\/luae059","type":"journal-article","created":{"date-parts":[[2024,4,23]],"date-time":"2024-04-23T05:58:15Z","timestamp":1713851895000},"source":"Crossref","is-referenced-by-count":6,"title":["Recombinant PTH Infusion in a Child With Sanjad-Sakati Syndrome Refractory to Conventional Therapy"],"prefix":"10.1210","volume":"2","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7563-9755","authenticated-orcid":false,"given":"Ibrahim","family":"Bali","sequence":"first","affiliation":[{"name":"Division of Pediatric Endocrinology, Department of Pediatrics, College of Medicine, King Saud University , Riyadh, Saudi Arabia, 11421- P. O. Box 800"},{"name":"Pediatric Endocrinology Unit, Department of Pediatrics, King Salman Bin Abdulaziz Medical City , Madina 42319 , Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5304-3528","authenticated-orcid":false,"given":"Reem","family":"Al Khalifah","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology, Department of Pediatrics, College of Medicine, King Saud University , Riyadh, Saudi Arabia, 11421- P. O. Box 800"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2024,4,23]]},"reference":[{"issue":"2","key":"2024042305580277300_luae059-B1","doi-asserted-by":"crossref","first-page":"193","DOI":"10.1136\/adc.66.2.193","article-title":"A new syndrome of congenital hypoparathyroidism, severe growth failure, and dysmorphic features","volume":"66","author":"Sanjad","year":"1991","journal-title":"Arch Dis Child"},{"issue":"1","key":"2024042305580277300_luae059-B2","doi-asserted-by":"crossref","first-page":"63","DOI":"10.1136\/jmg.37.1.63","article-title":"Confirmation of the assignment of the Sanjad-Sakati (congenital hypoparathyroidism) syndrome (OMIM 241410) locus to chromosome lq42-43","volume":"37","author":"Kelly","year":"2000","journal-title":"J Med Genet"},{"issue":"2","key":"2024042305580277300_luae059-B3","doi-asserted-by":"crossref","first-page":"145","DOI":"10.1136\/jmg.37.2.145","article-title":"Hypoparathyroidism, retarded growth and development, and dysmorphism or Sanjad-Sakati syndrome: an Arab disease reminiscent of Kenny-Caffey syndrome","volume":"37","author":"Teebi","year":"2000","journal-title":"J Med Genet"},{"issue":"2","key":"2024042305580277300_luae059-B4","doi-asserted-by":"crossref","first-page":"139","DOI":"10.1001\/archpedi.1986.02140160057032","article-title":"Hypercalciuria associated with long-term administration of calcitriol (1,25-dihydroxyvitamin D3). Action of hydrochlorothiazide","volume":"140","author":"Santos","year":"1986","journal-title":"Am J Dis Child"},{"issue":"5","key":"2024042305580277300_luae059-B5","first-page":"273","article-title":"Nephrocalcinosis in children and adolescents: sonographic evaluation during long-term treatment with 1,25-dihydroxycholecalciferol","volume":"9","author":"Weber","year":"1988","journal-title":"Child Nephrol Urol"},{"issue":"3","key":"2024042305580277300_luae059-B6","first-page":"e23032","article-title":"A staghorn calcium phosphate stone in a child with sanjad-sakati syndrome: an iatrogenic manifestation?","volume":"14","author":"Alomar","year":"2022","journal-title":"Cureus"},{"issue":"6","key":"2024042305580277300_luae059-B7","doi-asserted-by":"crossref","first-page":"2313","DOI":"10.1210\/jc.2015-3910","article-title":"Management of hypoparathyroidism: present and future","volume":"101","author":"Bilezikian","year":"2016","journal-title":"J Clin Endocrinol Metab"},{"issue":"2","key":"2024042305580277300_luae059-B8","doi-asserted-by":"crossref","first-page":"457","DOI":"10.1007\/s12020-019-02128-z","article-title":"Teriparatide (rhPTH 1-34) treatment in the pediatric age: long-term efficacy and safety data in a cohort with genetic hypoparathyroidism","volume":"67","author":"Tuli","year":"2020","journal-title":"Endocrine"},{"issue":"2","key":"2024042305580277300_luae059-B9","doi-asserted-by":"crossref","first-page":"345","DOI":"10.1016\/j.jpeds.2011.09.022","article-title":"Recombinant parathyroid hormone therapy for severe neonatal hypoparathyroidism","volume":"160","author":"Cho","year":"2012","journal-title":"J Pediatr"},{"issue":"6","key":"2024042305580277300_luae059-B10","doi-asserted-by":"crossref","first-page":"2680","DOI":"10.1210\/jc.2009-2464","article-title":"Long-term treatment of 12 children with chronic hypoparathyroidism: a randomized trial comparing synthetic human parathyroid hormone 1-34 versus calcitriol and calcium","volume":"95","author":"Winer","year":"2010","journal-title":"J Clin Endocrinol Metab"},{"issue":"3","key":"2024042305580277300_luae059-B11","doi-asserted-by":"crossref","first-page":"P1","DOI":"10.1530\/EJE-18-0609","article-title":"Standards of care for hypoparathyroidism in adults: a Canadian and International Consensus","volume":"180","author":"Khan","year":"2019","journal-title":"Eur J Endocrinol"},{"key":"2024042305580277300_luae059-B12","doi-asserted-by":"crossref","first-page":"115834","DOI":"10.1016\/j.bone.2020.115834","article-title":"Recombinant human parathyroid hormone (1-84) replacement therapy in a child with hypoparathyroidism","volume":"144","author":"Laurer","year":"2021","journal-title":"Bone"},{"issue":"16","key":"2024042305580277300_luae059-B13","doi-asserted-by":"crossref","first-page":"1687","DOI":"10.1056\/NEJMc063043","article-title":"Recombinant PTH for initial management of neonatal hypocalcemia","volume":"356","author":"Newfield","year":"2007","journal-title":"N Engl J Med"},{"issue":"3","key":"2024042305580277300_luae059-B14","doi-asserted-by":"crossref","first-page":"556","DOI":"10.1016\/j.jpeds.2014.04.060","article-title":"Effects of pump versus twice-daily injection delivery of synthetic parathyroid hormone 1-34 in children with severe congenital hypoparathyroidism","volume":"165","author":"Winer","year":"2014","journal-title":"J Pediatr"},{"issue":"11","key":"2024042305580277300_luae059-B15","doi-asserted-by":"crossref","first-page":"3308","DOI":"10.1210\/jc.2011-1359","article-title":"Long-term results of continuous subcutaneous recombinant PTH (1-34) infusion in children with refractory hypoparathyroidism","volume":"96","author":"Linglart","year":"2011","journal-title":"J Clin Endocrinol Metab"}],"container-title":["JCEM Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jcemcr\/article-pdf\/2\/4\/luae059\/57300710\/luae059.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jcemcr\/article-pdf\/2\/4\/luae059\/57300710\/luae059.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,4,23]],"date-time":"2024-04-23T05:58:25Z","timestamp":1713851905000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jcemcr\/article\/doi\/10.1210\/jcemcr\/luae059\/7655903"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,3,22]]},"references-count":15,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2024,3,22]]}},"URL":"https:\/\/doi.org\/10.1210\/jcemcr\/luae059","relation":{},"ISSN":["2755-1520"],"issn-type":[{"value":"2755-1520","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2024,4]]},"published":{"date-parts":[[2024,3,22]]},"article-number":"luae059"}}